Duodenal duplication is a rare congenital malformation and has been reported as a rare cause of recurrent acute pancreatitis. Hemorrhagic ascites has been reported in only one case of duodenal duplication.
An 11-year-old Chinese girl presented with abdominal pain, hematemesis and dark stools. On admission, an abdominal examination revealed a moderately distended abdomen with diffuse tenderness. Biochemical investigations showed increased serum levels of amylase, lipase, and urine amylase. An abdominal computed tomography scan and magnetic resonance imaging scan revealed an enlarged and heterogeneous pancreas with poorly delineated borders. There was a cystic lesion measuring 25mm × 48mm × 28mm, located between the descending portion of her duodenum and the head of her pancreas. There were massive effusion signals in her abdominal cavity. An exploratory laparotomy was performed. A tubular cyst measuring 32mm × 52mm × 30mm was found in the second part of the duodenum, next to the head of her pancreas. The anterior wall of the duplication cyst was resected and anastomosis of the remaining cyst to the duodenum was performed for drainage. Histopathological examination of the excised cyst wall showed duodenal mucosa, submucosa and muscle coats, indicative of a duodenal duplication.
It is important to be aware of duodenal duplication when evaluating a patient with recurrent acute pancreatitis accompanied by massive hemorrhagic ascites.
Acute pancreatitis; Child; Duodenal duplication; Hemorrhagic ascites
Current treatment options for visceral leishmaniasis (pentavalent antimony, amphotericin B, liposomal amphotericin B and mitelfosine) achieve long-term clinical cure in the majority of immunocompetent patients. Disease relapse is usually provoked by T-cell number or function impairment (corticosteroid or cytotoxic therapy, transplant recipients, advanced human immunodeficiency virus disease).
We report a case of visceral leishmaniasis with multiple relapses in a 75-year-old Greek immunocompetent man. Visceral leishmaniasis relapses occurred despite appropriate treatment with liposomal amphotericin B 3mg/kg/day on days one to five, 14 and 21 (for the first episode and the first relapse) and mitelfosine 150mg/day for 28 days (for the second relapse). The third relapse was treated with high-dose liposomal amphotericin B (10mg/kg for two consecutive days), followed by a secondary prophylaxis of 3mg/kg once per month, which prevented disease reappearance during one year of follow-up.
An unusual case of recurrent visceral leishmaniasis in an older immunocompetent patient was treated with high-dose liposomal amphotericin B and a monthly prophylaxis with no evidence of a relapse after one year of follow-up.
Leishmaniasis; Recurrent; Immunocompetent; Liposomal amphotericin B
Several macular complications related to abnormalities of the vitreoretinal interface have been classically attributed to retinitis pigmentosa of which cystoid macular edema is the most common. Other less frequent complications are as follows: epiretinal membranes, vitreomacular traction syndrome and macular holes.
A 64-year-old woman, with the previous diagnosis of retinitis pigmentosa, was referred to our department with a complaint of central visual loss in her left eye for 12 months. A fundoscopy and optical coherence tomography examination revealed the presence of a macular hole more than 500 microns in diameter. The patient underwent 20-gauge pars plana vitrectomy. Closure of the hole was observed after surgery, but reopening occurred at 2 years postoperatively.
The pathogenesis of macular hole formation in patients with retinitis pigmentosa is unclear. Surgical outcomes may not always be favorable, and the possibility of reopening must be taken into account, even after a long time.
A paradoxical reaction during antituberculosis treatment is defined as the worsening of pre-existing tuberculosis lesions or the appearance of a new tuberculosis lesion in patients whose clinical symptoms improved with antituberculosis treatment. The median onset time to the development of a paradoxical response has been reported to be about 60 days after the start of treatment. We report the case of a patient with a paradoxical reaction presenting as a psoas abscess after nine months of antituberculosis treatment. To the best of our knowledge, this manifestation has not previously been reported.
