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肾移植后早期皮肤杆菌性血管瘤病:病例报告及文献复习。

Early post-transplant cutaneous bacillary angiomatosis in a kidney recipient: Case report and review of the literature.

机构信息

Department of Infectious Diseases, Cleveland Clinic, Cleveland, OH, USA.

Transplantation Center, Cleveland Clinic, Cleveland, OH, USA.

出版信息

Transpl Infect Dis. 2021 Aug;23(4):e13670. doi: 10.1111/tid.13670. Epub 2021 Jul 8.

Abstract

Bacillary angiomatosis (BA) is an uncommon systemic disease caused by Bartonella henselae (BH) or Bartonella quintana (BQ) that occurs primarily in immunocompromised hosts. Few cases of BA recipients have been reported in adult solid transplant recipients over the years, with most cases presenting years after transplant. We describe a case of a kidney transplant recipient who developed cutaneous BA very early in the post-transplant period despite not having any exposures. Retrospective testing of donor and recipient's serum was performed and raised the concern for possible donor-derived infection. A literature review encompassing 1990 to present was also performed in order to better understand the clinical presentation, diagnostics and therapeutic approach of this unusual disease. Combined serology, histopathology and molecular testing (polymerase chain reaction [PCR]) were useful in diagnosing BA in our patient as serology alone might be unreliable. Macrolides or doxycycline for at least 3 months is the recommended therapeutic strategy; however, the optimal duration of treatment is not well established in transplant recipients. In our patient, we decided to use doxycycline for 1 year due to gradual resolution of lesions and ongoing immunosuppression. Patient responded successfully without any documented relapse.

摘要

杆菌性血管瘤病 (BA) 是一种由亨氏巴尔通体 (BH) 或五日热巴尔通体 (BQ) 引起的少见系统性疾病,主要发生于免疫功能低下的宿主。多年来,成人实体器官移植受者中仅报道了少数 BA 受者病例,大多数病例在移植后数年出现。我们描述了一例肾移植受者,尽管没有任何暴露,但在移植后早期即发生皮肤 BA。对供者和受者血清进行了回顾性检测,这引起了对可能供者源性感染的关注。还进行了一项涵盖 1990 年至今的文献复习,以便更好地了解这种不常见疾病的临床表现、诊断和治疗方法。联合血清学、组织病理学和分子检测(聚合酶链反应 [PCR])有助于诊断我们患者的 BA,因为仅血清学可能不可靠。至少 3 个月的大环内酯类或多西环素是推荐的治疗策略;然而,在移植受者中,治疗的最佳持续时间尚未确定。在我们的患者中,由于病变逐渐消退和持续的免疫抑制,我们决定使用多西环素治疗 1 年。患者成功应答,无任何记录的复发。

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