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免疫功能正常患者中表现为肾上腺功能不全的肾上腺组织胞浆菌病。

Adrenal Histoplasmosis in Immunocompetent Patients Presenting as Adrenal Insufficiency.

作者信息

Gajendra Smeeta, Sharma Rashi, Goel Shalini, Goel Ruchika, Lipi Lipika, Sarin Hemanti, Guleria Mridula, Sachdev Ritesh

机构信息

Department of Pathology & Laboratory Medicine, Medanta the Medicity, GURGAON, INDIA.

出版信息

Turk Patoloji Derg. 2016;32(2):105-11. doi: 10.5146/tjpath.2015.01349.

Abstract

OBJECTIVE

Histoplasmosis is an infectious disease caused by the dimorphic fungus Histoplasma capsulatum, endemic in central and eastern states of United States, South America and Africa. India is considered to be non-endemic area for histoplasmosis. Disseminated histoplasmosis may affect almost all systems. Disseminated histoplasmosis with asymptomatic adrenal involvement has been described in immunocompromised patients; whereas isolated adrenal involvement with adrenal insufficiency as the presenting manifestation of the disease is rare.

MATERIAL AND METHOD

Twelve patients from a non-endemic area with adrenal histoplasmosis, who were immunocompetent and diagnosed as adrenal histoplasmosis by cytology/histopathology between January 2012 to December 2014 were studied. 18F-FDG PET/CT (fluorodeoxyglucose positron emission tomography/computed tomography) was used to assess the extent of involvement.

RESULTS

There were a total of 12 immunocompetent males (mean age: 56.9 years). Ten patients had bilateral adrenal involvement and two had a unilateral left adrenal mass. All the patients had histopathologically/cytologically proven adrenal histoplasmosis. Two patients had simultaneous histoplasmosis of other sites, one in the epiglottis and the other in the alveolus. 18F-FDG PET/CT was performed in 10 patients showing high FDG uptake in the adrenals. All these patients received Amphotericin B and/or Itraconazole treatment that led to symptomatic improvement.

CONCLUSION

A diagnosis of invasive fungal infection requires a high index of suspicion, especially in immunocompetent patients who present with nonspecific symptoms, clinical signs, laboratory and radiological features that can resemble adrenal neoplasms. Clinical specimens must be sent for cytopathology/histopathology together with fungal culture for a definite diagnosis and appropriate management.

摘要

目的

组织胞浆菌病是一种由双相真菌荚膜组织胞浆菌引起的传染病,在美国中部和东部各州、南美洲和非洲流行。印度被认为是组织胞浆菌病的非流行地区。播散性组织胞浆菌病可累及几乎所有系统。免疫功能低下的患者中曾有播散性组织胞浆菌病伴无症状肾上腺受累的报道;而以肾上腺功能不全为首发表现的孤立性肾上腺受累则较为罕见。

材料与方法

对2012年1月至2014年12月期间来自非流行地区的12例肾上腺组织胞浆菌病患者进行研究,这些患者免疫功能正常,通过细胞学/组织病理学诊断为肾上腺组织胞浆菌病。使用18F-FDG PET/CT(氟脱氧葡萄糖正电子发射断层扫描/计算机断层扫描)评估受累范围。

结果

共有12例免疫功能正常的男性患者(平均年龄:56.9岁)。10例患者双侧肾上腺受累,2例患者左侧肾上腺有单侧肿块。所有患者经组织病理学/细胞学证实为肾上腺组织胞浆菌病。2例患者同时存在其他部位的组织胞浆菌病,1例在会厌,另1例在肺泡。10例患者进行了18F-FDG PET/CT检查,显示肾上腺有高FDG摄取。所有这些患者均接受了两性霉素B和/或伊曲康唑治疗,症状有所改善。

结论

侵袭性真菌感染的诊断需要高度怀疑,尤其是对于出现非特异性症状、临床体征、实验室和影像学特征类似于肾上腺肿瘤的免疫功能正常患者。必须将临床标本送检进行细胞病理学/组织病理学检查以及真菌培养,以明确诊断并进行适当治疗。

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