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A 60-year-old woman with athetoid cerebral palsy suffered a displaced femoral neck fracture of the left hip in 2004. She had been able to transfer from the bed to the wheelchair independently before the injury, but she lacked ambulatory ability in a community setting.\nRadiography revealed that the sharp angle of the left hip was 40° at injury []. The patient had no acetabular pathology prior to fracture. Five months later, she underwent bipolar hemiarthroplasty at another hospital and recovered uneventfully []. She had been able to return to her previous lifestyle, but she presented to us with a two-month history of severe pain in the left groin nine months after the hemiarthroplasty. Pain was aggravated by athetoid movement and increased muscle tone even at rest. Non-steroidal anti-inflammatory drugs did not relieve the pain.\nAt the first visit to our hospital, standard radiographs of the left hip demonstrated lateral migration of the prosthetic bipolar head []. Laboratory examinations revealed a normal C-reactive protein level and white blood cell count. Physical examination revealed mild restriction of left hip motion due to pain. She tended to adduct and extend her left hip with increased muscle tone. Antispasmodic drugs (p.o.) had little effect on pain.\nFour weeks later, in view of the severe continuous pain, we removed the outer head and placed the acetabular component with cement and without a bone graft. Intraoperatively, the lateral aspect of the acetabulum was found to be denuded of cartilage. We concomitantly performed release of the adductor muscles and medial hamstrings. A sample of clear synovial fluid taken from the joint at the time of surgery did not produce bacterial growth on culture.\nAt two years follow-up after the conversion, she could transfer independently between the bed and wheelchair with mild pain in the left groin, but the pain subsided at rest even with increased muscle tone. A plain radiograph revealed no signs of loosening or change in the position of the implants []. |
We present a case of a 40-year-old woman, gravida 4 para 1, with antenatal surveillance of her pregnancy in our institution. Her first pregnancy and labor occurred normally, fourteen years ago. Some years later, she started experience dysmenorrhea, pelvic pain, and heavy menstrual bleeding. The diagnosis of leiomyomata was made by ultrasonography and magnetic resonance imaging (MRI), showing a uterine volume of 580 cc with a dominant intramural fibroid of 105 cc volume in the anterior wall. She suffered two miscarriages after this diagnosis. After counseling about her different options of treatment, she always refused any type of surgery, trying to preserve her uterus and expecting a future pregnancy. Then, uterine artery embolization was performed with decrease in fibroid size of 24% after 6 months and improvement of symptoms. However, one year after, the symptoms recurred and uterine myomas returned to a previous size. She undergone for more 2 UAE in another center. The procedure was repeated in the second time because only 30% degree of fibroid ischemia was achieved after the first embolization. Moreover, MRI revealed some contributing circulation from the left ovarian artery.\nThree months after embolization she became pregnant spontaneously. On antenatal ultrasound scans several uterine intramural fibroids were evident including the dominant one placed at the lower segment of anterior wall measuring about 65 × 60 mm. The placenta was previa and the fetus was at transverse situation. For these reasons and because of vaginal bleeding incident, a cesarean section was performed at 37 weeks of gestation. Intraoperatively the lower segment was thickened due to underneath leiomyoma. A longitudinal hysterotomy was needed to access the amniotic sac. A 2970 g female fetus was delivered, with Apgar scores of 5 and 8 at 1 and 5 minutes, respectively. The placenta was delivered manually. After uterine contraction was noted, the prominence of the large leiomyoma with about 10 cm size in the anterior lower segment () and respective myomectomy was performed. A prophylactic Bakri balloon was left inside of endometrial cavity prior to complete hysterorrhaphy in three-layer suture. The remaining uterus was increased in size due to the presence of other smaller intramural leiomyomas, but its tonus was consistent.\nTwo hours later on postoperative recovery room, she developed atonic hemorrhage and became hemodynamically instable. A total hysterectomy was performed as a life-saving measure (). During surgery the patient was transfused with red blood cells, fresh frozen plasma, and platelets, in order to control the disseminated intravascular coagulopathy. Histopathology of the uterus showed adenomyosis, interstitial and subserous leiomyomas, and signs of previous embolization. The placenta showed no histopathological alterations.\nThe patient recovered well postoperatively and was discharged home on postoperative day 12. |
A 37-year-old woman visited our hospital with a bilateral hearing disturbance which had been aggravated for six years. She had a five-year history of otorrhea in both ears and had been treated conservatively with topical and oral antibiotics for the presumptive diagnosis of bilateral COM with cholesteatoma. She had a past medical history of longstanding allergic rhinitis and bronchial asthma and had been on medication for this for the past 10 years.\nAn otoscopic examination revealed turbidity of the bilateral tympanic membranes with middle ear effusion. Pure-tone audiometry (PTA) and the speech reception threshold test (SRT) revealed bilateral profound conductive hearing loss: 8 dB for the right ear and 5 dB for the left ear on PTA; 50 dB for the right ear and 48 dB for the left ear on SRT.\nTemporal bone CT (TBCT) performed at a private clinic two weeks ago revealed complete soft-tissue filling of the bilateral middle ear cavity without bony erosion and the sclerotic mastoid, resulting in bulging of the tympanic membranes (). TBCT also demonstrated mucosal thickening of the ethmoid and sphenoid sinuses, caused by inflammation. Temporal bone MRI for further evaluation showed T1 low and T2 intermediate signal, as well as enhancing lesion filling in the bilateral middle ear cavity. These MR findings are suggesting rich granulation tissue in the middle ear cavity rather than acquired cholesteatoma. The possibility of tumors such as adenoma or adenoid cystic carcinoma was very low from its bilaterality and from the findings of TBCT.\nThe patient underwent endoscopic sinus surgery and intact canal wall mastoidectomy on the right temporal bone within an interval of one month. On the surgical field, granulation tissue was found to be extruded to the external auditory canal from the middle ear cavity through the perforated tympanic membrane. The mastoid antrum and the air-cells were filled with purulent exudate and granulation tissue which had eroded the bony cortex of the mastoid air-cells. Histopathologic examination only revealed a large amount of granulation tissue with chronic active inflammatory cells. There was no histologic evidence of vasculitis or granulomatous inflammation. No micro-organisms were identified by the PAS, GMS, gram or Ziehl-Neelsen staining.\nApproximately four weeks following surgery, the patient visited the outpatient clinic again because of recurrent ear fullness. Otoscopic examination and follow-up TBCT revealed a recurrent, bulging mass from the right middle ear cavity and the mastoid protruding into the external auditory canal, which had gradually increased during the six-month follow-up. She underwent revision tympanoplasty and mastoidectomy on the right ear, which revealed chronic inflammatory infiltration with numerous eosinophils as well as Chartcot-Layden crystals within the inflammatory exudate. Based on these findings, this case was finally diagnosed as EOM. The patient was administered a high-dose steroid therapy to control the inflammation. During the five-month follow-up, there was neither recurrence of her symptoms nor bulging granulation tissue seen on the otoscopic examination. |
A 75-year-old man was referred to Kyung-Hee Dental Hospital from a local clinic for evaluation of paresthesia of the left lower lip and a firm mass on the left buccal side. He had a slight swelling localized in the area of the body of the left mandible. On palpation, the swelling was hard, attached to the body of the mandible, and slightly tender. Paresthesia was present on the left lower lip. An intraoral examination revealed a large erythematous mass in the lingual vestibular area (). The regional lymph nodes could not be detected by palpation. His medical history was unremarkable. According to his dental history, the patient had experienced dull pain in his left lower molar region 7 months earlier and underwent extraction of the left lower first molars because of a periapical abscess (). The patient underwent implant surgery approximately 4 months after the tooth extraction (). Two months later, explantation for peri-implantitis was performed. Since then, he had experienced continuous dull pain and swelling in the body of the left mandible and reported numbness of the left lower lip. The panoramic radiograph showed ill-defined, permeative bone destruction from the left lower premolar area to the second molar area. Comparison of this panoramic image with the one obtained from the local clinic on his first visit () showed that the lesion had expanded into the mandibular canal (). On the periapical view, there was infiltrative bone destruction and an enlarged soft tissue shadow (). Cone-beam computed tomography was performed to obtain more detailed information about the lesions. The sagittal images showed osteolytic destruction in the left mandibular body area and loss of cortication in the mandibular canal (). On the cross-sectional view, there was partial perforation and erosion of the buccal and lingual cortical plates (). An initial biopsy was performed under local anesthesia. A histopathologic examination revealed sheets of atypical plasmacytoid cells (). On immunohistochemistry, the tumor cells were negative for CD20, a B-cell marker (), but positive for CD138, a plasma cell marker (). While the kappa light chain was expressed in all of the tumor cells (), the lambda light chain was negative, indicating that the tumor cell population was monoclonal. The final diagnosis was solitary plasmacytoma. The patient is currently receiving radiotherapy. |
A 33-year-old man presented to our clinic with complaints of watering, redness, and purulent discharge in the right eye. The patient reported seeing a physician a year earlier in Georgia due to fatigue, nausea, vomiting, and diarrhea. After his diarrhea and vomiting had resolved, he had swelling of the lymph nodes on the right side of the neck. After returning to Turkey for treatment, he had received cephalosporin and penicillin for suspected pharyngitis. When night sweating and weight loss were added to his complaints, he had presented to another hospital where his treatment was changed to amoxicillin-clavulanic acid 1 g 3 times a day and ciprofloxacin 750 mg twice a day, and incisional drainage was performed on the lymph nodes of his neck. When his symptoms failed to resolve completely, he had presented to the department of infectious diseases of a different university hospital. Serum agglutination test was positive for F. tularensis at a titer of 1/1280 and he was prescribed streptomycin 1 g per day for 9 days followed by 1 g twice a day for 5 days for a total of 14 days, followed by doxycycline 100 mg twice a day for 1 week. Ultrasound examination of the neck had revealed multiple abscesses in the right submandibular region and pathological lymph nodes including multiple calcifications in the right cervical chain, while magnetic resonance imaging of the neck showed retropharyngeal abscess narrowing right nasopharynx and oropharynx and submandibular lymphadenopathies (LAP) including cystic and necrotic areas (). He reported that the LAPs had resolved after a few months with no recurrence, but complaints of watering, swelling in the lacrimal sac area, hyperemia, and pain in the right eye developed a few weeks later. The patient presented to our clinic with recurrent swelling around the lacrimal sac, hyperemia, and purulent discharge.\nOn examination his best corrected visual acuity was 20/20 in both eyes. Intraocular pressure measured by automatic tono-pneumometry was 15 mmHg in each eye. On slit-lamp examination, epiphora was noted in the right eye and the left eye was normal. There was swelling in the area of the right lacrimal sac (). Fundus examination was normal in both eyes. In nasolacrimal lavage, the patient’s right nasolacrimal duct was occluded and the common canaliculus was patent. Discharge of purulent material from the right lower punctum was noted after lavage. A sample of the purulent discharge was collected and sent to the microbiology laboratory for culturing and the patient was started on oral amoxicillin-clavulanic acid 1 g twice a day and topical ciprofloxacin drops 4 times a day. Antibiotherapy was discontinued because the culture was negative. Consultation from the otorhinolaryngology (ENT) department was requested to rule out any intranasal pathology. The patient underwent ENT examination, followed by nasal endoscopic examination. In addition, to rule out intranasal pathologies that may present an obstacle to surgery, the paranasal sinuses were examined using computed tomography. No intranasal pathologies were detected in ENT evaluation. Dacryocystorhinostomy surgery was recommended to the patient, but he refused the procedure. |
A 17-year-old male presented to the pediatric trauma hospital emergency department after an altercation with multiple gunshot wounds. As per advanced trauma life support protocol, life-threatening injuries were ruled out systematically. Orthopedic service was consulted to assess his injuries, which included three gunshot wounds of particular orthopedic interest. He sustained a gunshot wound to the medial aspect of the left thigh with profuse popliteal bleeding, gunshot to the right flank with missile air tracks down to the level of the posterior elements of the lumbar spine with resultant right L2 pedicle and laminar fractures, and finally, a left shoulder gunshot wound just inferior to the acromial margin in a lateral to the medial trajectory. Imaging revealed retained bullet fragments lodged in the posterosuperior quadrant of the glenoid (, ).\nAt initial presentation, while he was hypotensive and tachycardic, physical examination was notable for no sensation or motor control below the level of the umbilicus. Emergently, he was taken to the operating room for exploratory laparotomy as well as the vascular exploration of the left popliteal injury. Exploratory laparotomy revealed superior pole renal and caudate lobe hepatic lacerations which were hemostatic. The popliteal fossa was explored once proximal control was gained at the level of the groin and revealed considerable popliteal vein injury without arterial injury. Once hemostasis was obtained, fasciotomies were performed for the lower leg due to the significant time under tourniquet control before surgery. Osseous injuries to the spinal column were deemed stable, suitable for upright positioning, and the plan was to treat them conservatively.\nDue to the multiple injuries and critical status, his early care was dominated by intensive resuscitation, and once he was deemed adequately resuscitated for additional surgery, it was planned to address the retained bullet and shoulder joint arthroscopically. Of particular interest in addressing the left shoulder was the diagnosis of acute paraplegia with possible incomplete spinal cord injury, which would necessitate as much preserved upper extremity function as possible. The decision was made in concert with the surgical and intensive teams to continue intravenous antibiotic coverage until it was deemed safe to address the shoulder surgically. The pre-operative computed tomography (CT) of the shoulder () was carefully studied, and the bullet was located in the posterosuperior quadrant of the glenoid. The bullet measured 1.6 cm in diameter which is large and the normal arthroscopic graspers would not be adequate. Hence, various other instrument options were explored, including laparoscopic graspers. |
We report a case of a 30-year-old German woman who was first seen by the maternal-fetal medicine service for her first prenatal visit at 5 weeks of gestation at the Hospital Universitario Santa Fe de Bogota. This was her first gestation, and she had a history of recurrent urinary tract infections that required intravenous antibiotics. She was receiving a folic acid supplement and progesterone, and there was not any other relevant past medical or surgical. Her initial prenatal labs were within the normal limits.\nAt 9 weeks of gestation, she had her first ultrasound examination which revealed a normal fetal crown-rump length of 2, 13 cm and an abnormal location of the inferior body of the embryo, in the coelomic space (). These findings suggested a short umbilical cord syndrome. In order to confirm the diagnosis the patient was scheduled for a second ultrasound at 11 weeks of gestation. The obtained images confirmed the location of the inferior body in the coelomic space, with no visible bladder, absence of the right leg (), a severe abdominal wall defect, compatible with an omphalocele, and a short umbilical cord of 5 mm (). These last ultrasonographic findings were consistent with body stalk anomaly.\nBecause the malformation was considered to be incompatible with life, patient was offered to undergo voluntary termination of pregnancy. The embryo was sent to pathology after PG-induction. The embryo showed gross morphologic characteristics as follows: normal conformation of head with normal upper limbs, absence of the right leg, left leg bent toward the chest, and a severe omphalocele containing the bowels and liver.\nThe placenta had a measurement of 5 × 4 cm. Fetal umbilical cord was identified anteriorly, with central insertion and a short length of 2 cm and 0.3 cm diameter, wrapped by amniotic membranes that trapped the fetus. The fetus was immature and malformed. Head, part of the trunk, upper extremities, and left lower extremity were recognized. The face was symmetrical and the nose was flattened and lowered. The upper lip was not recognized, the lower jaw was maintained, and eyes had hypotelorism. The external ear structures were rudimentary and with low implantation. The trunk had scoliosis. There was a defect of the anterior and lower abdominal wall, with exposure of the intestine, liver, and spleen. The left leg was folded into the trunk. The upper extremities were normal. Findings were compatible with limb-body-wall complex (). The microscopic examination revealed immature chorionic villi that corresponded to the first trimester, with erythroid and nucleated forms. The membranes and the decidua were unaltered, and the fetal tissue was in continuity with amniotic membranes. The cytogenetic studies did not reveal numeric alterations in chromosomes 13, 18, and 21. |
An 80-year-old Japanese female noted swelling of the right side of the submandibular region and underwent a detailed examination at a local hospital. Based on ultrasonography, pleomorphic adenoma was strongly suspected. The patient was referred to our department for further detailed examination and treatment.\nOn initial presentation, an elastic soft and movable mass measuring 40 × 30 mm, with clear borders and healthy skin color, was palpable in the right submandibular area (Fig. ). No spontaneous pain or pain on pressure was present. Magnetic resonance imaging (MRI) revealed a round mass with relatively clear borders, measuring 29 × 24 × 40 mm, with the anterior edge bordering the right submandibular gland, whereas the posterior deep portion was compressing the right internal jugular vein. The margins were smooth. The mass exhibited a high-signal intensity area that was partially accompanied by a low-intensity signal area in the interior. There was no infiltration into the surrounding tissue, and no other significant lymph node enlargement was observed. Based on these findings, salivary gland tumor was strongly suspected (Fig. a, b).\nThe patient was hospitalized in our department. Two days later, tumor resection was carried out under general anesthesia. The tumor was round, encapsulated, and dark purple in color, and was relatively easily separated from the surrounding tissue. As the tumor was not contiguous with the submandibular gland, part of it was resected, and intraoperative frozen-section biopsy was performed. A large amount of blackish-brown, serous fluid was released from the tumor on resection. Inside the tumor, pale-yellow, papillary structures with a granular appearance were observed projecting toward the interior of the cavity (Fig. ). Intraoperative biopsy revealed cells that were suspected to have metastasized from the thyroid carcinoma. Based on this diagnosis, the tumor was separated from the surrounding tissue and excised intact. Two lymph nodes that were 12 × 8 mm and 10 × 6 mm in size adjacent to the underside of the tumor were also excised. No adhesion was observed between the tumor and surrounding tissue, and their separation was simple. The wound was washed with saline, and primary closure was performed to complete the surgery.\nThe final histological diagnosis demonstrated lymph node metastasis of PTC, with a positive reaction for thyroglobulin (Tg) on immunostaining. No metastasis was observed to the other excised lymph nodes. Based on the pathological diagnosis, postoperative examinations were performed, including neck ultrasonography, positron emission tomography (PET)-computed tomography (CT), blood tests [thyroid gland-related serological tumor markers such as Tg, thyroid-stimulating hormone, free triiodothyronine (T3), thyroxine (T4), and carcinoembryonic antigen], upper gastrointestinal digestive tract endoscopy, and plain chest radiography. The serum Tg level was high at 97.2 ng/ml, but no other abnormalities were observed.\nThe thyroid gland showed no abnormalities on CT and ultrasonography (Fig. ). After consultation with the Department of Endocrine Surgery at Tokyo Women’s Medical University Hospital, ultrasonography-guided fine-needle aspiration biopsy was performed twice in 2 months; however, carcinomatous components were not detected in the thyroid gland. In general, cases of cervical lymph node metastasis of occult thyroid carcinoma require total thyroidectomy. In this case, however, considering the slow progression of PTC, the patient’s age, past history of chronic cardiac insufficiency, hypertension, diabetes mellitus, and asthma, and overall physical condition, doctors at Endocrine Surgery could not get informed consent of the patient; therefore, close observational follow-up was selected. As of 8 years after surgery, no recurrence of the neck tumor or change in the size of the thyroid carcinoma has been observed, and the patient is progressing favorably.\nTumor cells had basophilic, polygonal, or cubic cytoplasm, and papillary proliferation. In the cytoplasm, or between cells, uniform structures considered to be slightly basophilic mucin were observed (Fig. a). Furthermore, separate from this area, one or two layers of similar tumor cells encompassed the surrounding area and proliferated in a follicular shape (Fig. b). Follicles were filled with uniform basophilic structures. Surrounding tumor cells often proliferated into the follicles. There was little mitosis or atypia in the tumor cells. Tumor cell nuclei were positive for thyroid transcription factor-1 (TTF-1), and Tg was positive in the cytoplasm of tumor cells and intrafollicles (Fig. a, b). These findings suggested PTC. No metastasis was noted in the other lymph nodes. |
A 16-year-old girl had surgery with the differential diagnosis of a median cervical cyst or thyroglossal duct cyst. The family history was otherwise unremarkable. Histopathology showed a papillary thyroid carcinoma. An evaluation with magnetic resonance imaging and radioisotope scanning of the thyroid gland as well as careful examination with regard to suspicious lymph nodes or metastases revealed no signs of further disease. Thus the girl underwent a reoperation four weeks later. The second operation appeared to be difficult at the upper thyroid lobe pole due to tight granulation tissue and scars. During the operation, magnifying, glasses were used. Both recurrent laryngeal nerves and all four parathyroid glands were identified. A total thyroidectomy with systematic dissection of the cervicocentral lymph node compartment was performed. One of the four parathyroid glands was autotransplanted into the left sternocleidomastoideus muscle. During the first day after the operation, the girl had symptomatic hypocalcemia, requiring intravenous substitution of calcium. On the fourth postoperative day, the girl developed a local erythema of the Kocher incision with fever and increased inflammation parameters. After opening of the wound, a small amount of pus was evacuated. Extensive lavage was performed daily. Fever and inflammation parameters were decreasing. At day 8 after the operation, while the girl was in good condition and the wound was unsuspicious, a sudden severe bleeding from the opened neck wound occurred spontaneously. Under manual compression of the wound, the girl was transferred to the operating room rapidly. During disinfection, the hemorrhage had ceased by the compression. Intraoperatively, the tissue was clean except fibrin layers on the muscles and the vessel walls. After careful separation of the planes in the depth of the wound, a severe bleeding developed. Digital compression and step-by-step exploration of the main vessels demonstrated a defect in the median wall of the right common carotid artery in the caudal third, about 6 × 3 mm in size with frayed margins. The distance from this defect to the origin of the carotid artery was scanty for the placement of a vascular clamp. Resection of the faulty artery segment was performed in an extension of 4 cm. An autograft from the saphenous vein was taken at the left proximal thigh. Blood flow could be detected to be 600 mL/min within the graft after reconstruction. In addition, a clamping time of the artery of 21 minutes was necessarily. As well as intra- and postoperatively, the girl did not have ischemic neurological defects all the time.\nBacteriologic culture of the excised tissue confirmed a localized staphylococcus aureus infection of the arterial wall. Histopathology of the specimen of the common carotid artery documented an acute purulent inflammation. After this operation the further clinical course was uneventful. Two weeks later the girl was dismissed. |
An 18-year-old male with history of bicuspid aortic valve presented to the emergency department (ED) the day after his senior prom with the chief complaint of right foot pain that had been gradually worsening over the prior three days. He had awoken at 4:30 am that morning and found that he could no longer bear weight on the right foot due to the intensity of the pain and had been using an old set of crutches to ambulate around his home. The patient indicated that the pain was primarily over the dorsal aspect of the foot, radiated up the back of the calf and was worse with bearing weight and movement. He had taken ibuprofen at home with no improvement in his symptoms. He denied any recent strenuous activity or injury to the foot, but had been dancing at his prom the previous night. He had been treated for plantar fasciitis in both feet by his podiatrist and had received a cortisone shot in the left foot three weeks earlier. Patient had also been having symptoms of fatigue, night sweats and fever for which his primary care physician had prescribed levofloxacin. He had completed one 10-day course 2–3 weeks prior to his presentation with only minimal improvement in his symptoms and had been started on a second 10-day course. The patient had been afebrile for the prior week, but he had continued to have problems with fatigue and night sweats and had recently developed exertional dyspnea. He denied intravenous drug use or recent dental procedures.\nOn initial examination, the patient was well appearing, but tachycardic at 118 beats per minute (bpm) with a grade 2/6 systolic murmur. He had normal breath sounds, was not tachypneic, and had a normal oxygen saturation. On examination of the right foot and ankle, he had tenderness over the dorsum of the foot and pain with range of motion of the ankle. Distal perfusion and sensation of the foot were intact. There were no overlying skin changes. The classic skin findings of endocarditis, including Janeway lesions, Osler nodes and splinter hemorrhages, were not present. A radiograph of the right foot was negative for fractures or dislocations. Basic labs including a blood count and basic metabolic panel were drawn that were significant only for an unexplained anemia with a hemoglobin of 8.9 g/dL. The patient was given a 1 L bolus of normal saline, but remained tachycardic on re-examination with a heart rate as high as 134 bpm. His electrocardiogram was otherwise normal. The decision was made to draw a D-dimer, which came back elevated at 721 μg/L. After a discussion with the patient and his mother, a computed tomography (CT) with contrast to rule out pulmonary embolism (PE) was ordered. Although negative for PE, his CT had concerning findings including an ascending thoracic aortic aneurysm measuring 4.1 cm, as well as ground-glass densities of the lungs. The patient was admitted to the hospital for an urgent echocardiogram, which revealed severe endocarditis affecting both the mitral and aortic valves (). Blood cultures were drawn and he was started on vancomycin and ceftriaxone.\nThe patient remained hemodynamically stable; however, three days after his admission, he developed multiple embolic phenomena manifesting as a left frontal infarct as well as an acute thrombus to the right internal jugular. After a negative magnetic resonance image of the ankle, a duplex ultrasound revealed the presence of an acute deep vein thrombosis of the right posterior tibial vein. Blood cultures were positive for Abiotrophia defectiva, but due to the difficulty in growing the organism the sample was sent to an outside facility and sensitivities were delayed. When sensitivities resulted, the organism was found to be sensitive to the empiric regimen. Notably, the organism was also sensitive to levofloxacin. The use of this antibiotic prior to his presentation may have contributed to his relatively benign initial presentation. The patient underwent bovine aortic and mitral valve replacements after which he was extubated and weaned off vasopressor and inotropic support. On post operative day 1, he developed a severe systemic inflammatory state with multi-organ system failure requiring re-intubation, escalation of vasopressors and inotropes, continuous renal replacement therapy and urgent venoarterial extracorporeal membrane oxygenation support due to refractory shock. A bedside echocardiogram revealed biventricular failure with ejection fraction of 5%. A biventricular assist device was later inserted, and approximately two months later the patient underwent successful heart transplantation. |
A 40-year-old male patient chronically maintained on phenytoin to treat seizure disorder, was diagnosed to have pleomorphic adenoma of left submandibular salivary gland for the last 1 year. The patient presented to surgery outpatient department with a chief complaint of swelling on the left side of the neck. The patient had a history of head trauma due to fall from a train with loss of consciousness and seizures but not of any bleed from ear, nose or throat, 20 years back. The patient had undergone some head surgery (probably craniotomy). The history of surgical procedure was unreliable because of unavailability of old records. After this, the patient was prescribed tablet phenytoin 100 mg thrice a day to manage seizure disorder, and the patient has been taking the same for the last 20 years. One year back patient noticed a small pea size swelling on the left side of the neck. The swelling had been increasing gradually in size since then to reach the size of a lemon at the time of presentation with relatively rapid growth in last month. Swelling was not associated with local pain, fever, anorexia, weight loss, cough with sputum, dyspepsia, or breathlessness. Besides above there was no history of any other chronic medical or surgical illness. On examination, patient was conscious and well oriented. His pulse rate was 76 beats/min, and blood pressure was 110/70 mm Hg. There was a visible around 3 cm × 3 cm, nontender swelling present in the left submandibular region. The overlying skin was without any redness, scar, sinus, fistula, or dialated and engorged vein. The swelling was mobile in both the directions and had no intraoral extension. It was not attached to underlying muscle and overlying skin. Local temperature was comparable to rest of the body temperature. There was no facial weakness and tongue deviation. Respiratory, cardiovascular, and central nervous system examinations were within the normal limits. Hematological (Hb 12.9 g/dL, total leukocyte count 7880 cells/μL, platelet 3.71 lac/μL) and biochemical (blood urea 28 mg/dL, serum creatinine 0.8 mg/dL, alanine aminotransferase 35 IU/L, aspartate aminotransferase 23 IU/L, alkaline phosphatase 176 IU/L, total bilirubin 0.5 mg/dL, Na+ 136 mEq/L, K+ 3.9 mEq/L) parameters were within the normal limits. Tri-dot and Mantoux tests were negative. Electrocardiogram and chest X-ray were also within the normal limits.\nFine needle aspiration cytology of the swelling, done on September 14, 2015, revealed pleomorphic adenoma of the left submandibular salivary gland.\nContrast-enhanced computed tomography neck showed heterogeneously enhancing mass lesion in the left submandibular region measuring 37 mm × 30 mm × 27 mm. Fat plane with parotid and surrounding muscles were maintained, no stranding seen. Ultrasound correlation showed well-defined hypoechoic mass in the left submandibular region medial to sternocleidomastoid muscle and anterior to carotid vessels, however, anterior part of submandibular gland appeared normal. For management of the condition submandibular gland excision was planned.\nAfter the patient was declared fit, surgery was performed successfully with adequate postoperative care. Peroperative findings revealed enlarged submandibular salivary gland involving both the superficial and deep lobe with normal surrounding viscera. The sample was sent for histopathological examination which did not show any malignant changes. There has not been a recurrence of the swelling yet at the end of 6 months during continuing follow-up. |
A 23-year-old female reported in the outpatient department with the chief complaint of an asymptomatic bony hard swelling in respect to the right posterior mandible, which gradually increased in size in the past 3 years to attain the present size of concern. Dental and medical history was insignificant. On clinical examination, there was evidence of a large non-tender, non-compressible, hard swelling measuring 2 × 2 cm on the right body of mandible, about 1 cm anterior to the posterior border of ramus, but no lymphadenopathy was noted. Overlying skin was normal in color, texture and consistency and was not adherent to the underlying swelling.\nIntraoral examination revealed a painless swelling in the right posterior mandibular vestibule from right mandibular second premolar to the distal aspect of second molar on the same side. The swelling was firm and covered by normal mucous membrane []. In place of the right mandibular first molar, only root stumps were present, but molar and premolar adjacent to it tested vital.\nRadiographically, it presented as a unilocular, radiolucent lesion, well circumscribed by a radiopaque border, about 3 × 2 cm, extending from the root of second molar posteriorly to the root of the first premolar anteriorly []. The lesion also caused a displacement of the right inferior alveolar canal toward the cortical inferior border of the mandible.\nThe lesion was perforated with a wide bore needle to rule out vascular lesion and aspirate the content. The aspirate was sent for pathological evaluation and the report stated it to be a cystic fluid with inflammatory exudates, confirming the clinical and radiographic diagnosis of radicular cyst in association with root stumps of 46.\nSo, enucleation of cyst with extraction of root stumps of first molar was planned. Under local anesthesia, an incision was made from distal of third molar to the medial of canine. Mucoperiosteal flap was raised and it was possible to see a thin expanded cortical plate which was removed with a scalpel. The cavity contained fibrous tissue wall full of liquid.\nThe lesion with all its fibrous tissue lining was enucleated carefully to ensure complete removal. The remaining bone tissue showed normal contour and consistency without any clinical signs of lesion. Root stump of first molar was extracted and mucoperiosteal flap sutured back after thorough curettage and lavage of bony cavity.\nThe histopathologic examination of the removed lining revealed UA contrary to the provisional diagnosis of radicular cyst []. Hence, root canal of 47 and 45 was done and radiologic observation of patient to monitor the relapse of lesion was chosen as the well-defined cystic envelope ensured the complete removal.\nPresently, the patient is under follow-up since 1 year without any signs of relapse and with bony restoration in the affected area []. |
A 33-year-old female who was previously healthy presented with swelling and pain over the right side of the neck that had been present for three weeks. The swelling was slowly increasing in size and associated with progressive right arm weakness. She also complained that the pain radiated to the right axilla as well as to the shoulder region. There was no history of fever or night sweats, but she reported losing about 5 kg in weight in the three months prior. She had no cough, and her clinical history was otherwise unremarkable. She had been a chronic smoker for the past 15 years with otherwise no relevant past medical or surgical history. Also, she was not taking any medications prior to the illness. On clinical examination, there was an area of diffuse swelling over the left side of the neck with marked engorgement of the superficial veins and was tender on palpation. The swelling also involved the upper part of the right shoulder (). Other than that, the clinical examination was normal. Endoscopic examination of the nasal cavity and larynx was unremarkable. Normal full blood count with a normal erythrocyte sedimentation rate of 4 mm/h and an elevated C-reactive protein of 30 units (normal range is 0–10 units) were found after the initial laboratory investigations. Thrombophilia screening, coagulation profile, urea and electrolytes, thyroid function tests as well as liver function tests were normal. She was referred for an ultrasound examination of the neck, which showed right cervical lymphadenopathy with right IJV and distal right subclavian vein thrombosis. The ultrasound revealed multiple enlarged right cervical lymph nodes involving the II, III, and IV levels. Ultrasound-guided core biopsy of the cervical lymphadenopathy was performed. She initially received intravenous antibiotics and was anticoagulated with low-molecular-weight heparin followed by warfarin. Consequently, she was subjected to computed tomography (CT) of the neck, chest, abdomen, and pelvis. Extensive right IJV thrombosis extending to the right subclavian, right brachiocephalic vein and into the superior vena cava was confirmed ( and ). CT also showed numerous right cervical lymphadenopathies at the II, III, IV, and V levels, and the right supraclavicular region showed extensive mediastinal lymphadenopathies (). Chest CT revealed a left lung mass with an irregular margin in the anterior segment of the left lower lobe measuring 2.3 x 2.6 x 2.5 cm. Another lesion with an irregular margin was also seen in the posterior segment of the left lower lobe measuring 1.3 x 1.3 cm with the liver, spleen, pancreas, adrenal glands, and both kidneys appearing normal. The histology of the lymph node core biopsy revealed lymphoid tissue exhibiting malignant tumor infiltrates. The tumor cells stained positive for cytokeratin 7 during immunochemistry, which is suggestive of metastatic carcinomas with possible differentials, including the lung and upper gastrointestinal tract. She was initially prepared for upper aerodigestive tract endoscopy and a CT-guided biopsy of the lung mass; however, the patient refused this procedure. Further investigations to establish a possible primary site, including gynecological and pelvic examinations, proctosigmoidoscopy, and tumor marker studies, were found to be normal. She was referred to the oncology department with a diagnosis of metastatic carcinoma of the lung for further management through chemo- and radiotherapy. |
A 26-year-old man was referred to our ENT clinic for unilateral hearing loss and ear pain in the right ear. Patient stated that his hearing was symmetric and normal prior to a wasp sting. Medical history taking revealed an initial presentation to the emergency department, approximately 29 days prior to our encounter in the clinic, for a sting to the right ear canal by an insect, and the development of unilateral hearing loss approximately 8 to 10 hours later. At that time, the patient complained of a foreign body sensation in the right ear. After inspection of the ear canal in the emergency department, the reported foreign body was removed and was revealed to be a wasp (Vespula vulgaris).\nInspection of the ear canal at the time of our evaluation revealed erythema above the right tympanic membrane and what initially appeared to be a small perforation in the left upper quadrant of the right tympanic membrane. Impedance testing were Type A bilaterally with normal volumes, so this could have just been a monomeric/dimeric portion of the Tympanic Membrane. Pure tone audiometry revealed a moderate hearing loss across all tested frequencies in the right ear. It was down-sloping with loss ranging from 40 dB at 0.25 kHz, down to 60 dB at 8 kHz (). Pure tone audiometry of the left ear was normal. Initial approach to management was a trial of oral steroids (Prednisone 60 mg for 1 week, followed by a taper over the following week). Patient was asked to follow-up within 14 days or sooner, but did not follow up until 42 days later.\nAt that visit after completion of the oral steroids, he denied improvement. Pure tone audiometry still showed a moderate hearing loss—approximately 40 dB across all tested frequencies, a slight improvement in the higher frequencies when compared with the previous audiogram (). An alternative approach using a intratympanic dexamethasone injection was tried. Topical phenol was applied to the right tympanic membrane and 0.5 mL dexamethasone (4 mg/mL) was injected through the anesthetized area.\nAt follow-up visit 7 days later, the patient reported a return of his hearing to baseline. There was a pinhole perforation to the right ear in the location of the intratympanic injection, but the previously described perforation/monomeric/dimeric area in the left upper quadrant had resolved. A repeat audiogram, now approximately 42 days from the initial onset of hearing loss, revealed a resolution of the sensorineural hearing loss. Bone conduction was all within normal range. Air conduction was also within normal range except for an air-bone gap of 20 and 10 dB at the 0.25 and 1 kHz, respectively, secondary to the small perforation from the intratympanic steroid injection site (). Impedance testing showed a large volume, as expected. |
