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A 70-year-old male presented at the Asago-Yanase Medical Center, (Asago, Japan) due to upper abdominal discomfort and dehydration lasting for three months. Contrast computed tomography scans revealed severe dilatation of the stomach, duodenum and upper section of the jejunum, and also revealed a tumor in the jejunum (). The tumor had induced wall thickening around almost the entire circumference of the jejunum at about 50 mm towards the anus from the ligament of treitz. Serologically, the carcinoembryonic antigen and carbohydrate antigen 19-9 titers were found to be within the normal limits.\nThe tumor was resected and a pathological examination of the tumor was performed. Macroscopically, the tumor was 60×52 mm and was classified as Borrmann type 2 (). Histologically, the tumor contained areas of adenoma and adenocarcinoma tissue ( and ). The adenocarcinoma was found to be in the center of the tumor, while the adenoma was at the edge (). In the adenocarcinoma area, the carcinoma showed various differentiating subtypes of adenocarcinoma, including mucinous adenocarcinoma, well-differentiated tubular adenocarcinoma, moderately differentiated tubular adenocarcinoma and poorly differentiated adenocarcinoma. Furthermore, the tumor had invaded the subserosa and had metastasized into the regional lymph nodes.\nThe tumor was immunohistologically analyzed for the expression of P53 and Ki-67. As shown in , in the normal mucosa of the jejunum, there were no P53-expressing epithelial cells and the Ki-67-expressing epithelial cells primarily formed a regular pattern at the base of the crypts. In the adenoma tissue, there was a greater number of Ki-67-expressing epithelial cells compared with the normal mucosa, and the cells were distributed in an irregular pattern. Certain adenoma cells expressed a low level of P53. In the adenocarcinoma tissue, there was a greater number of Ki-67-expressing epithelial cells compared with the adenoma tissue, and the adenocarcinoma tissue strongly expressed P53. Furthermore, in the adenocarcinoma, there were a greater number of Ki-67- and P53-expressing cells in the tubular adenocarcinoma area than in the mucinous adenocarcinoma area. |
The patient was a 58-year-old woman with no history of psychiatric disorder before the age of 58. When she was younger (approximately 10 years old), despite tending to exhibit inattentiveness and forgetfulness, she obtained good grades in school, and these tendencies did not interfere with her daily life. Although her family noticed certain behaviors—such as inattentiveness and forgetfulness, which was manifested as hitting or misplacing things—they assumed that these tendencies were inherited, since other family members who exhibited similar issues were not diagnosed with psychiatric disease and did not require medical treatment. After graduating from high school, she worked at a bank. Although she was aware of a tendency to forget to take notes or misplace her documents at work, she never made any errors that caused serious repercussions and did not experience problems with her colleagues. After she retired from the bank at 52 years of age, she started her own business.\nAfter starting her own business, she was much busier than she had been when she was simply an employee. Within a year, her attentional difficulties and memory issues gradually progressed, and at approximately 57 years of age, she became so inattentive and forgetful that it interfered with her work and family life at home. For example, she forgot meetings with important clients, transferred money to the wrong bank account, and these failures resulted in poor management of her company. At home, she experienced increasing difficulties with remembering prior commitments with her family and misplacing items, which her family members noticed. With the encouragement of her family and employees, who worried that she was suffering from dementia, she visited our memory clinic at 58 years of age.\nUpon visiting the clinic, she reported that she perceived her increased forgetfulness was problematic since it was bothering the people around her; therefore, she feared having dementia and prepared herself to retire from her own company. She did not report experiencing reductions in her levels of desires or interests, and no depressive symptoms were noted during the medical interview. No neurological abnormalities, such as parkinsonism or pathological reflexes, were identified. Neuropsychological testing, such as Mini-Mental State Examination, Alzheimer’s Disease Assessment Scale-Cognitive subscale and Rivermead Behavioural Memory Test, indicated that her memory was intact. Laboratory tests, including blood counts (e.g., full blood count, electrolytes, urea and creatinine, vitamins B1 and B12, and folic acid), blood biochemistry, and electroencephalography, did not reveal any abnormalities (Table ). Magnetic resonance imaging (MRI) of the brain did not reveal any clear cerebral parenchymal atrophy in the hippocampus or other regions, but single-photon emission computed tomography (SPECT) indicated mildly decreased blood flow in the posterior cingulate cortex. Pittsburgh compound B positron emission tomography (PiB-PET) did not show amyloid deposition in the cerebral cortex, and the phosphorylated tau/amyloid beta-42 ratio in the cerebrospinal fluid (CSF) was within the normal range (Fig. ).\nGiven that her condition at work and at home fulfilled eight of the DSM-5 diagnostic criteria for inattentiveness and three for hyperactivity/impulsivity, we diagnosed her with ADHD, and treatment with 18 mg of methylphenidate was initiated and continued this dose. Within a few days after starting the methylphenidate administration, pronounced improvements in her symptoms were observed; for example, she stopped losing her things, was able to concentrate for long durations, and could complete more tasks than she could prior to treatment, which surprised her family and subordinates. Thus, she no longer met the DSM-5 diagnostic criteria for ADHD (Table ). Although she thought she would have to retire from her business, she instead was able to return to work without issue. We conducted a follow up for 6 months with administration of 18 mg methylphenidate; during this period, her initial symptoms did not reappear. |
A 50-year-old man presented to the emergency department (ED) complaining of progressively worsening swelling over the angle of his left jaw associated with difficulty speaking, described as hoarseness and pain with phonation, as well as difficulty swallowing. It began spontaneously one day previously, right after he felt a snap or pop. There were no triggering incidents, although he was unclear if he was talking or swallowing at the time. It was associated with a non-radiating, moderate intensity pain, just below the angle of the mandible on his left side that increased in severity when he moved his jaw to speak, turn, or swallow. Over the course of 12 hours he developed worsening anterior cervical neck swelling and progressive odynophagia, dysphagia, and hoarseness. His medical history was significant for hypertension and left-sided Bell’s palsy since 2014, which was associated with ipsilateral tinnitus.\nOn examination, he was hypertensive with a blood pressure of 187/105 millimeters of mercury, heart rate 91 beats per minute, temperature 98.4° Fahrenheit, and oxygen saturation 100% on room air. The patient’s physical exam was unremarkable with the exception of palpable and visible swelling to the soft tissues below the left side of the mandible and clear discomfort on swallowing. Concern for a vascular abnormality such as aneurysm or thrombosis prompted immediate CT of the neck with IV contrast. This imaging revealed a fracture though an elongated, calcified left styloid process () with hematoma formation causing mass effect on the left lateral hypopharyngeal wall ().\nThe patient was preliminarily diagnosed with a spontaneous fracture of an elongated calcified styloid ligament with hematoma formation and was admitted to observation with otolaryngology (ENT) consultation for airway monitoring and the potential for surgical intervention if worsening airway impingement. He did well with no further progression of symptoms and was discharged after 24 hours. At follow-up with ENT services one week post-ED visit, swelling was markedly reduced and his pain had fully resolved. The spontaneous fracture was thought to be corrective and no further surgical intervention was required., He has had no further symptoms one year from his injury. |
An 81-year-old woman was found to have an abnormal shadow in the right lower lung field on chest radiography for a routine health check-up (Figure ). Her past medical history was unremarkable. Her smoking history was 1 pack/day × 50 years, and she had quit smoking at age 70. Chest computed tomography (CT) showed a tumor with an irregular border centered in the hilar region extending from segment 6 (S6) to the middle lobe of the right lung (Figure ). Based on stenotic and occlusive findings of the pulmonary artery on CT scan, the tumor was considered to have directly invaded the pulmonary trunk (Figure ). Although positron emission tomography showed fluorodeoxyglucose uptake with a maximum standard uptake value of 4.7 in the tumor region (Figure ), there was no fluorodeoxyglucose uptake in mediastinal lymph nodes or other organs. To closely examine the extent of proximal intravascular tumor invasion, angiography was performed, which revealed complete occlusion of the pulmonary trunk at the periphery proximal to the bifurcation of the posterior ascending branch, and a filling defect at the root of this branch (Figure ). Although bronchoscopy showed no mass lesion in the visible range, adenocarcinoma was diagnosed by bronchoscopic biopsy of the tumor. The level of carcinoembryonic antigen was elevated, at 5.7 ng/mL (institutional cutoff value, 5.0 ng/ml); however, no abnormalities were detected in other blood chemistry or tumor marker levels. The patient was otherwise healthy and asymptomatic. Her vital capacity was 1.92 liters, and her forced expiratory volume in 1 second was 1.40 liters. Because right pneumonectomy would be difficult given her pulmonary function and age, preservation of the right upper lobe was chosen as the surgical strategy.\nIntraoperatively, the tumor was found to be located on the interlobar surface of S6-middle lobe, tending to invade proximally and distally along the pulmonary trunk. Massive induration was felt inside the pulmonary artery at the cancer invasion site (Figure ). Cancer invasion was also observed at the root of the posterior ascending branch, and induration was felt in the lumen of this branch. Thus, it was ligated and divided on the peripheral side (Figure ). The pulmonary artery was clamped with vascular forceps at a site proximally peripheral to the bifurcation of the anterior trunk and without internal palpable induration; then, the artery was divided and sutured. As a result of these procedures on blood vessels, we were able to preserve the right upper lobe and perform middle and lower lobectomy (Figure ).\nGross examination of the specimen disclosed that the tumor was mainly centered in the hilar region at S6, measured 40 mm, and was partially grayish inside (Figure ). The tumor showed continuous invasion into the pulmonary artery, and the pulmonary trunk was occluded with a massive thrombus. Histopathologically, cuboidal tumor cells showed acinar, bronchioloalveolar and solid pattern with central fibrotic scar, leading to the diagnosis of adenocarcinoma with mixed subtypes (Figure ). Invasion of adenocarcinoma mainly composed of acinar pattern was observed in the pulmonary artery wall (Figure ), and the endothelium had also been replaced by adenocarcinoma cells in the same region (Figure ). Although a large thrombus was detected at the site of vessel invasion by adenocarcinoma in the pulmonary artery, there were no malignant findings in the thrombus itself (Figure ). No cancer invasion was observed at the surgical margins of either the posterior ascending branch or the pulmonary trunk (Figure ). There was no lymph node metastasis. The cancer was pathologically diagnosed as pT4N0M0, stage IIIA.\nThe patient's postoperative course was uneventful, and she has remained in good health since discharge. In consideration of her advanced age, no adjuvant chemotherapy was performed. For the 14 months to date, since surgery, the patient has remained free of lung cancer recurrence, and will continue to be followed-up on an outpatient basis. |
A 40-year-old male presented with complaints of decreased vision in the right eye of 4 months duration. There was pain in the right eye at onset and the visual loss progressed to complete loss of vision within 6 weeks of onset of symptoms. There was no history of seizures or focal limb weakness. There were no systemic features like fever, loss of appetite, or weight. Previous treatment history for the visual loss included a short course of oral steroids; no improvement was noted with the same.\nHe presented to us nearly 4 months after onset of visual symptoms and nearly 10 weeks after manifesting complete visual loss. On examination, there was no light perception in the right eye. Relative afferent pupillary defect and optic disc pallor were noted. Magnetic resonance imaging (MRI) brain and orbit at this time (16 weeks after onset of symptoms) revealed a homogenously enhancing, diffusely thickened optic nerve [] with nodular enhancing lesions in the right parietal and frontal lobes. [Figures and ]. Human immunodeficiency virus enzyme-linked immunosorbent assay (HIV ELISA) was negative. Cerebrospinal fluid (CSF) analysis including cultures was noncontributory. There was no significant lymphadenopathy. Chest X-ray and abdomen ultrasonography were noncontributary. As there was still significant enhancement of the optic nerve, possibility of an infiltrative optic neuropathy due to an infectious (tuberculosis, cysticercosis) or inflammatory condition (sarcoidosis) was considered most likely. Infiltrations from lymphomatosis, gliomatosis, and other compressive optic neuropathies were considered less likely especially considering the brain lesions. At this stage, he was asymptomatic for the brain lesions and had already completely lost his vision in the right eye. Initiation of empiric antituberculous therapy was strongly contemplated. However, considering the risk of missing other diagnosis in which different treatment will be required, it was decided to go ahead with a biopsy for definitive diagnosis. The options included an optic nerve biopsy versus brain biopsy.\nWhile awaiting the biopsy in the ward, mild impairment of dexterity of the left hand was noted. Antituberculous therapy (ATT) (isoniazid, rifampicin, pyrazinamide, and ethambutol) with dexamethasone was started after a repeat imaging. A repeat MRI brain revealed conglomerate ring enhancing lesions in right frontal and parietal lobes with perilesional edema, mass effect, and midline shift. [] There was also a 30 × 21 mm ring enhancing lesion in the right optic nerve [] with associated thickening. Biopsy from the right parietal lesion revealed necrotizing granulomatous inflammation with numerous acid fast bacilli. Pansensitive mycobacterium tuberculosis was grown on culture. ATT with steroids was continued. Decompression of optic nerve lesion was not done as he already had complete loss of vision with optic atrophy. The mild weakness of the left hand improved completely. Repeat imaging at 2 months follow up revealed subtle increase in the size of the right optic nerve necrotic rim enhancing lesion []. There was remarkable increase in the size, number, and perilesional edema of the brain lesions as well consistent with paradoxical worsening. [] The dosage of steroids was hiked up and ATT was continued. Repeat MRI at 18 months showed significant resolution of the intracranial lesions. [] The optic nerve lesion persisted, though there was a decrease in the size and degree of enhancement []. Hence, ATT was continued. |
A 33-year-old woman, gravida 3, para 3, non-obese and with no chronic diseases, underwent TLmRH as curative treatment for clinical stage IA1 cervical cancer. Two months later, she presented to our department with abdominal pain and genital bleeding after her first sexual intercourse after surgery from the previous day.\nWe sutured the vaginal cuff with absorbable sutures during initial surgery. We performed colpotomy with ultrasonic device and monopolar device in both cases.\nBoth patients were immediately diagnosed with VCD (Figure ). The prolapsed organ was found to be the intestine and it remained within the vagina without evisceration out of the vagina. The color of the intestine was normal, indicating that there was no ischemia present. After washing of the prolapsed intestine, we pushed back the prolapsed intestine, with sterilized gauze to prevent herniation outside of the vagina until operation. Vaginal approach repair (repair from the vaginal cavity), open approach repair, or laparoscopic approach repair were treatment choices. Suturing from the vagina could shorten vaginal length. To prevent recurrence, we thought it would be better to suture the peritoneum. We already resected the vagina about 2 cm in the initial surgery in both cases; thus, we would like to avoid further shortening. We thought that the open approach should be avoided considering its invasiveness if we could safely avoid this complication laparoscopically. Thus, we initially employed total laparoscopic repair.\nThe ureter was separated from the paravaginal tissue during initial cancer surgery, losing its normal anatomical position. This may cause ureteral damage during repair (Figure ). Thus, in order to avoid ureteral injury and to create a tight suture, the vaginal wall was separated by 1.0 cm, to the extent that concrete vaginal cuff suture could be made (Figure ), and suturing was complete (Figure ). The peritoneum was sutured to prevent recurrence, hematoma, and infection. The postoperative course was good. The cuff remained intact at 1-, 2-, 3- and 6-month examination. We suggested that sexual intercourse is safe 6 months after surgery.\nInterviews conducted on these cases after the 6-month examination revealed that normal sexual intercourse was performed without troubles in both cases. |
A 26-year-old girl reported with the complaint of dripping of fluids from the nose, difficulty in chewing food, and unesthetic appearance due to multiple missing teeth []. The past medical history revealed an operated case of bilateral cleft lip and palate. Multiple surgical interventions were carried out in the past for the correction of lip, palate, and nose defects. The past dental history included orthodontic treatment for 5 years and extraction of two maxillary posterior teeth. On general examination, the patient was moderately built and well oriented. On examination of the digits, the index finger on the left hand had clinodactyly and the left foot digits presented with brachydactyly as well as syndactyly of the fourth and fifth digits []. The right foot presented with clinodactyly of all digits []. On extraoral examination, the patient had an asymmetric face in vertical thirds and horizontal fifths with midface deficiency []. Other features noted were frontal bossing, hypertelorism, flattened nasal bridge, altered alar base morphology, scar in the philtrum region, and a concave profile. Intraoral examination revealed multiple missing teeth in the maxillary arch and a defect of the anterior hard palate (10 mm × 12 mm) in dimensions. The mucosal lining of the hard palate was irregular or corrugated. Family history revealed that the parents from a non-consanguinous marriage were healthy, had two girls, and a history of one miscarriage in the second trimester. Cone-beam computed tomography, radiograph foot, karyotyping, and gene sequence analysis were performed. A radiograph revealed multiple missing teeth, impacted maxillary right third molar, and mandibular left third molar. It also showed deficient bone in the maxillary anterior region with oronasal communication. Radiograph of the foot presented complete coalescence of metatarsals in all digits. Karyotyping was normal and gene sequence analysis revealed mutation at short arm of X chromosome at Xp22.23 site. A diagnosis of OFDS (Type – I) was made.[] In this case, since the surgical and orthodontic intervention had already been done, prosthetic rehabilitation was taken up. It was planned in three phases: Phase I – fabrication of interim obturator, Phase II – fabrication of definitive obturator, and Phase III – fabrication of foot prosthesis.\nIn the Phase I, an interim obturator was fabricated. Primary impressions of the maxillary and mandibular arch were made with irreversible hydrocolloid (Algitex, DPI), and the casts were poured using Type III dental stone (Kalabhai). Jaw relation, teeth arrangement, and try-in were completed. All conventional clinical and laboratory procedures were followed. The finished interim obturator was inserted in situ and evaluated for retention, support, stability, esthetics, and phonetics. The patient was called for follow-up for 2 months.\nAfter patient compliance and motivation with Phase I, the next phase included fabrication of a definitive obturator. Primary impressions and casts were made similar to Phase I, and the maxillary cast was evaluated on the surveyor. Designing for the metal framework was done by marking height of contour, selection of components, maxillary complete palate major connector with modified design, occlusal rests on the maxillary posterior teeth, cast circumferential clasp as direct retainer, and cingulum rest on the maxillary left canine providing indirect retention. Teeth preparation was completed as planned and a final impression was made using a two-step polyvinyl impression technique (Affinis, Coltene) and model was retrieved. Block out of undercuts was completed on master cast which was then duplicated using reversible hydrocolloid. A refractory cast was poured using phosphate-bonded investment material (Degugest). Designing of metal framework was completed using wax. Spruing, investing, burnout, and casting were completed to fabricate metal framework. The framework was retrieved, finished, and polished. Try-in was done to evaluate for fit, retention, support, and stability []. Jaw relation recording, teeth arrangement, and try-in were completed, and all conventional laboratory procedures were followed to fabricate a definitive obturator. The prosthesis was inserted in situ and evaluated for retention, stability, esthetics, and phonetics. The definitive obturator significantly improved form, function, esthetics, and the confidence of the patient [].\nAfter rehabilitating the intraoral defect, in the Phase III, the patient was rehabilitated with a foot prosthesis. An impression of the affected foot was made with a modified box technique using an irreversible hydrocolloid (Algitex) and working model was poured using dental stone (Kalabhai). The impression of the contralateral healthy foot was made by fabricating a custom tray with impression compound (DPI), and an impression was made using irreversible hydrocolloid material (Algitex, DPI). Modeling wax (Ruthinium Dental products) was heated in a wax bath and was poured into the impression to fabricate the wax pattern of the foot. The wax pattern was split into individual digits which were then reassembled on the working model to complete the wax pattern of the planned prosthesis []. Flasking of the wax pattern was done using a multi-pour method with orientation grooves at each pour using Type II and Type III gypsum product (Kalabhai). The assembly was placed in a warm water bath for dewaxing and a four-piece mold was obtained which helped in the fabrication of prosthesis with different shades on the dorsal and plantar surface []. Room-temperature-vulcanizing (RTV) silicone (Copsil T – 30 TN, COP) was used for the fabrication of prosthesis. A nylon stocking was placed on the working model over which the complete prosthesis was fabricated []. The nylon stocking not only eased the use of prosthesis but also the microroughness of the surface provided the retention to prosthetic material. Silicone was mixed with intrinsic colors following the manufacturer's instructions to closely match the shade of the patient []. After packing of silicone on the dorsal surface and ventral surface of the mold, the complete flask assembly was closed and left at room temperature for 24 h to complete curing. Putty index (Flexceed, GC) of nails of the patient's contralateral foot was taken and custom-shaped nails were fabricated using autopolymerizing polymethylmethacrylate (DPI). The prosthesis was retrieved, finished, characterized using extrinsic colors, and custom-made nails were attached using silicone adhesive (G611, Technovent) []. The prosthesis was inserted in situ and was evaluated for fit, esthetics, and gait of patient []. A comprehensive prosthetic rehabilitation not only improved form, function, and esthetics but also remarkably improved the confidence, psychological attitude, and social acceptance of the patient. |
A 48-year-old Caucasian woman presented with a painful abdominal wall mass in June, 2003. She did not have any history of trauma, surgery, or malignancy. Physical examination revealed a fixed and firm mass, about 5 cm in size, located around the umbilicus. The patient was afebrile. Laboratory parameters including erythrocyte sedimentation rate, complete blood count, C-reactive protein, and tumour markers were in normal values. Abdominal magnetic resonance imaging (MRI) showed an inhomogeneous mass composed of fibrous bands in the subcutaneous tissue in the periumbilical region, 6 × 5 cm in size, with no visceral involvement. An incisional biopsy was performed twice within a month under local anesthesia, where diffuse fibrosis and exudated suppurations were encountered. The histopathological examination indicated active chronic inflammatory changes, fat necrosis, and diffuse myofibroblastic activity corresponding to the diagnosis of an inflammatory pseudotumor. Microbiological examination and bacterial culture did not reveal any convincing findings. After initiating empirical antibiotic therapy with amoxicillin-clavulanic acid per oral, the patients was discharged, however, she was lost to medical follow up. In March 2005, she was readmitted to our clinic with the deterioration of her symptoms. Repeat MRI of the abdomen demonstrated the progression of the pseudotumor which reached a size of approximately 13 cm in diameter within the abdominal wall. Surgical exploration was decided on the basis of her medical history, previous histopathological, and the radiological findings. Following an abdominal transverse incision, intraoperative findings revealed a fibrotic mass of 12 × 13 cm within all layers of the anterior abdominal wall (). The omentum was adherent to the parietal peritoneum underneath the mass. The lesion was excised including the affected muscles and the omentum. The resulting abdominal wall defect was closed by implantation of a 20 × 20 cm intraperitoneal polypropylene mesh (Prolene, Ethicon, Johnson & Johnson, Somerville, NJ, USA). Histopathological examination showed acute and chronic inflammatory changes due to actinomycotic colonies (). Once the diagnosis was confirmed, a further questioning concerning her gynecological history revealed that she had been using the same IUCD without change for eight years. Gynecological examination was unremarkable except for an IUCD (Multiload Cu-375, Multilan, Organon, Oss, The Netherlands) which was removed consequently. Culture results from the IUCD revealed actinomycosis. She was prescribed a high-dose penicillin G (10 million units/day for three weeks) therapy and discharged on the seventh postoperative day without any complication. Oral penicillin was continued for further eight months. She recovered uneventfully and has been disease-free for four years. |
A 63-year-old woman with metastatic breast carcinoma presented to the ophthalmology clinic with diplopia in right gaze and head turn to the right. Medical history revealed that she was diagnosed with estrogen receptor (ER)-positive and progesterone receptor (PR)-positive invasive ductal carcinoma 1 year earlier with mediastinal lymph node and bone metastasis at the time of diagnosis. She was treated with zoledronic acid 4 mg monthly and paclitaxel 80 mg/m2 weekly for 12 weeks, followed by endocrine therapy with letrozole.\nOn ophthalmological examination, best corrected visual acuity was 20/25 in both eyes. Slit-lamp examination of the anterior segment and fundus was unremarkable other than bilateral posterior chamber intraocular lenses. On motility exam, abduction was totally limited in the right eye with globe retraction and narrowing of the palpebral fissure on attempted abduction (). Abnormal head position towards the right side was noted. Magnetic resonance imaging (MRI) revealed isolated enlargement of the right medial rectus muscle (). Clinical evaluation and laboratory studies were carried out for differential diagnosis. There were no clinical findings suggestive of thyroid eye disease and thyroid function tests were normal. Rheumatologic assessment for inflammatory and vasculitic diseases was not contributory. Biopsy of the right medial rectus muscle was performed to establish a definite diagnosis and initiate appropriate treatment.\nHematoxylin and eosin staining of the biopsy specimen revealed large, round to polygonal epithelioid tumor cells arranged in loosely cohesive clusters and sheets infiltrating fibrocollagenous tissue and muscle fibers (). Immunohistochemical analyses using streptavidin-biotin peroxidase complex method revealed panCytokeratin and cytokeratin 7 positivity (). ER, PR and human epidermal growth factor receptor 2 (HER2/neu) were negative (triple-negative). Based on the patient’s clinical history and the morphological and immunohistochemical features of the tumor, she was diagnosed with breast carcinoma metastasis to the right medial rectus muscle. Pathological examination demonstrating a triple-negative breast carcinoma indicated discordance with the primary tumor, which was ER- and PR-positive at the time of diagnosis.\nThe patient was referred to the radiation oncology department for external beam radiation therapy. The orbital mass was irradiated with 45 Gy in 15 fractions. Following radiotherapy, chemotherapy with docetaxel 100 mg/m2 once every 21 days was initiated. After 15 months of follow-up, abduction of the right eye has partially recovered; the patient is stable and continuing to receive palliative chemotherapy. |
A 33-year-old male, married patient from a rural background presented in the emergency unit of our hospital with a history of alcohol dependence from the past 10 years. The patient presented with a history of recurrent episodes of abdominal pain, tenderness in epigastric region, and weight loss from the past 6 years. The patient had an episode of alcohol withdrawal seizure 2 months back. His multiple clinical problems were manifestations of chronic pancreatitis and its complications which included raised blood sugar levels. His pancreatitis was initially precipitated by heavy alcohol use. The patient used to take heavy alcohol every day for the past 10 years. He was irritable and aggressive in nature. He used to have recurrent fights with his family members after taking alcohol. His vital signs at initial presentation in the emergency unit were clinically afebrile, pulse 77/min. Respiratory rate – 18/min, blood pressure – 135/73 mmHg. His oxygen saturation (SPO2) was 100% on room air.\nIn general, the patient was conscious and well oriented to time, place and person, thinly built, seemed uncomfortable, was well dressed but not well groomed. He kept on moving here and there in the emergency ward. He sat still and winced when we spoke about his clinical condition. His speech was in high tone and volume. Mood was irritable. Ears and throat examination revealed no abnormality. He had poor oral dentition. During general physical examination, no evidence of lymphadenopathy or a carotid murmur was observed. His abdominal examination revealed tenderness with palpation in the epigastric region. There was no guarding or rebound tenderness. His abdomen was not rigid. Bowel sounds were quiet. The patient was thoroughly investigated. His hemoglobin level was 12.3 g/dl. White cell count was 4.2 × 103/μL with 54% neutrophils. His metabolic panels were in normal range. Serum calcium levels were 8.2 mg/dl but serum amylase level was elevated at 290 IU/L, serum albumin level was 4.5 g/dl, triglycerides were 213 mg/dl, blood sugar levels 320 mg/dl, glycosylated Hb1c level was 8.1 (poor diabetic control). His viral markers were nonreactive for HIV and hepatitis B and hepatitis C serology. For detailed monitoring and investigations as well as treatment of alcohol dependence syndrome, the patient was admitted to psychiatry ward.\nInitially, the patient was managed with intravenous fluids and thiamine 100 mg/day, lorazepam 8 mg/day, and haloperidol 10 mg/day to control the alcohol withdrawal and aggressive behavior. On 2nd day of admission, an extensive workup, including consultation with surgeon and gastroenterologist was undertaken. His detailed ultrasonography was done which showed atropic pancreas along with large cystic lesion in lesser sac which was likely to be pseudocyst pancreas. His CT scan abdomen was undertaken which showed cystic lesion in the body and tail region of the pancreas as pancreatic pseudocyst.\nGradually patient was shifted to oral medications and oral hypoglycemic agents. As the patient was started with oral risperidone 4 mg/day, he developed extra pyramidal symptoms as drooling of saliva, slurring of speech, and tremors in hands. The antipsychotic medication was stopped, and the patient was started with trihexypenidyl 4 mg/day, but patient went in delirium after 24 h of medications. The patient was shifted to Intensive Care Unit setting and managed conservatively by medical intensevist. Patient started showing recovery in the resolution of delirium condition within 3 days. The patient was again shifted back to psychiatry ward after the recovery of delirium condition.\nUltrasonography of abdomen was repeated after 1 week of treatment which revealed pseudocyst pancreas size which was 89 mm × 63 mm × 132 mm with the volume 387 cc seen in the region of body and tail of pancreas with uniform thick internal echoes with wall thickness 2.5–3.5 mm with normal uncinate process, head and neck of pancreas with normal splenoportal vessels. CT of the whole abdomen was undertaken by taking 5/10 mm contagious section from domes of diaphragm up to the pelvic outlet. Pancreas showed a well-defined cystic lesions in the body and tail region measuring approx 12.7 cc × 7.2 cc × 8.4 cc toward the greater curvature of the stomach. No solid component was seen, and no internal septations were observed. No foci of calcification were revealed on CT scan abdomen. The main pancreatic duct was not dilated much and head, and uncinate process of the pancreas showed normal enhancement.\nThe surgical opinion was taken at regular intervals, and surgical intervention was planned after the patient became medically stable and his serum amylase level and blood sugar levels were in normal range. His family members wanted to postpone surgical options for next 2 months and consent for surgery was not given by the patient and his family members. Finally, the patient recovered well from all his symptoms, and he was discharged without surgical intervention under satisfactory condition after 1 month of treatment. On regular follow-up in outdoor, patient had abstinence from alcohol and started his field work and started looking after his family and recovery was unremarkable. |
Case 1 (Successful Treatment of Peri-Implant Bone Loss). A 48-year-old female, with no significant medical history, presented to the dental office as she was unhappy with her anterior cantilever bridge in the upper anterior region (Figures and ). Her initial appointment was for a consultation and periodontal exam. Her periodontal findings were normal with only isolated areas of mild gingival inflammation. Following the initial consultation, the patient was referred to a prosthodontist to section the bridge and to fabricate a provisional partial denture and a surgical template for implant placement (). The implant was placed using a standard protocol (). The implant used was a Straumann Roxolid® implant 3.3 mm in diameter and 10 mm in length. The area was sutured and the patient was given routine postoperative instructions. The patient was prescribed Amoxicillin 500 mg, 21 capsules to take 1 capsule three times a day for 7 days. Pain control included using Ibuprofen 600 mg every 6–8 hours for days 1 and 2 and then as needed after that. Healing appeared to proceed uneventfully (Figures and ). However, at second-stage surgery, which was done at 3 months following implant placement, vertical bone loss was noted on the mesial and distal aspects of the implant (). It was decided to treat the site using the following protocol: the use of titanium curettes to instrument the implant surface and application of ethylenediaminetetraacetic acid (EDTA) twice for 2 minutes. The implant surface was then rinsed with normal saline following each application. The site was grafted with freeze-dried bone allograft (FDBA) combined with Emdogain (). A similar post-op protocol was followed. Patient called the next day following surgery and indicated that she had fallen at home and hit her lip and that the site of surgery was bleeding. Following another visit, a second dose of antibiotics, Clindamycin 150 mg, 21 caps, 1 capsule three times a day for 7 days, was prescribed. Healing proceeded uneventfully. A periapical radiograph was taken at about 4 months (). The patient was referred back to the prosthodontist for the fabrication of a provisional crown (Figures and ). Following a period of about 4 months, the patient had her final restoration fabricated (Figures and ). She is currently on a 6-month dental prophylaxis schedule. |
A 71-year-old man with progressive angina was found to have severe stenosis in the circumflex artery and complex CTO of the left anterior descending artery (, Supplementary ). Coronary artery bypass surgery was offered to the patient but declined. He underwent uncomplicated stenting of the circumflex artery, followed by staged CTO recanalization of the LAD. A guidewire was passed relatively easily across the occlusion into the true lumen of the distal LAD beyond the bifurcation. Antegrade wiring of the diagonal artery was difficult. An epicardial collateral from the distal right coronary artery was used to access the diagonal branch retrograde, and this wire was steered into the antegrade guide catheter and externalized (). Both the LAD and diagonal were dilated with 3.0 mm noncompliant balloons. The diagonal was stented into the proximal LAD, and the distal LAD was rewired. The distal LAD was dilated through the stent struts to allow passage of stents into the distal LAD (Culotte technique). The distal LAD was stented using four everolimus drug-eluting stents. The stents were postdilated with 3.0 mm noncompliant balloons in the diagonal and distal LAD and 4.0 mm noncompliant balloon in the proximal LAD. Within the distal-most stent in the distal LAD, the balloon had a persistent waist until an inflation pressure of 12 ATM. At that point, the balloon suddenly expanded. Angiography revealed contrast filling of the right ventricle (RV), with the appearance of one or two focal jets of contrast extravasation at the location of the rigid lesion in the distal LAD, consistent with iatrogenic LAD-RV fistula (, Supplementary ). Of note, the flow beyond the stents in the distal LAD was not seen, likely due to shunt flow and “coronary steal.” The patient remained hemodynamically stable and was asymptomatic, and it was elected to manage the fistula conservatively. Serial echocardiograms revealed only trace pericardial effusion. He was discharged in stable condition.\nApproximately three weeks after the CTO procedure, the patient was presented to the emergency department with chest pain and dyspnea. He described several episodes of “tearing” sensation in the chest. Blood pressure and heart rate were 123/69 mmHg and 64 beats per minute, respectively. A 12-lead ECG revealed inferior T wave inversion. Troponin I was elevated to 0.06 ng/mL. Urgent coronary angiography was performed, which revealed patent stents in the proximal LAD and diagonal branch. LAD-RV fistula appeared relatively unchanged compared to during the CTO procedure (). The right coronary artery was normal and provided a very faint collateral to the apical LAD. Left ventriculography in the left anterior oblique projection revealed no evidence of a ventricular septal defect. The patient developed profound hypotension of unclear etiology during angiography. A right heart catheterization revealed normal filling pressures, normal cardiac output and ratio of pulmonic to systemic flow (Qp:Qs) 1.7. A transthoracic echocardiogram revealed preserved ejection fraction and normal left ventricular wall motion with turbulent flow signals at the LV and RV apex throughout the cardiac cycle (Supplementary ). There was no pericardial effusion. The patient was transferred to the cardiac intensive care unit in stable condition.\nHeart team evaluation was undertaken, and it was decided to perform coil embolization of the distal LAD. A standard 6 French left coronary guide catheter was used to advance a ProGreat (Terumo Medical Corp., Somerset, New Jersey) guidewire and 2.8 French catheter into the distal LAD. This was used to deploy two Ruby (Penumbra, Inc., Alameda, California) coils at the distal end of the stent.\nThere was still persistent shunt flow, so two Tornado (Cook Medical, Bloomington, Indiana) coils were subsequently deployed. Angiogram of the LAD confirmed complete cessation of flow into the distal LAD and absence of shunt flow (, Supplementary ). Hemodynamics postprocedure demonstrated a 20 mmHg increase in systolic blood pressure and normalization of Qp:Qs. A follow-up echocardiogram revealed the obliteration of apical shunt flow, normal left ventricular ejection fraction, and no left ventricular wall motion abnormalities. Creatine kinase eight-hour postprocedure was normal. The patient was discharged home in stable condition. At follow-up, one month later, the patient remained asymptomatic. |
