骨孢子丝菌病：巴西里约热内卢一家参考医院的 41 例病例。

Bone sporotrichosis: 41 cases from a reference hospital in Rio de Janeiro, Brazil.

机构信息

Pós-Graduação em Medicina Tropical, Instituto Oswaldo Cruz, Fundação Oswaldo Cruz, Rio de Janeiro, Brazil.

Instituto Nacional de Infectologia Evandro Chagas, Fundação Oswaldo Cruz, Rio de Janeiro, Brazil.

出版信息

PLoS Negl Trop Dis. 2021 Mar 17;15(3):e0009250. doi: 10.1371/journal.pntd.0009250. eCollection 2021 Mar.

DOI:10.1371/journal.pntd.0009250

PMID:33730026

原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8007180/

Abstract

BACKGROUND

Bone sporotrichosis is rare. The metropolitan region of Rio de Janeiro is hyperendemic for zoonotic sporotrichosis and the bone presentations are increasing.

METHODS

We studied a retrospective cohort of 41 cases of bone sporotrichosis, diagnosed from 1999-2016. The inclusion criteria was fungal culture isolation from any clinical specimen associated to bone involvement (radiography and/or computed tomography) compatible with fungal osteomyelitis or histopathological findings of bone material compatible with sporotrichosis. Molecular identification was performed when possible.

RESULTS

Male patients represented 58.5% of the cases, with a cohort median age of 43 years. Immunosuppressive conditions were present in 68.3% of the patients, mostly HIV coinfection (51.2%). Multifocal bone involvement (more than one anatomical segment) was diagnosed in 61% of the patients, while 39% presented unifocal involvement. The bones of the hands were the most affected (58.5%), followed by the feet (41.5%) and tibia (26.8%). Multifocal group was characterized by a higher proportion of males (p = 0.0045) with immunosuppressive conditions (p = 0.0014). Amphotericin B followed by oral itraconazole was the main treatment, with a median time of 16.7 months (1.5 to 99.2 months), and cure of 53.7% of the patients (84.6% of immunocompetent and 39.3% of immunocompromised patients). Sequelae occurred in 12.2% of the patients-amputations (7.3%) and ankylosis (4.9%), while 22% died in the course of the disease. Sporothrix brasiliensis was the causative agent in all the 9 (22%) performed cases.

CONCLUSIONS

Bone sporotrichosis is a chronic, challenging condition with prolonged treatment, often with poor results and sequelae.

摘要

背景

骨孢子丝菌病较为罕见。里约热内卢大都市区为动物源性孢子丝菌病的高度流行区，骨骼表现也在增加。

方法

我们研究了一个回顾性队列，纳入了 1999 年至 2016 年间诊断的 41 例骨孢子丝菌病患者。纳入标准为从任何与骨骼受累相关的临床标本（放射学和/或计算机断层扫描）中分离出真菌，且这些标本与真菌性骨髓炎相符，或骨组织的组织病理学发现与孢子丝菌病相符。当可能时进行分子鉴定。

结果

男性患者占病例的 58.5%，队列的中位年龄为 43 岁。68.3%的患者存在免疫抑制状态，其中 51.2%为 HIV 合并感染。61%的患者存在多灶性骨骼受累（超过一个解剖节段），而 39%的患者存在单灶性受累。手部骨骼最易受累（58.5%），其次为足部（41.5%）和胫骨（26.8%）。多灶性组的男性比例较高（p = 0.0045），且存在免疫抑制状态的比例也较高（p = 0.0014）。两性霉素 B 联合口服伊曲康唑是主要治疗方法，中位治疗时间为 16.7 个月（1.5 至 99.2 个月），53.7%的患者治愈（免疫功能正常者为 84.6%，免疫功能低下者为 39.3%）。12.2%的患者发生了后遗症，包括截肢（7.3%）和关节强直（4.9%），22%的患者在病程中死亡。在所有进行了检测的 9 例（22%）中，病原体均为巴西枝孢霉。