Drenth J P, Mariman E C, Van der Velde-Visser S D, Ropers H H, Van der Meer J W
Department of Medicine, University Hospital St Radboud, Nijmegen, The Netherlands.
Hum Genet. 1994 Dec;94(6):616-20. doi: 10.1007/BF00206953.
The hyperimmunoglobulinemia D and periodic fever (hyper-IgD) syndrome is typified by recurrent febrile attacks with abdominal distress, joint involvement (arthralgias/arthritis), headache, skin lesions, and an elevated serum IgD level (> 100 U/ml). This familial disorder has been diagnosed in 56 subjects worldwide. As the hyper-IgD syndrome resembles familial Mediterranean fever, one could speculate that both result from mutations in the same gene. The gene causing familial Mediterranean fever (MEF) has been located on chromosome 16p. We have studied 10 families with 19 affected and 28 non-affected subjects. The clinical findings and IgD determinations from these families are compatible with autosomal recessive inheritance. Using highly polymorphic markers surrounding the MEF gene, only negative Lod scores were obtained, whereas haplotype analysis excluded this locus as the cause of the hyper-IgD syndrome. In addition, no indication for linkage was obtained with markers from other candidate gene regions on chromosomes 17q and 14q.
高免疫球蛋白D血症和周期性发热(高IgD)综合征的特点是反复发热发作,并伴有腹部不适、关节受累(关节痛/关节炎)、头痛、皮肤病变以及血清IgD水平升高(>100 U/ml)。这种家族性疾病在全球56名受试者中得到诊断。由于高IgD综合征类似于家族性地中海热,人们可能推测两者都是由同一基因的突变引起的。导致家族性地中海热(MEF)的基因已定位在16号染色体短臂上。我们研究了10个家庭,其中有19名患者和28名未受影响的受试者。这些家庭的临床发现和IgD测定结果与常染色体隐性遗传相符。使用MEF基因周围的高度多态性标记,仅获得了负的连锁值,而单倍型分析排除了该基因座是高IgD综合征的病因。此外,未获得与17号染色体长臂和14号染色体长臂上其他候选基因区域的标记的连锁指示。