A 17-years old unmarried female from Bajura district of Nepal came to the dermatology out-patient department with a complaint of a single plaque over the right upper lip for the last 6 months. Lesion started as a small red papule which gradually increased in size with central crusting. No history of fever or weight loss was given. There was no significant family or medical history. Multiple over the counter treatment was used as trial without relief. There was no history of travel outside of her village until the time for consultation.\nOn examination, a single round to oval crusted plaque approximately 1.5 × 1.5 cm was noted over the right upper lip. Neither an induration nor easy bleeding was noted. No regional lymphadenopathy was present ().\nA slit skin examination was done for acid-fast and Giemsa stain which was negative for any organism. An incisional biopsy was done which showed multiple epitheloid type multinucleated giant cells suggesting Lupus vulgaris. However, a mantoux test revealed an induration of only 2 mm in 48 hours.\nThe morphology of lesion prompted us to investigate furthermore for cutaneous leishmaniasis before starting on anti-tubercular medications. We decided to do PCR for Leishmania species from the lesion. The sample was sent to the Laboratory of the Central Department of Biotechnology, Tribhuvan University, Kathmandu, Nepal due to unavailability of PCR in the National Academy of Medical Sciences, Bir hospital. PCR was done using the nested protocol previously described by Noyes, et al. in 1998. The DNA template for PCR was extracted manually from the lesional tissue. After second-round PCR, a band size corresponding to the size of approximately 700 bp was obtained that confirmed the presence of Leishmania parasite (). The band size corresponded to that typical of Leishmania donovani.\nA serological test conducted with an rk39 rapid diagnostic test kit also tested positive for this case.\nAn ultrasound of the abdomen and pelvis was normal. Liver function tests, renal function test, and blood sugar were also all within normal limits.\nPatient was diagnosed as a case of cutaneous leishmaniasis and was advised for follow-ups without any treatment. The patient came for follow-up in one month's time where the plaque had decreased significantly (). The patient was advised for regular follow-ups on a monthly basis till the lesion subsided. The lesion healed spontaneously within a couple of months. |
A 29-year-old woman with Marfan's syndrome was scheduled to undergo the elective Bentall's operation and thoracic aortic replacement with artificial graft. There were no abnormal findings or special histories on the preoperative evaluations.\nOn patient's arrival at the operating room, standard monitoring devices were applied including the left radial arterial cannulation. Anesthesia was induced and maintained according to institutional guidelines. After anesthetic induction, the right subclavian vein was cannulated with MAC™ Multi Access Catheter (ARROW international Inc., PA, USA) using the infraclavicular Seldinger technique. During this attempt to cannulate the subclavian vein, there was neither air aspiration nor arterial puncture. After ensuring the aspiration of dark and nonpulsatile blood, a guidewire was introduced without any resistance. This was followed by sliding the catheter over the guidewire, and the wire was withdrawn without apparent resistance. Thus, the catheter was placed without any difficulty during catheterization. After that, the patient received standardized management according to institutional guidelines including mechanical ventilation and cardiopulmonary bypass during intraoperative period.\nThe operation was performed uneventfully. The patient was transferred to the intensive care unit (ICU) and was very stable in vital signs and laboratory tests. On postoperative day 1, the patient was successfully weaned from mechanical ventilation. There was no evidence of hemorrhage including the volume of blood drained to chest bottles and follow-up chest X-ray (). On postoperative day 3, the patient was transferred to the general ward and the central venous catheter was removed. Ten minutes after the removal of the catheter, the patient complained of dyspnea and pleuritic pain on right side, at which time approximately 1,700 ml of blood was abruptly drained to chest bottles and the patient's chest X-ray confirmed a hemothorax on the same side as the cannulation. Although the blood pressure was stable with administration of intravenous fluid and norepinephrine, hematocrit values dropped from 32.0% to 22.6%.\nThe patient promptly underwent emergent thoracotomy and approximately 2,500 ml of clotted blood was evacuated during operation. The patient was maintained hemodynamically stable with infusion of 0.05 mcg/kg/min of norepinephrine, 3,500 ml of crystalloid and 4 units of packed red blood cell. There was an injury site by the central venous catheterization on the right side of superior vena cava, which was 3 cm distant from the junction of the right atrium and superior vena cava without any other injuries and reinforced by hemostatic materials. After transfer to the ICU, vital signs and other laboratory findings such as arterial blood gas analysis were stable without any pharmacologic supports. The patient was transferred to the general ward after a 4-day ICU stay and discharged on the 15th day after the thoracotomy without any complications. |
A 39-year-old woman, gravida 1, para 0, was diagnosed with invasive squamous cell carcinoma of the cervix following conization. Pathological findings showed carcinoma consistent with FIGO stage IA1 with lymphovascular invasion. She was referred to Keio University Hospital. The patient and her husband were informed of the treatment options, including AmRT and pelvic lymphadenectomy. The patient was told that the outcome of this procedure could not be guaranteed because an insufficient number of these procedures have been performed worldwide to yield reliable conclusions. The patient wished to preserve fertility, and she and her husband signed a written consent form agreeing to this treatment. Pathological findings after AmRT and pelvic lymphadenectomy showed no residual tumor and no lymph node metastasis. There was no finding of an ovarian tumor before surgery. A left ovarian cyst of 4 cm was identified during postoperative follow-up.\nAt 3 years and 6 months after surgery, the patient underwent IUI and then had fever and pain in her left lower abdomen 10 days later. At her first visit, her temperature was mildly elevated to 37.5°C. The patient's pregnancy was denied because a qualitative urine human chorionic gonadotropin (hCG) test was negative. A tumor with tenderness was palpated in the left adnexal area. A cystic tumor of 64x 41 mm was found by transvaginal ultrasonography (). Blood tests showed increases in white blood cell (WBC) count to 11900/μL and C-reactive protein (CRP) to 22.80 mg/dL. The patient was diagnosed with PID with ovarian cyst infection and hospitalized for treatment. Conservative treatment with antibiotics was initially used, but her symptoms did not improve. On hospital day 8, blood tests showed a further increase in WBC count to 23900/μL and CRP to 28.17 mg/dL, and pelvic CT showed that the ovarian cyst had grown to 10 cm in size ().\nWe decided to perform laparoscopic left ovarian cystectomy on day 8. Since the patient had a history of open surgery, adhesion was likely in the abdominal cavity. Before surgery, we asked the urologist to insert bilateral 6 Fr ureteral catheters because of possible difficulty identifying the ureters. The catheters were fixed to the thigh with tape (). The uterine manipulator was not inserted before surgery to avoid the risk of uterine perforation.\nCO2 pneumoperitoneum was established at 10 mmHg. Laparoscopic left ovarian cystectomy was performed using typical trocar placement. The left ovary was swollen to 10 cm and the fluid contents were purulent (). The left adnexa, posterior uterine wall, and retroperitoneum were firmly adhered (). The bilateral fallopian tubes were firmly adhered to the surrounding tissue and were unable to be identified. During surgery, by moving the catheter manually back and forth from outside the body, we were able to identify the ureters visually (). The 7 cm uterine manipulator was inserted during surgery under a laparoscopic view. The left ovarian cyst was excised, leaving the normal part of the ovary (). The operation time was 2 h and 58 min, and blood loss was 550 mL. No complications occurred during or after surgery. After the operation, symptoms improved rapidly and the patient was discharged 8 days after surgery. The excised specimen was pathologically an endometriotic cyst. A bacterial culture of the cyst fluid was positive for Prevotella bivia, Prevotella species, and Finegoldia magna. |
A 75-year-old man with a past medical history of hypertension but no past ocular history tripped on the sidewalk and hit his head on a branch. The patient was referred to the local emergency department, where the initial examination revealed an approximately 2-cm-long laceration in the lateral part of the left upper cheek with a wooden foreign object (Fig. ). Examination of the left globe was complicated due to periorbital swelling, hyperglobus, mechanical blepharoptosis, and difficulties in manually lifting the upper eyelid. There was a left-sided relative afferent pupillary defect, exotropia, exophthalmos, and no light perception (NLP). The motility of the eye was with significant limitation of all ductions, which most likely was due to compression of the globe and extraocular muscles by an orbital foreign body. Slit lamp examination revealed conjunctival injection, clear cornea, and normal depth of the anterior chamber without hyphema. Dilated funduscopy was without evidence of penetrating trauma or other posterior segment manifestations of injury to the globe. The intraocular pressure of the left eye measured with iCare was 23 mm Hg. The intraocular pressure of the right eye was not measured.\nA computed tomography (CT) scan of the facial bones revealed a cylindrical foreign body measuring 7 cm in length and 6 mm in diameter (Fig. ). It penetrated the left orbit inferolateral below the inferior rectus muscle. It was situated craniomedially through the posterior medial orbital wall into the sphenoid sinus, without penetrating the posterior wall (Fig. ). The minimal distance from the foreign body to the ICA was 6 mm. The globe and optic nerve were intact, but the patient had relative left-sided exophthalmos due to the displacement of the inferior and medial rectus muscles. There were fractures in the floor of the orbit, medially and caudally of the foreign object, penetration of the lamina papyracea, and air trapped in the soft tissue along the path of the wound.\nThe patient was transferred to the Department of Ophthalmology at our University Hospital and treated with intravenous antibiotics (vancomycin and ceftazidime). The following morning, the patient was transferred to our Department of Otorhinolaryngology, Head and Neck Surgery, and Audiology for joint venture surgery.\nThe removal of the object was performed by CT image-guided endoscopic surgery. Under visualization of and manipulation in the sphenoid sinus, a 7-cm-long stick was extracted through the wound in the lower eyelid (Fig. ). Significant amounts of debris and splinters were removed through the nose. The skin laceration was adapted and stitched together.\nThe patient showed no sign of infection, and the antibiotic treatment was adjusted to intravenous cefuroxime as monotherapy after consulting with the Department of Microbiology. The patient was discharged after 3 days with oral antibiotics, amoxicillin/clavulanic acid.\nSix months after the trauma, the patient had the last follow-up in the ophthalmology outpatient clinic. The visual acuity in the left eye had increased to 0.4, and there was still a relative afferent pupillary defect and affected color vision. Slit lamp examination and dilated funduscopy was unremarkable besides a pale atrophic optic disc. Spectral-domain optical coherence tomography of the peripapillary retinal nerve fiber layer (pRNFL) demonstrated marked thinning of the pRNFL (Fig. ). The motility of the left eye was normal except for reduced elevation to about 10°. There was no diplopia in spite of 20° of exotropia. The patient was diagnosed with traumatic optic neuropathy. |
A 65-year-old male came to the department for replacement of missing teeth and complained of sunken cheek appearance on either side. On examination, the patient has a completely edentulous maxillary and mandibular arch with a sunken buccal cheek []. The treatment plan decided was complete denture prosthesis with buccal cheek plumper on either side of the maxillary denture to improve esthetics.\nThe primary impression was taken with impression compound, and the primary cast was made with dental plaster. Self-cure acrylic resin special tray was fabricated, and border molding was completed using green stick compound, and the final impression was taken with zinc oxide eugenol impression paste. Record base was fabricated with self-cure acrylic resin, and occlusion rims were constructed. During the jaw relation procedure, it was found that the occlusal rim does not provide enough support for the cheek muscles. To improve the esthetics, the extra waxes were added on both sides of the occlusal rims near the first molar region until it provided enough support to the cheek muscles []. It was verified that the extra waxes did not cause dislodgment of record bases. Semi-anatomic teeth were selected according to the patient age, sex, and personality. The trial denture base was tried in the patient; the esthetics, phonetics, and jaw relation were verified. Facial esthetics was checked whether the wax buccal pad provided necessary esthetics without dislodging the wax trial denture base during functional movements of the cheek muscles. To reduce the weight of the prosthesis, the wax in the buccal pad was scooped out creating an open bulb with the border's thickness of 1.5 mm around the periphery []. The routine flasking, dewaxing, packing, and curing procedures were done. After deflasking, the complete denture was retrieved, trimmed, and polished. Since the open bulb could lead to food accumulation, the shellac base plate was used to form the lid for the buccal pouch []. After the extension and stability of maxillary complete denture with buccal pouch was checked in the patient mouth, lid with the shellac base plate material was replaced with heat cure acrylic, which was sealed with the hollow cheek plumper using self-cure acrylic resin [].\nThe maxillary complete denture with closed hollow bulb cheek plumper, and the mandibular complete denture was inserted []. The esthetics, the phonetics, and the stability of the prosthesis were checked. The patient was reviewed at 24 h, and the complete denture was found to be satisfactory both in esthetics and function. |
A 32-year-old parturient, Gravida 2, Para 1, came at 38 weeks of gestation for spontaneous labor. She had a history of obesity with a body mass index of 35.3 kg/m2, tympanoplasty, and gastric band surgery. During her first delivery, epidural analgesia was complicated with Horner's syndrome. Rapid dilatation did not allow the anesthetic team to replace the epidural catheter then, and the patient had spontaneous vaginal birth without any analgesia.\nAnesthetic consultation during her second pregnancy pointed out this complication.\nFor her second labor, an epidural catheter was placed without difficulty. Test dose (3 mL of 1% non-epinephrine lidocaine) was administered, and the aspiration test was negative. Twenty minutes after the first anesthetic bolus (50 mg of 1% non-epinephrine lidocaine), the pain was relieved, but the patient presented a left Horner's syndrome associated with a full and exclusive left sensory block (T4 level) without a motor blockade. Hemodynamic parameters remained stable during the whole process. The subdural catheter position was suspected, and the second placement of the epidural catheter was performed after a total regression of neurological signs and under cardiac monitoring. After a new test dose and negative aspiration, careful induction of epineural analgesia was performed, with 50 mg of non-epinephrine lidocaine divided into two injections. Fifteen minutes later, the patient presented a contralateral right Horner's syndrome associated with no sensory block. Again, hemodynamic parameters were perfectly stable during the whole procedure and surveillance. Due to the potential risks and the impossibility of using it in case of an emergency C section, the catheter was removed and not inserted again after the full disclosure of information to the patient who agreed with the anesthetic management. Once again, she had rapid labor and had no instrumental vaginal delivery with no analgesia 30 min later. Follow up did not reveal any complication, and she was discharged from the hospital 3 days later. |
A 23-year-old lady presented to our hospital complaining of an asymptomatic subcutaneous nodule over the dorsum of the nose which had started about 8 months ago. The nodule had progressively grown in size over the past few months. There was no history of trauma. She gave a history of similar nodules over the scalp and great toes. The nodules over the scalp healed after discharging a chalky material. The nodules over the great toes were left untreated as the patient did not give consent for its evaluation as they were not cosmetically detrimental. There was no family history of similar lesions.\nAt the time of presentation, she had no complaints of joint pain, muscle weakness, skin rashes, Raynaud's phenomenon, dysphagia or dyspnoea. She was not on any medication and had not undergone any surgery [].\nOn physical examination, we found a firm subcutaneous nodule measuring approximately 0.7 × 1 cm. Intra-nasal examination was normal. The systemic evaluations including the neurological examination were normal, and the patient had no symptoms such as muscle weakness or stiffness.\nThe laboratory investigations revealed within normal serum calcium levels of 8.6 mg/dl and phosphorus levels of 4.8 mg/dl.\nBiochemical examinations gave normal results for a complete haemogram, erythrocyte sedimentation rate, blood sugar, uric acid, electrolyte, and liver function and kidney function tests. Routine urine examination was normal. Screening tests for collagen vascular disease, including anti-nuclear antibodies, anti-deoxyribo nucleic acid, anti-SM, anti nuclear cytoplasmic antibodies, serum C3, C4 and CH50 levels, and rheumatoid arthritis (RA) test were within normal limits. A radiological examination of the nose showed calcification at the region adjacent to the lower one-third of the right nasal bone with no bony invasion or erosion [].\nThe nasal mass was excised via an external approach. The mass was noted to be arising from the skin over the nasal dorsum and was overlying the lower one-third of the right nasal bone. There was no attachment from the mass to the underlying bone. The mass contained a pasty white material within it. After the excision, the soft tissue defect over the nasal dorsum was augmented using silicone block prosthesis [].\nThe histopathology of the swelling (on haematoxylin and eosin staining) showed calcified nodules of varying size within the fibrotic tissue, and some areas were surrounded by foreign body cell reaction consistent with calcinosis cutis []. This was confirmed by von Kossa staining [].\nOn the basis of clinical, radiological and histopathology data, a diagnosis of ICC of the right nasal dorsum was made. Post-operatively, she was started on a bisphosphonate, ibandronate 150 mg once a month. The hospital stay was uneventful, and she patient was discharged after suture removal. She is presently asymptomatic and has no nasal deformity. |
A 53-year-old male with body mass index of 42 kg/m2 was referred for management of bilateral asymptomatic nephrolithiasis. His family history was significant for nephrolithiasis in his brother and sister. He had passed five calcium oxalate stones since the age of 30 years. He was a smoker with no other medical history.\nThe initial abdominal plain X-ray (kidney, ureter, and bladder radiograph [KUB]) showed a left lower pole calcification and a 1.5 cm calcification on the iliac crest (). A noncontrast CT of the abdomen and pelvis revealed a 1.8 cm right midureteral stone (1288 HU) with proximal hydroureteronephrosis, a thin right renal cortex, two right lower pole stones 5 mm each (). In addition, a dilated tubular structure measuring 9 × 4.3 cm connected to the cecum and containing a few dependent calcifications was described. This was thought to be an appendiceal mucocele compressing the right mid ureter below the midureteral stone (). On renal scintigraphy, the right kidney was obstructed and contributed to 32% of the global renal function. After a failed attempt of extracorporeal shockwave lithotripsy, right retrograde flexible ureteroscopy (URS) was performed, which revealed a tortuous right distal ureter up to the midureteral stone that was impacted on an inflammatory stricture. In the presence of the stricture and of a tortuous ureter, we thought it was not safe to proceed with laser lithotripsy because of increased risk of ureteral trauma. An indwelling ureteral stent was placed and the patient was brought back for second-look URS and laser lithotripsy. During the second URS 4 weeks later, the impacted midureteral stone was accessed with a semirigid ureteroscope, and the stone was fragmented with Holmium laser energy (1 J, 10 Hz) and the fragments were removed using a zero-tip basket. At this point, a mucous plug was found in the dilated tortuous proximal ureter. A ureteral access sheath was placed, and using a flexible ureteroscope, the lower pole stones were fragmented and basketed out. An indwelling ureteral stent was placed for 1 week. Urine cytology from the ureter showed mucoid material with rare benign urothelial cells. Six weeks later, a triphasic CT scan showed stone-free status on the right side with residual mild right hydroureteronephrosis and a left lower pole caliceal stone (). A diuretic renal scintigraphy showed a nonobstructed right kidney with a chronically dilated pelvicaliceal system and a 34% differential function in the right kidney. Two years later, his diuretic renal scan did not show deterioration of the differential renal function, indicating that there was no significant obstruction. Stone analysis showed 20% calcium oxalate dihydrate, 70% calcium oxalate monohydrate, and 10% carbonate apatite. Metabolic stone evaluation showed adequate urinary volume of 2.1 L, hypercalciuria secondary to hypernatriuria, hyperuricosuria, and hyperoxaluria. After following low salt, low purine, and moderate oxalate diet, his hypercalciuria persisted, he was started on hydrochlorothiazide and amiloride for prophylaxis against recurrence of renal stones.\nSimultaneous to the urologic investigations, he was assessed by general surgery for the appendiceal mucocele. Two weeks after the first URS, he underwent colonoscopy, which did not reveal involvement of the cecum; two sessile polyps were found and biopsied with final pathology analysis of tubular adenoma. Two months after his second URS, he underwent laparoscopic appendectomy with stapling at the base of the appendix to include a rim of cecum. Frozen sections indicated negative margins. At the time of laparoscopy, there were no signs of mucinous ascites nor were any signs of peritoneal deposits. The mucocele was not ruptured during manipulation. The final pathology report showed an unperforated low-grade appendiceal mucinous neoplasm of 13 cm without lymphovascular invasion. Since the whole mucocele was completely excised, it did not require any further follow-up. |
Herein, we describe a three-generation family of Italian origins. The proband is a 2 years old boy referred to our center for suspected Food-Protein induced Enterocolitis ().\nThe mother of the proband is a 31 years old woman. She reported allergic rhinoconjunctivitis and recurrent gastrointestinal complaints from a young age, described as frequent loose stools and abdominal pain, interpreted as food allergies. She also experienced dizzy spells, vertigo, and small skin rash. Laboratory findings included normal Ig levels, normal total IgE levels, and no specific Ig for known allergens, whereas increased basal serum tryptase was found. The maternal grandfather also reported non-specific gastrointestinal symptoms and elevated serum tryptase. The father of the proband had no relevant clinical history.\nThe proband was born at term via vaginal delivery from an uncomplicated pregnancy. The baby at birth weighed 2,750 g. He received mixed feeding for the first month and only breastfeeding in the following period. He then developed cradle cap and peri-auricular dermatitis. Gastroesophageal reflux was also diagnosed. The child started weaning at 6 months of age, and after the introduction of wheat at 8 months, he developed facial erythematous skin rash and diarrhea (5–6 episodes per day) starting 2 h after the aliment ingestion. After wheat reintroduction, the symptoms recurred, and so it was excluded from the diet. He then also experienced episodes of diarrhea after 3–4 h from the intake of milk derivatives and vomiting linked to the ingestion of egg yolk, which led to the exclusion of these two aliments. Skin prick test for alimentary allergens resulted negative. In the following weeks, reintroducing small quantities of wheat was attempted, but that prompted a severe episode of vomiting that required hospitalization. A diagnosis of Food-Protein induced Enterocolitis was therefore suspected. At 1 year of age, he presented atopic dermatitis and a maculopapular hyperchromic lesion with a positive Darier sign in the right wrist, compatible with solitary cutaneous mastocytoma (). Basal serum tryptase was 19.8 ng/ml. Levels of Vitamine D 25 OH, alkaline phosphatase, and parathormone were normal. Total IgE levels were also normal, but positive specific IgE antibodies for dust-mite were found in the absence of related respiratory symptoms. After the first trial of cow-milk reintroduction, the proband experienced upward and rightward gaze deviation followed by generalized hypotonia and lack of responsiveness lasting about 5 s, with normal vital signs. Brief episodes of gaze deviation recurred weekly in the following months, associated with forward movements of the neck and reduced responsiveness. They mainly happened when the child was tired or agitated. No other neurological alteration was found.\nThe patient presented also sleep disturbances, including insomnia, pavor nocturnus, and somnambulism. Polysomnography ruled out the epileptic origin of these signs. Basal serum tryptase levels fluctuated in the following months, from 15.8 to 18.8 ng/ml. In order to better characterize the reason for the persistent rise in serum tryptase, the proband, the mother, and the father underwent genetic testing on the TPSAB1 gene, using the same method as previously described in Case 1. Triplication (four copies) of the TPSAB1 gene was found in the proband and the mother but not in the father.\nAfter genetic testing, progressive reintroduction of food was attempted. Cow milk, wheat, and egg were successfully reintroduced in the diet without additional specific treatments. Neurological symptoms also receded with time. |
A previously healthy 19-month-old Amish female presented to her primary care physician with a 5-day history of right neck swelling, decreased oral intake, and decreased urine output. She developed a rash on the day of presentation, which prompted her parents to have her evaluated. There was no history of recent travel or sick contacts. She had no previous immunizations and was otherwise healthy. A CT of her neck, ordered by the primary care physician, suggested a retropharyngeal abscess, and she was subsequently transferred to our tertiary care facility.\nOn examination, she was afebrile and breathing effortlessly. Torticollis was noted. There was firm induration of the right neck, measuring approximately 7 × 5 cm in diameter. She was found to have edema of the right side of her soft palate and peritonsillar region, as well as an erythematous fullness on the posterior oropharyngeal wall. She had 2+ tonsils, clear rhinorrhea with erythematous nasal mucosa, and normal tympanic membranes. A red macular rash was noted on her forehead, neck, abdomen, and back with accompanying vesicles on some of the lesions. Her lungs were clear to auscultation bilaterally. She had a grade I of VI flow murmur that disappeared when supine. No abdominal masses were palpable, and her bowel sounds were normal.\nThe CT scan revealed a hypodense multiloculated mass consistent with an abscess in the right retropharyngeal space, extending from the soft palate inferiorly to the angle of the mandible ().\nShe was admitted to the otolaryngology service and started on intravenous ampicillin-sulbactam. She was taken to the operating room the next morning for intraoral incision and drainage of the abscess. After induction of general anesthesia, a draining abscess was visualized on the posterior pharyngeal wall. An incision was made in the posterior pharyngeal wall under general anesthesia, but evacuation of pus was limited. An 18-gauge needle was used to aspirate several locations along the right side of her posterior pharyngeal wall from the nasopharynx to the hypopharynx. Bacteriological cultures from the abscess grew group A beta-hemolytic streptococcus. Intraoperative scrapings were taken of her skin lesions and returned positive for Varicella zoster antigen.\nShe was continued on IV antibiotics until postoperative day two, at which time she was switched to oral amoxicillin-clavulanate for a total of 10 days and discharged. Two weeks later, the family reported by telephone that her rash had resolved and that she had resumed a normal diet and activities. |
A 71-year-old male was admitted to the Department of Otorhinolaryngology, Head and Neck Surgery, University Hospital of Geneva, Geneva, Switzerland, with a nodular lesion that was soft in consistency and located on the left paramedian part of the chin. It had been present for over 25 years and showed recent growth (6 months), producing purulent material after being incised 1 month before. Clinical examination showed a cutaneous lesion measuring 3 × 2 cm, and the clinical differential diagnosis was infected epidermoid cyst and subcutaneous neoplasm.\nThe patient's previous medical and surgical history included a cholecystectomy, and an upper gastrointestinal bleeding developed on a gastric ulcer. Laboratory examination was unremarkable. An ultrasound investigation revealed a dermal and subcutaneous, mostly well-encapsulated cystic lesion measuring 2.3 cm in its greatest dimension and 1.07 cm in thickness. The lesion showed an irregular, deep border lying in close proximity with the aponeurosis.\nAn incisional biopsy of the lesion was performed. Histologically, the specimen showed an atypical squamous cell proliferation arising from the epidermis and extending to the dermis, consistent with an invasive, moderately to well-differentiated SCC.\nA facial magnetic resonance imaging and a neck computed tomography scan were performed to assess any eventual tumoral extension in the maxillary bone and locoregional lymph nodes. The radiological analysis revealed a suspected, poorly circumscribed, ulcerated, cutaneous, and subcutaneous lesion of the chin invading the perimaxillary striated muscle tissue without any osseous involvement. Two bilateral parotid lesions consistent with Warthin tumors were also radiologically detected.\nSeventeen days after biopsy, a radical resection of the tumor followed by local reconstruction with a rhomboid graft was performed. Histologically, the excision material revealed an ulcerated squamous cell tumor consisting of some clear cells and microcystic structures filled with eosinophilic keratinous material. There were also micronodules showing some prominent ductal differentiation resembling MAC, associated with foci of calcification. The tumor invaded the whole dermis, subcutaneous fat, and striated muscle tissue in the form of atypical squamous cells, micronodules, and cords (Fig. ). The tumoral depth was evaluated to be 1.7 cm, and the resection margins were free of tumor. There was only 1 image of perineural tumoral invasion.\nThe tumor cells showed diffuse immunostaining for pancytokeratins and p63. The focal ductal structures were highlighted by the expression of epithelial membrane antigen and carcinoembryogenic antigen (Fig.). The tumor cells were negative for cytokeratin 7, cytokeratin 20, Ber-EP4 (Ep-CAM), and androgen receptor.\nThis histological aspect was highly reminiscent of cutaneous MAC but with areas with pure squamous differentiation consistent with an invasive SCC mimicking MAC.\nThere was no further treatment. A clinical and ultrasonographic surveillance were performed every 3 months. The patient was followed up at 3 and 6 months postoperatively and showed no suspected cervical lymphadenopathy. The parotid lesions remained unaltered. A squamous papilloma of the right anterior oropharyngeal pillar has been detected and biopsied at 6 months during the follow-up. |
A 32-year-old male patient was diagnosed as a case of lepromatous leprosy and was on multidrug therapy (MDT) for the last 1 year. He presented to us with complaints of joint pain, swelling, and stiffness of both wrist and ankle joints for the last 2 weeks. On examination thickening and tenderness of the tendons were noted and there was no evidence of joint deformity []. However, he gave a history of recurrent crops of the tender nodular lesion during and after MDT. Magnetic resonance imaging of the right ankle joint was done for evaluation of tenosynovitis which showed small focal peritendinous fluid collection along the flexor digitorium longus tendon along with minimal fluid collection around the ankle joint. Fine-needle aspiration cytology (FNAC) was done from the affected nerve and cytology revealed the presence of foamy macrophages and acid-fast bacilli. PCR from the isolates confirmed the presence of Mycobacterium leprae. A diagnosis of leprosy presenting with tenosynovitis and erythema nodosum leprosum (ENL) was made and standard WHO MDT treatment for leprosy was started. Earlier, some of the authors have shown similar presentations of leprosy and with similar proven technologies.[] Though he was given a repeated dose of steroids for a short duration, with which tender nodules subsided, tenosynovitis kept on progressing. The patient was given combination therapy of thalidomide 200 mg per day plus prednisone 50 mg per day and MDT was restarted. However, no response was seen after 6 weeks of treatment and he complained of intense pain. We started with infliximab after doing a pre-biologic workup including chest X-Ray and viral serologies. Infliximab was given at a dose of 250 mg diluted in normal saline with dosing schedule at baseline and weeks 2 and 6. The treatment was well-tolerated and good improvement in symptoms was noted. The patient was followed up for 6 months, no recurrence of ENL was seen. |
A 5-year-old, 14.8 kg boy was diagnosed with EM and primary hypertension 3 months prior to consultation by the pain management team. He had been symptomatic since the age of 2 years with restless leg syndrome. Consultation with the pediatric rheumatology service was sought when his lower extremities became progressively more painful, there was an increase in the restlessness of his legs, and erythema associated with color changes of the skin in the feet and lower legs appeared. No secondary cause of EM was found. Laboratory evaluation was normal, including a complete blood count, basic metabolic panel, liver function tests, antinuclear antibodies, and C-reactive protein. Genetic workup revealed SCN9A gene mutation of the voltage-gated sodium channels. He had recently been admitted to the hospital twice for pain management. His symptoms had been refractory to multiple medical management modalities and interventions, including gabapentin, pregabalin, mexiletine, clonidine, topical ketamine, and amitriptyline cream as well as intravenous (IV) lidocaine, ketamine, and nitroprusside infusions. At the time of consultation, his home medications included mexiletine 30 mg per os (PO) twice a day (bid), gabapentin 60 mg PO three times a day, and prednisolone 8 mg PO once a day. Intermittently, his pain would become severe and his feet would be very hot, edematous, and red. His pain was relieved by placing his feet in cool water or using a fan to cool his feet. He was spending an increasing amount of time with his feet in the bath tub and had developed skin breakdown. He had been evaluated by nephrology for hypertension and started on clonidine (50 µg) by mouth three times a day. Diagnostic evaluation for secondary etiologies of hypertension was negative. Pain in his feet had been persistent with multiple daily acute, severe episodes of pain. Pain intensity was rated as high as 10 on a 0–10 scale. He had cold, erythematous discoloration with skin breakdown on both lower extremities (). Muscle atrophy was noted in both calf muscles. Sensory examination was normal with no allodynia or hyperalgesia. His quality of life had been severely impaired and he spent most of the time in the bath tub or wearing cooling boots. His sleep had been severely affected by pain and he slept with cold wraps on his feet. There was a lack of response from all the medical interventions, with distressing adverse effects related to the medications. He was referred to the pediatric pain clinic, and following evaluation and a discussion with his family, he was scheduled to undergo computed tomography (CT)-guided LSB. After obtaining a written parental consent, the procedure was performed under general anesthesia with the patient in the prone position. Under intermittent CT fluoroscopy, 15 cm 21 G Chiba needles were advanced in tandem via bilateral posterior approaches through the psoas muscles. The needle tips were positioned just anterior to the L3 vertebral body in the expected location of the lumbar sympathetic chains (). Then, 5 mL of 0.2% ropivacaine, 30 mg clonidine, and 20 μg triamcinolone were injected after negative aspirate for blood. Each needle was then flushed with 0.5 mL of saline, and a small amount of contrast was injected to document appropriate drug dispersion. Contrast distribution along the lumbar sympathetic chain was confirmed with CT imaging (). Immediately following the placement of the sympathetic blockade, there was increased temperature of both lower extremities, with visible vasodilation and decreased discoloration of the lower extremities (). This resulted in an improvement in his pain symptoms, improved sleep, and no flare-ups for ~3 weeks. There were some complaints of residual pain on the right side, and a repeat LSB was performed 4 weeks after the first blockade. After the second LSB, he was discharged home on oxycodone (1.5 mg by mouth every 4 hours as needed for pain), but he did not require any doses. With his hospitalizations, he had developed extreme anxiety related to the hospital and medical professionals. To manage his anxiety, he was started on clonazepam 0.125 mg PO at bedtime as needed and escitalopram 7.5 mg PO once a day. In addition, carbamazepine was gradually titrated to 50 mg PO bid in between the two LSB procedures. Acupressure with ear magnets was also instituted. Two weeks after the second LSB, the pain had decreased significantly. His appetite improved, and he gained ~5 pounds. He was more playful and was sleeping well. The swelling in his feet decreased, and his skin looked increasingly better with no further skin breakdown. The clonazepam was tapered and discontinued. He also started engaging in trips outside the house and enrolled in piano lessons and swimming. He completely stopped the use of cold wraps around his feet and legs. He has not required hospitalization since his second LSB >9 months ago. |