A 23-year-old Japanese man presented to our hospital with lower abdominal pain. Computed tomography showed that he had mediastinal and abdominal para-aortic lymph node swellings. Fluorine-18 fluorodeoxyglucose positron emission tomography showed hot spots in these lymph nodes and in his right cervical lymph node, suggesting a lymphoma. The examination of an abdominal lymph node biopsy specimen showed lymph node tuberculosis, so antituberculosis treatment was started. However, after nine months of treatment, he experienced right flank pain. Abdominal computed tomography showed a right psoas abscess and abdominal para-aortic lymph node swelling. The abscess was treated by percutaneous drainage. After repeated drainage, the psoas abscess subsided and disappeared. The purulent fluid yielded no microorganisms, suggesting a paradoxical reaction.
Attention should be paid to paradoxical reactions occurring during antituberculosis treatment for systemic lymph node tuberculosis.
This report describes the apparent ‘resurrection’ of a patient in an emergency department setting. Befittingly named the ‘Lazarus phenomenon’, the recovery of spontaneous circulation after cessation of cardiopulmonary resuscitation is an extremely rare occurrence that was first described in 1982 and has been mentioned only 38 times in the medical literature. Our patient’s case is remarkable in that it helps illustrate many of the mechanisms of this rare phenomenon. It also serves as a reminder of our limitations in determining when to terminate cardiopulmonary resuscitation and suggests that cessation of cardiopulmonary resuscitation should be approached with more care.
An 89-year-old Caucasian woman with a medical history of hypertension, atrial fibrillation, hypothyroidism, aortic insufficiency, lymphedema and hypoxia secondary to partial lung resection presented to our hospital after a witnessed fall unassociated with head trauma or loss of consciousness. On examination, our patient was saturating at 85 percent and exhibited a decreased range of motion of the upper extremities and left hip. Radiographic images revealed a left femoral neck and left distal radius fracture. Our patient was stabilized on 100 percent fraction of inspired oxygen and was awaiting transfer to an in-patient unit when, at 3:30 a.m., she went into cardiac arrest. An advanced cardiac life support protocol was initiated, at which time our patient was intubated and administered epinephrine, vasopressin and sodium bicarbonate. Our patient remained unresponsive and asystolic so cardiopulmonary resuscitation was abandoned at 3:48 a.m. After five minutes a ventricular contraction was noted at 3:51 a.m. This progressed to sinus rhythm with a pulse at 3:53 a.m. Our patient was stabilized on norepinephrine and moved to our Intensive Care Unit. At 10:55 a.m., however, our patient again arrested and, despite resuscitative efforts, was pronounced dead at 11:03 a.m.
Our patient’s case clearly illustrates many of the proposed mechanisms for delayed return of spontaneous circulation including pulmonary hyper-inflation, hyperkalemia, delayed drug onset, and embolism dislodgement. Our patient represents a humbling and disturbing reminder that our medical acumen does not necessarily dictate the fate of our patients and that the decision to discontinue cardiopulmonary resuscitation should be approached with care by incorporating techniques such as end-tidal carbon dioxide, ventilator disconnect and passive monitoring.
Seizure disorders can have a wide variety of causes. In many cases, however, the underlying cause remains unknown. Vaccinations, for example, can trigger seizures, especially during childhood. In the literature, many cases have been reported in which febrile convulsions occurred after the administration of different types of vaccines, such as the measles, mumps and rubella vaccine or the tetanus and diphtheria vaccine. Only a few cases of epilepsy after vaccination have thus far been described in adults.
In the case reported here, a 44-year-old German Caucasian man working as a soldier had a seizure the day after he received a third dose of Japanese encephalitis vaccine. Before this vaccination, he had received multiple vaccines that he had tolerated well. He underwent several drug therapies at various institutions but has continued to experience different forms of seizures for more than 18 months. The intervals between seizures were approximately six weeks in length. The present work discusses our patient’s history, including all diagnostic procedures and results, as well as treatment approaches. None of the examinations revealed a possible cause for the seizures. Since no structural or genetic causes were detected, the seizures were deemed most likely to have been caused by the vaccinations, especially vaccination against Japanese encephalitis. To date, no medication has prevented our patient from having repeated attacks.