The patient is a 21-year-old female involved in a high speed, helmeted motorcycle accident. After initial evaluation at a community hospital she was transferred emergently to a Level 1 care center. On arrival she was evaluated by the orthopedic and trauma surgery teams, and noted to have a severely deformed left lower extremity with a significant soft tissue injury and active proximal bleeding. She was taken to the operating room where bleeding was isolated to small branches of the femoral artery at the level of an inguinal crease laceration. Hemostasis was obtained by the trauma surgery team. Concurrently, the orthopedic surgery team placed a spanning external fixator across her comminuted, Type IIIB open femur fracture. Proximal fixation was within the proximal femur and distal fixation in the tibia, spanning the knee joint (Figure ). Of the pins within the femur, the proximal pin was placed using standard percutaneous technique and the distal pin placed under direct visualization through the large soft tissue defect (Figure ). Prophylactic four compartment leg fasciotomies were performed as well.\nAt the time of her index procedure distal pulses were absent and an on-table angiogram was performed. Initially, this showed limited flow below the level of the trifurcation, which improved with application of local vasodilatory agents. Over the preceding 12 hours her dorsalis pedis pulse was lost and a CT angiogram performed. This study suggested the SFA was lying lateral to the distal of the two femoral ex-fix pins, becoming kinked as the anatomic course of the vessel is medial to the femur. The altered path of the SFA was also recognized on retrospective review of the initial angiogram (Figure ). We emergently returned to the operating room and confirmed the findings of the CT angiogram, with a palpable SFA pulse noted to the level of the pin then no distal pulse. The pin was removed after retraction of the vessel, then replaced through the same location in the bone allowing the soft tissues and SFA to lie medial to the pin. This relieved the kinking of the artery and provided palpable distal flow.\nThis patient had a protracted hospital course in treatment of her severe left thigh injury. She underwent femoral fixation with a locking lateral femur plate, which required later revision by shortening the plate and removing the devitalized intercalary segment. She eventually underwent split thickness skin grafting of the wound. No further vascular complications were noted during her care. |
A healthy 35-year-old woman, gravida 3 para 1, was referred to our unit at 21 weeks and 5 days' gestation because of CMS. The course of the pregnancy had been uneventful until 16 weeks with no history of amniocentesis or abdominal trauma. The patient had two previous pregnancies. The first pregnancy had been uneventful, resulting in a caesarean delivery at term. The second pregnancy was terminated by dilatation and curettage because of a missed abortion at 8 weeks'\ngestation.\nAn ultrasound examination revealed a single living fetus with an estimated weight of 419 g. However, the amniotic membrane appeared to be in a completely separated state from the chorion except the site where the umbilical cord was inserted into the placenta. The amount of amniotic fluid was decreased overall in the amniotic cavity, while the echogenicity of the amniotic fluid was increased. In contrast, the fluid between the separated amnion and chorion had low echogenicity (). We tried to perform a targeted ultrasound examination for fetal anomaly; however, it was difficult to scan the fetal structure in detail due to oligohydramnios.\nAfter obtaining the patient's consent, we removed 20 mL of amniotic fluid for chromosome analysis and cytology and then slowly infused 230 mL of warm saline and 40 mg of Indigocarmine (United Pharm, Seoul, Korea) into the amniotic cavity with a 22-gauge spinal needle. After infusion, we could confirm the fetus had no evidence of fetal anomaly, including in its urinary system. About ten minutes later, the amniotic fluid in the amniotic cavity started to decrease to the previous size. In contrast, the space between the chorion and the amnion had enlarged. After removal of 200 mL of amniotic fluid from the space, we completed the procedure. There was no evidence of leakage of amniotic fluid into the vagina. Based on this outcome, our diagnosis was CMS with amniotic membrane rupture. Chromosome analysis revealed 46XX and cytology showed there was no no white blood cell in the amniotic fluid and its glucose level was 24 mg/dL.\nAt 26 weeks and 6 days' gestation, she complained of regular uterine contractions. We used betamethasone for fetal lung maturation and magnesium sulfate as tocolytics. At 27 weeks and 4 days' gestation, fetal electric cardiac monitor showed minimal variability of heart rate and recurrent variable deceleration. An emergency caesarean section was performed. The newborn was female and weighed 930 g without gross anomaly. Apgar scores at 1 and 5 minutes were 4 and 6. She was intubated and transferred to the neonatal intensive care unit.\nAfter the delivery of the baby and placenta, a localized mushy surface measuring 1.5 cm in diameter was found on the fundus of the uterus. There was no evidence of placenta accrete. We put one hand through the uterine incision and guessed at the thickness of the squashy uterine wall with the other hand over the uterine surface. There was the defect of the uterine muscle and the thickness of the remnant wall was less than 5 mm (). We could infer that the defect was one of the convincing reasons for the CMS, and that the uterine muscle defect had been caused by the previous dilatation and curettage before this pregnancy. There was complete CMS on the placenta and amniotic membrane was only attached where the umbilical cord was inserted on the placental disc (); pathology revealed acute chorioaminonitis, deciduitis, and acute funisitis. After delivery, maternal body temperature decreased to a normal range. The patient was discharged without any complications. Although the baby was diagnosed with respiratory distress syndrome and treated in the neonatal intensive care unit, she was discharged without complications 14 weeks after birth. The baby's current age is 15 months, and she has grown well without any morbidity. |
A 48-year-old male patient reported to the department of oral medicine and diagnosis with chief complaint of pain and swelling in the upper right gums. The patient noticed the nodule some 2 months back and pain since 2 weeks. The patient had a habit of smoking but had no familial cancer background. The clinical examination revealed a large mass of 8 × 3 cm in dimension on buccal aspect of right maxillary alveolus involving marginal, attached, and interdental gingiva []. The growth was blackish gray with intact surface. The margins were well defined. Anteriorly, it extends from the gingiva of mesial surface of 22, to the gingiva in relation to 17 posteriorly. Superiorly, the lesion extended to involve the upper buccal vestibule. Medially, it extended to involve the palatal mucosa. 13 was missing and 12 was displaced laterally; while, 11, 12, and 21 exhibited mobility.\nThe palpatory findings revealed a firm consistency of lesion with mild pain. The regional lymph nodes were non-palpable. A complete examination of the lesion was done and no other primary site of the lesion was found. Correlating all clinical features, diagnosis of primary malignant melanoma of oral cavity was made and the patient was referred for further investigations. The radiographic features showed no evidence of destruction of underlying bone. A computed tomography examination of neck, chest, abdomen, and bone scanning and ultrasounds of liver and kidney were normal excluding any diagnosis of distant metastasis.\nAn incisional biopsy was done for the lesion under local anesthesia and the specimen was sent for histopathologic examination. The gross examination of tissue revealed a mass of 2 mm × 3 mm × 1 mm in size, which was black in color and firm in consistency. The hematoxylin and eosin-stained section showed a melanin-producing tumor, consisting of atypical irregularly elongated spindle and oval-shaped melanocytes, exhibiting uniformly dark, enlarged and irregular nuclei []. In the superficial layers of the tissue, a junctional nevus with pigmentation was found []. The diagnosis of an invasive melanoma arising most likely from a pre-existing junctional nevus was made and the patient was referred to the oral and maxillofacial surgery clinic for required therapy. As per the traditional approach, partial maxillectomy of the right side was performed. To reduce the defect and to reconstruct alveolus, microvascular fibula flap was used. The orbital floor near to maxilla was reconstructed with the help of premolded titanium mesh. The open connection between the nasal and oral cavities was treated with removable prosthesis. The histologic examination of the specimen confirmed the initial diagnosis of an invasive melanoma of the oral mucosa. The underlying bone was intact. The patient has been followed-up with no evidence of recurrence or metastasis either clinically or radiographically, 11 months after the tumor's resection. |
A thirteen-year-old female was referred to our department with a six-month history of intense anterior left knee pain. The pain was localized at the anterosuperior part of the knee. It was described as continuous and referred to be 3 on the visual analogue scale (VAS) [] with exacerbation during sport activity until a value of 7. During the last month, the pain worsened especially at night reaching a value of 8 on the VAS without any exacerbation during daily activity. The patient had sought medical attention for the first time six months earlier when she was diagnosed with a PFM and underwent a course of physical therapy without any benefit. Her past medical history was unremarkable and no previous trauma was reported. There was no fever or any other systemic findings. Physical examination revealed a normal Q angle with a complete and painless range of motion (ROM). Tenderness during palpation of the superior part of the patella at the quadriceps insertion point was noted. There was no patellar crepitus and the patellar apprehension test was negative. Laboratory studies including erythrocyte sedimentation rate, C-reactive protein, and basic biochemical tests did not show any abnormality. A previous plain radiograph of the patient's knee in two projections was considered to be negative (). Considering that the pain was especially nocturnal, the patient was asked to undertake a course of conservative treatment, which consisted of taking acetyl-salicylic acid (one aspirin) every night for a period of ten days. At followup, the patient reported significant pain reduction every time she took aspirin. A three-phase scintigraphy was taken and it revealed a picking up area on the superior part of the left patella (). Then a CT scan demonstrated a round, well-marginated sclerotic lesion of about 8 mm of diameter with a hypodense rim and a centrally calcified nidus. This image was consistent with an osteoid osteoma ().\nThe chosen surgical treatment was a CT-guided percutaneous drilling. The procedure was performed in the CT suite under femoral nerve block and without the use of a tourniquet. A 3 mm Kirschner wire was initially driven percutaneously into the nidus under CT guidance. A superior approach through the quadriceps tendon was used in order to minimise retropatellar articular cartilage injury. Therefore, a small skin incision (1 cm) was made to accommodate a 8 mm drill that was inserted along the K-wire in order to remove the nidus mechanically. Postoperative CT scan confirmed that the nidus was entirely removed ().\nThe patient was discharged the day after the operation. An antibiotic prophylaxis was given and the patient was suggested to use a knee sling for about two weeks. The pain disappeared immediately and completely since the night after surgery without any drugs consumption. The patient came back to the hospital for a checkup after four weeks, reporting no pain and a normal ROM of the knee joint. After 2 years of followup, the patient was still symptom free and no degenerative change of the patellofemoral joint was found. |
A 20-year-old Asian man was admitted with a three day history of central abdominal pain and decreased appetite but no history of vomiting or constipation. Examination of his abdomen revealed mild tenderness to the right of the umbilicus and bowel sounds were normal. All blood results including inflammatory markers were normal and an abdominal X-ray did not show anything unusual. He was started on conservative management.\nThe next day, the patient’s abdominal pain increased. He was tender in both the iliac fossae on clinical examination with no rebound tenderness or guarding. He however settled down by evening and was well the next day. The following day (4th day since admission), the pain recurred and he started vomiting. He was passing flatus but no faeces. On examination, he had a slightly distended abdomen and was tender on the right side of his abdomen. There was no guarding or rigidity. Blood tests showed rising inflammatory markers (neutrophil leucocytosis) (White cell count - 12.2 and C - reactive protein 22). An abdominal X-ray showed dilated small bowel loops. He proceeded to have a CT scan of his abdomen which showed evidence of small bowel obstruction with a cut off at the mid ileum ().\nIn view of his clinical picture, a decision was taken to perform a diagnostic laparoscopy. This however had to be converted to a laparotomy due to significant dilatation of small bowel loops. Operative findings included dilated small bowel loops till the mid-ileum where an ischaemic stricture with perforation of the small bowel had resulted as a consequence of an omental band. This omental band had encircled the small bowel and was adherent to a nodule arising from one of the appendices epiploicae. The appendix was slightly congested. The band was divided and a wedge resection of the small bowel was carried out. The nodule to which the omental band had stuck to was excised and sent for histology. The appendix was removed.\nThe patient developed a small pelvic collection post -operatively which was managed conservatively with antibiotics. He was discharged from hospital 12 days after surgery and did not have any further complications.\nThe histology of the excised nodule showed an infarcted appendix epiploicae. Small bowel showed acute ischaemic ulceration with focal infarction extending through the bowel wall and the appendix was normal. |
A 7-year-old, previously healthy boy, developed a severe cough with respiratory distress following accidental aspiration of an FB (piece of magnet) with which he was playing. He was initially treated with nebulization and oxygen therapy. Chest X-ray revealed a metallic FB in the right hilar area []. He was referred to a higher center for bronchoscopic removal of the FB, where he reported the next day and underwent rigid bronchoscopy under general anesthesia for removal of the FB. A large, bullet-shaped metallic FB was visualized in the BI. Attempts to grab it by any kind of forceps only pushed it distally. Several attempts to engage it in a dormia basket were also unsuccessful. After nearly 2 h of attempts at removal, the child was noticed to have developed subcutaneous emphysema in the neck which started progressing rapidly suggesting the possibility of airway injury. A tear could be seen in the BI which was partially traversed by the FB. The bronchoscopic procedure was abandoned, ventilation through the endotracheal tube was continued, and a 16F chest tube was inserted into the right chest. There was no fall in oxygen saturation, and he was hemodynamically stable. With a diagnosis of suspected BI injury, he was transferred to our hospital for surgical management.\nAt a presentation in our emergency department, the patient was on mechanical ventilation through an endotracheal tube with stable hemodynamic parameters. Subcutaneous emphysema was noted in the neck and upper chest. He had reduced air entry in the right lower zone. Consent for thoracoscopy/thoracotomy was taken from the parents, and the child was immediately taken up for thoracoscopy under general anesthesia. Single lung ventilation with collapse of the right lung was achieved by selective intubation of the left main bronchus under bronchoscopic guidance. The child was placed in left lateral position with 30° anterior tilt (semi-prone position). Three 5 mm ports were created: camera port in 7th intercostal space (ICS) posterior axillary line and two working ports with right one in 4th ICS slightly posterior to posterior axillary line and left one in 9th ICS in the vertical line from the tip of the scapula. A 5 mm high definition telescope was used for visualization through the camera port, and two atraumatic graspers were used for dissection.\nOn entering the right pleural cavity evidence of atelectatic collapse was seen in the right lower lobe with minimal fluid and formation of early fibrinous exudate over the lower lobe. The right upper lobe was not adequately collapsed because of being inflated by the leak from around the cuff of the endotracheal tube. At this point, the table was tilted anteriorly, and CO2 insufflation at the pressure of 4 mm of Hg was used to enable us to see the posterior hilum adequately. The mediastinal pleural over the posterior hilum and the BI was intact and was seen bulging with ventilation suggesting leak from the airway. The lung was retracted anteriorly, and the posterior mediastinal pleura was opened. The BI was visualized. A 1 cm × 0.5 cm rent was noted in the BI through which the blackish FB was seen []. Although it was seen easily, multiple attempts to hold and remove it with an atraumatic laparoscopic instrument through the defect were not successful on account of the conical part of the body being tightly impacted distally. The FB was finally removed by pushing over the distal BI with an atraumatic bowel grasper and using a Maryland forceps in the other hand to overturn the FB and delivering it out of the bronchus. It was confirmed to be a magnetic object because it stuck to metallic instrument []. It was placed in a glove finger and taken out through one of the ports []. An endotracheal suction catheter was passed through one of the ports and guided into distal BI. Mucopurulent secretions were sucked out. Thereafter, the defect in the BI was closed with interrupted stitches using 3-0 polydiaxanone suture by thoracoscopic intracorporeal suturing (three stitches). The cuff of the endotracheal tube in the left bronchus was now deflated to allow ventilation gases to escape to right bronchus to do underwater leak test on the repaired BI. There was no air leak. A 20 Fr chest tube was placed in the right chest cavity, instruments and the two working ports were removed. Moreover, the telescope was withdrawn to just allow a view of inside. The endotracheal tube was now withdrawn into the trachea under bronchoscopic guidance to check for inflation of the right lung. The right upper lobe expanded immediately whereas the middle and lower lobes expanded slowly. At the end, complete expansion of all the lobes was observed. The telescope was also removed, and all the ports were closed. The total operating time (incision to closure) was 45 min. The child withstood the procedure well, was extubated on the table and shifted to recovery room. The air entry was now good and equal on both the sides, there was no air leak in the chest tube, and postoperative chest X-ray showed good lung expansion. He was allowed orally 2 h later and discharged the next day on parent's request on oral antibiotics and pain killers with ICD in situ. He was reviewed in the outpatient department 2 days later, and the chest tube was removed. He is presently doing well. |
The second case is a 61-year-old male who presented with acid reflux symptoms in early May 2015. He underwent an EGD with a mass noted, and a biopsy was obtained confirming the diagnosis of gastroesophageal adenocarcinoma (Fig. ). Staging scans revealed pulmonary nodules which were biopsied and demonstrated the presence of metastatic adenocarcinoma. The tumor tissue was amplified for HER2, with overexpression noted. His performance status was 0 as measured by the ECOG scale. The patient started combination chemotherapy with docetaxel, cisplatin, and capecitabine plus trastuzumab (Herceptin). Unfortunately, the patient became neutropenic and a dose adjustment was made. PET CT performed in August 2015 demonstrated an excellent response with the previously affected nodal areas now completely regressed. Capecitabine was discontinued after 3 cycles due to severe fatigue and hand-foot syndrome but docetaxel, cisplatin, and trastuzumab were continued for an additional 5 cycles. Given the continued response, cisplatin was discontinued and the patient continued on docetaxel and trastuzumab for another 2 cycles. In February 2016 repeat staging scans revealed no evidence of active disease; therefore docetaxel was discontinued and the patient was continued on trastuzumab maintenance (6 mg/kg every 3 weeks). The patient tolerated this therapy well, and ECHO examinations did not reveal a decrease in cardiac ejection fraction. However, after 3 months of trastuzumab maintenance therapy a PET CT scan demonstrated disease progression in the left supraclavicular lymph nodes. Paclitaxel was introduced while trastuzumab therapy continued. However, after 2 months on this therapy he demonstrated slight progression of disease and the therapy was then changed to FOLFIRI plus trastuzumab. He completed 6 cycles of this combination with evidence of complete remission on PET CT scan. At that time (January 2017) he received consolidative radiation therapy (50.4 Gy) to the primary tumor field and previously involved nodal areas including the supraclavicular lymph nodes. The patient completed radiation therapy (with capecitabine as radiation sensitizer) in March 2017 and was continued on maintenance therapy of trastuzumab. PET CT in June 2017 failed to demonstrate any evidence of disease recurrence or active metastatic disease process (Fig. ). The patient has continued on trastuzumab maintenance therapy with continued evidence of a complete response 30 months after the diagnosis of metastatic disease (last evaluation with PET CT was in September 2017). |
The second case is a 72-year-old man with recurrent pancreatic cancer. His original diagnosis of pancreatic cancer was in 2007 in an outside institution, after which he underwent a distal pancreatectomy. He was seen postoperatively at our institution for adjuvant treatment and surveillance, during which time he was found to have a local recurrence on imaging. His annual CT scan showed a soft tissue mass, located between the hepatic artery and the portal vein, which encased the former vessel. Its maximal diameter was measured at 2.5 × 2 cm, with an adjacent necrotic node measuring 1.2 × 1.5 cm (). On positron emission tomography, only focal mild hypermetabolism was noted at the site of the suspected recurrence with no evidence of metastatic disease, which was confirmed on a CT chest.\nThe patient underwent neoadjuvant therapy (Gemcitabine with radiation therapy followed by Gemcitabine-Oxaliplatin). A CT scan to assess response showed stable disease.\nIn April 2013, the patient underwent an angiogram that detected a replaced right hepatic artery originating from the SMA (). An endovascular stent (32 mm proximal extension graft [Renu Stent Graft, Cook Medical]) was placed to cover the CA. Three weeks later, the patient underwent an exploratory laparotomy, which revealed the mass in the surgical bed, where the CHA was completely engrossed by the tumor with a margin of less than 1 cm before the takeoff of the GDA. Confirmation of effective collateral flow after clamping the CHA was demonstrated and further confirmed with intraoperative Doppler sonography. The mass was resected en bloc after clamping the CHA and transecting it just proximal to the GDA. In this patient's case, we were required to resect the CA flush with the aorta to ensure gross negative margins; this was made less complicated because of the presence of the endovascular stent. A partial portal vein resection was also performed with a longitudinal repair.\nThe patient did well postoperatively with no complications and was discharged home on postoperative day 5. His final pathology report showed moderately differentiated adenocarcinoma measuring 2.5 cm with focal involvement of resection margin. Twenty lymph nodes were examined and one was found to be involved by adenocarcinoma.\nThis patient was seen recently for his 18-month follow-up and was doing very well with no clinical or radiological evidence of recurrence of his disease. |
The patient, a 31-year-old woman, was admitted to the department of neurosurgery at The First Hospital of Jilin University due to a “right occipital mass for 5 months”. The patient was in good health, and her family members did not have similar lesions. A physical examination showed that the mass was in the right occipital region at the upper margin of the sternocleidomastoid muscle and exhibited slight tenderness without obvious pulsation or vascular murmurs during auscultation.\nA head CTA examination at admission suggested that the lesion was approximately 3 × 5 cm in size, was mainly composed of vascular components, presented as a “wool mass”, was located in a subcutaneous region, involved the muscles, and showed obvious enhancement. The lesion was mainly supplied by the occipital and muscular branches of the vertebral arteries. The venous drainage of lesion communicated with the suboccipital venous plexus and the paravertebral venous plexus, and the venous drainage communicated with the subcutaneous cervical superficial veins (Figs. and 2).\nA further DSA examination revealed that the blood supply of the lesion was mainly from the occipital artery and less from the muscle branches of the vertebral arteries (Fig. ). The lesion was considered an AVM based on its imaging characteristics and was recommended for surgical removal. The surgery was carried out under general anesthesia. The lesion could be touched when the occipital skin was incised during the operation. The lesion showed a clear boundary. The occipital artery touched the anterior margin of the lesion, was exposed and ligated, and was removed around the lesion. The lesion consisted of massive blood vessels, and the surrounding muscles were swollen, indicating IH.\nAfter the lesion was removed, the normal muscle tissue around the lesion was also removed. The specimen was sent to pathology, and postoperative pathology confirmed that the lesion was IH accompanied by thrombolytic recanalization. HE staining showed that the lesion almost purely consisted of capillaries with only rare cavernous components among the muscles. CD31 staining was positive in the vessel endothelium, indicating that the lesion was an IH (Fig. ). The patient achieved a good recovery after surgery and was discharged from the hospital. A head CTA review performed one year later showed partial residual IH (Fig. ). The patient had no obvious discomfort, and the hemangioma could not be touched from the surface, and the patient was recommended for radiation therapy. The patient refused radiation therapy. Follow-up 2 years later showed no enlargement of the IH. |
A 67-year-old man visited the emergency department of our hospital due to sudden paraplegia of both lower extremities caused immediately after slip down. On presentation to the department, both his lower limbs were observed to be almost paralyzed, with a motor grade of 2 or lower in both limbs. This paralysis showed a gradual aggravation. The patient had a history of renal cell carcinoma diagnosed 7 years prior, and achieved remission without any recurrence after total nephrectomy and adjuvant chemotherapy. He was also diagnosed with papillary thyroid carcinoma 5 years ago, and yet again achieved remission after surgical treatment. The patient signed an informed consent statement, and the study was approved by the Institutional Review Board of the Yeungnam University Medical Center.\nThe plain radiograph revealed the collapse of the T12 vertebral body. CT showed an acute burst fracture at the T12 vertebral body with an osteolytic mass-like lesion within the vertebral body and pedicle, causing severe encroachment of the spinal canal (Fig. ). Magnetic resonance imaging (MRI) revealed a bulging posterior cortex, with an acute fracture of the vertebral body and severe compression of the spinal cord (Fig. ). Considering his medical history and the MRI findings, a pathologic fracture with a tumorous condition was suspected. Therefore, the evaluation of the malignant potential and metastasis in the remote area was necessary. Hence, 18FDG PET/CT was performed, and 18FDG uptake at the T12 level was measured. A maximum standardized uptake value (maxSUV) of 1.7 was noted, with a central FDG defect on the vertebral body (Fig. ); this was interpreted as a benign fracture by a senior radiologist. Additionally, there was no positive uptake in the other body parts on PET/CT. Although there was a discrepancy between the interpretation of the spine fracture lesion on MRI and PET/CT, the patient required emergent surgical treatment owing to acute paraplegia caused by spinal cord compression, and we thus planned an initial surgery for the main purpose of cord decompression, temporary stabilization, and tissue biopsy. The surgical intervention was as follows. First, we performed T11-12 posterior laminectomy for the spinal cord decompression, followed by T9-L2 posterior instrumentation without a bone graft. Finally, tissue biopsy was obtained at the T12 pedicle and vertebral body. Histological examination confirmed the presence of a malignant solitary bone plasmacytoma (SBP) (Fig. ). Seven days later, a definite surgery with pedicle screw fixation and posterolateral bone graft from T8 to L2 without vertebral body corpectomy was performed.\nImmediately after the initial surgery, the patient's lower extremity motor function showed gradual improvement. After the surgery, the patient wore a thoracolumbar rigid brace for 2 months, and underwent continuous gait rehabilitation. Three weeks postoperatively, the motor function in his lower extremities had recovered sufficiently, and ambulation was possible with the assistance of a cane. Four weeks after the definite surgery, the patient underwent radiation therapy for 2 months. One year postoperatively, spine radiographs showed that the surgical segment had healed fully, and spine MRI revealed complete decompression of the spinal cord with no evidence of local recurrence (Fig. ). |
Patient B is a 70 year old white female who presented with a large left breast mass. The patient was unsure how long the lesion had been present. Nonspecific findings consistent with a fibroadenoma were noted in the same region on a mammogram obtained 5 years previous to her presentation. Her past medical history was significant for hysterectomy at age 52. She had no personal history of cancer, although her family history was significant for lung and pancreatic cancers. She had used an estrogen and progesterone combination for hormone replacement in the past, the duration of which was unclear.\nOn physical examination, the patient's left breast had a multilobulated and relatively firm mass that measured approximately 12 cm in diameter and essentially replaced the entire breast. No cervical, supraclavicular, or axillary lymphadenopathy was noted. The contralateral breast showed no signs of a mass. Core tissue biopsy taken at the time of presentation suggested a diagnosis of cellular fibroadenoma or phyllodes tumor. Pre-operative CT scan revealed a 7.5 × 11 cm mass in the anterior portion of the breast, with no apparent mediastinal, lung, neck, or axillary lymphadenopathy.\nLeft modified radical mastectomy with dissection of level I and level II lymph nodes was performed. A right mastopexy was performed for purposes of symmetry. An elliptical incision encompassing the entire mass and the overlying skin was made. Collaterals in the skin supplied the tumor, and no deep invasion was identified. The superior and inferior skin flaps included skin that had been overlying the tumor. The tumor was excised along with the pectoralis muscle fascia. Axillary dissection was undertaken because of the presence of palpable level II nodes intra-operatively. A tissue expander was placed before final closure, as the patient desired reconstruction.\nThe pathologic findings of this procedure were consistent with a benign phyllodes tumor. The tumor measured 10.0 × 8.0 × 5.0 cm ex vivo. Microscopic sections showed large branching ducts surrounded by a uniform, bland stroma (Fig ); areas of hyalinization and myxoid change were rare. As with patient A, the margin of resection was negative for the tumor with a tumor-free zone that ranged from over 0.3 to 1.0 cm (Fig ). The Ki67 proliferation index of the tumor from patient B was 0.8 and 4 for the epithelial and stromal component, respectively (Fig ). No significant cytologic atypia or mitotic activity was noted. Sixteen lymph nodes were obtained, all of which were benign.\nThe patient had an unremarkable post-operative course and was able to start tissue expansion 19 days after her surgery. She had exchange of her tissue expander for a permanent implant 6 months after her mastectomy. She had an uneventful recovery from these surgical procedures and is currently 6 years post-surgery without complication. |
A 60-year-old male was referred to the Asan Medical Center for a nodule in the left lower lobe (LLL). The nodule was incidentally detected during the preoperative evaluation of gallstone, for which he underwent laparoscopic cholecystectomy at an outside hospital three months prior to his presentation to us. On his visit, he did not have any respiratory complaints such as productive cough or dyspnea. Medical history was unremarkable except hypertension. Further, the patient had been a non-smoker for 10 years although he used to smoke half a pack a day for 30 years before quitting.\nWe found that the patient's physical examination was unremarkable except decreased breath sounds over the left lower lung field. The laboratory data were all within normal limits. Chest X-ray and computed tomography scan revealed a mass having the following dimensions: 20×10×5 mm. The mass obstructed the secondary bronchus entering into the LLL, which resulted in a total collapse of LLL (). A flexible bronchoscopy showed an endobronchial mass filling the basal segments of the LLL (). Further, a biopsy indicated a granular cell tumor.\nThe patient underwent a left lower lobectomy via left posterolateral thoracotomy through the 5th intercostal space. The left thoracic cavity showed neither pleural adhesion nor seeding suggestive of malignancy. The LLL was heavily collapsed due to the obstruction by the endobronchial tumor. We divided the LLL bronchus at the level of the left upper lobe spur and performed a left lower lobectomy. The medial side of the left main bronchus was repaired using an interrupted anastomosis of 3-0 Vicryl. The resection margin of the bronchial stump was clear from the tumor on the frozen section. All five lymph nodes that were biopsied were tumor-free. The patient recovered well postoperatively and was discharged on postoperative day 5. Immunohistochemical staining demonstrated the positivity for S-100 protein, and the Ki-67 labeling index was low (1%), supporting the current diagnosis. The final pathology report confirmed the diagnosis of the granular cell tumor (). As of the writing of this paper, the patient has been free of tumor recurrence for six months. |
A 33-year-old man presented to our clinic with complaints of watering, redness, and purulent discharge in the right eye. The patient reported seeing a physician a year earlier in Georgia due to fatigue, nausea, vomiting, and diarrhea. After his diarrhea and vomiting had resolved, he had swelling of the lymph nodes on the right side of the neck. After returning to Turkey for treatment, he had received cephalosporin and penicillin for suspected pharyngitis. When night sweating and weight loss were added to his complaints, he had presented to another hospital where his treatment was changed to amoxicillin-clavulanic acid 1 g 3 times a day and ciprofloxacin 750 mg twice a day, and incisional drainage was performed on the lymph nodes of his neck. When his symptoms failed to resolve completely, he had presented to the department of infectious diseases of a different university hospital. Serum agglutination test was positive for F. tularensis at a titer of 1/1280 and he was prescribed streptomycin 1 g per day for 9 days followed by 1 g twice a day for 5 days for a total of 14 days, followed by doxycycline 100 mg twice a day for 1 week. Ultrasound examination of the neck had revealed multiple abscesses in the right submandibular region and pathological lymph nodes including multiple calcifications in the right cervical chain, while magnetic resonance imaging of the neck showed retropharyngeal abscess narrowing right nasopharynx and oropharynx and submandibular lymphadenopathies (LAP) including cystic and necrotic areas (). He reported that the LAPs had resolved after a few months with no recurrence, but complaints of watering, swelling in the lacrimal sac area, hyperemia, and pain in the right eye developed a few weeks later. The patient presented to our clinic with recurrent swelling around the lacrimal sac, hyperemia, and purulent discharge.\nOn examination his best corrected visual acuity was 20/20 in both eyes. Intraocular pressure measured by automatic tono-pneumometry was 15 mmHg in each eye. On slit-lamp examination, epiphora was noted in the right eye and the left eye was normal. There was swelling in the area of the right lacrimal sac (). Fundus examination was normal in both eyes. In nasolacrimal lavage, the patient’s right nasolacrimal duct was occluded and the common canaliculus was patent. Discharge of purulent material from the right lower punctum was noted after lavage. A sample of the purulent discharge was collected and sent to the microbiology laboratory for culturing and the patient was started on oral amoxicillin-clavulanic acid 1 g twice a day and topical ciprofloxacin drops 4 times a day. Antibiotherapy was discontinued because the culture was negative. Consultation from the otorhinolaryngology (ENT) department was requested to rule out any intranasal pathology. The patient underwent ENT examination, followed by nasal endoscopic examination. In addition, to rule out intranasal pathologies that may present an obstacle to surgery, the paranasal sinuses were examined using computed tomography. No intranasal pathologies were detected in ENT evaluation. Dacryocystorhinostomy surgery was recommended to the patient, but he refused the procedure. |
In February 2014, an 11-year-old male presented with a history of recurrent respiratory tract infections and suspected tuberculosis following a case within his family. On physical examination, he appeared to be in good health. Chest auscultation did not reveal any specific pathological heart or lung sounds. Abdominal objectivity was negative. His parents did not refer any other relevant medical history about him. Tuberculin skin tests resulted positive and a chest X-ray showed a dishomogeneous parenchymal consolidation in the left lower lobe (). Suspecting active tuberculosis, a preliminary CT scan of the thorax was performed showing a heterogeneous consolidation with some cystic masses containing mixed fluid and air in the left lower lobe posterior segment, not in communication with the respiratory tract, and a probable expression of a dysplastic parenchymal area ( and ). These findings were suggestive of pulmonary sequestration, including a differential diagnosis of congenital pulmonary airway malformation, because of the presence of a cystic component within the consolidation. The injection of contrast medium showed an artery arising from the descending thoracic aorta that divided into two 1.2 cm after its exit, with both branches extending to the dysplastic area; therefore, a diagnosis of intralobar sequestration with associated aspects of bronchial atresia was made; superinfection and trapping of contiguous parenchyma coexisted. Just below the emergence of the previously anomalous vessel, another artery was detected that crossed the midline to achieve a healthy parenchyma in the right pulmonary base, configuring a pattern of aberrant systemic artery feeding a normal lung (–). Other congenital anomalies that appear to be related to pulmonary sequestration were absent; in particular, pulmonary venous drainage was regular through the pulmonary veins, there was no communication between the bronchus and the oesophagus, and no diaphragmatic defects or other gross pulmonary anomalies were identified either. Our patient responded well to antitubercular antibiotic therapy and his clinicians, together with his family, decided to keep him under control with clinical follow-up, avoiding surgery for the moment. |
A 54-year-old Japanese female visited an oral surgery clinic with a complaint of swelling in the inner region of the left cheek for the past one month. On clinical examination, a mobile swelling (size, 1 cm x 0.75 cm) with a clear boundary was observed on the left buccal mucosa. No associated pain was reported and the overlying mucosa was normal in appearance. The swelling was clinically diagnosed as benign buccal mucosa tumor. The tumor was excised under local anesthesia and was diagnosed as acinic cell carcinoma (AcCC) after histopathological examination. The margins were still positive for the tumor and further resection was advised. The patient reported to the Health Sciences University of Hokkaido Hospital for resection of the residual tumor two months after the initial surgery. Clinically, the patient was asymptomatic. The level 1B lymph nodes on both sides were palpable, bean sized, mobile, elastic, and soft. Intraorally, a surgical scar of about 7 mm was present on left buccal mucosa. There was no pain on pressure in the region of the scar (). The patient had a history of noninvasive ductal carcinoma (ductal carcinoma in situ [DCIS]; Tis N0M0) in the right breast, which was treated by excision and 57 Gy of radiotherapy five months ago. On investigation for oral lesion, no obvious abnormalities were detected on the computed tomography- (CT-) scan, contrast MRI, and ultrasonogram. Positron emission tomography- (PET-) CT did not suggest transition to and from any of the distant organs. The margin was resected under general anesthesia and sent for histopathological examination (). No relation to the parotid gland was found at the time of surgery.\nHistopathologically, the excised margin appeared as a fragmented tissue with no encapsulation. The tumor tissue was composed of cells with dominant microcystic structure with eosinophilic cytoplasm and eosinophilic secretory material. Papillary and tubular pattern of cell arrangement were also found but were limited to small area. A few vacuolar cells and some areas with hemosiderin deposition were observed. Furthermore, normal muscle tissue and atrophied salivary gland tissues were also seen ().\nThe secretory material was positive for diastase digested Periodic acid-Schiff (d-PAS), Mucicarmine, and Alcian Blue staining. No zymogen granules were found in the tumor cells. Immunohistochemistry (IHC) revealed strong positive reactions to vimentin, cytokeratin-19, and S100 protein. Mammaglobin was strongly positive, whereas discovered on gastrointestinal stromal tumors 1 (DOG-1) showed a negative reaction (). The histological sections of breast carcinoma were examined in suspicion of metastases; however, features of ductal carcinoma in situ that appeared completely different from those of buccal mucosa tumor were noted. Based on these histomorphologic and IHC profiles, the case was diagnosed as SC of the minor salivary gland in the buccal mucosa. |
