Papulonecrotic tuberculid--a rare skin manifestation in a patient with pulmonary tuberculosis.

Papulonecrotic tuberculid (PNT), a cutaneous manifestation of tuberculosis, is rare even in areas endemic for tuberculosis such as Taiwan. Concomitant pulmonary tuberculosis is uncommon in patients with PNT. We describe a patient with rare skin manifestations and simultaneous pulmonary tuberculosis. This 48-year-old woman had suffered from productive cough for 4 months, and pea-sized papules with central umbilication were noted over the extensor surface of her four extremities and lower abdomen 1 month prior to admission to our hospital. Chest roentgenography showed reticulonodular lesions with small cavitation over the lower right lung field, and sputum culture yielded Mycobacterium tuberculosis complex. PNT was diagnosed using the skin biopsy results and the papules healed with scar formation after antituberculous therapy.     

Penile tuberculosis associated with monoclonal gammopathy of undetermined significance.

Mycobacterium tuberculosis (TB) infection of the penis is a rare but serious problem. We report a case of penile TB in a 75-year-old man who presented with fever and dyspnea. No active lung lesions except pleural and pericardial effusion were found on chest X-ray. Monoclonal gammopathy of undetermined significance was diagnosed after serum and urine electrophoresis studies, and repeated bone marrow studies. Genital examination showed diffuse papulonecrotic skin ulcers involving the whole penile shaft, extending ventrally to the median raphe of the scrotum. Pus smear showed positive acid-fast stain, and culture yielded M. tuberculosis. Culture of pleural and pericardial effusion was also positive for M. tuberculosis. Anti-TB treatment was given with isoniazid, ethambutol, rifampin and pyrazinamide, and the cutaneous lesion was noted to be healed at follow-up 6 months later. Although rare, the possibility of TB as a cause of genital ulcer should be kept in mind.     

Acute respiratory distress syndrome due to tuberculosis in a child after allogeneic bone marrow transplantation for acute lymphoblastic leukemia.

We report the occurrence of tuberculosis in a 10-year-old Taiwanese boy, approximately 4 months after he received a matched-related bone marrow transplantation from his sister for acute T-cell lymphoblastic leukemia. After transplantation, grade III acute graft-versus-host disease developed and the patient was treated with prednisolone and cyclosporine. Marrow failure was noted on day 77 post-transplantation, however, after an episode of herpes zoster infection. Interstitial pneumonia, diagnosed on the basis of chest x-ray and computed tomography findings, occurred on day 120. Histologic examination of an open-lung biopsy specimen showed caseating granulomas and a few acid-fast bacilli. The patient died of acute respiratory distress syndrome, despite immediate implementation of antituberculosis therapy. Sputum cultures grew Mycobacterium tuberculosis 5 weeks later. This report demonstrates that the possibility of tuberculosis needs to be considered in immunocompromised patients, and that appropriate prophylaxis should be instituted in areas where tuberculosis is endemic.     

Congenital thrombotic thrombocytopenic purpura: Upshaw–Schulman syndrome: a cause of neonatal death and review of literature

Thrombotic thrombocytopenic purpura (TTP) is a rare disorder in children characterized by microangiopathic hemolytic anemia (MAHA) and thrombocytopenia. The classic Moschcowitz Pentads of TTP include hemolytic anemia, with fragmentation of erythrocytes, thrombocytopenia, diffuse and non-focal neurologic findings, decrease renal function and fever. We report a newborn who was diagnosed with congenital TTP. The newborn was admitted at age of 40 h, in our hospital, in view of respiratory distress with impending respiratory failure and red colored urine. On examination, the newborn was febrile, tachypneic, had deep icterus, pallor and no hepatosplenomegaly. Family history was significant with one unexplained neonatal death at age of 24 with symptoms of red colored urine. Examination of peripheral smear was diagnostic with the presence of fragmented RBCS, giant but fewer platelets consistent with a diagnosis of MAHA. The diagnosis of TTP was confirmed with low ADAMTS activity and gene analysis showed c 2203 G > T-p.Glu735X (domain TSP1-2) mutation in exon 18 of ADAMTS 13 gene. The newborn had rapid deterioration, with respiratory distress and refractory shock leading to death. Post-mortem bone marrow done showed marrow hyperplasia.     

