Cutaneous Penicillium marneffei Infection and Mycobacterial Spindle Cell Pseudotumor in a Patient with Rheumatoid Arthritis-A Case Report-

一類風濕性關節炎病人之皮膚嗎爾內菲青黴菌感染及分枝桿菌梭狀細胞假瘤－病例報告－

楊朝鈞(Chao-Chun Yang) ； 李玉雲(Julia Yu-Yun Lee) ； 許明隆(Mark Ming-Long Hsu)

《中華皮膚科醫學雜誌》 22卷4期 (2004/12) Pp. 338-344

摘要

嗎爾內菲青黴菌(Penicillium marneffei)會造成散佈性及進行性的感染，最常侵犯的部位為皮膚、血液、骨髓及淋巴結，并伴隨有發燒、貧血、體重減輕。分枝桿菌梭狀細胞假瘤(mycobacterial spindle cell pseudotumor, MSP)是一種特殊且罕見的分枝桿菌感染形式，多半由Mycobacterium avium-intracellulare (MAI)所引起，最常侵犯部位是淋巴結。MSP的病理上呈現梭狀細胞之增生，容易與其他梭狀細胞增生之腫瘤混淆。這兩種疾病大多發生在免疫不全的病人，特別是後天免疫不全症候群(HIV, AIDS)的病人，而且在臨床及病理表現上容易與其他伺機性微生物感染混淆。我們報告一例罕見的嗎爾內菲青黴菌感染及MSP於一位無人類免疫不全病毒感染，患類風濕性關節炎的病人身上。臨床上此病人的嗎爾內菲青黴菌感染於左胸壁表呈現多發性皮膚潰瘍；MSP則於右大腿以潰瘍性結節表現。前者的病理檢查發現有許多橢圓狀、具有中隔的雙細胞；後者則呈現瀰漫性的梭狀細胞浸潤，梭狀細胞具有空泡狀細胞質及許多酸耐受性桿菌。黴菌培養結果确定了前者的診斷；分枝桿菌培養及聚合酵素鏈鎖反應結果确定MAI的診斷。此病人在診斷MSP的前兩年期間，患有雙腳大拇趾慢性骨髓炎併引流瘻管形成，其病因不明。回頭重新查看病人曾經清創除下來的檢體，發現有眾多的酸耐受性桿菌，确定為分枝桿菌感染。此病例為第一例同時發生這兩種罕見的感染的病例。臨床及病理醫師在面對免疫不全的病人時必須對這兩種疾病提高警覺，以期能及時診斷及治療。

並列摘要

Penicillium marneffei infection is characterized by disseminated and progressive clinical course and mainly involves the skin, blood, bone marrow and lymph nodes with fever, anemia and weight loss. Mycobacterial spindle cell pseudotumor (MSP) is a peculiar and very rare form of mycobacterial infection mainly caused by Mycobacterium avium-intracellulare (MAI), and most commonly affects lymph nodes. MSP resembles spindle cells neoplasm pathologically and may be easily misdiagnosed. Both infections mainly occur in immunocompromised hosts, especially in patients with AIDS, and are easily confused with each other or other opportunistic infections clinically or/and pathologically. We reported a very rare case of cutaneous P marneffei infection on the anterior chest with a separate MSP on the right thigh caused by MAI infection in a HIV-negative immunocompromised woman with chronic rheumatoid arthritis. The P marneffei infection manifested multiple ulcers while the MSP presented as an ulcerated nodule. Biopsy specimens revealed numerous oval-shaped yeasts, some two-celled with a central septum (binary fission) in the former, and a dense diffuse infiltrate of spindle cells with vacuolated cytoplasm containing numerous acid-fast bacilli in the latter. The former was confirmed by fungal culture while the latter was proved by bacterial culture followed by polymerase chain reaction study. The cutaneous MSP was preceded by a chronic osteomyelitis with draining fistulae in both big toes for 2 years. The diagnosis of mycobacterial osteomyelitis was made retrospectively after reviewing the bone curettage specimens that revealed numerous acid-fast bacilli. This case represents the first case with two such rare infections. Clinicians and pathologists should be aware of these rare types of infection in immunocompromised hosts, so that the diseases could be diagnosed and treated in a timely fashion.

並列關鍵字

Penicillium marneffei ； Mycobacterial spindle cell pseudotumor ； Mycobacterium aviumintracellulare ； Skin ； Osteomyelitis