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FGFR3相关的低磷酸酯酶症:57例携带p.Asn540Lys突变儿童的纵向生长情况

FGFR3-related hypochondroplasia: longitudinal growth in 57 children with the p.Asn540Lys mutation.

作者信息

Arenas María Alejandra, Del Pino Mariana, Fano Virginia

机构信息

Department of Growth and Development, Garrahan Hospital, Combate de los Pozos 1881 (1245), Buenos Aires, Argentina, Phone: 0054 11 4122 6221, Fax: 0054 11 43085325.

Department of Growth and Development, Garrahan Hospital, Buenos Aires, Argentina.

出版信息

J Pediatr Endocrinol Metab. 2018 Nov 27;31(11):1279-1284. doi: 10.1515/jpem-2018-0046.

Abstract

Background Children with hypochondroplasia (HCH), who have FGFR3 mutations c.1620C>A or c.1620C>G (p.Asn540Lys) appear to have a more severe phenotype than those with HCH without these mutations. We describe the change in height, leg length and body proportions in a retrospective cohort of children with HCH related-p.Asn540Lys mutation and we compared them with Argentine population. Methods Anthropometric measurements were initially taken and followed up by the same observer, with standardized techniques. Sitting height/height and head circumference/height ratio were calculated as a body disproportion indicator. In order to make a comparison with the Argentine population height average, centiles of height, leg length and body proportions were estimated by the LMS method. Results The sample consisted of 57 HCH children (29 males and 28 females) between the ages of 0-18 years. The median (interquartile range) number of measurements per child was 8 (4.3, 13) for height, 7 (4, 12) for sitting height and 7.5 (4, 12.8) for head circumference. Leg length increased from 17 cm at birth to approximately 54 cm in adolescents, 25 cm shorter than the leg length in non-HCH populations. Sitting height increased from 39 cm at birth to 81 cm in adolescents, 7 cm below mean in non-HCH adolescents. Mean (range) adult height were 143.6 cm (131-154.5) and 130.8 cm (124-138) for males and females, respectively. Conclusions The disharmonic growth between the less affected trunk and the severely affected limbs determine body disproportion in HCH.

摘要

背景

患有软骨发育不全(HCH)且携带FGFR3基因c.1620C>A或c.1620C>G(p.Asn540Lys)突变的儿童,其表型似乎比未携带这些突变的HCH儿童更为严重。我们描述了携带p.Asn540Lys突变的HCH儿童回顾性队列中的身高、腿长和身体比例变化,并将其与阿根廷人群进行了比较。方法:由同一名观察者采用标准化技术最初进行人体测量,并进行随访。计算坐高/身高和头围/身高比值作为身体比例失调指标。为了与阿根廷人群的平均身高进行比较,采用LMS方法估计身高、腿长和身体比例的百分位数。结果:样本包括57名年龄在0至18岁之间的HCH儿童(29名男性和28名女性)。每个儿童身高测量的中位数(四分位间距)为8次(4.3,13),坐高测量为7次(4,12),头围测量为7.5次(4,12.8)。腿长从出生时的17厘米增加到青少年时期的约54厘米,比非HCH人群的腿长短25厘米。坐高从出生时的39厘米增加到青少年时期的81厘米,比非HCH青少年的平均水平低7厘米。男性和女性的成人平均身高(范围)分别为143.6厘米(131 - 154.5)和130.8厘米(124 - 138)。结论:受影响较小的躯干和受严重影响的四肢之间的不协调生长决定了HCH患者的身体比例失调。

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