Absence of peripheral blood chimerism in graft-vs-host disease following orthotopic liver transplantation: case report and review of the literature.

Absence of peripheral blood chimerism in graft-vs-host disease following orthotopic liver transplantation: case report and review of the literature.
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DOI:
10.1111/ijd.12149
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发表时间:
2014-11
影响因子:
3.6
通讯作者:
Shinkai K
Shinkai K
中科院分区:
医学4区
文献类型:
--
作者:
Schulman JM;Yoon C;Schwarz J;Vagefi PA;Mully TW;Shinkai K

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移植物抗宿主病(GVHD)是原位肝移植(OLT)中一种罕见且经常致命的并发症。皮肤通常在疾病进展的早期受累,但临床和组织病理学特征可能是非特异性的,这给诊断带来了挑战。虽然外周血嵌合检测已被提出作为olt后GVHD的诊断标准,但目前尚不清楚外周血嵌合是否为诊断的绝对要求。我们报告一例57岁的男性,他发展为olt后GVHD,并累及皮肤、肠和骨髓。我们也综述了肝移植后GVHD的流行病学、发病机制、临床表现、组织病理学、分子诊断技术和治疗。在我们的病人中,用短串联重复聚合酶链反应分析外周血没有检测到循环供体淋巴细胞。然而,在口腔黏膜中检测到供体淋巴细胞,证实了诊断。对先前发表的63例olt后GVHD病例的嵌合模式的回顾表明,这是首次报道的外周血中不存在嵌合但在其他部位存在嵌合的病例。olt后GVHD患者可能没有外周血嵌合现象。因此,需要结合临床、组织病理学和分子特征来做出这一具有挑战性的诊断。
Graft-vs-host disease (GVHD) is a rare and often fatal complication of orthotopic liver transplantation (OLT). The skin is frequently involved early in disease progression, but clinical and histopathological features may be nonspecific, presenting a diagnostic challenge. While the detection of peripheral blood chimerism has been proposed as a diagnostic criterion for post-OLT GVHD, it is not known whether peripheral blood chimerism is an absolute requirement for the diagnosis. We report a case of a 57-year-old man who developed post-OLT GVHD with cutaneous, enteric, and bone marrow involvement. We also review the epidemiology, pathogenesis, clinical presentation, histopathology, molecular diagnostic techniques, and treatment of GVHD following liver transplantation. In our patient, analysis of the peripheral blood by short-tandem repeat polymerase chain reaction did not detect circulating donor lymphocytes. Donor lymphocytes were detected in the buccal mucosa, however, confirming the diagnosis. A review of chimerism patterns in 63 previously published cases of post-OLT GVHD reveals that this is the first reported case in which chimerism was absent in the peripheral blood but present in another site. Peripheral blood chimerism may be absent in cases of post-OLT GVHD. A combination of clinical, histopathological, and molecular features is therefore required to make this challenging diagnosis.
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