Wali A, Ali G, John P, Lee K, Chishti M S, Leal S M, Ahmad W
Department of Biochemistry, Faculty of Biological Sciences, Quaid-i-Azam University, Islamabad, Pakistan.
Ann Hum Genet. 2007 Sep;71(Pt 5):570-7. doi: 10.1111/j.1469-1809.2007.00362.x. Epub 2007 Apr 19.
Alopecia with mental retardation syndrome (APMR) is a rare autosomal recessive disorder characterized by total or partial absence of hair from the scalp and other parts of the body and associated with mental retardation. Previously, we have reported the mapping of two alopecia and mental retardation genes (APMR1 and APMR2) on human chromosome 3. In the present study, after excluding both of these loci through linkage analysis, a whole genome scan was performed by genotyping 396 polymorphic microsatellite markers located on 22 autosomes and the X and Y chromosomes. A disease locus was mapped to a 10.9 cM region, flanked by markers D18S866 and D18S811, on chromosome 18q11.2-q12.2. A maximum two-point LOD score of 3.03 at theta= 0.0 was obtained with marker D18S1102. Multipoint linkage analysis resulted in maximum LOD scores of 4.03 with several markers in the candidate region. According to the Rutgers combined linkage-physical map of the human genome (build 36) this region covers 12.17 Mb. DNA sequence analysis of nine candidate genes including DSC3, DSC1, DSG1, DSG4, DSG3, ZNF397, ZNF271, ZNF24 and ZNF396 did not reveal any sequence variants in the affected individuals of the family presented here.
脱发伴智力发育迟缓综合征(APMR)是一种罕见的常染色体隐性疾病,其特征为头皮和身体其他部位全部或部分无毛,并伴有智力发育迟缓。此前,我们已报道了人类3号染色体上两个脱发和智力发育迟缓基因(APMR1和APMR2)的定位。在本研究中,通过连锁分析排除这两个基因座后,利用位于22条常染色体以及X和Y染色体上的396个多态微卫星标记进行了全基因组扫描。一个疾病基因座被定位到18号染色体18q11.2 - q12.2上,位于标记D18S866和D18S811之间的一个10.9 cM区域。标记D18S1102在θ = 0.0时获得了最大两点LOD分数3.03。多点连锁分析在候选区域的几个标记处得到了最大LOD分数4.03。根据人类基因组的罗格斯联合连锁 - 物理图谱(构建版本36),该区域覆盖12.17 Mb。对包括DSC3、DSC1、DSG1、DSG4、DSG3、ZNF397、ZNF271、ZNF24和ZNF396在内的9个候选基因进行DNA序列分析,未在此处呈现的该家族受影响个体中发现任何序列变异。