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We describe the case of a 34-year-old gravida II para l woman, with a gestational age of 26 + 3 weeks at admission, who had a relatively healthy 4-year-old child with her 40-year-old husband of non-consanguineous marriage. She had been on injectable contraception for 2 years and had regular menses for 6 months before the pregnancy. She had antenatal care at a local health center and was vaccinated with tetanus toxoid once and supplemented with iron for 3 months. She was screened for retroviral infection, hepatitis, and syphilis and it was documented nonreactive. She had no anatomic scan at early gestation. She came to Felege Hiwot Referral Hospital with the chief complaint of severe and persistent headache of a day’s duration which was occipital in location associated with blurred vision and generalized body swelling of 1 week’s duration. She had no other danger signs in pregnancy. Her past gynecologic history, medical history, and surgical history were uneventful. She is Amhara by ethnicity. She had no known family history of hereditary or chromosomal disorders.\nHer blood pressure at admission was 180/120 mmHg and pulse rate was 84 beats per minute; her respiratory rate was 22 breaths per minute and she was afebrile. She had pink conjunctiva and non icteric sclera, 24 weeks-sized gravid uterus, no abdominal tenderness, no organomegaly, no sign of fluid collection in her abdomen, and the fetal heart beat was positive. She had no vaginal bleeding or discharge. She had pedal and pretibial edema. She was conscious and oriented to person, place, and time. Her deep tendon reflex was +2 and her motor and sensory examinations showed no motor or sensory problems. Other parts of systemic examinations were normal.\nHer hypertension was controlled with intravenously administered hydralazine 5 mg two doses at our emergency department. In her complete blood count her white blood cells were 7300 cells/micL, hemoglobin of 13.4 g/dl, and platelet count was 169,000 cells/micL. Urine protein dipstick was +2, and liver and renal function tests were done: serum glutamic pyruvic transaminase (SGPT) 89 IU/L (elevated), serum glutamic oxaloacetic transaminase (SGOT) 102 IU/L (elevated), alkaline phosphatase (ALP) 229 IU/L, and lactate dehydrogenase (LDH) 288 IU/L. Total bilirubin was 0.24 mg/dl, albumin was 3.49 g/dl, blood urea and nitrogen was 12 mg/dl, serum creatinine was 0.69 mg/dl, and oral glucose tolerance test was in the normal range. Obstetric ultrasound showed a singleton, alive, intrauterine pregnancy with average gestational age of 26 weeks, there was a single large ventricle with partially formed midline structure (see Fig. ), amniotic fluid index was 13.4 cm, placenta was located anteriorly at the body of the uterus, and the presentation was breech; the fetus had normal four chambers of heart with normal outflow tract.\nAfter blood pressure was controlled (it took 2 hours), she was admitted with the diagnosis of late second trimester pregnancy and preeclampsia with severity feature plus semilobar HPE. Seizure prophylaxis for preeclampsia was given (magnesium sulfate according to World Health Organization guideline), methyldopa 500 mg orally every 8 hours was added, and she was counselled about options of management; the high incidence of associated anomalies, severe morbidities of survivors, and poor prognosis were discussed. Termination was decided and done with misoprostol 100 microgram every 3 hours at the third dose with outcome of 1.1 kg male, alive neonate. On examination of the neonate, there was cebocephaly, hypotelorism, single patent nostril which enabled nasogastric tube 6F, micropenis (8 mm), and unilateral right hand polydactyly with agenesis of middle phalanges of the fifth finger. There was rigidity involving all extremities which resisted extension and flexion (see Figs. , and ).\nAfter basic neonatal care was given (cord tied, airway cleaned, and newborn dried), he was transferred to our neonatal intensive care unit (NICU) but he died 20 minutes after admission to NICU. Immediate cause of death was not known. Following his death, further investigations were not possible for cultural reasons. At third postpartum day, maternal blood pressure was 130/90 mmHg, pulse rate was 78 beats per minute, and respiratory rate was 20 breaths per minute. Her complete blood count showed white blood cells of 12,000 cells/micL, hemoglobin was 11 g/dl, and platelet count was 122,000 cells/micL. Liver function tests showed SGPT of 35 IU/L, SGOT of 12 IU/L, ALP of 359 IU/L, and LDH of 254 IU/L; total bilirubin was 0.56 mg/dl, blood urea and nitrogen was 22 mg/dl, and serum creatinine was 0.8 mg/dl. After she was counselled to have preconception care and prenatal screening in next pregnancy, she was sent home relatively healthy. She was well at postpartum visits and methyldopa was discontinued at seventh postpartum day. |
A 38-year-old female reported to an outside private dental practice office with chief complaint of pain and discomfort on her left anterior mandible region 1 year prior to presentation to our office. Initial orthopantomography (OPG) was obtained and a radiolucent lesion measuring 15 × 10 mm surrounding the roots of mandibular left lateral incisor and left canine was noticed (Figure ). The lesion was hurriedly and erroneously diagnosed as a radicular cyst without conducting any further pulp vitality tests and the two above-mentioned teeth were endodontically treated. A few months subsequent to the initial root canal treatment, the pain did not subside and no change in the size of the radiolucent lesion was obtained; therefore, the left mandibular central incisor was endodontically mistreated additionally without yet again conducting any further pulp vitality tests. These sequelae of mistreatments and persistent pain and discomfort following a third root canal treatment forced the patient to be referred to our oral and maxillofacial surgery private office. There were no significant issues in the patient's medical history upon presentation.\nClinical examination revealed a mild, diffuse enlargement extending from the left mandibular first premolar to the right mandibular canine anteroposteriorly and slightly buccolingually. Overlying skin on the mental region was normal and devoid of any ulcerations, erythema, and rise in temperature. Intraoral examination did not reveal any breach in the mucosa and the integrity of it remained intact. On palpation of the oral mucosa, the enlargement was bony hard on lingual cortex but with a doughy consistency on buccal plate suggesting destruction of the buccal cortical plate, diffuse, and non-tender. Moderate mobility of five teeth including mandibular right lateral incisor to left canine was evident on palpation.\nOrthopantomography (OPG) and cone beam computed tomography (CBCT) were promptly ordered. They depicted a well-defined, multiloculated expansile osteolytic lesion with few small septa spreading inside the lesion. Expansion of the buccal cortex with areas of cortical destruction and root resorption of mandibular left central incisor, right central, and lateral incisors were also noticed. The lesion measured 31.2 × 21.0 mm extending from the left mandibular first premolar to the right mandibular canine (Figures and ). Radiographic preliminary differential diagnosis included central giant-cell granuloma (CGCG), odontogenic keratocyst (OKC), ameloblastoma, odontogenic myxoma, and fibroameloblastoma.\nAn incisional biopsy was then conducted and the histopathological specimen showed an irregular piece of soft tan-pink tissue with fragile consistency. Cut sections demonstrated homogenous myxoid pink surface containing hemorrhagic foci. The microscopic analysis revealed striated muscle fibers, some reactive new and pre-existing bone trabecula and presence of a neo-formed tissue composed of haphazardly arranged stellate and spindle cells with no atypia in an abundant loose myxoid stroma containing few collagen fibrils which was consistent with histopathological features of odontogenic myxoma and therefore a definitive diagnosis was made (Figure ).\nGiven the patient's age, noticeable size of the lesion, its invasive and recurrent characteristics, accompanied with buccal cortex expansion and destruction, enucleation and curettage, and peripheral ostectomy accompanied with concomitant burnishing of teeth roots was planned. After obtaining a written informed consent form from the patient and informing her regarding the poor prognosis of the remaining affected teeth, the surgery was performed under local anesthesia. Surgical procedure incorporated bilateral mental nerve anesthesia, followed by a full-thickness periosteal flap elevation with great vigilance in exposing and preserving bilateral mental nerves and subsequent excision of the lesion mass with curette and periosteal elevator in a piece by piece manner with extreme caution to preserve teeth roots (Figures and ). Teeth roots were then cautiously burnished. Afterward, peripheral ostectomy with a wide margin of 2–8 mm was conducted and the flap was reapproximated and sutured (Figures and ).\nThe excised specimen measuring at 2.5 × 2 × 1.5 cm accompanied with six irregular fragments of the same tissue and small bone particles aggregating to 2 × 1 × 1 cm was then placed in formalin and sent to pathology laboratory which further confirmed the definitive diagnosis of odontogenic myxoma.\nThe patient was informed of the recurrence tendency of the lesion and was advised for annual follow-ups for at least 5 years.\nThe patient's first- and second-year follow-up clinical and radiological examination revealed no recurrence of the lesion (Figures and ). |
A 66-year-old man presented with a 24-h history of severe nausea and abdominal pain. His medical history was remarkable for a rectal adenocarcinoma that was resected 4 months prior to presentation and had left him with a temporary ileostomy. He was otherwise healthy and took no medications. He underwent closure and reanastamosis of his ileostomy 5 days prior to his presentation. Upon arrival to the emergency department, his blood pressure (BP) was 80/40 mmHg and was tachycardic but displayed normal mentation. His examination was remarkable for diffuse abdominal pain and rebound tenderness, but no guarding. Laboratory work revealed acute kidney injury with a creatinine of 180 mmol/L. His pre-operative baseline value was 66 mmol/L. However, his arterial lactate and white blood cell counts (WBC) were normal. The patient received initial fluid resuscitation and underwent a computed tomography scan that showed a dilated cecum (8 cm) and an edematous colon. A Clostridium difficile toxin assay returned positive. He was admitted to our intensive care unit (ICU) for monitoring and medical therapy with intravenous metronidazole and oral vancomycin. The patient improved from a hemodynamic, renal as well as abdominal pain point of view, but his WBC and creatinine started to increase 3 days later without new hemodynamic compromise. An abdominal ultrasound (US) was performed and revealed evidence of thumbprinting (Fig. a), with the thickened colon wall measured at 17 mm (normal values: <3 mm when measured with high-frequency linear array probes) [], as well as pneumatosis intestinalis (Fig. b) In addition, peripherally located air speckles that follow the portal circulation were present (Fig. ). These findings were absent on the baseline US exam we performed upon admission. An abdominal X-ray as well as a repeat computed tomography (CT) scan were obtained to confirm the findings (Fig. d). The scan was performed with oral but not intravenous contrast in the context of acute kidney injury. A diagnosis of failure of medical therapy was made and a discussion with the surgical team led to a tentative colectomy-saving procedure, namely a redo-ileostomy in order to irrigate vancomycin locally. The patient improved within the next 48 h and was discharged from our ICU. |
The recipient was a 72-year-old male with a BMI of 29.5 kg/m2 and a history of type 2 diabetes for over 10 years, hypertension, and chronic hepatitis C, who was diagnosed with a biopsy-proven diabetic nephropathy in 2012. He had been on peritoneal dialysis for approximately one year prior to transplantation from a deceased donor kidney with adult polycystic kidney disease (APKD) in 2019. The recipient had no preexisting human leukocyte antibodies at either class I or class II on repeated screening. The recipient and his sister, who is a physician, were counselled in detail about the donor's diagnosis of APKD and the potential risk of complications, including the growth of the kidney and graft failure, before giving informed consent prior to surgery.\nThe deceased brain death donor was a 40-year-old female with a BMI of 26.5 kg/m2 and a history of APKD who was admitted to the hospital following an intracranial hemorrhage secondary to left cerebral artery aneurysm rupture. The serum creatinine on admission was 0.76 mg/dL, and urine output was around 100 cc/hr. Predonation computed tomography scan showed numerous renal cysts bilaterally, with the largest measuring 5 cm on the right and 3.8 cm on the left. The right donor kidney was used for transplantation and measured 15 × 7 × 4 cm, showed multiple cysts, and had a single vein, artery, and ureter (Figures and ). The donor's Kidney Donor Profile Index (KDPI) was 64%, which has been shown to have around a 75% graft survival rate at 5-year posttransplant []. The time zero biopsy (performed at the time of transplant) showed 4 glomeruli sclerosed out of 41 (9.7%), minimal chronic tubulointerstitial injury, minimal arteriolosclerosis, and mild acute tubular injury. The kidney was pumped on a hypothermic perfusion machine with a flow of 114 and resistance of 0.37 [].\nOur protocol induction therapy includes one dose of thymoglobulin 1 mg/kg, basiliximab 20 mg, and methylprednisolone 500 mg given intravenously at the time of implantation []. Our maintenance immunosuppression protocol includes a steroid-free regimen consisting of tacrolimus and mycophenolate mofetil starting on postoperative day 1. However, for this patient, it was changed to a maintenance immunosuppression consisting of mycophenolate mofetil, prednisone, and belatacept 5 mg/kg intravenous every month in efforts to protect the kidney from tacrolimus nephrotoxicity [].\nThe recipient was discharged home on postoperative day 9 without surgical complications. Renal ultrasound at 4-month posttransplant reported the transplanted kidney that measured 15.5 cm in length with multiple cysts; the largest cyst measured 4.5 × 5.2 × 4.5 cm. At 31-month posttransplant, he had a serum creatinine of 1.6 mg/dL. |
A 47-year-old previously healthy Sinhala female's right foot was bitten by a snake near the back door of her home in the Kegalle district, Sri Lanka. Within seconds, she felt burning pain ascending along that limb, and there was heavy bleeding from the site of bite. Within a couple of minutes, she felt dizziness, nausea, and numbness of the whole body, had profuse sweating and frothy salivation, and was screaming in pain from the site of bite. On the way to the nearby hospital, she started to clench her jaw tightly and limbs became rigid; she was frothing and was not responding for about 5 minutes, indicating a generalized seizure. She arrived at the hospital within 30 minutes. The doctor at the outpatient department decided to administer ASV and directed the patient to an internal medicine ward for that. Physical examination findings at the ward were a pulse rate of 100/minute and blood pressure of 150/90 mmHg, and lungs were clear to auscultation bilaterally with an arterial oxygen saturation of 95% whilst breathing air with no neurological deficit. By this time, the killed snake was brought in and doctors identified it as a HNV; thus, antisnake venom (ASV) was not administered. Even though there was bleeding at the site of the bite even on admission to the hospital, her 20-minute whole blood clotting time, platelet count, prothrombin time and international normalized ratio, and activated partial thromboplastin time and liver function tests were all normal. Urine sample obtained via the catheter showed 50–55 red cells per high-power field, arterial blood gases indicated a compensated metabolic acidosis, and serum sodium and potassium levels were normal. Her urine output was <100 ml for the first 24 hours and serum creatinine rose from 80 μmol/l to 277 μmol/l. She was transferred to the Teaching Hospital, Kandy, on day 2 for further management.\nOn day 2, a bulla developed at the site of the bite, and there was an edema and warmth at the right foot. Complete (full) blood count demonstrated neutrophilic leucocytosis, and the CRP level of the following day was 261 mg/l. Intravenous antibiotics was started to cover the wound infection. Serum creatinine was 377 μmol/l with oliguria on day 2. Serum sodium and potassium levels remained within the normal range from day 1–5. On the day 5, creatine kinase was 75.1 U/l. Regular hemodialysis every other day from day 2 to day 24 and fluid management were started. Oral sodium bicarbonate was started, and management of her acute kidney injury with collaboration of nephrology team continued.\nOn day 3, her blood pressure rose to 160/90 mmHg, and it was controlled by prazosin and nifedipine SR; however, it generally remained on or above 140/90 mmHg until her discharge. She developed bilateral lung crepitations on day 3 that remained for 7 days. She developed bilateral parotid swelling and edema of the right leg on day 3, and it lasted 3 days. Edema below her right knee persisted another 10 days. Her blood picture on day 2 did not show hemolysis and was suggestive of bacterial infection but blood picture on day 5 showed evidence of microangiopathic hemolytic anemia (MAHA), and same changes were there in a blood film taken on day 11, as depicted in .\nHer day 2 hemoglobin level of 10.8 g/dl dropped to 8.4 g/dl on day 5. On day 2, her platelet count was 104 × 109/l and that dropped to nadir of 29 × 109/l in day 6 and was <150 × 109/l until day 20. A consultant in transfusion medicine has assessed her, and blood transfusion and plasmapheresis was performed on day 7. Another four cycles of plasmapheresis followed. Local edema at the site of the bite increased with necrosis (); thus, wound debridement was done on day 7 and followed up by regular wound toilets.\nWe did an electroencephalogram (EEG) on this patient on the earliest available day (day 11) and that was normal. The 2D echocardiogram done on day 17 was also normal.\nThe offending snake's carcass was taken to the Peradeniya University, and an expert on HNV, Dr. Kalana Maduwage, has confirmed it as a Hypnale hypnale. is a photo of the offending snake.\nAs her daily urine output improved to >1000 ml, she was discharged on day 30 and asked to come for a review in five days. She defaulted treatment and was on alternative medication. After developing progressive bilateral ankle edema and exertional dyspnea, she came back again on day 46, and hemodialysis and supportive therapy were restarted at the nephrology unit. On day 49, she had an anterolateral non-ST-elevation myocardial infarction (non-STEMI), and she was managed at the cardiology unit. She had progressive impaired vision of the left eye starting from a few days after the snakebite and could not count fingers held 30 cm in front of that eye on the 46th day. She was referred to the eye unit, there was bilateral optic disc edema more on the left, the patient was diagnosed of left anterior ischemic optic neuropathy (AION), and steroid therapy was started. Her erythrocyte sedimentation rate and contrast-enhanced computed tomography (CECT) brain done on day 53 were normal. is a photograph of fundi of this patient.\nShe had two episodes of seizures on day 76, and we suspected a possible relationship to her envenomation. The opinion of the neurology team regarding three seizures was obtained. Repeated EEG and CECT brain were normal. Despite being on calcium carbonate 500 mg plus 0.25 μg 1-alpha-hydroxycholecalciferol daily from day 46, her serum calcium level was low (1.8 mmol/l). Last two seizures were attributed to hypocalcemia due to chronic kidney disease following HNV envenomation, and daily calcium carbonate dose was increased to 500 mg thrice daily. After three months, she was diagnosed of end-stage renal disease by nephrology team and on hemodialysis once in four days and was searching for a kidney donor at six months. |
A 47-year-old woman presented to our clinic and complained of a left upper eyelid lesion that had increased slowly in size over the past three years. The lesion was 1.0 cm in size. It was round shaped, circumscribed elevated and had brown pigmented color (Figure 1 ). The appearance was typical of a seborrheic keratosis. Her visual acuity and eyelid movements were normal.\nThe procedure is performed under local anesthesia with intravenous sedation and magnification. Upper eyelid tumor is marked with 2 mm margin. A line is drawn on the eyelid at the level of the lid crease. Then, the advancement flap of the anterior lamella is outlined with two Burow’s triangles marked for excision, one triangle medial or lateral to the defect and the second diagonal to the first, above the lid crease (Figure 2 ). An incision is then made through the skin and the subcutaneous tissue of the lesion. The lesion was excised with a 2 mm free margin. The triangles’ boundaries are cut with a scalpel, dissected, and mobilized with blunt scissors (Figure 3A ). The subcutaneous tissue at the edges of the defect is undermined in the subdermal plane to minimize the tension at the suture lines. An advancement flap of the skin and orbicularis of the upper eyelid was undermined, elevated, and advanced inferiorly over the defect (Figure 3B ). Interrupted buried 6/0 nylon sutures are used to approximate the dermis and subcutaneous tissue and close the defect completely (Figure 4 ). Topical antibiotic ointment is applied twice daily for 7 days. The sutures are removed in 10 days. Histopathological examination of the tumor revealed seborrheic keratosis and confirmed that the margin was free of tumor. The patient has been followed up for six months with no evidence of recurrence and has no concerns with eyelid function. Moreover, this treatment produces good aesthetic results (Figure 5 ) and increased patient satisfaction. |
A 40-year-old gravida five, para four woman presented for workup and management of abnormal uterine bleeding. Her past medical history was significant only for hypertension and anemia. On review of her social history, she admitted to drinking six packs of beer on the weekends but denied further substance use. She denied previous treatments for her bleeding including any previous intrauterine device usage.\nUltrasonography revealed a 7 cm fundal fibroid with otherwise normal pelvic anatomy. She was initially offered medical management of her bleeding. She declined any medical treatment and strongly desired definitive surgical treatment. She then underwent a total vaginal hysterectomy with adnexal conservation. Due to the large size of the uterus, a myomectomy was performed to facilitate vaginal removal. Her postoperative hospital course was relatively uncomplicated and she was discharged home on postoperative day three.\nOn postoperative day ten, she presented to the Emergency Department (ED) for fever, worsening abdominal pain, and new onset of nausea and vomiting. In the Emergency Department, she was tachycardic and tachypneic but afebrile. Her exam was significant for abdominal tenderness to minimal palpation, vaginal cuff erythema, and significant tenderness to palpation of the vaginal cuff. Lab work showed an elevated white blood cell count. She was admitted for management of presumed pelvic infection.\nA CT of the abdomen and pelvis was obtained and showed a 6.2 x 9.7 cm pelvic abscess adjacent to the vaginal cuff (Figures and ).\nInterventional Radiology placed a drain into the abscess and the patient was started on IV piperacillin/tazobactam. She was transitioned to oral amoxicillin/clavulanate potassium after four days on intravenous antibiotics and her drain was removed on hospital day 5. Vaginal wound cultures remained pending; however, due to continued clinical improvement on the oral antibiotic regimen, she was discharged home on hospital day 5 with a two-week course of amoxicillin/clavulanate potassium.\nThe patient then returned for her outpatient visit approximately one week later. The results of the vaginal wound cultures revealed a large growth of Actinomyces meyeri. The patient's case was discussed with an Infectious Disease (ID) specialist who recommended an additional two-week course of amoxicillin/clavulanate potassium.\nThe patient then returned to the ED on postoperative day 25 for pleuritic chest pain with mild cough but denied gynecologic complaints. She reported compliance with the oral amoxicillin/clavulanate potassium regimen. Exam and lab work were unremarkable. A chest X-ray showed left basilar heterogeneous opacities, likely subsegmental atelectasis. A CT angiogram was obtained due to concern for a possible pulmonary embolism (PE). The imaging was negative for a PE; however, it was concerning for possible pneumonia. The patient was discharged home with a five-day course of levofloxacin for treatment of pneumonia.\nOn postoperative day 27, the patient represented to the ED with worsening shortness of air and chest pain. Again, she reported compliance with her antibiotic prescriptions. Exam and lab work were again unremarkable. A repeat chest X-ray showed a slight progression of right basilar heterogeneous opacities thought to be infectious. Her antibiotic regimen was again discussed with ID specialists and an intravenous antibiotic regimen was felt preferable to an oral antibiotic course. She then completed an outpatient two-week course of IV ampicillin/sulbactam as recommended.\nOn postoperative day 37, a repeat CT of the abdomen and pelvis showed near complete resolution of the previous pelvic abscess. HIV testing was obtained and returned negative result. She reported significant improvement of her symptoms. She was placed on a six-month course of oral amoxicillin per ID recommendations with plans for continued follow-up in their clinic, as well as with gynecology. She has not shown any signs of recurrent infection after approximately 1 year of follow-up. |
A 32 year-old female presented with a gradually progressive right neck swelling since 4 months. Wedge excision biopsy revealed poorly differentiated metastatic carcinoma. Patient was referred for WB PET/CT study to look for a primary site. In addition, a complementary WB FDG PET/MRI was done after the PET/CT. WB PET/CT and WB PET/MRI revealed metabolically active right nasopharyngeal thickening [Figure and ] as the possible primary site and metabolically active bilateral cervical and retropharyngeal nodes which were metastatic [Figures and ]. In addition, MRI revealed ill-defined marrow changes involving the pterygoid plates [] with mild tracer uptake suggesting involvement, not appreciable on the CT images. Also, enhancing perineural thickening was seen in the right foramen ovale extending intracranially in the right cavernous sinus region [] which was also not discernable on CT images. A focal marrow lesion was identified in the lateral mass of C1 vertebral body with mild tracer uptake identified retrospectively on PET images and was possibly metastatic []. No identifiable lesion was seen on the corresponding CT images.\nNasopharyngeal carcinomas not only have the propensity for being locally advanced at presentation including intracranial extension by either eroding the skull base or perineural extension through the neural foramina in the skull base when the disease is staged as T4,[] they may also be metastatic at presentation in about 5-8% of cases, with the common sites being regional nodes, bones, lung, liver, and retroperitoneal nodes.[]\nMRI is an established modality for evaluating the extent of head and neck malignancies and supersedes CT in delineating lesions better because of its excellent soft tissue contrast and delineates its local extent better than any other modality, detects marrow lesions much better (with or without cortical involvement), and can detect unsuspected perineural intracranial extension.\nIn this case, an apparent T2N2 disease was upstaged to T4N2M1 due to the supplemental information from the PET/MR which also altered the disease prognosis. |
A 14-month-old girl presented to a private hospital with bilateral DDH. Plain radiographs showed a complete dislocation of both hips (Fig. ). She underwent staged open hip reduction and acetabuloplasty. The left hip was treated first, followed by the application of a hip spica cast for 6 weeks. The right hip was operated afterward, followed by immobilization in a hip spica for 6 weeks and then in a broomstick cast for another 6 weeks. After the removal of the cast, the right hip was dislocated again. The patient was treated with closed reduction under anesthesia and immobilized in a hip spica for another 2 months. Upon removal of the cast, the right hip had dislocated again, and there was stiffness in both hips.\nAt the age of 20 months, the patient was referred to our hospital with severe stiffness of both hips and a dislocated right hip (Fig. ). Because of the stiffness, we referred her to the physiotherapist to improve the range of motion (ROM) in both hips. Four months later, the ROM was acceptable (Table ).\nA computed tomography (CT) scan revealed a central acetabular bony prominence posterior to the triradiate cartilage that measured around 1.5 cm in width at its base and had an 8-mm long tooth-like end projecting into the joint. The femoral head was dislocated in the posterior-lateral direction (Fig. ). The parents were informed about the nature of their daughter's condition and the purpose of performing our novel operation, which, to our knowledge, has never been performed before, and they provided consent to perform our technique and for publication of the case.\nUnder general anesthesia, examination and arthrography of the right hip were performed and revealed an obliterated right acetabulum (Fig. ). We accessed the joint through the previous incision and excised all fibro-fatty tissue obliterating the acetabulum. Consecutively, we removed the bony prominence with its central spur and deepened the floor of the acetabulum using a dental bur to accommodate the head of the femur.\nWe decided to insert a spacer made of bone cement inside the acetabular cavity to prevent ankylosis. We formed the cement into a ball about the same size and shape of the head of the femur. We secured the position of the cement ball in the acetabulum with multiple non-absorbable sutures to ensure the healing of the acetabular surface (Fig. ).\nA hip spica cast was applied for 2 months to allow the acetabular bone to heal and to allow fibrocartilage to cover the bone so that the smoothened surface was ready to accommodate the head of the femur.\nEight weeks later, we removed the spacer and explored the acetabulum intraoperatively. We found that it was healed completely and covered with fibrocartilage of an optimal depth and congruency to accommodate the femoral head. The hip was reduced, and a capsulorrhaphy was performed. A hip spica cast was applied for 4 weeks, followed by a broomstick cast for another 4 weeks. After removing the cast, arthrography of the hip revealed a concentrically reduced hip with acceptable coverage of the femoral head (Fig. ).\nMild stiffness was noticed in both hips postoperatively, and we referred the patient again to the physiotherapist for ROM exercises. During the follow-up, a significant improvement in her gait and the ROM of both hips was noticed. Seven years after the surgery, she had almost full ROM of both hips with normal gait (Table ). She could cross her legs and perform daily activities without any restrictions (Fig. ). The radiographic assessment showed concentrically reduced hips with good acetabular coverage and no ankylosis (Fig. ). |
A 65-year-old African man presented to our surgical emergency with a week history of bleeding per urethra following nocturnal penile erections. He admitted to have introduced a broken transistor radio antenna into his urethra a week before to serve as an improvised ‘itchstick’ to ease a bothersome itchy urethral condition. The object subsequently migrated proximally out of reach. He made several unsuccessful attempts at self-retrieval prior to presentation and remarkably reported no difficulty with voiding. He had no history of psychiatric disorder, alcoholic or drug intoxication and had no prior history of similar or other self-injurious behaviour. He, however, had a bothersome perineal and itchy urethral condition which was worse at nights.\nPhysical examination revealed a hard rod-like mass in the proximal part of the penis extending to the perineum; the mass had only minimal mobility. A pelvic X-ray done showed a rod-like radio-opaque foreign body in the region of the penile and bulbar urethra []. A diagnosis of foreign body in the male urethra was made. After obtaining informed consent, removal of the foreign body was planned. Under caudal regional block with 2% Xylocaine gel instilled into the urethra, an attempt at ‘milking out’ the foreign body was made but failed. Endoscopic removal was considered but deemed inappropriate as the distal end of the object was jagged (irregular) and appeared impacted in sub-urethral tissue. External urethrotomy of the anterior urethra was performed with the incision made into the proximal penile urethra at the distal tip of the foreign body which was then easily extracted []. The extracted foreign body, an aluminium tube transistor radio antenna measured 13 cm in length [], urethral swab for microscopy and culture was taken and urethrotomy closed over a 16 Fr Foley's catheter. The patient was discharged after 48 hours and the catheter removed after a week at the clinic. The urethral swab grew Klebsiella pneumoniae and he was treated with sensitive antibiotics, tabs Augmentin for 2 weeks. At 1 month follow-up, he complained of a decreased but persistent urethral itch. Four-bottle urine microscopy revealed numerous pus cells, few epithelial cells and spermatozoa and only scanty growth of K. pneumoniae. No worm or ova was found. Antibiotics (Augmentin) were continued for a further 2 weeks and empirical treatment for pin worm infestation was given (Albendazole) resulting in resolution of symptoms. A psychiatric evaluation revealed him psychologically normal. At 9 month follow-up, he had no recurrent itch and normal voiding with good uroflow. |
A 46 years old woman was admitted for elective surgery with the diagnosis of multinodular goiter. The specialist endocrinologist established the indication for surgical treatment and the present euthyroid status of the patient.\nThe clinical exam revealed a rubberlike, rather irregular and big goiter, developed predominately on the left side, apparently going beyond the anterior margin of the sternomastoid, but with no signs or symptoms of local compression. No notable co-morbidities were present.\nThe operating team consisted of one consultant with special interest in endocrine surgery and medium – high volume of thyroid and parathyroid cases (50 – 60 operations per year) and 2 registrars.\nUnder general anesthesia, the surgical team proceeded to total thyroidectomy with routine identification of the inferior laryngeal nerve and, at least, superior parathyroid glands, with the vascular cutting and sealing done with Harmonic Ace CS14S. Although the left lobe was significantly bigger (), the dissection and identification of the recurrent laryngeal nerve were straightforward. On the right,we first mobilized the lobe anteriorly and medially by severing the middle thyroid vein and the vessels of the upper pole. We identified the inferior thyroid artery and its branches and, as we usually do, we commenced the search of the ILN, first cranially and then caudally, being unable to find any nervous structure resembling ILN in the tracheo-esophageal groove. Therefore, we thought of the possibility of the ILN being non-recurrent, identified the vagus nerve and followed it cranially. In this way, we encountered a transverse structure emerging from the vagus, approximately at the level of the cricothyroid junction and entering the larynx at the lower margin of the cricothyroid muscle. We carefully dissected its entire length and preserved this structure () that proved to be a non-recurrent ILN.\nThe postoperative course was uneventful, without any sign of hypocalcaemia, without dysphonia and the repeat indirect laryngoscopic exam was normal. |
A 67-year-old woman diagnosed with asymptomatic WM 3 years ago presented with gradual vision deterioration the past 3 months. Ophthalmologic examination revealed bilateral reduction in visual acuity (7/10) and bilateral optic disc swelling which was more prominent in the left eye. Clinical examination was otherwise unremarkable. The patient reported no weight loss or B-symptoms. At the time of initial WM diagnosis, the bone marrow biopsy showed 15% infiltration of M(κ)-clonal lymphoplasmacytic cells and the serum immunofixation was positive for monocloncal IgM, while serum IgM levels were 147 mg/dL. Laboratory examinations were within normal ranges with no signs of anemia or thrombocytopenia, whereas clinical and imaging assessment did not reveal any sites of lymphadenopathy or hepatosplenomegaly. Therefore, the patient was defined as asymptomatic and had been under follow-up every 4 months. Two years postinitial diagnosis a new diagnostic bone marrow biopsy revealed an increase in clonal infiltration up to 40% and genetic tests performed both in the bone marrow and in the serum cell-free DNA did not detect any MYD88 or CXCR4 mutations. The presence of mutations in the aforementioned genes was evaluated by allele-specific polymerase chain reaction and direct sequencing, as previously described.[ Serum IgM levels had been gradually increasing as well with no other concurrent symptomatology and reached at 723 mg/dL at the time of presentation with visual loss (serum M-peak = 0.61 g/dL) (Fig. A). Her medical history was also remarkable of right eye cataract surgery, thyroidectomy, and T4 hormone replacement therapy, but she had no history of migraine. She was also current smoker with mild hypercholesterolemia. Due to the presence of risk factors for cardiovascular disease, we performed initially an extensive workup including electrocardiogram, cardiac ultrasound, sonography of the carotid, and vertebral arteries that revealed no clinically significant abnormalities. Coagulation studies were within normal limits. No serum autoantibodies were detected. Apart from an erythrocyte sedimentation rate of 140 mm at 1st hour, there were no other clinical signs of giant cell arteritis. Although the IgM was relatively low, we evaluated serum viscosity, which was 2.0 cp and, thus, we ruled out hyperviscosity syndrome. MRI of brain revealed bilateral swelling of optic nerves extending from the retina to the optic chiasm and swelling of the left optic tract. Patchy enhancement of optic nerves was also shown upon intravenous contrast administration (Fig. A). MRI of the spine indicated no abnormalities. Subsequently, we performed a lumbar puncture; flow cytometry of the CSF revealed the presence of κ-light chain restricted, monoclonal B-lymphocytes accounting for the 86% of CD45 positive cells along with CD3(+)CD4(+) and CD3(+)CD8(+) T-lymphocytes. CSF protein electrophoresis showed a monoclonal band in the gamma region and immunofixation was positive for IgM and kappa light chain (Fig. B). MYD88 status was indeterminate. Thus, the diagnosis of BNS was established. Patient was initially treated with intrathecal methotrexate 15 mg twice weekly and systemic chemotherapy with HyperCVAD. However, after the course 1 of the 1st cycle she experienced grade 3 neutropenic fever that eventually resolved with granulocyte colony-stimulating factor and intravenous antibiotics. Following 2 intrathecal methotrexate infusions, CSF flow cytometry did not detect any cells, whereas the patient reported improvement in visual acuity. Therefore, we opted to start treatment with IV rituximab 375 mg/m2 every 4 weeks and per os ibrutinib 420 mg daily. Six months posttreatment initiation the patient has great visual improvement with resolution of papilledema and bilateral optic nerve swelling (Fig. B), whereas IgM levels have been reduced to 25.6 mg/dL, the serum protein electrophoresis (Fig. C) and immunofixation are negative for monoclonal component and bone marrow biopsy shows no evidence of disease infiltration. Therefore, the patient has achieved clinical, hematologic, and radiologic complete response. No adverse events have been reported except for transient diarrhea grade 1 probably related to ibrutinib. After completing 1 year treatment with rituximab, the patient discontinued rituximab and remains on ibrutinib monotherapy in complete response and with no major adverse events. |
