Department of Pediatrics, Osaka University Graduate School of Medicine, Osaka 565-0871, Japan.
Faculty of Health and Nutrition, Osaka Shoin Women's University, Osaka 577-8550, Japan.
Endocr J. 2023 Apr 28;70(4):435-443. doi: 10.1507/endocrj.EJ22-0477. Epub 2023 Feb 4.
Achondroplasia is a rare skeletal dysplasia characterized by rhizomelic short stature, whose prevalence is about 1 per 25,000 births. For some patients with achondroplasia, excess body weight is one of the major concerns due to an impaired linear growth. Epidemiological studies revealed a premature onset of cardiovascular or cerebrovascular events in achondroplasia. An association between obesity and cardiometabolic risk factors related to cardiovascular events remains unknown in patients with achondroplasia/hypochondroplasia. This cross-sectional study investigated anthropometric measurements, body compositions and cardiometabolic risk factors in pediatric patients with achondroplasia/hypochondroplasia. Thirty-two patients with achondroplasia and ten with hypochondroplasia aged between 1.9 and 18.7 years were enrolled in this study. Half of the participants presented at least one cardiometabolic abnormality. Elevated systolic blood pressure was the most common abnormality. None of the participants developed metabolic syndrome or type 2 diabetes mellitus. Body mass index-standard deviation score and hip/height ratio were strongly correlated with percent body fat assessed by dual energy X-ray absorptiometry although no significant association was found between anthropometric measurements or body fat mass and any cardiometabolic risk factors. No significant difference in body fat mass, as well as body mass index-standard deviation score and hip/height, was found between cardiometabolically normal group and cardiometabolically abnormal groups. These results suggest that not only weight gain and hip/height changes should be monitored but also individual cardiometabolic risk factors should be evaluated to avoid cardiometabolic events in the healthcare management of pediatric patients with achondroplasia/hypochondroplasia.
软骨发育不全症是一种罕见的骨骼发育不良,其特征为肢端短小,发病率约为每 25000 例出生一例。对于一些软骨发育不全症患者,由于线性生长受损,超重是主要关注点之一。流行病学研究显示,软骨发育不全症患者心血管或脑血管事件的发病年龄提前。肥胖与与心血管事件相关的心血管代谢危险因素之间的关联在软骨发育不全症/软骨营养障碍症患者中尚不清楚。本横断面研究调查了软骨发育不全症/软骨营养障碍症儿科患者的人体测量学测量、身体成分和心血管代谢危险因素。本研究纳入了 32 名软骨发育不全症患者和 10 名软骨营养障碍症患者,年龄在 1.9 至 18.7 岁之间。一半的参与者至少存在一项心血管代谢异常。收缩压升高是最常见的异常。没有参与者发生代谢综合征或 2 型糖尿病。体重指数标准差评分和髋高比与双能 X 线吸收法评估的体脂百分比呈强相关,但人体测量学测量或体脂质量与任何心血管代谢危险因素之间均无显著相关性。在心血管代谢正常组和心血管代谢异常组之间,体脂质量以及体重指数标准差评分和髋高比没有显著差异。这些结果表明,不仅应监测体重增加和髋高比的变化,还应评估个体的心血管代谢危险因素,以避免在软骨发育不全症/软骨营养障碍症儿科患者的医疗保健管理中发生心血管代谢事件。