This is the case of a 25-year-old male with a past psychiatric history of schizophrenia who presented to the behavioral health emergency department (ED) under an involuntary hold for psychiatric evaluation due to worsening psychosis, with symptoms including persecutory delusions, auditory hallucinations and aggressive behaviors, leading to concern for risk of harm to self and others. On arrival, the patient was observed appearing agitated and paranoid with disorganized and threatening behavior, including aggression toward law enforcement officers who escorted the patient to the ED. The patient appeared to be confused and had difficulty providing clear information to the treatment team upon initial evaluation in the psychiatric ED. The patient’s medical and surgical history was remarkable for right retinal detachment status-post seven eye surgeries, left eye blindness due to accidental traumatic injury during childhood, and chronic disc herniation with associated chronic back pain.\nAs the patient was unable to provide a clear history to the treatment team, his father was contacted for collateral information. On interview, he indicated that due to extenuating circumstances, the patient had been unable to obtain his medications for chronic back pain, including oxycodone 20 mg oral (PO) four times daily and transdermal fentanyl patch 50 μg, one patch applied to the skin daily. The father stated that the last dose was taken one week prior to the presentation. It was suspected that inadequate pain control contributed to nonadherence with psychotropic medication regimens including antipsychotic medication for psychosis as well as benzodiazepines for anxiety with alprazolam 1 mg PO three times per day. The state’s online prescription drug monitoring program corroborated this information. In combination, this all resulted in an acute exacerbation of chronic psychosis with aggressive and disorganized behavior, leading to the patient’s psychiatric evaluation.\nDue to the patient’s acute presentation in the context of abrupt discontinuation of opioids and benzodiazepines, the patient was sent to the medical ED for a more extensive medical evaluation. In the ED, the patient remained verbally aggressive and combative, prompting the administration of lorazepam 1 mg PO.\nOn examination, the patient was hemodynamically stable, and the physical exam was within normal limits other than the aforementioned visual deficits; the patient had been previously reported as retaining only approximately 25% of the vision in his right eye. Laboratory examination was grossly unremarkable. Additionally, CT of the brain without contrast was completed for evaluation of altered mental status, confusion, and agitation (Figure ). The radiologist’s read deemed the imaging significant for “evidence of colpocephaly with pronounced enlargement of the trigone of the lateral ventricles in comparison with the frontal horns, as well as the parallel orientation of the lateral ventricles. In addition, a normal corpus callosum is not clearly identified; this constellation of findings suggests agenesis/dysgenesis of the corpus callosum.” MRI of the brain was offered for further characterization but was not completed during an inpatient admission, as there were no acute concerns regarding this finding on the patient’s presentation.\nFollowing medical clearance, the patient was transferred back to the behavioral health ED. Upon presentation, the patient was calm and cooperative but still grossly psychotic and disorganized, stating "I am a commander of the army where we fight monsters and submarines." The patient then signed voluntarily for admission to acute inpatient psychiatry.\nOn admission, the patient was noted to be isolative but alert and oriented to person, place, and time. In an initial interview with the primary inpatient psychiatric team, the patient demonstrated poor insight into the current situation and events leading to hospitalization. He demonstrated paranoid delusions, reporting that there was a "rotor" that was used to monitor his whereabouts. He also demonstrated grandiosity discussing how he had been part of the Central Intelligence Agency (CIA) since the age of three and had traveled the world extensively as part of the military until age four or five. The patient used bizarre neologisms and was tangential at times. At this time, no psychotropic medications were started due to patient refusal and his voluntary, capacitated admission status. However, as the patient had abruptly discontinued home medications of alprazolam and oxycodone, he was placed on the Clinical Institute Withdrawal Assessment for Alcohol (CIWA) and Clinical Opiate Withdrawal Score (COWS) protocols to monitor for symptoms of benzodiazepine and opioid withdrawal. The only present physical complaint was his chronic back pain.\nOn interview the following day, the patient continued exhibiting poor insight into his illness, the purpose of treatment, and the risks of treatment refusal. He was then converted to an involuntary and incapacitated status with his father signing as a healthcare proxy (HCP). With permission from the HCP, the patient was restarted on a home medication regimen of sodium valproate 500 mg PO at bedtime for mood stabilization and risperidone 1 mg PO at bedtime for psychosis. After a later discussion with the patient's father regarding prior response to risperidone and paliperidone palmitate, the decision was made to discontinue sodium valproate and optimize risperidone dosage with a plan to convert the patient to paliperidone palmitate intramuscular (IM) injections to be administered monthly for improved medication adherence. Risperidone was titrated to 2 mg PO daily with good effect and was tolerated well, thus the patient received an IM injection of 234 mg of paliperidone palmitate on day four of hospitalization with the plan to receive a second loading dose injection of 156 mg four to 10 days after initial injection with a continued monthly dose for maintenance.\nThe patient was also seen by the acute pain consult team, who recommended the following regimen for the patient's chronic pain: celecoxib 200 mg PO daily, diclofenac gel four times daily, gabapentin 300 mg PO three times daily, which was further increased to 600 mg three times per day, baclofen 20 mg PO twice daily as needed, and oxycodone 30 mg PO every six hours as needed. The patient requested one oxycodone dose daily with adequate control of pain. Utilizing the CIWA and COWS protocols, no acute withdrawal symptoms were noted throughout admission; however, it is notable that the patient had run out of prescribed benzodiazepines and opioids about one week prior to admission. He additionally complained of anxiety during admission, for which he received lorazepam 0.5 mg once daily as needed for several days, which was discontinued prior to discharge, once other psychotropic medications were optimized based on symptom improvement.\nThroughout the rest of his hospitalization, the patient was cooperative with the treatment plan and did not pose any management problems. The patient did not require any emergency treatment orders for behavioral issues. After eight days of inpatient treatment, the patient exhibited and endorsed improvements in behavioral and psychotic symptoms, denied any suicidal or homicidal thoughts, had returned to his baseline level of functioning, and was therefore deemed psychiatrically stable for discharge. Prior to discharge, a safety plan including continued medication adherence, outpatient psychiatry follow-up, and abstinence from substances was discussed at length with the patient and he expressed agreement and understanding of this plan. |
A 71-year-old man was admitted to our hospital because of progressive dysphagia, for >5 months, and a slowly enlarging nodule in the left lung, detected for 2 months. The patient was diagnosed with a squamous cell carcinoma in the lower third thoracic esophagus and a nodule in the left superior lung lobe. These were considered to be synchronous double primary lesions, based on the imaging characteristics. Chest and upper abdominal computed tomography (CT) showed wall thickening in the lower esophagus, accompanied by a solitary nodule, 2.3 cm in diameter, in the superior lobe of the left lung (Figs. and ). Lymph nodes >10 mm in diameter were detected around the left gastric artery and gastric cardia. Additionally, a 5 mm lymph node was found with the suspicion of metastasis along the right recurrent laryngeal nerve. By esophagogastroscopy, an ulcerative mass, 35 to 40 cm from the upper incisors was identified. Using endoscopic ultrasound, the esophageal tumor was staged as T3N1. No distant metastases were detected in this patient prior to treatment.\nThe patient refused any treatment, except surgical resection, because of his advanced age. Therefore, an MIE and lobectomy was performed, via a left video-assisted thoracoscopic surgical approach. First, we performed thoracoscopic lobectomy and thoracic esophageal mobilization in the right lateral position (Supplemental video 1). The operator was positioned in front of the patient. Based on our prior experience, a 2 cm incision at the 8th intercostal space, along the middle axillary line, was selected as the observation port. The principal operating port, with a 3 cm access incision, was made at the 5th intercostal space, before the anterior axillary line. Another 2 cm assisting operating port was located at the 8th intercostal space, along the posterior axillary line. Dissection of the left superior lung lobe was completed at the beginning of surgery. The patient was then turned to a right lateral-prone position and the posterior mediastinum was exposed. The thoracic esophagus behind the aortic arch and the lower part were readily mobilized. Lymph nodes along the left recurrent laryngeal nerve and thoracic esophagus were also dissected (Fig. A and B).\nAfter that, laparoscopy was completed, using a standard procedure with the patient in the supine position, as previously reported.[ Stomach mobilization and upper abdominal lymphadenectomy were completed. The gastric lesser curvature was then trimmed with a linear endostapler and the gastric tube conduit was made extracorporeally. A cervical collar incision was made in the suprasternal fossa and elongated along the inner edge of the left sternocleidomastoid muscle (supplemental video 2). The bilateral recurrent laryngeal nerves were exposed under direct vision, as for thyroid surgery. The mobilization of the upper third of the esophagus and the lymphadenectomy along the bilateral recurrent laryngeal nerves were completed (Fig. C), via video-assisted mediastinoscopy, as previously described.[ The thoracic duct was exposed and ligated with a Hem-o-lock clip (Fig. D). The gastric conduit was pulled up, using a posterior mediastinum approach, and a triangular mechanical esophagogastric anastomosis was made in the neck to avoid lethal anastomotic leakage.\nThe length of the postoperative hospital stay was 11 days, with no postoperative complications observed. The final pathological examination revealed a moderately differentiated squamous cell carcinoma invading the esophageal adventitia (pT3). A total of 40 nodes were dissected, with 4 positive nodes, along the left gastric artery and gastric cardia, were detected (N2). The pulmonary nodule in the left superior lobe was identified as a moderately differentiated adenocarcinoma with no lymphatic metastasis (pT2N0M0). |
A 75-year-old male patient without a history of diabetes or hormonal medication use underwent tension-free repair of bilateral indirect inguinal hernia at a county hospital 10 years ago with a Mard Mesh & Perfix plug (BARD Company, NJ, USA), which is made of polypropylene material. His postoperative recovery was uneventful, with no significant pain in the bilateral inguinal area and lower abdomen for an extended time, and no symptoms of systemic fever. However, 3 months before admission to our hospital, he experienced redness, swelling, and pain in the right groin, with high local skin tension, accompanied by systemic fever. He continued to receive treatment at that hospital and was subjected to local puncture through which a yellow, thin pus was extracted; the pus had a foul odor and was considered to be indicative of abscess formation. One 1 cm incision was made, and approximately 50 mL of pus was released. Antibiotics were administered intravenously, and his body temperature returned to normal the next day. Since then, the dressing was changed every day, but the amount of pus drained daily did not decrease, and occasionally, it appeared that gas was emanating from the wound; gradually, a sinus tract was formed. Even after treatment for nearly 3 months, the sinus tract did not heal.\nAfter admission, the patient showed good appetite, stool, urine, good nutritional status, and no fever. Surgical incision scars were observed on both sides of the groin and included redness and swelling on the right side, an ulcer of approximately 1 cm on the scar, and active purulent discharge. Forceps were used to probe the sinus tract, and the depth of the sinus tract was approximately 4 cm. The patient had no peritonitis symptoms except for tenderness in the right inguinal region. According to the medical records provided by the local hospital, the hernia sac was dissociated, which was followed by excision of the excess hernia sac, but the hernia sac was not closed. Instead, the mesh plug was inserted into the abdominal cavity through the hernia ring opening; then, the plug and peritoneum were stitched together with nonabsorbable suture to close the hernia sac. Routine blood examination on admission showed a normal white blood cell count and an abnormal increase in C-reactive protein (67.5 mg/L); moreover, Streptococcus hemolyticus was cultured from the wound secretion. Plain and enhanced magnetic resonance imaging scans of the lower abdomen suggested right inguinal mesh and soft tissue infection as well as sinus tract formation (Fig. ). On imaging, adhesion was observed between the mesh plug and intestine, but no abnormal features of infection, such as an abscess in the abdominal cavity, were observed. After oral administration of methylene blue for 3 hours, the wound dressing pus was pale blue (unfortunately, no photos were obtained). Based on the comprehensive medical history, we diagnosed the patient with mesh plug adhesion to the small intestine resulting in low-flow EF combined with CMI.\nThe treatment was as simple as possible. Instead of entering the abdominal cavity, the infected mesh plug was removed through the original incision scar, and then, the isolated small intestine, surrounding scar tissue and aponeurosis of obliquus externus abdominis were repaired with absorbable suture, leaving a gap. The foam dressing (made of polyvinyl alcohol) of the vacuum sealing drainage (VSD; Wuhan VSD Medical Science & Technology Co, Ltd, Wuhan city, China) was inserted into the gap and covered the incision and the NPWT was performed with the pressure of 125 to 300 mm Hg in continuous mode.\nAfter 3 days of intestinal preparation, an additional 6 cm was cut from the original incision scar and the surrounding adhesion was dissociated. It was found that the plain mesh was closely attached to the transverse abdominal fascia and adhered to the mesh plug below. An abscess cavity had formed around the mesh plug from which thin pus was extracted. The area surrounding the mesh plug was relaxed, but the basilar part of the mesh plug was attached to the small intestine, where a small amount of digestive fluid was intermittently discharged. An EF was confirmed intraoperatively (Fig. 2A: The hole probed with forceps was perforated, and a thickened intestinal wall with adhesion to the mesh plug was observed). We continued to dissociate the base of the plug and removed the infected mesh integrally (Fig. B).\nThen, the local infected tissue was eliminated, the separated bowel was repaired with absorbable suture, and the wound was repeatedly washed with hydrogen peroxide and saline. The scar tissue was sutured, and absorbable suture was used on the aponeurosis of obliquus externus abdominis, leaving a space of approximately 2 cm for the insert of the VSD foam dressing. Most of the incisions were sutured, and the incision was then covered with foam dressing. The wound surface was continuously rinsed with saline for NPWT; the daily usage of saline was 500 to 1000 mL. On the first postoperative day, the VSD foam dressings appeared yellowish green (Fig. A). According to the instructions for use, each VSD device was used for 1 week. The color of the VSD dressing (Fig. B–F) and drainage fluid and the amount of drainage fluid (Fig. A–E) were observed on the third day. The wound conditions were observed after each VSD device change (Fig. A–E). When the VSD device was replaced, zinc ointment was applied to the wound for protection (Fig. F). Five VSD devices were used before wound healing (Fig. E), and the time from intervention to wound healing was 35 days.\nPostoperatively, the sensitive antibiotic levofloxacin was used to treat the infection for 10 days. The patient had no symptoms of abdominal pain, peritonitis, or fever except for the pain and discomfort from the surgical incision. The white blood cell counts were normal in a repeat examination. The patient had flatus on the 1st postoperative day and defecation on the 2nd day. On the 2nd day, he received nutrition support with oral enteral nutritional suspension instead of intravenous nutrition support. The daily amount of fluid leakage in this patient was approximately 50 mL, which indicated a low-flow fistula, and no gastrointestinal digestive fluid inhibitors such as somatostatin were used. The support time of oral enteral nutritional suspension was 1 month.\nSix months of follow-up (Fig. ) revealed no bilateral inguinal infection and no recurrence of hernia. |
An 89-year-old Japanese man noticed swelling of the left maxillary gingiva in November 2016. In December 2016, he was referred to our department because the mass had slowly enlarged. His past medical history included clear cell RCC in his left kidney 7 years prior, which had been treated by nephrectomy. Multiple pulmonary metastases of RCC had appeared 3 years after surgery. He had received molecular targeted therapy with sorafenib for 4 years, which suppressed the growth of pulmonary metastases. Intra-oral examination showed an 8 × 8 mm granulomatous tumor with palpable pulsation in the buccal side of the left upper gingiva (Fig. a). The lesion enlarged rapidly over 2 weeks (Fig. b), and our patient began to feel pain while eating.\nPanoramic radiography revealed resorption of the left maxillary alveolar bone (Fig. a). Enhanced computed tomography showed a tumor destroying the left maxillary bone as well as the anterior and lateral walls of the maxillary sinus (Fig. b). Magnetic resonance imaging showed a 47 × 31 × 22 mm mass in the left maxillary bone and maxillary sinus, which extended into the oral cavity (Fig. c). Computed tomography angiography demonstrated that the mass in the left maxillary bone had strong enhancement and was fed by the infraorbital artery, posterior superior alveolar artery, and sphenopalatine artery (Fig. d). Our patient was clinically suspected to have an arteriovenous malformation or neoplastic lesion in the left maxilla.\nIn January 2017, 5 days after intravascular embolization of three feeding arteries, our patient underwent maxillary tumor resection. Pulsation around the tumor was not palpable after embolization. Prior to surgery, a biopsy specimen of the left maxillary gingiva had been subjected to frozen study. The results indicated that metastasis of the previously treated RCC could not be ruled out. Subtotal maxillectomy was performed by an intraoral approach (Fig. ). Intraoperative blood loss was only 26 ml. After surgery, there was no problems with oral intake of the patient. Although no tumor recurrence was observed in the oral cavity, our patient died 17 months after surgery because of widespread pulmonary metastases.\nHistologically, the neoplastic cells were arranged in an alveolar pattern with intervening delicate vascular stroma (Fig. a). The tumor cells displayed round to polygonal nuclei, with mild to moderate atypia, and large amounts of clear cytoplasm (Fig. b). Periodic acid–Schiff-positive granules were found in some tumor cells (Fig. c). Immunohistochemistry staining revealed that the tumor cells were positive for CD10 and AE1/3 (Fig. d, e). The tumor was diagnosed on histopathology as a metastasis of clear cell RCC to the maxillary bone. |
A 3-year-old Middle Eastern boy presented with a defect in the midline of his neck. He was born at full term by normal vaginal delivery and had no significance in his past medical history. There was no family history of congenital defects or consanguinity. The anomaly was located in the ventral midline of his neck (Fig. ). The superior aspect was composed of a skin tag leading to a short mucosa-like raw surface. Inferiorly, there was a sinus present with a greenish, thick residue occluding the opening. There was no contracture of the neck. He did not appear to be troubled by the lesion and a full examination was otherwise normal, except for adenoidal hypertrophy.\nHe had an MRI done elsewhere, indicating a soft tissue mass without any fistula tract. Despite contrast material being injected through the opening at the caudal end of the lesion, the diagnosis of MCC was established. No evidence of any other neck anomaly was found (Fig. ). The sinus, less than 1 cm in length, was found to extend caudally to the suprasternal notch. There were no attachments to underlying structures.\nA surgical removal and immediate closure with multiple Z-plasties were performed. Surgical removal was done with an incision 1–2 mm from the periphery of the lesion, deepened down to the supraplatysmal plane (Fig. ). During the surgery, the sinus at the caudal end of the lesion was probed and followed caudally until it ended, which was found to be approximately 2 cm long. This underdeveloped fistula tract ended right above the thymus gland. The cranial end of the defect had a fibrous band extending up to the mandible and this band was resected together with the cervical lesion. The midline lesion was found to be superficial and hence the excision was done at the subdermal level. A double Z-plasty was found to be sufficient for the closure. Closure was done with 5–0 vicryl interrupted sutures at the subcutaneous level and 6–0 rapid vicryl interrupted sutures for skin closure (Fig. ).\nA pathological examination of the specimen confirmed our clinical diagnosis. The findings were consistent with stratified squamous epithelial cells covering the cleft with few adnexial structures at the dermal level (Fig. ).\nOne month follow-up examination revealed an uneventful healing period, with redness along the incision scar and some nodularities, which were most probably due to the subcutaneous suture material. He was able to move his head in all directions without any restriction or pain (Fig. ).\nA 14-month follow-up examination showed an acceptable level of scarring causing no restriction of neck movements (Figs. and ). |
A 35-year-old woman (gravida 5, para 2) was referred to our hospital due to placenta previa at 34 weeks of gestation. Her previous pregnancies involved two cesarean deliveries, one spontaneous abortion and one induced abortion. She had no medical history, and during her current pregnancy, the placenta covered the entire anterior wall of the lower uterine segment, and she was diagnosed with placenta previa marginalis. Ultrasonographic findings revealed loss of a clear zone between the placenta and myometrium. Magnetic resonance imaging (MRI) revealed loss of the uterine myometrium between the placenta and bladder walls and broad adhesion between the uterus and bladder (Fig. ). Based on these findings, the patient was considered to be at high risk for placenta percreta. Fetal growth was appropriate for the gestational age. The patient was counseled regarding the complications associated with cesarean hysterectomy and the conservative management of placenta percreta, and we informed her about the severity of complications associated with the failure of conservative management. The patient expressed a desire to undergo conservative management to avoid potential intraoperative complications and not to preserve fertility.\nA planned cesarean delivery was performed at 36 weeks of gestation. Laparotomy, which was started under combined spinal–epidural anesthesia, revealed large blood vessels and the placenta penetrating through the anterior uterine wall and strong and broad adhesion between placenta and bladder wall was observed (Fig. ). Therefore, we suspected placenta percreta and determined that separating the bladder from the uterus would be extremely difficult. We made a transverse uterine fundal incision to avoid an incision into the placenta; a healthy male infant weighing 2312 g was delivered successfully. A uterine fundal incision demonstrated minimal bleeding from the incision site, and could avoid the iatrogenic partial separation of the placenta. After cesarean delivery, the uterus was well contracted, and no bleeding was observed from the placental site (Fig. ).\nA multidisciplinary team comprising obstetrics, perinatology, gynecologic oncology, and urology diagnosed her with placenta percreta with bladder involvement, approaching the pelvic sidewall and filling the cul de sac. Accordingly, we decided on conservative management to minimize potential surgical morbidity. The intraoperative blood loss estimated by the weight of the swab was 200 mL. The patient was not administered postoperative prophylactic antibiotics. She had an uncomplicated intra- and postoperative course and was discharged on postoperative day 14 with her infant in good condition. The patient was followed up at weekly intervals and examined for general condition determined by blood tests and ultrasonography.\nHer clinical course was unremarkable until postoperative day 42 when blood tests revealed that her serum fibrinogen level had decreased to 114 mg/dL (normal range, 150–350 mg/dL). By day 46, her serum fibrinogen level had decreased further to 62 mg/dL. On day 47, she was admitted to our hospital. Significant coagulation abnormalities were observed in her blood laboratory parameters: serum fibrinogen level, 42 mg/dL; activated partial thromboplastin time, 37 s (normal, 24–39 s); prothrombin time, 59% (normal, 70.0%–125.0%); and D-dimer level, 40.35 μg/mL (normal, <0.5 μg/mL). These results suggested the onset of DIC. The results of other blood tests are shown in Table . A careful physical examination and blood tests showed no sign of infection or bleeding; therefore, we considered that DIC likely was caused by the residual placenta. MRI performed on postoperative day 48 revealed that the placenta remained and that its vasculature extended to the bladder wall (Fig. ). Although we considered delayed hysterectomy as high risk, continuing the conservative management posed an even higher risk. However, the patient strongly insisted on continuing conservative management despite being informed about the associated risks. Therefore, we decided to continue with conservative management instead of performing hysterectomy.\nFirst, anticoagulant therapy (unfractionated heparin, 10,000 units/day) was administered subcutaneously. However, her serum fibrinogen level did not improve; thus, she received a transfusion of 1200 mL fresh frozen plasma (FFP). However, fibrinogen level did not improve with FFP transfusion. Therefore, activated partial thromboplastin time (APTT)-adjusted intravenous unfractionated heparin (APTT: 1.5- to 2.0-fold increase) was initiated as anticoagulant therapy. Intravenous unfractionated heparin was used from postoperative days 48 to 69, and her serum fibrinogen level successfully improved to 100 mg/dL and heparin-induced thrombocytopenia was not observed (Table ). Although her serum fibrinogen level was approximately 100 mg/dL for 2 weeks, no symptoms were observed. By postoperative day 65, her serum fibrinogen level markedly improved to 206 mg/dL. However, she had fever (>38 °C), a markedly elevated white blood cell (WBC) count, and an elevated C-reactive protein (CRP) level (Table ). We initially considered the possibility of an infection; however, a careful physical examination and bimanual palpation of her uterus revealed no sign indicating the presence of infection foci. Blood culture and procalcitonin test results also were negative. We concluded that the fever was induced by absorption of the placenta and not by infection. Therefore, we did not administer antibiotics and treated her with antipyretics instead. Although her fever and markedly increased CRP level persisted, her general condition was good; no clinical signs of infection were observed. On postoperative day 77, she was discharged and careful outpatient observation was continued. T2-weighted MRI on postoperative day 92 revealed a decreased placenta size (approximately 6 cm), and gadolinium-enhanced T1-weighted MRI revealed the lack of gadolinium enhancement of the placenta (Fig. ). By postoperative day 103, her fever had resolved. Ultrasonography on postoperative day 121 revealed a normal-sized uterus and no residual placenta. This was confirmed by performing MRI on postoperative day 232 (Fig. ). She resumed menstruation 5 months after delivery without any complications. |
A 15-year-old Chinese boy presented with left-sided chest pain, loss of appetite and weight, and low grade fever for 6 weeks followed by cough and shortness of breath for a week prior to admission. On examination, he was pale and febrile with a temperature of 38.5ºC. Examination of his chest revealed reduction of chest movement on the left side with reduced air entry into the left lung. There were no masses palpable. Haematological investigation revealed haemoglobin of 8.0 g/dL and a normal white cell count. Other blood investigations were unremarkable. A chest radiograph showed a large pleural mass in the left hemithorax, with rib destruction and a pleural effusion but no significant shift of midline structures (). A CT examination of the chest showed a large heterogenously enhancing mass arising from left chest wall with lytic destruction of the fourth rib and coarse calcifications. There was a left pleural effusion with underlying lung collapse consolidation. There were no lung nodules in the right lung to suggest metastases (). Due to the presence of calcifications in the tumour, a provisional diagnosis of osteogenic sarcoma with a differential diagnosis of chondrosarcoma was made. Pleural aspirate yielded bloody exudate. Cytology was negative and culture of the aspirate showed no growth. A tru-cut biopsy of the mass revealed osteogenic sarcoma. Histopathology findings showed a cellular tumour composed of diffuse sheets of cells with pleomorphic nuclei and basophilic cytoplasm with presence of multinucleated cells with mitotic figures. There were also areas of calcification and osteoid formation. This was in keeping with a tumour originating from the bone.\nHe underwent three cycles of chemotherapy. Post-chemotherapy CT examination for assessment showed reduction of pleural effusion but insignificant improvement in the tumour size. He was commenced on second-line chemotherapy for sarcoma consisting of Ifosfamide and Etoposide and completed two cycles. A CT examination done after completion of this chemotherapy regime again showed no significant reduction in tumour size. A decision was made for surgical excision of the tumour. At surgery, a necrotic tumour mass measuring 9 cm by 13 cm in size arising from the antero-lateral region of the left chest wall and surrounded by thickened pleura was found extended from the third to the seventh intercostal spaces. It was adherent to the left hemidiaphragm and causing significant compression of the left lung. The third to seventh ribs on the left side was resected en bloc followed by total clearance of associated necrotic tissue and diseased pleura. A left thoracoplasty and reconstruction of the affected chest wall defect using a titanium mesh was done (). He recovered well from the surgery. There was no further chemotherapy given to the patient after surgery. A CT examination done 3 months later showed deformity of the left chest wall and the titanium mesh in situ (). There was no evidence of tumour recurrence, pleural effusion, enlarged mediastinal nodes or focal lung lesions. The patient remained well ten months after surgery. |
A three and a half year old boy was referred to Pediatric Infectious Disease clinic with a history of asthma, environmental allergies, multiple lung infections, long bone fractures, and hand-foot-mouth disease, for evaluation of recurrent skin abscesses and bullae. By the time of evaluation in Pediatric Infectious Disease Clinic a, referral to genetics had already been initiated to evaluate the child for Osteogenesis Imperfecta because of the history of recurrent long bone fractures.\nAccording to the patient’s mother, the abscesses and bullae usually occurred on the face and were generally of rapid onset. Most often, they began as a pimple on the cheek in the morning and progress forming large mildly erythematous bullae by the afternoon. Multiple lesions had been lanced and cultured, most often revealing Staphylococcal species. The mother first noticed very bad lesions on the patient’s face as a small infant, initially attributing these lesions to bad baby acne; however, the complexion was much worse than her other children, as well as other children who she knew. Additionally, the patient also experienced multiple, severe blisters over the leg when a cast was removed due to a distal tibia fracture. Additionally, approximately one and a half months prior to evaluation the patient was admitted to an outside hospital for recurring bullae on the medial aspect of the left foot. These bullae were surgically debrided and the fluid expressed was cultured, revealing budding yeast. The patient had a similar infection when he was three years old. At that time a culture from the lesion revealed yeast and the patient improved with oral fluconazole.\nThe patient was the product of a 37 week 2 day uncomplicated pregnancy, but was hospitalized in the NICU for ten days, requiring mechanical ventilation secondary to pulmonary edema.\nA review of the patients past medical history revealed a combination left tibia and fibula fractures at age 21 months, a combination left radius and ulna fracture at 29 months, and a fracture of the left tibia at 41 months. Each instance was due to minor trauma.\nLaboratory studies obtained from the outside hospitalization revealed a white blood cell count of 11.4 cell/cm, hemoglobin of 12.0 g/dL, and platelets of 385,000 cell/cm. The differential demonstrated 52% lymphocytes, 5% monocytes, 11% eosinophils, 31% neutrophils, and 1% basophils. The erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP) were both normal. A biopsy of the debrided tissue on his left foot revealed dermal changes consistent with a hypersensitivity reaction to a fungal infection. Direct immunofluorescence shows weakly patchy granular dermal-epithelial junction of IgM and C3.\nThe lesions responded to intravenous ambisome followed by Itraconazole. The subtyping of IgG were within normal limits, IgG1 of 894 mg/dL, IgG2 of 164 mg/dL, IgG3 of 17 mg/dL, and IgG4 of 133 mg/dL, with a normal antibody response to the Diptheria toxoid and Tetanus toxoid and T and B cell subtypes were normal.\nPartial immunizations had been given to date and the patient history revealed allergies to sulfa drugs, eggs, and some wheat products by skin test. His family history was significant for a paternal grandmother with CVID and asthma, asthma and allergies on the paternal side, and the patient’s mother and maternal grandmother had systemic lupus.\nOn physical exam, in Pediatric Infectious Disease Clinic at the University of Michigan, the height was in the 89th percentile, the weight is in the 96th percentile, and he appeared as a playful young boy in no apparent distress. There were multiple small, non-erythematous, hard papules over the cheeks and bridge of his nose. The facies were not dimorphic and the dentition was good. No blue tint of hue was noted in the sclera and the pupils were equal round and reactive to light. There was no cervical lymphadenopathy with no other masses in the neck were appreciated. The lungs were clear to auscultation bilaterally. The abdomen was soft, non-tender, non-distended with no hepatosplenomegaly. The medial aspect of the left foot had new granulation tissue with a lesion approximately 3 cm by 4 cm from a previous bulla. New bullae formation was occurring on the periphery of the healing lesion. Muscle mass, strength and tone were within normal limits and the joints were not hyper-extensible. The neurologic exam was grossly intact.\nLaboratory studies obtained from Pediatric Infectious Disease Clinic revealed an immunoglobulin panel with an IgA level of 214 mg/dL (normal 15-160 mg/dL), IgG level of 1260 mg/dL (normal 405-1160 mg/dL), IgM level of 117 mg/dL (normal 40-190 mg/dL), and an IgE level of >10,000 kU/L (normal 0-150 kU/L). The subtyping of IgG were within normal limits, IgG1 of 947 mg/dL, IgG2 of 92 mg/dL, IgG3 of 18.3 mg/dL, and IgG4 of 42 mg/dL, with a normal antibody response to the Rubella. A CH50 level was normal at 101 units (normal 52-128 units). The ESR and CRP were normal and a Gram stain and culture from the left foot were negative. |
We present the case of a 78-year-old male patient with no relevant medical history who presented in February 2015 because of onset of pain associated with tumefaction of the right leg (Fig. ). In March 2015, a contrast-enhanced MRI showed an ulcerated tumor lesion on the posteromedial surface of his right leg, measuring 46 × 30 × 18 mm (Fig. ). In June 2015, the lesion was biopsied, and the histological analysis revealed histomorphological features compatible with undifferentiated spindle cell sarcoma. Chest, abdominal, and pelvic CT scans did not show secondary metastatic lesions. We suggested radiochemotherapy treatment to the patient in a neoadjuvant setting. Due to an underlying cardiac disease, the patient was contraindicated to anthracycline therapy. Thus, concomitant external radiotherapy together with trabectedin chemotherapy was finally proposed to the patient. Treatment with trabectedin was started in September 2015 at the approved dosage and regimen: 1.5 mg/m2 was given as a 24-h infusion on day 1 of every 3-week cycle. Between September 2015 and March 2016, the patient completed a total of 9 treatment cycles.\nParallel to that, between November and December 2015, the patient underwent external radiotherapy with a total dose of 45 Gy in 25 fractions during 5 weeks, directed at the tumor lesion on the right leg. There were no significant toxicities during the whole period of neoadjuvant therapy. An MRI preoperative evaluation performed in April 2016 revealed the same tumor lesion in the subcutaneous tissue of the posteromedial face of the middle third of the right leg, whose dimensions were similar to those of the previous examination. In May 2016, surgical resection with flap was performed, and the tumor was histologically confirmed as a high-grade spindle cell sarcoma with skin ulceration reaching striated muscle tissue. The tumor showed a very good pathological response with 80% of necrotic area; the neoplasia was abutting one of the circumferential resection margins. No postoperative radiotherapy boost was performed, due to the long healing period that surpassed the optimal period for that procedure.\nThe patient resumed his therapy with trabectedin in August 2016, following the same treatment regimen. In January 2017, a follow-up CT scan showed no secondary/metastatic lesions. An MRI of the right leg in February 2017 showed a nodular heterogeneous lesion with ill-defined margins, measuring 3 cm in the longitudinal axis and 3.7 × 1.8 cm in the transverse diameters (Fig. ). This lesion was suggestive of tumor relapse and had ill-defined margins with subcutaneous and muscular edema. In February 2017, the lesion was biopsied, and the histological analysis revealed a neoplasm on the right leg, compatible with a relapse of the clinically known undifferentiated spindle cell sarcoma.\nAfter group discussion, we proposed above-the-knee amputation to the patient as well as continuation of trabectedin therapy. In March 2017, an above-the-knee amputation was performed, and the histological analysis revealed a local relapse of the undifferentiated spindle cell sarcoma in the medial surface of the right leg, occupying the full width of the soft tissues, causing cutaneous ulceration and involving vascular structures. The patient resumed his trabectedin therapy in April 2017 with the same dosage and regimen. In November 2017, a clinical and image re-evaluation revealed that the patient is locoregionally and distant metastatically disease-free (Fig. ). Currently, trabectedin therapy is maintained. Up to January 2018, the patient has received a total of 30 cycles, without significant toxicity which mostly consisted of reversible neutropenia and transient transaminase increases. |
