Friedman D F, Kwittken P, Cizman B, Argyris E, Kearns J, Yang S Y, Zmijewski C, Bunin N, Douglas S D, Monos D
Department of Pathology, University of Pennsylvania Medical Center, Philadelphia, USA.
Clin Diagn Lab Immunol. 1994 Sep;1(5):590-6. doi: 10.1128/cdli.1.5.590-596.1994.
Transfusion-associated graft-versus-host disease (TAGVHD) is a rare and usually fatal complication of blood transfusion which can arise when immunocompetent lymphocytes from the donor of a cellular blood product are transfused into a severely immunocompromised recipient. We describe the case of an 8-month-old male with a severe combined immunodeficiency syndrome who developed TAGVHD after receiving an unirradiated transfusion. Serologic HLA typing of the parents, the patient, and the blood donor demonstrated the foreign origin of circulating lymphocytes, confirming the diagnosis of TAGVHD. The manifestations of TAGVHD did not respond to medical immunosuppressive therapy, and bone marrow transplantation was planned to treat the underlying immunodeficiency as well as the TAGVHD. By using DNA-based class I and class II HLA typing, the child's HLA type was determined from nonhematopoietic tissues. This information proved critical in selecting the bone marrow donor. The child received immunosuppression, myeloablation, and a T-depleted, maternal bone marrow graft mismatched at one HLA class II allele. Trilineage hematopoietic engraftment occurred within 3 weeks, and the child remains clinically stable with no evidence of TAGVHD more than 2 years after the transplant. This case illustrates that TAGVHD can be successfully treated by allogeneic bone marrow transplantation and that DNA-based HLA typing can play a unique role in the diagnosis and management of TAGVHD.
输血相关移植物抗宿主病(TAGVHD)是一种罕见且通常致命的输血并发症,当来自细胞血液制品供体的免疫活性淋巴细胞被输注到严重免疫受损的受者体内时可能会发生。我们描述了一名患有严重联合免疫缺陷综合征的8个月大男性病例,该患儿在接受未经辐照的输血后发生了TAGVHD。对父母、患者和献血者进行的血清学HLA分型显示循环淋巴细胞来自异体,从而确诊了TAGVHD。TAGVHD的症状对药物免疫抑制治疗无反应,因此计划进行骨髓移植以治疗潜在的免疫缺陷以及TAGVHD。通过基于DNA的I类和II类HLA分型,从非造血组织中确定了患儿的HLA类型。这一信息在选择骨髓供体方面被证明至关重要。该患儿接受了免疫抑制、清髓治疗,并接受了一份在一个II类HLA等位基因上不匹配的、去除了T细胞的母亲骨髓移植。移植后3周内实现了三系造血植入,移植后2年多来患儿临床稳定,没有TAGVHD的迹象。该病例表明,异基因骨髓移植可以成功治疗TAGVHD,并且基于DNA的HLA分型在TAGVHD的诊断和管理中可以发挥独特作用。