The case presented is that of a 50 year-old Caucasian female with isolated recurrence of metastatic melanoma to the subcutaneous tissues of her left thigh. Ten years prior to her current presentation, she underwent a wide excision and skin grafting of her left distal thigh region and a superficial left groin lymph node dissection for a 2.3 mm malignant cutaneous melanoma with 22 negative lymph nodes. She received no adjuvant therapy and subsequently continued routine follow-up by her surgeon.\nTwenty-five months prior to her current presentation, she developed two skin nodules located approximately 4 cm distal to the previous skin graft on her distal thigh region. A wide excision of her left distal thigh region was subsequently performed. A diagnostic whole body 18F-FDG PET scan was performed, utilizing an intravenous injection of 13.9 mCi 18F-FDG, that revealed a solitary hypermetabolic focus within the anteriomedial left mid thigh region (peak SUV of 40.1) which was not palpable on clinical examination. No other hypermetabolic foci were identified elsewhere in her body. As a result, the patient subsequently (23 months prior to her current presentation) underwent isolated left lower extremity hyperthermic limb perfusion with melphalan and a concomitant left deep groin lymph node dissection.\nA six-month follow-up (17 months prior to her current presentation) diagnostic whole body 18F-FDG PET/CT scan was performed, utilizing an intravenous injection of 16.3 mCi 18F-FDG, and redemonstrated a solitary hypermetabolic focus within the subcutaneous tissues of the anteriomedial left mid thigh region, with a peak SUV of 7.6. No other hypermetabolic foci were identified elsewhere in her body. Subsequently (15 months prior to her current presentation), CT guided wire localization and wide excision of the nonpalpable subcutaneous focus of disease in her anteriomedial left mid thigh region was performed.\nThe patient continued routine follow-up by her surgeon. One month prior to her current presentation, the patient underwent a routine follow-up diagnostic restaging whole body 18F-FDG PET/CT scan (Figure ) on a Siemens Biograph 16 PET/CT unit (Knoxville, TN, USA) utilizing an intravenous injection of 14.8 mCi 18F-FDG. The scan demonstrated three foci of hypermetabolic activity within the subcutaneous tissues of the anterior left thigh region. Two closely approximated hypermetabolic foci (peak SUV of 25.3) were located in the subcutaneous tissues of the anteriomedial left mid thigh region and one hypermetabolic focus (peak SUV of 3.4) was located in the subcutaneous tissues of the anterior lower one-third of the left thigh region. No other hypermetabolic foci were identified elsewhere in her body. On clinical exam, no visible or palpable abnormalities were noted in the left thigh region or elsewhere.\nOn the day of surgery, a dose of 12.8 mCi 18F-FDG was injected intravenously into a peripheral vein at approximately 80 minutes prior to the start time of the surgical procedure, as per our 18F-FDG and PET/CT protocols previously described [,]. The patient was subsequently taken to the operating room. Intraoperatively, a handheld gamma probe (Neoprobe neo2000 unit, Neoprobe Corporation, Dublin, Ohio, USA) was used to attempt localization of the three areas of increased 18F-FDG uptake within the left thigh region. Initially, one predominant site of 18F-FDG activity was transcutaneously identified with the gamma probe within the anteriomedial left mid thigh region. Surgical excision of the subcutaneous tissue (measuring 16.0 × 7.0 × 3.0 cm in size) of this area was undertaken. Post-excision evaluation of the excision bed within this region was performed with the gamma probe and revealed residual increased 18F-FDG activity above background. Therefore, gamma probe directed excision of additional deeper subcutaneous tissue (measuring 3.5 × 2.0 × 1.0 cm in size) of this area was undertaken. Further post-excision reevaluation of the excision bed within this region was again performed with the gamma probe and revealed no 18F-FDG activity above background.\nAttention was then directed to the anterior lower one-third of the left thigh region where the third hypermetabolic focus was located in the subcutaneous tissues based on the previous diagnostic whole body 18F-FDG PET/CT scan. With the gamma probe, difficulty was encountered transcutaneously distinguishing a finite site of 18F-FDG activity that was distinct from that of the underlying background muscular and vascular blood pool 18F-FDG activity. Therefore, intraoperative ultrasound was performed using a Hitachi HI VISION™ 6500 ultrasound system (Hitachi Medical Systems America, Inc., Twinsburg, Ohio, USA) with a variable frequency linear transducer EUP-L54M (range 10.0 to 13.0 MHz) (Hitachi Medical Systems America, Inc., Twinsburg, Ohio, USA). An 8 mm hypoechoic ultrasound lesion (Figure ) was identified within the subcutaneous tissues coinciding with the area of generally increase 18F-FDG activity within the anterior lower one-third of the left thigh region seen on the previous diagnostic whole body 18F-FDG PET/CT scan. More localized increase 18F-FDG activity was verified within this same region with the gamma probe and surgical excision of subcutaneous tissue (measuring 4.2 × 4.0 × 1.0 cm in size) of this area was undertaken. Post-excision evaluation of the excision bed within this region was performed with the gamma probe and revealed no 18F-FDG activity above background. Ex vivo gamma probe evaluation revealed increased 18F-FDG activity within the excised subcutaneous tissue. Likewise, ex vivo ultrasound evaluation revealed the corresponding hypoechoic ultrasound lesion within the excised subcutaneous tissue.\nAll three resected specimens were then transported to the nuclear medicine department and imaged with the clinical PET/CT scanner (Figure ) at a time of approximately 210 minutes after the original 18F-FDG injection. Specimen PET/CT imaging revealed the presence of three hypermetabolic foci, corresponding to the three sites of excised subcutaneous tissue that represented the three hypermetabolic areas in the subcutaneous tissues of the left thigh region that were originally visualized on the preoperative diagnostic whole body 18F-FDG PET/CT scan. The specimens were then transported back to the operating room in order to be sent to and processed by surgical pathology for standard pathologic evaluation.\nPostoperatively, the patient was recovered uneventfully in the post-anesthesia care unit. After postoperative standard stabilization and recovery (at a time of approximately 120 minutes after the completion of the surgical procedure and at a time of approximately 340 minutes after the original 18F-FDG injection), she was subsequently taken to the nuclear medicine department and re-imaged with PET/CT scan without administration of an additional dose of 18F-FDG. The postoperative PET/CT scan demonstrated no residual sites of hypermetabolic activity, verifying excision of the all three previously visible sites of hypermetabolic activity within the left thigh region (Figure ).\nPathologic evaluation of the resected specimens revealed three separate sites of malignant melanoma. This included a 18 mm nodule of malignant melanoma that corresponded to the first excised focus, representing the larger of the two areas in the subcutaneous tissues of the anteriomedial left mid thigh region which demonstrated a peak SUV of 25.3 on the preoperative diagnostic whole body 18F-FDG PET/CT scan. Likewise, an additional 8 mm nodule of malignant melanoma was identified that corresponded to the second excised focus, representing the smaller of the two area in the subcutaneous tissues of the anteriomedial left mid thigh region which demonstrated a peak SUV of 25.3 on the preoperative diagnostic whole body 18F-FDG PET/CT scan. Finally, a 6 mm nodule of malignant melanoma was identified that corresponded to the third excised focus, representing the area in the subcutaneous tissues of the anterior lower one-third of the left thigh region which demonstrated a peak SUV of 3.4 on the preoperative diagnostic whole body 18F-FDG PET/CT scan.\nAt the time of the publication of this Technical Innovations report, the patient is currently six months out from the above-described innovative multimodality approach for tumor localization and verification of resection of all sites of hypermetabolic activity and appears to be without any evidence of further disease. |
A 37-year-old male patient presented to the Gulhane Military Medical Academy Haydarpasa Training Hospital (Istanbul, Turkey) with a one-year history of a growing nevus located in the right retroauricular region that had been undergoing changes in color. An excisional biopsy was performed on the suspicious skin lesion. Pathological examination suggested that the lesion was a nodular-type malignant melanoma, Clark level IV and with a Breslow thickness of 3 mm (). Neck ultrasonography revealed two lymphadenopathies in the right jugular chain, the largest measuring 11 mm. Thoracic and abdominal computed tomography and bone scintigraphy did not reveal the presence of metastasis. Lymphoscintigraphy of the right neck was performed using Tc99m. The procedure revealed uptake in two foci located in the right retroauricular region and the right upper cervical lymph nodes. The patient then underwent extensive re-excision and sentinel lymph node biopsy. Metastasis of the malignant melanoma was detected in one of the sentinel lymph nodes.\nThe patient subsequently underwent modified radical neck dissection and pathological examination revealed the presence of papilliary thyroid carcinoma metastasis in four out of 38 lymph nodes and also revealed reactive hyperplasia in the remaining 34 lymph nodes. The patient subsequently underwent total thyroidectomy and the pathological examination revealed a classical variant of papillary thyroid carcinoma (). Imaging examinations did not reveal distant organ metastasis associated with either primary tumor. The patient was initially intravenously administered with high-dose interferon therapy (20 MU/m2) five days a week, for four weeks, to treat the malignant melanoma. In order to treat the papillary thyroid carcinoma, the patient was administered with low-dose (5 mci) radioactive iodine therapy in the second week of the high-dose interferon therapy. The high-dose interferon therapy was followed by moderate-dose interferon therapy (10 MU/m2), which was intravenously administered three days a week for 48 weeks. The patient was also administered with high-dose radioactive iodine (150 mci) therapy in the eighth week of the moderate-dose interferon therapy.\nThe two primary tumors observed in the present patient were successfully treated. No serious side-effects were observed during therapy, with the exception of a fever caused by the high-dose interferon therapy. The patient continues to be followed up by the Gulhane Military Medical Academy Haydarpasa Training Hospital and is disease-free at present. Moderate-dose interferon therapy and thyroid hormone replacement therapy continue to be administered. |
A 36-year-old male presented to the outpatient department with a history of low backache for the past one year, associated with typical claudication symptoms, left-sided unilateral numbness, and paresthesia of the foot. He gave no history of radicular pain, but he complained of progressive reduction in claudication distance. He had experienced a sudden deterioration in gait along with urinary incontinence, following a trivial traumatic fall, one week before presentation. There was no history of constitutional features. On examination, he had a bilateral high-stepping gait due to foot drop. Symmetrical weakness of both the L4 and L5 roots (MRC grade 0/5) and partial weakness of the S1 root (MRC grade 3/5) was noted with nondermatomal sensory disturbances. Deep tendon reflexes of the lower limbs were absent bilaterally. Though anal tone was normal, saddle anesthesia was present. Postvoidal ultrasonogram of the urinary bladder revealed a residual urine volume of 250 ml, thus confirming a neurogenic bladder.\nPlain radiography of the lumbar spine did not show any gross feature of instability and was inconclusive (). Magnetic resonance imaging (MRI) of the lumbar spine revealed a solitary intraspinal posterior epidural lesion of 1.8 × 1.5 × 0.5 cm at the L2-L3 level with heterogeneous signal intensities and adjacent epidural fatty hypertrophy contributing to severe canal stenosis (3 mm). The cauda equina was severely compressed and was almost not visible (). Owing to the heterogeneous signal intensities, computerized topography (CT) was performed which revealed the presence of an osseous lesion attached to the right L2 inferior articular process causing severe secondary canal stenosis (). Considering the recent-onset neurological deficit, the patient was advised surgical decompression and excision biopsy at the earliest. The patient was positioned prone on a Relton Hall frame under general anesthesia. A standard midline posterior approach was employed and L2 and L3 lamina were exposed. The spinous process was removed and using a motorized burr, a rectangular trough was created surrounding the lesion. The lamina was thinned out using a burr to avoid further insult to the dural sac, and then using a Kerrison ronguer, laminectomy was completed all around the lesion under microscopic guidance (). A small osteotome was used to remove the attachment on the right side which required partial removal of the facet joint. The lesion was then held, and the adherent soft tissues beneath the lesion were removed, resulting in en bloc removal and complete decompression of the cauda equina. Fusion was performed, as the procedure involved partial facet joint resection. The lesion was sent for histopathological examination. There were no adverse events or postoperative complications. The patient was mobilized with bilateral orthoses, and bladder training was initiated.\nAt 4 weeks, there was an improvement in his urinary symptoms, and by 12 weeks partial neurological recovery (MRC grade 3/5 motor power) in bilateral L4 and L5 was observed. The S1 root power increased by one grade (MRC grade 4/5). His gait improved thereafter, and he was able to return to his normal activities by 6 months with further neurological improvement by 1 grade in all roots.\nThe gross specimen measured 2 cm × 1.5 cm × 1.5 cm. It was greyish white in color and firm to hard in consistency (). It had a well-defined capsule and had the feel of particulate materials on cut sections. Histopathological examination revealed the presence of bone, cartilage and ligamentous tissue, and zones abutting all these composed of mature adult-type encapsulated adipose tissue (Figures and ). This confirmed the diagnosis of benign osteolipoma. |
A 69-year-old male, known case of chronic angle closure glaucoma with advanced glaucomatous neuropathy, was on medical management for his raised intraocular pressures. He was highly noncompliant with his medications and subsequently went on to develop vascular occlusions (superotemporal branched retinal vascular occlusion in the right eye and central retinal vascular occlusion in the left eye) due to sustained high intraocular pressures in both his eyes in a sequential manner followed by NVG in the left eye.\nThe patient had undergone phacotrabeculectomy with Mitomycin C MMC in both the eyes for chronic angle closure glaucoma 6 months back. There was a diffuse functional bleb with an intraocular pressure of 12 mmHg in right eye. The bleb was flat and vascularized in the left eye and intraocular pressure in the left eye was 36 mmHg. Ahmed Glaucoma Valve with donor scleral patch graft was implanted along with intravitreal Inj Avastin for NVG in the left eye. One month postoperative follow-up of the patient revealed a well-functioning implant with an exposed scleral graft and overlying conjunctival melt []. The patient was put on Cap Doxycycline (100 mg bd) orally, eye drop Prednisolone acetate 8 times/day, E/D Tear substitute 8 times/day and continued eye drop Moxifloxacin 4 times/day, with a review on alternate days. After 2 weeks, there was complete epithelial healing over the exposed scleral graft [], anterior chamber was deep and the tube was functioning well. Doxycycline was continued once daily orally further for a period of 4 weeks and then stopped. During the 4 months follow-up period, the tube remained covered by intact conjunctival epithelium. On the last follow-up visit, the patient had a visual acuity of 6/18 in the left eye, the tube remained covered by intact conjunctival epithelium, and intraocular pressure was 13 mmHg on eye drop Brimonidine and the patient was advised a 3-monthly review []. |
An 18 yr old male patient presented with pain in the left upper posterior region of the jaw since 2 months. The pain was dull and intermittent in nature that aggravated on mastication or pressure and relieved on taking medication. It was occasionally associated with pus discharge. Past dental history revealed that he had undergone extraction of upper left posterior tooth due to caries. The patient also reported with good systemic health.\nThere were no conflicts of interest and no financial issues regarding this case.\nThere was no facial asymmetry seen extra orally. On intraoral examination, no significant swelling or vestibular changes were evident. Clinically, 27 and 28 were missing. [] The alveolar bone over that region appeared normal and was tender on palpation\nThe intraoral periapical radiograph showed irregular radiopaque masses distal to 26 in the alveolar bone. OPG showed a well-defined radiolucency present in the 27, 28 regions that extended from distal of 26 to maxillary tuberosity region surrounded by a white sclerotic border with intermittent irregular radiopaque masses within. Vertically impacted 28 were seen embedded in the left zygoma region. [] based on the clinical and radiological findings, a provisional diagnosis of an odontogenic tumour was wellthought of. Surgical intervention was performed and the tissue specimen was subjected to histopathological assessment [].\nOn histopathological examination, the H & E section revealed a highly fibrocellular connective tissue stroma consisting of numerous strands of odontogenic epithelial islands composed of peripherally arranged tall columnar cells. The stroma consists of collagenous bundles with plump shaped fibroblasts and endothelial lined blood vessels with extravasated RBCs. Deeper section showed the presence of few basophilic calcified areas []. Consider the clinical, radiological and histopathological features, final diagnosis of OF was given. The patient was kept under observation initially for 10 d and periodic follow-up was done for a year. No recurrence was reported. |
A 65-year-old female with no past medical history presented to the emergency department on 5/12/2021 with a complaint of periumbilical abdominal pain that radiated to the back. The pain was described to be tearing and sharp in nature, measuring 10/10 on the pain scale. The pain started suddenly after waking up earlier that morning. The patient presented six hours from the onset of symptoms. Upon presentation, the patient was evaluated by the emergency room staff vitals were obtained, and appeared to be within normal limits except for blood pressure in 150's systolic and over 90's diastolic. A full set of labs were obtained, which indicated a white blood cell count of 8.1 K/uL, hemoglobin of 15.5 g/dl, hematocrit of 45.4%, and platelets of 198 K/uL. Basic metabolic panel and coagulation studies were within the normal range.\nComputed tomography chest, abdomen, and a pelvis with intravenous contrast were obtained and indicated infrarenal abdominal aortic tortuosity as well as 68 mm abdominal aortic aneurysm with dissection. The infrarenal abdominal aorta was proximally tortuous leading into the fusiform infrarenal abdominal aorta aneurysm and short segment dissection with a wide intimal defect with equal opacification of the true and false lumen. The aneurysm extended to the level of the bifurcation. There were two left and one right renal arteries that were patent. Vessels were coursing along the left lateral aspect of the aneurysm (Figure -). There were concerns for impending rupture.\nGiven the concerns for impending rupture, the patient required immediate surgical intervention. Unfortunately, the patient was not a candidate for endovascular therapy due to the juxta-renal nature of the aneurysm and the lack of adequate proximal neck. Therefore, the patient underwent an open retroperitoneal repair, where she was found to have a large dissection with a prominent intimal flap as indicated in Figure . A 16 mm Dacron graft was placed and the aneurysm sac was closed over the graft using interrupted 3-0 vicryl sutures. The patient tolerated the procedure well, was successfully extubated, and was transferred to the ICU for hemodynamic monitoring. The patient did very well and was successfully discharged on post-operative day three. |
A 44-year-old female presented with a chief complaint of 3-year history of recurrent pain and intermittent swelling to the left mandibular region. The swelling was usually worsened by meals, extreme pain arising once a month. When the pain started, it lasted about 10 min, with an NRS (numeric rating scale) score of 10. She had recently begun to have pain every 4 h. On examination, there was a tense and sensitive submandibular salivary gland and visible swelling in the posterior part of the left side of submandibular area. No salivary flow was appreciated from the left submandibular duct. The radiograph showed an elongated radiopaque structure imposed on the left submandibular area (Fig. top). Computerized tomographic (CT) scan of the mandibular region showed the presence of multiple high attenuated materials and elongated sialolith located within the left Wharton’s duct. Also, very severe atrophic submandibular gland was found (Fig. bottom).\nPreoperative technetium-99m pertechnetate salivary gland scintigraphy revealed that other salivary glands were within normal limits, but with no definite radiotracer excretion in the Lt. submandibular gland (Fig. ).\nThe patient was placed on the operating table in supine position and was induced with short-acting paralytics to allow for monitoring of the branches of the facial nerve during dissection. General anesthesia was obtained via oral endotracheal intubation. The neck was extended with the placement of a shoulder roll, and the head was turned to the opposite side of the involved parotid. The patient was prepared and draped in a sterile fashion. The ipsilateral commissure of the mouth was prepared as readily visible. The incision line was marked (standard modified facelift incision). 2% lidocaine with epinephrine was injected within the subcutaneous tissues of the proposed surgical incision, involving a standard preauricular curvilinear incision which begins at the tragus, going around the inferior border of the lobule and then continuing backwards in the auriculomastoid groove. The superior aspect of the postauricular incision reached to the level of the superior aspect of the mastoid and then was extended posteriorly into the hair line of the neck for cosmesis (Fig. left).\nAfter skin incision, the subplatysmal skin flap is elevated just above the sternocleidomastoid (SCM) muscle using a monopolar electrocautery under direct vision. The greater auricular nerve and external jugular vein can be identified located superficial to the SCM muscle. The skin flap is elevated until the anterior extent reaches the midline of the anterior neck, the superior extent the inferior border of the mandible and the inferior extent the level of omohyoid muscle. Skin flap elevation below the mandible should be performed carefully to minimize injury to the nearby marginal branch of the facial nerve. Normally two assistant surgeons are required to comfortably lift up the skin flap with an Army-Navy retractor or a right-angle breast retractor. After obtaining a sufficient amount of working space (approximately 10-cm height), a self-retaining retractor is applied through the space and is secured [, ] (Fig. right). Dissection began at the lower border of the SMG using the da Vinci Xi surgical system (Intuitive Surgical Inc., CA, USA) with two endowrist arms (monopolar curved scissors & Maryland bipolar forceps) (Fig. left). The proximal facial artery was ligated with vascular clips, the lingual nerve was separated from the submandibular ganglion with monopolar cautery, and Wharton’s duct was ligated with a vascular clip. The lingual and hypoglossal nerves were well preserved. The specimen was well excised, the surgical bed irrigated with warm saline and bleeding control under both robot view and direct vision was performed (Fig. right). A close suction drain was inserted posterior to the hairline incision, and the wound was closed with Dermabond skin adhesive (Ethicon, USA) after subcutaneous layer suture. The pathologic report was sialolith with ductal atrophy. There was no postoperative complication. |
Our patient was a 54-year-old man who was originally diagnosed with a gastric SMT in 2008. He underwent an annual follow-up by upper GI endoscopy at another hospital but was referred to our hospital for further examination and treatment in September 2016, during which his main clinical symptoms were epigastric soreness, general malaise, and melena. He had an unremarkable family history.\nOn admission, he measured 161.6 cm tall and weighed 58 kg and his pulse was 115 beats/min, and blood pressure was 69/43 mmHg. There were no significant findings on abdominal examination, but laboratory analysis revealed a hemoglobin level of 5.6 g/dl. Other hematological and biochemical parameters were within normal limits. Chest X-ray, electrocardiography, and echocardiography results were normal. On admission, because he had advanced anemia and low blood pressure, he received blood transfusion (480 ml). On upper GI endoscopy, an SMT was identified with an associated ulcer on the anterior wall of the lower body of the stomach (Fig. ). There was an extrinsically compressing mass on the anterior wall of the greater curvature and the posterior wall of the stomach (Fig. ). However, no active bleeding from the SMT or ulcer was observed.\nWe diagnosed lipoma without malignancy based on the biopsy of the SMT and ulcer. Endoscopic ultrasound (EUS) confirmed a high-echoic submucosal lesion in the antral wall that extended to the stomach body (Fig. ), and computed tomography (CT) and magnetic resonance imaging confirmed a fat-containing mass spanning entire gastric walls of the stomach antrum and body, but excluding the lesser curvature, with a mass protruding on the anterior wall of the greater curvature (Figs. and ). CT images revealed no metastasis to lymph nodes or to other organs, and the serum tumor markers CEA and CA19-9 were within the normal limits.\nBased on our findings, we suspected a giant gastric lipoma and proceeded to perform a standard total gastrectomy. We considered the execution of resection of the stomach, but we decided to perform total gastrectomy because the range of the tumor was unclear and the risk of recurrence could not be determined. The specimen was opened along the greater curvature, revealing a mucosal surface that was smooth and diffusely elevated by the submucosal mass, but with no involvement of the lesser curvature (Fig. ). There was a compressing lesion associated with an ulcer on the anterior wall of the lower body. The gross pathology was of a yellowish adipose tissue with no fibrous capsules.\nGastric lipomatosis was confirmed by a histological examination of the resected specimen. Microscopy revealed adipocyte proliferation without nuclear atypia and mature adipocytes replacing the submucosal and muscle layers of the stomach. Lipomatosis was present only in the fundic gland zone, and there was no intestinal metaplasia or atrophic gastritis. Multiple hyperplastic polyps were observed on the mucosa overlying the area of diffuse lipomatosis. Immunostaining was negative for MDM2, CDK4, and p16. Moreover, there was no evidence of malignancy in the fatty lesion.\nThe patient recovered well following surgery and was discharged on the postoperative day 14. At the latest follow-up, he was continuing to do well and showed no evidence of recurrence in other organs.\nGastric lipomas are characterized by smooth, sharply marginated, and oval or spherical submucosal masses comprising well-differentiated adipose tissues surrounded by a fibrous capsule [–]. In contrast, gastric lipomatosis is characterized by multiple gastric lipomas or diffuse infiltration of mature adipose tissues into the gastric submucosal or subserosal layers. GI lipomas are extremely common benign colonic tumors, whereas gastric lipomatosis is particularly rare [, –].\nApproximately, 90–95% of lipomas are submucosal, and the rest are subserosal []. Most gastric lipomas are small (4–9 cm) and asymptomatic, occur on the posterior wall of the antrum, and are incidentally detected on radiographic or endoscopic examination of the upper GI tract. Smaller lesions are rarely symptomatic, but large tumors can present with symptoms of gastric ulcer, including epigastric pain, nausea, vomiting, and upper GI tract bleeding. Indeed, the most frequent clinical manifestation is GI bleeding, which is due to the ulceration of the overlying mucosa in 50% of the patients [, ]. When a large lipoma is present, venous stasis is probably the single most important factor underlying mucosal ulceration, which may lead to acute, and sometimes severe, upper GI hemorrhage. Several authors have reported anemia as the primary indicator of large gastric lipomas [–]. In the present case, we suspect that a part of the tumor rapidly increased in size and led to the collapse of the mucosal surface with subsequent ulceration and bleeding.\nCT is an excellent diagnostic tool because it allows the diagnosis of lipoma based on tumor fat density, precluding the need for an endoscopic biopsy. Indeed, a homogeneous mass with a fat density ranging between − 70 and − 120 Hounsfield units is considered pathognomonic of gastric lipoma []. Histologically, GI tract lipomas typically have well-differentiated adipose tissue structures. If a large submucosal tumor is detected on an endoscopic or upper GI examination, then a CT scan can be used to confirm the diagnosis and to inform therapy decisions. However, EUS is the most useful diagnostic tool for assessing neoplasia originating from the submucosa. In the present case, EUS showed a submucosal hyperechoic and homogeneous mass and diagnosed lipoma.\nAlthough conservative treatment is preferred for asymptomatic solitary lipomas, surgical intervention should be considered for symptomatic lipomas associated with ulcers or non-fatty elements. Endoscopic polypectomy is an option for submucosal lesions that are smaller than 3 cm, but larger broad-based tumors have a higher risk of perforation using this approach. In our case, we decided to treat the patient by total gastrectomy for three main reasons: first, a large and symptomatic lipoma was present at surgery; second, although biopsy suggested lipoma, we could not completely exclude malignancy; and third, anastomotic leakage and tumor recurrence were possible if partial resection was performed.\nEight reports of gastric lipomatosis exist in the literature, of which five describe a detailed pathology (Table ) [, , , –]. According to these reports, lipomatosis in the upper stomach can occur with multiple organ involvement or with multiple types of lipomatosis. However, no tumor has been reported in any other organ when lipomatosis occurs in the lower stomach. Most previous cases have reported multiple lipomas, and this is only the second case of gastric lipomatosis presenting as a diffuse lipoma []. In our case and in that by Jeong et al. [], the cases not only had similar specimens and histological findings but were also diagnosed as diffuse-type lipomatosis. In our case, it was noteworthy that the lipomatosis was present only in the fundic gland zone, without intestinal metaplasia or atrophic gastritis. Furthermore, multiple hyperplastic polyps were observed on the mucosa overlying the diffuse lipomatosis. We hypothesize that a relationship exists between hyperplastic polyposis and lipomatosis in some cases. |
A 72-year-old man was referred from a local hospital due to impaired consciousness after a generalized tonic-clonic seizure. He had been diagnosed with a large amount of CSDH in the left frontoparietal lobe and a midline shift to the right hemisphere (). A neurosurgeon at the local hospital had performed burr hole trephination in left frontal bone and he had failed to drain the hematoma sufficiently (). The hematoma was made up of yellow, mud-like material, and it had been misdiagnosed as subdural empyema in the operative room. The doctor immediately transferred the patient to our hospital in order to treat subdural empyema. A neurological examination at admission revealed drowsy mentality with a Glasgow Coma Scale score of 13. Hemiparesis of the right arm and leg was not detected. He could not recall any incident of head trauma. He had hypertension, diabetes, angina, and he had been taking clopidogrel for treatment of angina. A brain computed tomography (CT) scan showed many high density areas in the hematoma and the membrane, compared to usual CSDH. Focal calcifications were found in the membrane. Brain magnetic resonance imaging (MRI) showed a large amount of CSDH with a low, heterogeneous signal on T2-weighted images (WIs), an isodense signal on T1-WIs, and a low, homogeneous signal on diffusion-WIs (). There was no dense enhancement as with a subdural empyema. The membrane surrounding the hematoma was enhanced in a line. He was diagnosed with OCSH based on brain images and previous operative findings. We decided to treat by performing craniotomy, followed by removal of the organized hematoma and the thick membrane. We performed a frontoparietal craniotomy and incised the dura mater. After dural incision, we removed a thick, calcified outer membrane. Under the outer membrane, we found a large amount of yellow, sticky, mud-like organized hematoma with a thick inner membrane (), and no hemorrhagic fluid. The thick, hard, and calcified inner membrane covered the brain cortex. The inner membrane blocked the expansion even after adequate removal of OCSH. We gently performed inner membranectomy and confirmed pulsation in the normal brain cortex (). Pathologic examination revealed an old hematoma with focal calcification and chronic inflammation. Immediate postoperative brain CT showed adequate removal of the OCSH and the inner membrane. Clopidogrel was started as an antiplatelet therapy 1 week after the craniotomy after confirming the absence of acute hemorrhage. He was discharged from the hospital on clopidogrel, and he was scheduled to receive a neurological follow-up. A brain CT scan at discharge showed subdural fluid collection in the subdural space. One month after surgery, brain CT showed no recurrence of CSDH with subdural fluid collection (). He was in an alert state on follow-up examination, and he recovered completely. |
A 65-year-old man was referred by the otolaryngology department to our outpatient clinic due to sudden swelling and mild pain around the right eye. On examination, the patient exhibited what appeared to be severe edema encompassing the upper and lower lids of the right eye (). Crepitus was clearly audible on palpation of the eyelids. An attempt to open the lids was unsuccessful. Visual acuity and intraocular pressure could not be measured due to extreme lid swelling. The patient reported that he had undergone transnasal endoscopic nasal polypectomy through the right nostril 2 days earlier. He said he had been instructed not to cough or strain after the endoscopic nasal surgery and the sudden swelling occurred immediately after severe coughing and straining. We suspected that the sinus wall was weakened due to his endoscopic surgery and the increased pressure caused by straining had forced air in the nose into the periorbital area. B-mode ultrasonography showed trapped air in the periorbital area ().\nConsidering the patient’s anxiety, the severity of periorbital emphysema, inability to conduct a full ophthalmologic examination, and the risk of complications such as compressive optic neuropathy, the patient was re-evaluated for a surgical intervention. After consultation, it was decided to evacuate the air using a 21 gauge needle inserted in the subcutaneous tissue of the upper and lower lids. In sterile conditions, the eye area was cleaned with 10% povidone-iodine. A 21-gauge needle was passed through the skin and subcutaneous tissue of the upper and lower lids parallel to the tarsus about 1.5 cm from the lid margin. Evacuation of subcutaneous air was evident from a significant reduction in lid swelling during the procedure (). The patient’s vital signs were stable and the procedure was concluded. He was discharged with systemic antibiotics (cefuroxime axetil 500 mg twice daily) and moxifloxacin drops four times daily.\nOn follow-up examination the next day, the periorbital emphysema was substantially reduced and the globe could be examined (, ). He had full visual acuity in both eyes; intraocular pressure was 17 mmHg in the right eye and 16 mmHg in the left eye. Dilated fundus examination was normal. No restriction in eye movements was observed. Follow-up examinations at 1 week and 1 month revealed no pathological findings. |
A 34-year-old patient, gravida six para three, presented to our delivery suite at 37+0 weeks’ gestation in labor. She had two terminations of pregnancies, and three normal vaginal deliveries at 39 + 0, 36 + 4, and 38 + 6 weeks’ gestation in 2005, 2008, and 2016 respectively. Her babies were healthy and weighed between 2,500 to 2,925 g at birth. In May 2017, she underwent a laser cone biopsy for CIN 3 with clear margins achieved. Excisional biopsy was the treatment of choice as it was a type two transformational zone. A laser cone was performed instead of a loop electrosurgical excisional procedure (LEEP) in view of a large transformational zone. Subsequent follow up cervical smear and examination were normal. She had no other significant past medical history.\nShe conceived spontaneously in 2019 and presented at eight weeks gestation at her booking visit. A transvaginal cervical length scan, performed at 12 weeks’ gestation in view of her previous laser cone biopsy, estimated her cervical length to be 3.6 - 3.7 cm. She declined Down syndrome screening and her fetal anomaly scan at 20 weeks’ gestation was normal except for an aberrant right subclavian artery. Her pregnancy progressed uneventfully.\nThe patient presented at 37 weeks with spontaneous rupture of membranes followed by regular contraction pains. Speculum examination confirmed the presence of a definite pool of clear liquor but her cervical os was closed on vaginal examination. She was started on a Syntocinon infusion to augment labor, titrated to maintain her contractions every 2 - 3 min. Her cervix remained closed after 6 h and a decision was made to stop her Syntocinon infusion and insert a 3 mg dinoprostone (Prostin E2) tablet. A vaginal examination performed 6 h later revealed a full effaced and 1 cm dilated cervix. She was restarted on Syntocinon infusion thereafter but made no progress after 6 h of augmentation, with contractions felt and seen every 3 min. After discussion with the patient, another dinoprostone tablet was inserted, and Syntocinon augmentation recommenced 6 h later. However, after another 4 h, there was no change to her cervical findings despite regular contractions every 2 -3 min. A diagnosis of cervical stenosis was made.\nShe underwent an emergency cesarean section for failure to progress and delivered a healthy baby girl with a birthweight of 3,515 g and an estimated blood loss of 400 mL. Intraoperatively, her internal cervical os was found to be one cm dilated, thin and fibrotic. She had an uncomplicated recovery and was discharged well on the third postoperative day. |
A 46-year-old woman underwent cerebral angiography as a part of the diagnostic workup of an unruptured middle cerebral artery (MCA) aneurysm. An aneurysm of MCA, with a size of 15 mm × 12 mm, was detected on brain MRA during screening for headache. On admission, physical examination showed no signs of neurologic deficits. Her past medical history included hypertension, hypothyroidism, and polycystic kidney disease. Hemodialysis was initiated four years previously for chronic renal failure. She was a nonsmoker. Hemodialysis was performed on the day before the procedure. Under local anesthesia, diagnostic DSA showed a middle cerebral artery saccular aneurysm, with the size of 16 mm × 13 mm × 9 mm. The procedure lasted 20 minutes. A total of 80 ml of nonionic, hypoosmolar contrast medium “Iopamiro 370” (Iopamidol) was used, which is a standard in our hospital. This was the patient's first exposure to contrast medium. The contrast material was injected into the left and right carotid artery, as well as in the right vertebral artery, with no side effects noted. During injection of the contrast material into the left vertebral artery, the patient complained of blurring of vision which deteriorated to near-total blindness within minutes. The pupils were equal in size and responsive to light. The rest of the neurological examination was normal. Approximately 1 hour after the procedure, the patient developed an episode of a generalized tonic-clonic seizure. An emergency CT brain scan was requested and revealed bilateral symmetrical contrast enhancement in parieto-occipital cortex and subarachnoid spaces, as well as in thalami (). Based on the CT brain scan, the presumed diagnosis was contrast-induced encephalopathy and hemodialysis was quickly performed. Later on, over the next 24 hours, she was confused and agitated, with severe headache, vomiting, and no signs of improvement. A brain MRI, performed on second postprocedure day, demonstrated T2 and FLAIR bilateral symmetrical hyperintensities in basal ganglia, in parieto-occipital cortex, and in splenium of corpus callosum, and the DWI and ADC images demonstrated restricted diffusion in the same areas (). Daily hemodialysis was performed for the next 3 days, and she was treated with dexamethasone. Over the next few days, symptoms gradually started to improve progressing to complete recovery. On day 5, her neurological symptoms resolved completely. She was discharged home well. One month after discharge follow-up, brain MRI did not show any residual lesion, which correlates with the complete recovery (). |
A 31 year old female with a history of two cesarean sections, the last one of them three
years prior, presented with a painful mass in the left lateral side of the Pfannenstiel
incision which had been steadily growing. The same patient went a month ago to our
institution's urology clinic with pain in the inguinal region. After a detailed history
and physical examination an ultrasonography was performed that revealed a 20X12 mm
hypo-echoic nodular mass, neighboring the rectus abdominis muscle, with minimal
vascularization. The differential diagnosis included endometriosis and a possible
desmoid tumor. The patient was referred to our clinic for further evaluation. The pain
started on the left lower quadrant and radiated towards the inguinal region, and was
associated with menstruation. In physical examination a 2 cm wide mass was palpated in
the previously described localization. Magnetic resonance imaging was performed which
revealed a 20x11 mm mass which is slightly hyper-intense in the T1 sequence, and
contrast enhanced after IV gadolinium injection in the T2 sequence, with increased
signal intensity and nodular appearance in diffusion weighted sequences. These were
found to be consistent with an endometrial implant ( and ). Examination of the
uterine cavity showed effusion which was at most 15 mm in width when measured. A little
free fluid, indicating peritoneal irritation was present in between the intestinal
loops. No pathological lymph nodes were present in the lower abdomen.