A 55-year-old woman presented three days after a sudden onset of right-sided chest pain, pleuritic and positional in nature, associated with an acute onset of shortness of breath. She had gone to her primary care physician, who performed a chest X-ray and urged her to come to the hospital. Upon presentation at the emergency department, her oxygen saturation was above 95% on room air, and she was not in any respiratory distress, but her exam was significant for decreased breath sound on the right. A chest X-ray confirmed a large right-sided pneumothorax with small pleural effusion. A chest tube was inserted on the right side for the resolution of the pneumothorax, and subsequent computed tomography (CT) scan of the chest revealed bilateral diffuse bullous disease of the lung with multiple cysts (Figure -). The patient underwent video-assisted thoracoscopic surgery for right thoracoscopic wedge resection of a lung bleb and talc pleurodesis. Gross examination of the specimen revealed several dilated air-like spaces ranging from 0.2 cm to 0.4 cm in size. The hospital course was complicated by postsurgical pneumonia, but she recovered fully and was discharged to home with only minimal symptoms of dyspnea on exertion. Upon further investigations, she was found to have multiple small lesions of angiomyolipoma on the right kidney with diffuse retroperitoneal lymphadenopathy. One of the lymph nodes was biopsied, and pathology revealed predominantly spindle cells positive for HHF35 and smooth muscle actin, consistent with the diagnosis of leiomyoma. At the eight-month follow-up at the pulmonology clinic, her pulmonary function test (PFT) showed normal vital capacity and forced expiratory volume in one second (FEV1), but moderately reduced diffusion capacity, which may also be related to LAM. At her 12-month and 24-month follow-up visits, her PFT results showed improvements in peak flow and diffusion capacity, and the patient continues to report no symptoms other than minimal dyspnea on exertion. |
A 37-year-old pregnant (20th week), otherwise healthy female, reported a history of chronic low back pain for many years. During pregnancy, she noted some worsening of this back pain, albeit she was still able to easily control her symptoms with occasional use of paracetamol (acetaminophen). On the day of her initial presentation she awoke disoriented and with memory loss (as is typical for transient global amnesia). She enjoyed full memory recovery within several hours but complained of a marked but tolerable headache. She was examined by a local neurologist and was discharged home. Two days later she was admitted to a tertiary-care hospital because of symptom progression. On admission, the patient was suffering from cephalea, nausea, and had meningeal signs. MRI of the brain and cervical spine showed SAH in the prepontine and premedullary cisterns and on the convexity. No vascular abnormalities in the craniocervical junction or the cervical area could be identified on the MRI (A–D). Digital subtraction angiography of the cranial vessels did not reveal any aneurysms.\nAs the symptoms diminished, she was discharged home with the presumptive diagnosis of pretruncal non-aneurysmal SAH. After a period of 2.5 months and in her 32nd week of pregnancy, she underwent an MRI examination for moderate back pain. This identified a spinal vascular lesion with a large ectatic venous aneurysm at the level of L1. Dilated perimedullary veins could be seen in the T10 to L5 range. Cesarean section was recommended, leading to the delivery of a healthy child in the 33rd week of pregnancy. Two days later, as she was recovering in the obstetric ward, the patient developed progressive lumbar pain and lower limb weakness along with partial urinary incontinence. A repeat MRI examination revealed a conus medullaris hemorrhage at the L1 level, accompanied by thoracic myelopathy from T10 inferiorly (A,B). The patient was admitted to our hospital and spinal angiography identified a perimedullary AVF (PMAVF) type B centered at the L1 level, supplied by a pedicle from the dilated anterior spinal artery (ASA) (C) and by smaller pedicles from both posterior spinal arteries (PSA) (D,E). A large venous aneurysm was also identified.\nThe patient made a near-complete spontaneous functional recovery within several days. Microsurgical treatment was considered to be of higher risk compared to endovascular intervention. Therefore, transfer to a specialized endovascular center was scheduled. However, before her transfer during a bowel movement, she rapidly developed quadriparesis, bulbar syndrome, and dyspnea, requiring urgent intubation. Computed tomography (CT) showed a brainstem hemorrhage (A,B) and extensive cervical and thoracic myelopathy appeared on MRI (C).\nThoracolumbar MRI did not reveal any change in the intraconal hemorrhage. Second spinal angiography to close the fistula was attempted but the AVF feeding pedicle from the ASA was found to have spontaneously occluded. Both PSA (that were still supplying the PMAVF) were not amenable to percutaneous intervention (A–C). Neurosurgical intervention was postponed due to the critical state of the patient. Within 3 days the patient was extubated and her brainstem function recovered. However, the patient had severe paresis of the right hand and lower limb paraplegia accompanied by urinary incontinence and decreased sensation under the level of C7.\nA multidisciplinary team recommended surgical revision as the last-line treatment in hopes of preventing further, potentially fatal hemorrhage. During surgery, subdural and intraconal hemorrhage were visualized and were partially evacuated. A fistula was identified and obliterated. There was an arterialized venous aneurysm distal to the fistula, which was found to be the source of the hemorrhage.\nOver the course of the following year, the patient’s neurological deficit improved slightly, allowing her to ambulate with a walker (Aminoff 4). However, her urinary incontinence did not improve, nor was there any improvement in her diminished sensation from the T10 level inferiorly. Postoperative spinal angiography revealed complete obliteration of the vascular lesion with no residual filling (D–F) An MRI showed improvement with respect to myelopathy. |
The first case was of a 55-year-old, previously healthy, male patient. He presented with vague abdominal pain and discomfort after meals. He had a history of heavy alcohol consumption and smoking. His symptoms began 4 months before admission. Initially, he underwent an upper GI endoscopy that revealed an H. pylori-positive gastritis. Thus, he was treated with proton pump inhibitors (PPIs) and antibiotic administration. Although his follow-up endoscopy showed remission of the initial findings, the symptoms remained. He was afterward referred to our department. His physical examination revealed no particular findings apart from mild pain during the left upper abdominal quadrant palpation.\nPatient's laboratory evaluation revealed elevated serum γ-GT: 135 U/L, LDH: 250 U/L, and glucose: 165 mg/dL. Also, CA 19.9 was slightly elevated at 45 U/L. The rest of the laboratory values were within normal values. His upper abdominal ultrasound showed a mass located at the tail of the pancreas measuring 10 cm, and the computed tomography scan that followed revealed a cystic mass at the tail of the pancreas, with a max diameter of 12 cm (Figure ). Following these findings, an abdominal magnetic resonance imaging scan confirmed the previous studies, with a well-defined solid cystic lesion, unable to further specify character of the tumors. Also, the patient underwent endoscopic ultrasound (EUS) that showed a large solid, heterogeneous well-circumscribed pancreatic cystic mass, but failed to aspirate content for cytology examination. As the EUS-FNA performed could not exclude a potentially malignant cystic lesion, the suspicion of a cystic tumor of the tail of the pancreas leads the multidisciplinary team to suggest surgical exploration. Laparotomy revealed a cystic mass of the tail of the pancreas measuring 12 cm. In addition to this, the splenic vein was occluded by the mass prior to its confluence with the superior mesenteric vein. As a result, distal pancreatectomy and splenectomy were performed.\nPathology report revealed a multiloculated cyst with cystic walls lined by keratinizing stratified squamous epithelium. Beneath the squamous lining epithelium, lymphoid tissue, plasma cells, and germinal centers were found, as well as multiple lymphoid follicles, without signs of atypia (Figure ). Lymphoepithelial cyst of the pancreas was the conclusive diagnosis. Furthermore, submersions of the epithelium were identified to compose smaller cysts filled with mature keratinized material and surrounded by lymphoid tissue, along with lipoid tissue and lipoid-necrotic cysts filled with macrophages and Langerhans giant cells. The cyst adhered to the pancreatic tail, where dense fibrous reaction with ipsilateral sclerosis and atrophy of the pancreatic parenchyma was present. Postoperative course was uneventful, and he was discharged on the 15th postoperative day. On his 6-month follow-up, he was relieved from his symptoms and no sign of recurrence was observed at his abdomen CT scan. |
A 53-year-old Caucasian female presented with a chief complaint of “twisting” right breast pain, associated with the development of yellow-green bruising on the breast. The patient waited approximately 7 to 8 months before obtaining a mammogram and ultrasound of the right breast, which revealed a right breast mass and right axillary adenopathy (Fig. ). A biopsy was significant for cancer within lymphatic vessels in the skin, and the breast tissue showed grade 2 invasive ductal carcinoma. The tumor was estrogen and progesterone receptor- negative (< 1% each) and HER2+ (3+ by immunohistochemistry). During the initial visit, the patient complained of a productive cough with scant sputum production and mid-back pain for 2 months. The patient suffered from fatigue, and experienced night sweats for the past year with no weight loss. The patient admitted to noticing a change in breast shape and size, but no nipple discharge. The initial physical examination was significant for morbidly obese body habitus. The left breast was large, free of masses, and non-tender. The right breast was significantly larger compared with the left, and tender with a rash. A palpable 2-cm axillary lymph node on the right was appreciated. A positron emission tomography (PET)/computed tomography (CT) scan revealed hypermetabolism within the anterior aspect of the right breast consistent with inflammatory carcinoma, right axillary and supraclavicular lymphadenopathy, and several skeletal metastatic lesions predominantly within the cervical and thoracic spine. The primary tumor, regional lymph node, and distant metastasis (TNM) staging was T4b, N1, M1.\nAs indicated for the treatment of metastatic inflammatory breast carcinoma in a premenopausal patient, treatment was initiated with doxorubicin and cyclophosphamide every 2 weeks for 4 cycles. After the first week of chemotherapy, monthly zoledronic acid was added to the treatment regimen due to the presence of bone metastases. Treatment was then followed by paclitaxel and trastuzumab weekly for 12 weeks.\nAt the 5-month follow-up visit, a repeat PET/CT scan was significant for a positive response to treatment: the abnormal hypermetabolism within the right breast, axillary and supraclavicular nodes, and within the skeleton had resolved. At the 6-month follow-up visit, the patient had completed the above chemotherapy followed by the paclitaxel plus trastuzumab treatment course. The patient underwent a bilateral mastectomy with reconstructive surgery. Post-operatively, the patient had residual 0.1 cm of focal invasive ductal carcinoma, intravascular carcinoma with deep resection margins, and isolated carcinoma cells in two axillary nodes. The patient was subsequently enrolled into the Phase III open-label clinical trial of trastuzumab plus lapatinib maintenance therapy (trastuzumab 6 mg/kg every 3 weeks plus lapatinib 1000 mg/day), and was randomized to receive the combination treatment.\nAt the 16-month follow-up visit, a CT scan of the abdomen, pelvis, and chest with intravenous contrast was significant for sclerotic changes within the lumbar spine, thoracic spine, and ribs, consistent with metastatic disease. Within 1 month later, after continued trastuzumab plus lapatinib treatment, a nuclear medicine whole-body bone scan revealed increased activity correlating with treated metastatic disease of the thoracic spine.\nSubsequent CT and bone scan imaging over the next 3 years reflected stable metastatic disease, consistent with healed skeletal metastatic disease, and was unchanged. The patient continued to be treated with trastuzumab plus lapatinib, and at the 4-year follow-up visit (while still on trastuzumab plus lapatinib), the most significant complaint from the patient was chronic diarrhea (which was controlled with diphenoxylate plus atropine tablets 3 times a day) and a single syncopal episode (a magnetic resonance imaging scan revealed no acute infarct, hemorrhage, or mass). |
A 37-year-old female involved in motor vehicle accident (MVA). On arrival to the emergency room (ER), her vital signs were normal including O2 saturation 100% on facemask of full flow of O2; 10 min later, her level of consciousness started to deteriorate and O2 saturation dropped, which required securing the airway and ventilation, and intubation trial was attempted twice but failed. On the 3rd time, frova airway intubating introducer (FAII) (COOK) with its metallic stiffening cannula [] was used and inserted into the airway and then tracheal tube slided over FAII, which was reported as successful intubation. Immediately after intubation, the patient started to have extensive bilateral subcutaneous emphysema and severe hypoxia. Chest X-ray showed extensive surgical emphysema and right side tension pneumothorax, which was not relieved by bilateral chest tube insertion. Further, there was massive air leak from the right chest tube which required the second chest tube to be inserted [], after which the patient O2 saturation improved temporarily; which allowed to proceed for computed tomography scan chest, which showed near complete tear of the origin of the right main bronchus. There was also a significant amount of surgical emphysema dispersing into the mediastinum and the pleural spaces causing pneumothorax and soft-tissue emphysema [Figure and ].\nFiberoptic bronchoscopy (FOB) performed in ER which showed a complete transection of the right main bronchus at the origin from the carina, and also, there was a laceration 4 cm in length on the right side of the lower trachea.\nImmediately, the patient transferred to the operating room (OR); the tracheal tube was directed to the left main bronchus guided by FOB for lung isolation, ventilation, and better visualization. Right thoracotomy performed, and exploration showed that the mediastinal pleura was intact and not injured but there was underneath it complete transaction or avulsion of the right main bronchus from the carinal origin, which extends 4 cm upward to the lower trachea. Complete and direct repair of both the right main bronchus and the tracheal laceration were performed. During the procedure check, FOB repeated few times which was satisfactory. The patient kept ventilated in the Intensive Care Unit (ICU) for ten days, and after extubation, she remained in stable condition where she was discharged home on the 20th postoperative day in good and stable condition.\nRoutine follow-up in the clinic and check FOB were repeated in 3rd, 6th, and 12th month postoperative, without any abnormality. |
Our patient is a 40-year-old Asian man with complaints of bleeding and discomfort in his anus of 2 months’ duration. He was an employee with average income who did not smoke tobacco or drink alcohol. He had no weight loss or urinary symptoms, and no substantial family history. He denied any significant medical or surgical history. His abdomen was soft, non-tender, and non-distended, with normoactive bowel sounds. In examination, a mass could be touched by finger tips. The mass was large and bleeding. In subsequent examinations, blood was detected in a stool sample. His vital signs were: blood pressure, 130.77 mm Hg; respiratory rate, 18 breaths/minute; heart rate, 83 beats/minute; and temperature within normal limits. Oxygen saturation was 98% on room air on admission. In colonoscopy, a large lobular tumor was diagnosed at 4 cm above the dentate line, which was suspicious for malignancy. Various samples were taken from the tumor. The rest of his large intestine did not show a clear pathologic lesion in the colonoscopy.\nHigh-grade adenocarcinoma was reported in pathological examinations. In subsequent diagnostic procedures, his carcinoembryonic antigen (CEA) level was normal. Computed tomography (CT) scans revealed that metastatic lesions were not detected in his liver, abdominal viscera, and chest. In CT scans with or without contrast, and magnetic resonance imaging (MRI) scanning, an ectopic kidney was detected incidentally on his right pelvis without any prior urinary symptoms. The left kidney was in its original location, and both kidneys were functional. Renal function tests provided normal results.\nIn subsequent investigations done by MRI scanning for staging the tumor, a pelvic rectum tumor was reported to be interfering with the T3 N1 mesorectal lymph nodes (Figs. , ). The case was discussed in a multidisciplinary cancer team; afterward, our patient was regarded as a candidate for neoadjuvant radiotherapy. He underwent 45 GY radiation in 25 fractions to the pelvis along with capecitabine. He underwent total mesorectal excision (TME) surgery to maintain the ectopic kidney 6 weeks later. After abdominal exploration, his abdominal viscera were examined. There was no metastatic lesion in his liver and abdomen (Fig. ).\nAfter mobilization of the left colon and the splenic flexure, and the closure of the inferior mesenteric artery (IMA), in the avascular plane, the mesorectum was separated from the fascia propria, and the mesorectal lymph nodes and hemorrhoidal vessels in the anterior and pelvic nerves were fully mobilized, and the distal rectum was removed by an appropriate margin (Fig. ).\nHis right kidney was completely inside the pelvis, and while the kidney was carefully protected by the retractor, an attempt was made to minimize the damage to the ectopic kidney because there was a possibility of damage to the pelvic nerve and nephrectomy.\nThe blood of the right kidney appeared to be supplied by the right superior iliac artery. During the surgery, hematuria occurred to our patient, which was resolved by hydrating him. Then, coloanal anastomosis and temporary ileostomy were performed on our patient. He was transferred to our intensive care unit (ICU). He underwent laparotomy again due to anastomosis leakage a week following the surgery. As a result, a colostomy was performed. Postoperatively, after the reappearance of symptoms, stabilization, and healing of the wounds, he was referred to medical oncology and started adjuvant chemotherapy with 5-fluorouracil, folinic acid, and oxaliplatin (FOLFOX). Follow-up testing (for a year) included routine medical history and physical examination (every 3–6 months), blood tests such as serum CEA, colonoscopy, and radiologic imaging. He was dissatisfied with the permanent colostomy after the end of the treatment. However, the satisfying result was that his kidney was preserved (Additional file ). Before the surgery, the potential risks and damage to his ectopic kidney and the possibility of its removal were explained to our patient and his consent was obtained. His general condition is appropriate after 1 year and his quality of life has been reported to be satisfying despite the permanent colostomy. |
A 78-year-old male with multiple comorbidities, including hypertension, hyperlipidemia, and type II diabetes mellitus, presented to our emergency setting with complaints of recurrent bouts of abdominal pain and fluctuating fevers for the previous two weeks. The patient reported that the pain is a new manifestation of a previously dull aching pain that had waxed and waned over the last decade. His description alluded to a pain that was sharp and intermittent with localization in the right upper quadrant. He could not attribute the intermittent nature of his predicament to any aggravating or relieving influences. The pain was associated with fluctuating low-grade fevers (99°F-100°F), anorexia, and an associated 13-pound weight loss, which culminated in a visit to our clinical setup.\nFurther interrogation disclosed that the patient underwent a laparoscopic cholecystectomy in 2003. The ensuing year was relatively pain-free but was followed by recurrent bouts of right upper quadrant pain, albeit less upsetting than his current presentation. He was subsequently diagnosed in 2005 with gallstone spillage. The patient chose conservative treatment for his abdominal pain, rather than invasive interventions, which included the administration of acetaminophen and non-steroidal anti-inflammatory drugs (NSAIDs). This treatment modality was sufficient for the duration of a decade. He presented to another medical facility with similar complaints of fever and abdominal pain in 2016. A computed tomography (CT) scan of his abdomen disclosed the presence of a necrotic phlegmon, which was subjected to aspiration. Its composition included a combination of fibrous material, granulation tissue, and inflammatory infiltrate. The aspiration provided considerable relief of symptoms and he was discharged on a gabapentin prescription that was well-tolerated and produced sustained amelioration of his pain, with only occasional wavering with respect to his baseline.\nThe initial assessment showed an elderly gentleman, who was alert and well-orientated but under considerable distress due to the abdominal pain and accompanying chills. He had a fever of 104°F, a heart rate of 120 beats per minute, a blood pressure of 95/70 mm Hg, and a respiratory rate of 19 per minute. An abdominal exam revealed a non-protuberant, soft, and tender abdomen with marked sensitivity in the right upper quadrant. He had perceptible bowel sounds, an absence of dullness on percussion, and a clear rectal vault. Pertinent initial laboratory investigations included an elevated white blood cell (WBC) count of 16,900/µL, C-reactive protein (CRP) of 6.5 mg/dL, and a random blood glucose (RBG) of 280 mg/dL. The patient subsequently underwent a whole body CT scan, which revealed a large 19-cm sub-diaphragmatic, right retroperitoneal abscess, which was inferior and posterior to the right hepatic lobe (Figure ).\nIn lieu of these findings, the patient was admitted and started on a combination of intravenous fluids, intravenous (IV) vancomycin and piperacillin-tazobactam due to an underlying suspicion of sepsis secondary to a hepatic and/or perihepatic liver abscess caused by a lack of gallstone retrieval following his cholecystectomy. He was subsequently subjected to a percutaneous CT-guided drain placement, which allowed for the evacuation of approximately 700 mL of grossly purulent material and provided prompt pain relief. The fluid sample was sent for a gram stain, culture, and a bilirubin assay. The gram stain of the abscess fluid revealed branching gram-positive rods concerning for Actinomyces, Nocardia, and Streptomyces; therefore, the patient remained on IV vancomycin and piperacillin-tazobactam, with the addition of trimethoprim-sulfamethoxazole (TMP-SMX) to the antibiotic regimen. Remarkably, the culture growth was positive for Propionibacterium and ampicillin-sulbactam was added to the list of antibiotics. Two days later, the patient was shifted to the intensive care unit (ICU) following unrelenting fever and persistent tachycardia, even after his abdominal drain placement was supplemented with vigorous antibiotic and antipyretic therapy. His ICU stay was complicated by episodes of dyspnea, tachypnea, and pleuritic chest pain, while a chest auscultation unveiled a decrease in breath sounds at the right basal region. A repeat CT scan confirmed the reduction in the size of the abscess and showed a new right-sided pleural effusion, which explained the patient’s breathing difficulties (Figure ).\nThe patient underwent an ultrasound-guided chest tube placement, which evacuated 10 mL of purulent fluid, followed by two doses of lytic therapy with tissue plasminogen activator (tPA) via the catheter. The following morning, he described an aggravation in his pleuritic chest pain, and an episode of shortness of breath, whereby, the oxygen saturation dipped to 90%. A new chest CT scan was ordered, which showed an interval increase in the right basilar opacity with a moderately sized pleural effusion (Figure ).\nHe was subsequently upgraded to a larger catheter via a CT-guided approach that evacuated a total of 900 mL of purulosanguinos fluid. The patient tolerated subsequent lytic therapies well and showed vast clinical improvement following this procedure, with a reduction in dyspnea, pleuritic chest pain, and fever, as well as a down-trending of his inflammatory markers.\nA repeat CT scan was performed on the 10th day of admission, which revealed a near-complete resolution of his right-sided pleural effusion, and a decrease in his right-sided retrohepatic intrabdominal abscess (Figure ).\nThe chest drain was removed and the patient was subsequently discharged with a transhepatic drainage catheter, daily intravenous ampicillin-sulbactam, and acetaminophen, as needed for pain control. The patient was shifted to oral amoxicillin-clavulanate a month after the resolution of his pain, fever, and stabilization of inflammatory markers. The transhepatic percutaneous drain was left in place in order to allow for the formation of a chronic drainage tract, with hopes of localizing the perpetrating gallstones at its base.\nTwo months after discharge, the patient underwent a procedure for upsizing the drain in order to accommodate for a choledochoscope into the abscess cavity and the maturing sinus tract. He subsequently underwent an abscess cavity endoscopy, whereby a catheter and scope were advanced into the cavity for exploration. An abscess catheter study was performed through the existing catheter and showed no filling defects, after which the catheter was removed and an endoscope was inserted for direct visualization of the main cavity, as well as the two “fingers” branching out of the abscess cavity. Two small stones were expelled after flushing the main cavity multiple times. Following the expulsion of the gallstones, a replacement catheter was advanced into the cavity and a second catheter study was performed to rule out the possibility of any other stones. The patient tolerated this intervention well and had no further drainage from the intra-abdominal drain.\nFollowing a lack of drain output and resolution of symptoms after his last endoscopic procedure a month ago, the patient's intra-abdominal drain was removed. The patient is currently stable and is being followed as an outpatient to date. |
A 21 year old nulliparous female presented to the emergency department (ED) with the complaint of right lower quadrant abdominal pain. During her ED course, she admitted that her real reason for presentation was for examination of a mass that had been growing on her right labium. Following a brief physical examination by the ED physician, the gynecology team was consulted and verification was made of a right labial mass. The patient admitted that she first noticed a marble-sized “bump” on her right labium about six months earlier. The bump increased in size over the course of the six months until its current size on presentation. She reported being too embarrassed and afraid to address the growth but due to the polyp’s burdensome size she was forced to present for evaluation. She was on day two of her menstrual cycle and denied any nausea, vomiting or any constitutional symptoms. The patient denied any significant medical or surgical history. She denied any history of sexually transmitted disease or gynecology-related surgery. She reported a history of regular menstrual cycles with a menstrual index as follows - menarche at age 10, 30 day cycle intervals and 4 to 6 day cycle lengths with moderate flow. She had not been sexually active for over a year and was on no contraception. She was a non-smoker and denied alcohol or drug use. Her physical exam was remarkable only for a large, non-tender, skin colored, grapefruit-sized ulcerating pedunculated mass extending from the right labium majus. Transvaginal ultrasound showed normal anatomy of the uterus and ovaries but also described a broad-based encapsulated soft tissue mass of the right labium at the level of the clitoris. The patient was discharged home for follow up with outpatient Gynecology-Oncology. Physical exam by the oncologist again verified a right labial pedunculated mass. The stalk was noted to be vascular with an area of ulceration over the serosal surface of the polyp. There was no increase in the size of the mass with valsalva. An in-office procedure to amputate the polyp was completed at the oncologist’s office. The base of the mass was infiltrated with Xylocaine, 3 large Kelly clamps were placed across the base and the mass was excised. The pedicle was ligated with 0 Vicryl suture and adequate hemostasis was obtained. The patient received Percocet for pain control with instructions for a 1- week follow up appointment. At her follow up appointment the vulva was noted to be healing well and the stalk was retracted. The pathology report returned as a giant fibroepithelial stromal polyp and she was advised to return to her routine gynecologist for continued surveillance. |
A 74-year-old female patient presented with a locally advanced tumor of the proximal pancreatic body. The patient showed no signs of obstructive jaundice and did not require biliary stenting. Initially, her tumor was noted to involve the major visceral vessels, including the celiac artery, portal vein, and splenic artery and vein (). The tumor was deemed unresectable and the patient underwent extensive chemotherapy with multiple cycles of gemcitabine, Abraxane, 5-fluorouracil, Alloxantin, Avastin, and Xeloda. After showing a favorable response, she was referred for surgical re-evaluation. The post neoadjuvant chemotherapy CT scan () showed a poorly defined infiltrative pancreatic neck and body mass measuring ∼25 × 15 mm with ill-defined soft tissue encasing the proximal splenic artery, common hepatic artery (CHA), distal celiac axis, and superior mesenteric artery (SMA). The mass also partially encased the portal vein and superior mesenteric vein (SMV). There was no radiologic evidence of hepatic metastasis and there was mild pancreatic duct dilatation. Pre-treatment and post-treatment serum CA 19–9 levels were 46 and 9, respectively. The patient did not receive any additional studies to assess GDA flow or the need for preoperative coiling/embolization. The absolute need for resection of the celiac axis was not determined until the time of the operation. The patient's functional status, perioperative risk, and likelihood of response were weighed and she was determined to be a candidate for an attempt at a modified Appleby procedure. After being properly informed of her various treatment options, she elected to undergo the operation.\nExploration of the duodenum and pancreas revealed a firm mass in the body of the pancreas with a soft pancreatic neck. No gross evidence of metastatic disease was present. There was a benign appearing lesion in the superior aspect of liver segment II, which was excised in its entirety and confirmed to be benign. Cholecystectomy was performed, followed by Kocherization of the duodenum and dissection of the pancreas and major vessels. We were fastidious in preserving the GDA. The splenic artery was controlled distal to the tumor, leaving a normal CHA pulse. The spleen and pancreatic body and tail were then serially elevated out of the retroperitoneum. The aorta was exposed, taking down the diaphragmatic crura. The celiac artery was identified at its origin, tied, divided, and oversewn with 5–0 polypropylene suture. Abnormal soft tissue was palpated along the proximal CHA. In light of this vessel involvement, we divided the distal CHA near the GDA. We then dissected the specimen off the SMA successfully and divided the pancreatic neck.\nThe inferior mesenteric vein was then clamped and tied. The inflamed pancreatic neck and proximal body were then dissected free from the right lateral aspect of the SMV and portal vein. The splenic vein was taken flush with the SMV, and its stump was oversewn, leaving good forward flow of the SMV to the portal vein. The proper hepatic artery (PHA) and the GDA were preserved throughout the resection. A Doppler ultrasound probe was used to test the GDA and PHA. Both arteries had adequate signal and in fact, the PHA had a faint palpable pulse. The liver parenchyma was also found to have a strong arterial signal. The specimen had two short stitches and purple dye placed at the neck margin and the rest of the specimen was inked per our Jefferson protocol.\nPathology of the resected specimen showed ductal adenocarcinoma with marked treatment effects with invasion of tumor into the peripancreatic soft tissue. The excision margins were free of neoplasia and the specimen had no regional lymph node metastasis (0/26). The tumor was within 2.0 cm from the pancreatic resection margin and within 0.1 cm of the circumferential margin in the posterior peripancreatic tissue. The maximum diameter of the tumor was 3.0 cm in size.\nIn the initial postoperative period, the patient had a transient transaminitis. A postoperative hepatic vascular ultrasound showed good hepatic arterial flow through the PHA and the transaminases normalized. The patient was discharged on postoperative day 9. She remains well without evidence of disease 7 months postoperatively, and has elected to receive no further chemotherapy. |
A 30-year-old female patient who was a daily wages laborer was referred to the Department of Oral and Maxillofacial Surgery with a chief complaint of upper lip swelling, along with periods of exacerbation and remission, for the past 7 to 8 months. The patient gave a history of swelling which was sudden in onset with no history of trauma, pain or burning sensation. The swelling was noticed 8 months back, with discomfort in mouth opening for a year. She visited a local general practitioner for the same who prescribed medications for the swelling (topical steroids and antibiotics) which provided temporary relief. The swelling recurred again to which she was referred to our hospital for its management. The patient had no previous medical history and was not aware of any food or drug allergies.\nExtraoral examination revealed a diffuse swelling of the upper lip [] with no palpable lymph nodes. The swelling was nontender, noncompressible soft to firm in consistency, with no evidence of ulceration, fissuration, sinus or discharge, bruit or pulsations. There was no paralysis of facial muscles. Intraoral examination revealed slightly erythematous gingiva with granular appearance with respect to maxillary and mandibular arches. Bleeding on probing or tooth mobility was absent. The left buccal mucosa showed cobblestone appearance in the pterygomandibular raphae region extending anteriorly till the first molar, along the occlusal line []. There were no evident changes in the tongue and other parts of the mucosa. Based on clinical features, a provisional diagnosis of OFG was given. The other conditions that were considered under differential diagnosis included angioneurotic edema, Crohn's disease, tuberculosis, sarcoidosis, cheilitis granulomatosa, foreign-body reaction, fungal infections and contact allergy. We ruled out Melkersson–Rosenthal Syndrome because there was no facial paralysis and the tongue was clinically normal. Since there were no signs of anemia or symptoms suggestive of Crohn's disease, comprehensive gastrointestinal investigations were not justified in this case. The absolute eosinophil count was 400/mm3 suggestive of an allergic etiology and erythrocyte sedimentation rate was 35 mm in 1 h. Incisional biopsy of the upper lip was obtained for histopathologic examination which revealed an overlying parakeratinized stratified squamous epithelium with underlying stroma showing areas of vascularity along with circumscribed aggregates of noncaseating granulomas that composed of epithelioid histiocytes and Langhans-type giant cells with peripheral lymphocytes and plasma cells suggestive of a granulomatous lesion [Figure and ]. Staining for acid-fast bacilli (AFB) and periodic acid–Schiff (PAS) yielded negative results.\nBased on clinical history, laboratory investigations and histopathological findings, a confirmatory diagnosis of OFG was given. A change of toothpaste was recommended suspecting an allergy to some component in it as she was unaware of any other food/drug or material allergies. The tablets levocetirizine and topical corticosteroid were prescribed. The swelling of lips and the cobblestone appearance were noticeably reduced after the first follow-up. Subsequent visits by the patient revealed complete remission of the lesions with no recurrence. |
A seven-year-old girl was burnt by boiling water at home in a rural place in the Northwest of Iran. The burnt area was on the right elbow. Her mother left home to take a burn ointment from the neighbor and rubbed it over the burnt area. An hour later, the mother brought the girl to the local city hospital with an impaired level of consciousness and difficulty breathing. The family provided no more history than the burn. A general practitioner and afterward, a pediatrician visited the patient. She was unconscious without responding to any painful stimuli, had pinpoint pupils and apneustic respirations (respiratory rate: 6 per minute). Other vital signs were stable (pulse rate 90 bpm, blood pressure: 90/60 mmHg, and body temperature 36.5 °C). An oval shape second-degree burnt area with the dimensions of 3 cm × 4 cm (approximately 0.25% of the body surface area) was seen over the posterolateral aspect of the right elbow covered with a burn ointment (Zinc oxide 30% plus Fish oil 50%). With a clinical suspicion of opioid poisoning, intravenous (IV) naloxone 0.2 mg was injected four times at 2 to 5 minutes intervals (total dose: 0.8 mg), and the level of consciousness of the patient was improved. The burn site was washed using normal saline, and the burn dressing resumed. Nonetheless, considering other differential diagnoses, the essential laboratory examinations, including a brain computed tomography (CT) scan without contrast, and a lumbar puncture were performed. Then, the patient was referred to a tertiary care pediatric hospital located in the center of the province for further management.\nFour hours later, the patient arrived at the destination hospital emergency room, and she was immediately admitted to the pediatric intensive care unit. A pediatric resident and a toxicologist visited the patient. She was in a light coma (just poorly respond to painful stimuli) and had meiotic pupils and apneustic respirations. Her level of consciousness and breathing became better after receiving a bolus of 0.8 mg IV naloxone. Other treatments for the patient included: fluid therapy, continuous naloxone infusion (0.01 mg/kg/hour for 24 hours), ranitidine (50 mg IV every 12 hours), ceftriaxone (900 mg IV every 12 hours), and topical silver sulfadiazine 1% ointment (every 12 hours on the burnt area).\nLaboratory studies of the patient are summarized in . Furthermore, a chest x-ray and a brain CT scan were unremarkable.\nInitially, clinicians were unable to obtain any reliable information from the patient’s parents about the circumstances of intoxication. Further investigation revealed that after burning, the mother left home to seek for a burn ointment from the neighbors, and the patient was left alone with her heroin-dependent father. By observing the agitation of his daughter, the father sprinkled some heroin powder over the burnt area. Then the mother, unaware of everything, rubbed the burn ointment on the burn. Heroin gradually penetrated the damaged skin and led to the unintentional poisoning of the child. On the third day of hospitalization, the patient was discharged in good condition without any complications. |