A 66-year-old male with schizophrenia, moderate cognitive impairment, and type 2 diabetes mellitus underwent hemicolectomy for adenocarcinoma of the colon, after which he received several months of chemotherapy through a totally implanted right subclavian vascular access port and catheter. This device remained in place thereafter and he was clinically well during the intervening 6 years before presentation. He lived in a residential long-term care (LTC) facility managed by registered nurses with physician backup around the clock. His port and catheter device was sterilely flushed with normal saline and heparin every 60 days by a nurse manager during this interval to maintain patency although the device was no longer being used. There had been no known port-related infections. The patient had no other indwelling catheters, or implanted devices. He was edentulous and had no prosthetic devices. He had no history of renal or urinary tract disease. He had had a single seizure 23 years previously followed by a normal EEG. He had a long history of self-abusive head slapping which averaged several times each week, resulting in a traumatic cataract and loss of sight in his right eye. He was independently ambulatory and took care of his own activities of daily living. His current presentation began with back and leg pains and onset of a shuffling gait a month after his previous 60-day port flush, which had taken place without complications. A nurse and another caregiver at his LTC facility assisted him after lunch one day to walk steadily back several meters from the dining area to his bedroom. He had had no tripping or falls but his mentation seemed a little slow and he wanted to sleep. Before going to bed, his vital signs and finger-stick blood glucose were checked. He was afebrile, hemodynamically stable, and euglycemic. Fourteen hours later, he was discovered kneeling on the floor beside his bed, unable to follow commands or explain how he was feeling. His temperature was 39.2 degrees Celsius, pulse 136 beats/minute, respirations 54 breaths/minute, blood pressure 137/65 mm Hg, and oxygen saturation 89% on room air. His finger-stick blood glucose was 328 mg/dL. A mechanical body lift was used to get him off the floor. He was transferred to the community hospital emergency department (ED) where he exhibited no bruising or signs of pain, trauma, breathlessness, rigidity, or shaking. The skin covering his implanted chest port and subclavian catheter appeared normal, without redness, warmth, swelling, or tenderness. His heart was in sinus rhythm; his electrocardiogram (ECG) showed no ischemic findings. His leukocyte count was 9.3 × 109/L with a normal differential and hemoglobin of 15.2 g/dL. His initial troponin was mildly elevated but serial troponin values that followed were normal. His serum creatinine level was 1.36 mg/dL (reference range, 0.70–1.25 md/dL); however, his blood creatine kinase (CK) level, previously normal, was elevated at 529 U/L and later peaked at 23 961 U/L (reference range, 0–200). His electrolytes, alanine aminotransferase (ALT), and aspartate transaminase (AST) levels were normal. His hemoglobin A1c was 9.4%. Urine drug screen was negative, and his influenza A and B tests were negative. His urinalysis showed amber color urine, 3–5 RBC/hpf, rare epithelial cells, 1 + mucous, and 0 WBC/hpf. A noncontrast computed tomography (CT) scan of his brain revealed no midline shift, mass effect, herniation, or territorial infarct. The calvarium and scalp were normal. Chest radiographs identified his port and catheter but no infiltrates, nodules, pneumothorax, or abnormal findings (his routine annual purified protein derivative (PPD) had been done one week prior and had been negative). Due to his severe rhabdomyolysis, the patient was transferred to a regional tertiary medical center. Blood cultures from that admission were reported as negative on hospital day 4. He was discharged after 9 inpatient days, off all antibiotics, and no source of fever had been discovered. His gait was his usual. His CK level was 441 U/L and soon returned and remained within the normal reference range.\nDuring the first 4 months after his discharge from the medical center, he had some intermittent gait disturbances with loss of balance at his LTC facility but no known falls. In the 4th month, he had 2 days of fever (maximum 38.6 degrees Celsius) but no lab work was obtained, and he received no antibiotics. He complained of headache and back and leg pains. His nurses noted a slightly shuffling gait from time to time. In the fifth month, he developed a left-sided Bell's palsy 6 days after a routine sterile port flush with saline and heparin. He complained of a left earache. He was afebrile but was also noted to be having increased delusions and audiovisual hallucinations. His otoscopic exam was normal and revealed no vesicles or sores. He was alert, cooperative, and followed verbal instructions to sit in a chair and lift himself up to a standing position, which he did immediately without use of armrests or other assistance. He demonstrated that he could walk back and forth across the room slowly but steadily and sit back down. A left foot drag that was not known previously was noted by a physician who was examining him. He was started on valacyclovir and prednisone. The next day, the color of the patient's skin appeared sallow and he admitted that he was not feeling well. He was transferred to the community hospital ED where his vital signs were normal including oxygen saturation of 94% on room air. He was incontinent of urine but had no cough, dyspnea, icterus, or rash. He was noted to have a left-sided Bell's palsy, and his ear examination was normal. The skin over his chest port appeared normal. A repeat noncontrast CT scan of his brain and skull showed no interval changes and no mastoid pathology. His leukocyte count and hemoglobin were normal; his CK was normal (45 U/L); and his creatinine, electrolytes, bilirubin, and transaminases were normal. His influenza tests were negative and his nasopharyngeal polymerase chain reaction (PCR) for Severe Acute Respiratory Syndrome Coronavirus 2 (SARS CoV-2) test came back later as negative. He was given intravenous fluids, started on levofloxacin for possible urinary tract infection, and discharged back to his LTC facility.\nWithin 48 hours, he spiked a fever to 39.3 degrees Celsius, and his pulse was 120 beats per minute. His oxygen saturation on room air was 96% and he had no cough. His gait became unsteady, so he was transferred back to the community hospital ED, where his leukocyte count was 8.3 × 109/L, creatinine was 1.26 mg/dL, and urinalysis showed 10 WBC/hpf and 1 + bacteriuria. His blood pressure was stable. A third noncontrast CT scan of the brain and head demonstrated no acute changes. His CT scan with contrast of the chest revealed a 3.3 cm left-sided thyroid nodule; the port with its tip located in the right superior vena cava; no focal consolidation, effusion, or pneumothorax; normal heart size without pericardial effusion; normal thoracic aorta; moderate circumferential esophageal wall thickening; and no osseous injury or axillary adenopathy. His CT scan with contrast of the abdomen and pelvis revealed normal abdominal aorta and vascular tree; no focal liver, gallbladder, pancreatic, or splenic lesions; and normal adrenals, kidneys, and bladder. A right hemicolectomy was noted, and there was no sign of free air. He was transferred from there to a regional tertiary medical center for possible sepsis due to suspected urinary tract infection. Two sets of blood cultures were obtained, he was started on cefepime, and, over a few days, his condition improved. His urine culture came back negative. Five days after admission, an aerobic bottle from one of the two sets of blood cultures taken at admission turned positive with gram-positive cocci in chains; this was believed on that day to most likely be a contaminant. . The following day, the isolate was believed to be a Kinyoun-stain positive gram variable rod. The Oklahoma State Department of Health Laboratory utilized matrix-assisted laser desorption ionization-time of flight mass spectrometry (MALDI-TOF MS) to identify the isolate as Mycobacterium neoaurum. The colony characteristics of this and future blood culture isolates from our patient were consistent with M. neoaurum. A sample of the initial isolate was sent to the Advanced Diagnostics Mycobacterium Laboratory at National Jewish Health in Denver, Colorado, and reidentified as M. neoaurum, by rpoB gene sequencing. A month-long process of antibiotic susceptibility testing (AST) was started, using a Clinical and Laboratory Standards- (CLSI-) aligned method of broth microdilution minimum inhibitory concentration (MIC) tests.\nIn the interim, our patient finished a week of cefepime and had been discharged from the medical center. He was asymptomatic. Due to the isolation of Mycobacterium neoaurum from only 1 of the 2 sets of blood cultures (1 bottle) from his medical center admission, repeat blood cultures were drawn at his LTC facility 10 days after antibiotics had been stopped. One set of blood cultures was obtained from a peripheral arm vein and the other set drawn from his central line port. The patient looked and felt well at the time his blood was taken but about 15 hours later, he was incontinent of urine and had a fever of 39.1 degrees Celsius. He was also pounding his head with his fist and trying to throw himself out of bed. He did not answer questions, would not stand on his own, and slumped over if sat up. He was transferred again to the community hospital ED, where he had an unremarkable CT scan of the brain and an unremarkable chest radiograph and lab studies, including a normal leukocyte count. The skin over his port appeared normal, without erythema, swelling, and tenderness. He had no heart murmur and no lymphadenopathy. He was hydrated and observed in the ED while being started on levofloxacin for possible urinary tract infection. He became alert, afebrile, and ambulatory and wanted to return to the LTC facility. He was discharged to the LTC facility and doxycycline was added the next day to cover for possible M. neoaurum bacteremia. Two days later, both aerobic blood culture samples drawn from his arm and from his port before he was sent to the ED turned positive with a microorganism later identified as M. neoaurum.\nThe patient remained stable on dual antibiotic therapy and was admitted within 48 hours to the tertiary medical center and had surgical removal of his port and subclavian catheter. His central and peripheral venous blood was cultured the day before surgery and he was started on imipenem in addition to doxycycline and levofloxacin. The tip of his subclavian catheter was sent for culture and grew M. neoaurum. Three of the 5 peripheral blood cultures drawn the day before surgery were positive for M. neoaurum including 3 lyses centrifugation blood cultures. Blood drawn the day before surgery from the patient's central line port was also positive for M. neoaurum. Postoperative blood cultures drawn in the hospital 4 days after the removal of the port and subclavian catheter were negative.\nThe patient was discharged to his LTC facility on hospital day 6 and was continued on the combination of imipenem, levofloxacin, and doxycycline for about 4 weeks. He pulled out his peripherally inserted central catheter (PICC) line after 4 weeks of therapy, at which time we had just received the antibiotic susceptibility report with minimum inhibitory concentrations (MIC) from the National Jewish Health Mycobacteriology Laboratory in Denver, as follows:Amikacin MIC: ≤8mcg/mL = S Cefoxitin MIC: ≤16mcg/mL = S Ciprofloxacin MIC: ≤1mcg/mL = S Doxycycline MIC: ≤1mcg/mL = S Imipenem MIC: ≤2mcg/mL = S Linezolid MIC: ≤1mcg/mL = S Moxifloxacin MIC: ≤0.5mcg/mL = S Tobramycin MIC: ≤2mcg/mL = S Trimethoprim/sulfamethoxazole MIC: 1/19 = S\nBased on susceptibility data and resolution of symptoms after port removal and start of antimicrobial therapy, the patient was continued on levofloxacin and doxycycline totaling 6 weeks of therapy. A colonoscopy was performed about one month after completing the antibiotic course and revealed multiple benign tubular adenomas in the transverse colon. Our patient remains asymptomatic and free of signs of infection at 4 months after removal of his port and subclavian catheter. |
A 2-year-old female child patient visited the department of oral and maxillofacial surgery with a complaint of diffuse swelling in the right mid face since one year. The swelling was progressive with gradual increase in size, consequently involving the contralateral side causing a swelling in the left mid facial region after 6 months. Both the swellings exhibited gradual increase in size with evidence of nasal blockage ().\nOn examination, the swellings were diffuse extending on to the zygomatic arch, nontender with no secondary changes. Intraoral examination revealed a nontender, lobulated swelling which was firm to bony hard in consistency (). The swelling caused labial and buccal cortical expansion bilaterally and extended up to the pterygoid plates along with palatal bone thickening. The overlying mucosa was intact. The patient's family history revealed that the elder sister, who is currently five years old, had a similar complaint 3 years ago, which was then operated, and a histopathology report of ameloblastic fibroma was rendered. Presently she is free from any recurrence.\nComputed tomography with 3D reconstruction was performed which revealed a hyperdense mass involving the labial and buccal cortex and extending up to the pterygoid plates. The palatal bone also showed thickening with irregular surface (). Based on the clinical and roentgenographic findings, a presumptive preoperative diagnosis of odontogenic tumor was made. The lesion was excised and curettage of the adjacent maxillary bone was performed under general anesthesia. The surgical specimen was then sent for histopathological analysis. Macroscopically, the specimen measured 3.5 × 1.5 cm in greatest dimension and was firm in consistency with a smooth surface ().\nMicroscopically, the lesion showed proliferation of strands of ameloblastic epithelial cells within a moderately cellular connective tissue stroma that closely simulates the dental papilla. The epithelial islands, nests, and strands were composed of peripheral tall columnar hyperchromatic cells exhibiting reversal of polarity and loosely arranged central cells having angular to spindle shape. The mesenchymal component comprised evenly distributed plump ovoid and stellate cells in a matrix of loose myxoid tissue (Figures and ). |
A 23-years-old female who is a known case of RA from the age of 16 years and was on regular treatment for the same was referred to skin department by the rheumatologist, with complaints of loss of all hairs from the scalp and the body for the past 12 months. The patient denied having similar complaints before. According to the patient, she had been treated with multiple therapies for joint pains and swelling. Initially, she was managed with nonsteroidal anti-inflammatory drugs, along with oral methotrexate for approximately 4 years to which she was responding well. However, after 4 years, in spite of regular treatment, she gradually started developing deformities of hands and feet for which her rheumatologist started her on subcutaneous injection etanercept once fortnightly for initial 3 months and then once monthly for next 3 months leading to a marked improvement in her arthritis. After 6 months of the treatment, the patient gradually started losing her hair from the scalp followed by other body hairs. After observing this, her rheumatologist referred the patient to our clinic. The patient does not give any other history of triggering factors such as any kind of stress, infection, or trauma.\nHer dermatological examination revealed multiple patches of nonscarring alopecia along with the presence of few thin, pigmented hairs on the scalp and complete absence of hair in axilla, pubic region and other body parts as shown in Figures –.\nBiopsy of scalp showed the presence of miniaturization of the hair with peribulbar infiltrate as shown in . On doing dermoscopy of the scalp, it demonstrated the presence of black dots, yellow spots, and dystrophic hair []. The patient had a normal thyroid profile and normal vitamin D3 level. The patient was started on oral mini-pulse of steroid and topical treatment with minoxidil 5%. After 3 months, she started regaining hair of scalp in a patchy pattern, but no hair growth was observed in other body parts.\nIn our case, we made a diagnostic hypothesis of AU which is stimulated by the use of anti-TNF drug etanercept.\nThe decision to stop etanercept was made after consulting her rheumatologist. |
An 82-year-old man was brought to the hospital after sustaining a mechanical fall at home and was found to have a non-operable left greater trochanter fracture. On abdominal imaging, he was incidentally found to have a 9.2 cm dominant mass at the dome of the liver straddling the left and right hepatic lobes with a 6 mm right lower lobe pulmonary nodule, which was suspicious for metastatic intrahepatic cholangiocarcinoma. A subsequent liver biopsy confirmed moderately differentiated adenocarcinoma. Based on his age and performance status, the patient began treatment with single agent gemcitabine 1000 mg/m2 on days one and eight every three weeks in conjunction with pegfilgrastim.\nOn the fourth day of his first cycle with gemcitabine, the patient developed a blanchable maculopapular rash on his upper chest, which eventually resolved with loratadine and diphenhydramine. However, the patient became increasingly fatigued after day eight of gemcitabine. Due to these side effects, gemcitabine was dose reduced to 500 mg/m2 every other week. Although his fatigue improved on the days he did not receive chemotherapy, the patient complained of new onset of shortness of breath and a dry cough that persisted into his second treatment cycle. During an office visit, a pulse oximetry measurement registered the patient as breathing 92% on ambient air. A chest X-ray showed bilateral pleural effusions (). Even after initiating furosemide to facilitate diuresis and a short course of corticosteroids, the patient continued to have dyspnea on exertion. The patient, who had no significant history of cardiac or pulmonary disease, underwent a transthoracic echocardiogram, which revealed an estimated pulmonary artery systolic pressure of 35 mmHg assuming a right atrial pressure of 15 mmHg; this finding was consistent with pulmonary hypertension likely secondary to gemcitabine (). Computed tomography (CT) angiogram of the chest was performed to rule out pulmonary thromboembolism (PE), and it was negative. The patient was promptly treated with diuretics and gemcitabine was discontinued given the high suspicion of drug related toxicity causing pulmonary hypertension.\nThe patient could not tolerate any further treatment with gemcitabine at which point his regimen was changed to fluorouracil and leucovorin. Despite this, he endured worsening symptoms and ultimately opted for hospice care. |
Our patient was a 74-year-old Chinese man who had undergone left radical nephrectomy 10 years earlier. Postoperative pathological examination revealed clear cell carcinoma. One year later, he returned for laparoscopic cystectomy due to cholelithiasis, and we found a mass in the subcutaneous tissue protruding by about the size of a thumb into the abdomen. We did not resect the protruding mass, and we decided to see our patient on an annual follow-up basis. Nine years later, he came back to see us because he noticed the protruding mass in the subcutaneous tissue had grown larger within the last year. His physical examination revealed two masses, and enhanced computed tomography (CT) showed a 5 × 6-cm mass in the left side of the anterior abdominal wall and a 5 × 6-cm mass in the back of the right waist (Fig. ). Enhanced CT also revealed a hypervascular lesion in the pancreas (Fig. ). The patient’s tumor marker carcinoembryonic agent concentration was 6.0 ng/ml. Malignant tumors were suspected, and resection of the tumors was performed.\nIn surgery with the patient under general anesthesia, we first placed the patient in prone position to resect the tumor in the back of the right waist. Then, he was placed in supine position to resect the tumor in the left side of the anterior abdominal wall. Both tumors in the front and back were around 5 × 6 cm in size, and clear cell carcinoma was suspected. Later, we performed distal pancreatectomy with spleen preservation because enhanced CT showed a hypervascular lesion of approximately 3 × 3 cm in the pancreas. The size of the resected tumor in the left side of the anterior abdominal wall was 4 × 2.8 cm; the one in the right waist was 4 × 2.5 cm, and the ones from the pancreas were 1.8 × 1.3 cm and 1.9 × 1.5 cm. All resected tumors were of the clear cell type. Histopathological examination revealed they were paired box gene 8-positive (PAX8+), cluster of differentiation 10-positive (CD10+), RCC-positive, creatine kinase-positive (CK+), vimentin-positive, hepatocyte-negative, and thyroid transcription factor 1-negative (TTF-1−) (Fig. ).\nAfter surgery, our patient was seen in regular follow-up. One year later, our patient came back for a routine checkup, and CT showed recurrence in the pancreatic head. On the basis of our patient’s condition, our group offered him a palliative treatment plan, which is tyrosine kinase inhibitor (TKI) therapy. He refused any further treatment. The timeline of our patient’s case is listed in Table . |
A 21-year-old man who had previously experienced no pain in the legs, no muscle weakness, and no other disorders, developed mild pain in the right lower leg while playing baseball, but he was able to continue to playing. Three days later, he became aware of drop foot of the right leg, but did not seek medical care because he could tolerate the pain. Two days later, he presented to the orthopedic department complaining of persistent drop foot of the right leg. The initial clinical examination revealed mild swelling of the anterior and lateral right lower leg, with focal prominence over the lateral muscle compartment, as well as pain and tenderness. No pain was evident with passive plantar flexion of the ankle, and plantar flexion of the digits was elicited. Manual muscle testing revealed 0/5 muscle strength of the anterior muscle group (tibialis anterior and extensor hallucis longus), 5/5 muscle strength of the posterior muscle group, and 0/5 muscle strength of the peroneus muscle. The posterior tibial and dorsalis pedis artery pulses were both palpable. There was decreased sensation in the deep peroneal nerve area, but sensation was normal in the superficial peroneal nerve area. Lumbar diseases such as disc herniation were excluded because patella tendon and Achilles tendon reflexes were normal, and the straight leg raising test yielded negative results. Magnetic resonance imaging (MRI) of the right lower leg was performed because of swelling of the anterior and lateral lower right leg. MRI demonstrated hypointensity on T1-weighted imaging and hyperintensity on T2-weighted imaging that appeared to represent a cystic lesion with fluid–fluid level findings in the lateral compartment (Fig. a, b), and pressures in the anterior and lateral compartments were 42 and 120 mmHg, respectively. Based on these clinical findings, fasciotomy was performed based on a definitive diagnosis of anterior and lateral compartment syndromes.\nThe proximal lower leg was exposed through a longitudinal incision above the lateral compartment. Hematoma within the lateral compartment was evacuated. The tendon of the peroneus longus was found to be completely detached from its proximal origin. Although the hematoma was evacuated, lateral compartment pressure remained elevated (120 mmHg). The distal lower leg was exposed through an incision above the lateral compartment. The peroneus muscle was ischemic and swollen, but not necrotic. The skin was closed, because the skin was not tense. Reduced lateral compartment pressure was confirmed, and the operation was finished. The day after the operation, the patient complained of right lower leg pain. The wound was opened because lateral compartment pressure was again increased (120 mmHg). After the wound was opened, the patient noted pain relief.\nFourteen days later, he was taken back to the operating room for delayed primary closure. At the time of primary closure, tibialis anterior strength had recovered to 3/5, and extensor hallucis longus and peroneus strengths were 1/5. The patient was discharged 18 days after fasciotomy, requiring an ankle–foot orthosis for ambulation. Three months after fasciotomy, he was able to return to play baseball with almost complete recovery of muscle strength in the tibialis anterior (5/5) and extensor hallucis longus/peroneus (4/5). Clinical examination after 2 years revealed no hypesthesia and no muscle weakness in the territory of the deep peroneal nerve. |
A 19 year old man presented to the Accident and Emergency Department with a one hour history of the right upper eyelid injury from a wire fence, (figure ). The patient was walking across an allotment when he fell onto a damaged fence. The loose end of the wire penetrated the full thickness of the right upper eyelid. The patient was unable to extricate himself, requiring the Fire Brigade to cut him free. Of relevant past history, there was an injury to the same eyelid from a coat hanger two years earlier.\nUpon gentle examination with no external pressure, the patient was unable to open the eye himself. The wire passed from under the eyelid, through the centre of the upper lid, to the external surface. Approximately 15 mm of wire was superficial to the lid margin; the cut end was approximately 90 mm and taped to the cheek for security.\nThe patient had eaten ninety minutes previously so he was unfit for a general anesthetic. The decision was made to infiltrate with local anesthetic and remove the foreign body. This was complicated by the patient's inebriation and needle phobia.\n1% Lignocaine was infiltrated in to the upper lid, the lid averted, and the wire passed out through the defect, (figure and ). Post removal, the acuity increased to 6/9 and there was no intraocular penetration, (figure ). No abnormalities were detected in the anterior or posterior segments and intraocular pressure was within the normal range.\nAfter the application of topical anesthetic drops, the eye could be opened manually. Acuity in the right was counting fingers. The anterior chamber was formed and there were no pupil abnormalities. It was difficult to assess whether there was any intraocular penetration. In addition, the injured region was examined for any remains or other possible foreign bodies.\nAt follow up the next day acuity was still maintained and one week later, after a full course of antibiotics, examination was unremarkable with equal acuity bilateral |
A 19-year-old girl presented to our outpatient department with a swelling of the tongue of 1-year duration, which was initially slowly growing but later started progressing rapidly in the past 3 months. She was complaining of inability in the movement of the tongue leading to difficulty in speaking and chewing of food. This also resulted in nasal regurgitation of food. She was not suffering from any co-morbid conditions.\nOn examination, her general health was fair (Eastern Cooperative Oncology Group performance status was 1). On intra-oral examination, there was a lobulated swelling involving the entire tongue with an intervening ulcer and there was restriction of the tongue movement. Multiple bilateral level 1A and 1B nodes of varying sizes were palpable and which were fixed to the underlying structures. The largest node was around 3 cm in size, which was on the left side (clinical staging was T4bN2cM0).\nExamination of the skin, abdomen, respiratory system, cardio vascular and the central nervous systems were found to be normal. Magnetic resonance imaging (MRI) of the tongue showed an enhancing mass of more than 5 × 4 cm in its greatest dimension. The mass involved both sides of intrinsic as well as extrinsic muscles with loss of definition of lingual septum []. Extension of the mass was seen into the right-sided sublingual space with encasement of the neurovascular bundle which was also extending anteriorly to the tip of tongue. There was no bony erosion. No extensions were seen into the carotid and bucco-masticator spaces. Magnetic resonance imaging of the neck showed metastatic cervical lymphadenopathy at the level I, II, III, IV on the left and level II on the right []. Clinically, the diagnosis of the lobulated swelling with an ulcer on the tongue, was that of an infiltrative epithelial malignancy or a minor salivary gland malignancy. Limitation of punch biopsy in this case was the presence of a limited intervening area with ulceration on the tongue swelling, which may not be representative of the presence of definite malignant cells. However, punch biopsy from the ulcerative lesion showed solid sheets of moderately pleomorphic tumor cells with round to oval, central to eccentric nuclei and eosinophilic cytoplasm suggestive of undifferentiated malignant neoplasm with features of rhabdoid differentiation []. Immunohistochemistry (IHC) analysis was done to detect the expression of vimentin, desmin, pan cytokeratin (CK) and S-100 protein. Immunohistochemistry was positive for vimentin [] but negative for the expression of desmin, pan CK and S-100. The diagnosis of MRT was established after histopathological examination and IHC analysis. In this case, the patient was not considered for upfront surgery in view of the growth involving the whole of the tongue extending up to the vallecula; hence, there would be an absence of clear margins for resection. The patient was treated with six cycles of palliative chemotherapy with doxorubicine (50 mg/m2, D1) and ifosphamide (1500 mg/m2, D1-D5). Palliative chemotherapy was followed by palliative external beam radiotherapy (EBRT). There was no response to the treatment after completion of chemo-radiotherapy and the patient expired after 3 years from the date of diagnosis. |
A 71-year-old male attended haematuria clinic in 2017 with a short history of painless visible haematuria that first occurred on holiday in Vietnam. He has a background of a skin melanoma of the right leg which was completely excised. An ultrasound scan of the bladder and flexible cystoscopy was performed which showed a large bladder tumour on the left bladder wall. A staging computed tomography (CT) scan of the thorax, abdomen, and pelvis was negative for metastases, only the bladder tumour was observed (). A transurethral resection of the bladder tumour was performed which showed muscle invasive disease. He subsequently underwent 3 cycles of neoadjuvant chemotherapy (gemcitabine and cisplatin), followed by a radical cystoprostatectomy and ileal conduit urinary diversion. Histological analysis confirmed a high grade muscle invasive urothelial carcinoma of the bladder with perineural invasion (pT3aN0). Three months after his surgery, he attended a follow-up clinic complaining of a painful right hip and a swelling in his right thigh worse on flexion at the hip. There were no skin changes or neurovascular compromise to the limb. Clinical examination revealed a diffuse swelling in the lateral compartment of the thigh. An ultrasound of the right thigh revealed a 43 mm × 31 mm hypoechoic, well circumscribed lesion within the fibres of vastus lateralis (). A subsequent contrast enhanced magnetic resonance imaging (MRI) scan showed a 37 × 36 × 48 mm lesion of altered signal intensity with limited central enhancement, suspicious of a skeletal muscle metastasis (). He also developed bone metastases at the L3 vertebra and right pubic bone. An ultrasound guided biopsy of the right thigh lesion was identified as a poorly differentiated carcinoma with positivity for pankeratin and cystokeratin 7 consistent with a urothelial carcinoma and confirming the diagnosis of skeletal muscle metastasis. He was referred to oncology and underwent 4 cycles of anti-PD-L1 monoclonal antibody immunotherapy (IV atezolizumab 1200 mg one dose per cycle) as part of a clinical trial. Despite better pain control and reduced right thigh swelling, follow up imaging suggested a slight increase in the size of the metastatic deposit in the thigh. He subsequently received a course of palliative radiotherapy to the lumbar spine, pelvis, and right thigh (20 Gy in 5 fractions at each site) followed by six 14 day cycles of IV paclitaxel chemotherapy (175 mg/m2/day). The patient demonstrated partial remission as the skeletal muscle metastasis reduced in size to 22 × 34 × 52 mm (including an area of tumour necrosis) assessed by a repeat MRI scan in March 2018. However, the scan identified a new 53 × 40 × 18 mm subcutaneous UC metastasis over his left scapula which was fully excised, and the site was irradiated (20 Gy in 5 fractions). The patient continues to make good progress and he is mobile and self-caring. His disease to date has remained stable on follow up imaging and he is currently under CT surveillance. |
An 11-year-old girl presented to our A&E department after a fall on an outstretched arm with immediate pain experienced at the right shoulder. The X-ray demonstrated a nondisplaced fracture of the middle to proximal third of the right clavicle (). There were no other injuries, and neurovascular examination of the upper extremity was normal. The fracture was managed conservatively with sling immobilization for 6 weeks, and on clinical follow-up, it appeared to be healing well. The patient had no pain nor functional limitation, with adequate progression of periosteal reossification and callus formation seen on radiological examination. The patient was an amateur junior javelin thrower and was restricted from all sporting activity for a total of 3 months and until there was radiographic confirmation of callus formation ().\nThe patient remained completely asymptomatic for 12 months, after which time she presented with new swelling at the callus midline, associated pain, and shoulder function loss, in the absence of additional trauma. The X-ray at 12 months confirmed the clinical suspicion of a pseudoarthrosis ().\nFollowing a multidisciplinary discussion, we decided to investigate the suspected nonunion and need for surgical intervention using CT and MRI (as seen below). The initial topographic examination was important to validate the pseudoarthrosis, which clearly identified two areas of hypertrophy without signs of bridging. The MRI was useful in excluding an entrapment of surrounding articular structures and in evaluating the involvement of the neurovascular bundle with respect to the bone ().\nFollowing a discussion with the patient's parents, we opted for a surgical approach with open reduction and internal fixation and iliac crest bone graft from the ipsilateral hip 3 × 3 × 1 cm. The nonunion was confirmed intraoperatively with debriding of the fracture site, with a loss of bone substance of 2 cm. The bone sample taken from the iliac crest was customised, molded, and inserted into the remaining gap. Following bone tissue transplant, the clavicle was reduced and internal fixation performed with a 3.5 mm seven-hole locking compression plate (LCP) (Synthes) in compression ().\nFollowing a normal postoperative course, clinical and radiological follow-up showed complete isometric anatomical healing at 1 year from the operation (). The clinical examination showed no pain during shoulder movement, with a range of motion in antepulsion of 180°, abduction of 180°, internal rotation up to X thoracic vertebra, and external rotation of 80° (). |
A 39-year-old woman at 38 weeks of gestation received elective cesarean section at an obstetrics clinic. Her medical history included hypertension and no remarkable complications during pregnancy. Electrocardiography revealed no abnormality, such as QT elongation. Under spinal anesthesia with bupivacaine, the patient underwent surgery, and perioperative blood loss volume was approximately 300 ml. After delivery, gradual onset of bradycardia, loss of spontaneous respiration, and impalpable carotid pulsation were observed. Thus, CPR was immediately initiated. The initial electrocardiogram revealed ventricular fibrillation. Subsequently, tracheal intubation was carried out. The patient did not recover despite direct cardioversion and adrenaline administration. For further management and treatment, she was transferred to our institute while CPR was continued. The time from the onset of the CA to the departure from the clinic was 28 min. When the patient arrived at our institute, she did not recover despite continued CPR. Therefore, venoarterial extracorporeal membrane oxygenation (ECMO) through the right femoral vein and left femoral artery was started. The time from collapse to ECMO induction was 63 min (Fig. ). Eighty minutes after ECMO induction, return of spontaneous circulation was achieved. Although coronary angiography and computed tomography were carried out, we could not detect the cause of the CA. Because of postoperative bleeding, targeted temperature management for normothermia was administered by ECMO. In spite of the disseminated intravascular coagulation on admission, early replacement of the coagulation factors with blood products prevented hemorrhagic events (Fig. ). Levels of serum cardiac enzymes, such as creatinine kinase, were normal. The patient’s circulation improved over time, and ECMO was terminated on day 3 of hospitalization. Tracheal tube extubation was carried out on day 6. Although the head computed tomography scan on day 8 revealed a slightly hypoxic encephalopathy, the patient’s consciousness disturbance recovered gradually. She was transferred to a rehabilitation hospital 14 days after admission. Her cerebral performance category score was 3 at transfer. After 3 months of rehabilitation following discharge, cerebral function improved so that she could perform independent activities of daily life (cerebral performance category score: 2). While the patient was hospitalized, we tentatively diagnosed amniotic fluid embolism (AFE) as the cause of CA. However, zinc coproporphyrin I and sialyl-Tn antigen, the serum markers of AFE, were not found; therefore, anesthesia might have caused CA.\nAlthough a definitive diagnosis could not be established, clinical AFE, according to the Japanese criteria, could not be ruled out because the pathological evaluation of the uterus was not possible. |
We report the case of a 27-year-old Tunisian man who presented with dyspnea (New York Heart Association (NYHA) functional class II) associated with typical angina. He has a medical history of ventricular septal defect associated with pulmonary stenosis diagnosed at 15 years old, with surgical treatment refused by his parents and with interrupted follow-up since then. Currently, a physical examination was notable for a grade 3/6 systolic murmur over the second left intercostal space without signs of heart failure or cyanosis. His electrocardiogram showed sinus rhythm with incomplete right bundle branch block. His chest X-ray showed cardiomegaly and dilatation of his left pulmonary artery. Transthoracic echocardiography showed normal left ventricular systolic function without wall motion abnormalities. The right ventricle was hypertrophied, dilated but with normal contractility. Severe pulmonary valvular stenosis was also visualized. An exploration of the ventricular septum showed a closed ventricular septal defect without evidence of shunt, related to a spontaneous closure of the defect. Cardiac catheterization confirmed a tight valvular pulmonary stenosis with systolic right ventricular and pulmonary arterial pressures at 103 and 14mmHg respectively, resulting in a 95mmHg systolic gradient between the right ventricle and pulmonary artery. Selective pulmonary angiography showed a severe valvular pulmonary stenosis and a left pulmonary artery stenosis with a poststenotic aneurysm. Left ventricle angiography showed a 12mm ventricular septal defect completely closed and an aneurismal left coronary artery. Selective coronary angiography revealed a dilated left coronary artery and 3mm coronary fistula arising from the mid segment of the left anterior descending artery (LAD) to the right ventricle (RV) with occlusion of the distal LAD (Figure ). Percutaneous treatment of both lesions was decided starting with the CAF to prevent acute myocardial ischemia that may appear after acute coronary steal consequent to the decrease of the RV pressure after relief of the pulmonary obstruction. A successful transcatheter closure of the fistula using a 4/6 Amplatzer™ duct occluder II (ADO II) was achieved, with partial reopening of the fistula on the following day (Figure ). No electrocardiogram changes were observed during or after the procedure. Heparin therapy was started with 150mg aspirin and 75mg clopidogrel to prevent thrombosis of the occluded vessel and discontinued after evidence of partial reopening of the fistula on systematic echocardiography control performed 24 hours after the procedure.\nSerial electrocardiograms documented normal tracings without evidence of ischemia. Echocardiography showed normal left ventricular function with no pericardial effusion, allowing discharge 48 hours after the procedure. Our patient was on 150mg aspirin per day. Complete closure of the fistula was documented nine months later (Figure ) allowing a successful percutaneous treatment of the pulmonary stenosis, with reduction of the pulmonary gradient from 100 to 35mmHg, using a 23/40mm balloon, with an uneventful recovery after treatment. |
A 52-year-old male patient visited the emergency department in our hospital due to sudden dizziness and dysphagia. The patient was diagnosed with lateral medullary infarction using brain magnetic resonance imaging. Three days after onset, he was referred to the department of rehabilitation medicine for the rehabilitative treatment of dysphagia. The patient had right blepharoptosis and no muscle weakness except for ataxia of the right lower extremity. He was being fed by a gastric feeding tube. Water swallowing test demonstrated a typical wet voice and laryngopharyngeal retention despite the small amount of food intake. VFSS was conducted to identify the causative factors of dysphagia 4 days after the onset. The functional dysphagia scale score was 60 points, with no particular abnormality in the anterosuperior elevation of the oral cavity and larynx, atresia of the epiglottis, definite contraction of the upper pharynx, and retention of almost all foods in the pyriformis sinus and vallecular fossa after swallowing, with no passage past the upper esophageal sphincter (). During the test, compensation techniques, such as Mendelsohn maneuver, head turn, and head lean, were also conducted, but no particular difference was observed. The patient underwent rehabilitative treatments that included electrical stimulation for dysphagia, Shaker exercise, and training concerning a compensation technique including the Mendelsohn maneuver. An assessment was conducted using esophageal manometry 32 days after onset to more accurately identify the cause of dysphagia. By setting the upper esophageal sphincter as a reference point, the pressure changes in the pharynx above the upper esophageal sphincter, upper esophageal sphincter, and esophageal body were measured. Normal pressure change in the upper pharynx, decreased contraction of the lower pharynx, definitely decreased pressure upon the relaxation of the upper esophageal sphincter, retention in the upper esophageal sphincter due to the impairment of the coordination with the contraction of the lower pharynx, and no pressure changes caused by esophageal peristalsis were evident (). About four weeks later (33 days after onset), VFSS was conducted again. The functional dysphagia scale score was again 60 points (). As the patient was refractory to the one-month dysphagia rehabilitative treatment including electrical treatment for dysphagia, Shaker exercise, and the training of compensation technique, another treatment was applied.\nAs several previous studies reported that dysphagia was improved by increasing the neurotransmission and plasticity of the corticobulbar tract via rTMS of the cerebral cortex associated with swallowing [,], rTMS was used. The International 10-20 system and a bandana with lattice positioned at intervals of 1 cm were used to determine the stimulation site. The surface electrode was used to determine the site with the lowest motor threshold of the pharyngeal-laryngeal muscles, which was identified as an area 2 cm anterior and 5 cm lateral from the vertex of the head. rTMS was conducted with a frequency of 5 Hz and intensity of 120% of the motor threshold of bilateral hemisphere, and 10 cycles of 10 seconds stimulation and 50 seconds relaxation for 10 days (five times/week for 2 weeks) followed by VFSS and manometry to assess the result of the 2-week TMS treatment. The functional dysphagia scale was improved to 20 points. Compared to the result of the preceding VFSS, which showed that most foods were retained in the lower pharynx because of the inability to passing by the upper esophageal sphincter, at the follow-up most foods were able to passed by the upper esophageal sphincter. A small quantity of food was retained in the pyriformis sinus and vallecular fossa sinus after swallowing, and the laryngeal delay time was significantly shortened (, ). Meanwhile, compared to previous manometry results, the follow-up revealed a prolonged relaxation time of the upper esophageal sphincter of 2 seconds, which was more than twice the time of the previous results. This would be sufficient enough to enable foods to pass by the upper esophageal sphincter after the contraction of the lower pharynx. The pressure change was observed in the esophageal phase due to peristalsis ().\nAfter 2 weeks of rTMS, the patient began to consume small amounts of food in accordance with the schedule set by the authors. Regular chest X-ray and blood test revealed no complications, such as aspiration pneumonia, were observed. When the patient was able to consume 1,650 calories per day, he was discharged. He has been followed-up in our out-patient clinic and has been taking a regular diet without any complication for 2 years. |
