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托吡酯治疗一名患有先天性肾上腺增生症和偏头痛的严重肥胖青少年期间的体重减轻

Weight Loss During Topiramate Treatment in a Severely Obese Adolescent with Congenital Adrenal Hyperplasia and Migraine.

作者信息

Seagroves Amy, Ross Heather M., Vidmar Alaina P., Geffner Mitchell E., Kim William S., Hwang Darryl, Borzutzky Claudia, Fraga Nicole R., Kim Mimi S.

机构信息

Children’s Hospital Los Angeles, Center for Endocrinology, Diabetes and Metabolism, California, USA

University of Southern California, Keck School of Medicine, Department of Pediatrics, California, USA

出版信息

J Clin Res Pediatr Endocrinol. 2023 Feb 27;15(1):81-85. doi: 10.4274/jcrpe.galenos.2021.2020.0310. Epub 2021 Aug 23.

Abstract

Youth with classical congenital adrenal hyperplasia (CAH) due to 21-hydroxylase deficiency exhibit an increased prevalence of obesity, early adiposity rebound, and increased abdominal adiposity compared to unaffected youth. Current obesity management in CAH largely focuses on lifestyle modifications. There is evidence that topiramate therapy is effective in reducing body mass index (BMI), as well as visceral adipose tissue (VAT), in unaffected adolescents with exogenous obesity. However, little is known about the efficacy of topiramate in patients with classical CAH. We report on a 17-year-old female with severe obesity and salt-wasting CAH due to 21-hydroxylase deficiency, who demonstrated reductions in BMI, as well as abdominal visceral and subcutaneous adipose tissue (SAT) while on topiramate therapy. The patient was diagnosed with classical CAH as a newborn with a 17-hydroxyprogesterone 11,000 ng/dL. She had a BMI over the 95 percentile at 3 years of age, followed by unremitting obesity. At 17 years old, she was started on topiramate to treat chronic migraines. Following three years of topiramate therapy, her BMI z-score decreased from +2.6 to +2.1. After four years of therapy, her waist circumference decreased from 110 to 101 cm, abdominal VAT decreased substantially by 34.2%, and abdominal SAT decreased by 25.6%. Topiramate therapy was associated with effective weight loss and reduced central adiposity in an adolescent with classical CAH and severe obesity, without any side effects. Further study is warranted regarding topiramate therapy in obese youth with classical CAH and increased central adiposity, who are at higher risk for significant morbidity.

摘要

与未受影响的青少年相比,因21-羟化酶缺乏导致经典型先天性肾上腺皮质增生症(CAH)的青少年肥胖患病率增加、早期肥胖反弹及腹部肥胖增加。目前CAH的肥胖管理主要侧重于生活方式的改变。有证据表明,托吡酯治疗对外源性肥胖的未受影响青少年降低体重指数(BMI)以及内脏脂肪组织(VAT)有效。然而,关于托吡酯在经典型CAH患者中的疗效知之甚少。我们报告了一名17岁女性,因21-羟化酶缺乏患有严重肥胖和失盐型CAH,在接受托吡酯治疗期间BMI、腹部内脏和皮下脂肪组织(SAT)均有所降低。该患者新生儿期因17-羟孕酮为11,000 ng/dL被诊断为经典型CAH。她3岁时BMI超过第95百分位数,随后肥胖持续存在。17岁时,她开始服用托吡酯治疗慢性偏头痛。经过三年的托吡酯治疗,她的BMI z评分从+2.6降至+2.1。经过四年的治疗,她的腰围从110厘米降至101厘米,腹部VAT大幅下降34.2%,腹部SAT下降25.6%。托吡酯治疗使一名患有经典型CAH和严重肥胖的青少年有效减重并减少了中心性肥胖,且无任何副作用。对于患有经典型CAH且中心性肥胖增加、有更高严重发病风险的肥胖青少年,有必要进一步研究托吡酯治疗。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6ff4/9976166/0817f042c6bc/JCRPE-15-81-g1.jpg

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