Severe community-acquired pneumonia due to Legionella pneumophila Serogroup 6.

Legionella pneumophila is a common cause of sporadic community-acquired pneumonia, but culture-proven legionellosis is rarely diagnosed. There is no laboratory test for Legionnaires' disease that can detect all patients with the disease. Culture is the standard diagnostic method and should be initiated as soon as possible in suspected cases. We describe a rare case of community-acquired pneumonia caused by L. pneumophila serogroup 6. A 77-year-old man was admitted to a tertiary care hospital because of high fever, productive cough, and progressive dyspnea. Chest radiography showed bilateral pneumonia, which led to respiratory failure necessitating mechanical ventilatory support. Despite antibiotic therapy, his condition continued to deteriorate and acute renal failure also developed. Urine was negative for L. pneumophila. Culture of the sputum yielded L. pneumophila serogroup 6, although there was no elevation of the serum antibody titer. Pneumonia resolved gradually and he was extubated after treatment with levofloxacin followed by erythromycin. L. pneumophila other than serogroup 1 should be included in the differential diagnosis of patients with suspected atypical community-acquired pneumonia.     

Disseminated peritoneal tuberculosis mimicking advanced-stage endodermal sinus tumor: a case report

Abstract.   Dursun P, Ersoz S, Gultekin M, Aksan G, Yüce K, Ayhan A. Disseminated peritoneal tuberculosis mimicking advanced-stage endodermal sinus tumor: a case report. Int J Gynecol Cancer 2006; 16(Suppl. 1): 303–307.
It is well known that peritoneal tuberculosis may mimic advanced-stage epithelial ovarian carcinoma because of similar clinical, radiologic, and laboratory findings. However, disseminated peritoneal tuberculosis mimicking advanced-stage endodermal sinus tumor (ESS) has not been reported previously. An 18-year-old nulliparous woman came with the complaint of pelvic pain and weight loss. Imaging studies demonstrated that she had multiple peritoneal implants and left adnexial mass. Also, laboratory studies showed elevated CA125 and alpha fetoprotein levels suggesting an initial diagnosis of ESS. However, intraoperative frozen section examination showed caseous necrosis, and she was diagnosed as having disseminated peritoneal tuberculosis. Two months after the initial exploration, the patient required liver transplantation because of hepatic failure due to widespread hepatic involvement of the tuberculosis. Concomitant peritoneal and hepatic involvement of tuberculosis may cause false elevation of multiple tumor markers of gynecological cancers and may lead to misdiagnosis and mismanagement of patients. Elevation of these markers should be carefully investigated especially in premenopausal women. To our knowledge, this is the first reported case of peritoneal tuberculosis misdiagnosed as endodermal sinus tumor.     

Hepatosplenic actinomycosis in an immunocompetent patient

Hepatosplenic abscess caused by Actinomyces is rare and often misdiagnosed as malignancy. Herein, we report a case of hepatosplenic actinomycosis in a 37-year-old immunocompetent man with a 2-month clinical history of intermittent fever and upper left abdominal pain. Physical examination revealed a mildly ill-appearing man with a low-grade fever (38°C) and upper left quadrant abdominal tenderness. Abdominal sonographic examination showed the presence of a 6.3?cm?×?6.5?cm heterogeneous abscess with a hypoechoic center and honeycomb appearance in an enlarged spleen (8?cm?×?5?cm). Computerized tomography of the abdomen revealed a multiloculated splenic lesion, and laparotomy showed multiple hepatic nodules and a splenic abscess. Histopathological examination of the biopsy revealed filamentous branching bacilli and sulfur granules in the hepatosplenic abscess. The patient successfully underwent splenectomy accompanied by intravenous and oral penicillin treatment. Proper and prompt diagnosis of hepatosplenic actinomycosis is important because the therapeutic plan and prognosis of this pathogen are quite different from other microorganisms and malignancies.     