To the best of our knowledge there have been no previous cases reported in the literature where seizures occurred after multiple vaccinations in general or after vaccinations against Japanese encephalitis in particular. Although vaccines are tested before release, the appearance of new adverse reactions cannot be prevented in all cases. Seizure after vaccination is difficult to treat. In our patient’s case, different approaches have not led to a satisfying result to date.
Adverse reaction; Japanese encephalitis; Seizure; Vaccination
Burkitt’s lymphoma is a highly aggressive, small, non-cleaved B-cell non-Hodgkin's lymphoma. In the sporadic form of the disease that occurs in non-endemic areas around the world, most commonly in developed countries, patients usually present with an abdominal mass that frequently involves the ileocecal region of the bowel; ocular or orbital involvement is rare. Primary disease of the sinuses is uncommon and, to the best of our knowledge, that of the anterior septum has never been described. We report the diagnosis and successful management of Burkitt’s lymphoma originating from the nasal septum in a male patient.
An otherwise healthy 78-year-old Caucasian man who did not smoke cigarettes was admitted to our Ear, Nose and Throat outpatient clinic with the complaint of nasal obstruction due to left-sided nasal septal thickening. Paranasal computerized tomography revealed a well-circumscribed solid mass originating from his anterior nasal septum and obstructing his airway. The final diagnosis of Burkitt’s lymphoma was verified by immunohistochemical studies. Our patient had a good clinical outcome after chemoradiotherapy, with no problems reported to date in the second year of follow-up.
We provide what we believe to be the first report of a case of sporadic Burkitt’s lymphoma involving the nasal septum, and describe the efficacy of first-line chemotherapy. Being an original case report with broader clinical impact across more than one area of medicine, this case presentation has the potential to significantly advance our understanding of Burkitt’s lymphoma and we emphasize the need to include this disease in the differential diagnosis of patients presenting with a nasal septal mass.
Medulloblastoma, the most frequent brain tumor in childhood, also occurs with a wide range of characteristics in adult patients. Late relapse is common in adult medulloblastoma, and the overall survival of relapsed patients usually ranges from 12 to 15 months. Treatment at recurrence is still debated and after reoperation includes stereotactic or normofractionated radiotherapy, and high-dose chemotherapy with autologous bone marrow transplantation.
We report on the case of a 31-year-old Caucasian woman who underwent re-irradiation for a recurrence of medulloblastoma at nine years after first irradiation (56Gy), focusing on the radiobiological background and a review of previous studies involving re-irradiation of recurrent medulloblastoma. After surgical excision of the relapsed tumor and medical multi-agent treatment, the site of recurrence was treated using three-dimensional conformal radiotherapy to a total dose of 52.8Gy (1.2Gy/fraction/twice daily). A total biological equivalent dose of 224.6Gy (α:β = 2 Gy) was delivered to the posterior fossa (first and second treatments). No radionecrosis or local recurrence was evident at 18 months after re-irradiation.
Re-irradiation can be considered a possible and safe treatment in selected cases of recurrent medulloblastoma in adults. The reported radiobiological considerations could be useful in other cases involving re-irradiation of brain tumors.
Adult medulloblastoma; Biological equivalent dose; Re-irradiation
Sudden tetraparesis represents a neurological emergency and is most often caused by traumatic spinal cord injury, spinal epidural bleeding or brainstem ischemia and less frequently by medial disc herniation or spinal ischemia.
Here we report the rare case of an 82-year-old Caucasian man who developed severe tetraparesis four days after radical cystoprostatectomy. An emergency diagnostic study for spinal cord affection was normal. Brain magnetic resonance imaging revealed acute bilateral ischemic strokes in the precentral gyri as the underlying cause.
This case report underlines the need to also consider unusual causes of tetraparesis in an emergency situation apart from spinal cord or brain stem injury in order not to leave severe symptomatology unclear and possibly miss therapeutic options.