The patient was admitted for surgical removal of the mass. A 2 cm wide fibrotic
appearing mass was excised () and was sent
for pathological examination. The patient's complaints resolved after the surgery. She
was discharged with no complications two days after the operation. Four months after the
surgery, the patient came in for a follow-up visit, and had no complaints or
complications. Histopathological examination was consistent with glandular structures of
the endometrium with accompanying endometrial |
A 37-year-old woman, gravida 2 para 1, presented to a tertiary women’s hospital at 21 weeks gestation in her second pregnancy complaining of vaginal bleeding of 1-day duration changing three sanitary pads and colicky lower abdominal cramps. History taking revealed she had one previous full-term normal vaginal delivery that was complicated by pregnancy-induced hypertension 2 years earlier. She booked late at 19 weeks in her second pregnancy and was started on aspirin for this pregnancy to reduce the risk of preeclampsia. Antenatal bloods were normal. She declined maternal serum screening for chromosomal aneuploidies. Fetal anomaly screening at 20 weeks showed low risk with no fetal anomalies except for a low-lying anterior mid placenta.\nHer vital signs showed a temperature of 36.7 °C and maternal tachycardia of 107 beats per minute with blood pressure reading of 136/95. On examination, her abdomen was soft and the uterus was relaxed and not woody. Speculum examination showed no active bleeding with cervical os closed and a small clot evacuated. Her reflexes were normal with no signs of clonus suggestive of impending eclampsia. Fetal heart measured was normal at 147 beats per minute. Ultrasound scan showed a low-lying anterior placenta. The clinical diagnosis then was threatened miscarraige and low-lying placenta.\nSubsequently, she was admitted in hospital for further monitoring. Initial blood tests revealed a hemoglobin of 10.7 g/dL, low platelet of 90 × 109/L, deranged coagulation profile and mild acute kidney impairment with raised creatinine of 99 mmol/L. She was transferred to the high dependency unit for further management and was reviewed 4 h later when she suddenly complained of increasing vaginal bleeding and worsening generalized lower abdominal pain. Vital signs revealed blood pressure reading of 108/61 mm Hg with persistent maternal tachycardia at 110 beats per minute. On physical examination, the uterus was now tense and woody hard and vaginal examination using speculum revealed the cervical os was closed with active vaginal bleeding and clots evacuated. Atrans-abdominal ultrasound showed a heterogenous area anterior to the placenta suggestive of blood products with placenta abruption ().\nA repeat set of blood tests performed immediately showed a worsening drop in platelet levels from 90 × 109/L to 53× 109/L and a drop in hemoglobin from 10.7 to 7.8 g/dL. Coagulation profile remained severely deranged. The clinical diagnosis was consumptive coagulopathy secondary to placenta abruption. In view of the patient’s deteriorating condition, the patient subsequently underwent explorative laparotomy keep in view hysterotomy at 21 weeks gestation.\nUnder general anesthesia, a lower segment hysterotomy was performed via Pfannenstiel incision. Intraoperative findings revealed a Couvelaire uterus with clots noted on incision into uterus. The fetus and an entirely separated placenta were delivered complete. In total, there was estimated blood loss of 1,000 mL. The appearance of the uterus was consistent with Couvelaire uterus (). The uterus was closed in two layers and hemostasis was achieved. The patient recovered well subsequently with blood products replacement and her coagulation profile was normal after with post-operative hemoglobin of 9.2 g/dL. She was discharged well 3 days later. Histopathology of placenta showed retroplacental and retromembranous hemorrhage with intervillous hemorrhage consistent with placental abruption. |
A 21-year-old female, Caucasian, ASA I with no associated comorbidity, university student, presented at the dental service of the Universidad Austral de Chile on January 5, 2014, to get fillings treatment for superficial dental caries in teeth 35 and 36. Her clinical history did not show surgical interventions under general anesthesia. It reported previous dental filling treatments with the use of local anesthetic with no manifestation of any complication. The intraoral examination showed a good rate of oral hygiene, with no presence of gingivitis or any other injury of the delicate tissues. For the treatment, the dentist decided to use the troncular technique to the IAN or the Halstead technique according to the one described by Malamed[] using a carpule syringe with a 27-gauge 0.4 mm × 25 mm needle (Terumo Corporation, Japan) and only one vial of 1.8 ml of articaine hydrochloride 4% with epinephrine 1:100,000 (Nova–FL®, Brazil). The dental crown notch, pterygomandibular raphe, and the occlusal plane of the left mandibular molars were considered for the point of injection. Setting the body of the carpule on the opposite side, the dentist set the tip of the needle to the level of the retromolar space and injected approximately 0.3 ml of anesthetic solution into the mouth nerve and then penetrated with the needle into delicate tissue 25 mm approximately until osseous contact. Blood content was observed in the rest of anesthesia cartridge, verifying the positive aspiration of the intravascular tap [].\nAt the moment of the injection, the patient immediately informed of an itching and burning feeling in the nasal and palatine mucosa, without the pain and dizziness associated with the tap. In the clinical examination, numbness was recorded in the left genial region, endangering the lower eyelid, wing of the nose, and upper lip on the left side of the face. The intraoral examination revealed an ischemia of the left mouth mucosa, end of vestibule of the left upper lip, loose and stuck gum between the maxillary teeth 21 and 28 [], and palatine mucosa from the median raphe to the palatal alveolar processes []. Due to the clinical picture, the intervention was partially suspended. After 5 min, the patient showed dysesthesia of the lower lip, gum and half of the tongue on the left side; she did not have any visual or nasal complications. She did not present any ocular complications such as double vision, blepharoptosis, miosis, anhidrosis, anisocoria, or others.\nAfter 10 min, gradual recovery was observed of the normal skin color and a pale pink at the end of the buccal space and palatine mucosa. Fifteen minutes after administration of the anesthesia, the anesthetic effect on the IAN was verified through a caries probe in the gingival crevice of tooth 36. After the reported complication, the decision was made to treat teeth 35 and 36. Once completed, the patient received some oral instructions about preventive measures to consider with the lip and tongue anesthesia, soft diet, and to return for a check-up 3 days later. In light of the interesting nature of the case, the patient gave her informed consent to study the case and present it in this article. |
A 10-month-old male child, known case of tricuspid atresia, presented to our outpatient department (OPD) with complaints of facial deviation, dysphagia, nasal regurgitation of food, and nasal intonation of voice for the last 15 days. Symptoms were acute in onset, progressed initially for 5–6 days, followed by a static course. He did not have weakness of any limb. He was admitted in another hospital in view of pneumonia since last 20 days and he developed these complaints in the hospital duration only and was referred to our hospital for further workup. There was no history of any intramuscular injection, insect bite, head injury, or ear discharge. His immunization status was complete and has received three doses of diptheria pertusis tetanus and oral polio vaccine.\nOn examination, the child was fully conscious, alert, and responding. Physical examination showed facial deviation to the left along with drooling of saliva. Respiratory rate was 34 breaths/min, heart rate was 110 beats/min, SpO2 was 76%, and blood pressure was 90/60mm Hg. On central nervous system examination, left-sided lower motor neuron type of facial nerve palsy was noted along with 9th and 10th cranial nerve (CN) palsy. Rest all CNs were normal. Limb muscle’s power was normal, deep tendon reflexes were 1+, and plantar response was down going bilaterally. Sensory system examination was within normal limits. Fundus examination was normal. Nerve conduction study was carried out, which showed reduced amplitude and decreased conduction velocity in bilateral peroneal motor nerves and left tibial nerve. Study also showed reduced amplitude in bilateral sural nerves along with reduced amplitude but normal onset latency and conduction velocity in right tibial nerve. Routine blood investigations including complete hemogram were within normal limits. Workup for poliomyelitis was negative. Throat and nasal swab were sent, which showed no growth. As regurgitation of feeds was present and child vomited every meal, he was started on ryels tube (RT) feeding. Relatives were taught how to give RT feeding and once they were confident child was discharged on RT feeding after 1 month of hospital stay. Patient regularly visited OPD for changing feeding tube and detailed neurological examination at each visit []. There was no progression of the disease and slowly child improved with first recovery of facial nerve and complete recovery after 4 months of follow-up. Now patient is taking meals normally. Serological tests for antibodies were not carried out due to financial constraints. The diagnosis was kept as acute bulbar palsy plus syndrome––a localized variety of GBS on the basis of clinical course and nerve conduction study and negative workup for poliomyelitis. |
We present a 15-year-old boy who was admitted to our hospital with significant weight loss, malaise, and cold intolerance. Written informed consent for presentation was obtained from the patient and his parents.\nThe patient weighed 102 kg until four years ago, at which time, with the help of a dietitian, he started to lose weight. However, after losing 2 kg, he discontinued the dietary regime. Two years later, when he weighed 100 kg, he decided to lose weight again because he felt uncomfortable with the way he looked and felt very overweight. Within a year, he lost 20 kg by eating less and playing basketball every day for approximately two hours a day. At that time, he had to quit basketball due to a busy school schedule which led to a fear of gaining weight, causing him to restrict his diet even more. By restricting his daily intake to 500 kcal, he had lost 17.5 kg within the last two months before presenting to our clinic. The patient denied having body image problems but agreed that he had an intense fear of gaining weight. Past medical history was unremarkable except for an appendectomy performed when he was 7 years old. The family history revealed that two of his aunts have Hashimoto thyroiditis.\nAt admission, the patient’s body weight was 60.7 kg (50-75th percentile). Height was 186 cm (>97th percentile) and body mass index was 17.55 kg/m2 (<3rd percentile). His body temperature was 36.1 °C and respiratory rate was 22/min. His supine blood pressure was 100/60 mmHg and heart rate was 40 bpm. His standing blood pressure was 95/60 mmHg and heart rate was 66 bpm. Cardiac examination was normal except for the bradycardia, and other systems were also normal on his physical examination. Meeting the diagnostic criteria of the fourth edition of Diagnostic and Statistical Manual of Mental Disorders (DSM IV), he was diagnosed with AN-restrictive type and hospitalized due to bradycardia. His laboratory investigations which included complete blood count, liver and kidney function tests, glucose and electrolyte levels, sedimentation rate, cortisol, cholesterol levels, and urinary analysis were all within normal ranges. Thyroid function tests revealed very low TSH levels (0.025 uIU/mL, normal range: 0.27-4.20 uIU/mL), low free T3 (fT3) levels (2.87 pmol/L, normal range 3.10-6.70 pmol/L), and normal free T4 (fT4) levels (21.9 pmol/L, normal range: 12.00-22.00 pmol/L). Thyroid peroxidase antibodies and thyroglobulin antibodies were high, while TSH receptor antibodies were negative. With these findings, he was additionally diagnosed with Hashimoto thyroiditis. Thyroid ultrasonography confirmed the diagnosis.\nIn the inpatient unit, the patient was followed by an interdisciplinary team consisting of a child and adolescent psychiatrist, an adolescent medicine specialist, a pediatric endocrinologist, and a dietitian with special training and experience in adolescent EDs. During his three-week stay, he gained 4 kg. Despite the weight gain and the improvement in his nutritional status, bradycardia continued (40-50 bpm). Echocardiography findings were normal, and Holter monitoring only revealed sinus bradycardia. Thyroid functions were monitored closely without any medical treatment, along with his vitals. Before discharge, while TSH levels were still low (0.018 uIU/mL), fT3 levels were thought to be relatively high (3.76 pmol/L) considering his metabolic status. The patient was discharged with a weight of 64.7 kg. At his follow-up visit 2 months later, the boy had gained weight and weighed 75.1 kg. Due to the gradual increase noted in his TSH levels (from 8.91 to 28.84 uIU/mL), levothyroxine treatment was started. At that time, fT3 and fT4 levels were measured as 7.74 and 4.42 pmol/L, respectively. Two months later, it was learned that he had been using levothyroxine in doses three times higher than the recommended dose. Although monitored closely, due to the drug compliance problems and weight changes with severe body image issues, it was hard to maintain the thyroid levels within a stable course. Stabilization occurred after ten months of therapy when he started using a proper medication schedule and succeeded in preserving his target weight. The course of the thyroid function tests is given in . Bradycardia also improved with the recovery in thyroid hormone levels. |
A 67-year-old woman, with compensated hypothyroidism and hypertension, presented to another hospital with continuous low back pain and a low-grade fever of 3-month duration. She was diagnosed with subacute thyroiditis and treated accordingly; however, her symptoms did not abate. Computed tomography (CT) and magnetic resonance imaging (MRI) of the lumbar spine revealed destructive changes of the L1 and L2 vertebral bodies; these changes were surrounded by paravertebral abscesses (). A culture examination of the specimen harvested by CT-guided puncture of the paravertebral abscess revealed nontuberculous mycobacteria identified as Mycobacterium abscessus, which were noted to be resistant to antituberculous agents in vitro. The patient's pain level and spinal destruction gradually advanced. Additionally, there was a continuous discharge of pus from the puncture hole despite chemotherapy treatment with ethambutol, rifampicin, and isoniazid. The patient presented to our hospital 6 weeks after the diagnosis.\nUpon admission to our hospital, the patient had significant back pain without bladder or bowel dysfunction, or neurologic deficit of the lower extremities. Laboratory findings showed a white blood cell count of 4310/μL, C-reactive protein of 2.7 mg/dL, and an erythrocyte sedimentation rate of 53 mm/h. A chest CT showed no signs indicative of pulmonary infection. We changed the patient's regimen of antibiotics to include clarithromycin (800 mg per day, 400 mg orally twice daily), amikacin (600 mg per day, 300 mg intravenously twice daily), and imipenem (2000 mg per day, 1000 mg intravenously twice daily) for 3 weeks after admission to our hospital. However, her condition did not improve, and the continuous discharge of pus did not change. Smear examination of the pus discharged from the puncture hole did not reveal any types of bacteria.\nRadical debridement of the vertebral osteomyelitis of the L1 and L2 vertebral bodies and spinal reconstruction using iodine-supported instrumentation were performed using a right retroperitoneal approach. An autogenous bone graft harvested from the iliac crest and alpha-TCP paste mixed with imipenem were packed into an iodine-supported mesh cage. The mesh cage was inserted into the large defect after a radical debridement involving L1 corpectomy. Then, anterior fixation from T12 to L2 using antibacterial iodine-supported instrumentation was performed (). Antimicrobial chemotherapy using clarithromycin for 6 months and amikacin and imipenem for 3 months at the same dose of the preoperative period was continued after surgery, as these antibiotics have been proven to be effective against Mycobacterium abscessus, which was identified on the culture examination of the specimen harvested during surgery. A hard brace was applied for 3 months after surgery. Laboratory findings at 3 months after surgery showed no evidence of inflammatory signs; white blood cell count was 5000/μL, C-reactive protein level was 0.1 mg/dL, and erythrocyte sedimentation rate was 4 mm/h. Two years after surgery, the infected anterior site had healed, and bony fusion was successfully achieved (). The patient is now completely asymptomatic without a brace and has not had a recurrence of infection. No evidence of inflammatory signs has been apparent in subsequent laboratory examinations. |
A 58-year-old female walked into our clinic with intractable dizziness and nausea for 3 days. She was undergoing dialysis for her chronic renal failure caused by familial polycystic kidney disease. She had a history of neck clipping of unruptured left middle cerebral artery aneurysm some 13 years ago at another institution. Her GCS was E4V5M6 so WFNS grade was I. Head CT scan demonstrated small amount of SAH in the interhemispheric fissure []. CT angiography revealed 6 mm ACoA aneurysm, as well as complete clipping of left middle cerebral artery aneurysm []. Endovascular surgeons were reluctant from coiling because of wide neck of the aneurysm.\nOperation was performed via anterior interhemispheric approach. Dissection of interhemispheric fissure between rectus gyrus and genu of corpus callosum provided sufficient exposure of the ACoA aneurysm. During dissection, left olfactory nerve was missing and A1 segment of left ACA adhered to surrounding brain tissue, suggesting ACoA had been explored in the previous operation. The adhesion was more prominent around the aneurysm, and hypothalamic artery was encased in the aneurysmal wall with glue material []. Gentle and meticulous dissection was performed, but minor leak of blood occurred from the neck of the aneurysm adjacent to right A2. Any attempts for neck clipping only enlarged laceration of aneurysmal neck. Trapping of the aneurysm including the neck laceration was unavoidable to secure the aneurysm, with simultaneous revascularization of right A2 because right A1 was aplastic on initial CT angiography []. Temporary hemostasis of lacerated neck was obtained by compression with cottonoid under flow control for a few minutes. Back to A3 segment of ACA, A3-A3 side-to-side anastomosis was performed with bilateral A3 cross-clamping time 37 min []. The aneurysm, together with hypothalamic artery, was trapped with two aneurysmal clips []. Complete elimination of the aneurysm and reconstruction of right A2 segment of ACA was confirmed by Doppler ultrasonography, indocyanine green videoangiography, and postoperative CT angiography [].\nThe patient revealed good postoperative recovery with no apparent ischemic change on follow-up MRI []. She developed recent memory disturbance and topographical disorientation, which severely affected her social activity. However, after several months at home, her cognitive function recovered well to almost normal. Her postoperative hypophyseal function was normal. Her mRS at 12 months was 1. |
A 24-year-old male was admitted to the clinic due to recurrent episodes of bilateral lower limb weakness over 8 years, which had been aggravated during the past month. Eight years previously, he began experiencing bilateral lower limb weakness and gait instability, without pain or other symptoms, which were relieved by the oral or intravenous daily administration of 4–10 g of potassium supplementation. He experienced similar episodes approximately every 2 years.\nOne month before presenting to our clinic, the patient had rebound weakness upon the discontinuation of potassium. He was admitted to the hospital for further diagnosis. On admission, he was alert and oriented, with no sign of anemia. His vital signs were a temperature of 36.2°C, a pulse rate of 93 beats per min, 20 breaths per min, and a blood pressure of 127/87 mmHg. His waist circumference was 95 cm, and his body mass index was 26.12 kg/m2. His past medical history was positive for hepatitis B virus infection. No similar findings were present in his mother, elder sister, or his daughter. Family members had no history of consanguineous marriage, use of laxatives or diuretics, or alcohol abuse. This study conformed to human research guidelines stated in the Declaration of Helsinki, and all subjects gave their written informed consent.\nLaboratory tests. All the laboratory parameters were determined by Laboratory Medicine Department of West China Hospital, Sichuan University. Serum and urine electrolytes were tested by Cobas8000 automatic biochemical analyzer (Roche, Switzerland). Arterial blood gas was analyzed by GEM3000 blood gas analyzer (Instrument Laboratory, American). Thyroid hormones were measured by electrochemiluminescence immunoassay. Plasma renin activity and aldosterone levels were detected with radioimmunoassay.\nGene analysis. The proband's father died several years before and we were unable to reach his wife, so we could not collect samples of their DNA. Other family members provided peripheral blood samples, which were used to extract DNA with the QIAamp DNA BloodMini Kit (Qiagen, USA). Gene amplification was performed by polymerase chain reaction (PCR) of 3 ng/mL DNA samples with the Ion AmpliSeq™ Library Kit (Life Technology, USA). PCR was conducted using SLC12A3 primers (listed in Supplementary Table S1) on a GeneAmp 9700 PCR system (ABI) under the following conditions (,,\n): 5-min predenaturation and 30-s denaturation at 95°C, annealing at 57-64°C, and 90-s chain extension at 72°C; the above steps were recycled 30 times. All sample densities were of 3 ng/mL. Gene sequencing was performed and analyzed by ABI Personal Genome Machine (Life Technology). In addition, multiple ligation-dependent probe amplification (MLPA) was used to identify small base insertions or deletions. Gene amplification and sequence analysis were conducted at Division of Endocrinology and Metabolism, West China Hospital and Kingmed Center for Clinical Laboratory, Sichuan. The analysis of amino acid conservation was conducted according to the UniProt database. Three different in silico prediction tools, including PolyPhen 2, LRT and MutationTaster, were used to predict whether this mutation was pathogenic. |
A 32-year-old man with a diagnosis of right comminuted mandibular fracture, right comminuted zygomaticomaxillary complex fracture, right mandibular dentoalveolar fracture, and avulsive injuries of soft tissue and skin at his mandible due to gunshot after 18 days was hospitalized, and his injury was in category of penetrative and avulsive wound (Figure ). Right eye had become no light perception (NLP). Through his first surgery, it was decided to treat the fracture of right zygomaticomaxillary complex. Because of avulsive injuries at the mandible, we could not treat the mandible at the same time. After general anesthesia, hemicoronal incision done on the right side, to access the lateral orbital wall, body of zygoma and zygomatic arch. Via a subciliary incision, we had access to infra. Orbital rim and orbital floor and the segments have been reduced and fixed with two four-hole miniplates. Moreover, Medpor prosthesis was placed on the eye floor to reconstruct the defect. To the zygomatic buttress which has been reduced and through vestibular approach we had an access, and fractures fixed with two miniplates, arch bar and IMF has been used for temporary treatment of mandible fracture. Canfield's dressing with honey was used for avulsive soft tissue to regeneration.\nEight months after the first surgery, the second surgery was performed to remove the bullets from the maxillofacial area and reconstruct the mandible. A total sum of nine bullets were extracted from the maxillofacial area with the aid of navigation (Figure ). Then, the arch bars in two jaws were applied; after osteotomy of fracture segments, the teeth were manipulated in appropriate occlusion, and the fragments were fixed with a reconstruction plate and four miniplates. One fragment was fixed with a lag screw. Hence, the mandibular was fixed properly. Thereafter, the patient had the problem of bulky soft tissue in malar and buccal areas due to fibrosis after repair of avulsed soft tissue. One year after the second surgery, all plates in midface were removed to reduce the prominence of the cheek, the bone was shaved at zygomatic area, and debulking of the soft tissue of the cheek was carried out. Furthermore, there was a nonunion in mandible due to bone resorption bone defect causing the nonunion was reconstructed by calvaria bone grafts (Figures , ) Since the patient had facial nerve paresis and sagging of the lip at the corner of the mouth, the muscles of the corners of the lips were lifted upward and fixed. |
An abnormal shadow with anterior mediastinal calcification and an elevated left hemidiaphragm was incidentally observed in a 57-year-old man who underwent chest radiography during a periodic health examination (Fig ). Chest computed tomography (CT) revealed an anterior mediastinal mass with multiple ring calcifications (Fig ). He denied a relevant medical history and symptoms such as muscle weakness or red blood cell hyperplasia. Physical examination revealed no abnormal findings and no lymphadenopathy. Routine laboratory parameters were within reference ranges. He reported no history of other operations.\nBased on a high index of clinical suspicion for a non-invasive thymoma or teratoma, we decided to perform surgery for accurate diagnosis and treatment. After induction of general anesthesia, an extended total thymectomy and concomitant pericardial resection were performed via a median sternotomy. Using blunt and sharp dissection, the gland was freed from the pericardium and the adjacent mediastinal pleura. Although the tumor was observed to have invaded the pericardium and the left phrenic nerve, the extent of tumor invasion was unclear. A cardiovascular surgeon was consulted, and the invaded pericardium and the left phrenic nerve were carefully resected. The tumor had not invaded the heart, and the remaining thymic tissue was removed.\nThe resected tumor measured 8.0 × 7.0 × 3.0 cm in size (Fig a) and was observed to invade the pericardium directly. In gross sections, the tumor appeared to be a solid mass separated by multiple rings of calcification, and the surgical stage was identified as Masaoka–Koga stage III.\nHistopathological examination revealed that the tumor was encapsulated by a calcified and ossified layer with invasion of the pericardium and pericardial fat tissue (Fig a,b). A high-power view showed epithelioid cells intermingled with lymphocytes and perivascular lymphocytic infiltration (Fig c,d). Based on World Health Organization histopathological classification, the patient was diagnosed with an invasive thymoma type B2.\nThe patient's postoperative course was uneventful, and he was discharged on postoperative day 12. The patient is currently undergoing chemotherapy and radiation therapy to prevent tumor recurrence.\nThe human research ethics committee of Gyeongsang National University Hospital provided a waiver as approval is not necessary for single-case reports. The patient provided written informed consent for the publication of clinical details and images. |
The 8-month-old boy was born at term without any unusual birth history (38 weeks, 3,150 g, by Cesarean delivery) to a 45-year-old father and 36-year-old mother. He had one brother (12-year-old) and sister (8-year-old). None of the family members had any medical history during the growth period.\nHe was admitted to the pediatric department due to an initial seizure event following aspiration pneumonia and was referred to our clinic for the evaluation of unexplained neuroregression. Although he was hypotonic from birth, he achieved a social smile at 3 months and started head control during the first 4 months. He rolled over, and nearly grasped his toys with prone position at 6 months. Generalized tonic–clonic type seizures at 6 months were his first clinical symptom, a detailed history revealed delays in developmental milestones after that. Electroencephalogram (EEG) findings showed abnormal awake and sleep recordings due to slow background activity, suggestive of diffuse cerebral dysfunction with symptomatic or cryptogenic seizures. Magnetic resonance imaging showed cerebral hypoplasia especially in the frontal and temporal lobes at approximately 4 years of age. He was observed at the outpatient clinic for developmental delays associated with encephalopathy and seizure events, which occurred hundreds of times for 2 years and were fairly well-controlled with valproic acid, phenobarbital, and clonazepam.\nAt 26 months after surgery for bilateral cryptorchidism, progressive respiratory difficulty persisted and weaning from the ventilator was not possible; repetitive aspiration pneumonia occurred as he was unable to proceed with sputum expectoration. Therefore, tracheostomy was performed and night-time breathing using a ventilator was maintained subsequently. At the time of admission, repetitive hand flipping without purpose and lip smacking was observed during examination, although epileptiform discharges were not observed during EEG, we decided to proceed with additional evaluation other than that previously considered at this point. The various clinical features of the patient are described in Table .\nThere were no abnormal findings based on laboratory investigation, and genetic analysis of mutations including Prader-Willi gene, spinal muscular atrophy gene, and other chromosomal aberrations. Chromosome analysis revealed a 46, XY karyotype. A muscle biopsy also demonstrated no abnormal findings. |
In 2015, a 24-year-old male was referred to our hospital for evaluation of gingival swelling in the mandibular left posterior region. The patient was a non-smoker and had no underlying disease. He had first noticed the lesion about one year prior. On extra-oral examination, facial asymmetry with swelling involving the left side of the mandible was observed at the time of admission. Intraoral examination showed a discharge of pus in the posterior region.\nX-ray radiographs (panoramic radiograph and computed tomography [CT]) were performed.() Axial CT showed a fairly large (27 mm×34 mm×23 mm) and well-defined rounded cystic destructive lesion involving the left mandibular body. Absorption of the roots of #36 and #37 had occurred. Incisional biopsy was performed under local anesthesia, and the tissue was sent for histopathological examination, which revealed a suggestive benign cyst. Endodontic treatment of #36 was performed.\nAt the time of operation, extraction of #37 and #38 and enucleation of the lesion were carried out under general anesthesia. The iliac bone graft took place at the same time. After surgery, a newly excisional biopsy was performed, and the results did not show a benign cyst but instead were suggestive of unicystic ameloblastoma, mural type.() A follow-up check was done at three months, six months, nine months, and one year after surgery. Nine months postoperatively, a panoramic radiograph revealed favorable healing of the iliac bone graft in the left mandibular body.()\nAt one year and five months after surgery, implantation of #37 was performed. The implantation was done using a one-stage technique because the initial torque was favorable. () Based on the results of the initial histopathological examination, cyst enucleation and an iliac bone graft were performed simultaneously in this patient. Usually in cases of ameloblastoma, a bone graft is not carried out at the same time due to the high recurrence rate. In this case, non-invasive cyst enucleation and the iliac bone graft were performed simultaneously, and the healing was satisfactory without any evidence of recurrence. |
A 24-year-old pregnant woman (G2P1) was referred to us due to suspected bilateral ovarian cysts at 8 weeks of gestation. She had undergone ovarian cystectomy twice under open surgery: left and right ovarian cystectomy for mature cystic teratoma and mucinous cystadenoma, respectively. She had no additional medical history or familial medical history. Transvaginal ultrasound and magnetic resonance imaging (MRI) (Figures and ) revealed two pelvic cysts. The left-sided unilocular cyst was 9 cm in diameter. The right-sided multilocular cyst was 5 cm in diameter. We diagnosed this condition as bilateral ovarian cysts.\nAlthough the serum levels of tumor markers (CA125, CA19-9, and CEA) were normal for a pregnant woman, considering the large size of the cyst, cyst resection was attempted at 14 weeks; however, it was converted to probe laparotomy. Marked adhesion around the cysts, posterior uterus, and Douglas' pouch made cyst resection impossible as extensive adhesiolysis may cause uterine damage and also uterine contractions after surgery. Gross examinations revealed no metastatic lesions or lymph node swelling. Abdominal fluid cytology revealed no malignant cells.\nAt 32 weeks of gestation, MRI revealed that the left-sided cyst size had increased to 27 cm in diameter (Figures and ), although she was asymptomatic. As shown in , the right-sided multilocular cyst became very close to the left monocytic cyst. At this stage, the left large monocytic cyst appeared to merge with the smaller right multilocular cyst, forming a large cyst occupying the entire pelvic cavity, which was later confirmed by laparoscopic findings.\nThis large cyst showed no solid-part or papillary growth. The serum levels of tumor markers remained normal. Malignant ovarian tumor could not be ruled out but was considered less likely. We weighed merits and demerits between relaparotomy for tumor resection during pregnancy and a wait-and-see approach for several weeks; the former is likely to require extensive adhesiolysis and may cause preterm delivery. We decided on the latter strategy, since resection should be performed in the event of a size increase or images indicative of malignancy. The fetus normally developed without fetal growth restriction.\nCesarean section and tumor resection were performed at 37+4 weeks of gestation, yielding 3,012-g male infant with Apgar score 8/9 at 1/5 minutes, respectively. The infant did not have congenital abnormalities. After the completion of cesarean section, we ruptured the wall of this large cyst, with care to avoid the cyst content entering into the abdominal cavity. A large amount of serous fluid was drained. This large cyst was a multicystic cyst (5 cm), considered to be the right multicystic ovarian cyst that had been observed from the first trimester. The wall of the large cyst showed marked adhesion to the peripheral peritoneal cavity. We resected it as widely as possible together with right salpingo-oophorectomy (Figures and ). The left ovary was macroscopically normal, and thus there was no evidence of the left ovarian tumor. The resected tumor consisted of a large unilocular cyst with serous fluid and a mucinous cystadenoma (Figures and ). In the former, lining epithelium was absent in many parts () and mucinous epithelium was occasionally found in continuity with the cyst wall of the latter (right ovarian cystadenoma). No malignant cells were found in the resected specimen. Immunohistochemistry revealed focally positive staining for estrogen and progesterone receptors on the resected cyst wall (Figures and ). At 12 months after the delivery, left ovary remained normal and the retention cyst did not recur. An informed consent for this reporting was obtained from this patient. |
A 47-year-old female had a car accident and sustained fracture of both tibial bones. She underwent internal fixation of the fractured tibial bones under general anaesthesia. One week later she developed generalized muscle weakness and acute respiratory distress. She was transferred to the ICU where mechanical ventilatory support was given after intubation. Clinically she had bilateral lower motor neuron facial palsy and quadriparesis. The upper limb power was 3/5 bilaterally and lower limbs were 0/5 proximally and 2/5 distally. She had total areflexia of all four limbs and the plantar responses were mute. She was assessed by a neurologist and a diagnosis of acute GBS was suspected. Nerve conduction study (NCS) showed prolonged distal latencies, absent H reflex and reduced conduction velocities suggestive of acute demyelinating polyradiculoneuropathy. The blink reflex was abnormal with prolonged motor latency of both facial nerves. The patient refused lumbar puncture, hence cerebrospinal fluid examination was not done; as she complained of back pain. Other investigations including full blood count, blood biochemistry, autoimmune screen, protein electrophoresis, and porphyria screening were negative. Serological tests for Mycoplasma pneumonia, Epstein-Barr virus and hepatitis virus were negative. The clinical picture and electrophysiological findings were consistent with GBS. She was treated with intravenous immunoglobulin (IVIG; 0.4 g/kg for 5 days). While in the ICU she developed severe bulbar weakness. After the first course of IVIG, she started to improve slowly. The facial weakness and motor power in her limbs improved slowly. However, weaning from the ventilator was delayed due to bulbar weakness and inter-current respiratory tract infection. A second course of IVIG was given after 2 weeks. Chest infection was treated with piperacillin and tazobactam along with chest physiotherapy. Subsequently she recovered and after 3 weeks of ICU care, she was extubated. This patient improved well and was able to walk with minimum support and eventually discharged from the hospital. |
An 8-year-old boy with a history of improper treatment for asthma presented to the hospital with a 1-day history of fever, cough and dyspnea. On arrival, he exhibited wheezing, fever, shortness of breath and 90% oxygen saturation. We administered 3 L of high-flow nasal cannula therapy. On physical examination, the breath sounds of his left lung had almost disappeared, and wheezing was heard in the right lung. The WBC count was 16.5 × 109/L, the percentage of neutrophils was 84%, and the C-reactive protein level was 34.22 mg/L. Chest computed tomography (CT) showed double pneumonia, obstruction of the left main bronchus, emphysema in the left upper lung and atelectasis in the lower lung (). The day after admission, fiberoptic bronchoscopy was performed. During the operation, a thick and tough plastic cast was removed from the left principal bronchus, and a dendritic plastic cast was removed from the middle lobe of the right lung (). Pathological examination of plastic casts revealed inflammatory necrotic tissue with a large number of eosinophils, mononuclear phagocytes and a small number of neutrophils (). Multiple RT-PCR analyses of sputum and alveolar lavage fluid indicated positive influenza A virus H1. Sputum culture indicated Haemophilus influenzae. We administered oseltamivir, antibiotics, glucocorticoids and other treatments. Although his symptoms improved significantly, the boy showed mucus casts when he coughed violently. On the 7th day after admission, bronchoscopy was performed again, and a plastic cast was removed from the left lingual lobe bronchus. On the 9th day after admission, the patient appeared clinically healthy. Routine blood examinations were normal. Chest CT showed pneumonia in the left upper lung, emphysema had been relieved, and the left lower lung atelectasis had recovered (). The correct asthmatic treatment plan was given to the patient at discharge. After 2 months, chest CT showed obvious improvement in lung inflammation, and his asthma was well controlled. |