A 62 years old male presented with recurrent SCC of the left nasal bulbar conjunctiva. The patient's previous medical history detailed multiple interventions at this site. Initially, the diagnosis of a left nasal pterygium (a wing-shaped growth that starts on the conjunctiva and can spread across the limbus to the cornea affecting visual function) was made 3 years prior to the reported presentation, and was surgically removed with local excision. The lesion recurred 1-year post-surgery, and was subsequently managed surgically with a wide local excision. At this time, pathology revealed positive margins for SCC. A further recurrence 6 months later was treated with Plaque Therapy to a dose of 50 Gy in 5 fractions. Subsequently, the lesion was re-excised with adjuvant cryotherapy 7 months later.\nOn presentation 3 months post-cryotherapy, a recurrent lesion (10 × 5 × 2 mm) was detected on the nasal bulbar conjunctiva (Fig. ). The patient declined enucleation following this SCC recurrence. Subsequently, multidisciplinary consultation offered SXRT as an alternative treatment option (Fig. ).\nThe patient also presented with a history of Crohns Colitis (treated with immunotherapy) and a previous excision of a non-melanoma skin cancer from the right temple, and a subsequent skin graft of the area. The patient was on no other medications and had no allergies.\nThe patient was prescribed a dose of 48.4 Gy in 22 fractions, to a depth of 3 mm. This resulted in a skin surface dose of 54.4 Gy. A 2.0 cm diameter direct applicator was used with a 3 mm margin on the treatment region (Fig. ). To allow for an acceptable treatment margin that encompases the target volume and accounts for set up variability, the left inner canthus and tear duct were included in the treatment field. A single en-face beam of 2.0 mm Aluminium energy (2 mm Al) was used to meet the previously articulated dose prescription. The risk of ulceration, scleral perforation, ischemia and nasolacrimal duct obstruction was explained to the patient. This risk was further exacerbated in this case due to re-irradiation of the previously treated area, and a particularly sensitive region post-cryotherapy, surgery and plaque therapy. Topical anaesthesia (Tetracaine eyedrops) was administered to the left eye prior to retractor positioning, to ensure eyelids remained open throughout SXRT delivery. After positioning of the retractors the patient was instructed to maintain gaze on a target positioned to the left side of the room, and the eye was monitored throughout treatment delivery by video surveillance. Lubricating eye drops were prescribed for use prior to treatment administration to prevent drying of the ocular surface, and for the patients self-use between fractions to relieve dry eye symptoms. Total set up and treatment time for this patient was approximately 20 min, with the retractors in place on average less than 10 min. The anaesthetic drops and ocular surface lubricants induced limited sensation and the patient found it mildly uncomfortable. As a precaution, the patient was informed to avoid contact with the anaesthetised eye and wear a protective patch for 60 min post-treatment delivery daily. |
A 30-year-old non-smoker male, carpenter by occupation was detected to have end-stage renal disease due to chronic glomerulonephritis in 2006. He remained on intermittent hemodialysis till he received a renal allograft from his 50 years old mother in August 2008. He was not given induction therapy. He received triple immunosuppression that included oral prednisolone, mycophenolate mofetil and tacrolimus in therapeutic doses. Due to financial constraints, mycophenolate mofetil was replaced with oral azathioprine 100 mg a day after 3 months of renal transplantation. He maintained normal graft function. Four years after transplant, he presented with hoarseness of voice of 2 months duration. This was insidious in onset, progressively increasing and was not associated with neck pain, voice fatigue and cough. There was no relief with voice rest. There was no history of fever, hemoptysis and weight loss.\nHis general physical and systemic examination was essentially unremarkable. There was no tenderness or bruit over the graft kidney. The local examination of the nose, throat and ear did not show any gross abnormality. Flexible fiberoptic laryngoscopy showed an exophytic growth on the vocal process of the left arytenoid, extending anteriorly till the anterior commissure. The growth was excised under general anaesthesia and the base fulgurated. The tissue was sent for histopathological and microbiological examination, to evaluate for neoplastic and fungal pathology.\nOn investigations, the complete blood count (CBC) showed mild anemia with hemoglobin of 12.5 gm/dl and normal total and differential leukocyte and platelet counts. Urine examination showed absence of proteinuria and hematuria. The blood biochemistry revealed blood urea and serum creatinine of 40 mg/dl and 1.5 mg/dl respectively. His serology tests for hepatitis B surface antigen and antibodies to hepatitis C virus, Human immunodeficiency virus and cytomegalovirus were negative. Chest x-ray was normal. Biopsy from laryngeal mass lesion exhibited a polypoidal appearance lined by stratified squamous epithelium underneath which sheets of histiocytes containing numerous spores of cryptococcus of variable sizes were seen (). The PAS stain highlighted the fungus spores (). While, clinically there were no signs and symptoms of meningitis, the cerebrospinal fluid (CSF) examination was done that was negative for cryptococcal meningitis. Patient was treated with oral fluconazole 400 mg per day for 6 months and recovered completely. Since fluconazole has interaction with calcineurin inhibitors, the dose of tacrolimus was decreased to 1 mg twice a day maintaining the blood levels in the therapeutic range. |
The second patient is a 72-year-old woman with a past medical history including hypertension, type II diabetes, and obesity, who presented with weight loss and steatorrhea. As her symptoms were suggestive of pancreatic exocrine insufficiency, and with the associated weight loss, she underwent a CT scan for additional evaluation. This demonstrated a suspicious 2.5-cm complex cystic pancreatic head mass with associated pancreatic gland atrophy and a dilated pancreatic duct. She was also noted to have fatty infiltration of the liver (although not overt cirrhosis) and ascites.\nShe was then referred for further evaluation. Magnetic resonance imaging (MRI) and magnetic resonance cholangiopancreatography demonstrated diffuse dilation of the pancreatic duct up to 7.5 mm with an associated cystic mass in the head of the pancreas, suggestive of main duct IPMN (). EUS was attempted, but was unable to be completed due to severe tortuosity of her esophagus. Positron emission tomography (PET) demonstrated heterogeneous metabolic activity within the pancreatic uncinate process and to a lesser degree within the body and tail. Additional workup included a paracentesis to evaluate the ascites, which was unremarkable and ultimately deemed secondary to poor nutritional status from her exocrine insufficiency. Laboratory results were within normal limits with the exception of tumor markers and a mildly low albumin (3.4 gm/dL). Carcinoembryonic antigen was elevated at 6.0 ng/mL, and CA 19-9 was elevated at 46.7 U/mL. She was placed on pancrelipase, which resolved her symptoms of exocrine insufficiency and helped improve her nutrition.\nDue to the concern for main duct IPMN, she was referred for surgical resection. Again, it was unclear if the diffuse dilation of the pancreatic duct was secondary to involvement by main duct IPMN or due to proximal pancreatic duct obstruction. The patient was planned for a Whipple procedure with possible total pancreatectomy if high-grade dysplasia was noted intraoperatively at the resection margin. Upon transection of the pancreatic neck, IPMN with focal high-grade dysplasia was indeed noted at the margin. Due to the abnormal preoperative PET scan, in addition to the high-grade dysplasia at the margin, a completion pancreatectomy was performed. Due to her poor nutritional status preoperatively, a gastrojejunal feeding tube was placed at the time of the operation. She had an unremarkable postoperative course and was discharged with feeding tube supplementation.\nHer final pathology demonstrated a 1.1 cm well-differentiated mucinous colloid carcinoma arising in a background of IPMN (intestinal type) with foci of high-grade dysplasia and intermediate grade dysplasia throughout the remaining pancreatic duct (). Staining of the tumor was positive for MUC1, MUC2, and MUC5. All margins were negative. Zero of 24 lymph nodes was involved with tumor. Her final pathologic staging was pT2N0Mx. She was referred to medical oncology for discussion of adjuvant therapy and will be initiating gemcitabine adjuvant chemotherapy in the near future. |
A 49-year-old previously healthy woman with no history of trauma presented with intermittent gross hematuria and mild left flank pain of five days' duration. The patient described that her intermittent pain was aggravated by postural change and it gradually waned. The patient had no apparent respiratory, gastrointestinal or voiding symptoms. Her past medical history including hypertension, atherosclerosis and congenital diseases was non-contributory. She was afebrile and her physical examination findings were almost normal. Her blood pressure was 182/66mmHg. Urinalysis demonstrated 20–29 red blood cells per high-power field, 10–19 hyaline casts per field of vision and a sterile culture. Urine cytology was negative for malignancy. Laboratory examinations were within normal limits, except for the elevated C-reactive protein levels of 4.64mg/dL (normal, <0.30mg/dL). Ultrasonography and excretory urography did not reveal any definite structural abnormality in the urinary tract.\nWhen the patient visited our outpatient clinic the next day, urinalysis showed dark urine containing numerous red blood cells without dysmorphic changes. Cystoscopy revealed bleeding from the left ureteral orifice. Contrast-enhanced computed tomography (CT) demonstrated aortic dissection originating just distal to the left subclavian artery and continuing into the bilateral common iliac arteries with a re-entry site in the abdominal aorta, compatible with acute Type B aortic dissection. No apparent branch vessel ischemia was observed. The patient was transferred to the intensive care unit in order to provide medical treatment, and prompt attention was paid to stabilizing the patient by reducing blood pressure.\nOn the retrospective review of CT scans, the arterial phase demonstrated the left renal artery originating from the true lumen of the aorta and no apparent renal ischemia []. The venous phase showed compression of LRV between the enlarged aorta and SMA with distal dilatation []. A prominent left gonadal vein, implicating formation of collateral circulation, was also observed. Three-dimensional CT imaging demonstrated the compressed LRV and development of collaterals []. After the possibility of more common renal conditions such as tumors, urolithiasis and other medical renal diseases was excluded, we made a final diagnosis of NCP due to acute aortic dissection, explaining the gross hematuria. Invasive venography to measure the pressure gradient between LRV and the inferior vena cava was not performed.\nDuring the following month, her clinical symptoms subsided and urinalysis abnormalities normalized. However, one month after the diagnosis, CT findings of LRV compression and left gonadal vein dilatation remained unchanged. |
A 62-year-old woman presented with a one-month history of sudden painless visual loss in the right eye. On examination, best corrected visual acuity (BCVA) was 20/20 in both eyes. Intraocular pressure was 21 mmHg in both eyes. Dilated funduscopic examination in the right eye revealed retinal emboli inferior to the optic disc obstructing a small arteriole associated with retinal ischemia (Figures and ). The left eye was unremarkable. There was no prior past ocular history. The patient had hypertension and hypercholesterolemia and had an extensive smoking history.\nFluorescein angiography revealed delayed retinal perfusion along the inferior arcade in the right eye (Figures and ). Optical coherence tomography showed normal foveal contour with inner retinal ischemia and thickening, consistent with an acute inferotemporal branch retinal artery occlusion (). The patient was started on Latanoprost at nighttime in the right eye to lower the intraocular pressure in hopes to increase reperfusion of the retina. An extensive cardiovascular workup was done, and significant carotid artery stenosis of less than 70% was found. She was started on plavix and aspirin by her cardiologist. The patient continued with BCVA 20/20 OU vision and persistent highly refractile peripapillary emboli; retinal ischemia resolved. Her OCT showed inner retinal atrophy (Figures and ).\nUnexpectedly, the patient presented with a three-day history of sudden painless visual loss OD a year and a half after her initial presentation. BCVA was counting fingers (CF) in the right eye. Fundus exam revealed new superotemporal retinal ischemia associated with two new emboli. OCT demonstrated thickening and hyperreflectivity of the inner retinal layers consistent with an acute BRAO OD (). The patient refused fluorescein angiography at that time. Urgent workup revealed worsening stenosis to 80% in her right common carotid artery. The vascular surgery referral prompted a right carotid endarterectomy with a carotid stent placement approximately two months following her presentation with acute vision loss.\nAfter endarterectomy, vision in the right eye improved from counting fingers to 20/200 and 20/250 at 2 months and 6 months, respectively. Postoperatively, retinal whitening resolved and reduced intraretinal edema was noticed (). One year after endarterectomy, visual acuity was 20/30. The retinal swelling had resolved (). |
Mr. R, a 21-year-old gentleman from South India, who worked in a cellphone shop, was brought to the hospital with sudden onset breathlessness gradually worsening for the last 2 days. He also complained of generalized weakness and muscle pains. There was no premorbid illness or history of drug intake. On arrival to the hospital, he was found to have tachypnea, tachycardia, and was in hypotension. Chest examination revealed bilateral crepitations. Cardiovascular system did not reveal any murmurs. Abdomen and central nervous system (CNS) examination did not reveal any abnormality. His oxygen saturation was low, and the arterial blood gas analysis (ABG) was done which revealed type 1 respiratory failure with severe metabolic acidosis and lactic acidosis [].\nThe patient was immediately intubated and given fluid resuscitation and shifted to the medical intensive care unit (ICU). His initial hemogram and biochemical investigations revealed leukocytosis, with acute renal failure and deranged liver function tests [].\nChest radiograph revealed bilateral lower zone haziness and electrocardiogram revealed sinus tachycardia. Cardiac enzymes and echocardiogram did not reveal any abnormality.\nIn the ICU, he was investigated for his oliguric renal failure. His creatinine phosphokinase (CPK) was high, which was suggestive of rhabdomyolysis. In view of the worsening renal function and oliguria with severe metabolic acidosis, he was dialysed after consultation with the nephrology team. He continued to require daily dialysis in the ICU. His ventilatory parameters improved gradually, and he was extubated on the 7th day of the admission and shifted to the ward on the 9th day.\nHe was worked up extensively for his renal failure and rhabdomyolysis, including a muscle biopsy which was inconclusive. After ruling out infectious and autoimmune causes, a heavy metal screen was requested. The heavy metals were checked in the patients' blood by inductively coupled plasma mass spectrometer (ICP-MS), which can accurately detect the concentration of heavy metals in the biological materials. The abovementioned test revealed high levels of chromium in the blood (338 microgram/l) (normal range <10 microgram/L). Hence, a diagnosis of chromium toxicity was made. The exact source of chromium was not clear from history. On further questioning, the patient mentioned that he works in a mobile phone shop and has been handling mobile phone batteries for more than 2 years checking the power by putting the wires on his mouth. He improved gradually in the ward and required a total of 13 dialysis. He was in the hospital for a duration of 27 days and was followed-up in the outpatient department [].\nHe was discharged home in a stable condition. On follow up, his repeat chromium levels were 5.8 microgram/L (normal range: <10 microgram/L) and renal function test were in the normal range. The patient was educated regarding the possible source of his exposure and the related health implications. |
A 6-year-old, 25 kg boy was scheduled for strabismus surgery in both eyes. Neither the patient nor his family had any history of neuromuscular disease or a special family history. He had not previously received general anesthesia. The preoperative laboratory examinations were within the normal values. The patient received atropine 0.25 mg IM for premedication 30 min before induction. The preoperative vital signs were blood pressure: 90/50 mmHg, heart rate: 92 beats/min, respiratory rate: 24/min and axillary temperature: 36.4℃. The patient received ketamine 50 mg IV for sedation before induction in the waiting room.\nAnesthesia was induced with sevoflurane 2.5 vol% by mask ventilation in a mixture of nitrous oxide and oxygen (FiO2 0.5). Fifteen mg of rocuronium bromide was injected during induction. About 2 min after injection of rocuronium bromide, a size 5.0 cuffed endotracheal tube was inserted without any difficulty under direct laryngoscopy. Then 1 min after intubation, the heart rate of patient was increased from 160 to 195 beats/min. At first, the tachycardia was considered to be due to stimulation by the tracheal intubation, but the end tidal carbon dioxide concentration (ETCO2) was concurrently increased from 35 to 65 mmHg within 5 min. The oral temperature was increased to 38.9℃ within 5 min after induction. No significant changes in the muscle tone and skin appearance were noted. We suspected MH, so we stopped using sevoflurane and N2O. The patient was hyperventilated with 100% O2 through a new anesthetic circuit, and consequently the ETCO2 was decreased to 45 mmHg. The patient was administered midazolam 1 mg and sufentanyl 15 µg intravenously for sedation after discontinuing the sevoflurane and we started propofol infusion as maintenance because propofol is known to be a safe anesthetic agent in patients with MH.\nFor decreasing the body temperature, active cooling was immediately initiated by ice water massage and applying ice packs on the chest, back and axillary area. Arterial cannulation was done to continuously monitor the blood pressure and for the arterial blood gas analysis. This was done via external jugular cannulation for rapid infusion of cold IV fluid and central administration of drugs for resuscitation if it was need, and we placed a Foley catheter for checking the hour urine output. The arterial blood gas analysis was pH: 7.294, PaCO2: 41.2 mmHg, PaO2: 435.2 mmHg and the base excess: -10.1 mM/L. The myoglobin in the serum was normal and the urine was negative for myoglobin. The creatine phosphokinase and potassium levels in the serum were normal. We administered 8.4% sodium bicarbonate 10 ml for correcting the acidosis. Thirty minutes min after anesthetic induction, the patient showed an oral temperature of 38℃, a pulse of 160 beats/min, a blood pressure of 140/70 and we detected newly occurring ventricular premature beats (4-5/min) on the echocardiogram. So, we decided to stop the general anesthesia and surgery and administer dantrolene. But we didn't have dantrolene, so we requested dantrolene from the Korea orphan drug center.\nApproximately 90 min after the onset of MH, the patient recovered consciousness and the tracheal tube was extubated. The patient was transferred to the intensive care unit for further observation. About 120 min after onset of MH, we received the dantrolene. The patient showed an oral temperature of 37.5℃, a heart rate of 140 beats/min and a blood pressure of 140/80 mmHg, and he maintained the ventricular premature beats (4-5/min). The arterial blood gas analysis showed a pH of 7.390, a PaCO2 of 38.0 mmHg, a PaO2 of 89.5 mmHg and a base excess of -2.5 mM/L.\nDantrolene 25 mg was administered intravenously. Thereafter, the arrhythmia disappeared, and the patient maintained an oral temperature of 36.5-37.4℃ and a normal blood pressure and heart rate. But about 2 h after the administration of dantrolene, the patient again showed arrhythmia (ventricular premature beats) and an increased oral temperature of 38℃, a blood pressure of 130/100 mmHg and a heart rate of 130 beats/min. Dantrolene 25 mg was administered again. After the second administration of dantrolene, the arrhythmia, oral temperature, heart rate and blood pressure were all normalized, and the laboratory data and arterial blood gas analysis were normalized 2 days after anesthesia induction.\nFurther evaluations for MH were refused by the patient's parents. The patient was discharged from the hospital four days after the onset of MH without any problem. |
We present the case of a 60 year-old male smoker with a history of chronic obstructive pulmonary disease (COPD) who presented with a two-week history of increasing shortness of breath and occasional wheezing. Review of systems was otherwise negative. Past medical history was remarkable for mild COPD, but the patient had otherwise been free of health problems. Physical examination of the chest revealed clear lung sounds bilaterally, and no other abnormal physical findings were evident. Chest x-ray demonstrated a right pneumothorax, and a subsequent CT scan confirmed a right pneumothorax (less than 10% of lung volume), a small right pleural effusion, and two large blebs in the right lung. No mass lesions were apparent. A nuclear medicine lung perfusion and ventilation scan showed retention of radiotracer consistent with COPD and heterogeneous patchy areas of perfusion, most likely related to air-trapping. It was decided to procede with surgical exploration and possible excision of the patient's emphysematous blebs. A right mini-thoracotomy was performed, and a large bleb was readily identified within the anterior chest, which was resected. A separate aarea of possible abscess was noted in the right apex, which on dissection by the surgeon contained yellow viscous fluid. Otherwise, no mass lesions or findings suspicious for carcinoma were encountered.\nHistologic examination of the patient's bullous resection revealed emphysematous changes and mild subpleural fibrosis. Additionally, multiple glandular structures were noted within the pleural tissue (Figure ). These structures were composed of plump epithelioid cells with mild nuclear enlargement, fine chromatin, and inconspicuous nucleoli. No mitotic activity was evident. The glands were surrounded by fibrovascular tissue and showed no evidence of vascular or lymphatic invasion. The majority consisted of irregular small acinar structures, and there was no evidence of cribriforming or formation of solid structures. Immunohistochemical stains revealed positivity for TTF-1 (Figure ) and CK7 (Figure ), consistent with respiratory epithelial origin. Notably, calretenin (Figure ) was negative, arguing against mesothelial derivation. Stains for CK20 (Figure ) and CEA were also negative. The separately resected abscess showed fibrosis, subpleural bronchiectasis, and granulomata. There was no evidence of malignancy in any of the examined tissue.\nSix months after resection, the patient is in stable health, with no clinical or radiologic evidence of bronchogenic carcinoma. His respiratory status remains compromised secondary to chronic lung disease. We feel that the identified epithelial inclusions represent a benign process, likely associated with the background of chronic obstructive lung disease. |
The patient was a 13-day-old male infant 1,350 g at birth, who was born by cesarean delivery. Pulmonary surfactant was administered via an endotracheal tube twice because of respiratory distress syndrome and the newborn was provided with mechanical ventilation for 35 hours. The patient received oxygen by hood until the sixth day of life, and received 2.5 mg injections of aminophylline every 12 hours because of apnea diagnosed on the eighth day of life. Surgery was planned due to jejunoileal obstruction, diagnosed by a gastrograffin enema. Gavage feeding was discontinued and bile excretion was noted.\nBefore the surgery, the vital signs were stable. There were no abnormalities on the ECG or chest X-ray. On the day of surgery, the weight was 1,390 g and there was no premedication before the anesthesia. Standard monitoring (electrocardiogram, noninvasive blood pressure and peripheral pulse oxygen saturation) was performed. Injection of 500 ml of 10% dextrose water + NaCl 15 mEq + heparin 500 IU 8 ml/hr was carried out via the internal jugular vein using a 24 gauge catheter and was maintained at 10-0 ml/hr during the surgery. An 8 vol% of sevoflurane was administered and then maintained with a 3 vol% of sevoflurane. Fresh gas flow was maintained with O2 1 L/min and air 4 L/min. Injection of 1 mg of rocuronium was administered because there was no difficulty with mask ventilation.\nTwo minutes later, intubation with a 2.0 mm ID (internal diameter) endotracheal tube was attempted. After the endotracheal intubation, the patient could not be ventilated. The EtCO2 was measured at 0 mmHg. Therefore, the endotracheal tube was removed and mask ventilation was provided. The tube was checked and intubation attempted again with the same endotracheal tube, two minutes later. However, the second attempt failed and mask ventilation was again provided. Endotracheal intubation was attempted by a different anesthesiologist, however, all attempts failed.\nTracheal intubation with a 2.5 mm ID endotracheal tube was attempted. After the intubation, ventilation was successful and breath sounds present bilaterally. The tube was fixed at 7 cm the tube was cut from 12 cm to reduce the dead space. After cutting the tube, ventilation failed and the EtCO2 was 0 mmHg. There was no change in the depth; ventilation could not be provided and there were no breath sounds. The endotracheal tube was removed and mask ventilation was provided again. Intubation with the same endotracheal tube after two minutes was tried again. The ventilation was successful and breathing sounds could be heard in the lungs bilaterally. The tube was fixed at 7 cm and cut from 12 cm to reduce the dead space. After cutting the tube, ventilation failed and the EtCO2 was 0 mmHg again. One-lung ventilation was suspected, so manual ventilation was attempted, changing the depth of the endotracheal tube.\nIntubation with a 3.0 mm ID endotracheal tube was attempted. After the intubation, ventilation was successful and breath sounds were detected bilaterally, in addition, to regular capnography of the EtCO2. The tube was fixed at 7 cm and cut from 12 cm to reduce the dead space. However, this time the ventilation was successful. Manual ventilation was maintained without any additional problems. Twenty minutes before the end of surgery, spontaneous ventilation was encouraged. Injections with 0.05 mg of pyridostigmine and 0.01 mg of glycopyrrolate were provided and spontaneous ventilation occurred. After surgery, the patient was transferred to the ICU for potential apnea and mechanical ventilator care. The duration for anesthesia was 2 hr 30 min and the operation was 2 hr. There was no specific change in the BP or HR. The SpO2 was maintained at 95%. Two hours after surgery, the endotracheal tube was removed because the respirations were stable. Fifteen days later, an otorhinolaryngologist diagnosed tracheomalacia by laryngoscope examination. However, rigid bronchoscopy was not performed for confirmation, because there were no specific symptoms at that time. The patient recovered without any respiratory problems. |
A 65-year-old male was referred to the urology clinic in 2012 for suspected renal cystic disease discovered incidentally on CT. There were no significant findings on examination and the patient was asymptomatic.\nA contrast enhanced CT noted small SRCs bilaterally, the largest being 1.2 cm in width. A 10.5 cm upper pole simple cyst was also noted in the left kidney. It contained a small soft tissue focus in the lateral wall, however, no calcification, septations or convincing solid components were noted. The lesion fit the criteria for Bosniak stage IIF and the patient was placed on 12-month interval CT surveillance.\nIn 2013, significant increased global cyst size was noted in the left kidney, the largest being a 7.5 cm lesion in the left lower pole with a small focus of wall calcification. The left kidney upper pole cyst had increased in size to 10.7 cm with new focal soft tissue calcification noted. However, these findings were still consistent with Bosniak stage IIF. A decision was made to alter CT surveillance from 12- to 6-month intervals.\nIn 2014, the lower pole cyst in the left kidney had increased in size from 7.5 to 9.4 cm. A further 9.3 cm cyst had developed from the anterior midpole. The upper pole cyst had increased in size from 10.7 to 11.4 cm. The radiographic findings still indicated these cysts to be simple in nature. At this time the patient reported weight loss of 5 kg and fatigue over the last 6 months. eGFR had deteriorated to 29 ml/min. MRI corroborated CT results at this stage. MRI also demonstrated the left renal upper pole cyst to contain extensive thick walled septations. T1 gadolinium imaging demonstrated significant underlying haemorrhage. MRI findings upgraded the lesion from Bosniak IIF based on CT findings to Bosniak III. MRI and CT images illustrating these findings are outlined in Fig. .\nThe patient underwent an open radical left nephrectomy and a biopsy revealed clear cell carcinoma (Fuhrman grade 2). Resection margins were clear and overall the findings indicated good prognosis despite the large tumour size. Routine follow-up revealed no significant symptoms or complications. CT and MRI scanning indicated no sign of further complex renal cystic disease. |
An 81-year-old man presented with sudden abdominal pain and several days of nausea and abdominal discomfort. He was afebrile and had no diarrhea. Physical examination revealed a pulsatile mass in the central abdomen and a distended upper abdomen. Contrast-enhanced computed tomography (CT) revealed an infrarenal AAA with a diameter of 50 mm and a duodenal obstruction at the third portion between the AAA and the superior mesenteric artery (SMA). CT also displayed distension of the stomach and the first and second portions of the duodenum, accompanied by duodenal wall edema (). He was diagnosed with aortoduodenal syndrome.\nConsidering the patient’s acute symptoms, the possibility of AAA impending rupture could not be excluded completely; thus, emergency surgery was performed. Preoperative upper gastrointestinal examination with an oral contrast agent was not performed because of the emergent situation. Endovascular treatment was selected on the basis of the patient’s age, frailty (Clinical Frailty Scale 6), and anatomical suitability as his proximal landing zone was straight and over 2 cm long. Before the operation, a nasogastric tube was inserted and the intragastric fluid was suctioned completely to prevent vomiting during the procedure. Abdominal pain did not diminish after the aspiration of the gastric fluid.\nThe procedure was performed under local anesthesia and conscious sedation with dexmedetomidine to prevent aspiration pneumonia caused by vomiting on tracheal intubation. The AAA was successfully treated by fitting an endoprosthesis (Gore Excluder; W. L. Gore and Associates Inc., Flagstaff, AZ, USA). After the exposure of the bilateral common femoral artery, the main body and contralateral leg of the endoprosthesis were deployed. A proximal aortic extender was added because the digital subtraction angiography revealed a type IA endoleak. On the final angiogram, there was no endoleak (). The operative time was 96 min, and surgery was completed without complications. The patient’s abdominal pain was diminished after the operation, and a physical examination revealed a diminishment in aneurysmal pulsation.\nThe patient initially received parenteral nutrition and then began oral intake on the postoperative day (POD) 2 because of decreased nasogastric output. However, his oral intake was suspended and parenteral nutrition via a nasogastric tube was restarted on POD3 because of vomiting. CT indicated that the duodenal wall has remained edematous and the stomach and duodenum remained distended. His oral intake was restarted on POD8 after it was confirmed via CT that the edema of the duodenal wall had diminished, and distension of stomach and duodenum had improved on CT. His oral intake volume was gradually increased, and his meal form was gradually converted from liquid to solid from POD8 to POD30, and he continued oral intake without problems. A final CT showed the diminishment of the edema of the duodenal wall and no signs of duodenal obstruction, although the aneurysm sac remained unchanged in size (). The patient was discharged from the hospital without complications on POD32. He has been free from obstructive symptoms for 8 months since the operation, without aneurysmal shrinkage. |
A 32-year-old woman presented with concerns of intermittent right upper abdominal pain for one year. Her liver function tests were within normal limits. Ultrasonography (USG) revealed cholelithiasis along with choledocholithiasis. Endoscopic retrograde cholangiopancreatography (ERCP) revealed two small filling defects, and we extracted the stone via stent placement in the common bile duct (CBD). She underwent open cholecystectomy one month following ERCP. Intraoperatively, we noted the gall bladder (GB) was contracted and filled with multiple calculi, the CBD was dilated, and the hepatoduodenal ligament was frozen. Intraoperatively there was no suspicion of any bowel injury. An abdominal drain was placed in the right subhepatic region. She was discharged on postoperative day (POD) three with a drain in situ due to persistent serosanguinous discharge. The abdominal drain was removed on POD eight as output was decreased. A GB biopsy showed features of chronic cholecystitis.\nFollowing drain removal the next day on POD nine, she had concerns of abdominal pain and bilious vomiting. She was admitted and started on conservative management with nasogastric (NG) tube placement. USG of the abdomen revealed an 8 cm x 3 cm fluid collection in the GB fossa with multiple localized thick collections on the right side of the abdominal cavity. Given the sub-hepatic collection and localized peritonitis, we inserted an abdominal drain into the collection with the patient under local anesthesia. We drained 1.4 L of biliopurulent discharge on day one. She improved symptomatically but had persistent 500 to 600 mL bilious discharge from the drain and high NG output, so continued to be managed conservatively.\nGiven the suspected CBD/duodenal injury, she was referred to our center on POD 23 for further management. On initial examination, the patient’s vitals were stable with NG output of 500 to 600 mL/day, and the abdominal drain was draining 400 to 500 mL of bile each day. Blood investigations showed leucocytosis with normal liver function tests. Contrast-enhanced computed tomography (CECT) of the abdomen revealed multiloculated collection in the para duodenal region extending into the right paracolic gutter with a small perforation in the second part of the duodenum with abdominal drain and CBD stent in situ (Figures , ). She was given a trial of oral charcoal, which confirmed the presence of duodenal injury. Because of her delayed clinical presentation along with CECT findings, we planned for conservative management. Her drain was gradually withdrawn, and its output gradually decreased to 30 to 40 mL/day over one week. The abdominal drain was removed 30 days following insertion. Subsequently, her NG output also decreased, following which she was allowed oral liquids, which she tolerated well. She was discharged after a total hospital stay of one month, and the patient now has a normal follow-up course after six months. |
In February 2019, a 53-year-old female patient was admitted to the Department of Oral and Maxillofacial Surgery, “Dr. Carol Davila” Central Military Hospital, Bucharest, with an important facial asymmetry on the left side, caused by the presence of a swelling affecting the middle and lower thirds of the face, that was noticed by the patient about three months before admission (a). The lesion was asymptomatic, painless at palpation, overall of hard consistency except for small renitent areas at the intraoral examination. The patient reported no history of infectious exacerbations. The skin lining the affected area did not present any changes in color, temperature, and turgor and did not associate any motor or sensory deficit. The patient did not report any previously known conditions and did not take any chronic medication. General clinical and paraclinical assessment of the patient did not reveal any other significant pathological changes.\nA detailed dental history from the patient revealed having multiple teeth extractions in the third quadrant a few years before with no other history of swelling and pain until now. Intraoral examination showed a bilateral terminal edentulous lower arch, severe attrition, and a bicortical swelling on the left side of the mandible. The oral mucosa lining was of normal appearance, except for a small central area of translucent color, corresponding to the previously mentioned renitent area (b).\nThe initial panoramic X-ray showed a very large radiolucent lesion, extended from the region of the first molar–the angle of the mandible–vertical ramus–coronoid process, leaving intact only the left condyle. The contour of the mandible was altered, and areas of peripheral sclerosis were present. The projection of the left mandibular canal was displaced downwards. There were no teeth, impacted or erupted, in relation to the lesion (c).\nNative computed tomography (CT) revealed the presence of a large bony cavity extended in the left body and ramus of the mandible, measuring 80 mm × 35 mm in size. The cavity presented several areas of complete bone loss, with fenestration of the lingual and basilar cortical bone, deviation of the mandibular canal, and complete disruption of the superior wall of the canal (d,e).\nConsidering the major bone loss with a very high risk of mandibular fracture and the unknown diagnosis, we initiated a minimally invasive two-staged approach to treat this patient. In the first stage, the decompression of the cyst through marsupialization was performed. Under local anesthesia, we designed an ellipsoidal incision with the excision of an area of oral mucosa and adjacent cystic membrane, thus creating a fenestration of 1.5 per 1 cm between the cystic lesion and oral cavity. The cystic content, which had a greyish transparent aspect with small white inclusions, was evacuated and the edges of the oral mucosa were sutured to the cystic lining, using a technique of continuously running suture, thus preventing wound closure. The removed cystic tissue was sent for pathological assessment, which described the presence of inflamed non-keratinized squamous epithelium with cholesterol clefs and fibro-vascular tissues, suggestive for a residual cyst (f).\nThe patient was instructed on daily oral hygiene routine: tooth brushing and rinsing after every meal, as well as local chlorhexidine gluconate gel application for a period of three weeks, and was called every two weeks for follow-up visits. After 3 months, the patients’ appearance improved significantly, with almost complete restoration of facial symmetry (a,b) The imagistic examination—panoramic X-ray and cone beam computed tomography (CBCT)—evidenced an important reduction of the cystic dimensions, with the restoration of the normal mandibular contour and significant circumferential bone apposition, as a witness for active bone regeneration (c–e).\nAt 6 months, the decompression effects were stable, and the patient underwent the second stage of the treatment. Under general anesthesia, radical cystic excision was performed. The bone defect corresponding to the marsupialization window was reconstructed with a titanium mesh, fixed with screws, in order to prevent the formation of a chronic oro-cystic fistula (a). Postoperative care included local cold packs, soft diet, daily oral rinses with chlorhexidine gluconate mouthwash, antibiotic prophylaxis (ceftriaxone injection 1 g TID for 5 days), and anti-inflammatory drugs (ketoprofen injection 100 mg TID for 3 days). The sutures were removed two weeks after surgery, revealing a complete wound closure with no mesh exposure. At 12 months follow-up visit, clinical and imagistic assessments revealed a postoperative mucosa scar with no evidence of fistula formation and appropriate bone apposition, comparable to the contralateral healthy side of the mandible (b,c). |
