Kianifar Hamid Reza, Khalesi Maryam, Farid Reza, Badiee Zahra, Rastin Maryam, Ahanchian Hamid
Department of Pediatric Immunology, Ghaem Medical Center, Mashhad University of Medical Science, Mashhad, Iran.
Iran J Allergy Asthma Immunol. 2010 Sep;9(3):181-63.
Autoimmune lymphoproliferative syndrome (ALPS) is an uncommon nonmalignant lymphoproliferative disease which is characterized by chronic, persistent or recurrent lymphadenopathy, splenomegaly, hepatomegaly, immune cytopenia , hypergammaglobinemia and increased risk of lymphoma. We report a 2-year old boy with hepatosplenomegaly as first presentation. Petechial and purpuric rashes with massive cervical lymphadenopathies developed 10 months later.In laboratory tests anemia, thrombocytopenia and hypergammaglobinemia were observed. According to flocytometry increased double negative T cells and by apoptosis assay decrease apoptosis of lymphocytes accompanied clinical manifestations, thus diagnosis of ALPS was established. In conclusion; in all patients with massive lymphadenopathy and hepatosplenomegay; especially with cytopenia; ALPS should be considered.
自身免疫性淋巴细胞增生综合征(ALPS)是一种罕见的非恶性淋巴细胞增生性疾病,其特征为慢性、持续性或复发性淋巴结病、脾肿大、肝肿大、免疫性血细胞减少、高球蛋白血症以及淋巴瘤风险增加。我们报告一名2岁男孩,首发表现为肝脾肿大。10个月后出现瘀点和紫癜性皮疹以及大量颈部淋巴结病。实验室检查发现贫血、血小板减少和高球蛋白血症。根据流式细胞术检测,双阴性T细胞增加,通过凋亡分析发现淋巴细胞凋亡减少,这些与临床表现相符,从而确诊为ALPS。总之,对于所有有大量淋巴结病和肝脾肿大的患者,尤其是伴有血细胞减少的患者,应考虑ALPS。