An 18-year-old female engineering student belonging to middle socioeconomic status hailing from a rural area of Central India had presented to our psychiatry outpatient services with complaints of excessive fear of hens. At the age of 5 years, she had a terrifying encounter with a live hen. Subsequently, she started developing intense fear and anxiety amounting to full panic attack whenever she saw hens. Her thought process on seeing hens was that of fear of hen pecking or attacking her. Gradually, her fear extended to even pictures or films showing hens. She started experiencing anticipatory anxiety and avoided all circumstances involving hens including chicken dishes in restaurants, which is rarely reported and hence unique to our case. She recognized the irrationality of her fear but expressed inability to control her thoughts and subsequent fear and avoidance. Her interaction with family, friends, and schoolmates was otherwise normal, and no significant abnormality in other areas of life was noted. No history of other phobic/anxiety disorders or any other psychiatric disorder was elicited. She had no significant medical or surgical history. Her family history was not contributory. Her periods were regular, moderate, and painless. Premorbidly, she had a slow to warm up temperament with behavioral inhibition. Her physical examination and routine blood parameters were within normal limits. Based on her clinical symptoms, a diagnosis of specific phobia, animal type (alektorophobia), was made as per the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition diagnostic criteria.[] Since the patient preferred psychotherapy over pharmacotherapy, graded exposure therapy was initiated. A total of eight therapist-assisted sessions were conducted (once per week, each lasting 60–90 min). Initial sessions consisted of basic psychoeducation, relaxation training, grading of severity, and developing a hierarchy for graded exposure. The patient was exposed to various physical parts of hen through virtual audio-visual methods, followed by real exposure. Least anxiety provoking stimuli were tackled first, following the general principles of graded exposure. Complete remission was achieved after five sessions of graded exposure therapy and had remained symptom-free for the next 3-month follow-up. Homework therapy assignments with relaxation exercises were asked to be continued. Written informed consent was taken from the patient and her relatives for this case report and is available for review with the authors. |
A 66-year-old female with a medical history significant for end stage renal disease, diabetes mellitus type-2, hypertension, chronic obstructive pulmonary disease, hyperlipidemia and coronary artery disease initially presented to the emergency room with altered mental status. The patient quickly decompensated and suffered a cardiac arrest necessitating advanced cardiovascular life support (ACLS). The patient was intubated, received epinephrine 1mg, and calcium chloride 1g, yielding return of spontaneous circulation. In the intensive care unit, labs revealed a potassium level of 8.5 mEq/L, for which she received emergent dialysis. She was extubated on hospital day 4 and transferred to the progressive care unit and started on her routine home medications.\nShortly after being restarted on Lisinopril, the patient complained of shortness of breath, developed stridor, and severe angioedema. Concern of lost airway prompted re-intubation. She was transferred back to the intensive care unit for further medical management. Upon meeting criteria, the patient was extubated on day six, only to be intubated again for stridor. After 9 days of intubation, it was determined that the patient required a tracheostomy. Imaging studies prior to surgery could not rule out subglottic narrowing due to the presence of an endotracheal tube, however, paratracheal edema was noted ( and ). Clinically, her minimal air leak following deflation of the endotracheal tube cuff provided evidence for the latter finding. Intra-operatively, the Otolaryngology (ENT) surgeon noted that the posterior wall of the tracheal mucosa appeared grey and friable. The distal tracheal wall was inflamed and excoriated. The surgery was successful and there were no complications with ventilation in the operating room. Tracheal positioning was confirmed using end-tidal carbon dioxide monitoring. The patient was mechanically ventilated in the ICU, but approximately 30 minutes after returning from the OR, mechanical ventilation became difficult. The ventilator sensed an increased airway pressure, and there was distension of the abdomen with each breath delivered. The possibility of a TEF was immediately considered given the friable tracheal mucosa noted in the operating room, and abdominal distention despite the tracheostomy tube being in the trachea. ENT was bedside and confirmed positioning within the trachea by feeling the tracheal rings, and using a fiberoptic scope as multiple attempts to re-intubate from above proved to be difficult. The tracheostomy tube was changed to “extended-length tracheostomy tubes (Shiley ™ XLT) in attempt to bypass a possible TEF, however this also proved to be unsuccessful. The patient desaturated and eventually lost pulses and ACLS was initiated. She was successfully re-intubated following numerous attempts by experienced providers using a combination of gum elastic bougie, video laryngoscopy and fiberoptic larynogosopy. The endotracheal tube was advanced into the right main stem in effort to bypass any TEF. Unfortunately, the patient did not return to spontaneous circulation and was declared deceased at 36 minutes. Permission for an autopsy was denied by the family. |
This study was approved by the Ethics Committee of Orthopedic Surgery Department, Imam Khomeini Hospital, Tehran, Iran and a written consent was signed by the parents.\nA 7-yr-old boy, the only child of otherwise healthy parents was referred the Pediatric Orthopedic Clinic, Imam Khomeini Hospital, Tehran, Iran on July 2018 with the diagnosis of CP. The reason for referral was the parents’ concern about the increasing severity of disease despite regular occupational therapy.\nOn physical examination, the patient was developmentally delayed, unable to walk or stand, with obvious cognitional and gross and fine motor retardation. Flexion contractures were noted in elbows, wrists, knees, and hips. There was bilateral equinovarus deformity of feet and increased popliteal angle. Plantar reflexes showed extension response and DTRs were exaggerated. Spastic response of muscles was recorded after continuous stretching. Sitting balance was extremely unstable ().\nThe patient was the result of a consanguine marriage and normal pregnancy. Birth weight was 2950 gr and head circumference and height were 35 and 47, respectively. The few first months of his life showed normal weight gaining and development. He was able to hold his head in 5 months and roll over at 7 months age. The first time the parents had been told about the possibility of an abnormality was in a routine screening at 5 months age. The pediatrician noticed a decreased head circumference growth. Further investigation showed the head circumference reached a plateau (40 cm) in its growth around 12 months age (). His general and developmental condition seemed to experience a sudden pause with progressive delay in growth and development since then. He lost his ability to rolling over and never gained any gross motor milestones. His face became expressionless and his eyes started to sink into the orbits (). Other findings were: apparent cachectic dwarfism, microcephaly, loss of facial adipose tissue, pigmented retinopathy, thoracolumbar kyphosis, multiple joint contractures, senile appearance, photosensitivity, and thin and dry hair.\nAlthough physical examination had a lot of similarity to a patient with CP, the history was inconsistent with the diagnosis of CP in its almost all aspects. This made us reevaluate the diagnosis. After a thorough history taking, some clues were added to our knowledge which was critical to the correct diagnosis. These include rapid regression of all motor functions, regression of language and fine motor functions and facial changes which are not compatible with CP.\nAt 7-yr-old age, he was in a cachectic dwarfism condition. The progeroid appearance narrowed our differential diagnosis.\nOur first diagnosis based on clinical findings and progression of the disease was Cockayne syndrome. The diagnosis was later confirmed by molecular analysis for Cockayne syndrome. The patient was homozygous for ECCR6 gene (genotype: c.2551 T>A /p.W851R- c.2551 T>A /p.W851R). The parents were also heterozygous for the same gene. This was also true for the patient’s only sister. |
An ill-appearing 19-year-old male with the one-year history of asthma presented to the emergency room with non-specific symptoms including fatigue, dyspnea, numbness in the right leg, nausea, vomiting, and dizziness. Two months prior to presentation, he had a sinus surgery and within few days after this surgery, he developed cough and dyspnea, so he was admitted to outside hospital for possible pneumonia. He was treated with cefuroxime, Tamiflu, and oral prednisone. He improved momentarily with steroids. Two weeks later, he returned to the outside hospital complaining of right foot plantar numbness and dyspnea, he was discharged home on Levaquin as they thought he may have some residual sinus disease left. One week later he was seen by a pulmonologist at outside hospital and they noticed that one of the cultures grew staph, hence started on Bactrim. He took Bactrim for three days and his mother noticed that he developed some mental status changes, hence Bactrim was stopped. After this, no more symptom of mental status change was noticed. Over the next few weeks, the patient noticed tachypalpitations, continued to have fatigue, shortness of air, and fatigue so the family decided to come to our hospital's emergency department for further workup. While in the emergency room, he was found to be in atrial fibrillation with the rapid ventricular response and elevated troponins. The patient spontaneously converted into sinus rhythm within 10 minutes. His vital signs were stable except for tachycardia with a heart rate of around 100 beats per minute. Physical examination was unremarkable with a normal sensation on right leg and foot. He was admitted to cardiac intensive care unit for further workup due to elevated troponin.\nSalient laboratory values and electrocardiogram\nThe patient’s initial complete blood count was remarkable for white blood cell of 28,800/ul with eosinophil count of 12,960/ul (45%) in spite of the use of low-dose oral corticosteroids for a few days prior to admission. Erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP) were both elevated at 35 and 4.14, respectively. His admission troponin was 16.28. His initial electrocardiogram (ECG) showed atrial fibrillation with a heart rate of 161 beats per minute, non-diagnostic Q waves in the inferior leads, T-wave inversions in the inferior leads and no significant ST segment changes noted (Figure ). His repeat ECG 10 minutes later when he converted to sinus rhythm showed sinus tachycardia with a heart rate of 100 beats per minute, Q and T changes as noted earlier, as well and no significant ST segment changes noted (Figure ). Other labs, including TSH, UDS, BNP, lactate, and renal function, were unremarkable. Rheumatological workup including anti-nuclear antibody (ANA), perinuclear antineutrophil cytoplasmic antibody (p-ANCA), cytoplasmic antineutrophil cytoplasmic antibody (c-ANCA), rheumatoid factor, myeloperoxidase (MPO) antibody, serine protease antibody 3, and anti-cyclic citrullinated peptide (anti-CCP) IgG was inconclusive. However, the patient’s IgE and IgG were both markedly elevated. Several infectious causes, such as histoplasma, coccidioides, strongyloides, cytomegalovirus (CMV), human immunodeficiency virus (HIV), tuberculosis (TB), Epstein-Barr virus (EBV), hepatitis B, and hepatitis C, were explored and all were negative.\nImaging\nTransthoracic echocardiogram revealed an ejection of 55% with some apical hypokinesis. The transesophageal echocardiogram showed no evidence of endocarditis, thrombus, shunt, or atherosclerosis. Computed tomography angiography (CTA) of the chest with and without contrast showed moderate mediastinal and bilateral hilar adenopathy in addition to bilateral axillary lymphadenopathy, bilateral peribronchial thickening, and patchy ground-glass opacities most predominantly in the posterior lower lobes. There was no evidence of pulmonary embolism (Figure ). Cardiac magnetic resonance (CMR) showed several areas of delayed enhancement within the left ventricular myocardium and decreased perfusion in the mid to apical septal and inferior segments throughout the apex. It also revealed a small pericardial effusion and minimal hypokinesis of the lateral apical wall (Figures -). Due to the abnormal myocardial enhancement, a CT of the heart with coronary CTA was ordered which showed normal coronary artery anatomy with no evidence of stenosis, calcified plaque, or soft plaque (Videos -). Due to his reported neurologic symptoms, CT of the head without contrast was ordered and showed two areas of low-attenuation within right frontal white matter. MRI of the head was subsequently performed which showed many small bilateral punctate infarcts throughout cerebrum and a few additional ones in the cerebellum.\nBiopsies\nA bone marrow biopsy showed normocellular bone marrow for age and no concern for dysplasia; however, both the bone marrow biopsy and peripheral blood smear showed marked eosinophilia with leukocytosis. Several transbronchial cryobiopsies were taken from the left lower, upper lobes and lingula of the lung which showed patchy areas of eosinophilic venulitis with dense eosinophilic infiltrates involving many of the small venules. This process was happening in the background of chronic bronchiolitis with abundant eosinophils within small airways, smooth muscle hypertrophy, and goblet cell metaplasia (which suggests asthma). All these findings taken into consideration together suggested EGPA (Figure ).\nThe patient was initially started on 1000 mg of intravenous methylprednisolone for three days and then 1 mg/kg/day of oral prednisone for several months with a gradual taper. He was also started on cyclophosphamide for three to six months. Additionally, due to the patient’s young age, arrangements for sperm preservation were made prior to starting cyclophosphamide. The patient responded well to the treatment and at his one-month rheumatology follow-up, the patient continued to improve. His troponin-I reduced to 0.08 at one month visit. |
A 21-year-old female, who has underwent renal transplantation 1 week ago, was referred to the department of nuclear medicine for renal transplant scintigraphy. She had end stage renal disease as a consequence of nephritic syndrome for the past 4 years, and was maintained on regular hemodialysis. Both her previous right and left brachial-cephalic arterio-venous fistulas had become dysfunctional, and she had been on a renal transplant waiting list since then. A catheter had been implanted in her right internal jugular vein (IJV), and she underwent hemodialysis via this route for 10 months. The catheter has been removed a few days after transplantation. Dynamic renal scintigraphy was obtained in an anterior view after bolus injection of 7 mCi 99mTechnetium-diethylenetriaminepentaacetic acid (99mTc-DTPA) through an 18-gauge venous cannula in the right antecubital vein intravenously. The images were obtained with a single-head gamma camera (Siemens ecam) fitted with a low energy high-resolution collimator. In the angiographic phase, sequential 1-second images revealed an unusual vascular filling pattern along with patent iliac arteries and transplanted kidney (). In order to gather further information on the abnormal finding, an additional angioscintigraphy of the chest in the anterior view was performed following bolus injection of 5 mCi 99mTc-DTPA through the same venous cannula 4 hours later (). Sequential 1-second images demonstrated rapid filling of the right basilic and cephalic veins followed by the axillary and subclavian veins (). Unexpectedly, two parallel channels appeared and the right heart, left heart and aorta became visible in turn. Regurgitation into the lateral thoracic and IJVs was also noted (). All findings were suggestive of SVC obstruction, which was attributed to the prolonged IJV catheterization, accompanied by blood flow through the azygos system (). The patient did not have any symptoms or signs, except some dilated collateral veins over the chest and abdominal wall that were retrospectively detected on physical examination. Due to the risk of contrast nephropathy and the patient’s being asymptomatic, the referring physician did not agree to perform computed tomography (CT) angiography to obtain detailed anatomical information. |
A 68-year-old male patient, presenting with insulin-requiring diabetes which evolved over 30 years, was referred with a pancreatic cephalic ductal adenocarcinoma after prosthetic drainage for jaundice and biopsy by echoendoscopy. Thoracic and abdominal CT showed no metastases. Liver magnetic resonance imaging was normal. According to the guidelines of the National Comprehensive Cancer Network, the tumor was considered resectable[]. Abdominal CT without contrast showed multiple calcifications in the aorta and visceral arteries, as well as calcifications in the pancreaticoduodenal arcade (Figure ). In addition to the calcifications, the arterial phase of the CT showed: (1) a focal narrowing in the proximal celiac trunk with a “hooked” appearance characteristic of a MAL; and (2) arterial supply from the SMA to the common hepatic artery via the GDA, as well as a dorsal pancreatic artery (Figure and B).\nExploratory laparotomy showed no contraindication to resection. Para-aortic lymph node biopsy showed no metastasis. Peroperative ultrasound showed a large pancreaticoduodenal arcade and a large dorsal pancreatic artery. However, the preoperative CT scan had underestimated the local extension because evidence of tumor abutment on the mesenteric vein existed. We performed a MAL division using a lateral approach allowing for a progressive division of the right diaphragmatic crus on the right side of the abdominal aorta, and the right side and the upper edge of the CA was progressively freed of all dense fibrous tissue. An additional GDA clamping test with Doppler ultrasound monitoring showed unsatisfactory restoration of the liver blood flow through the CA. Thus, considering the tumor as “borderline” resectable, revascularization of the hepatic artery and PD were both postponed. The postoperative course was uneventful. The in-hospital stay was 7 d.\nThe patient received 4 cycles of neoadjuvant FOLFIRINOX before imaging reassessment and endovascular management. Endovascular revascularization was performed 45 d after the first surgical step, during the interval between 2 cycles of chemotherapy. A CT scan showed modification of the CA/aorta “angle” after MAL release, which allowed for the possibility of a much easier stenting. Selective arteriography of the CA showed a short and significant remaining proximal stenosis of the CA. A careful crossing of the stenosis allowed angioplasty followed by stenting (Figure and B). Subsequently, the CA blood flow was restored and the duodenopancreatic arterial supply disappeared.\nAfter 6 cycles of chemotherapy with a normalization of CA19-9 and an objective response on CT, a PD was performed without vein resection (Figure ). The hepatic arterial inflow was preserved after GDA and dorsal pancreatic artery clamping and division. The divided common bile duct was well vascularized.\nThe standardized pathological examination of the specimen showed a 20 mm yp T3N1 poorly differentiated pancreatic adenocarcinoma with perineural involvement (6/10 positive nodes; lymph node ratio: 0.6). The resection was R0 as the inked margins were all negative; SMA, venous and posterior inked margins were free of tumor with a more than 1 mm clearance.\nThe postoperative course was uneventful. The patient was discharged on day 15 after equilibration of the diabetes. Adjuvant chemotherapy was performed for 6 mo. After 18 mo of follow-up, the patient was well and recurrence-free. |
A 50-year-old woman visited the emergency room of our hospital for altered mental status 1 week after PTBD. She had a history of gallbladder cancer, which required cholecystectomy, liver wedge resection, and hepaticojejunostomy. The final pathologic stage was T2N0M0. Despite 3 courses of postoperative chemotherapy, endoscopic biopsy confirmed local recurrence in the intrahepatic duct 8 months after the operation. Follow-up abdominopelvic CT revealed multiple metastases in the liver with seeding in the right subphrenic and subhepatic spaces.\nDue to the presence of strictures in the anastomotic site and biliary duct caused by the local tumor recurrence and multiple hepatic metastases, PTBD was performed 3 times with biliary tract dilatation and stent placement. However, she presented with fever and pain at the PTBD insertion site with drainage of dark blood in the tube after 5 days.\nNeurologic examination revealed a semi-comatose state with muscle weakness (muscle strength grade I) in all extremities. Brain CT angiography revealed extensive pneumocephali in the sulci of the right cerebral hemisphere with air densities in the superior frontal sulcus of the left frontal lobe (Fig. A). No significant steno-occlusive lesions or filling defects were observed on the scanned neck and intracranial arteries. CT pulmonary angiography showed a large pulmonary thromboembolism in the left main pulmonary trunk and branches of the left pulmonary arteries (Fig. B). The patient underwent central venous catheter placement in the right internal jugular vein and hyperbaric oxygen therapy. However, follow-up neurologic examination showed no improvement.\nAbdominopelvic CT revealed a large necrotic mass containing air bubbles in the right hepatic lobe, and free air and fluid in the right perihepatic and subphrenic spaces, suggestive of metastatic hepatic tumor rupture (Fig. C, D). The PTBD tube was inserted into the B3 duct surrounded by a ruptured necrotic hepatic mass (Fig. C, D). Right portal vein thrombosis and multiple metastatic masses in the subcutaneous and muscle layers of the abdomen were also observed.\nDiffusion-weighted brain MRI taken after 6 hours revealed multiple high signal intensities (SI) in the right frontal, right parietal, and left frontal (i.e., superior and medial gyri) lobes, mainly in the cortex, with low SI on the corresponding apparent diffusion coefficient map (Fig. A, B). Fluid-attenuated inversion recovery showed increased SI with cortical swelling (Fig. C). Gradient-echo sequence revealed multifocal hypointense blooming dots in the right frontal and temporal lobes, suggestive of residual air bubbles (Fig. D). Arterial spin-labeling perfusion imaging showed decreased blood flow in the right cerebral hemisphere and left frontal lobe (Fig. E). These MRI findings were suggestive of acute infarction in the right cerebral hemisphere and left superior frontal lobe, probably due to cerebral air embolism. Magnetic resonance angiography showed no significant vascular abnormalities (Fig. F).\nFollow-up brain CT taken 12 hours later showed expansion of the infarct-related edema in the right cerebral hemisphere with left-sided midline shifting (Fig. ). Asymmetric enlargement of the left lateral ventricle was also observed, consistent with obstructive hydrocephalus. Despite the use of vasopressors, the patient's blood pressure continued to drop. She began to develop progressive oliguria, metabolic acidosis, and hyperkalemia. The patient died 12 days after admission. |
A 22-year-old female presented to the emergency room after a motor vehicle accident complaining of abdominal and left leg pain, with a pain level described as 8/10. She was a belted, backseat passenger when the car was struck broadside. Airbags were deployed. She denied any loss of consciousness. Chest and cardiovascular examinations were unremarkable; however, she was mildly tender to deep palpation of the abdomen and could not move the left lower extremity due to pain in the left pelvis. Her work-up included a chest X-ray and CT scan of the chest, abdomen, and pelvis that revealed left lung contusion with herniation of the stomach into the left hemithorax (Figures –). Aside from having mild asthma controlled with an albuterol inhaler, she lacked a significant past medical history that would suggest a diaphragmatic hernia. Additional injuries (CT images not shown) included several pelvic bone fractures, left sacroiliac joint diastasis, and a fracture of the left sacral ala.\nThe patient was taken to the operating room after adequate resuscitation, with a nasogastric tube already in place. The patient was intubated using a single-lumen endotracheal tube as the double-lumen tube was deemed unnecessary by the anesthesiologist. Exploratory laparoscopy was performed. Herniated bowel through a 9 cm posterior left diaphragmatic defect contained most of the stomach, loops of small bowel, and the transverse colon (Figures and ). We determined that a tension-free repair could be performed primarily without the use of mesh, with running silk suture followed by interrupted reinforcement sutures (). At the very last stitch, prior to closure of the diaphragmatic defect, a Valsalva maneuver was performed to fully expand the lungs and expel any remaining pleural air. Therefore, there was no need for a chest tube or pleural catheter, which was ultimately confirmed by the fact that the patient never developed a pneumothorax after surgery. Upon final inspection of the abdominal cavity, there were no other signs of injury. The patient tolerated the procedure without incident and was subsequently returned to the trauma admitting unit. Following orthopedic surgical repair of her other injuries, the patient was discharged, in stable condition, five days later. |
A currently 18-month-old boy, a product of a full-term vaginal delivery following an uneventful pregnancy with a birth weight of 3000 g, was referred to our neonatal intensive care unit (NICU) due to a ventral abdominal wall defect in the periumbilical region—gastroschisis () at the age of one day. The neonate had an immediate orogastric tube placed and was given intravenous fluid (IVF) expansion with subsequent IVF with antibiotics. Gauzes soaked with warm normal saline were applied around the bowel.\nThe neonate was transferred to the OR for POCS under general anesthesia. The stomach, transverse and descending colon, and terminal ileum were all outside of the abdominal cavity and dilated without membranous covering. Bowels were warmed using gauzes soaked with warm normal saline with a trial of reduction and primary closure. PCOS failed because the abdominal cavity was too small and the bowels were too swelled. The alternative management was to put the bowels into a silo bag filled with saline and suture the bag to the fascial edges for future repair. Since we did not have the standard silo bag, we used an IV normal saline bag to make a silo.\nThe neonate was connected to mechanical ventilation (MV) and kept nill per os (NPO) postoperatively. An echocardiogram showed a patent foramen ovale, mitral regurgitation, and an evidence of increased pulmonary pressure. Due to the congenital cardiac issues, the infant remained in the NICU for three months. During this time and on subsequent stages, we moved the bowels slowly inside the abdominal cavity and put clamps onto the silo bag to keep bowels in place (Figures and ).\nOnce the bowels were inside, we chose not to close the defect by the delayed primary closure with sutures due to the ongoing cardiac issues. We left the defect opened and covered it with nonadherent dressings for further closure by secondary intention. For the very first time, we saw that the normal skin was adhering to the granulation tissue forming a protective new layer. Therefore, we did not close the defect with sutures. The results of this technique were better than we expected. It made a more cosmetic appearance with a minimally visible scar (). When we searched the literature, we discovered this sutureless technique and learned that it is gaining wide acceptance across nations.\nAfter 3 months managing the coexisted congenital cardiac disease, the infant was able to be disconnected from the MV and reached full feeding capacity. The infant did very well and was discharged home. At routine follow-ups, the infant was gaining weight and doing well. At the age of 18 months, a follow-up showed a normal-appearing child with appropriate length and weight, although with a speech delay. There were no abdominal hernias (). |
A 58-year-old male patient presented with rapidly worsening dyspnea and total dysphagia occurring within several hours. Three years earlier the patient had noted bilateral neck swelling. The swelling grew slowly and one year later the patient developed mild dysphagia. As the patient was unaware of the potential severity of the symptom, a prompt examination and treatment were not provided. This symptom did not markedly progress until the occurrence of a traffic accident. Due to this accident, the patient underwent tibiofibular fracture surgery at a local hospital. Post-operatively, the patient recovered well and ate as usual. However, three days subsequent to the surgery, the patient suddenly developed rapidly worsening dyspnea and total dysphagia. Attempts at intubation failed, so a tracheostomy was performed under local anesthesia. The patient was then referred to the Department of Otolaryngology (Second Affiliated Hospital, Zhejiang University, Hangzhou, China). A physical examination revealed an extremely large, soft, non-tender mass measuring ∼11×10×8 cm, involving the bilateral neck. A laryngoscopy showed a retropharyngeal mass that was reducing the space of the pharynx. A computed tomography (CT) scan of the neck revealed a large, well-circumscribed, fatty, dense mass measuring 11×11×9 cm, which extended from the retropharyngeal space to the sides of the neck and from the level of the hyoid bone to the superior margin of the mediastinum. The mass displaced the trachea and larynx anteriorly and the carotid arteries laterally. The mass was not enhanced following contrast agent administration (). Due to the internal fixation of the leg, magnetic resonance imaging (MRI) was not an option. In addition, a CT scan of the chest and abdomen was performed and no similar mass was observed.\nAn ultrasound-guided core biopsy of the mass revealed histological components of fibrous, vascular and fatty tissues. An incisional biopsy of the lesion was then performed with the patient under local anesthesia. The histology indicated a vascular fibrous lipoma. Subsequently, a surgical excision of the retropharyngeal mass was performed under general anesthesia using an H-shaped incision. The tumor was well-encapsulated and adhered to the posterior pharyngeal wall. The tumor was subsequently resected completely and measured as weighing 401 g (). The posterior pharyngeal and esophageal walls were completely preserved. Microscopically, the lesion had components of mature adipocytes and lipoblasts with nuclear atypia (). A diagnosis of a well-differentiated liposarcoma was confirmed. The suggested adjuvant radiotherapy was not accepted by the patient.\nDuring the post-operative course, the patient developed vocal hoarseness. A laryngoscopy revealed right-sided vocal fold weakness, which the patient recovered from one month later. The patient was decannulated and the nasogastric tube was removed. Upon follow-up at 20 months, there were no signs of either local tumor recurrence or distant metastasis. |
A 30-year-old nulliparous woman presented to the emergency department with sudden onset of generalized lower abdominal pain, mild fever, and vomiting for one-day duration on a background of three days of diarrhea. She had associated generalized aches and headache. She had a past medical history of endometriosis. She was otherwise generally fit and well, worked in administration on a remote mine site, and had recently returned from a holiday in Bali. On assessment, her temperature was 37.9°C, and she was hemodynamically stable. An abdominal examination revealed tenderness over the suprapubic area. A vaginal assessment showed a normal sized uterus and severe right-sided adnexal tenderness with no abnormal discharge.\nHer inflammatory markers were raised (white cell count 23 × 109/L, neutrophil 21 × 109/L, C-reactive protein 233 mg/L). A computed tomography of the abdomen (CT abdomen) and pelvis () showed a possible tubo-ovarian abscess secondary to an ovarian cyst or endometrioma 111 × 118 × 95 mm in size. Her liver and renal function was normal. Endocervical swabs for chlamydia and gonorrhea polymerase chain reaction (PCR) were negative, and a high vaginal swab was sent for microbiology showed normal flora. Of note, a stool PCR was positive for Salmonella species; the significance of this was uncertain. The initial diagnosis was pelvic inflammatory disease, and she was treated with intravenous ceftriaxone, metronidazole, and azithromycin as per local guidelines. After three days of antibiotics, she was well and discharged with oral azithromycin to complete treatment for possible mild Salmonella enteritis. Follow-up in the gynecology clinic was planned to discuss the ongoing management of endometriosis.\nHowever, four days later, she presented with an acute surgical abdomen and septic shock requiring inotropic support. The CT of the abdomen and pelvis revealed a large pelvic mass (120 × 130 × 150 mm in size) with copious free fluid in the pelvis and edematous bowel (). She had an emergency laparotomy and drained 1500 ml of endometriotic fluid from the peritoneal cavity. There was inflammatory exudate present over the bowel wall, and when the endometrioma was laid open, it was noted to be purulent and malodourous. A peritoneal fluid was sent for culture and grew Salmonella senftenberg. A cystectomy of the endometrioma was performed, and histology of the cyst wall was compatible with endometriosis. Infectious disease consultation was sought. She had nine days of intravenous antibiotics and was discharged on a three-week course of high dose oral ciprofloxacin, to which the organism was susceptible. The patient had recovered well on review in clinic. |
A seven and half year-old boy visited the outpatient clinic of Pediatric Dentistry Department, Faculty of Dentistry, Cairo University in June 2015 with a chief complaint of pain on the lower right molar area. The patient’s mother stated that the pain was at times throbbing in nature, and child is not able to chew on this side.\nClinical examination showed a badly decayed, lower second primary molar with related localized intraoral abscess, where the lower first primary molar was intact. The patient had poor oral hygiene; he had not received any professional dental care, and was very apprehensive.\nRadiographic examination revealed root resorption and bone rarefaction related to lower second primary molar. The interesting finding was a considerable amount of root resorption of the distal root of the adjacent lower first primary molar (\n).\nThe case was managed by performing pulpectomy\n to the lower second primary molar, with root canals filled with calcium hydroxide paste with iodoform (Metapex, Meta Biomed, Republic of Korea). The tooth was then restored with high viscosity glass ionomer (GC Fuji IX GP capsule, GC corporation, Tokyo, Japan) (\n). The lower first primary molar was not touched and instead monitored. No antibiotics or analgesics was prescribed.\nUnfortunately, the patient’s mother did not want follow-up appointments in person, however, she was contacted on the phone, after 2 weeks, 3 months and 6 months, and she said everything was fine and there was no swelling or pain.\nAt about 8 months from the treatment appointment, the patient’s mother visited the outpatient clinic with the patient for other reasons, and decided to pass by the Pediatric Dentistry Department for patient follow-up. Clinical examination showed no signs or symptoms, occlusal restoration was intact, and radiographic examination revealed arrested root resorption, on both molars, and an increase in the density of bone although this was not at a normal level yet (\n).\nshows the patient’s timeline of symptoms, treatment and follow-up. |
The patient was a 72-year-old man who had undergone a deceased-donor liver transplant for hepatitis B-associated end-stage liver cirrhosis in 1994 in a foreign country. Renal cell carcinoma with pulmonary metastasis was diagnosed in 1997 at our hospital and was successfully treated with radiofrequency ablation and thoracoscopic superior segmentectomy. There were no new metastatic lesions or recurrence for 19 months post-operatively; however, gastric cancer was incidentally diagnosed under endoscopy during the regular follow-up period. The lesion was located in the pre-pyloric antrum and measured 1.5 cm (); it was histologically well-differentiated adenocarcinoma and was confined to the submucosa on endoscopic ultrasound ().\nThe patient had other co-morbidities including hypertension and hyperlipidemia, and his current drugs included the immunosuppressive drug FK506 (Tacrolimus; Fujisawa Pharmaceutical Co., Osaka, Japan) (2 mg) once per day without additional steroids and hepatitis B immunoglobulin (Hepabig, Green Cross Co., Yongin, Korea) once per month. Pre-operative computed tomography revealed a replaced common hepatic artery which branched from the superior mesenteric artery (); this suggested that the recipient common hepatic artery was anastomosed to the replaced common hepatic artery at the site of the liver transplant. Pre-operative liver function tests were within normal limits.\nThe laparoscopic gastrectomy and lymph node dissection (D1 + β) procedures were performed in March 2009 (). The patient was placed in a supine position under general anesthesia. A traditional chevron incision had been used for the previous liver transplantation and a right paramedian incision had been used for a previous open cholecystectomy. For the laparoscopic procedure, an 11-mm trocar was carefully inserted through an umbilical incision for the laparoscopic scope using the open technique; an additional two 5 mm trocars was inserted through the left upper and lower abdominal quadrants. Severe upper abdominal adhesions were present, particularly between the liver and duodenum, but there was no evidence of metastatic lesions upon laparoscopic examination. The adhesions were carefully dissected, and trocars (5 mm and 12 mm) were inserted into the right abdominal wall (). We maintained a CO2 pneumoperitoneum below 12 mmHg secondary to hemodynamic instability due to old age and underlying disease. Previous antecolic Roux-en-Y hepaticojejunostomy and jejunojejunostomy sites were located (). The harmonic scalpel (Ethicon Endo-Surgery Inc., Cincinnati, OH, USA) was primarily used for dissection of adhesions surrounding the liver and lymph nodes, and tissue manipulation near hepatic-associated vessels was minimized during lymph node dissection due to anatomical change after liver transplantation. The trocar incision was extended 5 to 6 cm transversely at the epigastrium after the intracorporeal procedure was completed; specimen removal and the Roux-en-Y gastrojejunostomy reconstruction proximal to the afferent loop jejunojejunostomy site was performed through this incision (). Surgical time was 300 minutes, and the estimated blood loss was 300 mL.\nFortunately, the final pathologic result was tumor confined to the mucosa without lymph node metastasis. Tacrolimus level was measured daily monitoring acute post-operative rejection; pre-operative levels measured at 2.7 ng/mL and dropped to 1.5 ng/mL on the 3rd post-operative day. However, there were no signs of acute rejection. Other liver function tests including alkaline phosphatase, bilirubin were within normal range during post-operative period. The patient re-started tacrolimus with soft diet on the 3rd post-operative day, and was discharged on the 5th post-operative day without complications. To our knowledge, the patient is in good general health and has suffered no post-operative recurrence. |
