<Emphasis Type=Italic>Histoplasma capsulatum</Emphasis> Sinusitis: Case Report and Review

Histoplasma capsulatum has not typically been associated with sinusitis in either immunocompetent or immunocompromised hosts. We report a case of sinusitis caused by H. capsulatum in a patient with chronic lymphocytic leukemia and discuss the reported cases of this rare clinical manifestation of histoplasmosis in the medical literature.     

Diabetes Insipidus Associated with Hehmophagocytic Lymphohistiocytosis: First Case Report

ObjectiveTo report the first case of central diabetes insipidus associated with hemophagocytic lymphohistiocytosis.MethodsWe describe the clinical course of a 75-yearold woman who developed febrile illness 1 month after receiving the H1N1 influenza vaccination. Shortly thereafter, she developed central diabetes insipidus, which responded to treatment with intranasal desmopressin acetate. She was then hospitalized with another febrile illness and diagnosed with hemophagocytic lymphohistiocytosis.ResultsMagnetic resonance imaging of the pituitary gland was significant for plaquelike meningeal enhancement and thickening surrounding the infundibulum, which has been previously reported as a manifestation of histiocytosis.ConclusionWe believe this is the first case of central diabetes insipidus associated with hemophagocytic lymphohistiocytosis. (Endocr Pract. 2011;17:e118-e122)     

Xanthoma Disseminatum: Case Report and Literature Review

ObjectiveTo report a case of a young normolipidemic woman with mucocutaneous xanthomas who developed neurogenic diabetes insipidus and hyperprolactinemia because of an inflammatory pituitary stalk lesion.MethodsThe clinical features, laboratory results, magnetic resonance imaging, and pathology findings are presented. In addition, the pertinent literature is reviewed.ResultsA 23-year-old woman presented with a 9-month history of polydipsia, polyuria, galactorrhea, secondary amenorrhea, and weight gain. Her previous medical history included chronic anemia and widespread mucocutaneous xanthomas. Laboratory tests showed hyperprolactinemia, normal electrolytes, and a normal lipid profile. The results of a water deprivation test were compatible with neurogenic diabetes insipidus, and cerebral magnetic resonance imaging showed pituitary stalk thickening. Histologic findings on a skin biopsy specimen supported the diagnosis of non-Langerhans histiocytosis. Treatment was initiated with cabergoline, nasally administered desmopressin, radio-frequency ablation of facial skin lesions, and surgical excision of other accessible lesions.ConclusionXanthoma disseminatum is a rare, benign proliferative disorder characterized by extensive cutaneous and mucous membrane xanthomas in normolipidemic patients. Central nervous system involvement is rare and usually occurs in the systemic variety. Pituitary stalk disease commonly causes hyperprolactinemia, diabetes insipidus, and various degrees of hypopituitarism. The natural history of xanthoma disseminatum usually is benign, but lesions in critical anatomic sites may result in morbidity and mortality. (Endocr Pract. 2010;16:1003-1006)     

Familial Paragangliomas: Case Report and Literature Review

ObjectiveTo report a case of a woman with poorly controlled hypertension who was found to have multiple paragangliomas and mutation in the B subunit of succinate dehydrogenase (mitochondrial complex II).MethodsThe pathogenesis of familial paragangliomas and case management are discussed, and the relevant literature is reviewed.ResultsIn a 68-year-old woman with a long-term history of hypertension, bilateral carotid body paragangliomas had been resected at 24 and 57 years of age. Current laboratory studies showed an elevated plasma normetanephrine level. Computed tomography and magnetic resonance imaging revealed 3 paragangliomas—1 in the middle mediastinum and 2 intra-abdominally. Surgical excision involved a 2-stage procedure with the patient under the same general anesthesia: (1) a median sternotomy without cardiopulmonary bypass and (2) a midline celiotomy for removal of the retroperitoneal paragangliomas. All 3 tumors were focally adherent to adjacent structures (the left atrium, the uncinate process of the pancreas, and the left renal vessels). All tumors were completely excised without complication, and frozen section analysis confirmed that they were paragangliomas. Genetic testing disclosed a germline mutation in SDHB.ConclusionFamilial paraganglioma is a rare autosomal dominant, tumorigenic disorder affecting sympathetic and parasympathetic paraganglia. (Endocr Pract. 2008;14:603-606)     

鞍区颗粒细胞瘤1例的病历分析及相关文献复习

目的：探讨伴有临床症状的鞍区颗粒细胞瘤（granular cell tumour,GCT）的病理组织学起源及其治疗方法。方法：回顾性分析我院收治的1例具有临床症状的GCT患者的治疗状况和病理结果,并结合近20年来关于GCT病理和治疗的相关文献报道,探讨GCT肿瘤病理组织起源和治疗要点。结果：该患者影像学报告怀疑鞍区炎性肉芽肿,给予抗生素治疗后复查MRI显示鞍区占位较前次无改变,增强MRI可见占位明显强化,诊断良性肿瘤,并采取翼点入路手术切除。术后病理证实为神经垂体颗粒细胞瘤。术后患者头痛缓解,尿量、尿比重、垂体内分泌激素等相关指标相继恢复至正常值。结论：GCT很可能起源于神经组织,伴有临床症状的GCT的治疗首选开颅手术,肿瘤残存的患者,术后不必放射治疗,定期复查即可。     

First Report of Greeneria uvicola Causing Bitter Rot of Grape in China

In 2013, bitter rot of grape was observed in Changbei Vineyard located in Nanchang City, Jiangxi Province, China. Greeneria species was consistently isolated from the diseased grape berries (Vitis labruscana cv. Kyoho) at approximately 91% of isolation rate in three independent experiments. The species was identified as Greeneria uvicola based on the morphological characteristics, cultural appearance and sequence analysis. Koch's postulates were fulfilled through pathogenicity tests on detached healthy Kyoho grape berries. To our knowledge, this is the first report of G. uvicola causing bitter rot of grape in China.     