Solitary spleen metastasis and amyloidosis in a patient with endometrial cancer

BACKGROUND: In contrast to autopsy findings, solitary splenic metastases from solid tumors are extremely rare. It may occasionally be the first manifestation of recurrent solid cancers, and in particular of gynecologic malignancies. Secondary amyloidosis is also found in malignancy. CASE: A 52-year-woman originally diagnosed with a Stage IB, grade 2 endometrial carcinoma presented two and a half years later with a paroxysmal non-productive cough, weakness, loss of appetite and daily afternoon fever. On clinical examination wheezing on forced exhalation and painful splenomegaly was found. After an extensive work-up the patient underwent an explorative laparotomy and a splenectomy was performed. Histologic examination showed solitary spleen metastasis with amyloidosis. All symptoms resolved. As the patient had received a full course of postoperative irradiation after a total abdominal hysterectomy, six cycles of combination chemotherapy were administered. The patient remains free of recurrence, 46 months later. CONCLUSION: A case of solitary spleen metastasis with amyloidosis in a patient with endometrial cancer is presented.     

Mononucleosis and hepatic failure associated with diphenylhydantoin treatment in an infant.

Diphenylhydantoin-induced hepatitis and mononucleosis are uncommon in children. The occurrence of these two diseases in the same individual, with progression to hepatic failure is rare and has not been reported in infants. This report represents a 6-month-old male infant who developed an infectious mononucleosis-like syndrome and hepatic failure 16 days after diphenylhydantoin administration. He took this anticonvulsant for controlling seizures after a head injury. Fever, skin rash, hepatosplenomegaly, lymphadenopathy, and atypical lymphocytosis led to the initial diagnosis of infectious mononucleosis. However, negative heterophil antibody did not support the diagnosis. Jaundice ensued in the following course and became more and more profound. Meanwhile, physical examination showed shrinking in liver size. Negative virology studies, including Epstein-Barr virus, cytomegalovirus, and hepatitis B virus, excluded them as causative agents. The patient lapsed into a stage I hepatic coma, but gradually recovered clinically and biochemically after eight successive exchange transfusions and supportive care. Two liver biopsies were performed 20 and 50 days after the onset of disease, respectively. Remarkable hepatic parenchymal loss, cholestasis, and fatty change were found on histologic examination of the first biopsy specimen, and portal fibrosis was noted on the second.     

Treatment of Toxoplasma brain abscess with clindamycin and sulfadiazine in an AIDS patient with concurrent atypical Pneumocystis carinii pneumonia.

Toxoplasmosis is the most common opportunistic infection of the central nervous system in patients with AIDS. The standard treatment for toxoplasmic encephalitis is pyrimethamine and sulfadiazine. There have been few reports of concurrent Toxoplasma brain abscess and cavitary Pneumocystis carinii pneumonia (PCP) in Taiwan. We report the case of a 26-year-old homosexual man with coexisting infection with Toxoplasma gondii and P. carinii who was successfully treated for brain abscess with clindamycin and sulfadiazine. The cavitary lung lesions, initially diagnosed as pulmonary tuberculosis, were proved to be PCP by lung biopsy. HIV infection and syphilis had been diagnosed 1 year before admission. He presented with general weakness, ataxia, nausea, blurred vision and fever for 2 weeks. Magnetic resonance imaging of the brain revealed multiple ring-enhanced lesions over the cerebrum and cerebellum. Chest roentgenography showed a 3-cm lesion with cavitation over the right upper lung field. Diagnostic computerized tomography-guided lung biopsy revealed P. carinii cysts. Clindamycin, sulfadiazine and trimethoprim (TMP)-sulfamethoxazole (20 mg/kg/day TMP) were given with good response. His CD4 count rose from 40 to 280/microL 4 months later. All antibiotics were discontinued after 4.5 months due to the development of a skin rash. He was well at follow-up 1 year later. This case suggests that the combination of clindamycin and sulfadiazine is an effective treatment for Toxoplasma brain abscess and highlights the importance of diagnostic lung biopsy for cavitary lung lesions, particularly in a region endemic for tuberculosis.     

