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[伴有肉芽肿病及弥漫性T细胞浸润的噬血细胞性淋巴组织细胞增生症,与播散性诺卡菌病及链霉菌属所致肺部感染相关]

[Hemophagocytic lymphohistiocytosis with granulomatosis and diffuse T-cell infiltration associated with disseminated Nocardiosis and pulmonary infection due to Streptomyces spp].

作者信息

Canouï E, Ingen-Housz-Oro S, Ortonne N, Lebeaux D, Rodriguez-Nava V, Godeau B, Mahévas M

机构信息

Service de médecine interne, hôpital Henri-Mondor, AP-HP, 51, avenue du Maréchal-de-Lattre-de-Tassigny, 94010 Créteil, France.

Service de dermatologie, hôpital Henri-Mondor, AP-HP, 94010 Créteil, France.

出版信息

Rev Med Interne. 2019 Jul;40(7):457-461. doi: 10.1016/j.revmed.2019.04.013. Epub 2019 May 15.

Abstract

INTRODUCTION

Hemophagocytic lymphohistiocytosis (HLH) is a rare syndrome frequently secondary to infectious disease, especially in immuno-compromised patients. We report a HLH secondary to disseminated nocardiosis and Streptomyces spp pulmonary infection.

CASE REPORT

A 69-years-old women had recent subcutaneous nodules of the forearms and loins associated with peripheral neuropathy and pulmonary nodule of the right upper lobe. Cutaneous biopsy revealed granuloma. Cutaneous lesions worsened and the patient developed a HLH with probable cardiac and neurological involvement, associated with cutaneous granulomatosis and diffuse polyclonal lymphocyte proliferation. Nocardia PCR was positive in cutaneous biopsy. Pulmonary samples revealed Streptomyces in culture and Nocardia in PCR. The evolution under antibiotic treatment was favorable.

CONCLUSION

Recent diagnosis of HLH without obvious etiology should lead to etiological investigation, including the search for infections with slow-growing bacteria such as Nocardia or Streptomyces spp.

摘要

引言

噬血细胞性淋巴组织细胞增生症(HLH)是一种罕见综合征,常继发于传染病,尤其是免疫功能低下的患者。我们报告一例继发于播散性诺卡菌病和链霉菌属肺部感染的HLH。

病例报告

一名69岁女性近期出现前臂和腰部皮下结节,伴有周围神经病变和右上叶肺结节。皮肤活检显示肉芽肿。皮肤病变恶化,患者发展为可能累及心脏和神经系统的HLH,伴有皮肤肉芽肿病和弥漫性多克隆淋巴细胞增殖。皮肤活检中诺卡菌聚合酶链反应(PCR)呈阳性。肺部样本培养发现链霉菌,PCR检测发现诺卡菌。抗生素治疗后病情好转。

结论

近期诊断为无明显病因的HLH应进行病因调查,包括寻找生长缓慢的细菌感染,如诺卡菌或链霉菌属。

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