Chiang Grace P K, Li C K, Lee Vincent, Cheng Frankie W T, Leung Alex W K, Imashuku Shinsaku, Imamura Toshihiko, Shing Matthew M K
Department of Paediatrics, Prince of Wales Hospital, The Chinese University of Hong Kong, Shatin, Hong Kong.
Division of Pediatrics and Hematology, Takasago-seibu Hospital, Takasago, Japan.
Hong Kong Med J. 2014 Aug;20(4):339-42. doi: 10.12809/hkmj134041.
Familial haemophagocytic lymphohistiocytosis is a rare but invariably fatal disease without haematopoietic stem cell transplantation. Genetic defect identification is useful for confirming a clinical diagnosis, predicting the risk of future recurrence, and defining haemophagocytic lymphohistiocytosis predisposition in asymptomatic family members. Notably, familial haemophagocytic lymphohistiocytosis type 2 associates with mutations in the perforin gene (PRF1) which is the most frequent subtype of familial haemophagocytic lymphohistiocytosis. Although perforin gene mutations have been described in Asians, they are largely reported from Japan. The case reported here is the first familial haemophagocytic lymphohistiocytosis type 2 patient in Hong Kong with an identified perforin gene mutation.
家族性噬血细胞性淋巴组织细胞增生症是一种罕见但未经造血干细胞移植则必然致命的疾病。基因缺陷鉴定对于确诊临床诊断、预测未来复发风险以及确定无症状家庭成员的噬血细胞性淋巴组织细胞增生症易感性很有用。值得注意的是,2型家族性噬血细胞性淋巴组织细胞增生症与穿孔素基因(PRF1)突变相关,这是家族性噬血细胞性淋巴组织细胞增生症最常见的亚型。虽然亚洲人中有穿孔素基因突变的报道,但大多来自日本。本文报道的病例是香港首例确诊穿孔素基因突变的2型家族性噬血细胞性淋巴组织细胞增生症患者。