Banerjee Indraneel, Trueman Julie A, Hall Catherine M, Price David A, Patel Leena, Whatmore Andrew J, Hirschhorn Joel N, Read Andrew P, Palmert Mark R, Clayton Peter E
Department of Paediatric Endocrinology, Royal Manchester Children's Hospital, UK.
Eur J Endocrinol. 2006 Jul;155(1):121-6. doi: 10.1530/eje.1.02184.
Constitutional delay of growth and puberty (CDGP) is a variant of normal pubertal timing and progress, often with dominant inheritance. It is likely that one or more genes will be associated with CDGP. Possible candidates are the leptin (L) and the leptin receptor (LR) genes, as the leptin axis links nutritional status to pubertal development. This study has assessed whether a) L or LR gene polymorphisms were associated with CDGP and b) the CDGP phenotype was influenced by these polymorphisms.
Case-control and transmission disequilibrium tests were used to test genetic association of L and LR polymorphisms with CDGP.
We genotyped L (3'CTTT repeat) and LR polymorphisms (Gln > Arg substitution, exon 6) in 81 CDGP children and 94 controls in the UK and 88 CDGP children from the US and assessed the effect of genotype on their anthropometric characteristics.
There was no association of these L or LR gene polymorphisms with CDGP. There was no difference in height or bone age delay within L or LR genotypes. However, UK CDGP children homozygous for the L short allele were heavier than heterozygotes and long allele homozygotes, with a similar trend in the US cohort. UK CDGP children with severe pubertal delay, who were thin, had significantly greater bone age delay and an increased frequency of parental pubertal delay than other groups and were less likely to be L short allele homozygotes.
There was no association of specific L or LR polymorphisms with CDGP, but L short allele carriage influenced the phenotype within CDGP.
体质性生长和青春期延迟(CDGP)是正常青春期时间和进程的一种变异,通常具有显性遗传。很可能有一个或多个基因与CDGP相关。可能的候选基因是瘦素(L)和瘦素受体(LR)基因,因为瘦素轴将营养状况与青春期发育联系起来。本研究评估了:a)L或LR基因多态性是否与CDGP相关;b)这些多态性是否影响CDGP表型。
采用病例对照和传递不平衡检验来检测L和LR多态性与CDGP的遗传关联性。
我们对英国的81名CDGP儿童和94名对照以及美国的88名CDGP儿童进行了L(3'CTTT重复序列)和LR多态性(第6外显子谷氨酰胺>精氨酸替代)基因分型,并评估了基因型对其人体测量特征的影响。
这些L或LR基因多态性与CDGP无关。L或LR基因型内的身高或骨龄延迟没有差异。然而,L短等位基因纯合的英国CDGP儿童比杂合子和长等位基因纯合子更重,美国队列也有类似趋势。英国严重青春期延迟且体型瘦的CDGP儿童,其骨龄延迟明显更大,父母青春期延迟的频率更高,且比其他组更不可能是L短等位基因纯合子。
特定的L或LR多态性与CDGP无关,但L短等位基因携带影响了CDGP内的表型。