Little information is available on the pathogenesis of heatstroke without strenuous exercise in younger patients. Here, we report the case of a 31-year-old man who developed heatstroke secondary to hyponatremia. His condition was initially misdiagnosed as classic heatstroke. We hope the detailed description of our patient’s case will provide valuable information for medical professionals faced with similar cases in the future.
A 31-year-old Japanese man who was regularly taking anti-hypertensive agents, including a thiazide, was admitted to our hospital owing to consciousness disturbance and a high-grade fever. A thorough examination revealed mild renal dysfunction, rhabdomyolysis, a hyper-coagulable state, and severe hyponatremia (114mEq/L). Because he worked in a hot environment, an initial diagnosis of heatstroke was established. His general condition improved rapidly with supportive measures, and he was discharged on the fourth day after admission. Interestingly, he did not recognize feeling hot while working in a hot environment. These findings suggest that the consciousness disturbance, which was probably attributable to acute hyponatremia caused by the thiazide, preceded the onset of the heatstroke.
Clinicians should consider the presence of underlying diseases, especially consciousness disturbance, when younger individuals develop heatstroke in the absence of strenuous exercise.
Hutchinson-Gilford progeria syndrome is a rare pediatric genetic syndrome with an incidence of one per eight million live births. The disorder is characterized by premature aging, generally leading to death due to myocardial infarction or stroke at approximately 13.4 years of age. The genetic diagnosis and special clinical manifestation in two Han Chinese siblings observed at our clinic for genetic counseling are described in this report. We screened the LMNA gene in these two siblings as well as in their unaffected parents. A homozygous mutation R527C was identified in the affected siblings, and both parents were heterozygous for this variant.
In case 1, the elder 10-year-old female sibling showed the classic physical and radiological changes of Hutchinson-Gilford progeria syndrome in addition to a considerable overlap with the phenotype of mandibuloacral dysplasia.
In case 2, the younger male sibling had begun to show some early physical changes at age six months.
The phenotypic findings in the patients we describe here widen the clinical spectrum of Hutchinson-Gilford progeria syndrome symptoms, providing further recognition of the phenotypic range of LMNA-associated diseases.
Adenocarcinoma of the seminal vesicles is a very rare malignancy, with less than 100 cases reported worldwide. It is documented to have a poor prognosis, with the majority of patients developing metastatic disease, most commonly in the prostate, bladder and rectum. Currently there is no standard treatment for metastatic disease and the limited reports of treatment with radiotherapy, chemotherapy and hormonal (anti-androgenic) therapy show that they are generally of modest benefit. The association between malignancy and an increased risk of autoimmune vasculitis has been demonstrated in a number of malignancies, but to date there have been no documented cases of adenocarcinoma of the seminal vesicles associated with anti-neutrophil cytoplasmic antibody vasculitis.
In this report we describe the case of a 55-year-old Caucasian man with metastatic adenocarcinoma of the seminal vesicles. He previously had received chemotherapy treatment for advanced testicular cancer and later presented with hemospermia. He subsequently developed c-antineutrophil cytoplasmic antibody vasculitis requiring intensive immunosuppression and renal dialysis.
Adenocarcinoma of the seminal vesicles is a rare diagnosis and our case is more unusual in that our patient previously had chemotherapy treatment for advanced testicular cancer and went on to develop severe antineutrophil cytoplasmic antibody vasculitis when diagnosed with metastatic seminal vesicle cancer. This case illustrates that autoimmune vasculitis can occur in any patient with malignancy and an early referral to the renal team combined with renal biopsy can assist in the earlier diagnosis and more successful management of these rare events. This case should be of interest to oncologists, renal physicians, urologists and general physicians who encounter patients presenting with hemospermia or vasculitis.
Similar to pheochromocytomas, paragangliomas can secrete catecholamines, although they are usually non-functional and clinical presentation is non-specific. We present a case of accidental, intra-operatively diagnosed neuroendocrine-active sympathetic paraganglioma, which was suspected and confirmed during elective retroperitoneal tumor removal.