A 72-year-old male with a past medical history of hypertension, chronic kidney disease, a solitary functioning left kidney (right kidney atrophic from unknown cause), and protein C deficiency (PCD) with baseline creatinine of 4 mg/dL presented with flank pain, and upon evaluation he was found to have severe left hydroureteronephrosis () with an elevation of his creatinine to 12.2 mg/dL. He underwent a left ureteral stent placement. During the procedure, a small stone in the bladder was identified, presumably passed from the kidney, and it was removed. The stone was sent for analysis and its constituents were unable to be identified, even after repeat analysis.\nHis renal function and hydronephrosis improved and the stent was removed 9 weeks later, assuming the issue of obstruction was resolved. Two months later, the patient presented with hematuria and flank pain and laboratory evaluation again revealed worsened renal function. A CT scan showed recurrent severe left hydroureteronephrosis with no definite cause of ureteral obstruction. Upon placement of the ureteral stent, a large efflux of bloody urine was noted from the stent.\nAfter his renal function returned to baseline, a diagnostic ureteroscopy was performed to assess for presence of ureteral stricture, mass, or an obstructing lesion as well as to remove any nonobstructing calculi within the collecting system. Ureteroscopy and pyeloscopy revealed a significant amount of debris and clot within the collecting system. Papillary necrosis was noted throughout the collecting system, especially notable in the central and lower calices. Floating necrotic renal papillary tissue was also observed in the lower pole of the kidney. There were no stones identified, only small blood clots present in the areas of missing papillae where there was a crater in the place where a papilla was expected. The kidney was irrigated free of all clots and debris. The necrotic material was removed with biopsy forceps to be sent for pathology analysis. Pathologic evaluation was consistent with necrotic renal papillae without any signs of invasion with cancer or infection (). The patient's protein C activity was measured to be 44 IU/dL, which is below the normal reference range (75–133 IU/dL) and considered to be mildly deficient. A ureteral stent was placed at the time of ureteropyeloscopy and removed 2 weeks later. At 6-month follow-up no recurrence was noted.\nTo our knowledge, this is the first case reported in the literature illustrating a unique presentation of renal papillary necrosis (RPN) in the setting of PCD and in the absence of a locally invasive infection or malignancy. |
The patient was a 54-year-old female with chronic severe right anterior and lateral leg pain with radiation above the knee and mild low back pain. Her pain score was 8/10 in the numeric rating scale (NRS). Prior to visiting our clinic, she had been diagnosed with multilevel lower lumbar degenerative changes, including a bulging disc and central canal stenosis at L3-4 and L4-5 on lumbar MRI. She received fluoroscopic-guided transforaminal epidural blocks repeatedly at the local spine center. However, the spinal blocks were not effective for her anterior thigh pain, and so lumbar spine surgery was planned. However, she refused to have lumbar surgery and visited our pain clinic. In her history, she was an officer and worked seated for long periods. She usually crossed her legs while sitting until the pain got worse. Although the patient could not remember the duration of the disease, she said that there was intermittent pain for about 25 years since childbirth and the pain had gradually exacerbated recently. There was no remarkable medical history or antecedent trauma. The patient continued to walk with a limp, which worsened with walking up a steep incline or climbing stairs. Her pain also was aggravated by movement and walking. Physical examination revealed mild tenderness over the right groin area. The patient was unable to fully extend her right hip joint to neutral in the supine position because it reproduced anterior thigh and low back pain and a sensation of tightness. The leg was not completely straightened. The Thomas test of the right side was positive. Her right hip joint showed a flexion contracture of about 15° due to limited range of motion in the supine position. The patient stated that she always slept with a pillow under her right knee or on the side. The Patrick and Ganslene test results were also positive on the right leg. The straight leg raising test was 80/80. Neurologic examinations revealed weakness of the hip flexor (grade 4/5 on the right side, grade 5/5 on the left side based on the MRC scale). We identified a weakness in the right hip flexors compared to the contralateral leg based on a resisted flexion test of the hip joint. No sensory changes were detected using pinprick and light touch sensation in the anterior and lateral aspects of the lower leg. No reflex abnormality of the legs was detected.\nOur clinical suspicion was that her right thigh pain was caused by other problems. Therefore, we evaluated her lumbar MRI taken at the local spine center, and were able to identify severe atrophy of the right psoas muscle besides degenerative spine ( and ). A plain radiograph of the hip joint was evaluated additionally, and right hip joint osteoarthritis with narrowing of the joint space was identified (). We performed ultrasound-guided intramuscular injection of the right psoas muscle with 10 ccs of 0.25% lidocaine for muscle tightness and nerve entrapment. Her pain improved on NRS 5~6/10, but she still complained of discomfort. Then, we performed ultrasound-guided, diagnostic, intra-articular injection of the right hip with 5 ccs of 0.5% lidocaine mixed with 10 mg of triamcinolone. At the time of a follow-up visit after one week, the patient reported that the pain level was 3/10 on NRS and that she had improved function, enabling her to straighten her leg. However, the flexion contracture of the right hip joint was not resolved completely. After two weeks, the anterior and lateral leg pain aggravated gradually, so the patient was referred to the Department of Hip Orthopedic Surgery for the evaluation of hip surgery. Informed consent for the publication was approved by the patient. |
A 42-year-old male patient had applied to our clinic with a claim of pain in his penis. It is understood according to patient's history that he had a multiple heated Vaseline injection done under penis skin one month ago for augmenting his penis. In patient's physical examination, it was palpated that there were soft-formed fixed and widespread mass lesions under penis skin. No deformation was found on penis skin surface (). Patient stated that he had normal erection but he felt pain during sexual relation. In preoperative examinations done on the patient, no biochemical or hematological disorder was detected.\nIn patient's superficial tissue ultrasonography, a thickening nearly 10 mm and obvious eco increase at extratunical level on penis root and penile shaft proximal section's dorsal side and tissue field with similar appearance on penile shaft distal section's ventral side were reported. No pathology was detected on both corpus cavernosum and corpus spongiosum.\nIt was decided to make an operation on the patient in order to pull out the foreign substance. Degloving was done to penis with circumcision incision after intraoperative urethral catheter was inserted and it was seen that tunica albugiane was intact. However, vaseline was spreaded among the subcutaneous layers to the highest degree and caused cohesion and granulomatous reaction. Vaseline was pulled out by being separated from tissues through dissection and necrotic fields which were excited. Layers were enclosed with anatomic plan after all foreign substances were pulled out and penis was bandaged up with an elastic bandage. On second postoperative day, the patient whose bandage was opened had been discharged from hospital as his wound appearance was seen better, with antibiotics and anti-inflammatory medication therapy. It was seen that penis skin was going under necrosis on the 7th postoperative day during patient followup and patient's consultation is done at plastic surgery clinic. Patient again hospitalized by plastic surgery clinic, was operated again as circular necrosis was reformed on penis skin during patient follow-up process, and defect repair was done with split thickness skin graft prepared from right-anterior femoral skin. |
A 10-year-old boy reported to the dental clinic with the chief complaint of large tooth in the maxillary anterior region of the jaw. No other member of the family was affected with similar dental anomaly and patient was mentally challenged. Intraoral examination revealed that the patient was in mixed dentition phase along with fusion of the maxillary right primary central incisor and primary lateral incisor with an additional (supernumerary) tooth []. Dental caries was evident on the incisal edge and proximal surface of supernumerary tooth. The child's dentition revealed Angle's class 1 molar relation on both right and left side. Maxillary right permanent central incisor was erupted, but it was in cross-bite relationship. A mid line diastema was also noted in maxilla. Clinical examination revealed missing right permanent lateral incisor and there was no history of previous extraction, of this tooth. Further investigation included intraoral periapical radiograph of maxillary anterior region []. Based on clinical and radiographical findings, the provisional diagnosis of fused triple teeth was made.\nThe treatment plan was aimed at removal of fused teeth. Under sedation, fused teeth were carefully extracted []. Macroscopically, root resorption was evident on apical portion of maxillary right primary lateral incisor. For histological examination, the extracted teeth were sectioned at three levels: The coronal, middle root and apical levels, respectively. These sections were visualized under a stereomicroscope at a magnification of ×50. Histological view of the coronal section revealed three teeth fused with each other with confluent dentin without intervening cementum and with separate pulp chambers []. The middle-third of the roots of three fused teeth also showed confluent dentin without intervening cementum []. The apical third of fused teeth showed merging of root canals of the fused teeth []. Based on these findings, a final diagnosis of incomplete fusion was confirmed.\nFurther treatment plan involved thorough oral prophylaxis and correction of anterior cross- bite. After oral prophylaxis and pit and fissure sealant application, a removable Hawley's appliance with Z-spring was fabricated for the correction of the anterior tooth cross-bite, but as patient was mentally challenged, cooperation from him was poor as the child was not ready to wear the appliance. Again Catalan's appliance was made and cemented on the lower incisors, but the attempt for the correction of cross- bite failed because of insufficient compliance of the patient as he was not occluding his teeth. |
A forty nine-year-old female presented to the emergency department due to episodes of sudden cramping, epigastric pain and nausea 2 months following endoscopic retrograde cholangiopancreatography (ERCP) where three gallstones were removed from the common bile ducts (). Before she underwent ERCP and endoscopic sphincterotomy, right upper quadrant abdominal ultrasound done on account of obstructive jaundice showed a noninflammed gallbladder with a large stone and a dilated common bile duct (). Her past medical history was also notable for severe aortic stenosis, pulmonary hypertension, diabetes, unstable angina, and morbid obesity with BMI of 60 kg/m2. Given her comorbidities, the plan during her ERCP visit was to perform her cholecystectomy at the same time she will undergo her gastric bypass, after optimizing her cardiopulmonary disease. At presentation, her abdominal computed tomography (CT) revealed evidence of small bowel obstruction with clear evidence of gallstone ileus (). A cholecystoduodenal fistula was also present (). Evidence of a decompressed gallbladder () and a clear transition point were noted. She was taken to the OR for laparoscopic exploration and small bowel examination. The segment that had the stone was identified and exteriorized through a small midline incision; the bowel was necrotic due to pressure ischemia; thus segmental bowel resection was performed with stapled anastomosis (). A large, calcified 3.5 cm × 3 cm gallstone was found in the small bowel impacted at 60 cm proximal to the ileocecal valve () with evidence of pressure necrosis to the bowel. The bowel was examined via laparoscopy from the Ligament of Treitz to the ileocecal valve with no evidence of additional stones. Patient recovered appropriately after surgery and was discharged home.\nA month later, she presented with signs and symptoms of small bowel obstruction. On physical exam, her abdomen was distended but with no tenderness. Patient was also noted to be in septic shock requiring vasopressors and unable to tolerate surgery; she was treated conservatively with Gastrografin challenge test (50 ml of water mixed with 100 ml of Gastrografin and given through the nasogastric tube, followed by abdominal X-ray in eight hours), which revealed contrast in the colon. Her symptoms completely resolved; she tolerated oral intake and was discharged from the hospital.\nPatient presented again one month later with similar symptoms. At this presentation, she had an abdominal CT scan, which showed dilated bowel with no clear transition point. She was treated conservatively for five days with mild improvement where she tolerated liquids but not solid food. During the same hospital stay, she developed sudden severe abdominal pain, shortness of breath, tachycardia, and hypoxia. Another CT scan was performed with pulmonary embolism (PE) protocol which was negative for PE, but positive for free air (). Patient was taken back to the OR for laparotomy, which revealed a second large non-calcified gallstone in the ileum impacted at the ileocecal valve distal to the previous anastomosis and perforated necrotic small bowel with a pocket of abscess. An intraoperative diagnosis of recurrent GSI was made. The abdomen was washed out; bowel resected; but anastomosis could not be performed given the severe inflammation. Patient was brought back to the OR for washout and closure; again, bowel anastomosis could not be performed, nor could the bowel be brought out as an end ileostomy due to severe obesity and shortened thickened mesentery. The bowel was placed in the upper part of the midline incision and the wound closed around it. Patient recovered appropriately and was discharged home. Subsequently, patient underwent elective surgery for cholecystectomy, takedown and primary repair of cholecystoduodenal fistula, bile duct exploration, suture repair of extra hepatic bile duct, and takedown of ileostomy with primary anastomosis. |
A 49-year-old female presented with a nail deformity of the left thumb which had been present for many years. With the increasing severity of the deformity, the patient had been experiencing increased pain severity. The patient was otherwise healthy, and had no family history of a similar lesion. Upon physical examination, a dystrophic left thumb nail was noted with prominent right axial curvature that caused compression of the distal portion of the thumb (). Radiographic examination showed no evidence of osteophyte formation at the base of the distal phalanx, and fungal nail infection was excluded by negative results of microscopic examination (KOH test) and fungal culture.\nInitially, two percent lidocaine without epinephrine was administered via the standard technique to achieve digital block and wing block at the left thumb. The thumb was wrapped with a surgical glove and the glove-finger was released from the finger tip down to the proximal part of the finger and fixed by a mosquito clamp to provide a finger tourniquet. The deformed nail plate was removed from the underlying nail bed by the rolling method. An oblique incision was made distal to the hyponychium in line with the most lateral aspect of the nail bed (), and a tunnel for the graft was created between the nail bed and the underlying phalanx with a periosteal elevator (). The dermal graft was harvested under local anesthesia from the thenar area of the left hand. The graft length ranged from the hyponychium to the paronychial fold and was at least one centimeter in width. The epidermis and subcutaneous fat were removed by fine scissors, and the donor site was finalized by primary closure. The graft was put into the hyponychial incision and drawn proximally in the tunnel more than one centimeter away from the nail matrix by passing the graft (distal to proximal) through the tunnel by nylon 4-0 sutured to the distal end of graft without incising the paronychial nail fold (). When the graft elevated the paronychial fold, it sutured the skin proximally to hold it within the tunnel. The excess portion of the graft was excised distally and the hyponychial incision was closed with nylon 4-0 (). are post-surgical results of the patient after 6 weeks, 5 months respectively. The patient has been doing well, and there was no relapse since in the 12 months following the procedure. Schematic surgical procedures have been provided in . |
A 41-year-old woman was diagnosed with complex regional pain syndrome (CRPS) using the "Budapest" criteria [] due to lower extremity pain after thigh contusion caused by dropping a heavy box on her thigh at work 28 months ago. She was admitted for a trial spinal cord stimulation (SCS) for treating her lower extremity pain that was resistant to multimodal pain treatments for 28 months after pain onset. Results of her blood tests including coagulation study were within normal ranges at the preoperative evaluation.\nDuring her first visit to our clinic, she complained of lower extremity pain with pain sites gradually extending to her upper extremities. She underwent surgery for a trial SCS at the lower thoracic vertebral segment to alleviate her lower extremity pain, and her lower extremity pain score decreased moderately from 9 to 5 on the visual analog scale (VAS). However, on the 3rd day of the trial SCS she was still suffering from upper extremity pain and her upper extremity pain score was increased from 7 to 9 on the VAS despite oral analgesics and intermittent intravenous opioid rescue medication. Cervical epidural catheterization was planned for continuous administration of analgesics to manage her upper extremity pain.\nThe patient underwent cervical epidural catheterization with a 20-gauge catheter through an 18-gauge Tuohy needle at C7-T1 with LOR using air under fluoroscopic guidance. Total amount of air instilled into the epidural space was uncertain; however, the practitioner (4th grade of anesthesiology residency) recalled administering about 7 ml of air.\nShe was comfortable and able to ambulate in the in-patient ward after cervical epidural catheterization. Six hours after insertion of the cervical epidural catheter, she suddenly complained of hypoesthesia and motor weakness of both lower extremities. Neurologic assessment revealed markedly decreased sensation to pinprick, cold, and touch below the T10 dermatome and slight contraction of the leg and foot muscles without movement (grade 1/5). The authors suspected an epidural hematoma around the thoracic spine possibly related to the previous epidural procedures and considered whole spine magnetic resonance imaging (MRI) although she did not have typical pain accompanying an epidural hematoma. However, she had received a trial SCS lead and underwent whole spine CT. Definitive findings of whole spine CT scan were multifocal air densities in the epidural space, intervertebral foramen, and paraspinal muscles between C1 and T9 and no obvious spinal cord compression (). While evaluating the patient and performing CT scan within 1 hour, the sensation below the T10 dermatome was totally absent and muscle power of both lower extremities was zero with no contraction (0/5).\nWe consulted a neurosurgeon for emergent decompressive surgery although there was no obvious spinal cord compression due to a mass-like lesion such as epidural hematoma or abscess. Within 3 hours after the paraplegia developed, decompressive operation was started by targeting the insertion site of the trial SCS electrode at the lower thoracic spine first because the neurologic deficit developed below the T10 level. However, there was no hematoma or abnormal findings in the epidural space where the trial SCS electrode was placed at T9–12. The radiologist and the authors thought that multifocal air collection in the epidural space and intervertebral foramen could be the cause of paraplegia, and a decompressive operation in the cervical spine was performed immediately after the closure of the thoracic spine. As soon as the cervical epidural space was opened, the neurosurgeon noticed escape of air bubbles with the naked eye and there was no hematoma or any other lesion.\nImmediately after the decompressive surgery, the patient underwent whole spine MRI for evaluation of the spinal cord and epidural space. There were no findings of spinal cord ischemia on the whole spine MRI. Her sensation and motor function recovered to normal immediately after she awoke in the post-anesthesia care unit. The following day, whole spine CT was performed and it revealed nearly complete disappearance of epidural and paravertebral air (). One month after this incident, she underwent SCS for the management of pain in the lower extremities. |
A 13-year-old boy was referred to our tertiary young adult hip service by a Paediatric Orthopaedic Surgeon for evaluation of left groin pain and a decreased range of movement in the left hip. He was a keen rugby player and had experienced an avulsion fracture of the AIIS during a rugby game which was played without a warm up six months previously. MRI just after injury showed a single bony fragment measuring 12 mm × 4 mm × 12 mm at the rectus femoris origin of the AIIS and it was retracted inferiorly 3 cm, anteriorly 1 cm and laterally 0.5 cm. Conservative treatment was advised by the Paediatric Orthopaedic Surgeon and he was referred to a physiotherapist for mobilisation and subsequently muscle strengthening.\nFive months post-injury he was progressing well with the physiotherapy, although, he still felt a sharp pain in his left groin when running with a ball while playing rugby. Flexion was restricted to 70° and a decrease in internal rotation in comparison with the opposite side was observed while abduction, adduction, extension and external rotation were comparable to the other side. There was no significant tenderness and no bruise in the region of the groin, and no distal neurovascular deficit. Plain radiographs demonstrated hypertrophic calcification in the region of the rectus femoris avulsion, whilst the hip joints were normal in appearance. A CT scan showed well corticated heterotopic bone formation at the site, measuring approximately 3.5 cm at maximum diameter (). Motion analysis using the three-dimensional CT datasets clearly showed the impinging area especially in 70° of hip flexion.\nBoth conservative and surgical management were suggested and discussed with the patient and his parents. They chose to go ahead with surgery, as he could not play rugby well because of the pain and restricted range of movement. The patient wanted to play rugby at a fairly high level, hopefully nationally, in the future. At arthroscopy, he was found to have a well corticated large lesion of heterotopic ossification, and it was dissected carefully from the muscles (). Once the dissection was completed, it was extracted via an incision to deliver the lesion. A dynamic impingement test was performed on the table to ensure there was no residual impingement and the impingement lesion was resected with a 5.5 mm arthroscopic burr. A thorough washout was carried out before closure.\nThe patient was advised not to perform any rotational activities in deep flexion for six weeks postoperatively and to follow the 16-week post-operative rehabilitation protocol. He was also advised prophylaxis against heterotopic ossification. Eight weeks following the procedure, the wound had healed well and there were no obvious signs of complications such as infection or deep vein thrombosis. Along with this, he demonstrated a pain-free, fully functional range of movement in his left hip joint and was delighted with his progress. Plain radiographs showed no further signs of calcification (). He was advised to continue to attend physiotherapy for at least another eight weeks and to engage in more gentle sporting activities like cricket and basketball, which he had enjoyed before the injury. At the 1-year follow-up, he remains asymptomatic with a full range of movement in his hip and continues to play rugby at a high level. |
A 42-year-old male came to our hospital with a complaint of defecation pain for 20 days and left lower abdomen pain for 3 days. Abdominal computed tomography showed thickening intestine wall at the recto-sigmoid junction, which leaded to an obvious dilatation of proximal intestine. And the density of intestinal mesentery increased as well as omentum (Fig. ). Through colonoscopy, we found an annular ulcer with irregular bottom and dirty surface near the recto-sigmoid junction, with pathological diagnosis of poorly differentiated adenocarcinoma. Without other obvious anomalies in the preoperative examinations, we performed an exploratory laparotomy on January 25. Massive serous ascites, a recto-sigmoid obstructive lump, and extensive nodules of implantation metastasis in abdomen, especially on the surfaces of intestinal tract and liver, were found during the operation. Considering the advanced stage of the tumor, we decided to perform a palliative Hartmann operation to relieve obstruction and adjuvant chemotherapy was chosen for following treatment. After operation, upper GI obstruction was well-relieved. But on February 10, 16 days after operation, the patient complained of discontinuous abdominal distention and pain again. Digital radiography of upper digestive tract showed a complete obstruction at the horizontal segment of duodenum, while the cavity was so narrow that the contrast agent could not pass through. We presumed that the obstruction was induced by the wide abdominal metastasis of the tumor. Conventional treatments for obstruction were performed, such as fasting, GI decompression, proton pump inhibitor, and parenteral nutrition. However, the intestinal obstruction was not relieved in the following days. Based on his situation, we assessed that the Eastern Cooperative Oncology Group Performance Status of the patient had already reached to 3 which was not suitable for high intensity of chemotherapy. So, targeted therapy was chosen as the further treatment instead of routine chemotherapy. By genetic testing, the wild type of KRAS gene had been found, for which cetuximab was very suitable. On February 27, an initial loading dose of cetuximab was given at 400 mg/m2; meanwhile, 250 mg/m2 was planned for following weeks. But on March 3, 4 days after the first course, the patient suddenly started spitting blood with the amount of 250 mL. We stopped the bleeding with thrombin, hemocoagulase, and somatostatin and suspended the subsequent treatment plan of cetuximab at the same time. On March 5, the patient threw up an 800-mL blood again while the diagnosis of UGIB had been made. Except for the previous general measures, antishock treatment was given immediately. After days of hemostasis and transfusion, the hematemesis was relieved gradually. The following endoscope revealed some old blood clot and an obvious narrowed cavity at the horizontal segment of duodenum, which mucosa was congestive and erosive seriously. Owing to the narrow cavity, endoscope could not pass through the horizontal segment of duodenum, leading to the failure in obtaining a clear image of overt bleeding. On March 14, when the condition was stable, we performed cetuximab treatment again. But a more severe hematemesis occurred immediately in just 40 h. And the patient was died of respiratory and circulatory failure caused by UGIB and advanced tumor on March 16.\nWe collected the blood coagulation spectrum and analyzed. However, no significant coagulation disorders were found after application of cetuximab. The changes of indexes related to coagulation during the course of treatment, including prothrombin time (PT), fibrinogen level, platelet level, and the d-dimer, were shown in Table . The scoring system of International Society of Thrombosis and Hemostasis (ISTH) was acknowledged to be appropriate for the diagnosis of disseminated intravascular coagulation (DIC). The total score of our patient changed from 2 to 3 points before and after the 2 application of cetuximab. They were all <5 points and did not meet the diagnosis standard of DIC. In addition, Rockall scoring system was constructed to assess the mortality of UGIB and risk of re-bleeding. In the scoring system, the risk of re-bleeding as well as death increased with the score mounting. Retrospectively using the Rockall scoring system, we concluded that the patient for first massive bleeding after endoscope scored 0 points for his age (46 years), 1 points for the systolic BP (108 mm Hg) and the pulse (126/min) recorded on May 5, 3 points for extensive abdominal metastasis found in the process of surgical exploration, 2 points for malignancy of upper GI tract diagnosed by endoscope on May 6, and 2 points for blood in upper GI tract detected by endoscope on March 6. The total score of our patient was 8 points which suggested the high-risk stage (Rockall score exceeds 5 points) with the highest risk of re-bleeding and death. |
A 53-year-old male patient presented to our institute with complaints of right flank pain of one year duration. General physical examination was unremarkable. On per abdomen examination, a vague lump was palpable in umbilical and hypogastric quadrant. USG abdomen revealed an ectopic right pelvic kidney with gross hydronephrosis with thinned out renal cortex. Routine hematological and biochemistry profile was normal. Renal dynamic scan reported nonfunctioning right kidney. To know the renal vascular anatomy, CECT abdomen with CT angiography was done which revealed grossly hydronephrotic right ectopic kidney placed in the pelvis with thinned out cortex (). There was single left renal artery arising from abdominal aorta at L1 level. The ectopic pelvic kidney was supplied by 2 arteries, one is from the abdominal aorta just proximal to its bifurcation and the other one is from the left common iliac artery. The patient underwent laparoscopic nephrectomy by SIMPLE technique and was operated under general anesthesia. Cystoscopy was done initially which showed bilateral ureteric orifice in normal position and bilateral ureteric catheters were placed. The right ureteric catheter was crossing the midline on left side and was placed in the renal pelvis of the right pelvic kidney. The patient was positioned in supine trendelenburg position. The patient was adequately supported and strapped, and all the pressure points were protected. Veress needle was used to create pneumoperitoneum. 2.5 cm incision was made in the umbilicus. We used conventional laparoscopic instruments during the surgery. A 10 mm port was inserted at the umbilicus, and the other 10 mm and 5 mm ports were placed adjacent to that in the same incision (). Posterior peritoneum over the pelvis was incised and right pelvic kidney was localized. The ureter was localized over the psoas muscle and dissected till pelviureteric junction. The renal pelvis was dilated which helped in dissection around the kidney (). Multiple vessels were present over the renal pelvis. The renal pelvis was decompressed with an externally placed needle. The renal vessels were dissected and isolated. The vessels were clipped and cut (). The kidney was mobilized all around and the final specimen was removed from the umbilical port site. Hemostasis was achieved and no drain was placed. The left ureteric catheter was left indwelling for drainage. The port site was closed with vicryl number 1 suture and skin staples were applied. Postoperative recovery was uneventful and per urethral catheter was removed on day 2, and patient was discharged on day 4. Final histopathology confirmed marked interstitial fibrosis, sclerosis, and tubular atrophy with absent glomerular structures suggestive of nonfunctioning kidney. |
A 44-year-old male presented with a mass on the right side of the neck known to have been stable for more than 10 years with a history of the increase in size from past 1 month associated with pain. There were no compressive symptoms and no history of flushing, cardiovascular disturbances, or diarrhea. Clinically, he was in euthyroid status. Past medical history was unremarkable. Physical examination revealed a nodule of 5 cm × 4 cm in the right lobe of thyroid gland with enlargement of level III cervical lymph nodes on the left side. Hematological, biochemical, and thyroid hormonal parameters were within normal limits. High-resolution ultrasonography of neck showed a 4.4 cm × 3.5 cm heterogeneous echo textured nodule right lobe thyroid with multiple calcific specks within. Left lobe also showed multiple small nodules with areas of coarse calcifications. Few lymph nodes noted along the left jugular vein largest measuring 1 cm × 0.7 cm, one of them showing calcific specks. Fine needle aspiration cytology (FNAC) performed showed features suggestive of anaplastic carcinoma with brownish-black pigment. A diagnosis of poorly differentiated malignancy with melanin pigment, possible malignant melanoma was made from the intraoperative frozen section of the tumor and hence a total thyroidectomy with removal of enlarged cervical lymph nodes was performed in one sitting. Gross findings showed enlargement of both lobes of thyroid with blackish discoloration of right lobe []. Microscopic examination of right lobe thyroid lobe revealed nodularity comprised of polygonal to spindle cells, showing organoid pattern with clumped chromatin, moderate to abundant cytoplasm, with areas of necrosis, focal hemorrhage, and abundant extracellular brownish black melanin pigment. The majority of tumor cells showed intracellular melanin deposition []. Sections from left lobe of thyroid showed features of medullary carcinoma of thyroid with amyloid production, without melanin pigment. Immunohistochemistry showed positivity with HMB45 in right lobe [] calcitonin in left lobe []. A final diagnosis of melanotic medullary carcinoma of thyroid with amyloid production was made. Excised lymph nodes showed metastatic medullary deposits.\nIn view of medullary carcinoma with melanin production, further work-up was done with serum calcitonin whole body iodine 131 meta iodo benzyl guanidine (I 131 MIBG) scan to outline any residual or metastatic foci with an intent to treat with high dose of I 131 MIBG. Serum calcitonin was 8 ng/L and I 131 MIBG scan did not reveal any residual or metastatic localization. Thus, due to nontherapeutic options with MIBG and known radio resistance to radiotherapy of melanotic medullary carcinoma thyroid,[] patient was kept on close follow-up. One year later, patient presented with diffuse boggy swelling in the right side of neck associated with severe pain radiating to the right ear, however, there was no puffiness of face, engorged veins on the neck or chest wall. Clinical examination revealed a firm diffuse fixed mass along the right jugular region. A F-18 FDG PET/CT was performed which revealed an intensely hypermetabolic hypo dense intraluminal filling defect with an standardized uptake value maximum of 16.61 in the internal jugular vein (IJV), external jugular vein (EJV), right subclavian vein confluencing at the right innominate vein with inferior extension into the superior vena cava (SVC) falling just short of the right atrium suggestive of tumoral thrombus. There was no residual thyroid in the thyroid bed, however, a small metabolically active pretracheal lymph node was noted []. There was no residual mass in the thyroid bed or elsewhere in the neck. There was no hematological evidence of any thrombotic or prothrombotic states. Serum calcitonin was elevated to 300 ng/L FNAC from the solitary pretracheal node revealed metastatic deposit of medullary carcinoma. With limited therapeutic options and impending cardiovascular catastrophe due to large tumor thrombus load, an immediate blunderbuss salvage radiotherapy was given, with F18 FDG PET/CT based IMRT. The field included the metabolically active mass in the IJV and SVC extending up to right atrium along with neck, sparing the adjoining trachea and carotid vessels to prevent carotid artery stenosis and blow out. A hypofractionated technique was employed delivering a cumulative dose of 50 Gy in 20 fractions instead of the conventional 28–25 fractions of 1.8–2 Gy/fractions. The patient tolerated the entire course without any hematological or vascular complications and became symptom-free by the end of the radiation treatment. Patient was kept on clinical follow-up and assessment with F18 FDG PET//CT done subsequently revealed complete metabolic regression in the intravascular tumor with significant reduction in the tumor thrombus and better contrast passage through the SVC []. Follow-up at 3 months patient continued to be symptom-free with complete disappearance of the boggy neck swelling. Color Doppler of the neck revealed significantly shrunken thrombus in the right IJV with flow void and normal flow through the left innominate vein []. A contrast CT chest showed non-enhancing hypodense filling defect reduced in extent with tiny calcifications in the right IJV, EJV, subclavian veins suggesting chronic bland venous thrombus []. Serum calcitonin levels came down to 10 ng/L. A patient being disease free is on replacement therapy with thyroxine and on follow-up. |
A 57 year old female patient visited the Department of Surgery, Incheon St. Mary's Hospital complaining of postprandial epigastric discomfort. An esophagogastroduodenoscopy (EGD) and abdominal computed tomography (CT) scan were performed. On EGD finding, a submucosal tumor was found at the posterior wall of the gastric antrum (), and it was revealed by CT scan to be a 10 ×8 cm sized heterogeneously enhanced, well circumscribed solid mass arising from the posterior wall of the gastric antrum (). There was no metastatic lesion in other organs. She was recommended an open resection, but insisted on laparoscopic resection. Therefore, she underwent laparoscopic resection after providing informed consent. Under general anesthesia, the patient was placed in the reverse Trendelenburg position with her legs apart. The operator was positioned on the right side, and the first assistant was positioned on the left side of the patient. The camera operator was positioned between the legs of the patient. A 10 mm trocar was inserted through the umbilicus for the camera. A 5 mm trocar and a 12 mm trocar on the right upper abdomen were used as working channels for the operator. Another 5 mm trocar and a 12 mm trocar on left upper abdomen were used for the first assistant. The greater omentum was opened with ultrasonic shears and the mass was identified. Great care was taken to avoid touching the mass directly with the laparoscopic instruments. There was no direct invasion to the pancreas (). The right gastroepiploic vessels were identified and doubly ligated with clips and the duodenum was resected with a laparoscopic linear stapler. The right gastric vessels and descending branches of the left gastric artery were identified and doubly ligated. The stomach was transected at 4 cm proximal from the upper border of the mass with a laparoscopic linear stapler. The specimen was placed into a plastic bag and retrieved through the umbilical port site which was extended to a 4 cm length (). As the consistency of the mass was soft, even a 10 cm sized mass could be extracted through a 4 cm length incision without any breakage (). The extended umbilical port site was partially repaired and pneumoperitoneum was reestablished. Intracorporeal Billroth-II reconstruction was performed. Oral feeding was started from the second postoperative day and the patient was discharged on the 7th postoperative day without any complication. The pathologic result was CD 117 and CD 34 positive gastrointestinal stromal tumor with 1 mitosis per 50 high power field (intermediate risk). |
A 19-year-old female sustained a closed spiral midshaft fracture of her right femur following a road traffic accident. The fracture was treated using a femoral intramedullary nail with one dynamic proximal screw and two lateral static distal locking screws. There were no intraoperative complications noted.\nAfter six months, she complained of difficulty walking. On examination she was noted to have an internally rotated right leg with her patella pointing medially. She also walked with a painful in-toeing gait on the right. No leg length discrepancy was found. Her wounds had healed well and she complained of no rest pain. Radiographs of her femur showed signs of union of the fracture. A CT scan of her right femur revealed approximately 30 degrees of internal rotation distal to the fracture site (see ).\nA decision was made to proceed with a derotational osteotomy, leaving the femoral nail in situ. At operation a lateral approach to the femur was performed. One K wire was inserted into the femur proximal to the fracture site and another inserted at 30 degrees of internal rotation distal to the fracture site. This angle was measured using a protractor normally used for the Maquet tibial osteotomy. A circumferential femoral osteotomy was made around the femoral intramedullary nail using a Gigli saw approximately 5 cm proximal to the distal interlocking screws. The bone was held above and below the osteotomy site using bone clamps. The lateral static distal interlocking screws were then removed and the malrotation was corrected. This brought the guidewires parallel, thereby correcting the malrotation deformity. Two antero-posterior static distal interlocking screws were used to secure the intramedullary nail and freeze-dried bone graft was inserted around the osteotomy site.\nThe patient went on to make a good postoperative recovery and at her six-month followup she had no pain and walked with a normal gait. The osteotomy site had united both clinically and radiologically (see ) and left the patient with no residual symptoms. |