Pituitary Macroadenoma in a Patient with Poems Syndrome in Conjunction with Castleman Disease: First Report of a Case and Review of Related Literature

ObjectiveTo describe the first reported case of a patient with POEMS syndrome (polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin changes) in conjunction with the endocrinologic manifestation of panhypopituitarism due to a large clinically nonfunctioning pituitary adenoma.MethodsWe present the clinical, laboratory, and radiologic details of the case and review the relevant updated literature.ResultsA 48-year-old man with hypopituitarism and progressive polyneuropathy presented to an outside hospital with confusion and diaphoresis. He also had diffuse lymphadenopathy, monoclonal gammopathy, and skin lesions consistent with a diagnosis of POEMS syndrome. Cytopathologic study of a lymph node showed findings consistent with Castleman disease. A large suprasellar mass was found to be the cause of the hypopituitarism.ConclusionPOEMS syndrome is a rare paraneoplastic condition, commonly associated with Castleman disease, that manifests with progressive distal polyneuropathy and a monoclonal plasma cell disorder, often accompanied by endocrinopathy, organomegaly, skin changes, sclerotic bone lesions, ascites, erythrocytosis, and thrombocytosis. Our current patient had all 5 classic features of POEMS syndrome along with some diagnostic elements of Castleman disease, sclerotic bone lesions, and thrombocytosis. To our knowledge, this is the first known reported case of a patient whose endocrinologic manifestation of POEMS syndrome was panhypopituitarism attributable to a large clinically nonfunctioning pituitary adenoma. (Endocr Pract. 2010;16:97-101)     

输尿管子宫内膜异位症伴肾积水1 例报告并文献复习

目的:探讨子宫内膜异位症累及输尿管的诊断和治疗方法。方法:术前诊断为右侧输尿管下段占位病变伴右肾积水的42岁女性患者,行下腹正中切口,探查右侧输尿管开口处可见淡黄色息肉样病变,突入膀胱,输尿管下段增粗并全程扩张积水,行输尿管下段并膀胱袖式切除,输尿管膀胱再植术。术后病理报告为输尿管子宫内膜异位症。结果:术后复查B超示右肾积水较术前恢复,术后予抑那通3.75mg/28d,随访6个月未见复发。结论:对于输尿管占位并上尿路积水的女性患者,除考虑肿瘤外还应考虑子宫内膜异位症可能。手术联合内分泌治疗是治疗输尿管子宫内膜异位伴肾积水的有效方法。     

Mycetoma in China: A Case Report and Review of the Literature

Mycopathologia - Mycetoma is a chronic granulomatous infectious disease that can affect the skin, subcutaneous tissue, fascia and bone. It can be caused by filamentous bacteria or fungi and usually...     

重症显微镜下多血管炎1例报告并文献复习

目的:显微镜下多血管炎(microscopic polyangiitis,MPA)通常易侵犯肾脏,很少累及神经系统,而同时累及肺及中枢神经系统的重症病例较少见。本文旨在探讨重症显微镜下多血管炎(MPA)的临床表现、诊断与治疗,为临床提供借鉴。方法:回顾性分析重症显微镜下多血管炎(MPA)的文献资料,结合本中心病例,54岁女性,尿检异常6年,咳血3年因肾功能衰竭合并心衰入院。结合实验室检查及影像学改变,诊断为重症AASV,考虑MPA可能性大。结果:入院后即给予激素联合环磷酰胺冲击治疗,病情明显好转;3周后出现病情反复,再次激素冲击治疗,效果不如前。继而出现脑出血,病情进一步恶化,最终因肾、肺、脑等多器官功能衰竭于入院5周后死亡。结论:对MPA患者关键在于早发现早治疗;而对缓解重症MPA患者病情,激素联合环磷酰胺冲击治疗在初始阶段有效。     

肺放线菌病1 例报告并文献复习

目的：分析肺放线菌病的临床表现、诊断及治疗,提高对肺放线菌病的认识。方法：回顾性分析我科收治的1 例肺放线菌病 患者的临床资料,并对相关文献进行复习。结果：本例患者,43 岁,男性,以咳嗽、咳血性痰为主要临床表现,胸部CT 提示右肺上 叶结节,经皮肺穿刺活检结果确诊肺放线菌病,青霉素治疗效果好。结论：肺放线菌病是放线菌感染引起的一种少见的呼吸系统 疾病,起病隐匿,呈渐进性过程,临床表现及影像学检查均无特异性,放线菌可在肺部引起化脓性肺炎,并经叶间隙、胸膜侵犯胸 壁、肋骨,形成窦道及破坏骨质。确诊有赖于病理学或微生物学证据,主要可采用青霉素抗感染治疗,在疑似肿瘤的情况下,需通 过外科手术治疗,既可以明确诊断也避免病变进一步引起肺、胸壁等组织的不可逆性破坏。