A 61-year-old Caucasian male with a past medical history significant for a remote history of Hodgkin's lymphoma status post splenectomy, pernicious anemia, type 2 diabetes, hypertension, hyperlipidemia, hypothyroidism, gastric carcinoid tumors, and morbid obesity (BMI 47) presented with an anomalous pancreatic mass on endoscopic ultrasound (EUS). The patient had a 20-pack-year smoking history, having quit 5 years prior to presentation. The patient's diabetes had some sequelae of chronic renal insufficiency (creatinine 1.6 mg/dl), microalbuminuria, and moderate peripheral neuropathy secondary to vitamin B12 deficiency in the past. His perioperative hemoglobin A1c level was 7.8%.\nEUS was being performed for surveillance of the nodular mucosa in the gastric fundus, of which biopsies revealed changes characteristic of autoimmune metaplastic atrophic gastritis including micronodular neuroendocrine cell hyperplasia. This study revealed an 8 × 5 mm anechoic cystic lesion in the pancreatic tail. Endoscopic sonographic findings were a simple cyst without septa or debris in close proximity to the pancreatic duct.\nMRI of the abdomen showed a focal duct cut-off near the junction of the pancreatic body and tail with associated mild pancreatic ductal dilatation. Fine needle aspiration of the lesion obtained via EUS revealed cellular smears with variably sized groups of cells with round nuclei and abundant cytoplasm (fig ). Immunohistochemistry showed the tumor cells had membrane reactivity for β-catenin but no nuclear reactivity. Chromogranin and synaptophysin were nonreactive. Ki-67 immunostaining showed a proliferation index of 5–10% (fig ). A diagnosis of acinar cell carcinoma of the pancreas was favored based on these findings.\nThe patient was deemed a suitable surgical candidate, and he underwent an attempted laparoscopic distal pancreatectomy. However, given his morbid obesity and intra-abdominal adhesions the procedure was converted from a laparoscopic to a bilateral subcostal incision. It was then possible to mobilize the pancreas easily. The lesion itself was not palpable; however, based on preoperative imaging, the lesion was known to be 8 cm from the end of the tail of the pancreas. Approximately 15 cm of the pancreas were mobilized up to the level of the superior mesenteric vein. The pancreas was then divided along the midline with an Endo GIA stapler. The pancreatic stump was oversewn with a 4-0 Prolene suture in a locking fashion, and two 19-Fr Blake drains were left in the pancreatic bed. An intraoperative frozen section revealed negative margins. The patient did well postoperatively, his drains were removed, and he was discharged home in good condition on postoperative day 5. On final pathology, a 1.3-cm malignant lesion was seen compressing the main pancreatic duct, composed of large epithelioid cells with abundant cytoplasm and frequent mitotic activity arranged in sheets and focal rosettes (fig ). Areas showed bile production by tumor cells, and a hepatocyte-specific antigen immunohistochemical stain was strongly positive (fig ). The final diagnosis was a hepatoid carcinoma involving the pancreatic body, confined to the parenchyma without extension into the peripancreatic adipose tissue and negative margins. The final clinicopathological stage was T1N0M0.\nThe tumor tissue was further evaluated with high-throughput gene sequencing to evaluate for specific mutations that would warrant further adjuvant therapy. With Caris Molecular Intelligence® 600 gene sequencing, the mutational analysis revealed two genes with known alterations. BAP1 is a BRCA1-associated protein and is a tumor suppressor gene that plays a role in protein deubiquitination metabolism and transcription regulation, as well as DNA damage repair. A Q590fs frameshift mutation in exon 14 was identified, and was thought to be pathological; it has been observed in melanoma, mesothelioma, renal cell carcinoma, and cholangiocarcinoma. Additionally, a presumed benign mutation was identified in NOTCH1, a protein in the Notch signaling cascade that regulates cell-cell interactions. The alteration was related to the rare single nucleotide polymorphism A1343V. In all, 37 of the genes tested had unclassified mutations based on current knowledge, and 16 genes had indeterminate results. No genetic alterations were identified in 539 genes, including 55 common genes associated with cancer, such as BRCA1, BRCA2, eGFR, c-KIT, JAK-2, Her2/Neu, KRAS, and several others. The genetic anomalies were mapped to potential adjuvant chemotherapeutic agents based on the current literature, but the tumor did not seem to have a genetic alteration pattern associated with a known benefit with adjuvant chemotherapy, albeit this approach has not been validated prospectively. The patient was not eligible for existing clinical trials. Adjuvant radiation therapy was deferred due to the negative nodal status, small tumor size, and negative surgical margins. In this context, the patient was followed with close clinical evaluation and surveillance imaging. At 6 months following resection, the patient had no evidence of disease and was clinically doing well with excellent functional status. |
A 51-year-old female with history of hypertension and human immunodeficiency virus (CD4 count 865, compliant on anti-retroviral therapy) presented to the emergency department for altered mental status. On initial examination, the patient was awake but confused, unable to follow commands, with dysarthria and noted to have diarrhea. Upon evaluation neurological exam was limited due to patient's confusion, however, the patient was moving all extremities, withdrawing pain stimuli, negative Babinski bilaterally, negative for facial droop, and intact pupillary reflex bilaterally. Workup was significant for right upper lobe consolidation seen on chest X-ray, leukocytosis with bandemia, transaminitis, elevated creatinine, hyponatremia, hypokalemia, and a significantly elevated creatine kinase level (Table ). The patient was started on antibiotics for empiric coverage for meningitis and respiratory illness. However, differential also included stroke and infectious etiology. Further imaging was ordered which included a CT head and MRI brain which were both unremarkable for any mass effect or acute infarct and did not warrant a neurology consult at that time. The patient was later admitted to the intensive care unit after found to be hypotensive and hypoxic. In the intensive care unit (ICU), the patient was started on broad coverage with vancomycin, cefepime, and azithromycin for worsening hypoxia and severe sepsis. Patient's hypotension was fluid responsive and did not require any vasopressors to sustain her mean arterial pressure. Workup performed in the ICU included a negative lumbar puncture, computed tomography angiography of the chest that showed an obstructive mass in the right upper lobe (Figure ), negative bronchoalveolar lavage, respiratory viral panel, and sputum culture. The pathogens tested included pneumocystis, tuberculosis, and fungal species. Cerebrospinal fluid was sent for herpes simplex virus testing and a culture of the fluid was performed which were both negative. Patient history of compliance on anti-retroviral therapy with a high CD4 count made human immunodeficiency virus associated infections less likely. Ultimately, the urine antigen was positive for Legionella.\nThe patient's antibiotics were changed to Levofloxacin once all other infectious causes were ruled out. During the patient's stay in the ICU, she required short-term dialysis due to worsening rhabdomyolysis and oliguric acute kidney injury. An electroencephalogram was also performed due to minimal improvement of cognitive status and further showed encephalopathy of nonspecific etiology. With appropriate antibiotic therapy and improvement in renal function with short-term dialysis, the patient's confusion resolved and she was later extubated to room air. On discharge, she continued to have persistent dysarthria, oropharyngeal dysphagia, balance and coordination deficits. This was contributed to encephalopathy secondary to acute infectious process and metabolic changes with mild improvement noted during the stay with resolution of confusion. The oropharyngeal dysphagia could be explained by the prolonged intubation. Physical therapy recommended short-term rehabilitation. Family and the patient at that time agreed to outpatient physical therapy and speech therapy. Per primary care follow-up, the patient's dysarthria was persistent for two months post discharge with gradual improvement. |
A 25-year-old male presented at our emergency department complaining of agitation, aggressiveness, delusion, and disorganized behavior that developed within a day after discontinuation of Internet use. Five days prior to his emergency room admission, he abruptly quit an online game that he had played daily for 8 to 12 hours for two years, partially of his own will and partially due to his parents' persuasion. However, he immediately became anxious and agitated; he could not concentrate or sleep. The next day, he still felt agitated and sometimes lethargic, and he reported watching a TV program that told him that electromagnetic waves could cause sexual dysfunction. This caused him to become excessively anxious and preoccupied with the idea that he had problems with erection. He reported removing all electronic cords to his computer and other electronic equipment and wandered around worrying about sexual dysfunction. He asked his sister to have sexual intercourse with him and demanded that his father follow any orders he made. On the day of admission, the patient waited behind the door of his house to ambush his father and he attacked his father with a knife, leading to an actual injury.\nAt the intake interview, he demonstrated continued persecutory delusions (i.e., the electromagnetic waves made him impotent), impaired reality testing, and psychomotor agitation. When describing his sexual problems to a doctor, he removed all of his own clothes.\nLaboratory tests, including drug screening and a brain computed tomography (CT) scan, revealed no abnormalities. He denied any personal of family history of mental illness. Although he showed a decline in psychosocial functions, such as difficulty maintaining a stable job and disturbed a sleep-awake cycle due to excessive gaming, there were no suspected previous psychiatric morbidities except IAD.\nWe began a rapid titration of quetiapine up to 800 mg to control his impulsivity and psychotic symptoms. We observed rapid improvement of symptoms within four days of treatment initiation. At first he claimed that he was not able to remember what had happened, but he gradually began to remember and explained his behaviors. He said that he became extremely anxious and restless within a short period of time. Without noticing, he was overwhelmed with the idea that he was sexually deficient for no specific reasons. He also acknowledged hearing voices of women and men after withdrawal from using the Internet, but could not remember the content. On the fifth day of admission, he had no psychotic symptoms and reality testing was intact. He became logical and relevant. A full psychological assessment was performed on the seventh day of admission that indicated no evidence of psychotic or mood symptoms. After two weeks of inpatient treatment, he was discharged.\nSince high school, the patient played online games for approximately five hours a day, either at home or at a PC bang (Internet café). However, two years ago, as he became more skilled at a massive multiplayer online role-playing game (MMORPG), called Lineage 2, he was able to collect many game items that could be exchanged for money. Hence, he spent an increasing amount of time more than 8-10 hours a day. During that time, he sometimes spent a day without eating, talking or sleeping. Of his own volition, he attempted to cut down his time spent gaming, but always failed. He said that the game gave him a sense of satisfaction and self-esteem, and it made him feel as though he was "being a god." He described playing the game as better than talking and meeting people in the real world.\nHe fulfilled the diagnostic criteria for IAD as described by Goldberg: tolerance for excessive time on the Internet; withdrawal symptoms (psychomotor agitation, anxiety, and irritability); craving and failure to decrease or discontinue the use; continued use despite negative life consequences; and social and occupational impairment.\nSix months after discharge from the hospital, he is currently employed with a part-time job and has remained on a small dose of medication (quetiapine 12.5 mg) for sleep. He has not played the online game since admission and has not shown any signs of psychosis. |
A 69-year-old Japanese female was admitted to our hospital due to thyroid tumors that had been increasing in number and size on routine thyroid ultrasonography (US). On physical examination, the patient was in a good nutritional state and in no acute distress. She had no symptoms of heart failure. There was a palpable soft mass in the thyroid gland, and the patient’s serum CEA level was a little high (5.1 ng/mL). Free T3 and free T4 levels were normal, but her TSH level was high (4.56 μIU/mL). Full blood count, blood chemistry, and serum thyroglobulin levels were normal. She had been taking thiamazole for 25 years for hyperthyroidism.\nShe had been operated on 10 years earlier to extirpate a left atrial myxoma (Fig. ). Multiple thyroid nodules were observed at that time. Thyroid scintigraphy revealed an increased focal uptake of 99mTc in the same lesions in the left lobe, being consistent with toxic multinodular goiter. These corresponded to the criteria of CNC, and she was diagnosed with CNC. Genetic studies found no mutation in the PRKAR1A gene. From that time on, she received annual brain MRI, abdominal CT, and thyroid US examinations. The thyroid nodules on thyroid US gradually increased in number and size. Cervical CT showed heterogeneously enhanced masses with microcalcifications in the thyroid gland, but no apparent lymph node swelling was found (Fig. ). Aspiration cytology revealed the nodules to be class III, but the possibility of malignant tumors could not be ruled out. We decided to perform a partial thyroidectomy with sampling of regional lymph nodes.\nThe resected specimen is shown in Fig. . The surface was rugged and slightly hard. It included multiple nodules with a maximum size of 4.0 cm. Microscopically, multiple follicular adenomas were present in the bilateral lobes. Most of them had the characteristics of the oxyphilic cell variant. Nodular hyperplasia was scattered in the background of the tumor (Fig. ). There was no malignancy, and lymph nodes were normal.\nThe patient was discharged on the 7th day after surgery following an uneventful postoperative course and has been well ever since.\nThe characteristic symptoms of CNC are myxomatous masses (cardiac, cutaneous, and breast), pigmented lesions (lentigines and blue nevi), and endocrine overactivity. Diagnostic criteria for CNC were proposed by Stratakis CA in 2001 (Table ) []. In Japan, since the first report of Akama, only 33 cases of CNC have been reported to date.\nThe major worldwide reviews of CNC [–] and the 33 Japanese CNC cases [–] are summarized in Tables and , respectively. Spotty skin pigmentation, cardiac myxomas, cutaneous myxomas, primary pigmented nodular adrenocortical disease (PPNAD), and pituitary adenoma were the common clinical manifestations of CNC globally as well as in Japan. Meanwhile, osteochondromyxoma, psammomatous melanotic schwannoma, and blue nevi were rare in Japan. The discrepancy may reflect genetic differences among racial groups. It is therefore important to consider these regional manifestations in making a differential diagnosis of CNC. Most CNC patients in Japan initially visited a dermatologist or endocrine physician, ultimately leading to a later CNC diagnosis. This trend reflects the chief complaints of these CNC patients, which were Cushing sign (33%), palpation of the subcutaneous or breast tumor (27%), and pigment deposition of the skin (24%). It is thus difficult to make a definitive diagnosis of CNC in a patient’s first visit, and we should keep in mind the potential existence of CNC when making a differential diagnosis.\nPRKAR1A genetic mutation helps with CNC diagnosis, and it is included in the supplementary diagnostic criteria. PRKAR1A was identified in 2000 as a causative gene of CNC type 1, but not of CNC type 2 []. PRKAR1A gene encodes the regulatory subunit type 1-α of protein kinase A (PKA, cAMP-dependent protein kinase). Activated PKA promotes phosphorylation of CREB (cAMP response element-binding protein) that relates to the copying of cells, metabolism, and cell cycle progress. PRKAR1A inhibits these PKA pathways, and the PRKAR1A gene is a tumor suppressor gene. Only 10 Japanese CNC cases, including our case, have received the PRKAR1A genetic test, and most of them were recent, between 2013 and 2017, indicating that the genetic test has not been widespread in clinical practice. Seven of the 10 CNC cases which included the PRKAR1A genetic test showed PRKAR1A genetic mutation (Table ), and these patients were younger than those without the mutation (mutation group 26.6 ± 20.2 years old, no mutation group 64.3 ± 11.7 years old, p = 0.0339). Our patient with no PRKAR1A mutation was diagnosed with CNC at the age of 59 years old.\nIn CNC patients, primary tumors are present synchronously and/or metachronously in multiple organs, including cardiac myxomas, pituitary tumors, breast tumors, adrenocortical tumors, and thyroid tumors, and they require repeated surgeries over several years. Six of the 33 (18%) Japanese CNC patients underwent surgery more than three times. The CNC patients should thus be monitored closely for clinical manifestations of the disease and be aware of necessity for polysurgery to improve prognoses.\nWorldwide, up to 60% of CNC patients had thyroid nodules; nonspecific cystic disease was present in 75%, follicular adenomas in 25%, and papillary or follicular carcinomas in up to 10% of the cases []. Whereas in Japan, only 5 of 33 (15%) CNC cases showed thyroid nodules, including carcinoma [–], as shown in Table . All patients were female, and the average age at detection of thyroid lesions was 56.2 years old. The CNC patients with PRKAR1A genetic mutation were more susceptible to the occurrence of thyroid tumors, and about two thirds of these cases showed thyroid disorder in infancy or adolescence [, ]. The age at onset of thyroid cancer in two Japanese CNC patients was 72 and 73 years old, in whom PRKAR1A mutation was negative in one and not available in the other. However, the thyroid tumors were often detected by chance during a general medical examination because most patients were not conscious of any nodule or swelling of the thyroid gland, except in our case.\nThe evaluation and treatment of thyroid lesions in the 5 Japanese CNC patients are summarized in Table . Although CNC patients usually show thyroid function within the normal range, two Japanese cases with thyroid lesions, including our patient, had a high TSH level (Table 5). Our patient had taken thiamazole for an extended period of time for hyperthyroidism; the negative feedback mechanism of which might have been involved in the high TSH level. The other case had a TSH-producing pituitary adenoma. Although TSH-producing pituitary adenomas account for only 0.2~1.0% of total pituitary adenomas, 3 of the 33 (9.1%) Japanese CNC patients had TSH-producing pituitary adenomas. The correlation between TSH value and occurrence of thyroid cancer has not been demonstrated in CNC patients, but the adenylate cyclase-PKA pathway located downstream of the TSH receptor may be involved in the development of thyroid cancer. Therefore, the monitoring of TSH levels may be important to allow for an early detection of thyroid cancer in CNC patients. During the long follow-up, any increase in the number, size, and calcification of the thyroid lesions could be a sign of malignancy. Our patient underwent surgery because the thyroid nodules gradually increased in number and size on follow-up thyroid US examinations. Four of 5 Japanese CNC patients with thyroid lesion underwent a partial thyroidectomy, 2 with cancer and 2 with adenoma, histologically. It is possible that thyroid adenomas in CNC could increase and grow over a long period and have the potential for malignant transformation based on an adenoma-carcinoma sequence. Cardiovascular disease is the leading cause of death for CNC patients []. Thyroid-related death has rarely been reported, and thyroid cancer in CNC has a good prognosis due to their well-differentiated nature.\nIn summary, the characteristics of thyroid lesions in CNC are as follows: (1) the thyroid lesions are present without major symptoms and are usually accompanied by other neoplastic syndromes in various organs, (2) adenomas are the predominant pathology of thyroid lesions, and they tend to increase in size and number during a long follow-up, (3) the age of onset of thyroid lesions is younger in cases with PRKAR1A genetic mutation, and (4) thyroid cancer develops in 6% of CNC patients, but they are well-differentiated cancers, i.e., papillary or follicular cancer, with favorable prognoses. |
A 55-year-old woman was admitted to our hospital on 27 July 2021 due to severe chest and back pain and weakness of lower extremities for 11 days. She also complained of urinary and fecal incontinence. Fever was not reported since the onset. She has had a history of hypertension, diabetes and depression. In addition, she used glucocorticoids and immunosuppressant for 2 months because of immune thrombocytopenic purpura. Physical examination revealed symmetrically completely paralysis in the lower extremities, decreased muscle tension, absence of knee and ankle reflexes, and the presence of bilateral extensor plantar reflex. Several subcutaneous lumps in the lower abdomen on the left side were also revealed. The patient’s blood routine, electrolytes, liver and kidney functions were normal, and the test for syphilis and human immunodeficiency virus (HIV) was negative. Albumin is reduced to 26.7 g/L (40–55 g/L as the normal value). Consecutive cerebrospinal fluid (CSF) examinations showed a normal range in white blood cell (WBC) count and significant increase in protein concentration.\nThe cranial and cervical spine, thoracic spine and lumbar spine magnetic resonance imaging (MRI) showed abnormal signal in the spinal cord and the pituitary. MRI examinations were performed for three times from July 29, 2021 to August 10, 2021 in Figs. and . The result shows abnormal signals in the entire spinal cord (cervical spinal cord and thoracic spinal cord) in Fig. A–G. Follow-up MRI of the thoracic spine (Fig. E VS Fig. H) shows cavitation changes in the thoracic spinal cord, suggesting that the lesion has changed in a short period of time, and necrosis and cavitation in the center suggesting infectious lesions. In addition, the posterior soft tissue of the cervical, thoracic and lumbar spine all showed patchy abnormal signals with enhancement, suggesting inflammatory changes. Meanwhile, two nodules were seen on the back of the pituitary gland with T2 hyperintensity in Fig. .\nThe patient underwent chest computed tomography (CT) examinations as shown in Fig. . Figure A–C is the initial chest CT on July 31, 2021, showing a patchy high-density shadow in the left lung and multiple nodules in bilateral pulmonary. Figure D–F is follow-up CT on August 5, 2021, which shows the patchy high-density shadow disappears of the left lung, and cavities appear in nodules suggesting that the lesions have changed in a short time, and nodules with cavity suggesting the infection.\nOn July 31, 2021, the patient underwent abdominal CT examination and found two nodules under the skin of the left lower abdominal wall, as shown in Fig. A, B. There are also strips of high density around, suggesting inflammatory changes. Figure C shows the spleen become large. B-ultrasound examination of abdominal wall nodules in Fig. D shows mixed echo bolus. After ultrasound puncture, the pathology in Fig. E shows a large number of inflammatory cells suggesting infection (HE × 400; Olympus, Tokyo, Japan). The acid-fast stain showed partially acid-fast bacilli in Fig. F (× 1000). Culture of the pus and blood fluid revealed an uncommon microorganism, termed Nocardia farcinica. Nocardia farcinica protein spectra obtained was analyzed by mass spectrometry in Fig. . The results confirmed that it is Nocardia with the coincidence rate 99.9%. No positive discovery was in the culture of CSF. However, second generation sequencing of the CSF revealed the same microorganism. |
A 30-year-old male was brought to the emergency and trauma care center with severe maxillofacial injuries sustained due to shattering of an improvised helmet used during welding process. The shattered helmet has produced multiple splinters which produced a destructive type of injury with lodgments at different parts of the body []. Initial evaluation revealed a semiconscious state with Glasgow Coma Scale of E3V3M4 maintaining oxygen saturation of 92% and blood pressure of 110/60 mmHg. Examination revealed laceration from the left side of chest, severe edema of face with laceration on medial aspect of the right eye with loss of vision [], and extensive lacerations over the left side of cheek and angle of mandible. There was bleed from oral cavity and medial aspect of the right eye. Occlusion was grossly deranged. Initial management of the patient was done as per the Advanced Trauma Life Support (ATLS) guidelines with the rapid assessment of airway, breathing, and circulation, and bleeding was managed with external pressure dressing.\nOnce stabilized, the secondary survey was done with radiographic examination of the head, face, and chest. Computed tomography scan and posteroanterior view skull radiograph revealed a homogenous radiopaque mass superimposing the sphenoidal sinus and multiple radiopaque masses at the angle of mandible suggestive of splinters []. Radiographic examination of the chest showed a homogenous radiopacity (splinter) at the right 7th intercostal space. Management was done in a multidisciplinary pattern for addressing the intracranial injuries, injury to the eye, comminuted fracture of mandible, and splinter in the chest wall. Individual was taken up for surgery under general anesthesia. Individual was intubated via nasal route. Repair of the soft and hard tissue was done in a systematic manner. Due to the transection of optic nerve on the right side, evisceration of right eye was done along with primary closure of the lacerated wound on the medial aspect of right eye. Removal of splinter in the sphenoidal air sinus was undertaken through endoscopic approach via transnasal route by the otorhinolaryngology surgeon. Splinter in the chest wall was not attempted for retrieval as it was asymptomatic and as advised by cardiothoracic vascular surgeon on consultation. The major impact of the shattering was on the face causing severe disfigurement with a large open wound on the left cheek. Intraorally, there was severe laceration of the left buccal mucosa, retromolar region on the left side with deranged occlusion. Aggressive debridement of intraoral wound was done. As the fracture was severely comminuted, a submandibular approach was used and exposure of the fracture fragments along with the splinters was done []. Splinters were removed and reconstruction of the continuity of the lower border of the mandible was done using reconstruction plate and miniplate [Figures and ]. Individual recovered well postsurgery. The patient developed sialocele postoperatively which was managed conservatively with pressure dressing and cerebrospinal fluid leak with medications and periodic review by a neurosurgeon. Individual was under regular follow-up postoperatively and was rehabilitated with ocular prosthesis on the right side by the prosthodontic team of our institute []. |
A 54-year-old right-hand-dominant woman presented with spontaneous loss of extension of the right thumb at the IPJ for the preceding 5 days. The patient denied any trauma to the hand. She stated that she was carrying less than 10 pounds of supplies at work when she felt a “pop” and discomfort in the distal dorsal forearm. The patient works as a physical therapist and performs dexamethasone and hydrocortisone iontophoresis. Her physical examination was notable for the absence of right thumb IPJ active extension. Magnetic resonance imaging (MRI) showed rupture of the EPL tendon, as well as evidence of synovitis at the third and fourth compartments of the wrist.\nIn the operating room, the patient had a ruptured EPL tendon with the proximal end in the mid-forearm and the distal end in the anatomic snuffbox (). Repair was performed with an ipsilateral palmaris longus tendon graft. The synovitis noted was resected. There was no obvious bony abnormality or rough edges of Lister's tubercle; however, the third compartment turned at a sharp angle at the distal end of Lister's tubercle ().\nAt the patient's 1-month follow-up, she stated she was doing well except that she had pain in her left dorsal thumb proximally. On examination, she had normal motion of the left thumb IP and no crepitus or pain at the left third compartment of the wrist. Given her history of right EPL rupture without pain, we obtained an MRI that showed first through third compartment synovitis. Given the patient's history of right EPL rupture, left EPL synovectomy and transfer out of third compartment was recommended.\nIn the operating room, the EPL tendon was examined within the third dorsal compartment. There was significant fraying of the tendon (<50%) along its radial aspect where it was in contact with Lister's tubercle (). Lister's tubercle was inspected, and no injury or rough edge was noted. The tendon was completely released and transposed radially. Synovium was removed and sent to pathology and microbiology for further evaluation.\nIn the ensuing 3-month period, the patient underwent splinting and hand therapy. Her recovery was uneventful. For both surgeries, the resected synovium showed mild chronic inflammation; all cultures were negative for growth. |
The patient gave her informed consent in order for her case to be presented.
A 73-year old postmenopausal woman presented with a small nodule on the left upper medial quadrant on a routine mammography. Her medical and surgical histories were null; no family history of any malignancy was reported as well. This nodule was not detected on clinical examination, whereas no axillary lumph nodes were palpated. Breast ultrasound scanning verified the existence of a solid nodule. The patient was admitted to our department for a j-wire excisional biopsy. Routine laboratory tests and CEA, CA15-3, CA125 and CA19-9 were within normal limits.
Histology of the specimen (Figure ) revealed a tumor measuring 1.1 cm in maximal diameter. The cut surface appeared whitish while the tumor had a nodular configuration and was hard on palpation. Microscopically the tumor was made up mostly of ovoid to round cells with variation in size, granular eosinophilic cytoplasm and nuclear pleomorhism arranged in irregular compact nests, distinct trabeculae or insular pattern of growth. Focally rosette formation was observed. Mitoses were relatively sparse. The intervening stroma was collagenised, in some areas heavily. Immunohistochemistry was positive for synaptophysin and chromogranin. There was no evidence of vascular or lymphatic invasion. The Hematoxylin-Eosin morphology assisted by the immunohistochemical expression profile confirmed the diagnosis of a carcinoid tumor with atypical features.
In order to exclude that being a secondary tumor stemming from a primary neuroendocrine tumor located elsewhere, a total body scan was performed after an intravenous administration of 6 mCi In111 - DTPA - ocreotide. There was no evidence of increased expression of somatostatin receptor subtype 2, subtype 3 and subtype 5 throughout the patient's body (Figure ).
Having ensured the diagnosis of a primary atypical carcinoid of the breast, the findings were discussed in the breast cancer multidisciplinary team (MDT) meeting in the view of deciding management options. A modified |
We discuss the case of a 25-year old woman suffering from progressive limb weakness and clinical presentation of a proximal tetraparesis with slightly reduced muscular tone and mild Trendelenburg’s sign. She has had difficulties in walking longer distances since the age of 14 when she underwent corrective osteotomy for congenital genu valgum. However during the last years she noticed a newly developed decrease in tone and strength of the proximal muscles, predominantly affecting the lower limbs, especially when climbing stairs. Muscular weakness had slowly progressed, being most prominent in the evening hours. For the past 6 months she had been dependent on walking aids (canes or crutches). Moreover she reported of exertion induced myalgia. Family history at this time was unremarkable.\nClinical examination showed a symmetric proximal tetraparesis particular of the limb girdle muscles (MRC grade 4+ / 5 on arm elevation, rotation and abduction and 4 / 5 on hip and knee flexion and extension on both sides). Strength of the distal muscles was unremarkable. Muscle tone was slightly decreased. There were no relevant muscular atrophies. Tendon reflexes were normal. Examination of gait revealed slight bilateral Trendelenburg’s sign. History and clinical features were consistent with signs of a neuromuscular disorder.\nOn diagnostic work-up, both cranial and spinal MRI scans were unremarkable, as well as the lumbar punction. ECG and echocardiogram showed no abnormalities. Electrophysiological examination showed neither signs of peripheral sensory or motoric neuropathy nor of central nervous disorders, however EMG revealed myopathic changes in the proximal muscles of the upper and lower limbs with few myotonic discharges. MRI of the thighs revealed fatty degeneration of the gluteal and quadriceps muscles ( and ). X-ray of pelvis, hip and knee joints showed no osseous destruction but moderate signs of osteoporosis. Computed tomography of the spine and pelvis measuring bone mineral density showed a Z-score of -4.0 compatible with generalized osteoporosis.\nBlood tests showed serum creatininkinase and myoglobine within normal range, however Alkaline phosphatase (ALP) was elevated (254 U/l, ref. < 104). Thyroid function tests were unremarkable including thyroid antibodies. Screening of autoantibodies showed elevation of antinuclear antibody (ANA) titer (1:320, ref. < 1:80) and positive antibodies against Sjoegren’s syndrom A (SSA-AB), whereas all other antibodies including p- and c-ANCA, antibodies against the acetylcholine receptor, the MuSK protein and Titinantibodies were unremarkable. We also found regular increase of lactate and ammoniac following physical exercise. Particularly remarkable results were found when testing for the parathyroid function, revealing excessively increased parathyroid hormone (PTH) levels (959 pg/ml, ref. < 72) along with decreased serum phosphate (1.8 mg/dl, ref. 2.7-4.5) and serum calcium levels (2.00 mmol/l, ref. 2.08-2.65). Further workup revealed markedly decreased vitamin D levels (< 4.0 ng/ml, ref. 31-100).\nBased on these results, we diagnosed secondary hyperparathyroidism with osteomalacia and a neuromuscular disorder as a result of presumably long standing vitamin D deficiency. Further rheumatological workup confirmed the diagnosis of Sjögren’s syndrome, but causal relationship with the neuromuscular symptoms was found to be unlikely. Endocrinologists recommended a therapy with high dose vitamin D substitution (single i.m. injection of cholecalciferol 200,000 IE, followed by weekly oral intake of cholecalciferol 20,000 IE) and oral substitution of calcium 2000 mg daily. Three months after the recommended therapy was started, serum calcium levels had normalized and serum PTH levels had decreased to 90 pg/ml. Moreover both neurological symptoms and clinical appearance markedly improved: the patient did not suffer from myalgia anymore and was able to walk without aid again. |
A 26-year-old male patient presented to the emergency department with a seven-day history of generalized intermittent crampy abdominal pain. The pain was accompanied by nausea and multiple episodes of vomiting. The pain worsened after meals. His last bowel motion was three days before the presentation; however, he continued to pass flatus. He had multiple similar episodes of this pain over the last two months that resolved spontaneously. He was otherwise healthy and had no history of previous abdominal operations.\nOn examination, she was in pain and her vital signs were as follows: pulse rate as 90 bpm, blood pressure of 121/76 mmHg, and respiratory rate of 20 bpm, and she was afebrile. Abdominal examination revealed a distended abdomen, with no tenderness or guarding. There was hyperactive bowel sounds all over the abdomen. No palpable masses or organomegaly and no external hernias were noted. The digital rectal examination was unremarkable.\nLaboratory studies showed hemoglobin of 13 g/dL, white blood cell count of 8,000/mL shift, and platelet count of 144,000/mL. Serum electrolytes, urea, creatinine, and liver function tests were all within normal limits. The chest X-ray did not reveal any abnormalities with no free air under the diaphragm. CT scan of the abdomen was performed (Figure ), which demonstrated sac-like clustered small bowel loops noted in the left upper quadrant, in the anterior pararenal space. The remainder of the large bowel is completely collapsed. These findings were suggestive of the diagnosis of left paraduodenal hernia, and the surgical team was then informed.\nGiven the aforementioned clinical and radiological findings, the patient was then prepared for diagnostic laparoscopy, which had confirmed the diagnosis of a left paraduodenal hernia. The surgery was performed under general anesthesia with the patient placed in a supine position, and three ports were inserted to carry out the procedure. After establishing the pneumoperitoneum and introducing trocars, careful adhesiolysis was performed. The entrapped bowel loops were noted, but it was difficult to reduce laparoscopically because of the dense adhesions. The surgery was then converted to open, and enterolysis was performed. The patient tolerated the procedure well with no complications and was discharged on the fourth postoperative day with no active complaints during the follow-up visit. |
History and physical
A 67-year-old male former smoker with a history of prior occupational asbestos exposure and recurrent bronchitis presented with progressive dyspnea and thoracic pain to the point that he could not lie down in bed. A computed tomography (CT) scan of the chest was performed, which was interpreted as right-sided pneumonia with right parapneumonic effusion. He was sent to his local emergency department, where he was admitted for antibiotics and thoracentesis, the latter which demonstrated the presence of atypical mesothelial cells with inflammatory cells. He was readmitted two weeks later for progressive thoracic pain, was found to have a recurrent right-sided pleural effusion, and was managed with partial right pleurectomy with pleural biopsy, and talc pleurodesis. Right pleural pathology demonstrated atypical mesothelial proliferation at the pleural surface, without true invasion or definitive pathologic evidence of malignancy. Following surgery, he felt substantially better, such that he could sleep in the bed again, and he was able to return to his baseline activity levels. He underwent repeat chest CT five months later, which showed right pleural thickening and a small loculated pleural effusion, favored to represent a combination of calcification, pleurodesis, and atelectasis. He remained clinically well for another five months until he presented with cough and sinus congestion unrelieved by guaifenesin, dextromethorphan, and antibiotics. He underwent repeat chest CT that showed extensive mass-like pleural thickening completely encasing the right lung, with prominent involvement of the mediastinal pleura, and probable mediastinal extension into the right paratracheal and precarinal space, with pericardial effusion and probable pericardial metastases. There was no definite invasion into the right chest wall and no evidence of disease outside of the thorax.
