Stevenson R E, Arena J F, Ouzts E, Gibson A, Shokeir M H, Vnencak-Jones C, Lubs H A, May M, Schwartz C E
Greenwood Genetic Center, Greenwood, SC 29646, USA.
Am J Hum Genet. 1998 May;62(5):1092-101. doi: 10.1086/301835.
Mutations in genes on the X chromosome are believed to be responsible for the excess of males among individuals with mental retardation. Such genes are numerous, certainly >100, and cause both syndromal and nonsyndromal types of mental retardation. Clinical and molecular studies have been conducted on the Mennonite family with X-linked mental retardation (XLMR) reported, in 1962, by Renpenning et al. The clinical phenotype includes severe mental retardation, microcephaly, up-slanting palpebral fissures, small testes, and stature shorter than that of nonaffected males. Major malformations, neuromuscular abnormalities, and behavioral disturbances were not seen. Longevity is not impaired. Carrier females do not show heterozygote manifestations. The syndrome maps to Xp11.2-p11.4, with a maximum LOD score of 3.21 (recombination fraction 0) for markers between DXS1039 and DXS1068. Renpenning syndrome (also known as "MRXS8"; gene RENS1, MIM 309500) shares phenotypic manifestations with several other XLMR syndromes, notably the Sutherland-Haan syndrome. In none of these entities has the responsible gene been isolated; hence, the possibility that two or more of them may be allelic cannot be excluded at present.
X染色体上的基因突变被认为是导致智力发育迟缓个体中男性过多的原因。这类基因数量众多,肯定超过100个,可导致综合征型和非综合征型智力发育迟缓。1962年,Renpenning等人报道了一个患有X连锁智力发育迟缓(XLMR)的门诺派家族,并对其进行了临床和分子研究。临床表型包括严重智力发育迟缓、小头畸形、睑裂向上倾斜、小睾丸以及身材比未受影响的男性矮。未发现重大畸形、神经肌肉异常和行为障碍。寿命不受影响。携带者女性不表现出杂合子表现。该综合征定位于Xp11.2 - p11.4,DXS1039和DXS1068之间的标记物的最大LOD评分为3.21(重组率为0)。Renpenning综合征(也称为“MRXS8”;基因RENS1,MIM 309500)与其他几种XLMR综合征有共同的表型表现,特别是Sutherland - Haan综合征。在这些疾病中,尚未分离出致病基因;因此,目前不能排除其中两种或更多种疾病可能是等位基因的可能性。