Growth hormone treatment and bone mineral density in pediatric patients with Prader–Willi syndrome

Author:

Oto Yuji1,Murakami Nobuyuki1,Inoue Takeshi1,Matsubara Keiko2,Saima Sohei3,Ogata Hiroyuki3,Ihara Hiroshi3,Nagai Toshiro4,Matsubara Tomoyo1

Affiliation:

1. Department of Pediatrics , Dokkyo Medical University Saitama Medical Center , Saitama , Japan

2. Department of Molecular Endocrinology , National Research Institute for Child Health and Development , Tokyo , Japan

3. Department of Psychiatry , Dokkyo Medical University Saitama Medical Center , Saitama , Japan

4. Nakagawanosato Ryoiku Center , Saitama , Japan

Abstract

Abstract Objectives Previous reports indicate that growth hormone (GH) treatment for Prader–Willi syndrome (PWS) improves bone mineral density (BMD) only when initiated at a young age and not when initiated in adulthood. However, there are no data on BMD during long-term GH treatment of Japanese children and adolescents with PWS. Thus, this study aimed to investigate BMD changes among patients with PWS, who were undergoing GH treatment from childhood to adolescence. Methods Sixty-seven pediatric patients with PWS who had GH treatment initiated during childhood between January 2003 and June 2020 were evaluated. To avoid underestimation, we used total body BMD, which was evaluated using dual-X-ray absorptiometry adjusted for the BMD z-score using patient height, sex, and age. Results In both sexes, age was negatively correlated with the BMD-standard deviation score (SDS) (male: r=−0.156 [p=0.042]; female: r=−0.197 [p=0.043]), which started to decrease in childhood. Conclusions The BMD-SDS of patients with PWS decreases gradually despite GH treatment. As there are no clear recommendations about monitoring of bone health in patients with PWS, further studies are needed to improve the guidelines for screening of BMD and treatment of patients with PWS.

Publisher

Walter de Gruyter GmbH

Subject

Endocrinology,Endocrinology, Diabetes and Metabolism,Pediatrics, Perinatology, and Child Health

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