He then established care at our institution’s mesothelioma and pleural disease multi-disciplinary program. Pathology review of the previously biopsied pleural tumor revealed that the pleural tumor cells were positive for Wilms' tumor-1 and calretinin, and negative |
A 12-year-old female patient presented to our OPD with complaints of swelling over the posterior aspect of her right elbow of 45 days duration. Swelling was sudden in onset, initially of a size of a one-rupee coin and gradually progressed to about 5 × 4 × 2 cm at the time of presentation. Patient did not give any history of trauma/prick injury. There was no history of any kind of immobilization or massage treatment. No similar swellings elsewhere in the body. Patient complained of pain during lifting weights or when direct pressure is applied.\nOn examination, swelling extended from the lower third of right arm to the elbow joint. It was globular in shape with the overlying skin being pinchable and erythematous. Swelling was firm in consistency with mild tenderness and no local rise of temperature. It is mobile in both horizontal and vertical directions suggestive of nonfixity to the underlying bone. All movements at elbow were unrestricted and pain free. There was no muscle wasting and sensations over the arm and forearm were intact without any distal neurovascular deficits ().\nSerum calcium (10.2 mg/dL), serum phosphorus (3.0 mg/dL), serum Alkaline Phosphatase (127 IU/L), parathyroid hormone levels, creatinine kinase, aldolase levels, ANA, Vitamin D levels, 24 hours urinary calcium, and inorganic phosphate were within the normal limits. An ultrasonographic scan revealed a well-defined and calcified intramuscular lesion measuring 4.1 × 3.4 cm in the lower end of triceps muscle, with no fixity to humerus. Radiograph showed well-defined calcified mass over the posterior aspect of the distal end of the humerus extending up to the elbow joint.\nWith the above clinical scenario, we opted for excision biopsy that revealed a single encapsulated grey white mass of tissue measuring 4 × 3.5 × 2 cm overlying the posterior aspect of humerus in the triceps muscular plane ().\nMicroscopic description showed large homogenous masses of calcified material separated by thick fibrous bands of septa. Numerous giant cells and macrophages were observed in the septa confirming it to be calcinosis cutis.\nPatient was followed up to a period of one year, where there were no signs of recurrence. |
A 38-year-old man of Korean descend and now living in Japan presented to the outpatient clinic of a local hospital with abdominal pain, melena and dizziness. Laboratory test showed that he had microcytic anaemia (haemoglobin 8.7 g/dL and mean corpuscular volume 71.7 fL). Faecal occult blood test was positive. His medical history included a previous duodenal bulb perforation which required surgery in his teens. Oesophagogastroduodenal endoscopy (OGD) and colonoscopy was subsequently performed, revealing no active bleeding but slightly deformity and linear scares in duodenal bulb. A plain CT scan later performed revealing no abdominal or pelvic masses. The patient received transfusion of two units of red blood cells which restored his haemoglobin to 11.5 g/dL, relieving his symptoms significantly. He was discharged on day 12 and later referred to our hospital, a metropolitan centre in central Tokyo, for further workup and treatment.\nIn the diagnostic workup, chronic iron deficiency anaemia caused by small intestinal bleeding was considered. BAE (single balloon, rectal approach) was hence performed in search of the culprit lesion. However, the scope failed to advance to the jejunum; mild inflammatory changes were found in the proximal ileum. Capsule endoscopy was further performed to inspect the small intestine. There was initially a delayed pylorus passing of the capsule endoscope due to stenosis in the duodenal bulb, but the capsule eventually reached to the small intestine. Images from the capsule showed small intestinal ulceration with surrounding erythematous mucosal changes in jejunum, which were likely to be the cause of the recent bleed (). Several days later, a plain abdominal X-ray was performed confirming capsule retention in distal small intestine. As a result, a secondary BAE (single balloon, rectal approach) was performed to retrieve the capsule. Despite the stenosis in area surrounding the jejunum–ileac junction, the scope was successfully inserted to the proximal ileum revealing geographic ulcers in the jejunal–ileac junction (). The retrieval of the capsule was successfully done. A Roentgen barium study was later performed, which revealed small intestinal morphological changes that were similar to the earlier endoscopic findings, such as mild stenosis with deformity of duodenal bulb () as well as stenosis in jejunal–ileac junction along with proximal dilatation ().\nBased on the clinical findings above, we were suspicious that this patient was likely to be affected with CEAS. In order for this condition to be diagnosed, his blood was obtained for genetic testing. NGS was performed in the laboratory, revealing point mutations of SLCO2A1 gene’s 7 exon (940+1G>A) and 13 exon (1807C>T) allele using the MiSeq method (Illumina corp). Additionally, a brain MRI showed mild pachydermia in the forehead which was later confirmed by a dermatologist (). There was minimal change on fingers, toes and long bones, but it is unlikely to be typical finding of clubbed fingers or other changes on hypertrophic osteoarthropathy, however.\nBased on the results of the NGS, morphological changes to the small intestine and clinical presentation, a diagnosis of CEAS was made. Since his presentation, the patient’s general condition has since improved, but still intermittently complaints about abdominal pain and diarrhoea. |
A 25-year-old male presented to emergency with a history of metallic ring stuck at the base of penis following insertion for sexual pleasure, two hours after the incident (Figure ). Initially, he was admitted to the urology department and was posted for its removal. Help was sought when they failed to remove it after their best efforts. The ring was approximately 1 cm wide and 0.25 cm thick and was made up of gold-plated platinum. The penis was swollen with a constriction at the base. The venous prominence was seen distal to the constriction ring. Mild edema at the glans penis was noted. The first attempt was made to cut the ring using a motorized cutting tool used in orthopedics. However, the speed and size of the saw were worrisome and there was every possibility of damaging the underlying soft tissue due to its high speed. Hence, this attempt was abandoned.\nThe second attempt was made using a Bard-Parker (BP) handle (Figure ) and a jumbo cutter (Figure ). This technique was fairly simple, as we passed the reverse end of the BP handle between the metallic ring and soft tissue at the base of the penis. The jumbo cutter was used to cut the ring over it. The advantage of using the reverse end of the BP handle blade was to ensure the prevention of soft tissue damage as one stabilizes it, while the other person can use a considerable amount of force over the jumbo cutter. After cutting the ring, the ends were opened out (Figure ) using nose pliers and the penis was freed with minimal soft tissue damage (Figure ) within an hour of emergency posting. The venous engorgement was quickly subsided and edema subsided after an hour.\nThe case was followed up in urology OPD at regular intervals. At follow-up one year later, the penis showed complete healing of ventral skin abrasions and no residual constriction (Figure ). |
A 59-year-old paraplegic male presented to the emergency department with progressive prolapse of the bladder through his SPC site. He had a gunshot wound in 1990, which resulted in spinal cord injury and paralysis below T8, and in 2006, he underwent SPC to manage neurogenic bladder.\nOn presentation, physical examination revealed malodorous and necrotic appearing bladder mucosa protruding through the SPC site. Patient had associated diffuse abdominal pain but denied any fevers, flank pain, hematuria, increase or decrease in urinary output.\nPatient consented to a cystectomy with an ileal conduit. A cystoscopy was performed prior to the procedure that revealed swelling and necrosis around the prolapsed bladder tissue. Multiple diverticula were noted, however, no stones or masses were appreciated.\nThe surgery was performed as a combined procedure by both the urology and the general surgery team without any complications. All of the necrotic tissue surrounding the prolapsed area were debrided until healthy tissue was encountered. The fascia was closed, however, the skin was left open due to the state of the prolapsed bladder through the skin. Packing was adequately placed with a wound vacuum assisted closure (VAC) over it. Patient was then transferred to the post anesthesia care unit in stable condition.\nThe histopathology of the bladder revealed stage IIIA (T3b) invasive and poorly differentiated urothelial cell carcinoma (Fig. ) with areas of mullerian and squamous cell carcinoma features (Fig. ). Histology also showed areas of necrosis and acute inflammatory changes.\nSix days after the operation, when changing the midline wound VAC dressing, bowel evisceration was noted at the lower aspects of the wound. Patient was taken back into the operating room that evening. The midline fascia was readily separated and remaining abdominal sutures were gently divided. Upon immediate inspection, the patient was noted to have necrotic fascial edges and subcutaneous tissue. Subsequently, using a combination of sharp dissection and electrocautery, the abdominal wall subcutaneous tissue was debrided. After successful debridement to attain healthy tissue margins, mesh was used to repair the fascial defect, as the fascia could not be re-approximated in a circumferential manner. After this was completed, the subcutaneous tissues were thoroughly irrigated and a new VAC dressing was placed. The abdominal tissue was sent to pathology and histology showed necrosis and microabscess formation.\nTwo days following the debridement and evisceration repair, the patient decompensated and a rapid response was alerted. At that time, the patient was febrile, tachycardic, hypotensive, short of breath and had altered mentation. He was intubated and started on fluids and antibiotics for treatment of acute respiratory failure secondary to septic shock.\nPatient was brought back to the operating room for further debridement due to possible necrotizing skin infection. Necrotic mesh was removed and after adequate tissue debridement, an open abdomen negative pressure therapy system (ABTHERA) was placed over the abdomen.\nPatient subsequently returned to the operating room on two additional occasions for further debridement secondary to continued necrosis of the abdominal wall. He remained intubated, sedated and on multiple pressors and broad-spectrum antibiotics.\nUnfortunately the patient continued to decompensate and remained hypotensive with multi-system organ failure. Escalation of care was refused and only supportive care was continued. |
A 25-year-old man with no past medical or drug history, presented to the emergency room with an acute pain of the right upper quadrant and flank evolving for three days associated with fever. The patient reported no chronic abdominal pain or transit disorders.\nPhysical examination showed that the patient had fever 39 °c. The abdominal examination revealed the presence of a 10 cm - regularly shaped, soft, and fixed mass in the right hypochondria and flank. The rest of the abdomen revealed no remarkable abnormalities.\nLaboratory tests showed a high c-reactive protein level (173.5mg/l) but a normal white blood cell count (7553/μl). No anemia nor hemostasis disorders were noticed. Furthermore, urine culture was normal.\nFirstly, we decided to perform an abdominal ultrasound that showed a cystic formation of the right pericolic gutter with a thick wall and several septa within. The mass had close relationships with the sixth liver segment and the ascending colon. At first, we suspected a hydatid cyst of the liver because Tunisia is an hydatid endemic area. However, a malignant tumor of the colon could not be ruled out.\nTherefore, we indicated an abdominal CT scan to characterize the mass and determine its exact location. It revealed a regular 10-cm- intraperitoneal cystic mass independent from the liver but repressing the ascending colon with heterogonous internal density. Moreover, the mass was multilocular edged by thick walls showing low contrast enhancement after intravenous injection. Peritoneal effusion was detected on CT scan in the pericolic gutter and douglas pouch, to boot (, ).\nAn infected cystic lymphangioma was then suspected besides the diagnosis of intraperitoneal hydatid disease or infected malignant tumor of the colon. We decided then to perform open surgery for diagnostic and therapeutic purpose.\nWe found a 10-cm-cystic-mass containing a serous bloody fluid in the right pericolic gutter depending from the ascending colon. The colon was not distended. No liver metastasis nor peritoneal carcinosis were noticed. An ileocolic en bloc -resection was performed with an immediate ileocolic anastomosis. ().\nMacroscopically, the 10 –cm colonic cystic mass had many septas and contained serous fluid. It was obviously arising from the ascending colon. In addition, histopathological examination revealed dilated lymphatic and vascular components lined with attenuated endothelial cells. These findings were consistent with the diagnosis of a cystic lymphangioma. The patient's post-operative course was uneventful. He was discharged on the 5th post-operative day. A 6-months clinical and radiological follow-up showed no symptoms or recurrence with good tolerance to regular diet. |
A 5-year-old female presented to our hospital in 2007 with a 3 - 4 month history of hemoptysis. Laboratory analysis demonstrated severe anemia, and a chest radiograph showed diffuse airspace opacities in the right lung with a small airspace opacity in the left upper lobe (). CT scan revealed ground glass opacity in all lobes of the right lung with ground glass opacity identified in the left upper and left lower lobes to a lesser extent, as well as dense right middle lobe consolidation (). Subsequent wedge biopsy of the right middle lobe demonstrated pulmonary hemorrhage with changes suggestive of pulmonary capillaritis. Testing for cardiac, rheumatologic, and infectious disease was negative. After cessation of hemoptysis and stabilization of her hemoglobin, she was sent home on prednisolone (1 mg/kg) and hydroxychloroquine. Within 1 month, hemoptysis recurred, and imaging was pursued to evaluate for active bleeding. In order to avoid more radiation exposure with subsequent CT, an MRI was performed to evaluate for pulmonary hemorrhage. MRI revealed increasing opacities in the left upper lobe compared to the prior CT, suggesting increased hemorrhage. Due to young age and ability, she was not able to perform comprehensive PFT at the time of initial diagnosis.\nIntravenous methylprednisolone (30 mg/kg daily × 3 days) and intravenous immunoglobulin (IVIG) (2 g/kg) were initiated, and then continued weekly and monthly respectively. As MRI demonstrated pulmonary hemorrhage before serologic changes, serial MRIs were performed to monitor disease progression and recurrence, especially as medications were weaned. Over a 5-year period, medications were weaned to monthly IVIG (1 g/kg) and prednisolone (5 mg every other day). With increasing age, her ability to perform more complex PFT improved, and demonstrated no evidence of restriction, obstruction, or diffusion limitation. Serologic markers remained negative, save for a modest increase in ANA (1:160 - 1:320). From July 2007 to February 2012, serial MRI images demonstrated stable disease. shows representative images during that time period.\nAfter 5 years of remission, she had recurrent hemoptysis, bilateral radiographic infiltrates, and decreasing hemoglobin. PFTs revealed an increase in DLCO consistent with an acute pulmonary hemorrhage. Repeat MRI revealed new patchy airspace opacities involving the majority of the right lung and the left lower lobe (). As recommendations had changed [], 1 g weekly intravenous methylprednisolone therapy was added to the monthly IVIG. Five months later, her hemoptysis recurred with similar laboratory and PFT changes. MRI revealed multiple peripheral small airspace opacities in the right lung (). IVIG was increased to 2 g/kg monthly, and azathioprine was started (2 mg/kg/day). She is stable on that regimen with normal PFTs and unchanged serologic markers. |
A 29-year-old man was admitted to our emergency surgery unit with a 2-day history of recurrent bilious vomiting, colicy abdominal pain and distension. He had no significant medical history.\nThe patient's vital signs were normal. The abdomen was distended, with increased bowel sounds, but no tenderness or mass were noted. Routine laboratory blood analysis was within normal limits. A plain upright abdominal radiograph showed air-fluid levels in the upper quadrants. Abdominal ultrasonography suggested mechanical obstruction. A computed tomography (CT) scan demonstrated the small bowel loops clustered to the left of the abdomen within a sac-like structure []. A decision for laparoscopic management was taken on the basis of these clinical and radiological findings.\nThe operation was performed using five trocars with Hasson's technique. At laparoscopy, the small bowel loops were seen dilated in the proximal part of the jejunum to a distance of approximately 40 cm from the duodenojejunal flexure. Beyond this point, the rest of the jejunal loops and the proximal ileum were conglomerated within a whitish fibrous sac. The other organs including the distal ileal loops were normal. Following extensive adhesiolysis using endoscissors and endograspers, the entire entrapped small bowel was freed from the sac [] and the sac was excised. There was a dense adhesion band in the proximal jejunum which caused the obstruction. Following the lysis of this band, the obstructed jejunal loops were relieved without bowel resection. The loops within the cocoon were also adhered to each other by thin flimsy adhesions which could be separated easily. Following thorough peritoneal cleansing, the operation was terminated with no intraoperative complications.\nHistopathologic examination of the sac showed proliferation of fibroconnective tissue with nonspecific inflammatory reaction. Cytologic studies and cultures of the ascitic fluid were negative for malignant cells and bacteria, respectively. Following an uneventful recovery, the patient was discharged home on the fourth postoperative day. During a six-month postoperative period, he showed no obstructive symptoms. |
A 49-year-old woman visited a neighborhood eye clinic complaining of a foreign body sensation and pain in her right upper eyelid of several days duration. She was referred to our hospital for further evaluation and treatment on the next day. Several years earlier, she had a tumor excised from her right eyelid, and the tumor was diagnosed as a spiradenoma. Our examination showed that her best-corrected visual acuity (BCVA) was 20/30 OD and 20/20 OS. A round to oval-shaped nodule with smooth borders was present in the right upper palpebral conjunctiva (Figure A), which was thought to be the same location of the previous tumor. The tumor was excised and evaluated histopathologically. The tumor cells were arranged in intertwining bands just beneath the conjunctiva. Two types of neoplastic cells were recognized; one had a small dark nucleus representing an undifferentiated cell, and the other was located at the center of the bands with large pale nucleus (Figure B). Levels of mitotic counts were low (0.3/10 HPF) and immunohistochemical analysis showed that the Ki-67 labeling index was 2.8% (Figure A). It was unclear if there were the tumor cells still in the incisional margin of the slices.One year later, she visited our hospital again complaining of pain in the right eye. A nodule of approximately 1–3 mm in size accompanying by enlarged blood vessels was observed at the right upper palpebral conjunctiva (Figure C). This nodule was regarded as a recurrence. Preauricular and cervical lymphadenopathy was not present. Laboratory data including blood chemistry, and serum level of tumor markers were within the normal range. No abnormal findings were seen in the chest X-rays. The tumor was excised with adjunctive cryotherapy and examined histopathologically. The solid and well-circumscribed nodule was located beneath the conjunctival epithelium. The tumor cells were slightly basophilic with clear cytoplasm and oval nuclei were arranged in a thick trabecular pattern (Figure D). The two cell pattern, which was recognized in the previous tumor, was inconspicuous. Mitotic activity was increased (5.5/10 HPF) throughout the tumor with no necrosis (Figure D). Immunohistochemistry showed a Ki-67 labeling index of 12.0% (Figure B). The tumor stained weak positive for epithelial membrane antigen (EMA), and negative for carcinoembryonic antigen (CEA), low molecular-weight cytokeratin (CAM5.2), synaptophysin, and chromogranin A immunohistochemically. Periodic acid-Schiff (PAS) stain was positive in the cytoplasm (Figure C), but the positivity disappeared after digestion by diastase (Figure D). The tumor was diagnosed as a recurrence of the spiradenoma. There has been no recurrence in the 6 months after the surgical removal and cryotherapy. |
A 63-year-old woman with metastatic breast carcinoma presented to the ophthalmology clinic with diplopia in right gaze and head turn to the right. Medical history revealed that she was diagnosed with estrogen receptor (ER)-positive and progesterone receptor (PR)-positive invasive ductal carcinoma 1 year earlier with mediastinal lymph node and bone metastasis at the time of diagnosis. She was treated with zoledronic acid 4 mg monthly and paclitaxel 80 mg/m2 weekly for 12 weeks, followed by endocrine therapy with letrozole.\nOn ophthalmological examination, best corrected visual acuity was 20/25 in both eyes. Slit-lamp examination of the anterior segment and fundus was unremarkable other than bilateral posterior chamber intraocular lenses. On motility exam, abduction was totally limited in the right eye with globe retraction and narrowing of the palpebral fissure on attempted abduction (). Abnormal head position towards the right side was noted. Magnetic resonance imaging (MRI) revealed isolated enlargement of the right medial rectus muscle (). Clinical evaluation and laboratory studies were carried out for differential diagnosis. There were no clinical findings suggestive of thyroid eye disease and thyroid function tests were normal. Rheumatologic assessment for inflammatory and vasculitic diseases was not contributory. Biopsy of the right medial rectus muscle was performed to establish a definite diagnosis and initiate appropriate treatment.\nHematoxylin and eosin staining of the biopsy specimen revealed large, round to polygonal epithelioid tumor cells arranged in loosely cohesive clusters and sheets infiltrating fibrocollagenous tissue and muscle fibers (). Immunohistochemical analyses using streptavidin-biotin peroxidase complex method revealed panCytokeratin and cytokeratin 7 positivity (). ER, PR and human epidermal growth factor receptor 2 (HER2/neu) were negative (triple-negative). Based on the patient’s clinical history and the morphological and immunohistochemical features of the tumor, she was diagnosed with breast carcinoma metastasis to the right medial rectus muscle. Pathological examination demonstrating a triple-negative breast carcinoma indicated discordance with the primary tumor, which was ER- and PR-positive at the time of diagnosis.\nThe patient was referred to the radiation oncology department for external beam radiation therapy. The orbital mass was irradiated with 45 Gy in 15 fractions. Following radiotherapy, chemotherapy with docetaxel 100 mg/m2 once every 21 days was initiated. After 15 months of follow-up, abduction of the right eye has partially recovered; the patient is stable and continuing to receive palliative chemotherapy. |
A 4-year old male child was referred to our hospital (St Paul’s Hospital Millennium Medical College located in Addis Ababa, Ethiopia) from a district hospital for further evaluation of recurrent chest infection and wheezing in 2018. He was admitted to the referring hospital eight times for chest infection in the first 2 years of his age. The first admission happened at the age of 2 weeks. Cough, fast breathing, and wheezing were the main presenting symptoms during the exacerbations. At each admission, he was treated with parenteral antibiotics and bronchodilators and was discharged from the hospital after he got improvement. He was apparently healthy in between the episodes of the respiratory illness. He did not have history of chocking episodes. He was delivered in a hospital at term, and pregnancy was uneventful. He had optimal development and was a kindergarten student at the time of writing this report.\nOn examination, he was not in respiratory distress and SPO2 was 94%–96% with room air. He weighed 14 kg, with a height of 106 cm (his anthropometric indices lie above –2 SD except his weight-for-height which lies between –2 and –3 SD). Chest examination revealed trachea shifting to the right side, decreased air entry over the right lung field, and relative dullness over the same area. Cardiac auscultation revealed normal heart sounds with regular rate and rhythm with no gallop. He was alert and playful, having good interaction during the examination.\nBasic laboratory examinations were within normal limits, and HIV status was negative. Chest X-ray showed hyperinflated left lung and reduced right lung volume with decreased vascularity and trachea shifted to the right ().\nEchocardiography demonstrated absence of the right pulmonary artery with no other cardiac lesion (). Subsequent computed tomography (CT) scan also showed hypoplastic right lung with absent right pulmonary artery ().\nThe case was evaluated by a team comprising pediatrician, pediatric pulmonologist, and cardiologist, and the team decided to follow the child regularly. |
The patient was a 43-year-old man who had been resuscitated from idiopathic ventricular fibrillation and who had undergone the implantation of an ICD through the left subclavian vein in a community hospital. The ICD was successfully implanted with transvenous active fixation of the dual coil shock lead (Riata, 8 French, model 1580; St. Jude Medical system, Sylmar, CA, USA) in the right ventricular (RV) apex and the bipolar pacemaker lead (Fineline, 5 French, model 4479; Guidant, Indianapolis, IN, USA) in the right atrial appendage.\nHe had undergone local debridement without the removal of the ICD system 3 times after the implantation due to ICD pocket infection. At 2 weeks after implantation, the first round of local debridement was performed due to warmth and erosion of the skin over the pocket site. At 4 months after implantation, the second round of local debridement was performed for wound disruption and purulent discharge. Because the wound was disrupted again at 14 months after implantation, a third round of debridement was performed with deep insertion of the generator into a more inferior location.\nAt the time of implantation, the pacing threshold of the RV Riata lead was 0.75 V/0.5 ms, the R wave amplitude was 12 mV, and the pacing electrode impedance was 510 Ω. At 3 years after implantation, the pacing threshold suddenly increased to 5. 0V/1.0 ms, with a decrease in the R wave amplitude 4.1 mV (pacing electrode impedance 475 Ω). After 5 years, although the ICD no longer captured the RV, even at the maximum pacing output with an R wave amplitude of 5.5 mV and a pacing electrode impedance of 420 Ω, attending physicians decided not to replace the ICD system because neither sensing failure nor significant lead displacement was observed on a chest radiograph. At 8 years after implantation, the ICD started to deliver several inappropriate shocks because the RV lead could no longer sense the R wave. The pacing electrode impedance was 330 Ω at that time.\nThe patient was therefore referred to our hospital for lead extraction. Chest X-ray images of the ICD showed significant displacement of the lead tip in comparison with the time of implantation (Figure ). A CT scan of the thorax (Figure ) showed perforation of the ICD lead through the anterior RV free wall and migration to the thoracic wall without pericardial effusion or pleural effusion. After discussion with the heart team, including cardiac surgeons, we decided to extract the lead transvenously under surgical backup with transesophageal echo monitoring and continuous blood pressure monitoring. The active fixation Riata lead was successfully unscrewed, but it could not be removed by simple traction using a locking stylet. Although some adhesion was observed between the leads, especially at the superior vena cava coil, the application of a laser to the atrial lead resulted in successful extraction. The Riata lead was easily moved by applying laser within only the left subclavian vein. The penetrating lead was successfully extracted via a percutaneous approach without any increase in pericardial effusion or pleural effusion. Because the patient had a history of ICD infection, we re-implanted a new ICD system via the opposite subclavian vein several days after the removal of the first device. |
A 70-year-old male with history of amyotrophic lateral sclerosis (ALS), dysphagia status post gastrostomy tube placement, chronic respiratory failure status post tracheostomy, and gastroesophageal reflux disease presents with six-day duration of melanotic stools. The patient was mechanically ventilated with gastrostomy tube and tracheostomy in place. His physical exam otherwise was benign. He was found to have acute onset anemia with hemoglobin of 7.1 g/dl. He was suspected of having peptic ulcer disease secondary to NSAIDs given the history of chronic musculoskeletal pain with NSAID use. Due to continual decrease in hemoglobin requiring daily blood transfusions despite conservative management, upper endoscopy was performed. It demonstrated a bleeding ulcer distal to pylorus in the duodenal bulb. Endoscopic technique was used to remove the clot, but the procedure resulted in bleeding that could not be stopped leading to poor visualization despite epinephrine injections. Due to his multiple comorbidities, he was deemed to be a poor surgical candidate. The patient was referred to interventional radiology for embolization of gastroduodenal artery (GDA).\nDuring the embolization of GDA, the celiac angiogram demonstrated no evidence of active extravasation, but contour irregularity at the level of the mid-GDA was noted. Prophylactic coil embolization of GDA was performed achieving GDA stasis. Post-embolization angiography of the SMA demonstrated no evidence of bleeding from IPDA branches. Incidentally noted was a replaced right hepatic artery arising from the SMA.\nDespite embolization of GDA, patient’s hemoglobin continued to decrease to 6.8 g/dl and the patient continued to have melena and hematemesis resulting in hypotension. The patient required an additional nine units of packed red blood cells (pRBCs), six units of platelets, and five units of fresh frozen plasma. Computed tomographic angiography (CTA) showed a large bleed from IPDA off the SMA immediately posterior to the origin of the replaced RHA (Figure ).\nThe patient was referred for urgent embolization by interventional radiology.\nDuring angiography, no active extravasation was identified and given that the GDA was already embolized, the most reasonable source vessel in the suspected territory would be the IPDA. The replaced RHA was selected and IPDA was identified as its first branch (Figures , ).\nWhile the branch arising directly from the replaced RHA represented the posterior IPDA, the anterior IPDA was seen as collateral vessel going back to the middle colic artery (Figure ).\nHowever, no focus of active bleeding could be identified from digital subtraction angiography from the posterior IPDA or the anterior IPDA. Cone beam computed tomography (CBCT) catheter angiography of the posterior IPDA was performed to identify the region of the duodenal bleed analogous to the region of active extravasation seen on abdominal CTA. The same location on the posterior wall of the duodenum seen on abdominal CTA was matched on CBCT angiography (Figure ).\nSubsequently, the posterior IPDA was embolized using n-butyl cyanoacrylate (NBCA) (Trufill n-BCA Liquid Embolic, Codman Neurovascular, Raynham, Massachusetts, USA) mixed with ethiodol 1:4 dilution ratio administered via the microcatheter (Figure ).\nPost-embolization angiography of the SMA demonstrated successful embolization of the posterior IPDA with sparing of the anterior IPDA. The patient became hemodynamically stable post procedure and no longer required blood products transfusion. His hemoglobin stabilized at 8.6 g/dl. The patient was successfully discharged to a skilled nursing facility to continue his recovery. At eight weeks follow-up the patient remained asymptomatic and he agreed to participate in this case report. |
A 58-year-old man had experienced recurrent abdominal pain and melena for 3 years, but repeated gastroenteroscopy and CTA had failed to find bleeding lesions.\nOn June 30, 2020, the patient presented to our department with a history of recurrent abdominal pain and melena for 3 years. In June 2017, he developed recurrent localized and dark pain in the upper abdomen without obvious inducement. It was tolerable, lasted for about 1 h, occurred about 3 times a day and was accompanied by thin, textureless melena, dizziness, and fatigue. His symptoms occurred repeatedly. He had gone to major hospitals across the country many times, but there was no clear diagnosis, and conservative treatment was used for alleviation. Two months before admission, the above symptoms progressively worsened. Melena was passed 4-5 times a day, with a total volume of about 200 g, and the dizziness and fatigue were significantly worse than before. After symptomatic treatment of hemostasis in the local hospital, the symptoms were improved, but still existed.\nHe was diagnosed with syphilis in 2008. Multiple treatments were successful, but the disease recurred shortly afterward. Because of heart valve disease, he underwent a mitral valve replacement and tricuspid valvuloplasty at Fujian Provincial Hospital In 2010. In 2019, he was diagnosed with atrial fibrillation and chronic pancreatitis. Currently, he is taking Betaloc 23.75 mg qd orally.\nThe patient had a history of unprotected intercourse with multiple sexual partners, a smoking history for 40 years (20 cigarettes a day), and a drinking history for 30 years. There is no family history of infectious or heredity diseases.\nOn admission, his temperature was 36 ℃, pulse was 66 beats/min, respiration was 18 breaths/min, and blood pressure was 120/80 mmHg. He was conscious and could cooperate in the physical examination. The skin of the whole body was slightly pale. Systemic superficial lymph nodes were not enlarged or tender, the conjunctiva was pale, the lips were slightly pale, carotid artery pulsation was normal, jugular vein was normal, liver jugular vein return sign was negative, and lung physical examination was not remarkable. There was a postoperative scar of about 20 cm in length on the chest, which had healed well. The heart rate was 80 beats/min, the heart rhythm was absolutely uneven, the first heart sound varied in intensity, a systolic murmur was heard in the mitral valve auscultation area, no obvious pathological murmur was heard in the other auscultation area, the fricative consonant was unintentionally included, the abdomen was flat and soft without tenderness or rebound pain, and the abdomen was not lumped. The liver and spleen were not touched, Murphy’s sign was negative, and there was no mobile dullness. Intestinal sounds (4/min) were normal.\nThe initial hemoglobin of the patient was 78.0 g/L and fecal occult blood was present (++). The syphilis toluidine red unheated serum test (TRUST) was positive; the syphilis titer was 1:2 positive. The white blood cell count, platelets, coagulation function, antinuclear antibody, antinuclear antibody profile, fecal routine, urine routine, alpha fetoprotein, carcinoembryonic antigen, glycogen antigen 199, total iron binding capacity, serum iron, unsaturated iron binding capacity, ferritin, folic acid, vitamin B12, purified protein derivative (PPD tuberculin test, tuberculosis antibody, and tuberculosis T-cell test showed no obvious abnormalities.\nThe patient’s upper abdominal magnetic resonance cholangiopancreatography revealed splenomegaly, and dilatation of the intrahepatic and extrahepatic bile ducts and pancreatic ducts (Figure ).\nAn electrocardiogram indicated atrial fibrillation (Figure ). Colonoscopy found multiple small ridges in the terminal ileum, and the pathology showed chronic active inflammation and erosive mucosa. Additional gastroscopy showed atrophic gastritis, and pathology of the gastric antrum indicated inflammation, and the Helicobacter pylori test was negative. Ultrasound gastroscopy showed dilatation of pancreatic duct with pancreatic duct stones, which was considered as chronic pancreatitis (Figure ). A capsule endoscopy was performed to further clarify the bleeding focus of the patient, and multiple intestinal erosions and ulcers were found (Figure ). The bone marrow puncture smear of the iliac spine showed normal hematopoietic tissue hyperplasia, presence of three-line hematopoietic cells, expanded islands of erythroid cells, hyperplasia of erythroid cells, a reduced granulocytosis ratio, and a scattered distribution of megakaryocytes, which was considered as hyperplastic anemia (Figure ). |
A 20-year-old, otherwise healthy, male presented to the Emergency Department (ED) after falling on his back while playing basketball. The patient landed on his right buttock on the hardwood floor. On presentation to the ED, he denied back pain, numbness, and weakness but did notice progressive swelling in the area of the fall. He denied any other injuries and had no previous bleeding or bruising history. His medical history was unremarkable for bleeding disorders or anticoagulation. He denied previous issues with hematomas, subcutaneous fluid collections, or abscesses.\nOn physical exam, the patient was not in acute distress and showed equivalent and intact strength and sensation in all four extremities. There was no weakness, numbness, or tingling in the lower extremities. All distal pulses were equal and palpable. The patient had mild pain in the right gluteal region with impressive swelling in the area. There was no lumbar, sacral, or pelvic tenderness to palpation. Point of care ultrasound demonstrated an 8 × 2.8 cm2 fluid collection in the right gluteal region which was incorrectly thought to be located within the muscle belly. He was given an ice pack, instructed to use Tylenol, and given a referral to Orthopaedics. He was instructed to return to the ED if the pain worsened or if he developed fevers, chills, nausea, vomiting, or other symptoms and was discharged the same evening.\nThe patient returned to the ED four days later for worsening pain and swelling of his buttock. He was unable to tolerate running and had worsening pain upon walking. The patient was concerned for a possible abscess at the injured area as it had become increasingly tender to touch since his previous visit to the ED. The skin at the area was intact and the patient denied drainage. He did not present with any constitutional symptoms including fevers, chills, nausea, vomiting, abdominal pain, lightheadedness, dizziness, or syncope. Physical exam was positive for myalgia in the right upper gluteal region. A complete blood count (CBC) revealed mildly microcytic RBCs of 78.3 fL (normal = 79.9–99.0), decreased mean corpuscular hemoglobin of 26.5 fmol/cell (normal = 27.0–32.0), and a decreased monocyte percentage of 5.8 (normal = 6.0–13.0). White blood cell count was 6.3 × 103 cells/mL (normal = 4.0–10.0), hemoglobin was 13.7 gm/dL (normal = 13.5–17.0), RBC count was 5.17 × 103 cells/mL (normal = 4.40–5.70), platelet count was 228 × 103 cells/mL (normal = 150–400), platelet thromboplastin time (PTT) was 25.1 seconds (normal = 22.0–32.0), prothrombin time (PT) was 10.9 seconds (normal = 9.5–12.0), and International Normalized Ratio (INR) was 1.0.\nA radiology-based musculoskeletal ultrasound was performed on the right upper gluteal region. The exam was notable for a large hypoechoic fluid collection deep to the subcutaneous tissue and superior to the gluteal musculature (Figures and ). The fluid collection had increased to 13 × 2 × 9 cm3. Internal echoes and fat globules were found within the hypoechoic fluid collection. These findings were suggestive of a Morell-Lavallée closed degloving lesion. Plastic Surgery was consulted and opted not to perform drainage of the lesion as there was no superficial skin breakdown. The patient was given a pressure dressing for the lesion and recommended to follow up with Plastic Surgery clinic for further management of the lesion. |
The case involved an 85-year-old female with congestive heart failure caused by severe aortic valve stenosis. Aortic valve replacement with a bioprosthetic valve was conducted electively. After the ascending aorta was unclamped, anesthetist noticed blood coming up from the endotracheal tube. Intraoperative flexible bronchoscopy confirmed massive hemoptysis coming up from all peripheral bronchi and mainly from the bilateral inferior lobes. The left chest was inspected through additional anterolateral thoracotomy connected perpendicularly to median sternotomy wound at 4th intercostal space. And the left hilum was exposed and lower bronchus was ligated externally to reduce the amount of intrabronchial bleeding. Repeated bronchoscopy showed persisting large-volume airway bleeding (Additional file : Video S1) and weaning cardiopulmonary bypass was impossible due to a reduced tidal volume of up to around 50 ml caused by severe peripheral airway obstruction. Groin cannulated veno-arterial ECMO with heparin coated circuit was initiated as a substitute for conventional cardiopulmonary bypass, and heparin was reversed precipitously with full dose of protamine sulfate. To treat the persistent airway bleeding, the chest wound was closed and the patient was shifted to a catheterization laboratory for bronchial artery embolization. Intraoperative total blood loss from the airway was over 4000 ml.\nAngiography confirmed massive bronchial artery bleeding into the left lower bronchus (Fig. ). However, diffuse bronchial artery bleeding was observed in the left upper lobe and right lower lobe as well. Embolization of the bilateral bronchial artery was conducted successfully. However, hemoptysis continued and repeated bronchoscopy confirmed ongoing diffuse bleeding. The right lower bronchus was blocked internally with an Endobronchial Watanabe Spigot (Nobatech, Cedex, France). Even after these aggressive interventions, diffuse hemorrhage persisted.\nEventually the amount of hemoptysis reduced on day four, and external ligation of the left inferior bronchus was finally released. The patient’s hemodynamics showed slight improvement on day five with, and a decision was made to convert to Venous-Venous ECMO. However, the patient crashed again and passed away from multi-organ failure on day six. |