A 25-year-old Caucasian Croatian man, American Society of Anesthesiologists status 1, underwent elective surgery for retroperitoneal tumor removal. The tumor had been discovered by chance during a routine examination and was suspected to be a sarcoma. Our patient had no history of previous medical conditions nor did he have symptoms characteristic of a neuroendocrine secreting tumor. The results of ultrasound and magnetic resonance imaging studies showed a large, well demarcated retroperitoneal tumor mass in his upper abdomen localized between the aorta and vena cava, measuring approximately 9×6×4.5cm. In the operating room an epidural catheter was inserted at the T7 to T8 level prior to induction of general anesthesia. Epidural analgesia was maintained by an infusion pump with local anesthetic and opiate mixture. During the surgical excision of the tumor, hemodynamic changes occurred, with hypertension (205/110mmHg) and tachycardia (up to 120 beats/minute). In spite of the fact that the surgical field of work did not include adrenal glands whose direct manipulation could explain this occurrence, there was a high degree of suspicion for the presence of a neurosecreting tumor. His clinical symptoms were relieved after administration of urapidil, esmolol and magnesium sulfate. After tumor excision, our patient developed severe hypotension. Hemodynamic stability was reinstated with aggressive volume replacement, with crystalloids and colloids, vasopressors and hydrocortisone. His post-operative course was unremarkable and on the eighth post-operative day our patient was discharged from hospital, with no consequences or symptoms on follow-up two years after surgery.
Our patient’s case emphasizes the need to consider the presence of extra-adrenal paragangliomas in the differential diagnosis of retroperitoneal tumors, despite their rare occurrence. In our patient’s case, invasive hemodynamic monitoring during combined general anesthesia and epidural analgesia and early recognition of catechol-induced symptoms raised suspicion of the existence of a paraganglioma, and this led to an adequate therapeutic approach and favorable outcome of the surgery. Pre-operative recognition of paragangliomas could lead to better pre-operative preparation, but even high clinical suspicion in undiagnosed forms during surgery and the availability of rapid and short-acting vasodilatators, α-blockers and β-blockers might favor good outcome.
Epidural analgesia; Invasive hemodynamic monitoring; Paraganglioma/pheochromocytoma; Therapeutic protocols
Health promotion and disease prevention are important aspects of primary health care. However, limited data are available concerning the opinions of older patients towards the respective services offered by family doctors. The aim of the present study was to evaluate an older patient's perception of the role of the family doctor in promoting his health, and identify those components that are difficult to examine in quantitative research.
A qualitative case study of an 80-year-old man using an in-depth interview was carried out. The interview transcript was analyzed thematically. Our patient was an 80-year-old university-educated man, with stable social and financial circumstances, living with his wife. He had retired early on grounds of ill health (tuberculosis) and had received a disability pension prior to formal retirement. At the time of the interview, his medical problems included mild prostatic hypertrophy, scoliosis and hypertension. He considered his health status to be satisfactory. He had changed family doctor five years prior to the interview, as he had been dissatisfied with the care provided.
We found that our patient expected the family doctor to be aware of, and to discuss, the following issues: physical activity, diet, management of stress and mental health, use of alcohol and tobacco, personal hygiene, health screening, use of medication, and social activity. At the same time, our patient perceived the doctor's role as supplementary to his own in terms of the appraisal and maintenance of his health.
Our findings provide evidence of what is important in the promotion of health among older people.
Case studies; Health promotion; Older people; Primary health care
Sunitinib is an oral multi-targeted tyrosine kinase inhibitor approved for first line treatment for metastatic renal cell carcinoma and imatinib-resistant metastatic gastrointestinal stromal tumors. Sunitinib administration can cause myelosuppression resulting in neutropenia and thrombocytopenia. Here we present the case of a patient with metastatic renal cell carcinoma who developed sunitinib-induced immune-mediated thrombocytopenia and who was treated with withdrawal of sunitinib and administration of intravenous immunoglobulin and steroids.