A 38-year-old man sustained injuries in both lower extremities in a motor vehicle accident. Bilateral closed femoral shaft fractures were present, along with bilateral tibio-fibular fractures and severe soft tissue injury. The right leg was amputated at the above-knee level on an emergent basis, due to extensive comminution and loss of circulation caused by the crush injury. The bilateral femoral shaft fractures were fixed with plates. Reduction and external fixation of the left tibia was performed along with titanium plating. Initially, the size of the soft tissue defect was 20 × 15 cm, and the fractured tibial segments were exposed. The patient was treated in the emergency intensive care unit for 2 weeks due to multiple rib fractures and hemothorax. A bacterial culture from the tibial defect site revealed the presence of Pseudomonas aeruginosa. Wound care was performed with betadine, saline solution irrigation, and betadine-soaked dressings. The patient was shifted to the general ward 2 weeks after the trauma, at which time debridement and ostectomy of the necrotic tibia were performed. The tibial defect measured 17 cm. A vascularized bone graft was required to reconstruct the tibia; however, the contralateral fibula was lost due to the trauma and the ipsilateral fibula had multiple segmental fractures. We conserved the defect by external fixation of the tibia until complete union of the fractured fibula was achieved.\nThe P. aeruginosa infection did not resolve despite intravenous antibiotic injections and debridement. The patient was transferred to the isolation ward due to neutropenic fever, 7 weeks after the trauma. Carbapenem and other analgesics that were assumed to have caused the neutropenic fever were discontinued. After 1 week of observation, the patient returned to the general ward. Thorough sterile irrigation and dressing, as well as debridement, were performed under general anesthesia twice in order to eradicate P. aeruginosa without antibiotics. During the conservative management period, union of the fractured fibula was observed on serial follow-up radiographs.\nFour months after the trauma, decrease of P. aeruginosa was observed in wound culture from a 20 × 15 cm skin defect, and a united fibula was noted on radiography (). We reconstructed the tibial defect using the ipsilateral fibula, including the united fracture segments.\nFirst, computed tomography angiography was performed before surgery to check whether the three main arteries of the lower extremities were intact. The fibula was approached through a curvilinear posterior incision. The lateral head of the gastrocnemius was mobilized from the distal femur and the soleus was stripped from its origin from the posterior aspect of the fibula and the interosseus membrane. The popliteal vessels were dissected distally to the tibial vessels and the peroneal artery, which supplied the pedicle to the fibula. The fibula was completely exposed and ostectomy was done containing the flexor hallucis longus muscle and the peroneal artery. The 17-cm vascularized fibular flap was transposed through the intermuscular septum. After the transposition of the vascularized fibular flap, a bipedicled flap was transposed from the medial to the anterior side to cover the area of bone exposure. The harvested fibula was affixed to the tibial defect site using a plate and the external fixator system was maintained for stable alignment of the tibia (). The size of the soft tissue defect was 20 × 15 cm, and it was covered with the bipedicled skin flap.\nTwo months after surgery, the flap was stable and bony callus formation was observed on radiography. The patient started non-weight-bearing and muscle-strengthening exercises of the left lower leg. Three months postoperatively, hypertrophy of the transferred fibula and complete healing of the femur was observed on radiography. Weight-bearing training was started with a lower limb prosthesis on the amputated side. The patient learned to take steps with the prosthesis and the reconstructed lower leg with the help of a walker. Two weeks after the start of weight-bearing exercises, the patient had a fracture in the tibia at the weak point of the proximal plate fixation site. After confirming fracture union, weight-bearing training was restarted with a walker. Subsequently, the patient began to ambulate independently without the walker and returned to routine activities 6 weeks after the second tibial fracture. Two years postoperatively, good bony union and fibular hypertrophy were observed (). We confirmed the patient’s satisfaction in performing daily living activities with the reconstructed leg at a 12-year follow-up (). |
Our patient was a 13-yr-old girl who was referred to our hospital because of acute abdominal pain. She had a fever for two weeks before being referred to us. Her menarche had begun one year before her referral. She had suffered from severe dysmenorrhea until then, and her last menstrual cycle had been two days before coming to the hospital. Her abdominal pain had increased during her last menstruation. The patient had a fever, chills, abdominal pain, and nausea at the time of admission to the hospital. She did not have a vaginal discharge with bad odor and had no particular problems in her medical history. Generalized abdominal tenderness was diagnosed in the physical examination, but mild vaginal bleeding and right lateral vaginal wall bulging were found in her vaginal and transrectal examinations. She underwent laboratory tests including leukocytosis (18,000 mm), platelet (441,000), hemoglobin (10.7), C-reactive protein (3+), normal liver and kidney function tests, and normal coagulate profile tests. Her abdominal ultrasound images showed uterus didelphys with a mixed-pattern cystic mass measuring 72 65 62 mm close to the right ovary plus a solid cystic mass measuring 100 58 mm in the right adnexa. The right kidney was not observed.\nWe decided to do abdominal and pelvic computed tomography scan to evaluate the cystic mass which was reported in abdominal ultrasound imaging. We had planned to do the surgery in an appropriate condition. This meant with patient's intestinal preparation after evaluating computed tomography results, and in the presence of an expert general surgeon. However, a few hours after the admission, we had to do an emergency surgery on the same night because she had a high fever, increased ESR and CRP, and a possible risk of septic shock. We administered many antibiotics before the emergency surgery because of the acute abdominal pain.\nDuring the first surgery, we did a physical examination while she was under anesthesia. The hymen was normal and intact. But a buldge was found in the right side of her vagina during the rectoabdominal examination. This was confirmed by vaginal examination with a speculum. The right longitudinal septum was revealed with a closed end. At first, we cut the closed end of the hemilateral obstruction of the vagina and then the vaginal septum was resected with an electrocautery device. Afterward, a 200-cc pus was drained out, and this sample was sent to a laboratory for culture and antibiogram.\nThe patient's abdomen was opened with a midline incision. There was a severe adhesion of the intestine and omentum to the abdominal wall. Since no intestinal preparation had been done and there was a high risk of intestinal rupture, the general surgeon decided that it would be better to end this surgery and prepare the patient for another abdominal surgery in better conditions.\nAbdominopelvic-computed tomography scan (with and without contrast which was done after the first surgery) confirmed the ultrasound imaging findings of uterus didelphys with multi-cystic mass with severe development in the right adnexa. A remarkable right hematocolpus hematosalpynx was seen that was due to longitudinal vaginal septum and hemivaginal obstruction. The right kidney was not observed (Figures 1, 2). Her problem was detected as HWW syndrome infected with pyocolpos.\nWe did the second laparotomy in less than 48 hr after the first surgery with the same incision in the presence of an expert surgeon. Our patient had severe intestinal adhesion and perforated appendix fistulated to the right fallopian tube beside distal small intestinal obstruction. We saw pelvic inflammatory mass beside endometriosis. We did an appendectomy, right salpingectomy, entrolysis, and abscess drainage. Cefepim (Maxipim) and cloxacillin (Jaber-ibn-Hayyan pharmaceutical co) were administrated intravenously as specific infectious disease drugs until seven days after the surgery according to the specimen culture. We ordered continuous low-dosage contraceptive drugs for preventing the progression of endometriosis. Our patient was discharged from the hospital after seven days with a good condition. |
Fifty-nine-year-old woman presented to a pulmonary outpatient department for management of recurrent pneumonias due to bronchiectasis diagnosed two years ago. She was found to have on an average of four to five episodes of lower respiratory tract infections (LRTI) per year during the same period. She was apparently well until about two years ago when she started developing LRTI which was initially treated with various antibiotics including fluoroquinolones, beta-lactam antibiotics and macrolides at different walk-in clinics. Chest X-rays obtained before the presentation were normal. Computed tomography (CT) of the chest which was obtained at our hospital revealed right lower lobe focal cylindrical bronchiectasis. This was presumed to be post-infectious based on LRTI history. Frequency of LRTI increased from an average of two episodes in six months to three to four episodes in six months. Repeat sputum cultures grew pseudomonas aeruginosa every time. She was managed symptomatically with airways mucus clearance and fluoroquinolones as needed.\nHistory for pulmonary childhood infections, immunodeficiency, severe allergies and other risk factors for bronchiectasis was negative. Serum immunoglobulin G subgroups and immunoglobulin E levels were normal arguing against immunodeficiency and ABPA. Upon closer review of the computed tomography of the chest, a possible endobronchial lesion was noticed resembling a polyp of about half a centimeter in length (Figure ) at the right lower lobe posterior segment. Flexible bronchoscopy was performed revealing thick yellow-green secretions originating from the right lower lobe with an endobronchial lesion in the posterior segment at the same level as the CT scan abnormality. Cultures from the bronchoalveolar once again grew pseudomonas aeruginosa and pathology from the biopsy of the endobronchial mass revealed acute inflammation with predominant neutrophils which we related to ongoing infection. Over the next six months, the patient had three hospitalizations due to LRTI. Department of cardiothoracic surgery was consulted and the patient underwent a video-assisted thoracoscopic surgery and a right lower lobectomy was performed due to BSI of 15. The resected lobe was sent to a pathologist for histopathological analysis. An impacted 1.3 cm non-surgical plastic foreign body was discovered in the right lower bronchus by the pathologist during the gross examination of the specimen (Figures , ). The histology of the airway and lung parenchyma surrounding the foreign body showed chronic inflammation and reactive benign tissue growth. Post-operative recovery in the hospital lasted four days and was uneventful. The patient had no recollection of aspiration or choking in the past which could explain the foreign body, nor did she have risk factors for aspiration such as dementia, alcoholism, drug use, stroke, medications, etc. Subsequently, the patient has had no recurrence of infections in the one-year follow-up. |
A 66-year-old male with a past medical history of well-controlled hypertension, hypothyroidism, and coal worker’s pneumoconiosis was noted to have rising serum creatinine levels on outpatient evaluation and was referred for hospitalization for further evaluation of his progressive kidney disease.\nHe reported generalized weakness, fatigue, nausea, occasional vomiting, anorexia, and onset of a petechial skin rash that involved both lower extremities including the anterior tibial regions, lateral aspects of the upper thighs, and the inguinal areas. He had intermittent bilateral ankle edema and bilateral pedal neuropathy for about 3 weeks prior to the presentation. He reported having mild vague chronic lower abdominal discomfort for several months prior to the presentation. He had been taking an over-the-counter analgesic powder containing aspirin, acetaminophen, and caffeine daily until 5 months prior to presentation when he was hospitalized for pneumonia and was advised to discontinue taking the analgesic powder. After he discontinued taking the analgesic powder, he started taking 2 tablets of over-the-counter ibuprofen daily for his chronic headache and low back pain.\nHe experienced unilateral visual disturbances in his left eye 3 months prior to admission, and on ophthalmological evaluation, he was diagnosed with posterior scleritis and was prescribed oral corticosteroids for 1 month. Despite completing the corticosteroid course, his visual disturbances continued to progress with complete loss of vision in his left eye. He preferred no further workup or treatment for his left visual loss.\nReview of systems was further remarkable for chronic cough with occasional production of small amounts of brown sputum, chronic exertional dyspnea, occasional episodes of epistaxis, and unintentional weight loss of about 35 to 40 pounds in the preceding several months. For these pulmonary symptoms, he received several antibiotic courses over a 6-month period.\nHis family history was notable for renal disease of uncertain etiology in his sister. He used smokeless tobacco in the form of tobacco dipping, but no alcohol or illicit drug use was reported. He had occupational exposure to fine particulate matter as a coal mine worker for several years. He had no history of any episodes of pancreatitis, hepatitis, or elevated transaminases. Vital signs were within normal limits. Physical examination revealed petechial rash over both shins, upper lateral aspect of both thighs and in the inguinal areas, and trace bilateral ankle edema. He was hemodynamically stable with no uremic symptoms with a good urine output, and a functional capacity more than 4 metabolic equivalents.\nA trend in his serum creatinine level months prior to current admission is described in . Workup 6 months prior to current admission is shown in .\nAt the time of current admission, his serum creatinine was at 9.0 mg/dL with a non-anion gap metabolic acidosis and hyperphosphatemia due to kidney disease. Plasma potassium levels were noted to be in the normal range. He had a baseline hemoglobin in the range of 11 to 12 g/dL, and his white blood cell count was chronically elevated for the preceding 6 months in the range of 10 500 to 22 900/µL. Elevated lymphocytes, granulocytes, and monocytes were found on the differential. Blood cultures were negative. Electrocardiogram did not show any significant abnormalities. Urinalysis revealed no red blood cells, white blood cells, casts, or proteinuria. Urine sodium level was 72 mEq/L, and urine creatinine level was 50 mg/dL. Hepatitis C antibody was reactive, while viral load was undetectable; hepatitis B surface antigen was negative. Further workup revealed low complement levels with low C3 and C4, positive rheumatoid factor, negative antinuclear antibody, negative antineutrophil cytoplasmic antibody, negative cryoglobulin, elevated free kappa and lambda light chains, elevated erythrocyte sedimentation rate, and C-reactive protein (). IgG subclass evaluation is described in .\nChest X-ray demonstrated progression of interstitial lung changes, which prompted noncontrast computed tomography (CT) scan of the lungs, which revealed multiple significantly enlarged mediastinal and right hilar lymph nodes, worsened from the previous examination with changes of chronic obstructive pulmonary disease and reticular interstitial changes. Significant worsening of infiltrates predominantly in both upper lobes and right lower lobe with a small right effusion were seen ().\nRenal ultrasound was unremarkable. CT scan of abdomen and pelvis without intravenous contrast revealed increasing infiltrates within both lung bases and pleural thickening when compared with radiographic imaging 6 months previously, mild retroperitoneal fibrosis with prominent soft tissue in the aortocaval region below the renal vessels, nonobstructing left renal calculi, and hepatosplenomegaly without portal venous enlargement ( and ).\nNephropathological evaluation of the renal biopsy was significant for a finding of IgG4-related tubulointerstitial nephritis consistent with IgG4-RD. There was no evidence of an immune complex–mediated or active glomerulonephritis. Light microscopy of the renal biopsy showed diffuse plasma rich inflammatory cell infiltrate throughout fibrotic interstitium with a focal storiform pattern, scattered lymphocytes, and few eosinophils within the inflammatory infiltrate. Immunohistochemical staining for IgG4 was strongly positive for staining within the interstitial plasma cell infiltrate. Immunofluorescence microscopy showed granular IgG staining along tubular basement membranes.\nHe was administered a single bolus of 500 mg intravenous methylprednisolone and was discharged on prednisone 40 mg by mouth daily with dose tapering to be determined on future outpatient visits.\nAt his outpatient nephrology follow-up visit a month after discharge, his serum creatinine levels showed a steady decline to 3.0 mg/dL. There was also a decline in inflammatory markers with C-reactive protein at 6.5 mg/L and erythrocyte sedimentation rate at 43 mm/h while on prednisone 40 mg by mouth daily. He will be followed closely in the outpatient setting for further prednisone dose tapering and renal function monitoring. |
Fifty-nine-year-old woman presented to a pulmonary outpatient department for management of recurrent pneumonias due to bronchiectasis diagnosed two years ago. She was found to have on an average of four to five episodes of lower respiratory tract infections (LRTI) per year during the same period. She was apparently well until about two years ago when she started developing LRTI which was initially treated with various antibiotics including fluoroquinolones, beta-lactam antibiotics and macrolides at different walk-in clinics. Chest X-rays obtained before the presentation were normal. Computed tomography (CT) of the chest which was obtained at our hospital revealed right lower lobe focal cylindrical bronchiectasis. This was presumed to be post-infectious based on LRTI history. Frequency of LRTI increased from an average of two episodes in six months to three to four episodes in six months. Repeat sputum cultures grew pseudomonas aeruginosa every time. She was managed symptomatically with airways mucus clearance and fluoroquinolones as needed.\nHistory for pulmonary childhood infections, immunodeficiency, severe allergies and other risk factors for bronchiectasis was negative. Serum immunoglobulin G subgroups and immunoglobulin E levels were normal arguing against immunodeficiency and ABPA. Upon closer review of the computed tomography of the chest, a possible endobronchial lesion was noticed resembling a polyp of about half a centimeter in length (Figure ) at the right lower lobe posterior segment. Flexible bronchoscopy was performed revealing thick yellow-green secretions originating from the right lower lobe with an endobronchial lesion in the posterior segment at the same level as the CT scan abnormality. Cultures from the bronchoalveolar once again grew pseudomonas aeruginosa and pathology from the biopsy of the endobronchial mass revealed acute inflammation with predominant neutrophils which we related to ongoing infection. Over the next six months, the patient had three hospitalizations due to LRTI. Department of cardiothoracic surgery was consulted and the patient underwent a video-assisted thoracoscopic surgery and a right lower lobectomy was performed due to BSI of 15. The resected lobe was sent to a pathologist for histopathological analysis. An impacted 1.3 cm non-surgical plastic foreign body was discovered in the right lower bronchus by the pathologist during the gross examination of the specimen (Figures , ). The histology of the airway and lung parenchyma surrounding the foreign body showed chronic inflammation and reactive benign tissue growth. Post-operative recovery in the hospital lasted four days and was uneventful. The patient had no recollection of aspiration or choking in the past which could explain the foreign body, nor did she have risk factors for aspiration such as dementia, alcoholism, drug use, stroke, medications, etc. Subsequently, the patient has had no recurrence of infections in the one-year follow-up. |
A 5-month-old girl, weighing 5.4 kg and measuring 26 inches, was presented to us with a large cyst along the floor of the right orbit since birth. She was born at a gestational age of 39 weeks. The parents of the patient were first cousins and the patient had three normal siblings. The grandmother of the patient had died of leukemia at the age of 50 years and the father had also developed leukemia at the age of 35 years and later died of it. A 17-year-old cousin of the patient also died of leukemia. On clinical examination, the patient had a unilateral, right bluish cyst, measuring 4 cm × 3 cm × 1 cm, covering the right eye completely []. Slit lamp examination of the left eye showed a normal anterior and posterior segment, an intraocular pressure of 14 mm of Hg, and normal angles on gonioscopy. The thumbs were absent in both the hands. Blood tests of the patient showed no evidence of any metabolic disorder or coagulopathy and were within normal limits. The computed tomography scan of the right eye showed a cystic lesion having fluid in it and covering a microphthalmic eye []. X-ray of the hands revealed the absence of thumbs in both hands and the radius in the right forearm []. Magnetic resonance imaging of the abdomen showed absence of the left kidney []. Cytogenetic studies were performed on peripheral blood lymphocyte cultures stimulated with phytohemagglutinin and induced with mitomycin C (40 ng/mL). Standard procedures were used to obtain chromosomal preparations, which were subjected to Giemsa-Trypsin banding. A high frequency of chromosomal breakage was revealed on chromosomal analysis (30% of the examined metaphases had chromosomal aberrations). The patient was diagnosed as a case of FA.\nThe cyst was excised with preservation of the microphthalmic. 6 years have passed since the patient was presented to us, and is being followed up on a regular basis with a pediatric ophthalmologist, medical specialist, rheumatologist, hematologist-oncologist, and a genetic counselor. Routine blood cell counts, surveillance of bone marrow failure, and development of malignancy is mandatory in this patient. The patient has been advised with protective polycarbonate glasses and is doing fine clinically to date except for the fact that she has developed a right-hand deformity following a trivial trauma. Two of her first cousins, aged 19 and 21 years, respectively, have been diagnosed with leukemia recently and are being treated for it. |
A 46-year-old woman presented with discomfort in her left hand and difficulty in carrying out particular activities, especially during her job as a beautician. The problem evolved rapidly with appearance of abnormal posture of the left hand during repetitive movement performed at work, sometimes associated with pain. This condition severely limited her business. Previous examinations, including brain and cervical magnetic resonance scanning, had excluded a peripheral or central nervous disease as the cause of her symptoms. The patient's medical history excluded the presence of any psychiatric disorder. She has no story of psychogenic disturbances and she had never taken any antidepressants or neuroleptics. EMG of her forearm muscles showed the presence of dystonic activation patterns, indicating a diagnosis of focal dystonia. Pharmacological treatment with clonazepam and gabapentin was prescribed; however, as this failed to ameliorate the symptoms and caused side effects (such as drowsiness and gastrointestinal disorders), the patient suspended pharmacological treatment and came to our attention.\nWhen she came to our attention, the clinical examination showed an abnormal posture of the left hand with hyperextension of the IV and V finger and flexion of the interphalangeal joints of II and III finger. This particular posture was more evident during the execution of movements similar to those performed during her job. We performed surface EMG that confirmed the diagnosis of focal dystonia: her muscles showed typical signs of hyperactivity (). We administered 100 U incobotulinumtoxinA diluted in 1 mL saline solution into the following muscles of her left hand: extensor digitorum communis (35 U), flexor carpi radialis (35 U), and flexor digitorum superficialis (30 U). Electrical muscle stimulation was used to guide the injections.\nOne month later a marked reduction in muscle hyperactivity was observed. The patient mentioned a clear-cut reduction of discomfort and disappearance of the dystonic posture at as little as a few days after the injection and was able to work again. We conducted clinical assessments at 1, 3, 6, and 12 months after the injection and each time observed complete abolition of the symptoms. Moreover, after 10 months, we recorded surface EMG activity again, confirming the beneficial effect of incobotulinumtoxinA (). No reinjections have been necessary, up until the final clinical evaluation, performed 12 months after the original injection. |
Two cases of STS of the lower limb characterized by DVT and pulmonary embolism (PE) as the initial presentation were observed in our Orthopedic department: a 47-year-old man diagnosed with an idiopathic DVT of the right distal femoral vein and the popliteal vein (Figure A-B) and a 44-years-old woman with a massive PE (Figure A). The patient with DVT was treated with therapeutic doses of subcutaneous enoxaparin for 6 months. After 2 months of enoxaparin, the case with PE was referred to our outpatient clinic for worsening of leg swelling and dyspnea. Neither of the patients had a past or family history of VTE. Physical and radiographic examination did not reveal any mass in either case, but only the signs of DVT or PE. Due to the persistence of symptoms and the inefficacy of anticoagulant therapy, patients underwent magnetic resonance imaging (MRI) of the lower limbs and excisional biopsy of the visualized mass.\nIn the first case (Figure C-D), MRI showed a large mass in the anterior muscle compartment of the right thigh, with inhomogeneous appearance after gadolinium administration. The lesion was associated with multiple lymphadenopathy in the inguinal and external iliac region, and thrombosis of the right common femoral vein involving the ipsilateral common iliac vein and the inferior vena cava until the confluence of the renal veins. An excisional biopsy of the mass was performed. The lesion was found to be adherent to the femoral vein. The dissection of the vein showed a thrombus that obliterated the lumen of the common iliac vein. The histological examination revealed a high-grade leiomyosarcoma. The patient subsequently underwent chemotherapy and radiotherapy. A computerized tomography (CT) scan performed 6 months after surgery showed multiple pulmonary metastases. The patients is currently receiving chemotherapy in the Oncology division of our hospital.\nIn the second case, MRI showed a large mass located in the middle third of the right thigh, with low signal intensity in T1- and T2-weighted sequences and enhancement after gadolinium administration (Figure B-C-D-E-F). The lesion was in close proximity to the superficial femoral artery that appeared deformed and was adherent to the superficial femoral vein that appeared compressed and showed signs of thrombosis. After arterial embolization, an excisional biopsy was performed, followed by intraoperative brachytherapy. The histological examination of the bioptic specimen revealed a high-grade leiomyosarcoma. At one year of follow-up the patient was asymptomatic and showed no evidence of recurrence of malignancy at MRI. |
The patient was a four-year-old boy who was the third child of healthy Somali parents. They belong to the same tribe, but are probably not closely related. After an uneventful pregnancy, he was delivered at term without complications in Somalia in 2001. Birth weight, length, head circumference and Apgar scores are not known, but the mother described him as a healthy and normal infant until he was 5 months old. From this age he started to have seizure-like movements, which according to the mother's description, could have been myoclonic epilepsy or infantile spasms. At first, the seizures appeared at night, but soon they also occurred during the day, with increasing frequency and duration. The seizures stopped at 8 months after he was examined in a local hospital and put on antiepileptic drug therapy. Despite becoming seizure free, the mother described him as becoming markedly less responsive and alert.\nGrowth and motor development were normal for his age. He crawled at the age of 9 months and walked unsupported at the age of 12 months. Speech development was significantly delayed. The family moved to Norway when he was 1 1/2 years old. Although he could say some words at 2 years of age, verbal language skills improved only little after this age. At the age of 4 years he entered a day-care centre for autistic children, as it was observed that he had limited communication and social skills, gave poor eye contact and displayed a repetitive pattern of behavior.\nClinical examination at 5 years did not reveal any dysmorphic features. His muscle tone and strength and coordination and deep tendon reflexes were normal without signs of peripheral sensory deficit. His height and weight corresponded to the 25th centiles, and his head circumference was at the 75th centile. Eye examination with fundoscopy, hearing test, electroencephalography (EEG), echocardiography of the heart and magnetic resonance imaging of the brain were normal. Blood glucose, urea, creatinine, and liver transaminases were normal. Chromosome analysis showed a normal 46, XY karyotype. A Southern blot test for Angelman syndrome was negative.\nSince the time of the diagnosis of SBCADD our patient was on a trial with protein restricted diet (1 g protein/kg/day) for 5 months. The diet was reasonably well adhered to. He was tested by a psychologist using standard tests before and after 5 months of protein restriction treatment. No observable improvement was demonstrated during the period.\nWhen examined with ADOS-G (Autism Diagnostic Observation Schedule-Generic), he obtained a score of 13, just above the cut-off score of 12 for childhood autism []. ADI-R (Autism Diagnostic Interview-Revised) could not be administered, due to his mother's limited knowledge of the Norwegian language. When tested with Bayley Scale of Infant Development at 4 years, his cognitive ability score corresponded to that of 2 years. Like most autistic children, his visual-spatial abilities were much better than his language abilities. WPPSI-R (Wechsler Preschool and Primary Scale of Intelligence) at 5 years demonstrated visual-spatial abilities corresponding to 3 years of age. The verbal tests could not be administered due to lack of verbal abilities. Psychometric testing concluded with moderate mental retardation. This was also in accordance with the Vineland-2 Adaptive Behavior Scale, reported by his teacher. Motor development appeared normal when using the Movement Assessment Battery for Children test manual. During this examination, he was not able to follow verbal instructions.\nBecause mental retardation and autism sometimes can be caused by inborn errors of metabolism [], plasma and urine samples from the patient were analyzed by a metabolite screening system [] including gas chromatography-mass spectrometry (GC/MS) (organic acids), amino acid analyzer, tandem mass spectrometry (MS/MS) (purines, pyrimidines, acylcarnitines), and capillary electrophoresis (transferrin variants). Considerable amounts of 2-methylbutyryl glycine were identified in his urine. The excretion of 2-ethylhydracrylic acid was unremarkable. The patterns of amino acids and purines/pyrimidines were normal, except for a slight increase in glycine. MS/MS analysis of acylcarnitines showed increased plasma level of C5-acylcarnitine (1.43 micromoles/L, reference range 0.05–0.38) and an increased C5/C3-acylcarnitines ratio of 2.83 (reference range 0.16–0.73).\nDNA was extracted from blood and cultured fibroblasts by standard methods. PCR amplification of all exons and part of the flanking intron sequences of the human ACADSB (SBCAD) gene were carried out by use of intron-located primers under standard conditions in an automated thermal cycler (Thermal cycler 480, PE) []. The PCR products were sequenced in both directions using a 3100-Avant genetic analyzer and a BigDye® Terminator v1.1 Cycle Sequencing kit (Applied Biosystems). Sequence analysis of all exons, and part of the flanking introns of the human SBCAD gene from patient genomic DNA, revealed an apparent homozygosity for an A > G mutation, which changed the +3 position of intron 3 of the SBCAD gene (c.303+3A > G). No other changes were observed. Sequence analysis of the mother showed heterozygosity for the same c.303+3A > G mutation. |
A 26-year-old man came to the Oral and Maxillofacial Surgery Department of Taipei Medical University Hospital (Taipei, Taiwan) in May 2011 due to swelling and pain in the jaw. The intraoral examination revealed a raised mass in the left mandibular buccal vestibular area extending to the labial area without secretory discharge. The color and appearance of the surrounding mucosa were normal. The teeth in this area also have no obvious periodontal pockets (). On extraoral examination, the patient's lateral view shows a protruding mandible, and the left and right sides of the face were also slightly asymmetrical. The neurological examination was normal, and there were no swollen and palpable lymph nodes in the neck (). A radiographic examination showed a wide range of radiolucent lesions in the mandible from the canine area on the right to the second molar on the left. The biopsy and pathology reports confirmed that the lesion was consistent with ameloblastoma (). The pathology report is presented as follows. Grossly, they are gray and brown and elastic with some bone chips. Microscopically, it shows an ameloblastoma with islands of odontogenic epithelium in plexiform and follicular patterns within a fibrous stroma. The odontogenic epithelium shows peripheral palisading and stellate reticulum. Neither cytological atypia nor increased mitoses is seen.\nMarsupialization was performed to reduce the size and destructiveness of the tumor for subsequent resection surgery. The marsupialization was firstly performed in the anterior and left posterior region of the mandible. Soft liner was applied immediately as the obturator materials, and regular visits were arranged to monitor the changes of tumor size and to adjust the obturator (). After 4 months, the tumor size was gradually reduced on the left posterior region, but remained unchanged over the anterior region. Thus, the marsupialization was performed again over the anterior region. It took a total of 12 months to reduce the size of the lesion and confirm the scope of the tumor (). After that, the segmental mandibulectomy combined with fibula free flap reconstruction was conducted. At that time, three-dimensional (3D) image processing and printing were used to analyze and guide dental implant design and construction. The more precise the reconstruction operation can be, the more the damage can be reduced. Additionally, the entire postoperative period will benefit from precision reconstruction.\nTherefore, computed tomography (CT) examination was taken to evaluate the precise extension and region of the tumor. And then, the 3D printing transferred from CT files/images was applied to create a solid model, and the actual scope of the resection could be simulated before surgery (). A fibula 3D model was also exported for model surgery, surgical template making, and titanium bone plate bending. During the surgery, the defect was first fixed with a prebent metal bone plate, and then, the fibula part of the removed free flap was divided into three sections according to the surgical template and bent to meet the needs of the defect. Soft tissue defects in the mouth were also reconstructed with free flaps (Figures and ). With the 3D printed models, the surgery can be completed more accurately, and the treatment time can be significantly reduced.\nDuring the healing period, the patient could only use the remaining teeth, 46 and 47, and the opposite tooth. After 9 months, alveoloplasty was performed to correct the uneven and sharp bony edges. The patient was referred to a prosthodontist 3 years after the first visit. At this point, the soft and hard tissues had healed completely and were ready for reconstruction ().\nAfter a prosthetic evaluation, the problems and preliminary treatment directions were revealed. The patient's treatment program was developed to address the following issues. Full mandible implants were necessary and feasible to meet the physical and psychological needs of the patient. Considering that teeth 46 and 47 were the only two teeth that could be used for chewing, a staged dental implant approach was adopted. The transverse discrepancy in the upper and lower jaws and a deficiency in the right maxilla resulting in asymmetric facial features required resolution but could not be repaired by dentures alone. The cross-bite on the right side required resolution, and the space was limited for restoration on the right side.\nA staged approach was conducted. First, four implants in tooth 33, 32, 43, and 44 position were placed in the mandible. After the osseointegration, implant-supported provisional crowns 3332xxx4344 were installed in position. Then, two implants in teeth 34 and 36 were placed on the left mandible and were fitted with 34 × 36 provisional crowns. Finally, teeth 46 and 47 were extracted (). The purpose of the patient's orthodontic treatment was to gain restoration space on the right side and correct the upper jaw deficiency. In the beginning, tooth 17 was extracted and followed by leveling and alignment to create space (). After rapid palatal expander (RPE) placement (), the right maxillary posterior segmental osteotomy (PSO) (), right mandibular alveoloplasty, and extraction of tooth 48 were performed in the same operation to achieve proper interocclusal relationship and gain enough restoration space over right posterior region. Next, the patient was asked to regularly rotate the screw of the conventional Hyrax expander. One rotation per day produces a 0.2 mm lateral expansion, which lasts up to two weeks but varies from individual to individual. After the desired position was achieved, there was no need to continue turning the screw. However, it was still necessary to continue wearing the device for at least three months to stabilize the expansion. PSO and RPE provided the required lateral expansion effect to transform the upper arch into a better shape. Alveoloplasty also increased restoration space (Figures and ). The implant surgery was completed with the implantation of teeth 14, 46, and 47. The provisional teeth were installed after osseointegration. The lower jaw was divided into three groups of cement-retained bridges, and tooth 14 was a screw-retained crown. The prosthesis was made with zirconia-based material. The pontic type was designed to be consistent with the original provisional crowns and appropriate for the patient's extensive mandibular reconstruction. This design did not allow any contact between the pontic bottom and the gums so that the patient could clean the teeth as efficiently as before, and the large-scale bottom hollow was not prone to food impaction. After postoperative treatment was completed, the upper and lower jaws presented smooth and symmetrical dental arches, dental function was restored, and the patient's face showed improved symmetry (). |
A 68-year-old Japanese man presented with weight loss of 6 kg over a 4-month period. His other medical problems included hypertension and hyperuricemia. The patient's family history was unremarkable. Physical examination revealed conjunctival pallor. Computed tomography (CT) revealed a 7-cm left renal mass, and renal biopsy confirmed a diagnosis of renal cell carcinoma (RCC), although no apparent adrenal metastasis was detected during the CT. Nevertheless, the patient underwent unilateral nephrectomy with ipsilateral adrenalectomy based on the current guidelines for RCC. The guidelines recommend ipsilateral adrenalectomy with radical nephrectomy for tumors that are >6 cm, based on their high risk of adrenal metastasis, even in the absence of radiographic evidence of metastasis.[\nThe surgery was uneventful, although the patient lost consciousness when he stood up for the first time on postoperative day (POD) 1. The patient's blood pressure could not be measured, and electrocardiography revealed ventricular fibrillation. The patient received 4 injections of epinephrine and 2 defibrillator shocks, which led to the return of spontaneous circulation at approximately 20 minutes after the ventricular fibrillation was detected. Pulmonary embolism was initially suspected, although CT failed to detect any emboli. The workup included coronary angiography, which revealed 90% luminal narrowing of the #6 branch of the lateral ascending artery, and the narrowing was attributed to vasospastic angina. The patient underwent percutaneous coronary intervention, which successfully stabilized his cardiopulmonary condition. Laboratory testing revealed marked increases in potassium levels and eosinophil counts from POD 1 to POD 2 (Table ), when the patient experienced the cardiopulmonary disturbance.\nTwo weeks after the percutaneous coronary intervention, the patient developed a retroperitoneal cystic infection, which induced fever and hypotension, and Pseudomonas aeruginosa was identified as the causative agent. He was successfully treated using meropenem and intravenous fluid, recovered without any further adverse events, and was discharged at 2 months after the adrenonephrectomy. At the time of discharge, he still had decreased appetite and general fatigue. However, the patient's fatigue and appetite did not improve with rest at home, and he was readmitted to our center at 1 month after his discharge (3 months after the surgery). The workup revealed a markedly elevated morning serum level of adrenocorticotropic hormone (ACTH; 151.4 pg/mL, normal: 7–50 pg/mL) and a mildly decreased morning serum cortisol level (6.4 μg/mL, normal: 7–28 μg/mL) (Table ).\nThe patient underwent ACTH and corticotropin-releasing hormone stimulation tests, which both revealed markedly decreased responses (Figs. and , respectively). His plasma renin activity, aldosterone level, and dehydroepiandrosterone sulfate (DHEA-S) levels were all within the normal limits (Table ). Postoperative CT of the remaining adrenal gland, magnetic resonance imaging of the head, and histopathological analysis of the resected gland revealed no abnormalities. On the basis of the patient's laboratory data and his clinical symptoms, primary adrenal insufficiency was suspected, and treatment was started using oral prednisolone (20 mg). The patient's appetite and fatigue significantly improved shortly after the initiation of prednisolone treatment.\nTo exclude 21-hydroxylase deficiency as an underlying disease, genetic analysis of the CYP21A2 gene was performed, although no mutations were observed. Testing at 6 months after the surgery, and before the morning prednisolone dose, revealed a lower morning serum cortisol level (2.2 μg/mL) and an elevated morning serum ACTH level (98.7 pg/mL). At the 1-year follow-up, the patient's morning cortisol level was 2.0 μg/mL and his ACTH level was 44.1 pg/mL before the morning dose of prednisolone, which was consistent with the diagnosis of chronic primary adrenal insufficiency.\nThe patient gave written informed consent. The ethical approval was not required because of the retrospective nature of the case report. |