甲羟戊酸尿症1例报道并文献复习

目的　报道1例甲羟戊酸尿症（MVA）患儿的临床特点、诊断、治疗及随访,以提高对该病的认识。方法　收集2006年7月于广州市妇女儿童医疗中心就诊的1例患儿的临床资料,进行分析并长期随防,复习文献总结分析其临床特点、诊疗及预后。结果　患儿男,6岁6个月,以反复发热,伴有淋巴结、肝脾大为主要临床表现,血沉增快,超敏C反应蛋白、白细胞计数、血IgA均增高,尿液气相色谱质谱联用技术（GC/MS）检测显示甲羟戊酸排泄明显增高,给予辅酶Q10、维生素C及维生素E等治疗,随诊4年8个月,发热发作渐有减少,约1～2次/月,伴有紫癜样皮疹、关节痛反复发作,淋巴结、肝脾大无进行性加重,智力轻度低下。结论　临床对不明原因反复发热患者,尤其是合并有淋巴结、肝脾大或神经系统症状时需要警惕MVA可能,进一步做尿GC/MS分析等早期诊断,以避免误诊误治。本病目前主要是对症治疗,部分患儿发热随年龄增长有减少趋势。     

经颈动脉超声诊断肥胖青少年早期动脉粥样硬化的探讨

目的探讨经颈动脉超声早期诊断肥胖青少年动脉粥样硬化价值。 方法于2006-07在丹东市中学随机抽取47例中、重度单纯性肥胖青少年并与36例正常青少年作对照,分别进行颈部血管超声检查。 结果肥胖组动脉内膜呈现不同程度的毛糙、不光滑、连续性差、限局性增厚，颈总动脉内膜中层厚度(IMT)明显高于对照组(P<0.05)，肥胖组颈 总动脉早期硬化的改变与肥胖程度正相关(P<0.05)。 结论青少年单纯性肥胖可以并发动脉粥样硬化，经颈总动脉超声检查可作为早期诊断动脉粥样硬化首选方法。     

Linfangioleiomiomatosis y embarazo. Reporte de caso

Lymphangioleiomyomatosis is a rare progressive cystic lung disease that occurs almost exclusively in women, generally between menarche and menopause. We report the case of a 33-year-old patient (gravida III, para II) with a 32-week pregnancy who presented with non-productive cough and progressive dyspnea. The physical examination was normal except for high respiratory frequency. Ultrasound examination revealed a viable live fetus corresponding to 31 weeks. High-resolution computed tomography revealed the presence of emphysematous and cystic changes in both basal lung segments and fluid in the bronchi. The bronchial biopsy report described diffuse and pronounced fibromuscular thickening and a sub-mesothelial layer with proliferation of smooth muscle cells and fibroblasts.     

Imaging findings of intradiaphragmatic bronchogenic cyst: a case report.

Isolated intradiaphragmatic bronchogenic cysts are extremely rare. Here, we report a case of intradiaphragmatic bronchogenic cyst with calcifications in a 34-year-old man presenting with no clinical symptoms. Imaging studies of the chest, including chest roentgenogram and computerized tomography (CT), revealed a left posterior mediastinal mass that abutted onto the left diaphragmatic crus with multiple calcifications and showed no enhancement after contrast administration on CT. The patient underwent surgical resection of the mass. Intradiaphragmatic bronchogenic cyst with calcifications was diagnosed by pathology. The patient was discharged 10 days after the operation, and no complication was found 3 months later at follow-up.     

Choledochal cyst diagnosed and conservatively treated during pregnancy

We report herein a rare case of choledochal cyst diagnosed at 14 weeks gestation and treated with percutaneous transhepatic drainage until postpartum. A 26-year-old primigravid woman at 14 weeks gestation presented with epigastric pain, slight fever, and nausea of 3 days duration. Abdominal ultrasonography revealed a 6-cm-diameter cystic mass between the porta hepatis and the pancreas head, which was diagnosed as a type I choledochal cyst. At 18 weeks of gestation, her upper abdominal pain became severe, and the size of the choledochal cyst increased to 12 cm in diameter. Laboratory data revealed a biliary obstruction. Percutaneous transhepatic drainage was performed immediately. She delivered a healthy male baby by elective cesarean section at 37 weeks of gestation. At 6 weeks postpartum, the patient underwent surgical excision of the choledochal cyst, cholecystectomy, and formation of a Roux-en-Y hepaticojejunostomy. Although choledochal cysts are rare during pregnancy, obstetricians should be familiar with the condition to ensure prompt diagnosis and adequate definitive management, given that the implications of missed or delayed diagnosis may be detrimental to both mother and fetus.     

