Case report: Multicentric Castleman disease as a manifestation of immune reconstitution inflammatory syndrome in Malawi.

Case report: Multicentric Castleman disease as a manifestation of immune reconstitution inflammatory syndrome in Malawi.
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病例报告:多中心卡斯尔曼病是马拉维免疫重建炎症综合征的一种表现。

DOI:
10.3389/fonc.2022.969135
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发表时间:
2022
影响因子:
4.7
通讯作者:
--
中科院分区:
医学3区
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--
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多中心Castleman病(MCD)是一种以全身炎症、淋巴结病和血细胞减少为特征的淋巴组织增生性疾病。由卡波西肉瘤疱疹病毒(MCD-KSHV)引起的MCD经常在HIV背景下出现。它可能与免疫重建炎症综合征(IRIS)有关,但MCD-IRIS在艾滋病毒和KSHV感染常见的撒哈拉以南非洲(SSA)很少报告。马拉维一名36岁女性艾滋病毒感染者接受抗逆转录病毒治疗(ART)9年,表现为疲劳、体重减轻和淋巴结病。淋巴结活检与HIV淋巴结炎一致,无明显KSHV-MCD,HIV RNA为4,244拷贝/mL。她转而接受二线抗逆转录病毒治疗,四个月后又回来了,淋巴结病恶化,发烧,盗汗,体重减轻和贫血。重复淋巴结活检证实了原始活检中不存在的明确KSHV-MCD特征。她的重复HIV病毒载量检测不到,她接受了化疗,随后在持续ART治疗24个月后缓解。这是来自撒南非洲的第一批报告的MCD-IRIS病例之一,这对艾滋病毒和KSHV高度流行的地区产生了影响。MCD-IRIS可能导致SSA患者开始ART治疗后的早期死亡率,需要提高认识,同时提高诊断和治疗能力。
Multicentric Castleman disease (MCD) is a lymphoproliferative disorder characterized by systemic inflammation, lymphadenopathy, and cytopenias. MCD caused by Kaposi sarcoma herpesvirus (MCD-KSHV) frequently arises in the context of HIV. It can be associated with immune reconstitution inflammatory syndrome (IRIS), but MCD-IRIS is rarely reported in sub-Saharan Africa (SSA) where HIV and KSHV infection are common. A 36-year-old woman in Malawi with HIV on antiretroviral therapy (ART) for nine years presented with fatigue, weight loss, and lymphadenopathy. Lymph node biopsy was consistent with HIV lymphadenitis without evident KSHV-MCD and HIV RNA was 4,244 copies/mL. She switched to second-line ART and returned four months later with worsening lymphadenopathy, fever, night sweats, weight loss, and anemia. A repeat lymph node biopsy demonstrated unequivocal KSHV-MCD features not present on the original biopsy. Her repeat HIV viral load was undetectable and she received chemotherapy with subsequent remission on continued ART for 24 months. This is among the first reported cases of MCD-IRIS from SSA, which has implications for a region where HIV and KSHV are highly prevalent. MCD-IRIS may contribute to early mortality after ART initiation in SSA, and increased awareness alongside improved diagnostic and treatment capacity are needed.
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