This case report describes a 70-year-old Aboriginal Australian with a diagnosis of metastatic renal cell carcinoma. Three weeks after the initiation of sunitinib he developed epistaxis and was admitted with thrombocytopenia (platelets 7 × 109/L) which was found to be refractory to platelet transfusion. Sunitinib was stopped and he was treated with intravenous immunoglobulin and steroids. His platelet count rapidly improved and returned to baseline in three weeks. Only two cases of sunitinib-induced immune-mediated thrombocytopenia have been described in the literature.
Clinicians should have a high index of suspicion for the potential of immune-mediated thrombocytopenia after the initiation of multi-targeted tyrosine kinase inhibitors such as sunitinib. This is a diagnosis of exclusion and can be safely treated by drug withdrawal.
Metastatic renal cell carcinoma; Sunitinib; Thrombocytopenia
Zollinger–Ellison syndrome is characterized by recurrent peptic ulcers and diarrhea that result from gastrin-secreting neuroendocrine tumors of the gastrointestinal tract; nevertheless, severe hypergastrinemia may also have alternative pathogenetic explanations.
A 61-year-old woman of Caucasian origin presented with a history of epigastric pain and early satiety, severe hypergastrinemia (approximately 2000 pg/mL) and a neuroendocrine polyp in the corpus of her stomach. Chronic atrophic gastritis and intestinal metaplasia was present, but she denied use of acid suppressant drugs and the results of tests for Helicobacter pylori as well as gastric parietal cell and intrinsic factor antibodies were negative. She underwent a radical gastric tangential resection. Six months later, serum gastrin was still elevated despite lack of recurrence of tumor.
The clinical picture was suggestive for a hypochlorhydria-related hypergastrinemia with subsequent development of a non-secreting carcinoid. We suggest a periodic endoscopic follow-up in patients with severe hypochlorhydria-related hypergastrinemia in order to earlier detect neuroendocrine polyps.
Carcinoid; Gastric polyp; Gastrin; Gastrinoma; Zollinger–Ellison syndrome
Arhinia, congenital absence of the nose, is a rare malformation. We present the third reported case of arhinia accompanied by hypogonadism and demonstrate that this is due to gonadotropin deficiency.
A 13-year-old Caucasian boy with congenital arhinia presented for evaluation of delayed puberty and micropenis. We examined genes known to be associated with hypogonadotropic hypogonadism for mutations and performed a chromosomal microarray to assess copy number variation.
No mutations in KAL1, FGFR1, PROK2, PROKR2, FGF8, CHD7 and GnRHR were identified in our patient and there were no copy number variations observed that would explain the phenotype. Though studies are limited in such patients, we suggest that hypogonadotropic hypogonadism is associated with arhinia and that the two entities likely result from a common genetic cause that affects early nasal development and gonadotropin-releasing hormone neuron formation or migration.
Acute cerebral edema is a significant cause of death in patients treated for diabetic ketoacidosis and hyperglycemic hyperosmolar syndrome.
We present the case of a 44-year-old African American woman admitted with acute severe headache and diagnosed with diabetic hyperglycemic hyperosmolar syndrome. Computed tomography of the head showed diffuse leukoencephalopathy, but sparing of the cortex. We were concerned for acute cerebral edema secondary to hyperglycemic hyperosmolar syndrome. Magnetic resonance imaging of the brain showed numerous collections of cystic spaces in the white matter of both hemispheres representing tumefactive perivascular spaces. Her headache improved with correction of the hyperglycemic hyperosmolar state.
Although the clinical presentation and head computed tomography were concerning for cerebral edema, the distinctive features on brain magnetic resonance imaging helped to clarify the diagnosis and differentiate it from other processes.
Tumefactive perivascular spaces; Cerebral edema; Hyperglycemic hyperosmolar syndrome
Leiomyoma of the mammary papilla is one of the most uncommon nipple tumors with only 50 cases reported in the literature until now. To the best of our knowledge we present the first report of a nipple leiomyoma that originated from a traumatic abrasion caused by breastfeeding.
A 35-year-old healthy Caucasian female with a cauliflower-like tender and pink nodular mass that was approximately 10mm in diameter presented to our out-patients department. The patient suggested that the mass originated from a traumatic abrasion caused by breastfeeding three years ago and it has been slowly growing ever since.