Hepatic abscess caused by Aspergillus fumigatus infection following splenectomy and immunosuppressive therapy.

Hepatic abscess caused by Aspergillus fumigatus infection is rare. The incidence of fungal hepatic abscess has recently increased. We report a case of Aspergillus fumigatus infection in a 66-year-old man with aplastic anemia who presented with intermittent high fever. He had received splenectomy about 2 years before this admission followed by treatment with anti-human thymocyte globulin, corticosteroids, and cyclosporin. Abdominal sonography and computerized tomography scan of the liver revealed a hepatic abscess and empiric broad-spectrum antibiotics were administered, but fever persisted. Culture of abscess aspirate yielded Aspergillus fumigatus. Amphotericin B was administered, but the patient died of sepsis. With the increasing number of immunocompromised patients, various fungal infections, including Aspergillus fumigatus, are increasingly common, and this infection can be very serious with fatal outcome. Alertness to the possibility that fungal infection may be present when an immunocompromised patient with hepatic abscess presents in hospital is important to decreasing morbidity and mortality.     

Gastric carcinoma diagnosed at the third trimester: a case report

Gastric carcinoma associated with pregnancy appears to be an extremely rare entity. It is usually diagnosed at advanced stages of the disease and presents a grave prognosis. Since the fatal outcome shows a rapid course within months of diagnosis, prompt gastroduodenal endoscopic examination following early clinical suspicion is mandatory. A case of 29 year–old pregnant woman with 31 weeks of gestation is herein presented. The women was diagnosed as gastric adenocarcinoma while being investigated for intractable nausea, vomiting, and a concomitant epigastric mass. She underwent radical gastric resection and received six cycles of adjuvant chemotherapy following vaginal delivery of a healthy 1950 g fetus. The patient was dead 20 weeks after the surgery due to pulmonary and hepatic metastasis.     

Emphysematous prostatitis caused by Klebsiella pneumoniae.

Emphysematous prostatitis is a rare condition that is characterized by gas and abscess accumulation in the prostate. We report a 60-year-old man with emphysematous prostatitis caused by Klebsiella pneumoniae. He had a history of recently diagnosed diabetes mellitus and a 16-year history of alcoholic liver cirrhosis. He was admitted due to fever, dysuria and difficult urination. Physical examination revealed lower abdominal tenderness and prostatic fluctuance on digital examination. Leukocytosis, pyuria and elevated C-reactive protein were found. Abdominal radiography disclosed a collection of abnormal air pockets in the lower pelvic cavity and computed tomography scans corroborated the existence of extensive air collection in the prostate. Under the impression of emphysematous prostatitis, the patient was successfully treated with transurethral incision of the prostate and antibiotics for 6 weeks; there were no urinary sequelae during 6 months of follow-up.     

Gastroschisis with fetal chromosomal abnormality: a case report

Gastroschisis is a rare anomaly and it is usually not associated with other syndromic or nonsyndromic anomalies. The first case of gastroschisis with aneuploidy (Turner syndrome) is presented. A fetal huge cystic hygroma was diagnosed by prenatal sonography at 12 weeks of pregnancy and chorionic villi sampling (CVS) was performed. Cytogenetic analysis revealed 45, X0. The pregnancy was terminated by induction of labor at 16 weeks of pregnancy. The female fetus had a big membrane of cystic hygroma surrounding the fetal neck. Additionally, a full abdominal thickness defect with multiple loops of bowel outside the abdomen, which could not be diagnosed on prenatal ultrasound scan, was detected on postnatal examination.     

Endometrial tuberculosis: a clinical case

Genital tuberculosis is a bacterial infection still frequent in less developed countries where lots of cases are not diagnosed nor treated. In this work we describe a rare case of primary endometrial tuberculosis in a woman of 50 years old. The diagnosis was confirmed by an ultrasonography of the pelvis and an endometrial biopsy followed by a histological examination. The patient after the diagnosis was put under antiturbecular treatment for six months with complete healing.