A 43-year-old female, with no previous medical history except asthma, suffered a prolonged cardiac arrest secondary to respiratory distress due to a status asthmaticus. After the return of spontaneous circulation, following advanced cardio-pulmonary resuscitation, she was admitted to the Intensive Care Unit (ICU) where she developed persistent myoclonus. A magnetic resonance imaging scan of the brain and an electroencephalogram were performed in the following days, and she was diagnosed with severe hypoxic encephalopathy. After 16 days, her neurological situation had not improved, and the decision was made with the patient’s family for WLST. Once this decision had been made, the option of organ donation was presented to the family, who agreed to proceed after considering that organ donation was consistent with the patient’s principles and values. The hospital’s Donor Coordinator considered the potential cDCD donor as a suitable candidate for multiorgan procurement including the heart. Because heart recovery was contemplated, our hospital was contacted, and the potential donor was transferred to our center with authorization from the donor’s family after specific and detailed information about the cDCD procedure had been given to them.\nOnce the patient was admitted to our ICU, a Swan-Ganz catheter was inserted through the right jugular vein and a transthoracic echocardiogram was obtained as part of the heart evaluation. After the heart and the abdominal organs were evaluated and deemed suitable for transplantation, she was transferred to the operating room where two femoral cannulas, artery and vein, were inserted using a Seldinger’s technique. This technique was also used to place a catheter in the contralateral femoral artery that would be used during TA-NRP to measure arterial pressures. During these procedures, analgesia and sedation were adjusted according to the donor’s needs. Once the cannulas and the femoral artery catheter were in place, the donor was anticoagulated using unfractionated heparin as previously reported []. Brain activity was monitored using the Bispectral Index (BIS™) in order to guarantee adequate levels of sedation during the WLST [].\nOnce the surgical field was prepared, the family was brought into the operating room. WLST and end-of-life care was conducted following the hospital protocol and performed by the intensive care physician in charge of the patient’s care. Cardiac arrest was diagnosed by the absence of a pulse wave in the femoral artery, and death was declared after a five-minute no-touch period.\nFollowing the declaration of death, a sternotomy was performed, the pericardium was opened, the supra-aortic trunks were clamped and TA-NRP was started with an ECMO flow of 3 L/min/m2, aiming for a mean arterial pressure > 50 mmHg and a T > 35 °C. At that point, a norepinephrine infusion was started, reaching a maximum dose of 0.1 mcg/kg/min during organ procurement. Simultaneously, the donor was intubated, and mechanical ventilation was started using a FiO2 of 1, a PEEP of 5 and a tidal volume of 6–8 ml per kg of predicted body weight. We consider the warm ischemia time (WIT) as the time from significant hypoperfusion, defined by a systolic blood pressure < 60 mmHg, until TA-NRP is started. A WIT < 30 min for the heart and liver and < 60 for the kidneys is considered valid []. The WIT for our case was 16 min.\nOne minute after the start of the TA-NRP, a spontaneous effective heartbeat was observed with a normal sinus rhythm. Cardiac output was measured using both the Swan-Ganz catheter and a transesophageal echocardiogram, which also addressed cardiac contractility. The absence of cerebral blood flow was confirmed by transcranial Doppler performed both in the anterior and posterior territories. Brain activity was monitored using the BIS™ which showed values of 00 and a suppression rate of 100 throughout the TA-NRP procedure (Fig. ). Blood samples to determine arterial blood gas, lactate, troponin I, hematocrit, hemoglobin and liver function parameters were collected every 30 min during TA-NRP (Table ).\nOnce heart function was assessed, the ECMO flow was brought down until it reached 0 L/min/m2 15 min after its commencement. Because the donor had a history of severe asthma, SpO2 and PaO2 worsened after TA-NRP was stopped. For this reason, we decided not to wean the donor off ECMO completely, and a flow of 1 L/min/m2 was maintained during organ recovery. The duration of organ procurement was 120 min, and the heart, the liver and the kidneys were recovered and transplanted.\nThe heart recipient was a 60-year-old male diagnosed with amyloid cardiomyopathy. Due to the novelty of the case, the recipient had previously been informed of the peculiarities of the transplant which he agreed to by signing an specific informed consent were the different aspects of the procedure were detailed. Cold ischemia time was 55 min. The surgery was uneventful. He was easily weaned off cardiopulmonary bypass, and effective heartbeat was achieved after one defibrillation. He was then admitted to the ICU on low dose norepinephrine (< 0.2 mcg/kg/min) and low dose dobutamine (< 5 mcg/kg/min) and was extubated after 36 h. Follow-up echocardiograms showed a normal biventricular function. Five months after the transplant he has resumed a normal life. |
A 45-year-old Haitian American female with no significant medical history presented with a six-month history of progressive solid food dysphagia and a one-month history of odynophagia. The patient had lost 15 pounds and was only able to tolerate pureed food or liquids. She had experienced no symptomatic relief on omeprazole 40 mg twice daily for the past two months and did not use tobacco, alcohol, or illicit substances. Her initial blood count and metabolic panel were unremarkable. An esophagogastroduodenoscopy (EGD) revealed erythematous and friable mucosa with ulcerations in the proximal esophagus (). There was a stricture encountered at 15 cm from the incisors through which the gastroscope could not be traversed. Biopsies were taken from the inflamed esophageal mucosa and the proximal lumen of the stricture. Brush cytology was collected through the stricture as there was a concern for malignancy. A subsequent barium esophagram and upper GI series demonstrated 2 cm irregular narrowing in the cervical esophagus, but no abnormalities in the rest of the esophagus, the gastroesophageal junction, stomach, duodenum, or proximal jejunum. Contrast-enhanced computed tomography (CT) of the chest showed no acute esophageal, mediastinal, pulmonary, or cardiac pathology. The esophageal biopsy indicated acute and chronic inflammation with filamentous sulfur granules consistent with Actinomyces; rare fungal hyphal elements were additionally identified (Figures , , and ). The cytology was negative for malignant cells and the acid-fast bacilli (AFB) stain was negative, ruling out Nocardia as a potential pathogen. The patient was started on intravenous (IV) Penicillin for a diagnosis of esophageal actinomycosis and oral Fluconazole for a presumed Candida coinfection given concurrent fungal elements. Her human immunodeficiency virus (HIV) status was negative and her fasting blood glucose was within the normal range. She was discharged on Fluconazole 200 mg daily for 2 weeks and IV Penicillin G 3 million units every 4 hours for 6 weeks followed by oral Penicillin to complete a total of 6 months of antibiotics. Her esophageal culture eventually grew normal oropharyngeal flora and rare Candida albicans. The patient returned to the hospital 6 weeks later due to an acute right upper extremity deep venous thrombosis (DVT) associated with her peripherally inserted central catheter (PICC). Her odynophagia had improved but she was still not able to advance her diet. Repeat EGD was performed, which showed resolution of her esophagitis, but a remaining stricture in the proximal esophagus (). During an attempt to dilate the stricture, a small mucosal tear was induced. The dilation had to be deferred at that point pending mucosal healing. A repeat barium esophagram illustrated the previously identified stricture with no perforation. The patient's PICC was removed and she was discharged on oral amoxicillin 875 mg twice daily to complete the 6-month course. The patient was given a follow-up appointment in our GI clinic where she would be reassessed for esophageal stricture dilation. Unfortunately, she was lost to follow-up. |
A 13-year-old female patient with an avulsed right central incisor was referred to Ege University, School of Dentistry, Department of Restorative Dentistry and Endodontics after a motorcycle accident [Figures and ]. Avulsed maxillary central incisor had been kept in saline solution from the moment of trauma until its replantation 3 h later. The crown of the avulsed tooth was intact and the root had a closed apex. The intraoral and radiographic examination also revealed complicated crown fracture of the left central incisor []. No other oral injury was detected clinically. The left central and lateral incisors showed negative response to vitality test.\nThe patient was immediately anaesthetized, the alveolar socket was washed with saline solution to remove the blood clot and the avulsed maxillary right central incisor was replanted back into the alveoler socket with the help of finger pressure []. The tooth then splinted to the adjacent teeth with composite resin and the gingival lacerations were also repaired with sutures [Figures and and ]. The patient was instructed about her biting habits and oral hygiene implement. A 7-day course of systemic penicillin was prescribed, and the patient was referred to the medical practitioner for an antitetanus booster.\nThe sutures were removed one week later the replantation. The splinting was removed 10 days later and the root canal of the replanted tooth was mechanically prepared. Calcium hydroxide paste was used as an intracanal dressing during the endodontic treatment (6 months) to prevent inflammatory root resorption []. The intracanal dressing was renewed by one month intervals. Maxillary left central incisor with complicated crown fracture and left lateral incisor were also treated endodontically due to pulp necrosis [Figures and and ] and the teeth were restored with composite resin after root canal treatment [Figures and ].\nRadiographic and clinical examinations were performed during 27 months follow-up period. During the 15 months follow-up period, the tooth remained in a stable functional position and did not reveal clinical ankylosis or replacement resorption [Figures and ].\nThe replanted incisor developed mild infraocclusion (of about 1 mm) and replacement root resorption 21 months after the replantation [Figures and ]. Nevertheless, it remained functional stability and was aesthetically acceptable after adjusting the left central incisor with polishing discs []. Twenty-seven months after the replantation, the tooth still remained in a stable functional position. Infra-occlusion was about 0.5 mm [] and the radiographic image was not considerably different from the previous control radiography [].\nBoth the patient and her parents were satisfied with the treatment outcome and wanted to avoid complicated treatments, so it was decided to follow-up the case and keep the replanted incisor as long as possible. The patient and her parents were informed about the fact that infra-occlusion could become more severe as the patient grew and the complicated treatment (e.g., prosthetic solutions, implant) would be necessary if the root was totally resorbed. |
This is a 75-year-old man with a history of choroidal melanoma of the right eye diagnosed in 1984 and treated by radioactive Co-60 plaque. Thirty years later, he presented with progressive abdominal distention, early satiety, and weight loss of 20 pounds over a period of 6 months. He was seen by his primary care physician who requested a CT scan of the abdomen that showed a large hepatic mass measuring 34 cm by 26 cm, replacing the majority of the liver without retroperitoneal or mesenteric lymphadenopathy (). Hepatic tumor biopsy revealed metastatic melanoma consistent with his primary choroidal melanoma. While completing his diagnostic workup, the patient developed generalized weakness prompting his hospital admission due to acute renal failure, hyperkalemia, and spontaneous tumor lysis. He started hemodialysis promptly followed by the administration of weekly nab-paclitaxel 150 mg/m2 and then reduced to 75 mg/m2 thereafter due to severe neutropenia. The patient recovered his renal function as serum creatinine improved from 4.93 mg/dl to 0.69 mg/dl (normal values 0.60–1.20 mg/dl) and demonstrated clinical improvement of his generalized weakness, abdominal distention, and edema of the legs after three doses of nab-paclitaxel. A repeat abdominal CT scan one month after the therapy revealed a good response to treatment with significant decrease in tumor burden. This is donated by full clinical recovery and total resolution of tumor lysis manifestations. However, according to RECIST criteria, the response can be minimal followed by maintained stable disease. CT scan of the abdomen after 4 cycles of nab-paclitaxel revealed shrinkage of the hepatic lesion to 24 × 15 cm in maximum diameter (approximately 7% decrease in the largest lesion per RECIST criteria) (). This patient is still alive and continues to have excellent functional status, ECOG performance status of I, and no signs or symptoms of disease progression for 32 months now.\nOur patient with this metastatic uveal melanoma with extensive liver metastases with GNQ-209P mutation on the tissue biopsy (Figures and ) and undetectable mutations on the peripheral blood molecular profiling in serial follow-up samples suggests marked response to nab-paclitaxel. This can be understood by the dramatic tumor response on CT scans which was accompanied clinically by spontaneous tumor lysis syndrome followed by very prolonged disease control up to 30 months indicating nab-paclitaxel efficacy. All other patients with metastatic ocular melanoma, who did not have the GNQ-209P mutation, did not respond and did not have prolonged survival when treated with nab-paclitaxel.\nOur patient has received 8 cycles of Abraxane with initial minimal response followed by no increase and stable tumor size in the following imaging scans. In an attempt to achieve further response, the patient received an anti-PD-L1 in a clinical trial for 9 cycles. No further reduction in tumor size was achieved, and the patient was disqualified from the study after he developed sarcoidosis/interstitial pneumonitis. He was then restarted on Abraxane, achieving clinical and radiological stabilization of his disease with no major toxicities, and remains fully functional. He has received to date 12 cycles of Abraxane (in addition to the prior cycles of Abraxane received initially). |
A 79-year-old Chinese Han man with a history of squamous cell carcinoma of the esophagus was treated with surgical excision 3 months before admission. He denied having any family history of digestive disease.\nHe presented with progressive dysphagia and esophageal metallic stent placement (20 × 70 mm, the upper edge was 24 cm from the incisors) for anastomotic stenosis (27.5 cm from the incisors) 1 month before. At this time, he was transferred to our emergency department due to hematemesis and tarry stool. Abdominal computed tomography (CT) revealed only postoperative changes in esophageal cancer and esophageal metallic stent, which was placed close above the aortic arch (Fig. ). Upon arrival at the intensive care unit, the patient vomited approximately 500 mL of fresh blood. On physical examination, he was oriented and diaphoretic with a pale conjunctiva. He had no heart murmur, clear breathing sounds, and an old operative scar over the left chest, and the abdomen was soft without tenderness. Blood pressure (BP) was 72/56 mmHg, heart rate was 108 beats/min, respiratory rate was 28 breaths/min, and oxygen saturation was 91% in room air. Hemoglobin level was 6.8 g/dL. Large volumes of blood products were rapidly transfused, including 7.5 units of packed cells and 340 mL of fresh frozen plasma, and noradrenaline were administered simultaneously. When the patient's BP reached 90/60 mmHg, gastroscopy demonstrated large quantities of fresh blood and blood clots in the esophagus and stomach, so the source of the bleeding could not be identified. A vascular rupture was highly suspected. CT angiography (CTA) of the aorta was performed, which disclosed an aortic arch with possibly localized rupture (Fig. ). A cardiovascular surgeon performed an emergent angiography of the aorta, which showed bleeding from the wall of the aortic arch, so a diagnosis of AEF was confirmed, and an long covered stent (26 × 200 mm) was implanted (Fig. ) for thoracic endovascular aortic repair (TEVAR). The patient underwent fasting and received antibiotic treatment (intravenous cefodizime [2.0 g] once every 12 hours for 2 weeks and then replaced with intravenous combination of levofloxacin [0.5 g] once a day and piperacillin-tazobactam [4.5 g] once every 12 hours for another 2 weeks). The patient recovered well and was admitted to the department of gastroenterology on post-admission day 2. On post-admission day 9, esophagogastroduodenoscopy showed neither endoleakage nor peptic ulcer, and a jejunal feeding tube was established to provide enteral nutrition. Re-hematemesis occurred abruptly and massively on post-admission day 26, and the patient eventually died. The son of the patient provided informed consent for the publication of the case. |
A male patient, 67 years old, was admitted to the hospital via the emergency room, complaining of perianal pain he had been experiencing for the last month. The patient had also been suffering from constipation associated with hemorrhoids for a long time, and his symptoms, based on the criteria of chronic constipation, could be categorized as the Rome III criteria by using his medical history [,].\nAbout 30 years earlier, the patient had received injection sclerotherapy from an unlicensed therapist as treatment of his condition, but such injection caused fecal incontinence. Although the fecal incontinence was quickly resolved soon, constipation became even more severe and required medical treatment from other hospitals. Nonetheless, the symptoms did not improve; rather, they began to deteriorate about 10 years ago. Due to this progression, he received a hemorrhoidectomy twice at clinics of private practitioners. Notwithstanding such surgical procedures, the patient's symptoms deteriorated to the extent of having difficulty in defecation so that he had to use his finger, cotton rods, or tap water for evacuation of bowel contents.\nAt the time of visiting the emergency room, his blood pressure was 110/74 mmHg, his heart rate was 90 times/min, his respiratory rate was 16 times/min, and his body temperature was as high as 38.5℃. From the physical examination, abdominal tenderness was discovered, but other than that, there were no findings such as rebound tenderness or abdominal rigidity. On visual examination, fibrotic tissues of about 1.5 cm in size were found to be blocking the anal canal, so the doctor could not use his little finger for a digital rectal examination (). The patient had diabetes mellitus and benign prostatic hypertrophy as underlying diseases, and denied having any surgical history than the two hemorrhoidectomies about 10 years earlier. From hematological assay, the white blood cell count was 13,960/µL (neutrophils, 80.9%), hemoglobin was 14.9 g/dL, platelet count was 224,000/µL, and high sensitivity C-reactive protein was 7.74 mg/dL (reference range, 0 to 0.5 mg/dL). The abdominal computed tomographic scan taken at the time of visit to the emergency room showed fecal impaction in the colon and rectum with a large quantity, and focal perforation of the posterior rectal wall was confirmed by using the extraluminal gas shadow associated with soft tissue infiltration ().\nThe patient underwent emergency surgery based on the above examination results. Under general anesthesia, an anal examination was undertaken to check the conditions at the lithotomy position; then, a resection of the fibrotic tissues occluding the anus was performed, which enabled an approach through the anal canal. After the feces had been mechanically evacuated as much as possible, the rectum was washed using saline solution, followed by betadine solution. Thereafter, the patient was repositioned supine, and the sigmoid loop colostomy was constructed at the left lower abdomen for temporary colonic diversion due to rectal perforation. The patient was permitted to sip water on the second day after the operation, began to have a liquid diet on the third postoperative day, and was discharged on the fourth postoperative day. On the first postoperative visit to the out-patient department two weeks after the discharge, a digital rectal examination showed that feces still remained inside the anus. The patient had been undergoing conservative treatment with a warm-water Sitz bath at home, and the symptoms were quite improved in comparison to his preoperative conditions. |
Patient 10 (Tables and ), a 76-year-old female, was referred to the department of neurology in July 2012 because of progressing cognitive decline over the last 12 months, loss of weight, nausea, gait disturbance and tremor. She was seen on May 2011 for the first time by a neurologist with a 3-month history of dull holocephalic headache who ordered a cranial magnetic resonance imaging (MRI) and diagnosed a tension-type headache and a depressive disorder. Treatment with an antidepressant (duloxetine) was started. The patient experienced no improvement and a second examination by another neurologist was undertaken 2 months later. Again no focal neurological signs could be detected. Due to the weight loss, an occult neoplasm was suspected but not detected during an extensive inpatient investigation at a medical department during February 2012; however, the MRI showed bilateral white matter lesions (WML) and an old lacunar lesion located at the left striatum, the latter was not seen in the previous MRI from May 2011. Since the patient also suffered from mild hypertension, vascular encephalopathy was thought to be the cause of the progressive cognitive decline. Extensive neurocognitive testing was carried out in a rehabilitation centre in May 2012 and disclosed a severe decline of attention, memory and executive functions corresponding to subcortical dementia (Fig. ). When the patient was seen for a further diagnostic work-up at the SMZ-Ost-Donauspital in July 2012, the weight was 47 kg and a weight loss of 20 kg was reported over the past year. The gait was insecure with postural instability and with a tendency to fall when turning around. Frontal signs were positive, the voice was quiet, the tonus was mildly elevated and showed a slight hesitancy (“Gegenhalten”), tendon reflexes were brisk, paresis and pyramidal signs missing. There were no signs of ataxia, but a mild bradykinesia. Action tremor was more distinct than a mild resting tremor. Again, neurocognitive testing and gait disturbances were consistent with subcortical dementia (Figs. and ). Regarding the mild signs of parkinsonism, dementia with Lewy bodies (DLB) was also suspected but excluded by a dopamine transporter (DAT) scan. Fluorodeoxyglucose positron emission tomography (FDG-PET) demonstrated hypometabolism in the left striatum and in the left frontotemporal cortex (Fig. ). Cerebrospinal fluid (CSF) showed signs of a chronic lymphocytic inflammation. The CSF markers for dementia, total tau protein and phosphor-tau were within the normal range, while beta-amyloid 1-42 and the Innotest-amyloid-tau index (IATI) were found to be below the reference values (beta-amyloid 1-42: 290 pg/ml, reference value > 500 pg/ml; IATI 0.6, reference values > 1). Finally, LNB was diagnosed when further CSF examinations disclosed a highly elevated Bb-specific-AI indicating local intrathecal Bb-specific antibody synthesis (Table ). The patient was treated with 2 g ceftriaxone daily for 3 weeks.\nNeurological symptoms and impaired cognitive functions, although persistent for a year, recovered rapidly within a few weeks (Figs. , and ) and so did the pathological CSF findings (Table ). A follow-up FDG-PET examination showed the left frontotemporal hypometabolism in remission, while this was not the case for the cystic lacunar lesion in the left striatum. A new and clinically silent small right thalamic lesion was detected that was not present in the pretreatment MRI (Fig. ). The Consortium to Establish a Registry for Alzheimer’s Disease (CERAD) test battery at the last follow-up in April 2014 scored within the age-dependent normal range with the exception of verbal learning and semantic verbal fluency (Fig. ). In a telephone call in February 2018 at the age of 82 years, the patient reported no gait problems or cognitive impairment and had just returned from a trip to Cuba. |
A 22-year-old woman was referred to our institution with a past medical history of wedge resection of the left lung for two incidentally detected pulmonary nodules at another hospital fifty months earlier. The initial outside multi-phase dynamic CT scan obtained at that time using a multidetector CT scanner (Sensation 16, Siemens, Erlangen, Germany) after intravenous injection of contrast media was available for review, although the information on the amount and the brand name of the contrast media was not available. The CT images revealed two nodules each in the left upper and lower lobes. These nodules were round in shape with a well-circumscribed margin, and maximum diameters of 22 and 20 mm, respectively. On non-contrast CT scan, the nodules revealed no calcification. On multi-phase dynamic CT images, the nodules showed peripheral nodular enhancement at the early arterial phase obtained 1 minute after intravenous administration of contrast media and a subsequent "central filling-in" enhancement pattern on the delayed scans (obtained 2 and 4 minutes after contrast enhancement) (), which was identical to the well-known enhancement pattern of hemangiomas of the liver. Wedge resection of those left pulmonary nodules was performed at the other hospital, and the review of the pathologic specimen revealed pulmonary capillary hemangiomas.\nAt 50 months follow-up, new pulmonary nodules were detected on a chest radiograph, and the patient visited our institution for further evaluation. The CT scan obtained at our institution using a multidetector CT scanner (LightSpeed VCT, GE Healthcare) after intravenous injection of 100 mL (3 mL/s) of nonionic contrast media (Iopamiron 300; Bracco, Milan, Italy) revealed two recurrent pulmonary nodules each in the left lower lobe and the right lower lobe with maximum diameters of 30 and 12 mm, respectively. These nodules also showed a round shape with a well-circumscribed margin. The larger one detected in the left lower lobe especially showed the prominent "peripheral nodular enhancement" pattern at post-contrast CT images obtained 40 seconds after contrast administration (), which was the same as the previous CT finding.\nA left lower lobectomy and wedge resection of the right lower lobe was performed, and the gross specimens of the resected nodules showed well-demarcated hemorrhagic or vascular nodules (). Histopathologic examination revealed vascular tumors of a capillary growth pattern. These nodules were composed of anastomosing capillary vascular channels filled with red blood cells. These capillary lumina were divided by narrow trabeculae of hyaline stroma (), which were identical to those of the previously resected pulmonary nodules at the other hospital. The final diagnosis was recurrent pulmonary capillary hemangiomas, and the tumors showed no evidence of histologically malignant features such as cellular atypia or mitosis. |
A 36-year-old Indian female who had immigrated to Saudi Arabia 3 years prior was brought to the emergency department after a 3-min episode of generalized tonic–clonic seizure that occurred during sleep 6 days after the patient had a normal vaginal delivery at 37 weeks of gestation. In the postictal state, she was drowsy for about 15 min and did not complain of headache, fever, dizziness, vertigo, visual disturbance, nausea or vomiting. The patient had no history of chronic cough, chronic diarrhea, weight loss or decreased appetite and had no history suggestive of diabetes, hypertension or tuberculosis. The patient's most recent trip was to India 1 year prior. She had a history of epilepsy when she had two episodes of generalized tonic–clonic seizure at the age of 8 years and was treated with carbamazepine for 2 years, after which medication was stopped because there were no seizure episodes.\nOn physical examination, the patient was alert, conscious and cooperative, with a Glasgow coma scale of 15/15. Her cardiovascular, respiratory, musculoskeletal and gastrointestinal systems were functioning normally. Neurological and fundoscopic examinations were normal, with no signs of meningeal irritation.\nThe complete blood count, erythrocyte sedimentation rate, electrolytes and blood sugar levels were within the reference range. The results of a purified protein derivative skin test (Mantoux test) were negative. Chest X-ray and electroencephalogram were normal. The patient refused to receive a lumbar puncture. Stool examination was normal.\nComputed tomography (CT) scan of the head revealed multiple calcified nodular lesions [Figure and ]. T1-weighted magnetic resonance imaging with contrast showed a ring-enhancing lesion in the left frontoparietal area, which informed suspicions of NCC []. Because the suspected NCC presented during the patient's postpartum period, venous sinuses thrombosis was considered as a possible diagnosis; however, absence of headache or blurred vision ruled this out. While a recurrence of the patient's epilepsy, triggered by the stress of her delivery, was also suspected, the serum (qualitative) enzyme-linked immunosorbent assay (ELISA) results having returned positive for immunoglobulin G (IgG) antibodies for cysticercosis – in addition to the CT findings – excluded this diagnosis, and thus, a diagnosis of intraparenchymal NCC was confirmed.\nThe patient was administered dexamethasone (4.5 mg/kg intravenously every 6 h) for 4 days to alleviate the intraparenchymal inflammation and perilesional edema in addition to levetiracetam 500 mg orally twice daily. This treatment resulted in successful control of her seizures during the hospitalization period. Ophthalmology consultation confirmed the absence of any orbital involvement. At the 3-month follow-up, the patient's condition had significantly improved, and no seizure recurrence was observed. |
A 71-year-old African American woman with a past medical history of uterine cancer and no medical history of diabetes mellitus presented after a ground-level fall and a syncopal episode. She was initially admitted to the trauma service but transferred to the internal medicine service after being cleared by trauma. Her electrocardiogram showed significant ST depression, and laboratory investigations revealed markedly elevated troponin (13.67 ng/mL) as well as acute kidney injury. She was admitted with a diagnosis of non-ST segment elevation myocardial infarction, but an urgent cardiac catheterization revealed normal coronaries with an ejection fraction of 70%. Her initial venous BG was noted to be 128 mg/dL, while a bedside POC BG, when checked around the same time, was 453 mg/dL. Her POC-BG the following day was reported to be consistently higher than 500 and 600 mg/dL. Her beta hydroxybutyrate (BHB) level was elevated (>13.5 mg/dL) with high anion gap metabolic acidosis (anion gap of 40). She was presumed to be in DKA and was started on the DKA protocol with insulin infusion. Two hours after starting the DKA protocol, a “code stroke” was called on the patient with reports of left-sided facial paralysis, right hemiparesis, and decreased responsiveness. Urgent brain computed tomography (CT) did not reveal any acute changes, and CT angiogram showed normal perfusion. While in CT, she developed ventricular fibrillation arrest, for which she received cardiopulmonary resuscitation with return of spontaneous circulation. She was later intubated for airway protection. She developed cardiogenic shock following cardiac arrest, which required multiple pressor support. She was also started on continuous renal replacement therapy for acute kidney injury but with very minimal ultrafiltrate retrieval due to hypotension. While she remained on the DKA protocol for 2 hours, her venous BG was consistently and persistently low with readings ranging between 16 and 34 mg/dL, while simultaneous POC-BG reading were consistently high at 500 to 600 mg/dL. Insulin infusion was stopped after the discrepancy was noted but dextrose water (D5W) was not started immediately even though venous glucose was reported to be low. The insulin infusion was withheld for 6 hours after the discrepancy was noted but was restarted after 1 of the POC-BG read of >600 mg/dL. An endocrine consult was requested the next day, the insulin drip was discontinued permanently, and continuous D5W was started at this point. Unfortunately, the patient had sustained prolonged hypoglycemia for about 18 hours before D5W was started (see ).\nSurprisingly her HbA1C remained normal 5.1% (32 mmol/mol). Brain MRI brain was suggestive of hypoxemic ischemic encephalopathy, and unfortunately, she continued to deteriorate neurologically, with minimal purposeful response. She coded again a few days later with pulseless bradyarrhythmia. Cardiopulmonary resuscitation was initiated but resuscitation was called off upon family request, and the patient was pronounced deceased after a complicated 8-day hospital course.\nThe strong discrepancy between venous and POC-BG values led to a suspicion of interference with the POC glucose meter. We performed a retrospective analysis of the case with a thorough chart review and discovered that patient had been on IV high-dose vitamin C infusion as a natural remedy for her uterine cancer. This was confirmed via a telephone conversation with her son. She received her last dose one day prior to presentation at our hospital. He also reported that the patient had been losing a lot of weight recently with poor oral intake and progressive weakness.\nIndeed, we realized that this was an instance of a mismanaged case, and we alerted our hospital administration about changing POC-BG meters that were being used in our institution. Unfortunately, as of now, we do not have any alternate POC-BG meters in the hospital. We presented this case as a morbidity and mortality review with our faculty, residents, and hospital administrators and hope to be able to introduce safer and more reliable POC-BG meters in the future. We are also planning to design a questionnaire for asking patients about their recent use of medications that can interfere with POC-BG meters. |
Patient III:1 in Family 1 was a 27-year-old male and first child of consanguineous healthy parents (first degree cousins) of Lebanese ancestry. Since childhood, he has had thin muscles especially affecting the lower legs without apparent weakness. Between 20 and 24 years of age, he had experienced episodic muscle pain, weakness and fatigue. These episodes were most evident in his arms and legs in the beginning of the day, lasting from one month to several months and without any apparent triggering or associated factors. Neurological examination at age 27 revealed no muscle weakness, but the lower legs were thin, the medial parts of the gastrocnemius muscles were atrophic (). He had a wide neck, anteverted shoulders, prominent trapezius and latissimus dorsi muscles and a tendency to thoracic kyphosis. He had slight shortening of the Achilles tendons, and slight contractures of his long finger flexors. Slight syndactyly was observed between fingers and a tendency to sandal gap widening between the first and second toe. EMG of the vastus lateralis was borderline myopathic as found on the turns amplitude analysis of the interference pattern. EMG was normal in the deltoid muscle and the interosseus dorsi muscles of the hands. At age 20 years, he was examined because of recurrent sharp chest pains. His ECG showed Q waves in aVL, I and aVR, T-inversions in aVF and III and increased QRS amplitudes in the chest leads, suggesting left ventricular hypertrophy. A cardiopulmonary exercise test (CPET) was prematurely terminated due to leg fatigue and showed reduced exercise capacity, 192 W, 70% of the reference value. Twenty-four-hour Holter monitoring and echocardiography were normal. A cardiac MRI (CMR) at age 27 showed increased basal wall thickness (30 mm) consistent with hypertrophy and a small ventricular diverticulum, but otherwise adequate systolic ventricular performance and normal valvular function. There were no signs of atrial abnormalities.\nLaboratory tests revealed a persistent increase in high-sensitive cardiac troponin T 20–30 ng/l (reference value <14 ng/l), serum creatine kinase (CK) 18–22 μkat/l (reference interval 0.80–6.7 μkat/l), CK-MB 32–36 μg/l (reference value <5 μg/l) and myoglobulin 178 μg/l (reference value <90 μg/l). N-terminal prohormone of brain natriuretic peptide (NT-proBNP) was normal 130–250 ng/l (reference value <300 ng/l).\nMRI showed a symmetrical representation of all muscles. In the right semimembranosus muscle, there was a marked high signal on the fat-suppressed T2-weighted sequence representing oedema, with a slight fatty infiltration in the medial half of the muscle. On the left, there was a superficial marginal oedema in the same muscle without fatty infiltration. Otherwise, no evidence of intramuscular oedema or fatty degeneration could be found. The distribution of the muscle bulk corresponded with the clinical assessment, and there were no signs of fatty replacement of the relatively thin muscles of the lower legs and distal arms. |
An 87-year-old woman visited the emergency department for worsening severe dyspnea and chest discomfort. On admission, the blood pressure was 150/90 mmHg, the respiratory rate was 36 per minute, the pulse rate was 112 per minute, and the peripheral oxygen saturation was 80%. Fine crackles were auscultated in both lower lung fields and an apical grade 2/6 holosystolic murmur was present. A chest radiograph revealed cardiomegaly and pulmonary congestion (). The electrocardiogram showed voltage criteria consistent with LV hypertrophy, and T wave inversion in leads V5 and V6 was compatible with LV strain ().\nThe CK-MB and troponin-I levels were normal (1.87 and 0.022 ng/mL, respectively). The B-type natriuretic peptide was markedly elevated (1,850 pg/mL). Other laboratory parameters were unremarkable.\nThe patient had a history of an acute MI during a previous admission; the electrocardiogram showed ST segment elevation in leads III and aVF and Q waves in leads II, III, and aVF. Coronary angiography revealed severe coronary artery stenoses (total occlusion of the proximal right coronary artery, total occlusion of the proximal left circumflex artery, and a 40% stenotic lesion in the distal left main artery). Thus, primary percutaneous coronary revascularization of the right coronary artery was performed.\nTwo-dimensional echocardiography at the previous admission showed decreased LV systolic function (ejection fraction, 40%) and a mild pericardial effusion. The regional wall motion abnormalities with akinetic basal to the mid-inferior and posterolateral walls of the LV were observed. In a color Doppler study, mild mitral regurgitation was noted in systole. The continuity of the myocardium of the mid-posterior wall was disrupted and a small sac (22 × 11 mm) with a narrow neck was seen which was suspected to be a rupture of the free wall with a thrombotic plug (). A LV pseudoaneurysm was diagnosed and contrast echocardiography was performed to evaluate further blood leakage through the ruptured myocardium and sac. Contrast echocardiography revealed that the pseudoaneurysm on the LV posterior wall was clearly defined and did not communicate with the pericardial space (). Cardiac magnetic resonance imaging (MRI) also showed a small bulging sac-like lesion with a neck portion in the mid-posterior wall of the LV without definite myocardial tissue ().\nThe patient and her family declined to undergo surgery for the LV pseudoaneurysm. The patient was discharged after a few days of medical therapy and did not return for follow-up.\nDuring the admission, two-dimensional echocardiography revealed an increase in the size of the LV and decreased LV systolic function (ejection fraction, 30%). A large cavity in the posterior area of the mid-posterior wall of the LV (> 80 × 55 mm) was noted which was diagnosed as a small LV pseudoaneurysm 1 year earlier (). Blood flow across the hole from the LV to the cavity in systole () and from the cavity to the LV in diastole was observed by color Doppler study (). Mild-to-moderate mitral regurgitation in systole and diastole was noted in a color Doppler study () that was more severe compared with the previous admission. A small LV pseudoaneurysm after an acute MI progressed to a huge pseudoaneurysm with significant mitral regurgitation in 1 year without any treatment. Surgical management was strongly recommended, but she declined again and was discharged when dyspnea had improved with medication for congestive heart failure. |