An excision biopsy was performed. The histological and immunohistochemical examination confirmed the diagnosis of leiomyoma. There were no postoperative complications or any sign of local recurrence four years postoperatively.
Leiomyoma of the mammary papilla is a rare benign neoplasm that usually appears as a solid tender nodule. Differential diagnosis comprises breast carcinoma, leiomyosarcoma and myoid hamartoma. The recommended treatment is complete excision of the tumor with histologically confirmed tumor-free margins otherwise recurrence is possible. A detailed history of the patient’s disease can reveal the original etiology. This is an original case report that will have particular interest to plastic surgeons, dermatologists, and pathologists. The pathogenetic mechanism was trauma of the nipple. According to our review of the literature this particular information has never been reported and we think that it may advance our knowledge of this very infrequent tumor.
Benign tumors; Nipple; Leiomyoma
Benign neonatal hemangiomatosis is a condition in which multiple cutaneous hemangiomas appear at birth or shortly thereafter; visceral complications are absent. Here, we report a case of a consumption hypothyroidism in an Egyptian baby with benign neonatal hemangiomatosis.
An 8-month-old Egyptian boy with benign neonatal hemangiomatosis was referred to our institution for evaluation of developmental delay. Initial examination revealed a quiet baby who was able to sit only with support. He had hypotonia, a large anterior fontanelle, puffy eyes, cold extremities, hypothermia, bradycardia, and abdominal distension. An examination of his skin revealed more than 100 dome-shaped red-purple cutaneous hemangiomas that varied in size from 5 to 10mm on the back, the abdomen and the extremities without mucus membrane involvement. He had low serum free thyroxine concentration and triiodothyronine levels and high thyroid-stimulating hormone and reverse-triiodothyronine levels. A work-up that involved appropriate imaging ruled out visceral involvement. Based on the above mentioned data, a diagnosis of consumptive hypothyroidism due to benign neonatal hemangiomatosis was made. He was started on oral thyroid medication which was gradually increased to 90μg L-thyroxine daily (15μg/kg/day). After three months of treatment, he was able to sit alone without support and he had normal levels of thyroid-stimulating hormone and serum free thyroxine.
Thyroid function should be assessed periodically in babies with benign neonatal hemangiomatosis, especially if symptoms of hypothyroidism appear or the size and number of hemangiomatosis increase rapidly. Moreover, high doses of L-thyroxine may be needed to achieve euthyroidism during the infancy.
Tuberculosis is still endemic in Morocco and the urogenital form is common. This form is characterized by clinical polymorphism. However, the isolated ureteric form is very rare. The differential diagnosis might be raised in tumoral cases while undertaking surgical excision which is the realistic choice. Hence, we report an isolated ureteric tuberculosis case, and we discuss the clinical, imaging, diagnostic and therapeutical features.
A 30-year-old Moroccan man consulted us for left back pain associated with urinary frequency and a few macroscopic episodes of hematuria for the past six months. A computed tomography urography revealed a left hydronephrosis and hydroureter secondary to focal wall thickening of the left lumbar ureter. Hence, we had diagnosed a ureteral tumor. However, a clinical examination showed irritative voiding symptoms and epididymal disorders associated with prostate infection suggesting a Koch’s bacillus assessment of the patient’s urine of which the results proved strongly positive. The treatment consisted of establishing a double-J ureteric stent to drain the left kidney, followed by antituberculous antibiotics.
Urogenital tuberculosis is common in endemic countries, however isolated ureter affection is rare. It is important to consider a ureteral tuberculosis diagnosis whenever ureteral thickening is revealed in a patient living in a country in which tuberculosis is endemic.
Pseudotumor; Tuberculosis; Ureter
Although blunt trauma to a hernia-containing bowel is known to cause bowel perforation, this report documents the first incident of a small bowel transection following a non-traumatic event.
