Standen G R, Lillicrap D P, Matthews N, Bloom A L
Q J Med. 1986 Apr;59(228):401-8.
A kindred with X-linked hereditary thrombocytopenia in association with elevated serum IgA and a mild nephropathy is described. Thirteen males with thrombocytopenia were identified in three generations amongst 49 family members who were available for screening. Serious infective sequelae were absent but five patients had suffered from severe eczema since infancy. The platelet volume as measured by an automated counter and electron microscopy was reduced compared with normal and in vitro tests demonstrated minor abnormalities of immune function in three patients. The disorder is identified as a novel variant of the Wiskott-Aldrich syndrome and comparisons are made with previously described kindreds showing different patterns of expression.
本文描述了一个与X连锁遗传性血小板减少症相关的家族,该家族伴有血清IgA升高和轻度肾病。在49名可供筛查的家庭成员中,三代内共鉴定出13名患有血小板减少症的男性。这些患者没有严重的感染后遗症,但有5名患者自婴儿期起就患有严重湿疹。通过自动计数器和电子显微镜测量的血小板体积与正常情况相比有所减小,体外试验表明3名患者的免疫功能存在轻微异常。该疾病被鉴定为维斯科特-奥尔德里奇综合征的一种新型变体,并与先前描述的表现出不同表达模式的家族进行了比较。