An 82-year-old male was admitted to the emergency department for worsening shortness of breath and hypoxia. He was admitted a week after he was diagnosed with a left ninth rib fracture secondary to a fall. He had long-standing history of chronic obstructive pulmonary disease, coronary artery disease, and peripheral vascular disease. Chest radiographs revealed a left pleural effusion and possible infiltrate. The patient was initially treated with a nebulizer, prednisone, and empiric antibiotic coverage with ceftriaxone and azithromycin. The patient failed to improve with the medical interventions and a therapeutic thoracentesis was performed. The thoracentesis was completed with ultrasound guidance, with the puncture made above the 11th rib at mid chest on the left. The pleural effusion was found to be frank blood. No immediate complications were noted, and the patient was taken to recovery. The next day the patient was found to be in respiratory distress. A chest x-ray revealed an opaque left hemithorax that was likely rapid accumulation of pleural fluid (Fig. ). A follow-up contrast-enhanced computed tomography (CT) of the chest performed during the arterial phase revealed a left intercostal pseudoaneurysm with hemothorax and adjacent compressive atelectasis (Fig. ). Ultrasound of the left chest wall was performed (Fig. ) directly over the thoracentesis site and doppler flow revealed bidirectional fluid flow, indicating the presence of a large pseudoaneurysm (Fig. ).\nFollowing identification of the left intercostal pseudoaneurysm, the patient underwent a thoracic aortogram and multiple-level left intercostal angiogram (Fig. ) under IV conscious sedation. Selective catheterization of the T5, T6, and T7 intercostal arteries was unsuccessful in identifying the pseudoaneurysm. Selective catheterization of T10 and T11 intercostal arteries was performed with a C2 Cobra catheter, following multiple catheter exchanges due to the patient’s atherosclerotic vessels. The pseudoaneurysm was ultimately found to have a left T10 origin and the C2 Cobra catheter was exchanged for a microcatheter. Once access was gained, coil embolization of the pseudoaneurysm was performed with a series of 15 Axium micro coils. Significant room was left on both sides of the pseudoaneurysm and a follow-up angiogram was performed via the microcatheter, then a 5-French Cobra catheter. The follow-up angiogram demonstrated no further filling of the pseudoaneurysm (Fig. ). The catheter was removed and a Perclose device was placed in the left groin for hemostasis. Following completion of the procedure, the patient was taken to recovery. The patient proceeded to return to his baseline following medical management during the remainder of his hospital stay and was discharged home after 5 days. |
63-year-old woman who had suffered from the right medial knee pain for 5 years and was not responsive to conservative treatment was admitted to our clinics. 30° varus-valgus stress X-ray indicated that the patient had an intact MCL and LCL. After the detailed physical examination and reviewing of X-ray images, it was decided that UKR would be the most suitable option for the patient with anteromedial knee osteoarthritis. After spinal anesthesia application and sedation, the UKR surgery was performed with a standard minimal invasive midline vertical incision and a medial parapatellar approach; the patella was removed laterally but not dislocated or everted. The patient received a medial partial knee implant with a mobile-bearing insert (medium size with 4 mm thickness; Oxford®, Zimmer Biomet Inc., Warsaw, IN, USA). Following the UKR surgery (), weight bearing was allowed as tolerated by the patient and a standard postoperative physiotherapy was started on the first postoperative day. The patient was discharged at postop 2nd day when she met the following criteria: independent ability to get dressed, to get in and out of the bed, and to sit and rise from a chair/toilet; independence in personal care; and mobilization with crutches. After discharge, a home-based exercise program was given to the patient. At postoperative follow-up, our patient acquired a full knee RoM in the postop 1st month and returned to independent daily activities without any external support in the postop 3rd month.\nAt postoperative 1st year after first UKR application, the patient fell down while getting on a public bus; this caused that the right knee of the patient was exposed to the valgus force vector. After that moment, the patient heard a pop sound and felt an incredible pain that prohibited the flexion and/or extension of the medial side of the right knee. And then she was admitted to our emergency department. The first evaluation was performed, and the patient was diagnosed with a grade 3 MCL rupture and the UKR insert dislocation (). Having completed the preoperative preparations, the patient was operated on the same day. After anesthetic administration, a surgery with a standard minimal invasive midline vertical incision and a medial parapatellar approach (to a previous incision site) was performed to change the mobile-bearing insert with the same size (medium-sized mobile-bearing insert with 4 mm thickness; Oxford®, Zimmer Biomet Inc., Warsaw, IN, USA). After having changed the mobile-bearing insert, the MCL structures were repaired and anchored to its femoral origin with a 5 mm titanium anchor. Following the surgery, weight bearing and full RoM with a hinged knee brace were allowed as tolerated by patient and a standard postoperative physiotherapy was started on the first postoperative day. Crutches were recommended for 2 to 3 weeks to enable the patient to regain a normal gait. The brace was used continuously for 4 weeks and thereafter during the day for 2 weeks. After the physiotherapy program administration, the patient was discharged at postop 1st day.\nThe patients were evaluated regarding pain intensity (Numeric Pain Rating Scale (NPRS)), active range of motion (RoM), and quality of life (Short-Form 12 Health Survey (SF-12 Health Survey)). Functional capacity was evaluated using the Iowa Level of Assistance Scale (ILAS), Iowa Ambulation Velocity Scale (IAVS), Hospital for Special Surgery (HSS) knee score, and Timed Up and Go (TUG) test. Rehabilitation program and outcome evaluation were conducted by one clinical physiotherapist at preoperative period (before the first UKR application), at discharge (postop 2nd day after the first UKR surgery), and at postop 2nd year (after 2 years from the MCL repair and the insert change). The evaluation results are shown in . |
A 41-year-old married female applied to our clinic with a left inguinal mass. The patient had been primary amenorrheic, but had not consulted a doctor. Clinical examination showed a bulging mass of 15 cm × 10 cm on the lateral side of the left inguinal canal and another 3 cm × 4 cm mass in the right inguinal region []. The vulva and perineum were normal in appearance, the vagina was 6 cm long but ended blind, pubic and axillary hair was sparse; however, the patient had normal breast development as well as a normal female phenotype. The abdominal ultrasonography (USG) and magnetic resonance imaging revealed the absence of the uterus and ovaries. They also showed a 13.7 cm × 8 cm × 12.4 cm polypoid cystic mass with a regular surface in the left inguinal region extending from the hip joint to the level of labia majus, and a solid 3.2 cm × 2.8 cm mass in the right inguinal region. In hormonal analysis, follicle stimulating hormone was found to be 9.68 mIU/mL, luteinizing hormone was 15.74 mIU/mL, estradiol was 56.07 pg/mL, and testosterone was 1.83 ng/mL. The tumor markers including carcinoembryonic antigen, CA125, alpha-fetoprotein, lactate dehydrogenase, and alkaline phosphatase were within normal ranges. The chromosome test revealed a 46, XY karyotype. The diagnosis of CAIS was made based on these findings.\nAfter the diagnosis, we performed an exploratory laparotomy. Internal female organs were absent. The right testis was of normal size, and the left one was swollen with a cyst. A gonadal tumor in the left inguinal region measuring 13 cm × 8 cm × 12 cm and a 3 cm × 4 cm one in the right inguinal region were extirpated. Vaginoplasty was not performed as the vagina was 6 cm long, and this was thought to be adequate for sexual intercourse.\nHistopathologic examination revealed immature seminiferous tubules surrounded by immature germ cells and sertoli cells in the mass from the right inguinal region, and a serous papillary cystadenofibroma in the one from the left. No signs of spermatogenesis or leydig cell hyperplasia in the interstitial tissue were seen in both gonads [Figures -]. Estrogen therapy was initiated postoperatively. |
A 29-year-old female without any notable past history visited for further evaluation of elevated alkaline phosphatase (ALP) and a mass was found at the lower right thyroid gland during a regular checkup. A relatively firm mass of about 3 × 2 cm was found at the right anterior portion of the neck upon examination. The mass was movable, and there was no palpable lymph node.\nA clinical laboratory study performed before the operation resulted in the following: blood urea nitrogen 6.3 mg/dL, Creatinine 0.8 mg/dL, serum calcium 11.1 mg/dL, phosphorous 2.1 mg/dL, ALP 1,072 U/L, and iPTH 1,476 pg/dL.\nA round, hypoechogenic mass sized about 2 × 1.7 × 3 cm with well-defined margin separate from the right lower thyroid gland was observed under ultrasonography, and accompanied by signs of increased blood flow (). Some lymph nodes appearing to be positive reactive changes were observed inferior to the mass at level VI of right neck. Fine needle aspiration revealed Hurthle cell change of cell clusters, implying the possibility of Hurthle cell neoplasm of the thyroid gland or a parathyroid neoplasm. The immunohistochemistry resultsshowed iPTH and the clinical pattern confirmed the suspicion of parathyroid tumor, and therefore, a diagnosis of parathyroid tumor was made (). Sincecytologic study alone was unable to determine the benign or malignant nature of the tumor, a need for a frozen section of the specimen upon surgical excision was suggested. Imaging studies showed enhancement upon contrast injection, making the determination of the exact nature of the tumor more difficult ().\nThe operation was performed in June 25th, 2010. The right lower parathyroid gland did not show any direct invasion to neighboring organs on surgical examination, and a round shaped mass of about 3 × 2 cm with a smooth capsule easily separable from the thyroid gland and the surrounding muscles was found. The cut surface was yellowish, smooth, and homogenous (). Therefore, a right lower parathyroidectomy was performed, and an excisional biopsy of the surrounding lymph nodes was also performed simultaneously for frozen-sectionevaluation. The pathologic result of the frozen section reported high possibility of benign parathyroidoma, and examination of the excised lymph nodes revealed reactive hyperplasia. Therefore, no further operation was performed. The serum iPTH level immediately after the excision was 199.7 pg/dL, which became 18.54 pg/dL 1 hour later and 43.84 pg/dL 3 hours later.\nThe patient complained of a tingling sensation 2 days after the operation. Positive Chevostek sign was observed, but the patient did not show any spasmodic muscle contraction. Serum calcium, phosphorous, and iPTH at this period was measured to be 7.2 mg/dL, 1.9 mg/dL, and 102.5 pg/mL, respectively. Intravenous calcium chloride solution and 4 tablets of oral calcium agent (Dicamax 1000 tab, Dalim Biotech, Seoul, Korea) divided into 2 doses twice a day, were concomitantly administered thereafter. The serum calcium level was maintained at below normal value despite a continuous supply of calcium, and serum calcium and iPTH 8 days after operation was 7.1 mg/dL and 226.1 pg/dL, respectively. Dicamax1000 tab was replaced by Healthcal tab (Dong Wha Pharm Co., Seoul, Korea), with a daily dosage of 6 tablets divided into 3 doses three times a day.\nThe patient was discharged 18 days after operation since she did not show any particular symptoms, and laboratory study performed 174 days after surgery reported iPTH at 65.73 pg/dL, ALP at 172 U/L, serum calcium level at 9.1 mg/dL, and phosphorous at 4.3 mg/dL (). Healthcal tab was replaced by Dicamax 1000 tab with daily dose of 2 tablets divided into 2 doses twice a day.\nThe final pathologic diagnosis of the patient was minimally invasive parathyroid carcinoma, and no metastasis to surrounding lymph nodes was found (). |
EA was a 20-year-old known SCD female undergraduate in one of the Nigerian Universities. She was diagnosed of sickle cell anemia (HbSS) when she was 8 years and has been receiving medicare at sickle cell center, Benin City, Edo State, Nigeria since childhood. She was regular with her routine follow-up visits at sickle cell center.\nShe was admitted to the Central Hospital Benin City, Nigeria on April 05, 2019 on account of anemia and chronic leg ulcer of 11 months' duration. There was no improvement in the healing process as a result of poor wound care. She has been on hydroxyurea, oral Vitamin C, zinc tablets, folic acid, and antibiotics (oral ampiclox). She was admitted with a packed cell volume (PCV) of 15%. Her stable PCV was 22%. Her urine analysis, electrolytes, and urea were normal.\nThe hematologists were consulted to see her. On examination, she was pale, dehydrated, and jaundiced. Her pulse rate was 112 beats/min while her blood pressure was normal. The abdomen and chest were essentially normal. The musculoskeletal system showed an extensive ulcer at the lower one-third of the left leg over the lateral malleoli. The ulcer measured about 12 cm by 22 cm with no raised edges and having necrotic debris on the floor.\nSome parts of the ulcer were yellowish and there was obvious fluid exudation from the ulcer []. Wound swab was taken by this method: The surrounding skin was decontaminated with 70% alcohol. Wound was cleansed with sterile saline and debrided of nonviable slough and necrotic material prior to sampling for microbial culture. The margins of the wound was separated to avoid touching the wound edge with the sterile swab. The tip of the swab stick was extended deeply to reach the base of the lesion and rotated over the area of viable tissue, using sufficient pressure to extract fluid from the wound tissue and transported immediately to the laboratory for microbial culture. Wound swab for microscopy, culture, and sensitivity (m/c/s/) yielded growth of Staphylococcus aureus which was sensitive to clavulanic acid and amoxicillin. This necessitated her being placed on oral Augmentin 625 mg bid and had wound debridement. She was transfused with freshly donated packed cells, the donated or transfused blood were screened for sickle cell trait using standard methods[] to ensure only blood without the S-hemoglobin were transfused. She had red cell transfusion twice weekly till her PCV rose to 30% (hemoglobin concentration of 10.0 g/dL) as a part of hypertransfusion regimen. This was maintained for 4 months with top-up transfusions. The wound was dressed twice daily with original unadulterated honey procured from local bee-honey vendors/farmers. The wound dressing with honey was reduced to once daily after 6 weeks following a remarkable improvement with new granulation tissues and epithelization with consequent cessation of fluid exudation.\nAfter 4 months, there was total healing and closure of the ulcer []. The patient was seen at the hematology clinic on Tuesday September 10, 2019, and she was doing very well. Most importantly, she was tutored and counseled against recurrence which has been documented to occur in 80%–97% of cases after 2 years of wound healing.[] The counseling included the use of appropriate foot wears, strict adherence to prescribed medications, personal hygiene, and nutrition and regular follow-up visit to the hematology clinic. |
A 66-year-old female patient visited the Department of Oral and Maxillofacial Surgery, Seoul National University Dental Hospital, with a complaint of severe difficulty in eating due to limitation of jaw function and trismus. The patient had a history of multiple TMJ surgeries for reduction of condyle fracture of both sides and arthroplasty/coronoidectomy due to trismus. Clinically, the patient exhibited the maximum mouth opening of 8 mm and relapse of severe trismus (Fig. ). After CT scan was taken, type IV TMJ ankylosis on both sides was confirmed (Fig. ). Since the glenoid fossa and condyle of both sides were completely fused, the anatomic structures were indistinguishable. Gap arthroplasty or interpositional arthroplasty could not be applied to this patient because bony TMJ ankylosis in both sides was complete and recurrent. Therefore, we decided to use the 3D virtual surgical planning, CAD/CAM-fabricated surgical guide, and stock TMJ prosthesis system (Biomet, Jacksonville, FL, USA) for total joint reconstruction.\nThe 3D virtual surgery was performed with a specific navigation surgery software (FACEGIDE, MegaGen implant, Daegu, South Korea). As the first step, both condyles and their surrounding hyperplastic bone were removed, while making it sure to avoid the mandibular foramen. The second step was virtual placement of the condyle and fossa components of the stock TMJ prosthesis. After placement of the fossa and condylar components, any areas of interference were investigated by virtually opening the jaw.\nAfter confirmation of the position of the fossa and condyle components, the surgical guides were fabricated by using the CAD/CAM technology. The surgical guides consist of the upper and lower parts. The upper part is designed to be adapted onto the root of the zygoma, which guides the location of upper margin for resection of the root of the zygoma and the position of the fossa component of the stock TMJ prosthesis. The lower part is designed to be adapted onto the mandibular angle area, which guides the location of lower margin for the resection of the ankylosed condyle and has several drill holes for fixation screws of the condyle component of the stock TMJ prosthesis (Fig. ).\nUnder general anesthesia, the total joint reconstruction surgery was performed via preauricular and submandibular incisions. Osteotomy and placement of the stock TMJ prosthesis were carried out according to the 3D virtual surgical planning (Fig. ). At 2 months post-operative follow-up (Fig. ), the patient was able to open her mouth up to 30 mm without complication. She could undergo dental treatments that had been unavailable due to severe limitation of jaw function and trismus. One year after surgery of follow-up, the patient stated that she was living without any change of mouth opening range and side effects. |
A 53-year-old lady presented to the otolaryngology outpatient department with lateralizing neck pain and previous swelling at the site of preceding acupuncture. She had chronic migraines for which she had been attending a chiropractor. At a chiropractic consultation she underwent manipulation of her cervical spine, following which she had worsening cervical pain. On review by the chiropractor she was offered acupuncture to see of this would improve her symptoms. Immediately following the acupuncture she had acute onset right level 1/2 neck swelling, pain and bruising. She attended her general practitioner who arranged an ultrasound Doppler of the neck. This was normal with no evidence of vascular injury or pseudo aneurysm of the common, external or internal carotid artery. While the swelling subsided she had ongoing neck discomfort so a routine referral to ENT was made.\nOn review she complained of persistent right lateralizing neck pain, worse on movement but always present. She denied red flag symptoms such as dysphagia, odynophagia, dysarthria or weight loss.\nExamination showed subtle isolated hypoglossal nerve palsy with deviation to the ipsilateral side. There was no visible tongue fasciculation or muscle wasting. The remaining cranial nerves were normal as was flexible nasendoscopic examination of the tongue base, pharynx and larynx. Palpation of the neck revealed no lymphadenopathy or other swellings but there was some residual localized tenderness over the previous acupuncture site. She had no symptoms of dysarthria and was unaware of the deviation herself.\nHer past medical history included possible Sjogrens syndrome (not confirmed by rheumatology), varicose veins and chronic migraines. Her headaches had been extensively investigated by neurology and were intractable to most medication. She had been referred for greater occipital nerve block, but had self-attended a chiropractor while awaiting review.\nAn MRI scan was arranged to further investigate the apparent cranial nerve injury to ensure there was no cranial or oropharyngeal pathology. The MRI head and neck showed no mucosal lesions and no abnormality of note to explain her symptoms.\nOn review 4 months later her partial hypoglossal nerve palsy had completely resolved. She has a further review appointment to ensure no new signs or symptoms develop. |
In the period of treatment, JL was a 38 years old single man. At the time of the brain injury, 3 years earlier, JL lived alone, at what was once the family home. He had been educated to Master’s degree level, and was employed as a professional. Three years before treatment, as a result of a complex medical condition related to diabetes, he had experienced an anoxic brain injury, and upon admission to hospital he suffered three cardiac arrests in quick succession. Consistent with the common neuropsychological consequences of such damage, JL then presented with a marked impairment in laying down new episodic memories (anterograde amnesia), but a largely preserved capacity to recall events prior to it (retrograde memory).\nThe rehabilitation service’s intake report for JL indicates that before the accident, there were significant premorbid systemic issues in his background. JL had two older brothers, two younger sisters and a younger brother. His life had been fractious and unsettled, with the family moving quite frequently for work – often internationally. There was one long distance move, where the family settled for approximately 10 years. On admission to the rehabilitation services, JL reported using and abusing alcohol from the age of thirteen. However, during treatment, he informed the starting date as even earlier (10 years of age).\nJL was 20 years old when his parents separated, acrimoniously, and he recalls having to leave the country quickly, with his mother and siblings relocating to their country of origin, and living off the kindness and generosity of friends and family. JL met this adversity quite well, working during the day as an office clerk, and returning to education at night in order to obtain a Masters degree. He also trained for, and eventually worked in, a professional position. However, within a few years of returning (when JL was in his early twenties) both parents passed away. His father was the initial loss, followed 3 years later by his mother. JL spiraled into a full blown alcohol and cannabis addiction, which also led to deterioration in work performance, and subsequent loss of a series of work positions. At the same time JL began to neglect his personal well-being, his living arrangements, and more importantly his diabetes, which had been diagnosed when he was sixteen. This resulted in a series of heart attacks, and eventually the anoxic brain injury, as a result of a complex medical condition related to diabetes, a disorder often associated with mismanagement of insulin.\nSince the injury, JL has lived in a residential unit for neurological patients. Prior to this he had spent a year in a rehabilitation hospital, which he appeared to have much preferred to the current accommodation. At the hospital his days were structured and interesting. At the residential unit he became the youngest service user, and did not have much in common with the other patients. He had (and has) an older brother who lives nearby, visiting JL once a week. However, JL had little contact with the rest of his family, who live abroad. At the residential unit, his routine was highly repetitive, with much of his day consisting of listening to music, doing jigsaws, and making models. There are further activities organized by the residential unit, and the rehabilitation services which he attended twice a week. Because of his diabetes he was monitored closely by the ‘sisters’ (as JL described them), in reality nurses. This lack of freedom was a great source of frustration, and JL expressed a strong desire to live independently, or to be moved to assisted housing.\nAt the initial consultation JL was dressed in jeans, and wearing a football jersey and training shoes – which were in need of cleaning. His hair was long and greasy, and came to just above the shoulder, around his neck hung a collection of odds and ends, suspended from what looked like a piece of climbing rope, attached to a large climbing clip. The objects were nail clippers, keys, pens, a miniature torch, a bottle opener, and a mobile phone. The latter JL used as a memory aid. He also wore glasses, attached to a cord around his neck. He had a day’s growth of beard, where large areas beneath the chin had been missed when last he shaved. His fingers were nicotine stained, and the nail on the little finger of his left hand had been allowed to grow much longer than its neighbors. This idiosyncrasy endured for the remainder of the therapy, a remarkably continuous feature despite JL’s amnesia.\nUpon meeting JL for the first time, he presented as polite and friendly, and spoke both confidently and clearly, with a foreign accent. He appeared to be lucid and oriented for both time and space. When entering the consulting room he examined it with what seemed like an intelligent curiosity. However, after exchanging introductions, the amnesia presented itself almost immediately, with JL repeating information to the analyst within 5 min. The following vignette shows the impact of JL’s memory impairment in the continuity of the session, as well as the preservation of other abilities, such as his insight:\nP (Paul Moore): Maybe I’ll take some notes while we’re talking… maybe just start by just telling me about yourself.\nJ (JL): I was born in_____… left for ______ in 1981 till 1994 returned to Ireland and went to (University) at night time. I worked full time and studied part-time. I did that for 6 years… got my degree ……. and worked in the profession and after that it was a bit of mystery for me.\nP: What do you remember?\nJ: How do you mean?\nP: What happened to you?\nJ: What happened me? I don’t know.\nP: What do you remember of your early life?\nJ: Oh yeah, I remember being brought up I remember everything up to about 2 years ago…3 years ago (Suddenly the alarm goes off somewhere in the room and we both cannot find the source for a while, and analyst and patient share laughter).\nP: So you don’t remember anything after 3 years ago?\nJ: No\nP: So what happened?\nJ: I have no idea.\nP: And would you like to tell me about your life before 3 years ago?\nJ: I used to be a (professional position)…. (i.e., JL had already forgotten that he mentioned his work.)\nJ: I can’t read… I can’t get the information to stay in my head. And I don’t know why I could read and read and read and read and I would have to go back and start again… just have to keep repeating it over and over and it just won’t stay in my head (He seemed to be aware of his memory problems, but it did puzzle him why this was so).\nP: And what about listening to audio tapes, watching TV?\nJ: I can watch TV, but ask me about half an hour of watching and I haven’t got a clue about what I’ve been watching so (JL did not appear to be unduly upset about this).\nP: You can’t hold the information?\nJ: Yeah\nP: It gets dropped.\nJ: Mmm mmm it goes in one ear and out the other\nP: What is that like for you?\nJ: It doesn’t bother me at all… if there is a film on I will watch it and I will really enjoy it but don’t ask me about the film a half an hour after I was watching it. I wouldn’t even know if I was watching it or not.\nJL’s clinical presentation during the following sessions offers additional information regarding the extent of his amnesia. When collected from the reception area, JL appeared not to remember the analyst’s name, the location of the consulting room where they have met the week before, nor the layout of the building. During the initial sessions he did not formulate questions regarding why he was meeting with the analyst, nor who the analyst was. Neither were there ever any explicit references to the previous session, suggesting some level of temporal discontinuity. Nevertheless, JL could bring back topics that had been discussed during the previous meeting, most notably sports, which seemed to have been ‘preserved’ somehow in memory. Interestingly, even though this topic was similar, the emphasis put by JL was different on each occasion. The repetition of certain topics across sessions was a cardinal feature of the therapeutic process:\nJ: Did you watch any sport over the weekend? (This was a topic discussed during the previous session.)\nP: I didn’t…Did you?\nJ: No, not really. I did watch Rory McIlroy though.\nP: Do you like golf?\nJ: Yes, I used to play it…but I was never really any good at it. That’s going back a couple of years now.\nP: Did you enjoy it?\nJ: Oh yeah\nP: How are things in Bellevue (his residential unit, not the actual name. Name is intentionally changed to protect anonymity)?\nJ: Same old same old… ticking along… (2 min silence) … Did you watch any of the golf just on there now?\nP: No, I didn’t see any of it.\nJ: Rory McIlroy did very well. I don’t know what happened to Tiger Woods. He’s gone off the radar completely.\nJL’s premorbid level of general intellectual functioning was estimated to have fallen within the average range (WTAR, ). In contrast, his post-injury general intellectual functioning appeared within the low average range (Total IQ = 89, WAIS-III, ). There was no observable discrepancy between verbal and visual abilities (Verbal IQ = 88, Performance IQ = 87) and a normal performance on Verbal Comprehension, Perceptual Organization, and Working Memory Indexes from the WAIS-III. Only Processing Speed appeared below the normal range according to age and educational level (scaled score = 6).\nMemory assessment confirmed the clinical presentation of profound anterograde amnesia, characterized by a marked impairment in encoding and recalling verbal and visual information after a delayed period of time. In the Logic Memory Task (WMS-III, ) immediate recall was moderately impaired (scaled score = 6) while delayed recall was severely impaired (scaled score = 1). A similar picture was observed in the deficit recalling information, both immediately (5/50 units) and after a delayed period of minutes (3/50 units). In the Rey–Osterrieth Complex Figure (), JL only managed to recall five elements in the delayed condition, thus suggesting a similar profile to the one observed in the Logic Memory Task. It is important to note that JL did not improve significantly his performance when cues were provided in the Logic Memory Recognition task (18/50), thus suggesting that his memory impairment was a consequence of a deficit in encoding (taking in) and consolidating (learning) new information and not retrieving -as is commonly observed in patients with dysexecutive syndromes after frontal lobe damage. JL’s adequate performance on executive tasks that assess working memory (Spatial Span WAIS scaled score = 12; Digit Span WAIS scaled score = 8) as well as planning, reasoning, and problem solving (Twenty Question Task scaled score = 10, D-KEFS, ) supports this conclusion. This ‘hippocampal’ profile of memory impairment was consistent with JL’s preserved ability to focus his attention and hold onto information for seconds, on its immediate presentation. However, JL’s difficulty encoding new information meant that any distraction during the task resulted in a failure to retrieve information after the interference. |
The present 7-year-old boy was the third offspring of healthy non-consanguineous parents. He was born at 38 weeks of gestational age, weighing 3376 g. No asphyxia or other perinatal events were noted. He was able to control his head at 5 months, sat independently at 8 months, and walked at 14 months of age. When he was infected with influenza at 1 year and 5 months of age, generalized hypotonia and flaccid paralysis rapidly developed within a few hours on the second day of illness. When he was taken by ambulance to the hospital, his respiratory conditions and heart rate were unaltered, but all of his voluntary movements of the eyes, mouth, limbs, and trunk had disappeared. Both eyes remained open, but no verbal or non-verbal responses were made to external stimuli. Bouncing and rapid oculogyric movements in random directions were also prominent signs on admission. Muscle hypotonia was remarkable, while deep tendon reflexes were absent. Hypoglycemia, acidosis, and unbalanced electrolytes were excluded based on the findings of blood gas analyses, blood cell counts, and serum chemistry. Brain magnetic resonance imaging (MRI) showed no parenchymal lesions or atrophy (Fig. ). Single-photon emission tomography detected laterality in cerebral blood perfusion on the 22nd day of admission (Fig. ). Electroencephalogram (EEG) did not show any high-voltage slow waves, epileptiform discharges, or other signs of encephalopathy (Fig. ). From the third week of admission, he began to show voluntary movements in his mouth and hands. Concurrently, choreoathetotic movements in the upper and lower extremities appeared and subsequently continued for over 6 years, until the present. Accordingly, his muscle strength slowly recovered within 2–3 months after the onset, but it never returned to the prior condition. The rapid and random ocular movements were substantially ameliorated within a month and eventually disappeared at 5 years. No signs of cerebellar ataxia were evident throughout the initial admission and thereafter.\nHe had experienced three episodes of recurrent attacks with flaccid paralysis on febrile illness at 1.9, 3.3, and 5.7 years of age (Additional file : Figure S1). In each episode, the involuntary movements disappeared while generalized paralysis persisted for a few weeks. Electroencephalogram (EEG) and MRI studies did not show signs of encephalopathy or neuro-degeneration (data not shown). He had occasional anuresis without signs of paralysis or fever, but his vesicorectal functions were evaluated as normal. At present, he is unable to stand or walk alone, and his daily activity is limited since he uses a wheelchair (Fig. ). His verbal skill is severely affected by dysarthria, but he can compose sentences and perform single-digit calculations using a touch-panel display and keyboard. As such, his language perception, social skills, and other cognitive functions were considered minimally affected. He has had no arrhythmic episodes or shown any abnormal features on electrocardiography.\nAfter filtering the polymorphic variations from the WES dataset, we identified two de novo mutations in the coding regions of ATP1A3 (c.2266C > T:p.R756C) and TOM1L1 (p.Gly4Alafs*16) and one intronic deletion in C3 (c.1976-22_20TCTdel). All of these mutations were validated by the Sanger method (Fig. and Additional file : Figure S2). We considered that the de novo missense mutation of ATP1A3 was likely pathogenic in this case, whereas the effects of the other two events remain to be determined. The variant sequence in ATP1A3 encodes the protein with an amino acid substitution of Arg756 with Cys. The amino acid residue is located within a conserved sequence across species and was predicted to be deleterious with the Polyphen-2 (), Sift (), and Mutation Taster () programs (Additional file : Table S5). We further ensured that this mutation was absent in more than 500 healthy individuals. Across the whole protein structure of ATP1A3 (), the Arg756 residue was located close to the junction of the largest cytoplasmic loop and the fifth trans-membrane domain (Fig. ).\nThe p.R756H mutation, previously reported, causes atypical phenotypes of RPD []. Recently, another case carrying a de novo p.R756C mutation was shown to have similar clinical features to the present case []. To compare the phenotypic spectra of ATP1A3-assocaited disorders, we summarized the clinical features of the reported cases and those of AHC, RDP, and CAPOS (Table ). The three cases carrying mutations of p.Arg756 to Cys or His all shared the core symptoms of recurrent encephalopathy and acutely developed paralysis followed by prolonged hypotonia, dystonia, and choreoathetosis. We also verified that these cases presented with the mixed phenotypes of AHC, RDP, and CAPOS.\nPrevious studies suggested that RDP-causing mutations were associated with unstable expression of mutant ATP1A3 proteins in cultured cells. We therefore investigated whether or not the p.R756C mutation of ATP1A3 might be expressed at a lower level in HEK293T cells than the wild type and the typical AHC-causing mutant protein (p.D801N). Western blotting showed that the wild type and the two mutant ATP1A3 proteins (p.D801 and p.R756C) were expressed at comparable levels (Fig. ). |
A 75-year-old man was referred to our hospital for abdominal fullness and nausea since 2 months. He had a medical history of hypertension and hyperlipidemia and a surgical history of the right inguinal hernia. The patient's laboratory findings were within normal limits. Abdominal computed tomography (CT) revealed a well-demarcated oval isodensity mass of 25 mm at the tip of his appendix. Contrast-enhanced CT revealed a lesion with gradual homogeneous contrast enhancement from the arterial phase to the equilibrium phase (). No abnormal findings were found in the root to the middle of the appendix. Abdominal ultrasonography (US) revealed a well-demarcated hypoechoic tumor. The tumor size was 22 mm × 18 mm × 18 mm, with some cystic area and blood flow (). Colonoscopy findings were normal. The patient's symptoms naturally alleviated during examination period.\nPreoperative diagnosis indicated appendiceal neuroendocrine tumor (NET) G1 or gastrointestinal mesenchymal tumors, such as GIST. Malignancy could not be ruled out; therefore, laparoscopic ileocecal resection with D3 lymph node dissection was recommended. Intraoperative findings revealed a well-demarcated tumor at the tip of the appendix, with no invasion into the surrounding tissue. This observation was similar to the preoperative imaging findings. According to another intraoperative finding, dissecting the adhesion between the terminal ileum and the peritoneum, which was the effect of the past herniorrhaphy, was necessary. The operation time was 167 min, and the amount of blood loss was 100 ml.\nPathological findings revealed a well-demarcated tumor originating from the muscular layer at the tip of the appendix and spindle-shaped heterotypic cells proliferating in a bundle. Vascular invasion and lymph duct invasion were not detected. No tumor cells were found in the dissected lymph node. Immunohistochemical studies revealed negative values for KIT and CD34 and positive values for S-100 protein (), which confirmed the schwannoma of the appendix. The patient was discharged on the 9th day after surgery without any complication requiring medical treatment. The patient is presently doing well without any evidence of recurrence at 3 months after surgery. |