We report the case of a 49-year-old African American man with a chronic incarcerated inguinal hernia awaiting elective repair. He presented to the Emergency Department with abdominal pain following an episode of coughing. On examination, he was found to have peritonitis. He underwent exploratory laparotomy, and had a complete small bowel transection. A bowel resection with primary anastomosis was performed, as well an inguinal hernia repair.
Chronic hernia incarceration can lead to weakening and ischemia of the bowel, and minimal trauma can lead to perforation of the weakened segment. In such presentations, bowel resection and repair of the defect with a biological material is safe and feasible.
Bowel transection; Hemoperitoneum; Inguinal hernia
Linezolid-induced black hairy tongue has been rarely reported. The purpose of this paper is to report a case of linezolid-induced black hairy tongue and review the literature.
A 56-year-old Caucasian man was admitted with community-acquired pneumonia that failed to respond to levofloxacin 750mg daily. He was started on linezolid and meropenem and was subsequently discharged home on oral linezolid 600mg every 12 hours and intravenous ertapenem 1g daily. On a follow-up clinic visit, day 14 of linezolid therapy, he complained of dysgeusia and his tongue examination was consistent with black hairy tongue. After he finished his antibiotic course, his complaints resolved with regular tongue brushing.
Black hairy tongue is characterized by abnormal hypertrophy and elongation of filiform papillae. Five reported cases of linezolid-induced black hairy tongue were identified in a MEDLINE search (from January 2000 to June 2012). The Naranjo Probability Scale revealed a probable adverse drug reaction of linezolid-induced black hairy tongue. Potential contributing factors included other antibiotics, drug–drug interaction and poor oral hygiene. Health care professionals should be aware of the possibility of linezolid-induced black hairy tongue. Thorough history for other possible contributing factors should be obtained. Patients on linezolid should be counseled to perform good oral hygiene.
Oxazolidinone; Linezolid; Back hairy tongue
Common features of autoimmune polyendocrinopathy-candidiasis-ectodermal dysplasia include candidiasis, hypoparathyroidism and hypoadrenalism. The initial manifestation of autoimmune polyendocrinopathy-candidiasis-ectodermal dysplasia may be autoimmune hepatitis, keratoconjunctivitis, frequent fever with or without a rash, chronic diarrhea, or different combinations of these with or without oral candidiasis.
We discuss a profoundly affected 2.9-year-old Caucasian girl of Western European descent with a dramatic response to immunosuppression (initially azathioprine and oral steroids, and then subsequently mycophenolate mofetil monotherapy). At four years of follow-up, her response to mycophenolate mofetil is excellent.
The clinical features of autoimmune polyendocrinopathy-candidiasis-ectodermal dysplasia may continue for years before some of the more common components appear. In such cases, it may be life-saving to diagnose autoimmune polyendocrinopathy-candidiasis-ectodermal dysplasia and commence therapy with immunosuppressive agents. The response of our patient to immunosuppression with mycophenolate mofetil has been dramatic. It is possible that other patients with this condition might also benefit from immunosuppression.
APECED (autoimmune polyendocrinopathy-candidiasis-ectodermal dysplasia); APS (autoimmune polyendocrinopathy syndrome); Autoimmunity; Immunosuppression; Endocrinopathies
Posterior epidural migration of thoracic disc herniation is extremely rare but may occur in the same manner as in the lumbar spine.
A 53-year-old Japanese man experienced sudden onset of incomplete paraplegia after lifting a heavy object. Magnetic resonance imaging revealed a posterior epidural mass compressing the spinal cord at the T9-T10 level. The patient underwent emergency surgery consisting of laminectomy at T9-T10 with right medial facetectomy, removal of the mass lesion, and posterior instrumented fusion. Histological examination of the mass lesion yielded findings consistent with sequestered disc material. His symptoms resolved, and he was able to resume walking without a cane 4 weeks after surgery.
Pre-operative diagnosis of posterior epidural migration of herniated thoracic disc based on magnetic resonance imaging alone may be overlooked, given the rarity of this pathology. However, this entity should be considered among the differential diagnoses for an enhancing posterior thoracic extradural mass.
Intervertebral disc herniation; Posterior migration; Thoracic spine