A 77-year-old woman presented with a two-month history of increasing dyspnea on exertion, fatigue, and severe anasarca extending into the lower extremities. She has no other complaints except for mild symptoms associated with postprandial GE reflux, mild paroxysmal nocturnal dyspnea, and orthopnea. The patient denied any history of cardiac or respiratory issues, as well as fevers, night sweats, and unintentional weight loss. During physical examination, vital signs were stable except for an oxygen saturation of 92%, indicating hypoxia. No abnormal physical examination findings were observed in the patient other than pitting edema, moderate erythema, and warmth of the lower extremities. Laboratory studies, including a complete blood count and basic metabolic panel, were within normal limits. A subsequent chest radiograph demonstrated pulmonary edema, a small left pleural effusion, and a prominent hiatal hernia (Figure ).\nA CT scan of the chest was performed, which demonstrated a type IV hiatal hernia involving the stomach and a portion of the pancreas, which also displaced the aorta and inferior vena cava (Figures , ).\nThe patient was then admitted to the hospital for further work-up and appropriate treatment of hypoxia, as well as pitting and peripheral edema. The patient underwent a transthoracic echocardiography during her hospital course where a preserved ejection fraction was appreciated. Her symptoms of increased dyspnea on exertion, postprandial GE reflux, and mild hypoxia were attributed to chronic aspiration pneumonia secondary to the massive hiatal hernia. Oral diuretics were used to treat the patient’s peripheral edema. The massive hiatal hernia displacing the aorta and inferior vena cava seen on the CT scan of the chest could be a factor in her symptoms; however, it was not considered one the sentinel cause. The patient was kept on oral diuretic therapy and nasal cannula oxygen during her hospital stay, and later referred to gastroenterology and pulmonology for evaluation of her type IV hiatal hernia. |
A 16-year-old, 60 kg, female patient presented to our plastic surgery department for cosmetic correction of hollowness of right cheek. Past history revealed the presence of hemifacial dysmorphism with speech and swallowing difficulty since birth with decreased neck movements as the child grew. As a child, the patient had undergone seven corrective surgeries elsewhere. During the last surgery, done 8 years back for correction of nasal deformity, patient was mechanically ventilated postoperatively for 48 h in view of aspiration of blood, leaving a young child traumatized mentally. Past medical records revealed the absence of any cardiac anomaly. The detail of techniques followed for securing the airway during previous surgeries was not mentioned in discharge summaries.\nPhysical examination revealed several facial deformities, prominently on the right side of face with unilateral mandibular hypoplasia. Mandibular protrusion was restricted, micrognathia was significant with an inter-incisor gap of about 3 cm. Unilateral ankylogossia present on the right side prevented protrusion of the bulky tongue, attached to the floor of the mouth with a fleshy septum giving rise to speech and swallowing difficulty []. The neck was short with a fixed flexion deformity restricting neck movement []. Hematological and biochemical investigations were unremarkable, along with normal 12-lead electrocardiogram.\nWe anticipated difficulties in securing airway and intubation through the nasal route, retrograde intubation and surgical route were all contraindicated. Despite the presence of a difficult airway, awake procedures were not possible as our patient refused to cooperate.\nAn 8 h of fasting were advised and patient received tablet ranitidine 50 mg and tablet metoclopromaide 10 mg 1 h before shifting to the operating room. She was nebulized with 4% lignocaine, preoperatively, for 20 min. Inside the operating room; monitoring was established as per American Standards Association standards. Injection glycopyrrolate 0.2 mg was administered after securing intravenous access. Lignocaine spray (10%) was administered over the posterior pharyngeal wall, but glossopharyngeal nerve, superior laryngeal nerve, and transtracheal blocks could not be given in view of unidentifiable anatomical landmarks. Patient was given supplemental oxygen and 1 mcg/kg of injection dexmedetomidine was administered intravenously over 10 min, followed by infusion at the rate of 0.2 mcg/kg/min. Although patient had effective spontaneous ventilation, she did not allow significant airway manipulation at this dose. Thus, we gradually increased the dose of dexmedetomidine infusion up to 3 mcg/kg/h. After the patient was adequately sedated, oral fiberoptic bronchoscopy was done, 2 ml of 2% lignocaine was sprayed over the glottic opening and the patient was successfully intubated with a 7.0 mm flexometallic endotracheal tube. After securing the airway, dexmedetomidine infusion was stopped and the patient was given 100 mcg of injection fentanyl, 80 mg of injection propofol and neuromuscular blockade with intravenous atracurium 30 mg. Anesthesia was maintained with nitrous oxide (66%) and oxygen (33%) along with propofol infusion. The intraoperative course was uneventful. At the end of surgical procedure, which lasted for 1.5 h, neuromuscular blockade was reversed and patient's trachea was successfully extubated. |
A 24-year-old male, who has been using cannabis in dependent pattern presented with an insidious onset and continuous illness of 2 years' duration, characterized by the delusion of reference, delusional percept, delusion of love, auditory (commenting, commanding, and discussing), and tactile hallucinations. At presentation, on mental status examination, he was untidy and ill-kempt, had blunt affect; however, there was no formal thought disorder. When asked about his psychopathology, he ascribed the voices heard to one of his female teacher, who would express her love toward him in the conversations heard as part of the auditory hallucinations. He also ascribed the tactile hallucinations to the same teacher. He described this as a sensation of vibration, which he would feel all over his body, more so in his thighs, and genital region. This would occur mainly at night, when he would be fully awake or asleep and alone, would feel aroused, and simultaneously hear the voice of his teacher claiming to be responsible for these sensations. As per him, he would hear that she is in deep love with him and wanted to have sexual intercourse with him, would feel his penis to be touched, and in the process would have erection and occasional ejaculation. He would be unable to describe exactly how she would be doing this but was convinced that it was she who was doing so. He also believed that his body is under the control of his teacher, who forces him to indulge in sexual activities, against his will. He would deny drawing any pleasure out of these activities and would say that he was not able to avoid this experience. This experience was not associated with any active cannabis use or abstinence from cannabis. At the time of mental status examination, his cognitive functions were preserved, and he had poor insight.\nDue to all these symptoms, he had to discontinue his education and became homebound. Based on the available information, a diagnosis of schizophrenia and cannabis dependence syndrome was considered, and he was sequentially treated with olanzapine, risperidone, Trifluoperazine, and the combination of olanzapine and fluphenazine decanoate, in adequate doses for adequate duration. He showed 40%–50% improvement in auditory hallucinations but no significant change in tactile hallucinations with these medications. Finally, he responded to clozapine in the dose of 200 mg/day. After being treated with clozapine, his insight improved, he would acknowledge that all his previous symptoms were unreal and part of the illness. |
A 38-year-old man was found to have aortic root dilatation and moderate aortic valve stenosis with bicuspid valve by transthoracic echocardiography. Enhanced computed tomography () showed aortic root dilatation (55 mm), right aortic arch, and an abnormality at the origin of the left subclavian artery (LSCA), which were diagnosed as KD. The diameter of KD including the aorta was 40 mm, longitudinal diameter of KD was 16 mm, diameter of the ascending aorta was 40 mm, aortic arch had severe angulation, and arch vessels measured 6 to 7 mm. We therefore decided to perform aortic root reconstruction and total arch replacement (TAR) using FET technique for KD.\nWith the patient in supine position, a horizontal incision was made below the bilateral clavicle to expose the axillary arteries, and both were connected to an 8-mm graft. The mediastinum was accessed via a median sternotomy. An arterial cannula was inserted into the artificial blood vessel connected to the right axillary artery and the right common femoral artery. A cannula for venous drainage was inserted into both the superior and inferior vena cava from the right atrium, and total extracorporeal circulation was established. A left ventricular vent tube was inserted from the right superior pulmonary vein, and a guidewire was inserted through the femoral artery and advanced to the ascending aorta.\nWe used a knitted quadrifurcated graft (InterGard™ Quadrifurcated, MAQUET Cardiovascular, La Ciotat, France) to reconstruct the four cervical branches. After the rectal temperature decreased to 28°C, the left common carotid artery (LCCA) was ligated and transected, and an end-to-end anastomosis was performed using the inner leg of the quadrifurcated graft. The right common carotid artery was anastomosed in the same way. The LSCA was ligated and clipped, and cerebral perfusion was initiated through the artificial blood vessel connected to the left axillary artery. After the ascending aorta was opened, cardiac arrest was obtained by retrograde cardioplegia. Then, the ascending aorta was incised longitudinally to the origin of the LCCA, and the proximal part of the aortic arch was transversely dissected. We inserted the device for the FET (Frozenix®, Japan Lifeline, Tokyo, Japan) using a guidewire under trans esophageal echocardiography (TEE) guidance. A synthetic graft with one branch (which was part of the valsalva graft) was anastomosed end-to-end to the stump of the distal aortic arch before antegrade systemic perfusion was begun. A composite graft consisting of valsalva graft and mechanical valve was used to replace the aortic root. Then, the proximal and distal valsalva grafts were sutured in an end-to-end fashion. The main body of the quadrifurcated graft was sutured to the valsalva graft in an end-to-side fashion. Finally, the outer legs of the quadrifurcated graft were anastomosed to the grafts that were connected to the axillary arteries in an end-to-side fashion. Surgical time was 655 min, cardiopulmonary bypass time was 379 min, cardiac arrest time was 245 min, and rectal temperature was 27.5°C. Postoperative computed tomography (CT) showed good exclusion of the KD, and the arch branches were reconstructed in the shape of an antler (). The postoperative course of the patient was uneventful and he had no complications at the time of discharge. |
A 54-year-old male patient was admitted to the Zhejiang Cancer Hospital on October 16, 2007, due to a phyma at the left frontal brow. The phyma was 1.5 × 1.0 cm in size, with mild tenderness and no local redness. The patient presented with no abnormal coughing, sputum or fever. A surgical resection was performed on October 26, 2007, and the phyma was identified as a left frontal metastatic poorly-differentiated adenocarcinoma by pathology (). Further examinations were performed in order to identify the primary tumor, and a chest computed tomography (CT) scan disclosed the presence of two lesions. One was a lump shadow with a small burr of ~2.0 cm in size in the upper right lung, while the other was also in the upper right lung, but was a patchy shadow with an unclear edge, thereby indicating a high possibility of lung cancer (). Using emission computed tomography (ECT), several lightly visible restricted radioactivity concentration sites were detected at the left temporal bone, the 7th and 10th thoracic vertebra and the right ileum, while no evident abnormalities were observed in the brain. There were also no evident abnormalities in the pancreas, spleen, kidney or double-adrenal gland, which were examined by an ultra-B scan. The gastroscopic results and blood carcinoembryonic antigen (CEA) levels were normal and the patient had no history of smoking or high blood pressure.\nThe general health of the patient was fine and the performance status (PS) was scored as 0. A surgical scar was observed on the left frontal area of the face. There was no enlargement of the two supraclavicular lymph nodes and heart and lung auscultation heralded negative results. No abnormality was observed by abdominal examination, routine blood tests, blood biochemical examination or routine urine tests. The patient was diagnosed with lung cancer combined with subcutaneous T3N0M1 metastasis.\nThe patient was selected for the SAiL clinical trial. According to the SAiL scheme, the patient was initially administered 15 mg/kg (900 mg)d1 bevacizumab monoclonal antibody (mAb), 175 mg/m2 (300 mg)d1 paclitaxel and the area under the concentration time curve 6.0 (770 mg)d1 carboplatin.\nGrade I bone marrow suppression and peripheral nerve toxicity and grade III hair loss and epistaxis developed following the chemotherapy. Following two chemotherapy cycles, the tumor in the upper right lung was observed to be slightly shrunken by chest CT, with the therapeutic evaluation of a stable disease (SD; ). On January 11, February 1, February 28 and March 25, four chemotherapy cycles were applied as planned, resulting in six cycles in total. Compared with the pre-treatment evaluation, the therapeutic evaluation was of SD (). On April 23, 2008, the patient was initially administered 15 mg/kg (900 mg/d1) bevacizumab mAb and then maintenance treatment every 3 weeks (). The final bevacizumab mAb treatment was administered on January 16, 2012. The total bevacizumab mAb treatment time lasted 50 months, with a cumulative dose of 54 g. During the bevacizumab maintenance treatment, the patient was examined by chest CT every three months and by brain magnetic resonance imaging (MRI) every six months for the therapeutic evaluation. On January 16, 2012, the efficacy evaluation review using CT scans revealed the stable status of the tumor (). The patient required to be withdrawn from the SAiL clinical trial due to long-lasting 3+ proteinuria. On May 23, 2012, a chest CT indicated that the upper right lung lesions were significantly increased compared with the previous data (). The patient did not cough blood and had no fever, sputum, chest pain, hemoptysis, chest tightness, shortness of breath or headache. No abnormalities were identified from the brain MRI, bone ECT or abdominal CT, and the blood CEA was 33.2 ng/ml. A lung tumor biopsy was performed and cancer cells were identified. Considering the progress of the disease, the PFS was 54 months. On May 31, 2012, one chemotherapy cycle of gemcitabine (1250 mg/m2) combined with cisplatin (75 mg/m2) was administered and the patient developed grade III neutropenia and grade IV platelet decline. On July 17, 2012, a palliative treatment was performed on the right-sided lung cancer. Pathological lung nodules with poorly-differentiated adenocarcinoma cells combined with disintegration and necrosis were discovered forming two tumors, one small, sized 2 × 2 × 1.5 cm, and one large, sized 3.5 × 3 × 2.8 cm. Tumor invasion of the visceral pleura, infiltration and metastasis statuses were: bronchial roots lymph node 1/6, intrapulmonary bronchial lymph node 1/2, the 7th groups of lymph nodes 0/2, the 9th groups of lymph nodes 0/1 and the 10th lymph nodes 1/4 (). G/C chemotherapies were performed on August 15, and September 6, 2012. The current Eastern Cooperative Oncology Group (ECOG) PS score was 1. Approval for the study was obtained from the ethical committee of Zhejiang Cancer Hospital and informed consent was obtained from all the participants.\nDuring the bevacizumab treatment there was no sign of hemoptysis. During the 1–2 cycles of chemotherapy, grade I epistaxis was observed, but no high blood pressure. During cycles 1–17 of bevacizumab treatment, there was no significant proteinuria. Weak positive proteinuria was diagnosed from the start of the 18th cycle, but from cycles 33–39, the proteinuria was 1.0–2.0 g/l (++). Following several 24-h interval examinations, the total urine protein was shown to peak at 1.38 g/24 h. During cycles 40–57, the proteinuria was 2.0–4.0 g/l (+++). The total urine protein measurement at the 57th cycle was 2.284g/24 h on January 17, 2012. The urinary protein level in the patient demonstrated a positive correlation with the cumulative dose of bevacizumab (P=0.000; ). Subsequent to withdrawing the medication and pausing for more than a month, proteinuria was diagnosed [<0.15 g/l (−)] on May 25, 2012, and was kept at a constant negative from then on. In this patient with advanced non-small cell adenocarcinoma and a PS score of 0, bevacizumab combined with paclitaxel and carboplatin was able to stabilize the tumor condition. The bevacizumab maintenance therapy lasted for 50 months. At three months after treatment withdrawal the tumor progression restarted. Overall, the bevacizumab therapy demonstrated a good long-term effect with the major adverse reaction of proteinuria correlating with the drug cumulative dose, which may be completely restored following withdrawal. |
A 54-year-old woman was admitted to the hospital because of blood in the sputum and hemoptysis.
A 54-year-old woman was admitted to the hospital because she had seen blood in her sputum for more than 4 mo and had had hemoptysis for 1 wk. The hemoptysis occurred without a clear reason. The coughed-out blood was bright red in clots and was accompanied by a large quantity of white and nonviscous sputum. The patient did not sweat excessively either during the day or night, did not feel cold or have a fever, did not feel chest tightness or shortness of breath, and had no dizziness or headache.
The patient had undergone surgical removal of gallbladder polyps more than 20 years ago, and had received minimally invasive surgery for breast fibroma more than 2 years ago.
The patient had no history of smoking or consuming alcohol, and there was no family medical history.
On admission, the patient's temperature was 36.7°C, with a heart rate of 78 beats/min, blood pressure of 110/68 mmHg, and respiratory rate of 16 breaths/min. The patient exhibited clear consciousness and compliance throughout the examination. Her trachea was in a neutral position, there was no cyanosis in her mouth or lips, and no swollen lymph nodes were identified in the supraclavicular region of either side. The breathing sound of both lungs was clear; no obvious rale was heard. The heart rhythm was regular; no obvious pathological noise was heard. The patient’s abdomen was flat and soft; no tenderness or rebound pain was identified. Edema in her lower extremities was not evident, and no clear pathological signs were found in the nervous system.
Her hemoglobin level was relatively low at 98 g/L; the remaining routine blood examination results, C-reactive protein level, liver and kidney function indicators, and electrolyte levels were all within the normal range. A laboratory examination indicated that the |
This patient was a 57-year-old right-handed traffic policeman with a history of diabetes mellitus and hypertension. He was first admitted to a local hospital due to urgent hematochezia and upper abdominal discomfort. Emergency gastroscopy proved duodenal ulcer bleeding. After blood transfusion, he presented paraphasia, walking instability, and loss of bimanual coordination, characterized by the left hand performing opposite movements to the right hand. He also complained that his left hand did not belong to him but to someone else. More importantly, the patient showed loss of coordination as well as mirror movements between the lower limbs. For instance, he had difficulty going to the toilet by himself, because when his right leg moved forward, his left leg moved back at the same time. This phenomenon disappeared gradually and then mirror movements appeared. Sometime when his right leg moved forward, his left leg did the same action. Two weeks later, he was admitted to our hospital because he suffered from anesthesia of the left side of his body.\nOn examination, the patient was alert and oriented. There was only mildly dysarthric speech. Hypesthesia of his left side of body and face was observed. His motor strength and tone were both normal. But deep-tendon reflexes in all extremities reduced symmetrically and frontal lobe releasing signs such as reflexive grasping were absent. Other general physical examinations were all within normal limits. The brain magnetic resonance imaging (MRI) showed a few regions with low signal intensity in the genu, body, and splenium of the right corpus callosum on the T1-weighted image and high signal intensity in the same area on diffusion-weighted images (DWI), T2-weighted, and fluid attenuated inversion recovery (FLAIR) images (Fig. ). Computed tomographic angiography of the cerebral vessels did not show any obvious stenosis in cerebral arteries. Our patient got 23 and 12 scores in the Mini-mental State Examination and Montreal Cognitive Assessment, respectively. No positive results were found in Line bisection tasks, Letter cancellation, Star test, Line cancellation, and Copy a daisy. During neuropsychological test, when he used his right hand to move toy bricks, the left hand would interfere by taking them back (the intermanual conflict). Our patient exhibited difficulties in naming the left fingers as well as pointing to named parts of the body with his left hand, especially in the nonvisual condition, on the test of body identification by the method of Nagumo and Yamadori.[ The accuracy of naming the fingers with eyes closed and with hand behind body was 40%/30% (left fingers) as well as 80%/90% (right fingers), respectively. Correct rate of pointing to the named body parts with the left hand and the right hand was 86% and 100%, respectively. In addition, he performed the test of praxia, including verbal commands, imitation, pantomime to visual presentation of objects, and object use by the method of Lausberg et al.[ Our patient showed prominently left hand ideomotor apraxia, such as brushing teeth and combing his hair, which was worse on verbal commands and pantomime to object use to visual object presentation, while improved on imitation and actual object use. Types of errors included spatial errors, hand position errors, and hand shape errors. He received symptomatic and supportive treatment in our hospital. After 6 months of hospital discharge, our patient improved apparently and could drive a car by himself. These abnormalities on brain MRI completely disappeared. |
A 64-year-old woman with a history of hypertension and hypercholesterolemia under treatment, presented to our emergency department with acute onset of substernal chest pain radiating to the neck and jaw. The pain had emerged 2 hours earlier when she had experienced near-drowning and fear of imminent death.\nPhysical examination on admission revealed a heart rate of 100 bpm, her blood pressure was 150-90 mmHg and her oxygen saturation was 97%. A grade 1–2/6 systolic murmur and a fourth heart sound were heard.\nECG demonstrated ST-segment elevation in leads V2–V6. Echocardiographic evaluation depicted decreased LV ejection fraction (40%), with new regional wall motion abnormalities, i.e. hypokinesis of the basal and mid segments of the LV (fig. , , ). It was noted that these changes had not been noted on a previous routine echocardiogram. However, subsequent coronary angiography (CAA) revealed neither major atherosclerotic lesions nor coronary spasm, while throughout her hospitalization there was only a slight increase of troponin I (cTnI) levels. Magnetic resonance imaging (MRI) was performed and showed no evidence of myocarditis. The patient had no increased inflammatory markers. No endocrine diseases or other serious concomitant disorders were present.\nDuring her hospitalization the patient remained asymptomatic. Ventricular systolic function recovered completely and the wall motion abnormalities resolved by the 3rd day following admission. Her ECG evolution showed T-wave inversion. The patient was discharged after six days, under medical treatment with a b-blocker and an angiotensin converting enzyme inhibitor. The prior occurrence of a stressful event, the normal CAA findings accompanied by cTnI levels that were disproportionate to the extent of hypokinesia and, finally, the fast LV recovery, lead us to believe that our patient had experienced a variant form of acute reversible stress cardiomyopathy.\nDuring the next two months the patient experienced two more episodes of prolonged chest pain, both after emotional stress, bearing the same characteristics as before. However, the echocardiogram on both these occasions depicted no akinetic segments. There were neither abnormal ECG findings nor cTnI elevation.\nTo-date, she remains asymptomatic, with normal LV systolic function and VO2max. |
A 52 year old man with a history of T3N1M0 (Dukes C) rectal adenocarcinoma treated with neo-adjuvant chemo-radiotherapy (5FU + radiotherapy) followed by anterior resection and adjuvant chemotherapy (5FU) presented 18 months post completion of therapy with rising serum CEA level (2.3 μg/l post resection of primary rectal malignancy to 9.7 μg/l) and a 15 mm left lower lobe pulmonary nodule on computerized tomography (CT), suggestive of a metastatic deposit (figure ). He had no other significant past medical history and no family history of malignancy. Physical examination was unremarkable, with no obvious mass lesions in the abdomen or palpable local recurrence in the rectal stump.\nAn 18F-FDG whole body Positron Emission Tomography (PET) scan was performed to further evaluate the nature of the left lower lobe pulmonary nodule and to evaluate any other possible sites of metastatic disease. The whole body PET scan demonstrated significant 18F-FDG uptake in the left lower lobe pulmonary nodule compatible with a metastatic deposit (figure ). An intensely FDG-avid right lower lobe thyroid nodule was also noted, which corresponded with a large partially calcified well circumscribed nodule on CT.\nAn ultrasound guided fine needle aspirate of the right lower pole thyroid nodule was performed which revealed malignant cells with features suggestive of a primary papillary thyroid cancer. A right hemi-thyroidectomy was subsequently performed. Macroscopically, the resected thyroid specimen demonstrated a well-circumscribed dominant thyroid nodule measuring 32 mm in diameter with a pale tan capsule less than 1 mm in thickness. The cut surface had a variegated appearance with pale tan friable tissue intermixed with foci of yellow tissue and dark brown foci. Histological examination revealed cells typical of metastatic adenocarcinoma of the colon intermixed in a background of a poorly differentiated papillary thyroid carcinoma (figure ).\nA wedge resection of the left lower lobe pulmonary nodule was subsequently performed which confirmed metastatic colorectal adenocarcinoma (figure ). Serum CEA level normalized post operatively (1.9 μg/l) and no further surgery was contemplated. Despite no further overt metastatic disease on CT, the patient was commenced on a course of chemotherapy (5 FU/Oxaliplatin) to treat presumed low volume metastatic colorectal disease and thus decrease the risk of developing overt recurrence. No further thyroid cancer specific treatment has been initiated. |
An active 48-year-old male patient with a body mass index of 21 presented with anterior knee pain. The patient suffered from Charcot Marie Tooth Type 1b and bilateral hip dysplasia, which had been treated with multiple surgeries. On first presentation, the patient was found to have patellar dysplasia and patella alta, but with normal patellar tracking, and there was a full-thickness chondral defect on the lateral facet of the patella. In addition, there was also an old complete tear of the anterior cruciate ligament (ACL), and the patient was complaining of intermittent instability.\nThe patient underwent an ACL reconstruction combined with a tibial tuberosity advancement osteotomy, to correct the patella alta. The chondral defect on the lateral facet of the patella was found to measure approximately 2 cm × 1 cm. The edges of this defect were stabilized by arthroscopic radiofrequency chondroplasty and the base was treated with microfracture.\nThe patient underwent slow and cautious rehabilitation, remaining non-weight-bearing for the first 6-week period followed by a gradual return to full-weight-bearing as comfort allowed, but avoiding any heavy patellofemoral loading until 6 months post-operatively.\nThe patient then reported that the knee felt stable and initially there was reduced anterior knee pain. However, over the course of the following 3–4 years, the patient once more began to complain of increasing anterior knee pain again, particularly with any activity that involved patellofemoral loading. A magnetic resonance imaging (MRI) scan at 4 years post-microfracture showed poor filling of the patellar lesion with minimal fibrocartilage ().\nAfter a detailed discussion of the various potential options, the patient consented to further surgery. The knee was opened with a mini-medial parapatellar arthrotomy and the patella was everted. The base of the chondral defect on the lateral facet of the patella was curetted down to the subchondral bone plate and microfracture was performed according to the protocol of Steadman et al. A resorbable PGA–hyaluronan-based implant (chondrotissue®—BioTissue AG, Zurich, Switzerland) was cut to size, placed into the defect, and fixed in place with two bioabsorbable ActivaNail™ pins (Bioretec Ltd, Tampere, Finland) (). The implant was seen to immediately soak up the blood from the microfracture. Furthermore, platelet-rich autologous fibrin glue (Vivostat PRF®—Vivostat A/S, Alleroed, Denmark) was prepared from 120 mL of the patient’s blood used to cover the implant as additional sealing ().\nPost-operatively, the patient was initially kept non-weight-bearing with his knee in a hinged brace locked in full extension for the first 2-week period followed by commencement of regular unloaded range of motion exercises. Weight-bearing was commenced at 6 weeks post-operatively, followed by a gradual return to light cardio fitness exercises, but excluding any heavy patellofemoral loading, such as squats, lunges, the leg press or loaded leg extensions.\nThe treatment proceeded without any intra- or post-operative complications such as inflammatory reaction, swelling, infection, arthrofibrosis, or undue pain. To evaluate the cartilage repair, MRI was performed on a 3T scanner 6 months post-operatively. The MRI () demonstrated good formation of new tissue on the lateral facet of the patella, with evidence of the biodegradable pins having partially dissolved and with what appeared to be growth of new articular cartilage. By this stage, the patient had reported a significant decrease in his anterior knee pain but he was complaining of a clunking sensation anteriorly with knee flexion. Second-look arthroscopy ( and () performed 9 months after the patellar chondrotissue implantation, revealed the formation of new cartilage repair tissue filling the whole defect, which was stable and which exhibited normal cartilage-like texture on probing. Some scarring of the anterolateral aspect of the lining of the joint was indentified, and this required just a minor debridement. Six weeks after the second-look arthroscopy, the patient reported a reduction of the previous catching sensations that had been present beforehand and an overall significant improvement in his anterior knee symptoms that had been present prior to the PGA–hyaluronan scaffold implantation procedure. The patient reported satisfaction, with improved function and a return to regular non-impact exercise. |
25 year old lady presented with swelling and pain of left thigh of seven years duration. This gradually increased in size without weight loss or proximal muscle weakness. She did not have difficulty in walking and her activities of daily living were not affected by the pain. Examination of the left thigh revealed bowing of the left femur and a soft tissue swelling not attached to the bone. The size of the swelling was 2 × 3 cm, mobile and mildly tender to palpation.\nShe had attained menarche at the age of 13 years and had normal menstrual cycles. She was married for the past one and a half years and had not conceived yet. Her body mass index was 25.1 kg/m2 with height of 150 cm. She did not have any bony deformity or café au lait spots. On biochemical evaluation she had normal alkaline phosphatase, normal serum calcium, phosphorous and 24 hrs urine calcium, phosphorous and creatinine. She had a low level of 25 hydroxy vitamin D of 4.85 ng/ml (20.0-32.0 ng/ml) X-rays of pelvis and hip showed expansile bony swelling with cortical thinning of both tibia []. She underwent a bone scan which showed abnormal, irregularly increased tracer activity in left scapula, left tibia, left femur, pubis and left sacroiliac joints, L5 vertebra, left distal humerus and multiple ribs [], suggestive of polyostotic fibrous dysplasia. MRI of the left thigh showed well defined intramuscular mass lesions []. These lesions were hypointense on T1 weighted image and hyperintense on T2W. They were partly exophytic and measured: 28 × 41 mm in the anteromedial aspect of right adductor longus, 16 × 23 mm in the right adductor magnus at the level of mid-thigh and 9 × 10 mm in the gluteus maximus A bone biopsy [] from the left femur revealed fibrous dysplasia and a biopsy of the soft tissue mass from left thigh was confirmed as intramuscular myxoma []. |
A 38-year-old male with a three-year history of slow, progressive right-sided nasal obstruction came to the ear nose and throat (ENT) clinic. He had a history of dental filling procedures for one right upper tooth cavity a year and a half back. It was associated with right-sided facial fullness and pain as well as occasional purulent nasal discharge mainly from the right side with post-nasal secretions. He had no change in smell. His past medical history was free of any co-morbidities, his past surgical history was negative for surgeries or traumas. Before his complaint that started three years ago, he had only complained of minimal intermittent alternative nasal obstruction which did not affect his daily life or sleep.\nOn physical examination, he had a nasal polyp descending lateral to the middle turbinate and running posteriorly. In addition, he had nasal septal deviation to the right side. On the left side, no polyps or discharge were seen. Throat examination showed clear post nasal drip and no polyps could be seen. His head and neck exams were unremarkable. Endoscopic nasal examination showed a protruding soft glistening polyp with no obvious dilated vessels in the right middle meatus extending toward the posterior choana with severe septal deviation to the right side, endoscopy on the left side showed the choanal part of the right antrochoanal polyp (ACP) and absence of any polyps in the left nasal cavity.\nComputed tomography (CT) of the nose and paranasal sinuses revealed a large polypoidal soft tissue mass obliterating the right nasal cavity and extending posteriorly to the right choana. Besides, the right maxillary sinus showed complete soft tissue obliteration with a central area of calcific density seen within the right maxillary sinus (Figure ).\nThe patient was sent for surgery under general anesthesia. At first, septoplasty was done, followed by right-sided Functional Endoscopic Sinus Surgery (FESS) (Figure ). An uncinectomy with a right wide middle meatal antrostomy was then performed, then polypectomy was done. After that, the maxillary sinus was noticed to be full of cheesy and clay-like debris and a fungal ball was noted in the anteroinferior wall of the maxillary sinus. The pedicle of the polyp was attached to the posterolateral wall of the right maxillary sinus. The origin site of the polyp was removed using giraffe forceps and curved debriders. Irrigation and wash of the maxillary sinus was done to clear the debris. Opening of bulla ethmoidalis on the right side was also done but did not show any disease within it.\nThe excised samples (Figure ) were sent in two different specimens for histopathological examination. The first of them, the right nasal polyp, was consistent with histology of antrachoanal polyp (Figure ) a benign ciliated pseudo-stratified columnar epithelium and composed of proliferated mucus salivary glands in markedly oedematous vascular chronically inflamed stroma and rich in eosinophils with no fungi or granuloma or malignancy. The second specimen, the right maxillary sinus content, was consistent with aspergillus (Figure ); there were many colonies and masses of numerous closely packed fungal septate hyphae with positive periodic acid shift (PAS) test and negative vascular invasion.\nThe postoperative course was uneventful. Complete resolution of the main patient’s symptoms was achieved after surgery. No recurrence was noted in eight weeks follow-up visits post-operatively.\nTissue culture was not sent because it was not expected to find fungal elements during surgery. However, it should be stressed that even if most fungal balls are made of Aspergillus, cultures are often negative []. |
A healthy 24 year-old male presented to the Oral and Maxillofacial Surgery Department at Tufts Medical Center for evaluation and treatment of dental malocclusion with skeletal facial discrepancy by his orthodontist. The patient was diagnosed with masticatory dysfunction secondary to maxillary hypoplasia and transverse discrepancy as well as mandibular prognathism resulting in a severe class III malocclusion and elected to undergo orthognathic surgery for its correction. Before the surgery, he underwent leveling and alignment of both maxillary and mandibular arches and removal of all four third molars. The preoperative evaluation and work-up were performed by the surgical team and planned for a 2-piece Lefort I osteotomy for maxillary advancement of 3mm, a bilateral sagittal split osteotomy for mandibular setback of 6mm, an advancement genioplasty of 8mm and maxillo-mandibular fixation (MMF).\nTwo weeks prior to the procedure, he underwent a preoperative evaluation by the consultant physician and the concerned anaesthetist and was medically cleared by his primary care physician. On our preoperative assessment, the patient weighed 89 kg, was alert, awake, and oriented to person, place, and time. He denied any cardiovascular, pulmonary, renal, hepatic, gastrointestinal, genitourinary, hematologic, endocrine, or psychiatric disorders. He denied taking any medications and had no known reported drug allergies. Pertinent social history included being a fourth-year medical student with a supportive family structure, social alcohol use, and no tobacco or other illicit substances.\nIntravenous access was obtained in the preoperative holding area and the patient was transferred to the operating room. Intravenous induction was performed by the anesthesia team and the patient was easily intubated through the right nares with a number 8 size nasoendotracheal tube which was taped at the 29 cm mark by the oral and maxillofacial surgery team. Bilateral breath sounds and capnography confirmed tube placement. The patient underwent a 2-piece Lefort I osteotomy for advancement of 3 mm, a Bilateral Sagittal Split osteotomy for setback of 6 mm, an advancement genioplasty of 8mm and fixation of the osteotomized segments with 2.0 mm Titanium miniplates and 6.0 mm long titanium screws, followed by maxillo-mandibular fixation with elastics without complication. The surgical procedure lasted 3 hours 56 minutes. Based on the clinical evaluation of the patient at the conclusion of the procedure, we noted positive hemostasis at all incision sites and significant postoperative edema of the face and oral cavity and made the decision to keep the patient intubated and sedated overnight in the post-anaesthesia care unit (PACU). The patient was extubated the following morning at 7 am without complications. The remaining postoperative course in the hospital, a 2-day stay, was uneventful and the patient was discharged home and followed up in the oral and maxillofacial surgery clinic 1 week postoperatively. At the 1-week postoperative check, the patient noted features suggestive of mild bilateral paresthesia of cranial nerve V3 distribution and was optimizing his nutritional intake. Neither the patient nor his family observed any change in voice during the period that the patient was on MMF. At the 2 week postoperative evaluation, maxillo-mandibular fixation was removed. Two days after MMF removal we received a call from the patient's father with questions about changes in the quality of his son's voice, as the patient and his family noticed a change in his voice soon after the MMF was removed and the patient started speaking. We evaluated the patient and confirmed hoarseness of his vocal quality and noted mild coughing when taking thin liquids. He had no difficulty with swallowing thicker consistency preparations or any dyspnea. The otolaryngology voice department at Tufts Medical Center was consulted for evaluation of vocal cord function. The otolaryngology service performed a flexible fiberoptic laryngoscopy that showed normal abduction and adduction of the right true vocal cord, normal abduction of the left true vocal cord but very limited adduction. This left the patient with significant glottal incompetence and secondary muscle tension dysphonia. At that time the patient was offered a temporary vocal fold injection to help with his hoarseness but denied having the procedure at that time. We continued to follow the patient on a weekly basis, and 6 weeks postoperative the hoarseness completely resolved without medical or surgical intervention. |
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