A 40-year-old woman with schizophrenia attempted suicide by falling from the 4th floor of a building. She had multiple fractures involving the calcaneus bilaterally, spine, and the right distal tibia and fibula. Plain radiographs of the right ankle showed a Gustilo–Anderson type IIIA open pilon fracture with a large segmental bone defect approximately 10 cm in size at the distal tibia (Fig. ). Because neurologic symptoms resulted from the spine fracture, the patient had to undergo an emergency spine operation. Massive irrigation and wide debridement of the right leg were performed at the same time. For the length and stability of the right leg, which was shortened by a large segmental bone defect, open reduction and internal fixation (ORIF) in the fibula were performed using a 1/3 tubular plate. And an Ilizarov external fixator was applied to the right distal tibia (Fig. ). In so doing, we maintained the length and stability of the leg.\nTo manage the large segmental bone defect, we decided to use a fibular strut allograft because massive iliac bone graft or autogenic fibular bone graft was impossible due to multiple fractures. Because the allograft is very vulnerable to infection, we delayed the operation after complete wound healing and normalized laboratory findings, such as the erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP) level. Minor debridement was performed under local anesthesia due to mild serous discharge from open wound site. The wound was completely healed after 4 weeks. Then, the external fixator was removed, and we performed ORIF using fibular strut allograft. First, the large segmental defect was exposed via an anterior approach. Second, the fibular strut allograft was impacted to the medullary canal of the tibia. Then, the fixation was performed using a periarticular distal tibia locking plate (Synthes®, Switzerland). Finally, the iliac auto-bone and calcium sulfate (bone substitute) were added to enhance bone union (Fig. ). We obtained the normal alignment and rigid fixation postoperatively (Fig. ).\nThe patient was encouraged to do active ankle motion with non–weight bearing for accelerating rehabilitation immediately after ORIF. One month later, the patient was discharged without wound problems. From 2 months after ORIF, the patient started partial weight-bearing ambulation. Four months thereafter, the patient could ambulate without any assistance and had a full range of ankle motion. At the 2-year follow-up, the patient ambulated well without specific problems (Fig. ). |
The patient, a 55-year-old male with a 10-year history of hypertension, was admitted to the thoracic surgery department of our hospital for dysphagia. Chest enhanced computed tomography (CT) revealed cancer in the lower esophagus and cardia. 1 week later, cardia cancer surgery was performed, and pathological examination showed highly differentiated adenocarcinoma; pathological classification was T4N1M0. The patient recovered after the operation and was discharged from the hospital without additional radiotherapy and chemotherapy.\nThree months later on March 5, 2019 (2019/03/05), he experienced a sudden episode of dizziness, incoherent speech, and weakness of his right limb during rest. His vital signs were stable after admission, and electrocardiography (ECG) showed no atrial fibrillation. His National Institutes of Health Stroke Scale (NIHSS) score was 6. The results of first and subsequent coagulation function tests are shown in , and accompanying inflammatory indicators (CRP) are shown in . Head CT showed an ambiguous boundary between gray and white matter in the left parietal lobe (), and chest CT showed changes in the area of gastric tumor resection – namely, multiple enlarged lymph nodes near the descending aorta. Other blood biochemical indices were normal. The patient was treated by IVT with 0.6 mg/kg rtPA 156 min after symptom onset. By the end of thrombolysis, the right limb weakness had improved, with an NIHSS score of 4; 24 h after thrombolysis (2019/03/06), CT showed multiple infarction in the left parietal lobe and multiple blood foci in the right occipital lobe (). At this time, the patient did not have obvious discomfort, and the dizziness and speech symptoms were slightly improved. Magnetic resonance imaging (MRI) performed on the 3rd day after thrombolysis (2019/03/07) revealed a large infarction area in the left temporoparietal lobe and hemorrhage in bilateral occipital lobes and the left cerebellar hemisphere (). A second coagulation function test was performed. Enhanced MRI showed that there was no tumor metastasis in the brain, and no atrial fibrillation or other arrhythmias were observed upon ECG examination. No thrombus was found by CT angiography (), and no obvious plaques were detected in the neck and lower extremities by color Doppler ultrasound. The patient has both cerebral infarction and cerebral hemorrhage, considering that antiplatelet drugs can aggravate the risk of bleeding, so he did not have antiplatelet treatment during the first hospitalization. The patient’s blood was hypercoagulable, suggesting cerebral embolism; however, a series of tests did not reveal the location of the thrombus. Therefore, a diagnosis of TS with advanced gastric cancer was made. The symptoms of dizziness and speech incoherence improved (NIHSS score of 4), and the patient was discharged from the hospital.\nOne week after discharge (2019/03/21), the patient was re-admitted to the hospital with sudden right limb weakness and speech confusion. MRI showed a large new area of scattered infarctions (), and then he was given aspirin 100 mg, clopidogrel 75 mg, and atorvastatin 40 mg. After treatment on day 13 after admission (2019/04/02), the patient showed improvement despite an episode of sudden dizziness and vomiting on day 4 (2019/04/06), and was transferred to the rehabilitation department for further treatment. The transformation of bleeding after a new infarction was observed by head CT (). ECG revealed no obvious abnormalities, and the patient was transferred to our department for further treatment on 2019/04/08. On day 3 after admission (2019/04/10), MRI revealed new ischemic foci in the left cerebellum and absorption of hemorrhage in the right occipital lobe (). A third coagulation function test was performed. The patient had a history of recurrent cerebral infarction and hemorrhage after infarction that was complicated with abnormal blood coagulation function. In consultation with the hematology department, the patient was treated with cryoprecipitate (6 U) and reexamined 2 days later (the fourth coagulation function test). Blood coagulation function was improved, and 6000 U low-molecular-weight heparin was administered to the patient by subcutaneous injection along with oral administration of Rivaroxaban (15 mg) daily for 3 days. On 2019/04/24, the patient was in a stable condition and as the brain CT showed that the bleeding focus had been resorbed, he was discharged from the hospital.\nOn 2019/05/08, the patient had sudden obliquity of the mouth and incontinence and died 2 days later, 66 days after the first ischemic stroke. No autopsy was performed. |
A 10-year-old girl presented with multiple asymptomatic papules over right lower abdomen present since the age of 2 months. The lesions were gradually progressive with a rapid enlargement in the last 1 year. There was no history of any neurological, ocular or skeletal abnormality. On examination there were multiple erythematous papules and coalescent plaques (0.5 cm to 5 cm) in a linear pattern over the right lower abdomen []. Most papules were discrete, pink, dome shaped with a few papules showing central umblication and crusting. The papules coalesced to form numerous papillomatous plaques of varying sizes (1 to 5 cm). Yellowish slough and crusting was seen on some of the lesions. The discharge from the lesions foul smelt. No regional lymphadenopathy was present. The routine hematological and biochemical tests, chest radiograph and abdominal ultrasonography were normal.\nThe skin biopsy revealed cystic invagination of epidermis with papillary projection lined by two rows of cells. There were columnar cells towards the lumen and cuboidal cells in the outer layer. Decapitation was seen in the luminal columnar cell layer. The fibrovascular core of papillae showed mixed inflammatory infiltrate comprising of plasma cells, lymphocytes and neutrophils. The underlying deep dermis showed apocrine sweat glands []. The histopathologic features were consistent with the diagnosis of syringocystadenoma papilliferum. Immunohistochemistry (IHC) staining using epithelial membrane antigen (EMA), CD56, CK 19, CK 5, P 63 and smooth muscle antigen (SMA) revealed EMA positivity in columnar cells. Focal positivity of CD 56 was seen in columnar cells. CK 19 was positive in both columnar and basal cells. CK 5 and p63 were positive in basal cells. SMA was focally positive in basal cells []. These findings were consistent with the diagnosis of SCAP of apocrine origin. There was no evidence of malignant transformation. The patient was referred to Plastic surgery for surgical excision with skin grafting. However, the patient was lost to follow up. |
The patient was a 34-year-old man who came to our hospital on May 12, 2016, with complaints of poststernal burning pain that lasted for 1 hour without relief. Physical examination and questioning revealed that the patient had chest pain lasting >1 hour which radiated to the neck, coupled with chest tightness, excessive sweating, and nausea. Emergency electrocardiography showed sinus bradycardia and ST-segment elevation in leads II, III, and aVF. This was followed by an immediate coronary angiography that showed coronary artery dilatation lesion with diffuse angiectasia, with significantly dilated vessel diameter at the dilatation site. The maximum diameter of the proximal right coronary artery (RCA) dilatation was up to 8 mm, and all 3 vessels including the left anterior descending (LAD), left circumflex (LCX), and RCA were involved. In addition, the patient's RCA showed coexisting tumor-like expansion, stenosis, and occlusion. The dilated coronary artery showed slow blood flow, and no thrombosis was found in the dilated lumen during the operation. Based on the patient's medical history, physical examination, and related auxiliary examinations, he was hospitalized for coronary heart disease, inferior wall AMI, CAE, arrhythmia, sinus bradycardia, and level I heart function (Killip classification). As the guidewire could not passthrough owing to the severe coronary artery occlusion, it was impossible to perform coronary stent implantation. The patient was hence admitted to the intracardiac intensive care unit for further monitoring and treatment. With conservative drug treatment, regular postoperative cryptogram sulfate, atorvastatin, isopropanol fumarate, the patient's condition was adequately controlled. The patient had no history of hypertension or diabetes. He had undergone left varicocele surgery 8 years ago, had a 10-year history of smoking, and consumed alcohol occasionally. The patient's father had coronary heart disease and his mother had hypertension. There was no family history of genetic disease or infectious disease.\nThe results of the emergency coronary angiography performed on May 12, 2016, are presented in Figure . The results showed RCA was the dominant artery. The LM artery terminal showed neoplastic expansion; the lumen of the LAD was irregular without obvious stenosis or obstruction. The LCX lumen was irregular without obvious stenosis or obstruction. Aneurysmal dilatation of the proximal lumen of the RCA was observed, with 100% vessel occlusion from the first turning point. The opening occluded right coronary artery was tried. Because the guidewire could not pass through the second bend, stent implantation was abandoned. Repeated angiography showed that the distal blood flow could not be restored.\nAfter admission on May 12, 2015, the patient underwent emergency green channel coronary angiography and percutaneous coronary intervention (PCI) to assess and dredge the stenosis and occlusion of the RCA lumen, to improve the treatment of myocardial perfusion, save the dying myocardium as much as possible, reduce the risk of death in the acute phase, and improve long-term prognosis. However, because the catheter could not pass through the second turning point of the RCA, stenting was not performed. The patient was transferred to the intensive cardiac care unit (ICCU) for conservative treatment. Aspirin enteric-coated tablets, clopidogrel sulfate tablets, tirofiban hydrochloride, and low molecular-weight heparin calcium was given for anticoagulation and antiplatelet therapy. Atorvastatin calcium tablets were used to regulate blood lipids. Perindopril and spironolactone were used to inhibit the renin-angiotensin-aldosterone system (RAAS) to reverse myocardial remodeling. Calcii Dibutyry-ladenosini Cyclophosphas for injection and Danhong injection were used to improve microcirculation and nourish the myocardium. After the blood pressure and heart rhythm were stable, ACEI and beta-blockers administered to resist ventricular remodeling and improve cardiac function and prognosis. After active rescue treatment, the patient recovered and was discharged. Three months later, the patient's condition was stable without chest pain and any other discomfort.\nDouble-source coronary computed tomography angiography (DSCT-CA) was performed on January 3, 2019. The results showed that the RCA was the dominant artery, and there was no obvious abnormality in the LCA and RCA openings (Fig. ). The proximal part of the LAD ramus and the initial part of the LCx ramus showed local expansion. The wall of the proximal tube of the LAD branch was thickened with the formation of mixed plaques, and the stenosis rate of the adjacent lumen was 30%. The wall of the proximal segment of the left circumflex branch was thickened with mixed plaque formation, which corresponded to about 20% stenosis. Multiple non-calcified plaques and mixed plaques were found throughout the RCA, and the corresponding luminal stenosis was mild. The RCA was locally dilated at the proximal and distal ends and measured about 8-mm wide. The lumen at the second turning point of the RCA showed severe stenosis, and the lumen before and after the second turning point of the RCA expanded. An arc-like, low-density shadow was seen in the inferior wall of the left ventricle, confirming inferior wall AMI.\nThe study plan was approved by the Humanistic Ethics Committee of Affiliated Hospital Jining Medical University (2016-FY-076), which strictly followed the principles of the Helsinki Declaration. The patient provided written informed consent. Peripheral whole blood samples (3 ml) were collected from the patient on July 17, 2020. Genomic DNA was extracted by using a DNA extraction kit (QIAGEN, Valencia, CA, USA) according to the manufacturer's instructions and stored at −80°C until further use. Briefly, 2000 bases upstream of the transcription start point of the ATG16L1 gene was selected from the GenBank database (NCBI: NC_000002.12) and the PCR primers for the promoter of the ATG16L1 gene was designed according to the sequence. The sequences of the 2 primers were ATG16L1-F: 5’-CCCAAACAAACCACAAAACC-3’ (20 bp) and ATG16L1-R: 5’-GGAGCTCACCTCCACACACT-3’ (20 bp). The polymerase chain reaction (PCR)-amplified product was a 1075 bp DNA fragment, and the primers were synthesized by Shanghai Sangon Biotech (Shanghai). The target fragment amplified by PCR was sent to Shanghai Sangon Biological Co. Ltd. for gene sequencing by the Sanger method. Chromas and DNAMAN software were used for gene sequence analysis. At the same time, the TRANSFAC database was used to carry out comparative analysis of the mutation sites in the ATG16L1 gene promoter region, to further study the influence of gene mutations on the binding of transcription factors.\nAfter sequencing and analysis of the target gene, the results showed that there were 3 mutation sites (g.233250963T>C, g.233251039T>C, and g.233251699T>G) in the promoter region of the ATG16L1 gene in the patient. The chromosomal locations of the 3 mutation loci are illustrated in Figure . Cytogenetic location: 2q37.1, which is the long (q) arm of chromosome 2 at position 37.1. In addition, after searching the NCBI database, we identified the 3 mutation sites as 3 single-nucleotide polymorphisms (SNPs) (rs1816753, rs12476635, and rs2289477) as shown in Figure . The gene sequencing chromatograms of these 3 mutations are shown in Figure . The predicted transcription factor binding sites of these mutations in Homo sapiens are presented in Table . The SNP [g.233250963T>C (rs1816753)] may create the binding sites for TRPS1 and SPDEF, and abolish the binding sites for GLI2. The SNP [g.233251039T>C (rs12476635)] may create a binding site for AP-2, and abolish the binding site for BARX2, HOX, and DLX. The SNP [g.233251699T>G (rs2289477)] may create the binding site ZNF, and abolish the binding site for NFIB, CHURC1, and HSF4. In this patient, the ATG16L1 gene was found to have heterozygous nucleotide variations of g.233250963T>C, g.233251039T>C, and g.233251699T> g, which may lead to the binding of transcription factors to the gene promoter, thus affecting transcription level, autophagy, and possibly the occurrence and development of disease. To the best of our knowledge, the pathogenicity of these mutations has not been reported. |
A 64-year-old post-menopausal female presented to our hospital with belching and bloating sensation. The patient had undergone vaginal hysterectomy 8 years back for a benign condition. Physical examination revealed gross ascites. On investigation, CA125 was 1,240 U/mL. Whole body PET CT scan revealed metabolically active lesions in both adnexa, multiple peritoneal lesions, retroperitoneal nodes, mediastinal nodes and left supraclavicular nodes. Ascitic fluid cytology was positive for malignant cells. She was diagnosed to have stage IV ovarian cancer. She received six cycles chemotherapy: initial three cycles of cisplatin and paclitaxel and subsequently three cycles of carboplatin and paclitaxel, as there was toxicity to cisplatin. Whole body PET CT scan at the end of six cycles of chemotherapy showed regression in metabolic activity of adnexal lesions, retroperitoneal nodes and supraclavicular nodes with no metabolic activity in mediastinal nodes and reduction in CA125 levels to 16 U/mL (). Subsequently she underwent optimal cytoreductive surgery including bilateral ovariectomy, total omentectomy, bilateral pelvic lymph node dissection and excision of all macroscopic peritoneal nodules except for < 1 cm multiple nodules in the bilateral subdiaphragmatic peritoneum. Postoperative period was uneventful.\nMacroscopically right ovary measured 4 × 2 × 1 cm and the left was 4 × 3 × 1 cm. Cut sections of the ovaries revealed solid areas with focal necrosis (20%). Largest peritoneal nodule over sigmoid mesocolon measured 1.5 × 1 × 0.8 cm. Largest left subdiaphragmatic nodule was 1 × 0.8 × 0.4 cm. Microscopically, monomorphic histology was noted in both the ovaries and dimorphic histologies in the sigmoid mesocolon nodule, omentum and left subdiaphragmatic nodules. One histology showed solid sheets and cohesive highly pleomorphic neoplastic cells with occasional formation of acini, slit like spaces and papillae suggestive of high grade papillary serous carcinoma (). The second histology demonstrated solid aggregates of neoplastic cells with a biphasic morphology consisting of cytotrophoblasts admixed with multinucleated syncytiotrophoblasts, a picture characteristic of choriocarcinoma (). Because of this rare combination, immunohistochemistry for beta-HCG was performed in the areas showing choriocarcinomatous area, but it was negative. Serum beta-HCG estimated postoperatively was < 2.0 IU/mL. Patient was advised for further chemotherapy in view of residual disease; however, she was unwilling for intravenous chemotherapy and hence started on oral chemotherapy (endoxan and topotecan), drugs which are also active against choriocarcinoma. Metronomic chemotherapy continued and patient is on regular follow-up for the past 1 year with stable disease. |
A 23-year-old man (weight 65 kg, height 175 cm, and BSA 1.8 m2) with a diagnosis of primitive right atrial enlargement from foetal age was referred to our Centre for cardiological evaluation. Cardiac examination showed increased heart size on percussion and a grade II/VI Levine systolic murmur. No significant pathological findings were found on pulmonary examination. Electrocardiography showed a regular sinus rhythm with a rate of approximately 60 beats/min associated with an abnormal morphology and duration of P wave (enlargement of P wave with duration of 130 msec), together with a low amplitude of QRS complexes in the limb leads. All routine laboratory studies were within normal limits. Chest radiography showed an abnormal cardiac silhouette with increased convexity in the lower half of the right cardiac border and cardiomegaly ().\nTransthoracic two-dimensional echocardiography demonstrated a huge right atrium of about 6.2 cm and a volume of 230 ml/m2, with a thick smoke pattern and mild tricuspid regurgitation. The pulmonary arterial pressure was normal (). The tricuspid valve was normal without significant annular dilation. No stenosis or abnormal displacement of the tricuspid valve leaflets was detected. No significant regurgitation of the tricuspid valve was found despite a partial distortion of the anterior leaflet and compression of the right ventricle inflow. The right ventricle appeared small and compressed anteriorly by the right atrium (area of RV: 11 cm2).\nCardiac magnetic resonance imaging showed a marked right atriomegaly (right atrium area: 66.50 cm2, volume: 220 ml/m2) and normal size of the left atrium (left atrium area: 7.02 cm2). The right ventricle was regular in size and global contractility but was partially compressed and dislocated posteriorly, due to the massive enlargement of the right atrium. The left ventricle was regular in dimension, thickness of the wall, and global/segmental contractility (FE VS = 61%). No evident transvalvular jets or areas of late gadolinium enhancement were found. The pericardium was visualized without focal abnormalities or pericardial effusion ().\nDue to the high risk of arrhythmias and thrombus formation in the right atrium, which is a potential risk for pulmonary embolism, the patient underwent cardiac surgery. Through a median sternotomy, cardiopulmonary bypass was established with standard aorta and bicaval cannulation. After the pericardium was opened, the entire anterior surface of the heart was found to be covered with a thin wall in continuity with the right atrium. No atrial appendage as such was apparent. The right atrium was fully opened. The inferior border of the atriotomy was sewn around the anterior part of the tricuspid annulus, and the superior border was brought over the lateral wall of the right atrium as a flap and sewn near the interatrial groove. This provided adequate reduction of the atrial size and reinforcement of the atrial wall ().\nThe histology of the resected atrial wall showed focal hyperplasic areas of smooth muscle cells with polymorphic nuclei surrounded by a few scattered areas of hypertrophic fibrous tissue.\nPostoperative transesophageal echocardiogram showed a significant reduction of the right atrium area (23 cm2, volume: 93 ml).\nThe patient was extubated 11 hours after surgery. Complications arose postoperatively with the early appearance of pericardial effusion with leukocytosis and elevated inflammatory markers. This was resistant to conventional medical therapy, which in the end required surgical drainage. Medical therapy of the postpericardiotomy syndrome (ibuprofen 600 mg/TID and colchicine 1 mg/OD) was continued over the subsequent 6 follow-up months without further recurrence of pericardial effusion. |
A 51-year-old female with a history of rheumatoid arthritis and a 10.5-pack-year smoking history presented with an aspergilloma in her right lung. After failing medical management, she was treated with a right pneumonectomy at an outside institution. This was complicated by BPF and empyema of the pneumonectomy cavity. She underwent two additional thoracotomies requiring rib resection, and placement of serratus anterior and later latissimus dorsi flap to close the fistula. Seven months following her last operation, she presented to us with stridor, persistent cough, and dysphagia, concerning for postpneumonectomy syndrome. Review of last computed tomography (CT) imaging from three months after the pneumonectomy revealed a multiloculated pleural space, with air fluid levels in the pneumonectomy cavity. An updated CT scan showed interval progressive rightward mediastinal shift with nearly complete obliteration of the pneumonectomy cavity by the heart (Fig. ). A bronchoscopy was performed, which demonstrated narrowing of the left mainstem bronchus (Fig. a) and stenosis of the lower lobe bronchial orifice due to external compression of the airways. Results of a previous complex right-sided BPF with two areas of disrupted bronchial staple line were noted (Fig. b).\nThe patient elected to proceed with operative correction of her postpneumonectomy syndrome. A thoracotomy in the fifth intercostal space was performed and dense adhesions in the chest with rotation of mediastinal structures were faced. Upon entering the pleural space, a small loculated serous fluid collection was encountered. To rule out an infected field, the pleural rind and fluid samples were sent for intraoperative gram stains, which returned negative. Cultures were also submitted. The mediastinum was mobilized from the chest wall, taking care to avoid damage to the muscle flaps, which had previously sealed the BPF. As a result, only the mid and inferior portion of the mediastinum was mobilized. A saline immersion test was performed to ensure the integrity of the muscle flap seal over the right mainstem bronchus. The implants were sized based on measuring the amount of saline instilled in the chest, and close hemodynamic monitoring of arterial and central venous pressures. Before placing the implants, the thoracotomy was closed temporarily after placement of implant sizers, monitoring hemodynamics to ensure there was no right heart compression. Two implants (250 mL and 100 mL) were placed into the pleural cavity, and the thoracotomy was closed. The postoperative recovery was uneventful. The patient was discharged on post-operative day 5. She noted complete resolution of her stridor, cough, as well as dysphagia four weeks post-operatively. Her post-operative chest radiograph showed partial medialization of the inferior mediastinum with persistent rightward deviation of the proximal trachea (Fig. ). At time preparation of this manuscript, the patient continues to have full resolution of symptoms at fourteen months following surgery. |
A 32 year-old woman with a history of hypothyroidism and pre-eclampsia initially presented to an outside hospital with acute onset dense left hemiplegia, right gaze preference, and left-sided neglect. Her initial National Institute of Health Stroke Scale (NIHSS) was 14 and she had an admission Glasgow Coma Scale (GCS) of 10. A computed tomography (CT) angiogram of her neck revealed complete occlusion of the right cervical internal carotid artery (ICA). She was outside the time window for intravenous thrombolysis; however, she underwent mechanical thrombectomy using a stent retreiver device and aspiration (Penumbra System®, Alameda, CA). Immediately after the procedure, there was a successful restoration of the blood flow to the distal ICA, proximal middle cerebral artery (MCA), and to the anterior cerebral artery (ACA), with residual distal M2 occlusion. She was intubated for the procedure and was extubated in the following days. Her left-sided weakness persisted and a repeat CTA showed re-occlusion of the right cervical ICA. No further intervention was done and she was treated with aspirin and statin for secondary stroke prophylaxis. The stroke was deemed cryptogenic after work-up for a potential source was negative including an echocardiogram which demonstrated a normal ejection fraction, normal left atrial size, and negative bubble study. A workup for prothrombotic and hypercoagulable states were negative as well. Magnetic resonance imaging (MRI) of the brain was done which showed a large area of diffusion restriction with corresponding decreased apparent diffusion coefficient (ADC) and T2 hyperintensity in the right frontal, parietal, temporal lobes and in the basal ganglia with areas of hypointensities on gradient echo sequencing, which suggested infarction in these areas with some areas of hemorrhagic conversion (Figure ).\nSubsequently, she was discharged to an inpatient rehabilitation center. While at the rehabilitation center, about four weeks after her stroke, she developed moderate to severe insidious onset headache. A repeat MRI, done four days after the onset of headache, showed diffusion restriction (with corresponding decreased ADC) and a ring-enhancing lesion in the right basal ganglia which involved part of the previous ischemic stroke. An extensive area of T2 hyperintensity was seen around this lesion consistent with vasogenic edema (Figure ).\nWith a recent invasive procedure along with the MRI findings, the possibility of an abscess was entertained, even though she did not have systemic signs of an infection (afebrile, WBC count 7800/mm3, negative blood cultures). She was empirically started on broad-spectrum antibiotics (vancomycin, cefepime, and metronidazole) and admitted to our institute for further management. On day three of admission to our hospital, she developed a high-grade fever and had an acute deterioration in her mentation that progressed to coma. An MRI was repeated to evaluate for any progression of the disease and to obtain stereotactic images for drainage. In addition to the previously mentioned ring-enhancing lesion, the post-contrast sequences now demonstrated enhancement of the right lateral ventricular wall which was suggestive of ventriculitis (Figure ).\nShe underwent a stereotactic drainage of the lesion, which aspirated purulent material. The patient was continued on broad-spectrum antibiotics. Vancomycin was discontinued after 10 days. Cefepime was switched to ceftriaxone, which along with metronidazole, was continued for a total of six weeks. An extensive laboratory workup was done which did not reveal a potential source of infection or immunocompromised state. Due to the high suspicion for an abscess and the purulent aspirate, a bacterial DNA probe was carried out on the aspirate, which revealed the presence of Fusobacterium necrophorum. Since Fusobacterium necrophorum is the implicated organism in Lemierre's syndrome, a surveillance of signs were carried out on the patient but failed to reveal neck pain or thrombosis of the internal jugular vein (imaged with an ultrasound of the neck). On post-drainage day one, her mental status improved and she progressed to her baseline prior to her discharge from the hospital. |
Two brothers presented to us, both with similar symptoms. Our first patient was an eight-year-old male who presented with an inability to stand or walk since the past two months, along with bilateral foot deformities. According to his father, the patient had developed a difficulty in walking and in climbing stairs, accompanied by frequent falls - about six months back. Gradually, he had lost the ability to walk even with support and was mainly confined to his bed-although he could sit up and crawl.\nThe patient’s intelligence was unaffected by the illness; he had no history of trauma, fever, fits, incontinence, or syncope and did not display vision, speech, or hearing abnormalities. A detailed review of the gastro-intestinal, genitourinary, respiratory, and cardiovascular systems showed no abnormality. The patients’ parents were first cousins, albeit unaffected by the disease themselves. However, out of five siblings, two of the patient’s sisters (12 and 14 years of age) and one brother (five years old) were affected by a similar illness. The patient had had an unremarkable birth history, had reached all the relevant milestones timely and was said to be taking a nutritionally adequate diet. As per the parents, all his vaccinations were complete and the past medical history was clear.\nOn general examination, the patient was well oriented in time, place, and person with his vitals, height, and weight all within the normal ranges. Regarding system-wise examination, the central nervous system examination showed no signs of wasting or abnormal tone in the upper limbs, the power in both the upper limbs was 4/5, and the deep tendon reflexes were normal when elicited. However, the bulk of both the lower limbs was decreased, with the right lower limb being slightly more wasted than the left. The tone was decreased as well and power in both the lower limbs was 2/5. The deep tendon reflexes of the lower limbs were absent. On further examination, Babinski sign was negative and the pupils were bilaterally and equally, reactive to light. The gait of the patient could not be assessed as he could not stand. However, there were no signs pointing towards cerebellar or cranial nerve dysfunction and mental functions and speech proved to be intact. On sensory examination, a higher threshold to pain and temperature was noted.\nOn examination of the musculoskeletal system, the patient had marked wasting in the anterior compartments of both legs. He demonstrated a bilateral foot drop with pes cavus (Figure )-more pronounced on the left side - and his feet were kept in a plantar, fixed position in the relaxed state. Contractures on the knees and Achilles tendon were seen. The upper limbs did not show any marked abnormality other than contractures over the interphalangeal joints of the fingers, with the skin prominently thicker there. The examinations of all the other systems were unremarkable.\nAs per laboratory investigations: the complete blood count, electrolytes, and creatine-phosphokinase levels were all within the normal ranges. The nerve conduction velocities were markedly decreased. A sural nerve biopsy was carried out and a subtotal reduction in myelin fibers was noted, along with focal endo-neuronal edema. No granuloma formation or inflammatory component could be identified. Based on the clinical, hereditary, and investigative findings, the patient was diagnosed with CMT disease, type 2.\nIn terms of management, no specific medical treatment is available, but the patient was referred to the orthopedic and rehabilitation departments to manage the foot deformity.\nOur second patient, brother of the first patient, was a five-year-old male who presented with difficulty in walking and frequent falls since the past two months. Gradually, his condition had progressed and his distal weakness had worsened, due to which he had been unable to walk without support since the past two weeks. Unlike his brother, the patient could still walk with support, although by dragging his feet. There was no significant difference in the history and examinations of this patient when compared to his brother and similar investigations were carried out, wherein the sural nerve biopsy showed nerve bundles with adequate myelin. Based on the above information, a diagnosis of CMT disease was made here as well and the child was similarly referred to the orthopedic and rehabilitation departments.\nConcerning the apparent autosomal recessive mode of inheritance, the parents of the boys were counseled accordingly regarding the implications of having more children in future and the manner in which their children could further propagate the condition. Additionally, they were counseled to bring in their affected daughters with reportedly similar symptoms for an evaluation and any possible rehabilitation as well, even though the parents described their status as nonambulatory. |
An 85-year-old Caucasian female nonsmoker with a past medical history significant for angiosarcoma presented to emergency department with a 2-week long history of progressively worsening shortness of breath. The patient’s angiosarcoma originated from the scalp and had extension into the face with metastasis to the lung. The patient previously had surgery to remove the angiosarcoma in the scalp and had reconstruction done. The patient was further treated with radiotherapy for the metastasis to the lung, with the latest treatment administered 2 weeks prior to her presenting to the emergency department. She had received a total of 12 MeV electrons delivered to the site with a dose of 57.2 Gy fractions in 20 fractions over a several week interval. She reported that her shortness of breath had been getting progressively worse and was associated with a productive cough. She denied orthopnea, paroxysmal nocturnal dyspnea, chest pain, palpitations, fevers, chills, or changes in weight. On physical exam, blood pressure was 182/89 mm Hg, pulse was 110 bpm, respiratory rate was 22/min, saturation was 88% on room air, and temperature was 98.3 °F. She was in moderate respiratory distress during examination. There was a large mass on the right cheek and a graft with acanthosis partially healing over the scalp. There were areas of erythema with mild bleeding and raw surfaces on the scalp as well. Lungs examination revealed a resonant chest with a tympanic node overlying the anterior apices, diffuse resonance bilaterally, decreased air entry, and severely diminished breath sounds. Laboratory data showed a hemoglobin of 11 g/dL, hematocrit of 34%, white count of 11,000/μL, platelets of 371,000/μL and an INR of 1. Basic metabolic panel was within normal limits. ECG showed sinus tachycardia. Chest X-ray showed bilateral pneumothoraces at least 50% on the left and 70% on the right (). The patient was diagnosed with radiation-induced spontaneous pneumothorax and bilateral chest tubes were placed by the cardiothoracic surgeon. Patient was discharged home after few days and she was doing fine after 6 months of follow-up. |