A 16-year-old male patient presented with a pulmonary mass in the upper lobe of the right lung for 10 days. Lab examination results show NSE was high (16.93 ng/ml). Lung enhanced CT showed a round soft tissue density shadow in the upper lobe of the lung, the size was about 6.3 × 6.2 cm, and the edge is smooth. The enhanced CT scan showed the value of the mass was about 46 HU and the adjacent bronchus is compressed. The distal lung tissue showed strips of ground-glass opacity shadow ().\nDuring the surgery, no adhesion was found in the thoracic cavity. The pleura was smooth and there was no significant effusion in the thoracic cavity. Pulmonary fissure developed normally. The tumor was located at the upper lobe of the right lung with a size of 6 × 5 cm. The tissue texture is hard and red. The mass was closed to the pleura. The tumor mass was relatively large, therefore, right upper lobectomy combined with lymph node dissection was performed.\nOne lobe from lobectomy. A mass is seen in the lung. The size of the lung lobe is about 11 cm × 9 cm × 4.5 cm. The mass is located 0.6 cm away from the end of bronchial anastomosis, close to the lung capsule with a total size of 7 cm × 6 cm × 5 cm. The section surface is gray and gray-red, the texture is slightly brittle, the margin is not clear, bleeding, shows a cystic change, and easy to be broken. The gross image of pulmonary mass includes the following: the section is gray and gray-red, bleeding, and shows a cystic change (). Under the microscope, the tumor cells are extreme poorly differentiated, round, medium size, nested, or scattered. It is similar to the morphology of small cell carcinoma or lymphoid epithelioid carcinoma. Few fibrous connective tissues were seen between the cell nests. There are multiple foci and slice-like necrosis. The adhesion of tumor cells is relatively poor. Most nuclei is round or oval-round shape and medium size. The size of cells is about two to threefold over lymphocyte. The ratio of the nucleus over cytoplasm is high; the nuclei are irregular and atypical. The chromatin is fine or granular, vesicle shaped, and its nucleoli are prominent. Mitotic figures and apoptotic body are commonly seen; however, there is no significant “squamous differentiation” pattern in the stained slice. Focal infiltration of interstitial lymphocytes is seen and arrange in a flaky, scattered, or nested pattern ().\nImmunohistochemical staining was performed on a broad spectrum of biomarkers (). In addition to NUT, several positively stained biomarkers have been identified. CKpan (partial+), NUT (nuclear positive), P63 (mostly+), TIF-1(few+), CK7(few+), CK5/P40(few+), CD5/6(few+), Syn (partial+), CD30 (few weak+), EMA (few weak+), Vimetin (+), CD99 (few weak+), HMB45, MelanA, CD3, CD20, CgA, CD31, CD34, Desmin, LCA, MyOD1, S-100, WT-1, PLAP, α-inhibin, CD138, CD38, MUM-1, TdT, ALK, Ki-67 index (80%), and florescence in situ hybridization EBEB (−). Some of the representative stainings are shown in .\nQuantitative PCR was performed, and the result indicated that the patient was positive for NUT carcinoma. Dye-based qPCR was used to identify NUT gene product, and lung adenocarcinoma and water were used as reference. The design of amplification probes: forward probe is located in exon 10 and reverse probe is located in exon 2 of the NUMT1 gene. The result showed that the CT value of sample is 26.34 which indicated a strong amplification. Moreover, the dissolution curve showed the product is specific. The CT value of reference sample is ≥34 which indicated a weak amplification. The dissolution curve showed nonspecific amplification which mostly was primer dimer. The results of Sanger sequencing showed positive for NUT rearrangement. The probe design: forward probe is located in exon 10 of the BRD4 gene and reverse probe is located in exon 2 of the NUMT1 gene. Amplification Sanger sequencing further confirmed that the translocation occurs between the NUMT1 and BRD4 genes ().\nUsing the high-throughput gene sequencing technique, we have identified KIFB-related heterozygous mutations and gene changes that are associated with multiple tumor-related pathways (). Some signaling pathways are associated with NMC. To be specific, most of the KIFB mutation leads to a dysfunction of those signaling pathways that are related to cell metabolism and differentiation. The dysfunction of these genes would lead to an increase in the risk of tumor development. Since NMC patients are often diagnosed in advanced stage, the surgical intervention is usually not an option. Thereby, biopsy sample from the surgery is relatively few. In 70% of the cases, translocation occurs with the juxtaposition of NUT (15q14) and BRD4 (19p13.1) genes or BRD3 (9q34.2, 6% of cases) and other unknown genes (24%). Usually, gene sequencing test was performed to evaluate the possible gene variants () in order to find possible gene mutations.\nThis patient was transferred to multiple places in Ji Nan (Shandong Province) and Beijing for medical consultation, and he received two traditional chemotherapies. The outcome of this case was unfortunately fatal, and the patient died 3 months after diagnosis. |
A 56 year old man presented to the Emergency Department after passing bright red blood mixed with dark clots per rectum. He had vague, crampy abdominal pains for the previous two days. Past medical history included hypertension, type 2 diabetes and ischaemic heart disease. One year previously, he was admitted to hospital with vague, intermittent central abdominal pain, which resolved following observation for 5 days.\nOn admission, he was tachycardic and hypotensive, with no abdominal tenderness or palpable masses. Rectal examination revealed bright red blood and clots on the glove. Admission haemoglobin was 8 g/dl. Serum ferritin was low at 19 μg/L. He was resuscitated and stabilised with intravenous fluids. Computed tomography (CT) scan demonstrated uncomplicated sigmoid diverticular disease and no other pathology to explain his symptoms. He underwent urgent upper gastrointestinal endoscopy, which was normal to the second part of the duodenum, with no signs of haemorrhage. Subsequent colonoscopy showed a colon full of fresh blood and clots up to the caecum, with no obvious bleeding source. Intubation of the small bowel and examination of the terminal ileum showed fresh blood filling the lumen, with a likely bleeding point in the proximal small bowel beyond the reach of the endoscope. At this stage, the patient became haemodynamically unstable and a decision was made to take the patient for an urgent exploratory laparotomy.\nAt laparotomy, blood was seen to fill the entire large intestine. The small bowel was filled with blood from the terminal ileum up to the proximal jejunum. The first 100 cm jejunum, after the ligament of Trietz, was fixed to the retroperitoneum with the rest of the proximal jejunum lying to the right of the midline (Figures &). There were no palpable masses or visible inflammatory pathology. The bleeding source was presumed to be in the proximal jejunum. The blood in the small bowel was emptied manually and a series of soft bowel clamps were applied to observe and confirm the site of the bleed. Blood was seen to fill the proximal jejunum, in the segment which was abnormally fixed in the retroperitoneum. The malrotated segment of jejunum was mobilised from the retroperitoneum. A segmental small bowel resection (75 cm) was performed, centred on the presumed point of haemorrhage. A primary side-to-side jejeno-jejeunal anastomosis was fashioned. The small bowel was examined again, with no further haemorrhage noted.\nSix units of blood were transfused during the operation. The patient was managed on the high dependency unit for 48 hours and was transferred to the surgical ward. His recovery was complicated by an infection of his central venous catheter site and Clostridium difficile-associated diarrhoea. He was discharged 14 days following surgery, with no evidence of further gastrointestinal bleeding or cardiovascular instability. Histological examination of the resected small bowel demonstrated focal dilatation of vessels within the mucosa, submucosa and muscularis propria layers, with areas of erosion, in keeping with the likely source of haemorrhage (Figure ). There was no evidence of thrombosis, vasculitis or neoplasia. The patient remained well at three month follow-up with no further drop in haemoglobin or signs of gastrointestinal bleeding. |
We present the case of a gentleman in his late sixties who was referred to our spinal clinic by his general practitioner (GP). He first attended the clinic six months after a fall from standing height, complaining of ongoing back pain. Plain radiographs arranged by his GP three weeks after his fall revealed a T12 fracture, and over the next several months, his pain failed to settle. He had no radicular symptoms and no change in bowel or bladder habits. His medical history included a radical oesophagectomy for oesophageal carcinoma eight years previously, chronic obstructive pulmonary disease (COPD), asthma, and morbid obesity. He lived alone in a bungalow, normally mobilised without any walking aids and was independent with his activities of daily living (ADLs). Following the fracture, he occasionally used a stick to mobilise and reported that his pain was preventing him from carrying out his ADLs.\nPlain radiographs were arranged by his GP three weeks following his fall and revealed a minor wedge fracture of the T12 body with uncertain chronicity. Repeat imaging three months later suggested worsening of the fracture. Upon receipt of referral to our services, six months following his original injury, and prior to clinic attendance, he underwent an MRI scan on which there was evidence of fracture non-union (Figure ).\nFollowing assessment in the clinic, standing thoracolumbar radiographs and CT scan were arranged (Figure ). The standing thoracolumbar radiographs were felt to be sub-optimal, but within the limitations of the study, the T12 fracture appeared to have worsened from initial radiographs, and with multi-level degenerative disc disease in the form of prominent anterior osteophyte formation was noted. On the basis of this imaging, it was felt that instrumentation was the best option to achieve adequate analgesia through spinal stabilisation and fracture union.\nThe procedure was performed under general anaesthetic by consultant spinal surgeon Daniel Rolton, with great care taken over patient positioning due to the risk of iatrogenic injury. Surgical stabilisation was achieved using a minimally invasive technique, through the use of posterior percutaneous pedicle screws, spanning three levels on either side of the fracture (from T9 to L3) and 5.5 mm titanium rods bilaterally. Pedicle screws were 6 mm × 40 mm for thoracic vertebrae and 6 mm × 45 mm for lumbar vertebrae (Viper, DePuy-Synthes). The operation was tolerated well and check imaging revealed satisfactory metalwork placement and satisfactory fracture reduction (Figures , ).\nOur patient remained in hospital for two days post-operatively, with no complications. He was followed up in our clinic at four-month intervals from six weeks post-operatively to eighteen months post-operatively, at which point he was discharged back to the care of his GP. His surgical wounds healed well, with no metalwork complications, and there was radiological evidence of union. At four months post-operatively, he required no mobilisation aids and returned to work. When seen twelve months following his operation, he described himself as "85% better" compared to his pre-operative status, with no pain routinely, having discontinued his analgesia. However, he reported some backache when standing and walking for long periods. |
A 13-year-old boy was referred to our tertiary young adult hip service by a Paediatric Orthopaedic Surgeon for evaluation of left groin pain and a decreased range of movement in the left hip. He was a keen rugby player and had experienced an avulsion fracture of the AIIS during a rugby game which was played without a warm up six months previously. MRI just after injury showed a single bony fragment measuring 12 mm × 4 mm × 12 mm at the rectus femoris origin of the AIIS and it was retracted inferiorly 3 cm, anteriorly 1 cm and laterally 0.5 cm. Conservative treatment was advised by the Paediatric Orthopaedic Surgeon and he was referred to a physiotherapist for mobilisation and subsequently muscle strengthening.\nFive months post-injury he was progressing well with the physiotherapy, although, he still felt a sharp pain in his left groin when running with a ball while playing rugby. Flexion was restricted to 70° and a decrease in internal rotation in comparison with the opposite side was observed while abduction, adduction, extension and external rotation were comparable to the other side. There was no significant tenderness and no bruise in the region of the groin, and no distal neurovascular deficit. Plain radiographs demonstrated hypertrophic calcification in the region of the rectus femoris avulsion, whilst the hip joints were normal in appearance. A CT scan showed well corticated heterotopic bone formation at the site, measuring approximately 3.5 cm at maximum diameter (). Motion analysis using the three-dimensional CT datasets clearly showed the impinging area especially in 70° of hip flexion.\nBoth conservative and surgical management were suggested and discussed with the patient and his parents. They chose to go ahead with surgery, as he could not play rugby well because of the pain and restricted range of movement. The patient wanted to play rugby at a fairly high level, hopefully nationally, in the future. At arthroscopy, he was found to have a well corticated large lesion of heterotopic ossification, and it was dissected carefully from the muscles (). Once the dissection was completed, it was extracted via an incision to deliver the lesion. A dynamic impingement test was performed on the table to ensure there was no residual impingement and the impingement lesion was resected with a 5.5 mm arthroscopic burr. A thorough washout was carried out before closure.\nThe patient was advised not to perform any rotational activities in deep flexion for six weeks postoperatively and to follow the 16-week post-operative rehabilitation protocol. He was also advised prophylaxis against heterotopic ossification. Eight weeks following the procedure, the wound had healed well and there were no obvious signs of complications such as infection or deep vein thrombosis. Along with this, he demonstrated a pain-free, fully functional range of movement in his left hip joint and was delighted with his progress. Plain radiographs showed no further signs of calcification (). He was advised to continue to attend physiotherapy for at least another eight weeks and to engage in more gentle sporting activities like cricket and basketball, which he had enjoyed before the injury. At the 1-year follow-up, he remains asymptomatic with a full range of movement in his hip and continues to play rugby at a high level. |
The patient is a 13-year-old male who initially presented at the age of 4 years with seizures, marked by staring, twitching of the left eyelid and projectile vomiting. He experienced anywhere between 10–60 seizures a day. Starting at the age of 7 years, after multiple failed attempts to control the seizures using various pharmacologic regimens, the patient underwent the first of 7 surgical resections. The first two surgeries were performed outside the United States and consisted of resections of the left hippocampus and neocortex and the left anterior temporal lobe and hippocampus. Nonspecific diagnoses of gliosis were reportedly made in examining the resected tissue from the first two surgeries. A third surgical resection for continued seizures at the age of 10 years included resections of the left frontal lobe and portion of the left temporal lobe; the pathologic diagnosis was focal cortical dysplasia, Palmini type IA []. The patient was seizure-free for 14 months postoperatively but eventually developed seizure recurrence. Three subsequent surgeries within a 3-week period ensued and included resections of the left temporal lobe, orbitofrontal region, and insula. The pathology in all three cases was interpreted as representing Palmini type IA focal cortical dysplasia. Seizures recurred within 2 weeks of the sixth surgery and persisted for the next 5 months. A seventh surgery included a left frontoparietal resection.\nHistopathologic examination of tissue from the final surgical resection showed focal evidence of contusional damage, related to previous surgery. Areas of cortical architectural disorganization marked by an abnormal layering pattern with focal absence of cortical layer two and occasional enlarged and dysmorphic neurons characterized by an atypical distribution of Nissl substance in the cytoplasm were observed (). The findings were interpreted as being consistent with a Palmini type IIA pattern of focal cortical dysplasia. Balloon cells were not identified. Additionally, multiple foci of perivascular meningeal and parenchymal chronic inflammation consisting of benign appearing lymphocytes were observed (). Most of the lymphocytes stained with T-cell antibody to CD3 (prediluted, Ventana, Tucson, AZ, USA) (); only rare CD20 (1 : 25 dilution, Dako, Carpenteria, CA, USA) positive staining B lymphocytes were present. Infiltration of the parenchyma by scattered lymphocytes was highlighted on the CD3 immunostain. Distributed primarily within the cortex were small collections of microglial nodules, highlighted with an CD68 immunostain (1 : 60 dilution, Dako, Carpenteria, CA, USA) (). Viral inclusions were not identified. Neutrophils, eosinophils and granulomatous inflammation were not observed. Focally, prominent cortical atrophy with gliosis was observed (). The histopathologic findings were interpreted as representing Rasmussen's encephalitis associated with focal cortical dysplasia (Palmini et al. type IIA pattern or ILAE focal cortical dysplasia type IIId) []. |
An 82-year-old female patient, was reported to the department of Oral Medicine having history of facial pain which was right sided, electric shock like, and confined to the right half of the lower lip and also lower half of the face on same side with duration of 10 months []. The patient had previously visited 3 dentists who were unable to relieve patients pain later for which she underwent extraction of decayed lower right back molar thinking it to be the root of cause [Figures and ]. Following the extraction, pain was still not relieved in the area. Then various pharmacological remedies were given like Tergretol 100mg, TID along with Gabapentin, where the pain was temporarily relieved, but patient persisted to have the shock like facial pain. The patient had a medical history of hypertensive since 15yrs and was on medication.\nAt the outset of the consultation and history, the patient described her pain as a dull aching pain in the area of 46, 47 and 48 (which the patient had got it removed) that was exacerbated with chewing. Clinical evaluation demonstrated an edentulous arch in relation to 46, 47 and 48 regions with attrition and cervical abrasion in relation to tooth 44 and 45. The tooth responded normally to temperature. Infrequently, pain of short duration radiated through the right lower half of the face. The patient stated that the pain occurred spontaneously at times – while speaking, laughing and when exposed to cold wind (especially while traveling on a 2 wheeler) and described it as electric shock like, which lasted for 2–3 s, and was so severe that the patient had tears in her eye during the attack phase.\nIn light of the patient's history and clinical examination, a provisional diagnosis of residual infection-induced neuropathic pain was given.\nThe patient was advised to get an OPG done to rule out if any residual infection was present within the alveolus, but to our surprise, the OPG revealed an impacted supernumerary tooth within the body of the mandible, that was surrounded by an well-defined unilocular radiolucency, with a sclerotic border, that was measuring approximately 2.5 cm × 1.5 cm in size and seemed to be encroaching on the mandibular canal; supernumeraries were also present in the 1st and the 3rd quadrants []. To rule out any cortical plate expansion, a mandibular cross-sectional occlusal radiograph was taken, which did not reveal any expansion [].\nBased on the radiological appearance a diagnosis of dentigerous cyst was made, with a differential diagnosis – Odontogenic Keratocyst& cystic Ameloblastoma was given.\nShe was further investigated with CBCT to rule out, if the lesion was impinging on the mandibular canal. The CBCT reports revealed the involvement of the nerve [Figures – and ].\nFollowing the radiographic findings including CBCT reports, the patient was advised excision of the lesion. She underwent medical workup with complete hemogram, liver function test and peripheral smear, which were within normal limits and a physician consent was obtained.\nThe surgical procedure ENUCLEATION was done in toto under local anesthesia. The patient was given antibiotics and anti-inflammatory drugs for 5 days [Figures –].\nPostoperative healing was uneventful, but the patient had paresthesia of the right half of the lower lip and the patient was advised to take neurobion forte daily for 30 days.\nThe excised specimen [] was sent for histopathologic examination which revealed fibrocollagenous tissue lined by stratified squamous epithelium exhibiting palisading of basal epithelium and luminal surface containing wavy parakeratotic epithelial cells. Subepithelium shows mixed inflammatory cell infiltrate along with congested blood vessels [].\nDuring follow-up, there was a drastic improvement in her symptoms; hence, carbamazepine was tapered and stopped after a month. |
A 78 year-old man was admitted to the outpatient clinic of our department of otolaryngology with complaints of sudden pain of left auricle and ear canal. The patient also showed facial paralysis on left side (House-Brackmann grade III). He didn't have any underlying disease and showed a social history of smoking with 60 packs years and no history of alcohol intake. According to the physical examination, there was found no dermatological lesion such as rash, vesicle or crust near the ear drum, external auditory canal and near the external ear. The patient showed a sensorineural hearing loss of high frequency in both ears on pure tone audiometry. The hearing ability showed no worsening pattern as the symptoms were developed. The pain that was shown in this patient was of great severity compared to that generally shown in Bell's palsy, and he complained of a stronger, deep pain in the external auditory canal rather than in the areas near the auricles. Accordingly, beside to Bell's palsy, we considered an atypical type of Ramsay-Hunt syndrome that doesn't show dermatological lesions, as our possible differential diagnosis. We started with high doses of steroid (prednisolone, 60 mg/day) and antiviral drug (Acylovir 1800 mg/day) after the patient was hospitalized. Even though it's rare to present severe otalgia from primary or metastatic tumors of the skull base, we performed a CT scan of the temporal bone to rule out these possibilities. The findings from the CT scan showed a bony destruction of the temporal bone of the left, anterior attic and sphenoid bone (). Consequently, we could assume that the facial paralysis occurred due to damages of the first geniculate ganglion of facial nerve. We performed a MRI of the temporal bone in order to find out the etiology of the bony destruction of temporal bone and we observed a neoplastic lesion that is invading the base of the skull and left sphenoid bone. It was observed that the neoplastic lesion invaded the middle cranial fossa, temporalis muscle, left side of the foramen ovale and the left cavernous sinus and it could be diagnosed as a metastatic tumor of the temporal bone (). On the 8th day after hospitalization, we performed a positron emission tomography-computed tomography (PET-CT) scanning in order to confirm the presence of other metastasis to other organs and the existence of an originating region of the metastatic tumor. The PET-CT scan show-ed a high fluorodeoxyglucose uptake in the left middle cranial fossa, both ribs, left scapula and the left adrenal gland. We also observed a lesion of 3 cm in the upper lobe of the right lung () and tried to perform a biopsy on the upper lobe of right lung in suspicion of lung cancer with multiple areas of metastasis. However, it was not allowed as the guardians of the patient didn't want any invasive examinations or treatment, considering the patient's age and recovery rate after the treatment. Currently, the patient is under conservative treatment including pain management and he's under observation for 1 year so far. |
A 26-year-old male, unmarried, educated up to 10th standard, a laborer by occupation but unemployed for the past 4 months, was brought to the psychiatry OPD by his brother with complaints of poor concentration, excessive thoughts, repeated checking, difficulty in performing work, disturbed sleep for the past 15 years, and increased severity of symptoms for the past 4 months. The patient was asymptomatic 15 years back. He reports that he was sexually abused by his cousin brother who was 6 years elder to him. His brother forced him to have sexual intercourse on multiple occasions. The patient reported that he eventually started enjoying those incidents until his cousin went away for work. After the abuse, he started having repetitive thoughts about the act. He was not able to concentrate on studies and dropped out of the school. After that, he started doing small works nearby to support his family. However, he could not sustain one job since he had difficulty in concentrating on work. He uses to have repetitive sexual thoughts and sexual urges to an extent that he even made sexual intercourse with animals (goat). After these instances, the patient felt guilty about those acts and moved to the city where he was removed from multiple jobs because he made multiple mistakes and was last to finish his task. Over the past few months, he had excessive sexual thoughts and urges, to an extent that he even had thoughts about having sexual contact with children. As these thoughts started increasing, he eventually told his brother that he feels that he has some problem with his mind and he was subsequently brought by his brother to psychiatry OPD. The patient's mother was a diagnosed case of schizophrenia and was under regular treatment for the past 15 years. On MSE, his mood was sad and he had an anxious affect, his thinking was goal directed, he had repetitive, excessive, intrusive, blasphemous, and sexual thoughts along with compulsive acts of checking things. A diagnosis of OCD was made, he was treated with capsule fluoxetine 60 mg with behavior therapy, he showed significant improvement over the span of 6–8 months, and he regained his socio-occupational functioning. |
A 35-year-old male presented 6-month postexenteration for invasive fungal infection. There was complete loss of the orbital contents and medial orbital wall along with the upper and lower eyelids with a naso-orbital fistula leading to escape of inspired and expired air through the orbital defect. The exposed orbital cavity had unstable lining with history of recurrent bleed from the orbital surface. The patient also gave history of recurrent upper respiratory tract infections []. On evaluation, loss of both eyelids with orbital contents and lining with a large communication with the nasal cavity was observed. The patient was investigated in the form of routine blood investigations along with three-dimensional computed tomography scan of the face []. The aim of the reconstruction was to release the socket contracture, give stable lining to the orbital wall, close the naso-orbital fistula, and provide supple matching soft tissue cover for anophthalmic orbit amenable for external prosthetic reconstruction.\nSince the patient had a naso-orbital fistula, this condition was a challenge for the anesthesiologist as induction was not possible with routine bag and mask ventilation due to the escape of inhalational agents through the fistula. Thus, obliteration of orbital route was first accomplished with dry pad packing, and it was only then the patient could be put effectively on inhalational agents []. Socket contracture was released, unstable lining was excised, and the orbital defect was measured. The dimensions of the defect were 5.2 cm in its depth, medial to lateral wall measured 6.4 cm, and distance between supraorbital ridge and infraorbital rim measured 3.5 cm []. Radial artery forearm free flap (RAFF) was chosen as the preferred method of reconstruction. The flap was planned and outlined on the left forearm after performing Allen's test []. The central skin paddle was used to create the matching eye patch which would be subsequently used for eyelid reconstruction by an external prosthesis and the peripheral adipofascial component used for filling of the orbital defect. Under tourniquet control, a curvilinear incision starting from the inner boundary (central skin paddle) of the flap was given along its axis to reach the antecubital fossa. The skin incision for the flap was limited to the dimensions of the central skin paddle. The skin was elevated off the peripheral component of the flap till its outer dimensions where it was deepened to incise the deep fascia. The flap was then carefully raised keeping in view of the adequate pedicle length. A tunnel of adequate size was created superficial to the facial muscles for the pedicle to be anastomosed with facial artery and vein in the neck. Adipofascial component was placed into the defect, and the central paddle was sutured with the skin margins. The donor site was closed with a split skin grafting cover.\nThe patient has been followed for 6 months with complete fistula correction and well-settled flap []. Further reconstructive procedures such as eyelid reconstruction and placement of orbital prosthesis are yet to be performed. The donor site has healed well with no loss of function. |
An 18-year-old male patient reported from another private clinic to the Department of Oral Surgery in our hospital, complaining of pain in the mandibular anterior region since the past 6 months. Patient revealed past history of trauma in the same region 4 months back. The patient was asymptomatic 3 months following trauma; however, he noticed discharge from the lingual aspect of the permanent mandibular right canine since 15 days. The assessment of the previous medical history was non contributory. On extra oral examination, there was no observed swelling or lymphadenopathy. No expansions of the cortical bone or neurosensory defects were evident. Intraoral examination revealed pain on palpation in the mandibular anterior region. The overlying mucosa was intact with no signs of infection and sinus tract formation. The mandibular anterior teeth were intact and not mobile. An intraoral periapical radiograph revealed well demarcated unilocular radiolucency in the region of parasymphysis. The panoramic radiograph showed radiolucency extending from tooth 33 to the mesial root of 46 []. There was no resorption of the roots of the teeth involved in the area of the lesion. Pulp vitality tests were performed with respect to tooth numbers 31, 32, 33, 41, 42, 43, 44, 45 and 46, and they were unresponsive to electrical and thermal stimuli. Based on the history, clinical and radiological features, a preliminary diagnosis of periapical cyst was established.\nAccess opening was done and root canal treatment was performed for all the non-vital teeth involved in the area of the lesion. However, the patient reported again after a period of 2 months, with the complaint pain and swelling which had gradually increased. The swelling was 3 × 2 cm in size, with well defined borders and was hard on palpation. It extended antero-posteriorly from the tooth number 32 to 45, and superior-inferiorly involved the entire alveolus only sparing the inferior inferior border of the mandible, and caused expansion of the buccal cortical plates. Patient complained of pain on palpation in the area of the swelling. Fine needle aspiration biopsy revealed no significant pathology. As the lesion had not responded to endodontic therapy, the diagnosis of inflammatory periapical cyst was excluded. Other non inflammatory periapical lesions such as central giant cell granuloma, aneurysmal bone cyst, odontogenic keratocyst and ameloblastoma were considered in the differential diagnosis. The patient was referred to the department of oral and maxillofacial surgery and surgical enucleation of the lesion under local anesthesia with adrenaline was advised. A full thickness flap was raised, and an encapsulated lesion was located between the enlarged buccal plates. After enlarging the bony access, the entire cyst was enucleated by performing curettage of the lesion and protecting the mental nerve. The excised specimen was then submitted for histopathological examination. The patient's post operative recovery was uneventful with no signs of recurrence within 1 year follow up examinations.\nMicroscopic examination revealed a plexiform ameloblastoma predominantly composed of epithelium arranged in long anastomosing strands and cords []. Under higher magnification these epithelial cells were bounded by columnar ameloblast like cells and were surrounded by stellate reticulum like cells. The supporting stroma was loosely arranged and vascular in nature [Figure , ]. Thus, based on these findings, a final diagnosis of plexiform ameloblastoma was made. |
A 30-year-old female with middle aortic syndrome, diagnosed at the age of 15, presented to us with hypertension and significant buttock numbness after a brief period of sitting down. Past cardiac history was also remarkable for a bicuspid aortic valve and mild mitral regurgitation. She was medically managed with an angiotensin-converting enzyme inhibitor and β-blockers for hypertension and was a nonsmoker who expressed strong wishes to become pregnant. Her obstetrician was concerned that her condition would not support her pregnancy owing to lack of blood flow to the pelvis during pregnancy. The patient had two prior spontaneous miscarriages. Physical examination was remarkable for hypertension, with systolic blood pressure ranging from 160 to 180 mm Hg. Strong palpable pulses were noted in her upper extremities, with weaker palpable pulses in her lower extremities.\nPreoperative work-up included a magnetic resonance (MR) angiogram which revealed a normal appearing thoracic aorta that tapers from 2.0 cm down to 8 mm in size. There was no evidence of stenosis at the origin of the renal arteries or in the mesenteric vessels (\n). A dynamic cardiac MR revealed a maximal gradient of 37 mm Hg at the level of the diaphragm.\nThe patient underwent a tunneled descending thoracic aorto-left common iliac artery bypass, avoiding the need for a traditional large thoracoabdominal incision. The thoracic aorta was accessed via a 10-cm left thoracotomy incision and the left common iliac via an 8-cm left paramedian incision. A retroperitoneal tunnel was then created, and an opening in the diaphragm was made to enter the thoracic cavity. The bypass was performed using a 22 cm × 9 mm Dacron graft between the descending thoracic aorta and the proximal left common iliac artery tunneled through the neo-diaphragmatic hiatus. She did very well in recovery and was discharged from the hospital on postoperative day 5 without any complications.\nAt her 3-week follow-up visit, the patient reported complete resolution of her buttock pain and was able to discontinue her antihypertensive medication. The patient subsequently became pregnant and delivered a full-term baby. At 1.5 years' follow-up, the patient continues to do well. |
A 50-year-old male patient admitted to the emergency because of an electric shock which occurred 15 minutes before (alternative current, 50 Hz, 220 V). The patient indicated that as soon as he touched the wall plug to switch on light, he was shocked from his right hand and found himself lying on the floor of his home. He stated that he had lost his consciousness with the shock and after recovery he had found out he had a tightening chest pain radiating to his left arm. His pain had continued increasingly. Arterial blood pressure ratio was measured as 140/80 mmHg on examination of the patient. Pulse was arrhythmic and tachycardic. No lesion was observed regarding the electric shock entry and exit points. Sinus tachycardia, increase in the amplitude of the T-wave at inferior derivations, and ventricular premature beats were monitored in the performed ECG ().\nAfter establishing a vascular access, routine blood tests were performed and the patient was monitored by connecting to a defibrillator. Nearly after ten minutes, atrial fibrillation developed with a sharpened chest pain. In the performed ECG, ST segment elevation in inferior derivations and ST segment depression in V1–V3 derivations were observed (). In order to maintain the ventricular rate under control, intravenous beta-blocker was applied to the patient whose blood pressure was stable. Due to continuous ST segment elevation, ASA 300 mg and clopidogrel 600 mg were given and enoxaparin 0.6 cc was applied subcutaneously. In an effort to exclude a potential Type 1 myocardial infarction, the patient was immediately transferred to a center where primary angioplasty could be performed in 120 minutes. Before coronary angiography in the hospital where he transferred, the patient's chest pain disappeared, ST segment descended to isoelectric line, and the patient returned to sinus rhythm approximately 100 minutes after the detection of symptom. In the performed coronary angiography, normal coronary arteries were determined and dominancy of right coronary artery was observed. The patient was thought to have right coronary arterial vasospasm (Type 2 MI) which was probably triggered by electric shock. A moderate increase in troponin and CK-MB levels was seen in the monitorization of the patient. In echocardiographic examination, EF was estimated as 58% and segmental motion defect was not detected. The patient was discharged with full recovery after four-day-long observation in the hospital. |
A 13-year-old boy was referred to our tertiary young adult hip service by a Paediatric Orthopaedic Surgeon for evaluation of left groin pain and a decreased range of movement in the left hip. He was a keen rugby player and had experienced an avulsion fracture of the AIIS during a rugby game which was played without a warm up six months previously. MRI just after injury showed a single bony fragment measuring 12 mm × 4 mm × 12 mm at the rectus femoris origin of the AIIS and it was retracted inferiorly 3 cm, anteriorly 1 cm and laterally 0.5 cm. Conservative treatment was advised by the Paediatric Orthopaedic Surgeon and he was referred to a physiotherapist for mobilisation and subsequently muscle strengthening.\nFive months post-injury he was progressing well with the physiotherapy, although, he still felt a sharp pain in his left groin when running with a ball while playing rugby. Flexion was restricted to 70° and a decrease in internal rotation in comparison with the opposite side was observed while abduction, adduction, extension and external rotation were comparable to the other side. There was no significant tenderness and no bruise in the region of the groin, and no distal neurovascular deficit. Plain radiographs demonstrated hypertrophic calcification in the region of the rectus femoris avulsion, whilst the hip joints were normal in appearance. A CT scan showed well corticated heterotopic bone formation at the site, measuring approximately 3.5 cm at maximum diameter (). Motion analysis using the three-dimensional CT datasets clearly showed the impinging area especially in 70° of hip flexion.\nBoth conservative and surgical management were suggested and discussed with the patient and his parents. They chose to go ahead with surgery, as he could not play rugby well because of the pain and restricted range of movement. The patient wanted to play rugby at a fairly high level, hopefully nationally, in the future. At arthroscopy, he was found to have a well corticated large lesion of heterotopic ossification, and it was dissected carefully from the muscles (). Once the dissection was completed, it was extracted via an incision to deliver the lesion. A dynamic impingement test was performed on the table to ensure there was no residual impingement and the impingement lesion was resected with a 5.5 mm arthroscopic burr. A thorough washout was carried out before closure.\nThe patient was advised not to perform any rotational activities in deep flexion for six weeks postoperatively and to follow the 16-week post-operative rehabilitation protocol. He was also advised prophylaxis against heterotopic ossification. Eight weeks following the procedure, the wound had healed well and there were no obvious signs of complications such as infection or deep vein thrombosis. Along with this, he demonstrated a pain-free, fully functional range of movement in his left hip joint and was delighted with his progress. Plain radiographs showed no further signs of calcification (). He was advised to continue to attend physiotherapy for at least another eight weeks and to engage in more gentle sporting activities like cricket and basketball, which he had enjoyed before the injury. At the 1-year follow-up, he remains asymptomatic with a full range of movement in his hip and continues to play rugby at a high level. |
A female infant was born by Caesarean section at a gestational age of 30 weeks and 5 days to a healthy 29-year-old mother. There were no abnormalities demonstrated on routine antenatal ultrasound scans. The birth weight was 1,545 g, and the Apgar scores were 7 and 9 at 1 and 5 minutes, respectively. At 3 days of age, heart murmur and cardiomegaly were detected. Doppler echocardiography done at 4 days of age revealed normal intra-cardiac structure and a suspected PDA measuring 4 mm in diameter between the left subclavian artery and the main pulmonary artery (). Two cycles of oral ibuprofen treatment (10 mg/kg for the first dose, followed at 24-hour intervals by 2 additional doses, 5 mg/kg/dose) were given, but failed to close the PDA. At 1 week of age, small hemangiomas involving the left periauricular, temporal, and infraocular area were detected. The hemangiomas subsequently developed into large facial hemangiomas that threatened the left eye (). Surgery at 2 weeks of age was performed because of the failure of oral ibuprofen treatment and persistent cardiomegaly. Surgery showed an atypical PDA arising from the left subclavian artery and entering the main pulmonary artery, which was closed by surgical clipping. A subsequent three-dimensional cardiac computed tomography (CT) scan and angiography not only confirmed this diagnosis, but also revealed complicated cardiovascular anomalies, including a right-sided aortic arch and the tortuous left subclavian artery. In detail, a three-dimensional cardiac CT scan and angiography showed a mirror image right-sided aortic arch. The first branch of the right-sided aortic arch was a left innominate artery which was divided into a left common carotid and subclavian artery. The second was the right common carotid artery, and the third a right subclavian artery. The PDA arising from the base of the left subclavian artery was closed by surgical clipping, and did not form a vascular ring. A small collateral artery between the thoracic aorta and the left subclavian artery was observed (). Because of the large size of the facial hemangioma, oral prednisolone (1 mg/kg/day) and topical clobetasol-17-propionate were initiated at 1 month of age. At 6 weeks of age, brain magnetic resonance imaging (MRI) and angiography (MRA) were obtained because PHACE association was suspected. These showed the presence of the Dandy-Walker variant with hypoplastic cerebellar vermis and multiple intracranial hemangiomas in left cerebellopontine angle (). No definite abnormalities in the intracranial large vessels were noted on the brain MRA. At 7 weeks of age, laryngoscopic examination revealed a hyperemic lesion on the uvula, and oropharyngeal hemangioma was suspected (). Ophthalmologic and audiometric examinations showed no abnormal findings. No ventral developmental skin defects were noted. The thyroid profile was normal. In addition, no specific findings were detected in the chromosomal analysis, including array comparative genomic hybridization (CGH) to identify gene copy number variations. The prednisolone therapy was gradually tapered and oral propranolol was added at 10 weeks of age. Thereafter, she was treated with intralesional triamcinolone and pulsed dye laser in addition to oral propranolol. This appeared to reduce the proliferation of the hemangioma. No significant changes were noted on a subsequent laryngoscopic examination done at 3 and 8 months of age. At the follow-up examination at 12 months of age, the hemangioma in the infraocular area was significantly decreased. Neurological examinations at 6 and 12 months of age demonstrated no developmental delay for her corrected age. Seizures were not noted. In addition, physical measurement at 12 months showed a significant growth retardation in weight, height, and head circumference (<5th percentile for her age). |
