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

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Veröffentlicht/Copyright: 23. Juni 2021
Journal of Pediatric Endocrinology and Metabolism
Aus der Zeitschrift Journal of Pediatric Endocrinology and Metabolism Band 34 Heft 9

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.

Keywords: bone mineral density; growth hormone treatment; hypogonadism; Prader–Willi syndrome
Corresponding author: Yuji Oto, Department of Pediatrics, Dokkyo Medical University Saitama Medical Center, 2-1-50 Minamikoshigaya, Koshigaya City, Saitama 343-8555, Japan, Phone: +81 48 965 1111, Fax: +81 48 965 8363, E-mail:

Funding source: JSPS KAKENHI

Award Identifier / Grant number: 19K17375

Acknowledgments

We are grateful to the PWS patients and their families for their willingness to participate in this study.

  1. Research funding: This work was supported by JSPS KAKENHI Grant Number 19K17375.

  2. Author contributions: All the authors have accepted responsibility for the entire content of this submitted manuscript and approved submission.

  3. Competing interests: The funding organization(s) played no role in the study design; in the collection, analysis, and interpretation of data; in the writing of the report; or in the decision to submit the report for publication.

  4. Informed consent: Written informed consent was obtained from the parents of all patients.

  5. Ethical approval: This study was approved by the Ethics Board of Dokkyo Medical University Saitama Medical Center (No. 2007) and was conducted according to the Declaration of Helsinki.

References

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Received: 2021-01-25
Accepted: 2021-04-27
Published Online: 2021-06-23
Published in Print: 2021-09-27

© 2021 Walter de Gruyter GmbH, Berlin/Boston

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  2. Review Article
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  4. Original Articles
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  16. Short Communication
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  18. Case Reports
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https://doi.org/10.1515/jpem-2021-0061
Schlagwörter für diesen Artikel
bone mineral density; growth hormone treatment; hypogonadism; Prader–Willi syndrome

Artikel in diesem Heft

  1. Frontmatter
  2. Review Article
  3. Effects of aerobic exercise on obese children with metabolic syndrome: a systematic review and meta-analysis
  4. Original Articles
  5. Evaluation of clinical, endocrine and metabolic findings in obese children with and without hepatosteatosis
  6. Anxiety, depression, and glycemic control during Covid-19 pandemic in youths with type 1 diabetes
  7. Monogenic diabetes in Pakistani infants and children: challenges in a resource poor country
  8. Hereditary hypophosphatemic rickets and craniosynostosis
  9. Acromesomelic dysplasia-Maroteaux type, nine patients with two novel NPR2 variants
  10. The presentation of congenital adrenal hyperplasia in an unscreened population
  11. Presenting status of children with classical congenital adrenal hyperplasia over two decades (1999–2018) in the absence of newborn screening in Sri Lanka
  12. Diagnostic re-evaluation and predictors of congenital hypothyroidism with eutopic thyroid gland in Jiangxi, China
  13. Neonatal maple syrup urine disease in China: two novel mutations in the BCKDHB gene and literature review
  14. The treatment and clinical follow-up outcome in Iranian patients with tetrahydrobiopterin deficiency
  15. Retrospective evaluation of patients with X-linked adrenoleukodystrophy with a wide range of clinical presentations: a single center experience
  16. Short Communication
  17. Growth hormone treatment and bone mineral density in pediatric patients with Prader–Willi syndrome
  18. Case Reports
  19. SLC35A2-CDG: novel variants with two ends of the spectrum
  20. Genome analyses and androgen quantification for an infant with 5α-reductase type 2 deficiency
  21. Neurological and gastrointestinal symptoms as an initial presentation of pediatric thyroid storm: report of three cases
  22. Rapid molecular diagnosis of ALB gene variants prevents unnecessary interventions in familial dysalbuminemic hyperthyroxinemia
  23. MAN1B1-CDG: novel patients and novel variant
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