A 72-year-old male patient who had a history of hypertension, hyperlipidemia, and thrombocytosis presented with generalized seizure. T2-wighted magnetic resonance imaging (MRI) at admission revealed focal edema in the left posterior temporal lobe []. Susceptibility-weighted imaging revealed the presence of microbleed within the edematous area in the brain []. The seizure was controlled by the oral administration of levetiracetam (1000 mg/day). DSA revealed dAVF in the superior wall of the left transverse sinus, which was fed by a single artery (the left occipital artery [OA]) and drained into a single vein (the left temporal cortical vein), without drainage into the left transverse sinus [-]. Left internal carotid artery angiography revealed segmental stenosis of the left transverse sinus, which was adjacent to the shunting point []. This finding indicated the presence of thrombosis in the left transverse sinus, which can be a cause for the development of Cognard Type III dAVF. Since endovascular access to the shunting point was difficult due to the tortuosity of the feeding artery, surgical interruption of the intradural draining vein was performed in the hybrid operating room.\nAfter the induction of general anesthesia, a 4-F catheter was placed in the left common carotid artery (CCA) through the right radial artery. Then, the patient was placed in prone position. Left CCA angiography that was performed before craniotomy revealed the presence of dAVF []. Left temporo-occipital craniotomy was then performed, and the left OA was excised during craniotomy. The shunt was not observed during the second DSA conducted after craniotomy []. However, after dural opening, the draining vein was discovered as a red vein, indicating the presence of the remaining shunt []. Abnormal arterial vessels were accumulated in the dura adjacent to the shunting point. IA-ICG videoangiography was performed using the same catheter used in DSA using the microscope with integrated ICG technology (ZEISS KINEVO 900, Carl Zeiss CO., Oberkochen, Germany). ICG videoangiography after craniotomy revealed early venous filling of the draining vein, which clearly indicated the remaining shunt []. We resected the shunting point along with the sinus wall and arterialized draining vein []. The shunt was not observed during IA-ICG videoangiography conducted after resection [].\nPostoperative T2-weighted MRI revealed rapid improvement of the edema [] and magnetic resonance angiography showed the elimination of the shunt []. The postoperative course was uneventful, and the patient was discharged without any neurological deficits. |
A 70-year-old woman presented with a 6-month history of asymptomatic multiple yellowish plaques on both lower extremities, including thighs and legs. She had been diagnosed with mycosis fungoides 7 years ago and had been treated with PUVA therapy, narrow-band UVB therapy, and acitretin medication for 5 years (). She had been treated with PUVA therapy firstly for 25 months and then treated with UVB therapy for 21 months. The phototherapy was done once a week or twice a week. The total number of exposure of PUVA was 72 and the cumulative PUVA radiation dose was 216.9 J/cm2. The total number of exposure of UVB was 98 and the cumulative UVB radiation dose was 110.35 J/cm2. Finally, she reached complete remission of mycosis fungoides (). However, new yellowish lesions started to appear 1 year after discontinuing the phototherapy. A physical examination revealed multiple yellowish plaques on both lower extremities (). The plaques were well circumscribed and slightly elevated (). Laboratory tests, including a complete blood cell count, differential leukocyte count, erythrocyte sedimentation rate, and blood chemistry studies were all normal. A biopsy specimen showed multiple nodular deposits of eosinophilic amorphous material in papillary dermis and upper reticular dermis (). The deposits represented apple green birefringence on Congo red stain viewed under polarized light microscopy (). The acellular small nodules in the upper dermis consisted of randomly oriented, non-branching, non-anastomosing 6.67~12.7 nm thick amyloid fibrils on electron microscopy (). Therefore, we confirmed the diagnosis of secondary cutaneous amyloidosis. We reviewed past biopsy slides to determine when the amyloid had formed. Dense atypical lymphocyte infiltration in the dermis and epidermotrophism without evidence of amyloidosis were observed at the mycosis fungoides diagnosis 7 years ago. We found small multifocal deposits of faintly eosinophilic amorphous material confined to the papillary dermis but no evidence of mycosis fungoides 2 years ago during complete remission of the mycosis fungoides. |
An 8-year-old girl with insulin-dependent diabetes mellitus was referred to the Pediatric Neurology Department for gait problems. She had been asymptomatic till 7 years of age when she developed polyuria, polydipsia, and acute onset vomiting along with high blood sugars, increased urine and blood ketones, and metabolic acidosis. A diagnosis of diabetic ketoacidosis with underlying diabetes mellitus was made, and she was started on insulin along with dietary and lifestyle modifications. The child responded well and was on regular follow-up with well-controlled sugars and normalization of hemoglobin A1c levels. Around a year later, she complained of gait problems. She had developed gradual onset progressive imbalance for 3 months, had frequent falls, and could not climb stairs without support. She could use her upper limbs well and did not have problems with handwriting. There was a history of slipping of slippers but no sensory loss. Examination revealed an ataxic gait with an inability to tandem walk or stand on one leg. There was no vision, hearing, speech, or cognitive issues. Deep tendon reflexes were absent in the lower limbs and power was 4/5 in distal lower limb muscles and normal elsewhere. Plantar reflexes were normal. She had loss of vibration sense in the lower limbs, but other sensations were preserved.\nNerve conduction velocity studies revealed an axonal type of generalized sensory neuropathy []. The presence of diabetes mellitus along with early-onset sensory neuropathy raised the suspicion of FA, and a two-dimensional echocardiogram was ordered. There was left ventricular (LV) concentric hypertrophy with infiltration (LV mass – 76.1 g) with impairment of LV function (ejection fraction – 52.5%). Trinucleotide repeat study for FA showed abnormally expanded GAA repeats on both alleles (>200), thus confirming the diagnosis. The patient was started on idebenone and enalapril along with continued management for diabetes. Ophthalmological and orthopedic screening examinations were normal. Parents were found to be heterozygous carriers for the mutation. She has been started on physical and occupational therapy and is on regular follow-up. Genetic counseling has been provided to the family. |
A 46-year-old Bahraini female diagnosed as premature ovarian failure at the age of 29 years treated with hormonal replacement therapy presented with a history of epigastric abdominal pain and vomiting at the age of 37 years. Biochemical and radiological assessment showed features of acute pancreatitis in terms of elevated pancreatic enzyme level, and CT abdomen finding showed edematous pancreas with normal ductal system. It was attributed to hormonal replacement therapy after thorough investigation. Although the patient had stopped the implicated medications, she still had recurrent attacks of acute pancreatitis.\nSince there was no obvious cause found for her recurrent episodes of pancreatitis, autoimmune pancreatitis was suspected.\nThen, she underwent endoscopic ultrasound in 2015 which revealed mass swelling at the duodenal ampulla, and biopsy was taken. The biopsy showed ampullary adenoma with high-grade dysplasia (Figures and ).\nThen, the patient decided to go abroad for further assessment where she underwent Whipple's procedure and histopathology confirmed the presence of ampullary adenoma with high-grade dysplasia.\nUnfortunately, she continued to have recurrent episodes of pancreatitis despite the removal of the ampullary adenoma.\nIn 2016, while she was admitted under care of a surgical team for another episode of pancreatitis, she was reviewed by the rheumatology team to rule out autoimmune condition. Therefore, IgG4 level was tested (1.49 g/L (149 mg/dl)). The biopsy was reassessed and found to have increased IgG4-positive plasma cells around 30–40 per high-power field with the background of adenoma with high-grade dysplasia. Accordingly, she was diagnosed to have both IgG4-related disease and ampullary adenoma.\nShe was started on oral prednisolone 0.5 mg/kg and rituximab therapy with significant improvement over 1 year of follow-up as the pancreatitis attacks have reduced from around once in every month to around once in every 3 to 4 months after 3 months of rituximab therapy, and currently she remained attack free for around one year. |
A 28-year-old woman initially presented with blurry vision that developed over the span of approximately one month. The blurry vision was initially most prevalent on horizontal gaze but progressed to include vertical gaze. It resolved with closure of one eye. She reported a history of gradually worsening headache over the past several years. Her headaches both worsened in intensity and increased in frequency, until it was quite debilitating and occurred daily. She described the headache as an intense pressure in both the front and back of her head. She also noted a “whooshing” sound in her right ear. She denied any nausea or vomiting and had not had any syncope, numbness, weakness, facial droop or slurred speech. Furthermore, she had no history of bladder or bowel dysfunction.\nHer medical history was pertinent only for obesity with a body mass index (BMI) of 39. On physical exam she was noted to have papilledema. Her neurological exam was unrevealing with the exception of a subtle sixth cranial palsy.\nA magnetic resonance image (MRI) was obtained which showed a T1 hypointense and T2 hyperintense cystic lesion arising from the pineal gland measuring 2.0 x 1.1 cm in the sagittal plane with mild mass effect on the tectum and partial effacement of the cerebral aqueduct (Figures , ). The lesion demonstrated a thin rind of contrast enhancement and had thin enhancing internal septations. The lateral ventricles were mildly enlarged. There was no restricted diffusion and no loss of gray white differentiation. Cine flow study noted cerebral spinal fluid (CSF) flow through the cerebral aqueduct. Based on the radiographic images, the most likely diagnosis was an atypical pineal cyst.\nGiven the rapidity of the vision changes, the decision was made to pursue surgical intervention. An endoscopic third ventriculostomy (ETV) with pineal cyst fenestration was performed without complication. A computed tomography (CT) scan obtained post-operatively noted questionable decompression of the lateral ventricles but the patient reported no improvement in symptoms (Figure ). Ophthalmologic evaluation noted worsened papilledema. At this time the patient underwent a lumbar puncture, which noted an opening pressure of 32 cm H2O. Subsequent catheter venography noted severe stenosis of the right transverse sinus associated with a 9 mm Hg trans-stenosis gradient (Figure ). Placement of a venous sinus stent obliterated the pressure gradient (Figure ).\nAt six-month follow-up, her blurred vision, headaches and papilledema had all resolved. She reported complete resolution of her symptoms and plans were made for continued annual follow-up to monitor symptoms and ensure patency of the stent. |
An 86-year-old man with chronic acid reflux, cryptogenic cirrhosis complicated by hepatic encephalopathy, ascites, and nonbleeding esophageal varices presented to the emergency department with progressive epigastric pain. A portable chest radiograph revealed a 20-mm, disc-like radiopaque foreign body within the middle one-third of the esophagus, which was seen migrating into the gastric fundus 1 hour later on sequential noncontrast abdominal computed tomography imaging (Figure ). The patient denied foreign-body ingestion, including button battery or coin. Given the history of hepatic encephalopathy and the resemblance of a button battery on imaging, the patient underwent an emergent push upper endoscopy in adherence with guidelines from the American Society for Gastrointestinal Endoscopy on prompt removal of suspicious ingested foreign bodies. The foreign body was not found on endoscopy to the proximal jejunum. After procedure, follow-up radiographs no longer demonstrated the foreign body, and it seemed to have vanished.\nFurther detailed review of the patient's medications identified tablets of bismuth salicylate with calcium carbonate, which the patient admitted to swallowing whole routinely, including just before presentation. Bismuth is notably radiopaque (Figure ). We suspect that the tablet simply disintegrated in the stomach before the esophagogastroduodenoscopy based on the similar appearance of additional tablets he had on his person, detailed review of the computed tomography scan demonstrating subtle ill-defined edges of the ingested object not appreciated on initial chest radiographs, and absence of foreign body on the subsequent examination. The patient had an uneventful postendoscopic course and was safely discharged on a proton pump inhibitor for dyspepsia from the emergency department.\nSome medications, including barium, lead, bismuth, iodine, and arsenic, are readily opaque on imaging and should be considered in evaluating vanishing radiopaque objects in the GI tract. This case highlights lessons learned of a rare situation in which ingesting whole tablets of bismuth subsalicylate can mimic a button battery, where a thorough history and knowledge of radiopaque medications could have mitigated the need for invasive procedures. |
The patient is a 39-year-old male with a clinical picture that began at six years of age. He presented muscular weakness of progressive establishment, with a frequency of one episode per month, which worsened with intense physical exertion, associated with intercurrent febrile symptoms that limited his ability to perform physical activity. The crisis was variable in intensity, some of them presenting only with mild to moderate weakness and some others with complete paralysis. During infancy, nonpotassium alteration was documented during the crisis. Although the situation was periodic, the patient did not suffer motor development delay, and he achieved all his milestones without difficulty.\nCrisis worsens in adulthood, being more severe and related to physical exercise of moderate intensity. No other triggers were identified as drugs or anesthetic events. Many of the episodes required short-term hospitalizations lasting two or three days. During hospitalizations, several electrocardiograms, brain MRI, and toxic profiles were normal. Other paraclinical tests, such as renal and thyroid function, hepatic function, screening for Pompe disease, and levels of lactic acid and pyruvate, were within normal limits.\nDuring the last crisis, the only positive findings were mild to moderately increased CPK levels (values ranged between 209 and 873 U/L). Also, potassium measurements reached levels until 5.3 mEq/L in blood samples analyzed during the weakness and paralysis episodes. The electromyography study performed by multiMUP (multimotor unit potential) showed no abnormalities. These preliminary findings were interpreted as compatible with muscle fiber disease.\nHe did not present with important personal antecedents, although he stated that a paternal great-uncle died at age 50 due to a condition associated with motor impairment; however, in that case, there was no specific diagnosis.\nOn physical examination, the patient presented weakness of proximal predominance in all the four extremities, with 4+/5 strength in shoulder abductors and adductors, elbow flexors, and extenders; in lower extremities, he presented 4/5 strength in hip flexors and extenders, as well as knee flexors. The rest of the muscle groups were within normal limits. He also showed four limb hyporeflexia, normal muscle tone, and gastrocnemius hypertrophy (see ).\nA muscle biopsy performed at sixteen years old showed isolated muscle necrosis with reparative changes. A second biopsy performed in adulthood showed changes of denervation, regenerating fibers, increased lipid deposition, and intracytoplasmic glycogen deposits.\nConsidering the histopathological findings of the second biopsy, an NGS (Next-Generation Sequencing) genetic diagnosis panel was requested, which included the PYGM, PGAM1, PFKM, and LDHA genes for glycogen storage disease. The NGS panel showed no alterations.\nLater, due to the course of the disease and the presence of relapses suggestive of periodic paralysis associated with abnormal potassium levels, it was decided to request a new genetic panel for periodic paralysis, which included the CACN1S, KCNA2, KCNJ2, KCNJ5, and SCN4A genes.\nThe panel demonstrated a missense variant in SCN4A c.4483A > G (p.Ile1495Val.). This variant has not been previously reported in the literature. However, it is considered likely pathogenic according to the ACMG/ACP standards [] and Sherloc criteria []. None of the parents have the same variant, and none of them or any other family members were symptomatic.\nBased on clinical presentation, the increased levels of potassium during the crisis, and the likely pathogenic variant found in gene SCN4A, we decided to start treating the patient as a case of hyperkalemic paralysis, prescribing hydrochlorothiazide 25 mg daily, subsequently increasing it 50 mg daily. A comprehensive rehabilitation plan tailored to the specific pathology was also established and specific diet recommendations. With this treatment, the patient presented a marked improvement in both weakness symptoms and functionality, and the frequency of weakness episodes was reduced. |
A 35-year-old, right dominant handed man, involved in road traffic accident while driving two wheeler had sustained injury to left forearm, resulting in obvious clinical deformity. No neurovascular deficit was evident. Radiograph revealed comminuted, multifocal radial shaft fracture in combination with displaced two part diaphyseal fracture of the ulna(). Within 24 h, an open reduction and internal fixation of the fracture was performed.\nUnder general anesthesia, using a direct subcutaneous approach to the ulna, the ulna was reduced and fixed with a seven-holed Recon plate. The radius was exposed through Henry’s approach[], manual reduction was achieved and two square nails were passed retrograde. Careful handling of the soft tissues was paramount and extra care was taken to avoid devascularization ofany of the bone fracture segments. The decision for squarenails was taken because of commination. Incision length for ulna was 8 cm and for radius 5 cm. After wound closure, an above-elbow back slab wasapplied with the elbow held in 90° of flexion. The forearm was held elevated in a sling and our patientwas monitored for signs of compartment syndrome. Our patient was discharged from hospital after 48 h of observation in a broad arm sling; there were no immediate post-operative complications. Post-operativeX-ray was satisfactory().\nAfter 2 weeks, the sutures were removed. There was no neurovascular deficit. Back slab was continued for total 3 weeks from surgery and assisted a range of movements was started after suture removal. At 6 weeks follow-up, our patient showed further functional improvement. Results as seen on radiographs were satisfactory. After 3 months, our patient returned to work as an administrator. Patient was able to perform his routine activities with his left forearm without any difficulty except terminal supination and was started on physiotherapy. Patient was followed till 2 years and showed good functional and radiological outcome (). Our patient completed the disabilities of the arm, shoulder and hand (DASH)[] questionnaire and scored 52.1 (measures scaled on a 0-100 scale: A higher score indicates greater disability). |
A 77-year-old man attended our services with exertional dyspnoea secondary to aortic valve stenosis. He received an orthotopic heart transplantation (HTx) in 1994 for idiopathic dilated cardiomyopathy (DCM). Unfortunately, we have no records of the patient's transplant operative data given the fact that his procedure was done 23 years ago. He remained asymptomatic during follow-up except for paroxysmal atrial flutter for which he received a single chamber pacemaker in 2008 and later, atrial flutter ablation in 2010. Patient was adherent to his medication regimen and did not show any signs of transplant rejection on several cardiac biopsies. His post-transplant cardiovascular risk factors included systemic hypertension, dyslipidaemia, and stable stage 4 chronic renal dysfunction (eGFR 23 mL/min/1.73 m2). Serial transthoracic echocardiography (TTE) performed in our institution showed progressive degenerative aortic valve disease.\nAt presentation, his TTE showed degenerative bicuspid aortic valve with fusion of the right and left coronary cusps by an incomplete raphe. The appearance of the valve was consistent with severe aortic stenosis which was confirmed by hemodynamic Doppler assessment that revealed a peak gradient of 65 mm Hg, aortic valve area of 0.9 cm2 derived from the continuity equation and a dimensionless velocity index (DVI) of 0.24. Left ventricular function was normal with an ejection fraction (EF) of 59% by Simpson's method. Further evaluation of the aortic valve and aorto-iliac anatomy was pursued by a Multi-detector computed tomography (MDCT). It confirmed the morphology of a heavily calcific BAV, the absence of associated aortopathy, and suitability for transfemoral approach. The maximal aortic annulus dimension was measured as 25 mm with an aortic root diameter of 32 mm at the level of the sinuses of Valsalva. Coronary angiography was performed to screen for cardiac allograft vasculopathy (CAV) which did not show any evidence of obstructive coronary disease.\nIn addition, he was noted on admission to be bradycardic with episodes of second-degree mobitz type 2 atrio-ventricular (AV) heart block. Electrophysiology service was consulted and decided the need to upgrade his pacemaker to a dual-chamber system following the TAVI procedure.\nHis case was discussed at the Heart Valve Team meeting with a consensus that TAVI would be the optimal intervention strategy being a high-risk surgical candidate with a Society of Thoracic Surgery (STS) predicted risk of 30 days mortality of 7.035%.\nThe TAVI procedure was performed according to the standard local TAVI protocol. Vascular access was obtained with ultrasound guidance under local anesthesia and conscious sedation. Heparin (6000 units) was given intraoperatively to achieve an activated clotting time (ACT) greater than 250 seconds. A balloon expandable 29 mm Edwards Sapien 3 transcatheter heart valve (Edwards Lifesciences, Irvine, CA, USA) was advanced via the right femoral artery through the calcified, transplanted native aortic valve without prior balloon aortic valvuloplasty. Final positioning was confirmed by fluoroscopic guidance. Under rapid ventricular pacing, by temporary pacing wire via the left femoral vein, expansion of the prosthesis over the stenotic valve was accomplished with excellent results and no immediate complications. The total amount of contrast used was 60 mL and subsequent renal function tests were stable. His pacemaker was electively upgraded to a dual-chamber system the following day as planned earlier due to pre-existing high degree heart block. Pre-discharge TTE revealed a well-positioned aortic valve prosthesis with a peak and mean trans-prosthesis gradients of 14 mm Hg and 12 mm Hg respectively. There was no evidence of valvular or paravalvular regurgitation on color flow Doppler and the LV systolic function remained normal.\nPatient showed immediate symptomatic and hemodynamic improvement and was discharged from hospital 48 hours post index procedure. He was maintained on his regular medication including the immunosuppressive therapy. At the routine 1-month clinic follow-up the patient was doing well and did not report any symptoms with no limitation of his physical activity (NYHA 1). |
A 42-year-old woman, who was pregnant with twins, was scheduled for cesarean section at 37 weeks of gestation under combined spinal and epidural anesthesia. The woman had atopic dermatitis but no past history of drug allergy. After arrival at the operating room, intravenous administration of hydroxyethylated starch was started. Combined spinal and epidural anesthesia was administered in the right lateral position, at the L3–L4 and the Th12–L1 interspaces, respectively. After local infiltration of 6 mL of preservative-free 1% mepivacaine, the epidural space was identified by loss of resistance to saline, and an epidural catheter was placed. An aspiration test was confirmed as negative, and a test dose of 1% mepivacaine (3 ml) was administered via the catheter. During spinal anesthesia, maternal blood pressure became unmeasurable with a noninvasive blood pressure monitor. Lumbar puncture was successfully performed, and 10 mg of 0.5% hyperbaric bupivacaine and 20 μg of fentanyl were intrathecally administered. After returning the patient to the supine position, her face was swollen, and she exhibited erythema all over the body. Maternal blood pressure and heart rate were 74/56 mmHg and 112 beats/min, respectively. The mother was diagnosed with anaphylaxis and immediately received infusion of Ringer’s solution with left uterine displacement. Intravenous phenylephrine was intermittently administered. Despite hemodynamic instability, the mother’s breathing remained stable at 98% of oxygen saturation on room air, and oxygen was administered via a face mask. Fetal heart rate monitoring revealed sustained fetal bradycardia of 80 beats/min. Maternal systolic blood pressure remained around 80–90 mmHg under repeated administration of phenylephrine and transfusion. Fetal bradycardia was not recovered. After confirming the sensory block level of Th4, it was decided to proceed with cesarean delivery. The infants were delivered 17 and 18 min after anaphylaxis onset, both without spontaneous respiration, and were intubated and transferred to the neonatal intensive care unit. At 1 and 5 min, the apgar scores were 2 and 4 for the first infant and 2 and 5 for the second infant, respectively. Analysis of the umbilical artery blood revealed a pH of 6.842 for the first infant and 6.775 for the second infant. After delivery, the mother’s vital signs were recovered and remained stable. A two-phase allergic reaction was prevented through administration of 500 mg of methylprednisolone. After surgery, the mother was continuously monitored in the maternity ward, and her clinical course remained uneventful, being discharged 6 days after surgery. The first infant was extubated 2 days after birth and discharged 13 days after birth, whereas the second infant required further examination and treatment after having seizures. He was discharged with an oral anticonvulsant 16 days after birth, after which he remained seizure-free. The anticonvulsant was discontinued at 6 months of age. Currently, the twins are 4 years old and exhibit no developmental problems.\nWe suggested the patient to be examined to determine the causative agent of anaphylaxis. The patient agreed to be subjected to the basophil activation test (BAT), but not the skin test. The BAT was performed 9 months after the operation for mepivacaine and hydroxyethylated starch, providing a positive result to mepivacaine (Fig ). The patient was then suggested once more to undergo the skin test to confirm the accuracy of the BAT results and to investigate cross-reactivity with other local anesthetics. The skin prick test was performed 10 months after the operation, for latex, hydroxyethylated starch, procaine, lidocaine, bupivacaine, and mepivacaine, providing a positive reaction to both lidocaine and mepivacaine (Fig ). |
A 58-year-old man visited our clinic with a complaint of unsteady posture. In March 2001, he was found to have an aneurysm in the left anterior communicating artery, and he underwent the clipping procedure for this aneurysm. After surgery, the patient reported a tremulous feeling in the right leg when he was standing, but not when walking or lying. The right leg’s tremulous feeling during standing soon disappeared. However, due to a subsequent syncopal episode, he was given a brain MRI in July 2006. This MRI showed cerebromalacia in the left frontal area and total occlusion in the distal left internal carotid artery (). In February 2007, the patient visited our clinic due to disequilibrium during standing. Although his posture appeared abnormal during standing and turning, he had never fallen while standing or walking. He had no family history of neurological or psychiatric disorders and his past medical history was unremarkable. On examination, his speech was normal and he had no rigidity or bradykinesia. However, he showed all the frontal lobe release signs, including glabellar, snout, and palmo-mental signs. With regard to motor power, he showed mild weakness in the right upper extremity and hypethesia (2/10) on the right side of his body on the touch and pinprick test. Deep tendon reflexes were normal. His routine laboratory results were within normal limits and his brain single photon emission computerized tomography showed decreased blood flow in the left frontal area (). When he stood up from a chair, his posture appeared unsteady; that is, for a while he would experience a bouncing posture, sometimes mixed with an ataxic posture. However, his feet moved forward readily, there was no freezing feature during walking. His stride and stance were also normal during walking, but when he halted, especially while turning, the bouncing and/or ataxic posture would re-appear. On the pull test, his bouncing posture became aggravated but he did not fall down. We performed a placebo test to evaluate the patient for psychogenic disorder. One minute after the placebo was injected, his abnormal bouncing posture diminished a little, and five minutes later, his posture on standing was normal. However, nine minutes after the placebo injection, his abnormal posture re-appeared (). We suggested that he had a psychogenic balance disorder and referred him to the psychiatric department. |
A 56-year-old male presented to the emergency department (ED) with significant substernal chest pain starting 30 minutes prior to arrival. The patient was immediately brought back to an exam room after an electrocardiogram (ECG) was performed and was seen by a provider within 10 minutes of registration (Figure ).\nThe initial ECG revealed atrial fibrillation with a rapid ventricular response, rate of 147, with minimal ST depression within the lateral leads but was without apparent ST-segment elevation. On initial assessment, the patient had point-of-care labs immediately drawn (a basic metabolic panel and troponin), and a chest X-ray performed to evaluate for a possible aortic dissection which revealed no gross abnormalities (Figure ).\nWith a non-diagnostic chest X-ray alternative diagnoses were pursued. The ultrasound fellow in the department was consulted for an immediate cardiac ultrasound for evaluation of right heart strain secondary to pulmonary embolism. During the bedside cardiac ultrasound, the patient experienced ventricular fibrillation (Video ), and CPR was immediately started.\nThe ACLS algorithm was followed for pulseless ventricular fibrillation, and the patient received multiple rounds of epinephrine, 450 mg of amiodarone (300 mg and then 150 mg), and three conventional defibrillations with increasing joules at 150 J, 200 J, and 200 J (the departmental defibrillators are biphasic and have a maximum output of 200 J). The patient continued with ventricular fibrillation throughout the ACLS algorithm, and the decision was made to attempt DSD. The patient had a second set of pads applied in the anterior-posterior orientation in addition to the conventional right upper chest and left lateral chest with successful conversion of the ventricular fibrillation. The patient was additionally given Lidocaine, 100 mg, due to a wide-complex tachycardia and apparent non-responsiveness to the previously given amiodarone (Figure ). An improvement was noted after the use of Lidocaine, and a Lidocaine drip was started.\nOnce the patient was stabilized, he was taken for computed tomography (CT) imaging to further evaluate for the possibility of a pulmonary embolism. In the CT room, he developed bradycardia and subsequently lost his pulse. CPR was again started, the patient was given atropine, and return of spontaneous circulation (ROSC) was achieved shortly after. The CT scan did not reveal any evidence of aortic dissection or pulmonary embolism and a repeat ECG was performed which showed a persistent wide complex tachycardia with no obvious ST-segment changes. Due to the morphology of the QRS complexes and length of resuscitation time from initial arrest (nearing 90 minutes), tissue plasminogen activator (tPA) was used as a thrombolytic for what was presumed to be a large vessel occlusion myocardial infarction. Hundred milligram of tPA was administered (50 mg as a bolus and 50 mg as a drip given over 60 minutes) with an apparent reperfusion rhythm followed by a "normal" appearing sinus tachycardia. Another ECG was repeated which revealed sinus tachycardia, at a rate of 114, now with ST-segment elevations present in aVR, V1, V2, V3, and V4 with depressions in leads II, III, and aVF (Figure ).\nGiven the patient's persistent elevations despite thrombolytic therapy, interventional cardiology was consulted, and the patient was transferred to a tertiary care facility for cardiac catheterization revealing a thrombotic occlusion in the proximal left anterior descending coronary artery.\nAfter transfer to the tertiary care facility for cardiac catheterization, the patient developed cardiogenic shock. The patient was started on ionotropic medications with no improvement, and he was placed on venous-arterial extracorporeal membrane oxygenation (ECMO) therapy (~20 hours after his cardiac arrest). Before initiation of ECMO, the patient was awake, alert, and following simple commands (although still intubated). Unfortunately, despite ECMO support, his cardiac function did not improve, and the patient was not a candidate for cardiac transplantation. Seventeen days after the patient's initial presentation to the ED, the family decided to withdraw care. The patient was extubated, had ECMO discontinued, and time of death was documented shortly after. |
A 90-year-old woman was referred to the Osaka University Hospital with acute onset vitreous hemorrhage of unknown origin. She reported a sudden onset of blurred vision in her left eye. On examination, her best-corrected visual acuity was light perception in the left eye and 20/16 in the right eye, and her intraocular pressure was 13.0 mm Hg OD and 12.0 mm Hg OS. Fundus examination of the left eye by B-mode ultrasonography revealed dense vitreous hemorrhage without retinal detachment. A bulge on the optic disc was also detected (Fig. ). Additionally, she had a history of hypertension, but her blood pressure was well-controlled with oral medication. Results of preoperative systemic examinations revealed a thoracic aortic aneurysm. A pars plana vitrectomy was performed to treat the dense vitreous hemorrhage in her left eye. After excision of the vitreous hemorrhage, a two-disc-diameter hematoma, which looked encapsulated, appeared on the optic disc and was promptly removed (Fig. ). Because the source of the hemorrhage was arterial and pulsating, the first vitrectomy could not achieve hemostasis (Fig. ). Aside from the optic disc hematoma, other areas of the eye, such as the macula, arcade vessels, and the peripheral retina, showed no lesions. Five days after the first surgery, we performed a second vitrectomy, which revealed subretinal hemorrhage along the superior and inferior arcade vessels and a macular hole (Fig. ). To prevent recurrent bleeding, we trimmed the edges of the hematoma on the optic disc to minimize its size. Because the macular hole, caused by the retinal arteriolar aneurysm rupture, was refractory [, ], we closed it using the indocyanine green-assisted inverted internal limiting membrane flap technique (Fig. ). The macular hole, which was almost closed (Fig. ), reopened 41 days after the second vitrectomy (Fig. , c, d). The patient did not wish to undergo further surgeries. The left eye was actively monitored at subsequent follow-up visits. |
A 36-year-old woman complained of an insidious onset of generalized myoclonus that first became apparent at age 27 years. She had no perinatal problems and her development was normal in childhood and juvenile periods. Adult-onset myoclonus had worsened progressively from right hand to four extremities, tremulous voice and gait disturbance developed after 3 years from disease onset, and she could not continue working as a nurse. She did not complain decreased visual acuity and hearing difficulty. She had no history of febrile convulsions or seizure, infectious disease in the central nervous system, exposure to toxic materials, or intake of herbal drugs.\nHer younger brother aged 40 years old also had progressive generalized myoclonus, which was detected 6 years ago at age 34 years (). He had normal intelligence and did not complain decreased visual acuity. Other family members except younger brother were reportedly healthy.\nA physical examination did not reveal any dysmorphism or evidence of hepatomegaly. The patient was alert and oriented, and her Mini-Mental State Examination score was 30. She did not have gaze palsy, and her vision and hearing were normal; however, her voice was tremulous and generalized positive myoclonus was observed at four extremities and body. Negative myoclonus, dystonia, tremor, and rigidity were not detected, and both motor and sensory functions were intact. Deep tendon reflexes were normal, and Babinski’s sign was not present. There was no evidence of cerebellar dysfunction, and she did not have an ataxic or parkinsonian gait, although she staggered slightly because of myoclonus.\nExamination of the eyes revealed cherry-red spots (), but her electroencephalogram was normal. Levels of serum electolytes, creatinine, and liver enzymes were normal. No white matter lesion or cerebellar atrophy was detected in an MRI of her brain ().\nNeuraminidase, hexosaminidase A, and β-galactosidase activities in the leukocytes and cultured fibroblasts in patient and younger brother were normal. |
A 73-year-old female with a past medical history of hypertension, peripheral arterial disease, asthma, gastroesophageal reflux disease, and diabetes mellitus presented to our otolaryngology clinic with the complaint of a “nodule” behind her left ear. She stated that the mass had been slowly increasing in size over the last 5 years. It was described as nonpainful and she had never experienced any discharge from the area. Her only other complaints were decreased hearing, xerostomia, and hoarseness; no dysphagia or weight loss was reported. Past surgical history was significant for angioplasty and stent placement in her legs. Social history was significant for a 20-pack-year tobacco history. No significant family history was reported. On physical examination pertinent findings consisted of a left posterior auricular mass approximately 2 centimeters (cm) that was fixed and nontender. All cranial nerves, most notably the facial nerve, were intact. No cervical adenopathy was palpated. Nasopharyngeal laryngoscopy demonstrated arytenoid edema consistent with reflux but the true vocal folds were mobile and no other lesions were visualized. Treatment plan at that time consisted of smoking cessation and a computed tomography (CT) scan with contrast of the neck.\nThe CT scan revealed a multilobulated and cystic mass in the left parotid gland with an infiltrating appearance consistent with neoplasm. There was no pathologic cervical lymphadenopathy reported. The differential diagnosis at this time included benign mixed tumor, Warthin's tumor, epidermoid tumor, and adenoid cystic carcinoma. A fine needle aspiration was performed at this time and the results were nondiagnostic. The decision was made to proceed with a left parotidectomy.\nLeft superficial parotidectomy with facial nerve monitoring was performed approximately one month after presentation. A 2 × 2 cm lesion was removed from the tail of the left parotid gland. The gross description was an encapsulated, lobulated mass with a brown-yellowish coloration. The specimen was sent for analysis by the pathology department. The tumor was composed of neoplastic myoepithelial cells arranged in a fascicular nested pattern separated by collagen stroma (). There was an epithelioid pattern of uniform cells with central small nuclei with fine chromatin, inconspicuous nucleoli, and an eosinophilic cytoplasm. Mild degree of nuclear atypia without mitosis was noted (). Immunohistochemical staining of the specimen was positive for calponin (), CK5/6 (), GFAP (), p63 (), S100 (), CK7, vimentin, and SMA. CK 20 was negative. No malignant features were identified and the diagnosis of epithelioid myoepithelioma was made.\nThe patient recovered from the surgery without complications and her facial nerve was functioning well. The patient had no sign of recurrence at 10 months and is currently being seen regularly for routine monitoring. |