A 39-year-old gravida nine para two was referred at 30 weeks of gestation for evaluation of MAP. Her history included two uncomplicated low transverse cesarean deliveries, four curettage procedures for pregnancy termination, and two spontaneous abortions. Pelvic imaging showed an anterior placenta with obliteration of the uterine interface, presence of intraplacental vascular lacunae, and increased vascularity proximal to the bladder and loss of the myometrial margin. Findings were suspicious for placenta percreta possibly involving the urinary bladder. The patient was counseled extensively regarding the management of MAP, including conservative management and its associated risks, and comprehensive informed consent was obtained. Planned delivery at 35 weeks and 6 days of gestation occurred under general anesthesia. Immediately prior to delivery, the patient underwent uncomplicated placement of bilateral hypogastric artery balloon catheters and bilateral ureteral stents. At the time of stent placement, systematic inspection of the bladder was noted to be normal on cystoscopy. Intraoperatively, an area of tortuous blood vessels on the uterine serosa was noted extending to a ballooned right broad ligament and to the border of the bladder reflection on the left side, confirming a placenta percreta with broad ligament and possible bladder invasion (). Given the extent of abnormal placentation, conservative management was elected. Uncomplicated cesarean delivery of a neonate weighing 3,150 grams via a fundal hysterotomy then followed. After high ligation of the umbilical cord, the placenta was left adherent to the myometrium. The hysterotomy was repaired in three layers and total estimated blood loss was 1000 cc. The patient was then transferred to the interventional radiology suite in stable condition where balloon catheters were removed followed by bilateral internal iliac embolization using a slurry of absorbable gelatin compressed sponge (gelfoam). Postembolization arteriography was performed showing stasis within the uterine arteries.\nShortly after arrival to the postanesthesia care unit the patient reported buttock numbness but was otherwise asymptomatic. The epidural catheter was removed on postoperative day one and shortly thereafter she reported a burning sensation in the right buttock. Physical exam revealed a tender 9 × 11 cm area of ecchymosis in the medial aspect of right buttock with extension to the gluteal crest. Given recent embolization, there was concern for end-artery compromise and potential soft-tissue and skin necrosis. Interventional radiology, vascular surgery, and plastic surgery teams were consulted. Computed tomography angiogram demonstrated patent internal iliac arteries with patent proximal pelvic branches. Over the following week the lesion blistered, necrosed, and unroofed to reveal healthy tissue beneath. The lesion was treated with topical silver sulfadiazine cream and pressure relief. The patient was discharged home on postoperative day seven with close outpatient follow-up.\nThe patient was planned for interval hysterectomy at six to eight weeks postpartum but was briefly lost to follow-up. Shortly thereafter, she presented and was admitted to the hospital when she noted umbilical cord tissue prolapsing from the vagina. She subsequently underwent hysterectomy at ten weeks postpartum []. At that time the buttock wound was noted to be healing well with improved epithelialization, scar contraction, and healthy granulation tissue throughout (). She was recommended continued use of daily hydrated polymer dressings until the wound was fully closed. |
A 12-month-old boy was admitted to the hospital because of recurrent pulmonary infection and combined severe respiratory distress. The patient had already been diagnosed with congenital pulmonary agenesis on the right side based on pre-natal ultrasonography and a postnatal computed tomography (CT) scan. After admission, a follow-up CT scan and bronchoscopy were performed to evaluate the condition of the lung and trachea and their relationship with surrounding structures. Bronchoscopy found tracheal narrowing at the level of the aortic arch, and chest CT confirmed that the trachea was compressed between the aortic arch and the spine. We thought that the patient’s respiratory distress was caused by extrinsic tracheal compression. The cause of tracheal stenosis was mediastinal deviation that developed due to congenital lung agenesis (). To relieve mediastinal deviation, diaphragm translocation was performed.\nThe operation was performed under general anesthesia, with the patient in the left lateral decubitus position. Posterolateral thoracotomy was performed at the level of the sixth intercostal space (ICS), and an additional mini-thoracotomy incision was made at the level of the ninth ICS to facilitate identification of the margin of the diaphragm. We approached the thorax through the extrapleural space. Upon evaluation of the pleural space, the parietal pleura and mediastinal pleura were found to be intact, although no structure was associated with the lung and bronchus in the pleural space. The diaphragm was detached along the costal margin, from the sternum to the spine. The peritoneum was preserved in the costal area; however, around the tendinous portion of the diaphragm, the peritoneum needed to be dissected for sufficient mobilization of the diaphragm. Several interrupted Prolene sutures reinforced with pledgets were applied to anchor the harvested diaphragm to the chest wall. The diaphragm was fixed at the level of the third rib laterally, at the level of the fourth rib anteriorly, and to the adjacent ribs posteriorly to achieve tension-free attachment of the diaphragm (). Finally, two separate soft drains were inserted into the pleural cavity and peritoneal cavity. No unexpected events occurred during the operation.\nThe patient was transferred to an intensive care unit (ICU) with mechanical ventilation because he had suffered severe preoperative respiratory distress due to a large volume of secretions. The following day, the patient was extubated and the drains were removed at postoperative day 12. The postoperative course was favorable; however, bronchitis developed and the patient suffered from a large amount of sputum and mild dyspnea. The patient received conservative care at the ICU and was transferred to a general ward at postoperative day 7. A follow-up CT scan performed on postoperative day 14 revealed improved mediastinal and tracheal deviation. Furthermore, because of the rearrangement of the mediastinal structure, tracheal narrowing seemed to have improved. The patient was discharged on postoperative day 17 without any complications. The patient underwent follow-up at the out-patient department until 1 year after surgery. A follow-up CT scan and bronchoscopy revealed improvement of the tracheal narrowing (), and the patient’s symptoms were much improved. |
A 48-year-old man presented with a chief complaint of hypersensitivity and unacceptable esthetic appearance of his anterior teeth. The patient went to see a dentist at a private clinic to receive dental treatment for his anterior teeth erosion. The dentist performed history taking and found no obvious intrinsic or extrinsic causes of the erosive lesion. Thus, the patient was referred to the Prosthodontic Clinic at the Faculty of Dentistry, Prince of Songkla University, Thailand for consultation and joint management. Medical and dental history taking were performed to verify whether the patient was exposed to any cause of dental erosion or not. The patient mentioned that he rarely consumed acidic foods or beverages and had no sign of acid regurgitation presented. Apparently, it was found that the patient had been routinely swimming for 3 months. Since then, he noticed that his anterior teeth had been eroded. He stopped swimming 2 weeks ago because of the extreme sensitivity of the anterior teeth. The patient was asked to collect a sample of pool water from the swimming pool that he routinely went to swim in. The sample of pool water was evaluated for a pH value using a pH meter (Precisa, pH900, Precisa Gravimetrics AG, Dietikon, Switzerland). The pH value for the pool water was 4.5, which was lower than the critical value for enamel dissolution. Moreover, the patient reported that he spent more than an hour swimming each time, which meant that he had been exposed to low pH pool water for more than an hour every day over 3 months.\nIntra-oral examination revealed loss of enamel on the labial and incisal surface of the maxillary anterior teeth. Four mandibular incisor teeth presented with loss of enamel on the facial surface and there was excessive reduction of the incisal edges. Diastemas appeared between most of the teeth, and the incisal plane was uneven (). The patient's esthetic appearance was affected by the irregular incisal edge position of the remaining tooth structure. Thus, he had no confidence to smile. The erosive lesion appeared with well-defined finish lines along the free gingival margins resembling veneer preparation (). There was no dental caries which existed and his periodontal health was fine. The pulp vitality test showed no signs of pulpal pathology. The occlusal function and vertical dimension of occlusion appeared to be normal since the patient had adequate and stable posterior stops. A significant finding in this case was the maxillary-mandibular relationship, which appeared to have anterior open bite due to the loss of tooth structure. However, the patient had no difficulty in pronouncing an "S" and "F" sound during function. During the comprehensive oral examination, the patient complained that his teeth were extremely sensitive to air blow. The diagnosis in this case was severe anterior tooth surface loss, resulting from long-term exposure to low pH in swimming pool water.\nThe treatment goals in this case were to restore the damaged tooth structure and reestablish the esthetics of the smile. Definitive treatment modalities for excessive tooth erosion include direct composite resin restorations, porcelain laminated veneers, and complete coverage crowns. Since the remaining tooth structures were insufficient to support composite resin restorations and porcelain-laminated veneers and the optimal clinical crown height needed to be restored, complete coverage metal-free crown restorations were the treatment of choice in this case for long-term durability and esthetic satisfaction.\nIrreversible hydrocolloid impressions were performed for fabricating diagnostic casts. A face-bow transfer was made and the casts were mounted in maximum intercuspation on a semi-adjustable articulator (Denar Mark II, Whip Mix, Louisville, KY, USA). Diagnostic wax-up was made on the casts with the optimal tooth proportion and with 1 mm horizontal and vertical overlap. The wax-up was transferred to the patient's mouth using the direct intra-oral mock-up procedure with bis-acryl provisional material (Protemp 4, 3M ESPE, Seefeld, Germany). The mock-up () was adjusted to obtain desirable esthetics, phonetics and function, and It was subsequently duplicated for using as a guideline for fabricating provisional restorations. The splinted provisional restorations were made with heat-polymerized polymethyl methacrylate (Namilon, American Tooth Industries, Oxnard, CA, USA) prior to the tooth preparation.\nIt was decided to use a novel hybrid ceramic (Vita Enamic, Vita, Zahnfabrik, Germany) for constructing metal-free restorations in this case. Vita Enamic is an interpenetrating phase composite material that combines the properties of ceramic and polymer. This material consists of a fine microstructure of feldspathic ceramic matrix and an acrylate-based polymer network. As a result, wear characteristics, flexural properties, and elasticity of this material are similar to dentin., The minimum thickness required to ensure clinical success of restorations made from VITA ENAMIC is at least 0.8 mm circumferentially. Therefore, the amount of tooth reduction required is less than tooth preparation designs for other all-ceramic materials. Maxillary incisor and canine teeth were prepared with a 1 mm wide chamfer finish line and mandibular incisor teeth with a 0.8 mm wide chamfer finish line. No additional incisal reduction was required since the incisal edges were eroded. Internal line angles were rounded in all prepared teeth. A double cord gingival retraction was performed and final impressions were taken using a polyether impression material (Impregum Soft, 3M ESPE, Seefeld, Germany).\nShade selection was made using the Vita 3D Master System. Splinted provisional restorations were relined and temporarily cemented (Temp-Bond NE, Kerr, Orange, CA, USA). The impressions were poured in Type IV stone (Silky Rock, Whip Mix, Louisville, KY, USA) and pindexed. The pindexed casts were mounted on an articulator in maximum intercuspation. Laboratory prescription for Vita Enamic crowns were completed and sent to an outside laboratory (T Dental Lab, Bangkok, Thailand). Final restorations were recieved from the laboratory ().\nClinical try-in and adjustment were performed prior to permanent cementation. Comprehensive esthetics, phonetics, and function were evaluated. The occlusion was assessed to eliminate occlusal interferences, especially on the maxillary and mandibular canines. The edge-to-edge occlusal relationship was established in this case to avoid excessive coronal crown height in both maxillary and mandibular anterior teeth. The intaglio surfaces of the restorations were treated with 5% hydrofluoric acid (VITA CERAMICS ETCH, Vita, Zahnfabrik, Germany) for 60 seconds according to the manufacturer's instruction. The etching gel was completely removed using water spray and the restorations were dried for 20 seconds. Silane was applied to the intaglio surfaces of the crowns and dried completely. Self-adhesive resin cement (Rely X Unicem, 3M ESPE, Seefeld, Germany) was used to cement restoration following the manufacturer's recommendations. Excess cement was removed completely and the special care instructions for ceramic restorations were given to the patient (). After completion, the patient's discomfort and tooth sensitivity disappeared. The patient has been back to swim in a swimming pool with proper chlorination. Esthetics, phonetics, and function were satisfactorily restored. After 6-months follow-up, the restorations were in place with no complications. The periodontal health appeared to be normal. The patient has been maintaining good oral hygiene care of his teeth. Post-treatment monitoring will be followed in every 6 months. Since there have been insufficient clinical report regarding using hybrid ceramic in this type of case, long-term follow-up will be performed for at least 3 years. Further long-term clinical performance will be provided. |
Our patient is a 53-year-old male who worked as a coal miner. In spring of 2017, he noticed enlarging scalp nodules and right hip pain. He also complained of fatigue but was otherwise asymptomatic. Because of the hip pain and nodules, he came to the emergency department where a CT scan showed an enlarged right frontal scalp mass that measured about 4 cm (). MRI also revealed a right frontal scalp mass and extensive vasogenic edema localized to the right frontal lobe with a 5 mm midline shift at the foramen of Monroe (). A subsequent CT chest, abdomen, and pelvis was performed and showed a lytic lesion in his ilium and a single enlarged right hilar lymph node (not shown). A follow-up nuclear medicine bone scan demonstrated multiple areas of increased uptake concerning for metastasis (). One of the largest areas was in the right frontal scalp. After discussion with the patient and the need for a definitive diagnosis, he was consented for subtotal resection of the right frontal scalp mass.\nHe was taken to the operating room where a right frontal scalp incision was performed for subtotal resection of likely metastatic cancer. The mass was primarily located within the right frontal scalp but extended through the skull into the right frontal cortex. It did not however appear to be involved with the brain parenchyma. The resection consisted of removing scalp mass and taking a core biopsy from the specimen. Histopathology of the biopsy showed a diffuse and monotonous infiltrate composed of small cells with irregular nuclei, condensed chromatin and inconspicuous nucleoli (). On immunohistochemistry, the cells are CD20 positive B-cells and co-express CD10 and Bcl-2 but do not express CD3, 5, 23, 43 and cyclin D1 (). CD21 was present highlighting a disrupted follicular dendritic cell meshwork. Ki-67 proliferation rate was low (less than 20%). By flow cytometry, a dim kappa light chain-restricted B-cell population was identified expressing CD10. The FISH assay revealed t(14;18) IGH (immunoglobulin heavy chain locus)-Bcl-2 fusion. These findings support the definitive diagnosis of follicular lymphoma, WHO grade 1, diffuse pattern stage IV.\nAfter definitive diagnosis was made, the patient was then referred to radiation oncology for follow-up. By time of presentation to radiation oncology, the patient was showing symptoms of lower extremity numbness, personality changes, and some memory loss. The radiation oncologist discussed the diagnosis with the patient and his family and came up with a treatment plan to combat the unusual presentation of follicular lymphoma with the intracranial extension from the calvarium. The treatment plan consisted of repeat MRI’s; local (palliative) XRT (4 Grays over two fractions) and systemic chemo-immunotherapy (6 cycles) followed by rituximab maintenance. Lumbar puncture was done a month after resolution of vasogenic edema to assess leptomeningeal spread later proven negative for malignant cells. MRI showed complete resolution of the right scalp mass post-radiotherapy and after the patient had already underwent 3 cycles of chemo-immunotherapy. PET/CT done approximately 3 months since therapy initiation showed no suspicion for abnormal hypermetabolic activity, which suggested the absence of active malignancy. |
A 46-year-old man with a smoking history of 15 pack-years presented with cough and dyspnea. He had been treated for a malignant mediastinal germ cell tumor, the original part of which had been surgically resected with part of the left lung and the left phrenic nerve 10 years earlier, resulting in paralysis of the left diaphragm. He had also undergone treatment for bilateral iliac metastasis with chemotherapy and heavy ion radiotherapy 5 years before and metastasis to the right lung with surgical wedge resection 6 years before. Nine months before the outpatient visit, the patient had undergone stereotactic body-radiation therapy with 56 Gy of radiation in seven fractions to treat another right lung metastasis measuring 3.5 cm in diameter and located close to the right hilum (Fig. a, b). On presentation, computed tomography revealed right bronchial stenosis over the range of previous radiotherapy (Figs. b, c; a, b). Bronchoscopy showed broad necrosis of the right bronchus, ranging from the right main bronchus to the orifices of the bronchi of the middle lobe and the superior segment of the lower lobe (Fig. a–d). Pulmonary function was low with 2.6 L of vital capacity and 1.7 L of forced expiratory volume in one second. He was repeatedly treated with bronchial debridement of the necrotic tissue, culture of which revealed infection with Aspergillus fumigatus and other various bacteria. Bronchoscopic biopsies were performed several times, showing no malignancy but only fungal hyphae. Despite intensive antibiotic therapy with voriconazole and repeated debridement, the necrosis was progressive. Neither balloon dilatation nor stenting seemed to be an option because of the fragility of the bronchial wall and serious concern regarding formation of a bronchopulmonary arterial fistula. The patient eventually developed sepsis and renal failure, which necessitated urgent surgical removal of the necrotic bronchus. However, he was unable to endure right pneumonectomy because of his low respiratory function caused by left phrenic nerve paralysis. Preoperative pulmonary perfusion scintigraphy using technetium-99m-labeled macroaggregated albumin showed moderate-to-severe decrease of blood flow into right upper lobe, and flow volume into right lung was 47%. We therefore performed right lung autotransplantation with preservation of the right basal segment, the orifice of which appeared to still be viable (Fig. d).\nDuring the surgery, we first prepared a pedicled omental flap through an upper median laparotomy in the supine position. We continued the surgery in the left decubitus position, and right lateral thoracotomy was performed through the fourth intercostal space. Right pneumonectomy was performed with staplers placed on the right main pulmonary artery and right pulmonary vein, while the right main bronchus was trimmed to the carina because of the broad bronchial necrosis. On the back table, we removed the upper and middle lobes and the superior segment of the lower lobe, then flushed the remaining basal segment anterogradely and retrogradely using extracellular-type trehalose-containing Kyoto (ET-Kyoto) solution in the same manner as in our living-related lung transplantation protocol. Next, we implanted the basal segment by direct anastomosis of the bronchus to the carina, followed by end-to-end anastomoses of the pulmonary arteries and anastomosis of the basal pulmonary vein to the superior pulmonary vein (Fig. a). The arterial anastomosis was technically challenging because of the distance and extensive dissection of the main pulmonary artery centrally behind the superior vena cava was necessary, while the venous anastomosis was easy after dissecting the pericardium around left atrium in the same manner as regular lung transplantation. We also wrapped the bronchial anastomosis with an omental flap to facilitate bronchial healing and lower the risk of a bronchopulmonary arterial fistula (Fig. b). The operation took about 18 h due to extensive tight adhesions derived from the previous treatment, refractory bleeding from adhered regions and anastomosis of pulmonary artery, and repetitive bilateral lungs ventilation. The back table surgery took about 2 h and graft ischemic time was around 5 h. After operation, the bronchial mucosa of the transplanted basal segment appeared pale distal to the bronchial anastomosis (Fig. e).\nOn postoperative day 9, the patient developed left-sided pneumonia and required temporal support with extracorporeal membrane oxygenation for 2 weeks. One month later, minor bronchial dehiscence at the bronchial anastomosis was observed and was successfully treated with antibiotics and nutritional support. Three months after the operation, the right bronchial anastomosis showed stenosis due to overgrowth of granulation tissue, which was treated with repeated bronchoscopic dilatation and eventual placement of a silicone stent (Fig. f). The patient was discharged home 5 months after the operation in a stable condition. At discharge, his pulmonary function was even lower with 1.26 L of vital capacity and 0.88 L of forced expiratory volume in 1 s although he was free of supplementary oxygen.\nThree months after discharge, however, the patient developed massive hemoptysis. He was resuscitated on venoarterial extracorporeal membrane oxygenation, and hemostasis was obtained. Because he had fully recovered from the condition, we started discussing further surgical intervention to segregate the bronchial anastomosis and pulmonary artery. Regrettably, however, he soon developed a second massive hemoptysis episode and died. |
A 7-year-old boy along with his parents reported to our department for treatment. The chief complaint being as presented by the mother was pain in relation to the child's upper left teeth since 2 days. Since the child's appearance and head size was not normal, the family and medical history were taken in detail before referring the child to the pediatrician and psychologist for their expertise. The pediatrician and psychologist did diagnose the child with Crouzon's syndrome associated with mild to moderate mental retardation. Review of medical history was unremarkable, specifically, the mother reported normal labor and delivery. There were no anomalies in any siblings or near relatives reported. The child was not on any medications and denied any medical allergies. He has never been to a dentist before and this was the first dental consultation. Further medical history revealed that the enlarged size of the head was noted by the mother ever since he was 6 months and the severity has gradually increased.\nExtra-oral examination revealed elliptical-shaped head, with dolichofacial growth pattern, and convex facial profile. The presence of prominent eyeballs, which is the characteristics of the Crouzon disease triad, can be observed [ and ].\nIntra-oral examination showed that the patient is in early mixed dentition, with all primary teeth present in the maxillary and mandibular arch, the chronology of eruption and eruption status was normal for the child's age. The child also had a high arched palate which is typical of this syndrome. The deciduous teeth that were present were grossly decayed owing to the poor oral hygiene maintained because of lack of awareness on part of the parents and also partly due to the mental condition of the child. The permanent first molars which were erupted were in occlusion and healthy. The orthopantamograph (OPG) which was taken confirmed these findings. The OPG also showed the presence of all succedaneous teeth which looked healthy but the teeth on the left mandibular quadrant appear to have macrodontia which again is a feature which has been reported [].\nKeeping in view the mental condition and behavior pattern of the child and also the fact that so many teeth were to be extracted and restored we decided to carry out complete oral rehabilitation under general anesthesia followed by fabrication of functional space maintainer and a regular preventive regimen for which the parents would have to get the child to us on a periodic basis.\nThe rehabilitation was completed with an oral prophylaxis, the extraction of all the grossly decayed primary teeth, and the restoration of all the deciduous canines with glass ionomer cement. Impressions of the maxillary and mandibular arches were made for the fabrication of a functional space maintainer [Figure and ]. The teeth extracted were all grossly decayed which could not be saved and also anterior teeth which showed preshedding mobility were extracted.\nThe child was discharged on the following day and recalled after a week for follow up. The healing of the extraction sockets was uneventful and no other complications were reported. The removable functional space maintainer which was made with the child's favorite color was delivered to him.\nThe preventive regimen consisted of regular follow up every 2 months wherein based on the presenting eruption status, the functional space maintainer would be modified by trimming the same. A specially designed handle for his toothbrush was fabricated using self-cure resin so that he was comfortable using it []. At the subsequent follow ups for the next 2 months, topical fluoride varnish was painted on his permanent teeth and also on the remaining of the primary teeth. The child is also found to be coping with his space maintainer appliance quiet successfully. The child is been considered for further treatment by the oral and maxillofacial surgeons, pediatricians, and psychologists for his requirements.\nWe also will try and use interceptive orthodontics at the right time to treat his mid-face deficiency thereby improving his facial profile and appearance as early orthodontic intervention will prevent many malocclusions from developing. |
A 61-year-old female was admitted to our department with complaints of right facial pain for three years with coexisting severe cervical pain and itch for one month. The onset of the right facial pain was June 10, 2010. The patient stated that the pain was not serious (verbal pain scale, VPS 2) and that she never needed any analgesics to relieve it. As the pain did not change, she paid no attention to it, and no imaging studies were performed until one month previously. New symptoms of right facial and cervical pain, as well as right cervical itch, developed on July 5, 2013. The pain (VPS 9) and itch were severe, and drugs (tramadol, carbamazepine, and antihistamines) did not relieve the symptoms. Physical examination showed only paresthesia of the right cervical area, with no abnormal observations of the limbs, the central nervous system, or the right cervical skin. Magnetic resonance imaging (MRI) of the head was performed one day after admission. Unfortunately, the head MRI showed that the right trigeminal nerve was compressed by the right posterior cerebellar artery (). A trigeminal nerve microvascular decompression was planned; however, the right cervical pain and itch were not explained by the trigeminal nerve compression. After a group discussion involving the whole department, a cervical MRI was performed. The cervical MRI demonstrated a round intramedullary lesion located at the C2 level, exhibiting a core of increased signal intensity and surrounded by a low signal intensity rim on the T2-weighted images ().\nA debate was held on which surgery should be performed first: microvascular decompression or intramedullary lesion resection. Considering that the right cervical pain and the itch were recent developments, we opted for the removal of the C2 intramedullary lesion first. After the preoperative examinations were completed, the patient was sent to the operating room. Microsurgical extirpation by posterior middle incision under general anesthesia was performed. After the skin and muscle were dissected, a laminectomy at the C2 level was performed under the microscope, assisted by neuroelectrophysiology. The dura was opened and an expanded cord a dark red lesion was clearly visible under the microscope (). A myelotomy was performed directly above the lesion. The cavernous angioma was completely resected ( and ), and was confirmed by a postoperative pathological examination. Interestingly, two weeks after the operation, the itch was effectively relieved and the patient stated that the right facial pain was also relieved, with her pain score decreased to 3 (VPS). Microvascular decompression was not performed because during the one-year follow-up period, the symptom of right facial pain was successfully relieved and no new symptoms occurred. No lesions recurred during the follow-up period ( and ). |
Here, we present a case of a 55-year-old Caucasian female with a history of substance use disorder and a comorbid bipolar disorder, who presented to the local general hospital with a history of the fragmentation of a single personality into different personalities under emotional stress and under the influence of a drug. Multiple aspects of her personalities were reported, including the following: a personality of a seven-year-old child, a personality that would behave as a teenager, and another that acted like a male person in addition to her normal 55-year-old personality. She reported that she had been constantly dominated by her alternate personalities and became aware of their existence when people around her informed her, usually after a situation ended. She reported that stressful situations and substance abuse could aggravate the fragmentation of her personality. This was found to be mostly an involuntary phenomenon with seldom memory of the event.\nWhile transitioning between these personalities, she was found to be violent even to people who were close to her. This could range from being suicidal to homicidal for which she was arrested twice in the past. She had to be isolated and restrained by being locked in a room and calling the police. As a result, she was hospitalized in a mental institution for a significant period at least two to three times in the past. Under the influence of stress or substances like marijuana or cocaine, her personality would split into various personalities. These states were very different from one another in terms of age or gender.\nOne of her alternate personalities behaved as a seven-year-old child and would show the same interests and choices that included becoming moody or a self-arrogant personality. While in these states, she could hurt herself or had weeping spells if her wants were not met.\nAnother personality acted as a teenager with some sharp choices and dressing. Increase in substance abuse, alcohol use, and smoking would lead to multiple cases of fights or homicidal attacks, with some incidents of self-harming events. Multiple scars were found on the dorsal side of her right hand. Her speech was found to be pressured and she would repeat the same words/ conversations.\nThe next personality was diagnosed to be a temporary transition to the opposite gender (a male). There was a change in voice and behavior. This included male dressing, language, a perception of male body parts, choices of friends, and attraction towards females, including sexual behavior.\nThe normal state of a 55-year aged female was the default personality that made her feel most comfortable. She reported that she had anxiety during a personality state transition, as it could occur at any time, and involuntarily, but mostly in stressful situations and during substance abuse. More violent and harmful events were reported when someone tried to meet the patient alone rather than in a group.\nThe treatment included psychotherapy with cognitive behavioral therapy addressing stress and substance use disorder. The psychotherapeutic treatment lasted for at least six months. The dual treatment of drug therapy was also involved to calm her down. The patient was prescribed escitalopram to reduce her anxiety symptoms. She believed that the anxiety pills were really helpful. After six months, the patient's condition was not drastically different. However, she believed her stress was getting better. The patient was further followed up for the next one year and the treatment continues to date. |
A 38-year-old male patient attended our Plastic Surgery outpatient clinic because of left knee soft tissue defect associated with the exposed proximal patella (). Physical examination of the left knee region revealed exposed left patella with questionable vitality and 4 × 4 cm skin defect with visible patella fixation sutures.\nEight years ago, the patient was involved in a traffic accident driving a motorcycle and sustained left patella fracture. He underwent surgery which was not carried out in our hospital. Few months following the initial surgery, osteosynthesis metalwork was removed because of the infection. After the metalwork removal, the wound healing was complicated with wound dehiscence. Therefore, negative pressure wound therapy, antibiotics and physical therapy were all used for wound healing.\nThe wound healed with elongated callus formation at the fracture site and the patient was able to straight leg raise. Two months before the patient presented in our clinic, his orthopaedic surgeon performed the resection of the callus and patella osteosynthesis. The procedure was complicated by the proximal patella necrosis and soft tissue necrosis. After proximal patella debridement, resection and reinsertion of the quadriceps muscle tendon with transoseal sutures was performed and the exposed bone was covered with a local flap. Post-operatively, the local flap used to cover the soft tissue defect became necrotic, so it was debrided and negative pressure wound therapy was applied.\nTo cover the soft tissue defect and to preserve the remaining patella, we planned to use medial gastrocnemius flap with medial sural artery and vein pedicle. Instead of well-known reconstructions of quadriceps muscle tendon using fascia lata [] or semitendinosus tendon [], we decided to use gastrocnemius muscle fascia.\nAfter skin-sparing debridement of the anterior aspect of the left knee, medial head of gastrocnemius muscle was fully mobilized by cutting free gastrocnemius muscle origin and insertion. The gastrocnemius muscle fascia was harvested and a tubular structure was made out of it (, ). A transverse tunnel (2.5 mm in diameter) was made through the patella. The tubular fascia was pulled through the formed tunnel and, more proximally, fixed to the remaining part of the quadriceps femoris tendon. Furthermore, the patellar tendon was reinforced on the anterior aspect with the remaining gastrocnemius muscle fascia ().\nThe soft tissue defect was reconstructed with a gastrocnemius muscle flap and the flap was covered with a split-thickness skin graft (Thiersch). Redon drain was put in the donor site and negative pressure wound therapy dressing was applied to the knee wound.\nPostoperatively, the patient had no complications. Negative pressure wound therapy dressing was removed on the third post-op day with skin graft acceptance and vital muscle flap. On the 10th post-op day wound looked healthy and above knee backslab was applied. One month following the surgery, the patient started having passive physiotherapy and three months after the surgery active physiotherapy was started with gradually putting weight on his left leg.\nThe patient was last seen in our clinic 12 months after the surgery and he had full active knee extension and limited flexion up to 110° with excellent cosmetic outcome (). |
A 50-year-old male patient who underwent total thyroidectomy and neck dissection with the diagnosis of thyroid papillary carcinoma 17 years ago presented to our clinic with the complaint of swelling in a region corresponding to the thyroid compartment on the left side. A 4 × 3 cm sized hard mass was palpated on the left side at the level of cricoid on physical examination. Endoscopic examination of the larynx and vocal cord movements were normal. Ultrasonographic examination revealed a solid mass with heterogeneous internal structure that was about 4.5 × 2.5 × 4 cm in size invading the laryngeal structure. Fine-needle aspiration biopsy taken from the mass was found to be compatible with the thyroid papillary carcinoma. Neck CT showed a malignant mass lesion extending to the larynx in the cranial direction in the thyroid compartment and destructing cricoid cartilage and thyroid cartilage (). The patient was scheduled for a laryngectomy and postoperative radioactive iodine based on these findings.\nUnder general anesthesia, the previous operative thyroidectomy incision was entered and skin flaps were elevated. After the excision of infrahyoid muscles invaded by tumor tissue, the larynx was reached. Left half of the thyroid cartilage and the region towards the left side from the midline of cricoid cartilage up to 5 mm in front of cricoarytenoid cartilage were found to be invaded by the tumor. When the left half of the thyroid cartilage was elevated, left paraglottic space adjacent to cartilage was found to be minimally involved. The patient first underwent supracricoid laryngectomy. Full thickness excision of invaded part of cricoid cartilage was performed by preserving inner mucosa. The upper right half of the thyroid cartilage not invaded by tumor including the superior cornu was shaped and sutured to the site of the resected part of the cricoid, and cricoid framework was recreated and protected inner mucosa was mounted in this framework (). The larynx was closed by cricohyoidoepiglottopexy. There were no complications postoperatively. The nasogastric tube was removed on the 16th postoperative day and the patient was decannulated on the 17th postoperative day. The result of the pathological examination was papillary carcinoma invasion of the thyroid cartilage and cricoid cartilage. Then radioactive iodine treatment was given. Narrowing of the laryngeal air passage and local recurrence were not detected during the 3-year follow-up period. The control CT scan of the neck has shown no evidence of recurrence, and the cartilage used for reconstruction was found to be in the appropriate position allowing an adequate airway passage (). |
Our patient, a 58-year-old Caucasian male, presented to the emergency department with the acute onset of quadriplegia extending from C5 throughout the rest of the pan neuroaxis while awakening from bed the morning of presentation. Upon initial physical examination there were no signs of trauma noted that were significant to the presenting symptoms, additionally, patchy sensation was noted in the upper and lower extremities with clonus in the legs only and hyperreflexia in both arms and legs. Initially, our patient was administered one intravenous dose of methylprednisolone and allowed to enter a state of permissive hypertension which improved our patient's arm strength bilaterally to 2/5, but had no positive effects on the lower extremity paraplegia. As a result of minimal improvement, immediate imaging of the cervical spine was ordered and highlighted a severe cervical stenosis from C3 to C7, as seen in Figure , due to extra-axial posterior compressive spinal mass with cord signal change.\nAdditional imaging of the brain was completed as a precautionary measure and was normal. Upon obtaining full patient history and medical records from nearby hospitals, it was found that our patient was on a prolonged regimen of warfarin for many years due to a prior diagnosis of congestive heart failure without regulation and regular INR/prothrombin evaluation. Our patient also did not follow up with his primary care physician throughout the duration of the warfarin regimen. At presentation, our patient's INR was found to be 5.0 necessitating the prompt correction with intravenous injection of fresh frozen plasma (FFP) and vitamin K. Additional magnetic resonance imaging (MRI) of the thoracic and lumbar spine revealed large compressive acute epidural hematoma posteriorly compressing the thoracic spinal cord from T6 to T10, visualized in Figure , with cord signal change, as well as L4-S1 posterior acute epidural hematoma compressive of the cauda equine, visualized in Figure .\nUpon a complete review of the patient history, presenting symptoms, and radiological imaging results our patient was deemed a prime surgical candidate that would significantly improve both motor function, sensation, and ultimate restoration of our patient's quality of life. Our patient was emergently taken to the operating room for stage 1 of a two-part surgical procedure. Stage 1 involved a posterior T3 to L1 decompression, epidural hematoma evacuation and instrumented fusion. Three days later, our patient was taken to the operating room again for stage 2 of the planed procedure which involved a posterior cervicothoracic C2 to T2 decompression and instrumented fusion with evacuation of acute epidural hematoma. |
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