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由分枝杆菌属感染导致的硬化性肠系膜炎:一例报告。

Sclerosing mesenteritis due to Mycobacterium genavense infection: A case report.

机构信息

Infectious Diseases Unit, Internal Medicine Department, Hospital Universitari Son Espases-IdISBa, Palma, Spain.

Radiology Department, Hospital Universitari Son Llàtzer-IdIsBa, Palma, Spain.

出版信息

Medicine (Baltimore). 2022 Sep 9;101(36):e30351. doi: 10.1097/MD.0000000000030351.

Abstract

RATIONALE

Since its first identification in the early 1990s, Mycobacterium genavense has been considered and opportunistic pathogen. It mainly causes gastrointestinal symptoms, but also disseminated infections in severely immunosuppressed patients. Sclerosing mesenteritis is a long-term complication with high morbidity and mortality. As it is a rare condition, there are no specific guidelines for its management. We report a challenging case of persistent M. genavense infection, and propose surgery as an alternative treatment strategy.

PATIENT CONCERNS

A 38-year-old Caucasian man presented to the emergency room with fever, abdominal pain, and night sweats for 3 months. HIV screening revealed a previously unknown HIV-1 infection, with a CD4 cell count of 216 cell/µL and viral load of 361.000 copies/mL at diagnosis. A body CT-scan showed mild splenomegaly as well as mesenteric and retroperitoneal enlarged lymph nodes. Fine needle aspiration revealed the presence of acid-fast bacilli, but mycobacterial cultures were negative. In the second sample, 16S RNA sequencing yielded a diagnosis of M. genavense infection. Despite 2 years of corticosteroids and antimycobacterial treatment excluding rifampicin due to a severe cutaneous reaction, there was no clinical improvement and an increase in the mesenteric lymph node size was observed, with a sclerosing transformation of the mesentery. A surgical approach was proposed to release small bowel loops and to remove fibrin. A second surgery was required due to an acute peritonitis ought to yeyunal segmental isquemia and perforation. Finally, the patient evolved favorably, and antimycobacterial drugs were suspended without relapse.

LESSONS

Despite a prolonged multidrug strategy, some patients develop persistent M. genavense infection. Once sclerosing mesenteritis is established, clinicians have few treatment options. Surgery should be considered in patients with sclerosing mesenteritis or bowel obstruction. The combination of medical and surgical treatment could be a potential cure for these patients.

摘要

背景

自 20 世纪 90 年代初首次发现以来,分枝杆菌属一直被认为是一种机会性病原体。它主要引起胃肠道症状,但也会在严重免疫抑制的患者中引起播散性感染。硬化性肠系膜炎是一种长期并发症,发病率和死亡率都很高。由于它是一种罕见的疾病,因此没有针对其治疗的具体指南。我们报告了一例持续性分枝杆菌属感染的挑战性病例,并提出手术作为替代治疗策略。

患者关注

一名 38 岁的白人男性因发热、腹痛和盗汗 3 个月就诊于急诊室。HIV 筛查显示他以前未知的 HIV-1 感染,诊断时 CD4 细胞计数为 216 个/μL,病毒载量为 361.000 拷贝/ml。全身 CT 扫描显示轻度脾肿大以及肠系膜和腹膜后淋巴结肿大。细针抽吸显示存在抗酸杆菌,但分枝杆菌培养为阴性。在第二份样本中,16S RNA 测序诊断为分枝杆菌属感染。尽管使用了 2 年的皮质类固醇和抗分枝杆菌药物(由于严重的皮肤反应排除了利福平),但没有临床改善,肠系膜淋巴结大小增加,并出现肠系膜硬化性转化。提出手术方法以释放小肠环并清除纤维蛋白。由于急性回肠节段性缺血和穿孔,需要进行第二次手术。最后,患者病情好转,抗分枝杆菌药物停药后无复发。

教训

尽管采用了长期的多药策略,但一些患者仍会出现持续性分枝杆菌属感染。一旦发生硬化性肠系膜炎,临床医生的治疗选择就很少了。对于有硬化性肠系膜炎或肠梗阻的患者,应考虑手术。医学和手术治疗的联合可能是这些患者潜在的治愈方法。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/48ca/10980495/bdc807fb7608/medi-101-e30351-g001.jpg

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