A previously healthy 6-year-old boy presented to a pediatric hospital with a 3-week history of torticollis. He had symptoms of an upper respiratory tract infection four weeks prior and had 2 days of documented fever at home during that time. He had been treated with a 7-day course of amoxicillin by the primary care physician for suspected streptococcal pharyngitis. Four days into the course of antibiotics, he woke up from sleep with pain on the left side of his neck. Despite taking ibuprofen and acetaminophen, he presented to the Emergency Department 3 weeks later due to persisting torticollis. Pain was worse with movement. There was no history of head/neck trauma. At the time of presentation, the infectious symptoms had resolved. Some fatigue was noted but he remained generally active, continuing to play hockey. There was no history of rash, peripheral joint pain, or weight loss. Past medical history and family history were unremarkable.\nOn examination, the patient was afebrile with normal blood pressure for age and a maximum heart rate of 110 beats per minute. The patient's head was tilted to the right with chin rotation to the left. No lymphadenopathy or masses were noted on palpation of the neck. There was no tenderness to palpation of bilateral sternocleidomastoid muscles. There was a limited range of motion in all planes of rotation of the neck secondary to pain, particularly in lateral flexion. Bilateral injected conjunctivas were present. The oropharynx was normal with no erythema or mucus membrane changes. Cardiovascular exam revealed normal peripheral pulses, a quiet precordium with normal heart sounds, and no murmur. Respiratory exam was normal. The abdomen was soft with no distension, tenderness, or hepatosplenomegaly. There were no bruits heard on auscultation of major vessel regions. There were no rashes or desquamation of the skin. Neurological exam was normal.\nAt the time of presentation, laboratory investigations revealed an elevated white blood cell count of 17.4 × 109/L with a neutrophil count of 14.1 × 109/L. Hemoglobin was normal for age at 110 g/L. Inflammatory markers were elevated including platelet count of 860 × 109/L and CRP of 38.5 mg/L. Renal function (BUN and creatinine) and liver function (ALP and ALT) were normal for age. Because of the unexplained elevated white blood cell count and evidence of inflammation, a chest X-ray was performed which revealed normal lung fields but an enlarged cardiac silhouette. X-ray of the cervical spine was normal with no atlantoaxial rotary subluxation demonstrated. Ultrasound of the neck revealed mild thickening of the left sternocleidomastoid muscle and no lymphadenopathy. Abdominal ultrasound with Doppler was normal.\nAdditional investigations included a normal throat swab for group A streptococci and a negative anti-streptolysin O antibody titer. High-sensitivity troponin was elevated to 176 ng/L. Creatinine kinase was normal. ANCA was normal. Electrocardiogram showed normal sinus rhythms without evidence of chamber hypertrophy. The patient underwent an echocardiogram to further characterize the enlarged cardiac silhouette identified on the chest X-ray. This revealed massive ectasia and aneurysmal dilatation of the right coronary artery, left main artery, left anterior descending artery, and circumflex arteries, as seen in . Left ventricular function was normal. The aortic arch was normal as were the proximal neck vessels.\nBecause of the dilated coronary aneurysms, the patient was diagnosed with KD. Despite lack of fever, given the evidence of ongoing inflammation and initial presence of bilateral nonsuppurative conjunctivitis, in addition to the coronary artery changes, the patient was treated with high-dose IVIG (2 g/kg) and started on daily low-dose aspirin. Low-molecular-weight heparin was started as antithrombotic therapy and once stabilized, daily atenolol was initiated. Activity was restricted as much as possible.\nInflammatory markers were followed. Platelets revealed a peak of 952 × 109/L and CRP a peak of 54.6 mg/L. After treatment, both platelet and CRP levels normalized.\nThe patient's neck pain and the limited range of movement resolved immediately after treatment, as did the bilateral conjunctivitis. The patient was stable and appeared well at time of discharge. His aspirin, low-molecular-weight heparin, and atenolol were continued. The CT angiogram performed after discharge revealed massively dilated and aneurysmal coronary arteries, as shown in .\nIn follow-up cardiology and rheumatology clinics, he has been doing well with no further neck pain or stiffness. He did not develop desquamation during follow-up, and the repeat echocardiogram one month after discharge was unchanged. He will continue long-term anticoagulation therapy with low-dose heparin with a target level greater than 0.5 IU/ml. He will also continue low dose aspirin and atenolol. His family was advised to have the annual influenza vaccine. |
A 70-year-old female presented with a chief complaint of swelling in relation to the upper front teeth since 3 days. She gave history of pain which was of pricking type radiating to the upper half of the face. Extraoral examination revealed a single diffuse swelling measuring 5 × 4 cm2 involving the upper lip and the surrounding structures (). Skin overlying the swelling appeared normal. On palpation the inspectory findings were confirmed and there was no local rise in temperature and no pulsation; the swelling was firm and tender. There was no numbness or paresthesia in relation to the swelling.\nPatient was from a low socioeconomic background, was malnourished, and had a poor oral hygiene. Intraoral examination revealed diffuse necrosis of soft tissues in labial vestibule in relation to teeth 11, 12, 13, 14, 21, 22, and 23. The area was soft and tender on palpation. The anterior part of the hard palate showed necrosis and the mucosa covering it was completely detached exposing the underlying bone (). A sinus opening was seen present on the mucosal surface of upper lip in relation to tooth 11. Live grayish white maggots were seen crawling through the opening of the sinus. PNS view revealed radiolucency in relation to teeth 11, 12, and 21 suggestive of bone resorption (). The hematological report was normal.\nThe patient was treated by removal of the maggots, debridement, and irrigation. The wound was debrided under local anesthesia and roller gauze impregnated with turpentine oil was inserted into the cavity created as a result of tissue necrosis. 30–40 live maggots were harvested from the affected region (). Copious irrigation with normal saline and povidine iodine was performed. Under antibiotic coverage with oral penicillin, the patient underwent debridement again until the maggots were completely removed. On entomological examination the maggots were found to be of species Chrysomya bezziana (Figures and ). |
A 13 year old African female presented at our service with a 3 week history of progressive painful abdominal distention and loss of vision of the left eye, associated with anorexia and weight loss. She had no fever, headache, vomiting, bloody urine nor weakness of limbs.\nOn physical examination, her vital signs were within normal limits. She weighed 37 kg, and height 147 cm (surface area of 1.2 m2). There was no pallor, no lymph adenopathy and no sign of malnutrition. External evaluation of the left eye revealed complete blindness with a mydriatic pupil that was non- responsive to light. Gastrointestinal examination revealed a circular scarification mark on the abdomen as well as a huge visible abdomino-pelvic mass measuring about 20 cm above the supra-pubic region (Fig. ). The mass was hard with irregular contours, fixed, moderately tender on palpation and extending to the epigastric region. She had no ascites. The liver, spleen and kidneys were non palpable. A non-tender left breast mass was also palpated, while her mouth, teeth and jaws were normal and likewise the rest of the systemic examination.\nA full blood count done revealed lymphocytosis (70%) with normal total white cell count and haemoglobin level of 10.2 mg/dl. She had negative test results for malaria and HIV and her urine analysis was normal. An abdominal ultrasound scan showed a huge hypoechoic abdominopelvic mass, with normal homogenous liver and spleen, while both kidneys had moderate hydronephrosis. Bone marrow aspirate revealed normal trilineage maturation with erythroid hyperplasia and negative for lymphoma or other malignancy. Cerebrospinal fluid (CSF) cytology was negative for malignancy (lymphocytic pleocytosis). Fine needle aspiration of the tumour was done and histopathology analysis of sample showed no malignant cells. A CT scan and MRI were requested but not done due to financial constraints.\nDespite the negative bone marrow aspirate results, a presumptive diagnosis of Burkitt’s lymphoma was made based on the clinical picture and abdominal ultrasound findings. The patient was placed on the induction phase of the Burkitt’s lymphoma chemotherapy protocol as adapted from the Malawi 2003 protocol [].\nAs per this protocol the patient was due 3 pulses of cyclophosphamide and intrathecal methotrexate and hydrocortisone on days 1, 8 and 15 while maintaining good hydration.\nFollowing initiation to treatment, and prior to day 8 (second day of chemotherapy), the breast and abdominal tumours had rapidly regressed. By the end of the induction phase, the left breast mass was no longer palpable though it still appeared bigger than the right whilst the abdomino-pelvic mass had reduced significantly to a small pelvic mass about 6 cm in diameter, non-tender, smooth and firm. The patient however remained blind on the left side. She was then placed on the intensification phase of the chemotherapy protocol. At the end of the chemotherapies, the right breast was now same size with the left, the abdominal mass had completely regressed (Fig. ) but with persistence of the blindness.\nAt 6 months follow-up, the primary disease was in remission but there was still blindness of the left eye. |
A 40-year-old Hispanic man with a past medical history of human immunodeficiency virus (HIV) was brought to the emergency department complaining of right upper extremity (RUE) weakness and numbness for four days with associated bitemporal headache and generalized fatigue. The patient reported first time use of intranasal cocaine and heroin, after which he lost consciousness and woke up approximately four hours later with new onset RUE and headache. His cluster of differentiation 4 (CD-4) count was reported above 500 cells/mm3 and viral load (VL) was undetectable. The patient did not have any known CNS complications in the past.\nOn physical examination, his blood pressure was 151/97 mm Hg and pulse was 82 and regular. He was alert and cooperative. His cranial nerves were intact. His motor exam, however, was abnormal in the RUE with 3/5 arm strength and wrist drop; the strength and tone of the other extremities were normal throughout. Deep tendon reflexes were normal bilaterally, but his gait could not be evaluated. His sensory function decreased to pin sensation at the RUE and normal sensation was noted in the rest of the extremities and face. Laboratory testing was normal except for an elevated creatinine of 6.9 mg/dl, creatine phosphokinase (CPK) of 7855 IU/l, alanine transaminase (ALT) of 139 IU/l, and aspartate transaminase (AST) of 109 IU/l. Urine toxicology was positive for metabolites of cocaine and heroin. Magnetic resonance imaging (MRI) of the brain was done and it revealed two areas of increased T2/FLAIR signal within the medial aspect of both basal ganglia, measuring 16 mm in the right and 12 mm on the left involving each globus pallidus and the genu of the internal capsule, as can be seen in Figures -. His chest radiography was normal, computerized tomography (CT) of the brain, as can be seen in Figure , and cervical spine were normal. His electrocardiogram was normal.\nIn the subsequent days, his kidney function and rhabdomyolysis improved. The patient remained fully awake, alert and oriented, but the weakness of his RUE persisted. The patient decided to leave against medical advice despite full explanation of the risk of leaving.\nThe patient was contacted over the phone and he informed us that he followed up with his primary care physician and reported improvement of the weakness. He received physical therapy and was independent in all activities of daily living and functional mobility. His only limitation was a moderate decrease in fine motor coordination of the RUE. |
Case 1 is a 3-year-old boy with a bleeding in a left frontotemporal arachnoid cyst. He was treated with a craniotomy with fenestration and marsupialization of the arachnoid cyst. Half a year later, a subduroperitoneal drain without valve was implanted because of a persistent headache due to a large subdural hygroma at the location of the former arachnoid cyst (Fig. ). During the following years, the patient developed variable postural headaches attributed to liquor hypotension. Removing the drain led to immediate severe headache, and we decided to implant a new subduroperitoneal shunt with a PS medical medium valve. Subsequent shunt revisions over the years were performed with changing the valve to a Delta II, back to PS medical medium and finally to a Miethke 9/29. The latter valve resulted in an acceptable clinical condition (Fig. ) with symptoms of occasional mild headaches during the following years. At the age of 14, he developed a different kind of headaches, characterized by short attacks of headaches relieved by vomiting. An MRI scan showed an acquired Chiari I malformation due to thickening of the cranial vault with a subsequent decrease of intracranial volume especially within the posterior fossa, without signs of syringomyelia or spinal CSF leakage (Fig. ). We decided to augment the volume of the posterior fossa in combination with decompression of the Chiari without creating a risk of a descending cerebellum. Therefore, we modeled the thickened occipital planum to a normal size and subsequently placed back the thinned bone. During the same procedure, a standardized C0 augmentation, C1 laminectomy, and a dural patch were given. The patient has remained without symptoms now for more than a year. A control MRI showed an adequate decompression (Fig. ). The volume of the posterior fossa was estimated on the T2-weighted scans using Brainlab iPlan 3.0 Cranial. The tentorium, occiput, McRae line, and clivus served as boundaries of the posterior fossa. There was an increase in the size of the posterior fossa (188 cm3 preoperatively, 205 cm3 postoperatively). |
A transsexual 35-year-old Caucasian male without known relevant medical history of cardiac disease presented to the emergency room with a sudden onset of typical anginal chest pain. Physical examination revealed a hemodynamically stable patient with resting pulse of 70/min and blood pressure of 110/70 mmHg, with no signs of acute cardiopulmonary decompensation. Cardiac examination was uneventful with normal heart sounds without pathological murmurs. Respiratory, abdomen, and neurological examinations remained normal. Active smoking accumulating to 20 pack years and a history of daily alcohol consumption were significant risk factors. Drug history revealed a hormonal therapy with intramuscular testosterone undecanoate for gender conversion over the past 6 years, with an initial dosage of 1000 mg per month over a period of 1 year from 2011 to 2012 and later at a dose of 1000 mg every 3 months to date. An electrocardiogram showed sinus rhythm with ST-Segment elevation in the inferior leads ().\nAn emergency coronary angiography revealed normal coronary arteries with an occlusion of the distal right coronary artery (). The appearance of the remaining coronary arteries was smooth, not indicative of an advanced atherosclerotic coronary artery disease. We suspected a thromboembolic origin as the primary cause of the myocardial infarction by the appearance of the coronaries. Recanalization of the vessel was achieved by catheter-driven direct thrombectomy using an aspiration catheter. This was followed by intracardiac lysis with 20 mg tissue plasminogen activator, Alteplase, and subsequently the intravenous glycoprotein IIb/IIIa inhibitor Tirofiban. A short episode of reperfusion-related ventricular fibrillation and subsequent third-degree atrioventricular block (av-block) were overcome by defibrillation and intravenous administration of 2 mg Atropine, respectively. Finally, a successful recanalization was achieved with TIMI-3 flow () with immediate resolution of the patient's symptoms. A stent implantation was not necessary. Cardiac biomarkers were subsequently elevated with Troponin I up to a maximum of 46.63 ng/ml (Reference range: 0.00–0.06 ng/ml). Echocardiography revealed a good systolic left ventricular function with postinfarct inferior wall hypokinesia. The patient remained haemodynamically stable without complications during the hospital stay.\nSubsequently, investigations were conducted with the aim to identify an underlying cause for the suspected embolic myocardial infarction. The transthoracic echocardiography did not reveal intracardiac thrombus or valve vegetations as evidence for infective endocarditis. A deep venous thrombosis could also not be detected by ultrasonography, and the D-Dimer levels were negative.\nThe initially performed transoesophageal echocardiography under sedation (Midazolam) raised the suspicion of a possible small persisting foramen ovale (PFO). We performed a second contrast-enhanced TOE without sedation. No crossover of microbubbles over to the arterial circulation were observed even under Valsalva manoeuvre. Thus, a haemodynamically relevant right to left shunt could be ruled out.\nNo evidence of paroxysmal atrial fibrillation was reported in a 72 h Holter-ECG Monitoring. Cardiac embolic sources were thus deemed unlikely.\nOther manifestations of atherosclerotic vessel diseases could not be seen in the colour-coded Doppler Ultrasound scans of the extracranial arteries or pelvic/lower limb arteries.\nLaboratory tests for Antiphospholipid Syndrome (APS), Factor-V Leiden, Protein C and Protein S deficiency, Antithrombin III deficiency, and Hyperhomocysteinemia as possible causes for a hypercoagulable state were also negative.\nIn conclusion, the work-up for the definitive cause of the infarction remained inconclusive. The drug history of the patient with the use of high-dose hormonal therapy with testosterone for gender conversion was one of the significant factors in the patient's history.\nThis background of high-dose testosterone therapy for the purpose of gender conversion and its procoagulant associations led us believe this to be the factor leading to the coronary embolism.\nA follow-up of the patient was performed at 3 months and 1 year. The echocardiography and ECG showed no signs of long-term detrimental effects following the myocardial infarction with resolution of the initial wall motion disturbances. |
Lower back pain and lower extremity numbness for 10 h.
A 70-year-old man with a history of lumbar spinal stenosis, hypertension, and gout presented to our emergency department because of severe lower back pain and lower extremity numbness. He has been receiving intermittent physical therapy and medical treatment for lower back pain and lower extremity numbness. On the physical examination, he had tenderness in the lower back, and no obvious decrease in skin sensation around the anus. He had 4/5 of strength in both legs and decreased sensation below the knees. Lasègue signs were negative. The bilateral knee and Achilles tendon reflexes were normal. He had joint deformity and gout nodules between the fingers and toes. Babinski’s sign and other pathological reflex signs were negative. Laboratory testing was largely unremarkable, aside from a uric acid level of 462 μmol/L. X-rays showed degenerative changes of the lumbar spine and the L4 vertebral body had slipped forward slightly. He was diagnosed with spinal stenosis by an orthopedic surgeon and admitted to the Department of Spinal Surgery for further workup.
After admission, he was treated with steroid injections, analgesia, and nervous system nutrients for symptom relief. MRI demonstrated lumbar spinal stenosis (L4/5). Based on these findings, he was diagnosed with lumbar spinal stenosis. After conservative treatment failed, he underwent transforaminal lumbar interbody fusion. Postoperatively, his paraesthesias and muscle weakness did not improve markedly and he reported numbness and weakness in both upper extremities. On postoperative day 2, he had 2/5 of strength in his upper extremities and 1/5 of strength in his lower extremities. Deep tendon reflexes (for example triceps reflex, biceps reflex, and knee and Achilles tendon reflexes) disappeared. He also reported dysphagia and numbness in the upper extremities. We requested a neurology consultation. Careful review of the patient’s history, as provided by his family and community doctor, revealed that he |
A 48 year-old male veteran sustained an injury to his right leg in 2006. While undergoing aqua-therapy, he injured his right great toe, which subsequently became infected. Following drainage of an abscess and removal of the great toe’s nail, the patient developed the following CRPS symptoms in his right lower extremity: swelling, allodynia (pain to normal touch), color change, temperature change, and some weakness. By 2007, the patient developed moderate CRPS symptoms in his upper extremities. In 2008, he developed blisters and skin ulceration in his right lower extremity (Fig. ). At this time, the patient was being treated with opioids, pregabalin, and duloxetine.\nBy 2009, the patient’s pain had become severe enough that he could not ambulate without assistance. He developed muscle spasms in the right upper extremity. In 2010, he underwent a cardiac bypass surgery for coronary artery disease. His CRPS symptoms became widespread after this surgery, spreading to his upper chest, upper arms, and forearms. In 2011, the patient developed significant dystonic spasms to both upper extremities, resulting in hyperextension of his fingers. From 2008 to 2012, the patient underwent multiple treatments with anticonvulsants, antidepressants, physical therapy, psychotherapy, topical and systemic analgesics, including but not limited to opioids. The patient also currently has diabetes mellitus type II, hypertension, hyperlipidemia, as well as coronary artery disease.\nIn August of 2011, the patient began low-dose intravenous ketamine infusions. He reported a good initial response, but the relief was not sustained. He continued to have ketamine booster infusions at intervals of 4 to 6 weeks. Although the patient engaged in aggressive physical therapy during this time, his maximum interval of relief from pain following a given ketamine infusion decreased to an interval of 3 weeks. In January 2012, his use of the opioid oxycodone was changed to tapentadol. This narcotic was removed for 1 week prior to starting low-dose naltrexone, which was started and maintained at 4.5 mg per day (1 dose at night). Additional medications included: metformin, tramadol, valsartan, cloazepate, simvastatin, fish oil, and vitamin C.\nImmediately before LDN treatment, the patient had patchy areas of allodynia to the medial and dorsal aspect of his right foot, extensive areas of dysesthesia in his right lower extremity below the knee and heel of his foot, as well as bilaterally dysesthesia in the upper extremities. There were significant color and temperature changes in the right foot compared to the left foot, as well as pitting edema in the right foot. A triple-phase bone scan revealed significant reuptake in the right foot, characteristic of CRPS.\nBy March of 2012, the patient’s requirements for the lower dose intravenous ketamine infusions were not as frequent (6 week intervals, pain spikes not as high). The patient recovered from CRPS flares more quickly, felt more energetic, and tolerated pain better. He became physically more active, and his sleep improved significantly. Within 2 months after starting LDN, the patient’s dystonic spasms discontinued, although he still had moderate pain in both upper extremities. The patient was able to walk without a cane (Fig. ), which he had used continuously since 2006. His pain was an average of 8 to 10 on the Numeric Rating Scale (NRS) before starting LDN. It dropped down to an average of 5 to 6 on the NRS after starting LDN. After LDN therapy, the patient’s pain symptoms have reduced in severity, but not in their distribution. His current mood state is good. No side effects of LDN were noted. |
A 45-year-old woman underwent distal gastrectomy for AGC at our hospital in July 2006 (pT2bN0M0, stage IB, according to the UICC classification system\n[]). A histological examination of the primary gastric cancer lesion revealed a signet ring cell carcinoma that had infiltrated the subserosal layer. There was no lymph node metastasis in any of the 44 retrieved nodes, and the proximal resected margin was 2.0 cm. The patient remained well until 1 year after surgery, when recurrent remnant gastric cancer invading the distal pancreas was detected during a follow-up evaluation. After no other metastasis was identified, a complete, total gastrectomy with distal pancreaticosplenectomy was performed in July 2007. Postoperative adjuvant combination chemotherapy included six cycles of 5-FU and cisplatin. She remained well for 2 years following her second operation until she experienced mild swallowing difficulty. A CT scan and barium swallowing series showed a recurrent mass around the jejunojejunostomy site that compressed the Roux limb and resulted in a luminal obstruction. A third operation was performed in July 2009. En bloc resection of the recurrent tumor, including the Roux limb, was carried out, and a repeat esophagojejunostomy with jejunojejunostomy was performed. She refused postoperative adjuvant chemotherapy and remained well for 3 years. However, a 3-cm left adrenal mass found on a CT scan showed intense FDG uptake on PET-CT scans (Figure \n). In this patient, sufficient clinical information was available for surgical resection and other alternative treatments, so surgical resection was planned with the patient’s informed consent. A fourth operation was performed in May 2012, including a left nephroadrenalelctomy, transverse colectomy and resection of the Roux limb because of direct invasion of the adrenal tumor into the renal capsule and adjacent jejunal mesentery and mesocolon (Figure \nA). A histopathological examination revealed a 6 × 3–cm adrenal tumor with metastatic signet ring cell carcinoma, which is the same histologic finding as that of primary and recurrent lesions (Figure \nB). The regional lymph nodes, including jejunal mesentery (0 of 13), transverse mesocolon (0 of 1) and renal parenchyma were free from metastasis or direct tumor invasion. The patient’s postoperative course was uneventful, and she was discharged on postoperative day 10 without any complications. She received second-line chemotherapy with seven cycles of 100 mg/day S-1. During the 12-month follow-up period, no definite evidence of tumor recurrence was found. |
A 20-year old gentleman was injured in a high velocity road traffic accident while riding a motorcycle. A sheet of metal went through his right knee. He was brought to the emergency department nine hours following the trauma. He had an 8 x 6cm grossly contaminated transverse laceration over the anterior aspect of his knee exposing a comminuted fractured patella and radiographs further revealed a bicondylar Hoffa (AO 33-B3) fracture ( and ). He had no distal neurovascular deficits.\nHe underwent emergency debridement and removal of the loose patella fragments. The laceration was extended and the distal femur was visualised through the patella fracture. The bicondylar fracture was found to have a large medial and a smaller lateral condyle fragments. The quadriceps, patella tendon, collateral, cruciate ligaments and menisci were found to be intact. Minimal internal fixation was done in the initial stage in view of the contaminated nature of the injury. The bicondylar Hoffa fracture was stabilised with two posteroanterior lag screws (Synthes 6.5mm partially threaded cancellous screws), one for each condyle along with a cerclage 16-gauge wire for the patella ( and ). At elective relook debridement 48 hours later, two more lag screws (Synthes 6.5mm partially threaded cancellous screws with washers) were added for the larger medial condyle fragment. The fractures were found to be stable following fixation.\nThe wound healed uneventfully and he was immobilised in an above knee cast for six weeks in view of the comminuted patella fracture. He was subsequently started on active knee range of movement (ROM) exercises and graduated weight bearing. At three months, he underwent manipulation under anesthesia as he had some residual knee stiffness with flexion up to 90°. Following manipulation, his ROM improved and he was advised regular follow-up. Three years later, he presented with anterior knee pain and implant prominence over the knee. It was then decided on implant exit for the patella. However, in view of the extensive intra-articular extent of the fracture, unavoidable consequences like knee stiffness and early arthritis in the future, the distal femur hardware was removed in the same sitting. Radiographs following implant exit showed a well-united fracture without arthritic changes in the knee ( and ).\nAs this was an open intra-articular fracture, he was advised clinical follow-up of atleast once in three years following union of the fracture. He was very compliant and was on regular visits. At latest follow-up (15 years since the trauma), he was asymptomatic, although there were Kelgren-Lawrence grade 2 osteoarthritic changes of the right knee visible on radiographs ( and ). The gait was normal and both knees were in alignment (). He had no pain or extensor lag and his knee was clinically stable with a flexion of 130° which was 5° short of the normal side ( and ). He could squat, sit cross-legged ( and ) and had resumed his normal level of activity which included playing badminton at the club level. His Oxford Knee Score was 47 and his knee and functional score components of the Knee Society Score were 85 and 100 respectively. |
A 29-year-old male farmer from Sigmo District, Jimma Zone, Ethiopia presented to the Eye Clinic with a complaint of right side severe periocular swelling of 10 days duration. The swelling started as a small reddish raised lesion on his right upper eye lid which was later followed by vesicle formation and dark ulcerative wound with progressive swelling involving the upper lid, lower lid and cheek on the same side. He slaughtered an ill ox in his village a week before the start of his illness and he also ate the uncooked meat. There was no history of trauma or surgery. He was given oral antibiotics from the local pharmacy which he took for a week prior to presentation to the Eye Clinic.\nOn examination, the patient had diffuse non-pitting edema of both the upper lid and lower lid with ulcerated central dark tissue. It was not tender to touch and there was no abscess (Figure ). The patient was unable to open the affected lids and it was thus difficult to record visual acuity at presentation but the cornea looked normal. Vital signs were normal and his body temperature was 36.1°C; and the white blood cell (WBC) count was 7, 800 × 103.\nWith the clinical diagnosis of severe preseptal cellulitis/periocular cutaneous anthrax/, the patient was admitted to the Eye Ward and started on intravenous ceftriaxone 1gm BID and cloxacilline 500 mg IV QID for 2 weeks and then he was put on oral doxycycline 100 mg BID for 6 weeks. After 3 weeks the lesion resolved and visual acuity was 6/6. There was persistent cicatricial ectropion of the right eyelids (Figure ) for which full thickness skin graft was done at 2 months of follow up. On further follow up at one year, there was lid disfigurement on the nasal part of both lids which warranted surgical correction. |
A 33-year-old female presented to the clinic complaining of a polyp on top of a red plaque in the left side mons pubis for 1 year. It was a kind of round flaky red bulge lesion in the skin of mons pubis. On top of this, it is shown sarcomatoid hyperplasia covering with the yellow-green crust, and the surrounding skin had no obvious abnormality and elcosis. The plaque was slightly bulgy without pressing pain, covered with a thick yellow-white crust (Fig. ). The plaque was 2.2 × 1.8 × 0.5 cm, and the polyp was 2.5 × 2 × 1.5 cm. The width of the pedicle was 1.4 cm. There were no other systemic abnormalities or any palpable lymphadenopathy, ultrasound B was applied to evaluate the superficial inguinal lymph nodes. She had no significant history of medicine, surgery, irritation, and trauma. Before the lesion appearance, the patient had no discomfort, thus she did not pay attention to the skin lesion, and had not used other external medicines or ask for help from professionals. Until nearly a month, she found that the lesion was prone to bleed after friction, thus she came to our hospital for treatment. The lesion was removed surgically and the histopathologic examination was performed. The possibility of skin tumor was considered through dermoscopy, then the histopathological examination was performed to make the confirmation. Skin biopsy was made on December 31, 2019, and further immunohistochemical reports have been done on January 17, 2020. Detailed information was shown below.\nThe dermoscopic images showed the dark red background, covering a thick yellow-and-white crust, with spot-shaped and polymorphic vascular structures which focally distributed. In some areas, white homogeneous structures could be seen, as well as dark red clumps. No typical pigment structure was seen (Fig. a, b). The skin lesions were considered as the diagnosis of skin tumors using dermoscopy.\nThe excisional biopsy was carried out, and the specimen tissue was fixed in formalin, and embedded in paraffin. The histological examination showed: the skin lesion at the left side of mons pubis was spindle-shaped, the lesion size was 2.2 × 1.8 × 0.5 cm. There were gray-brown and mushroom-shaped protrusions on the epidermis, and the size of the protrusions was 2.5 × 2 × 1.5 cm, the pedicle width was 1.4 cm, and the transaction of the protrusions was grayish-white and grayish-red. Besides, Breslow thickness was about 9.5mm, and Clark level was IV. No tumor embolus was detected in the vessels. The tumor involvement had been found in the incisal edge of long-axis two sides and base of the tumor sample.\nMicroscopically, the polyps were lined by melanocytes, with pale cytoplasm (Fig. a–d). It was represented as atypical cells and heteromorphic nuclei, with different cell sizes and abnormal mitosis of 5–7 counts /10HPF. Each slide was reviewed by 3 different pathologists, and the diagnosis was made as the melanocyte tumor.\nThe immunohistochemistry (IHC) studies revealed that the expressions of S-100, HMB-45, Melan A and Cyclin D1 were stained positive, while CD-117 was focally immunoreactive and CD31, P16, PCK and LCA were stained negative. The final histopathological diagnosis was made: melanocyte nevus malignancy, nodular malignant melanoma. This patient underwent a complete local excision and she recovered well recently.\nThe patient was required to re-examined regularly after surgery, and recent follow-ups showed that she recovered well and there was no sign of recurrence till now. |
A 32-year-old Caucasian woman was diagnosed with breast cancer and treated with radiation and mastectomy with immediate flap reconstruction and implant 1 year before the presentation of this case report. One month following the mastectomy, a pericardial effusion was identified during an out-patient thoracentesis for radiation-induced pleural effusions. The patient was admitted to a hospital where she was treated with pericardial window drainage of the effusion. She was discharged home on intravenous antibiotics (vancomycin, fluconazole) and analgesia. At home, she continued to use unspecified naturopathic therapies that she had been taking since the mastectomy and of which further details are unknown. Ten days post-discharge, the patient suffered a cardiac arrest at home and was admitted to hospital where mechanical ventilation was initiated and bilateral chest drains inserted. Eleven days later, a tracheostomy was performed. Two tracheal aspirate samples taken on ventilation days 24 and 26 grew C. indologenes. The isolate was susceptible to co-trimoxazole, ciprofloxacin, and levofloxacin. The patient received a diagnosis of ventilator-associated pneumonia and was treated with levofloxacin. Her white blood cell count, which had remained within normal limits until ventilation day 24, peaked at 19 × 109/L on ventilation day 26. The patient died from cardio-respiratory failure and complications of stage IV metastatic breast cancer 40 days after admission.\nOnly five cases of non-ophthalmic and one case of ophthalmic C. indologenes have previously been reported in the USA. In 2001, a 77-year-old man with a history of curettage for squamous cell carcinoma of the leg developed cellulitis of the leg. Wound cultures grew C. indologenes that was susceptible to trimethoprim-sulfamethoxazole and levofloxacin. The patient recovered following treatment with oral levofloxacin [].\nIn 2004, a case occurred in a man aged 54 years with a metastatic squamous cell carcinoma of the nasal tube. After radiation and chemotherapy, he was fed through a gastrostomy. On hospital day 46, he experienced a fever but had a normal white blood cell count. Blood cultures obtained through a peripheral venous catheter were positive for C. indologenes, and the patient was successfully treated with piperacillin-tazobactam and removal of the catheter [].\nA case reported in 2006 was in a Caucasian woman aged 57 years with metastatic breast cancer. An indwelling catheter through which she received chemotherapy had been colonized by C. indologenes, resulting in pea-sized nodules and yellow discoloration of the skin surrounding the catheter. This patient recovered after treatment with ciprofloxacin [].\nIn 2007, the first case of C. indologenes central nervous system infection was reported in a 13-year-old boy with congenital hydrocephalus and a lumboperitoneal shunt. The patient was successfully treated with combination treatment with trimethoprim-sulfamethoxazole and rifampin [].\nThe first case of subcutaneous port-related C. indologenes infection in a liver transplant recipient was reported in 2013 []. The patient, a 26-year-old woman had received a liver transplant 6 years prior to the infection and was on an established immunosuppressive regime. Blood cultures drawn from the port grew C. indologenes; susceptibility testing revealed resistance to piperacillin-tazobactam, other broad-spectrum β-lactams and aminoglycosides. The patient was treated with intravenous levofloxacin and oral trimethoprim-sulfamethoxazole. However, device removal was also necessary. |
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