Abstract
Background
Prader-Willi syndrome (PWS) is associated with marked obesity that can lead to severe complications such as diabetes mellitus. Early adiposity rebound (AR) is associated with future obesity and an increased risk of diabetes mellitus and metabolic syndrome. Previous reports have shown that the onset of AR occurred earlier in diseases that cause obesity. However, there have been no studies focusing on the timing of AR in PWS, or on the effect of growth hormone (GH) treatment on AR. The aim of this study was to explore AR in PWS patients and to analyze the effect of GH treatment on AR.
Methods
This retrospective study evaluated 48 patients, with 16 of the patients found to have AR prior to GH treatment. AR was constructed for each patient using Microsoft Excel, and the exact point of the nadir of body mass index (BMI) following the initial peak was determined. We additionally analyzed the relationship between GH treatment and the timing of AR onset.
Results
AR onset for patients found to have AR before starting GH treatment was 16.0 (13.0–21.0) months. In contrast, AR onset for patients found to have AR after starting GH treatment was 27.5 (23.8–36.3) months. The difference between the two groups was statistically significant (p=0.0001). A positive correlation was found between the GH treatment period and AR (p=0.00013).
Conclusion
The median age of AR onset in PWS patients was 16.0 (13.0–21.0) months, and GH treatment might delay the early AR onset.
Acknowledgments
We are grateful to the PWS patients and their families for their willingness to participate in this research. We would like to thank Forte (https://www.forte-science.co.jp/) for English language editing.
Author contributions: All the authors have accepted responsibility for the entire content of this submitted manuscript and approved submission.
Research funding: None declared.
Employment or leadership: None declared.
Honorarium: None declared.
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.
References
1. Cassidy SB, Schwartz S, Miller JL, Driscoll DJ. Prader-Willi syndrome. Genet Med 2012;14:10–26.10.1038/gim.0b013e31822bead0Suche in Google Scholar PubMed
2. Ma Y, Wu T, Liu Y, Wang Q, Song J, et al. Nutritional and metabolic findings in patients with Prader-Willi syndrome diagnosed in early infancy. J Pediatr Endocrinol Metab 2012;25:1103–9.10.1515/jpem-2012-0167Suche in Google Scholar PubMed
3. Koyama S, Ichikawa G, Kojima M, Shimura N, Sairenchi T, et al. Adiposity rebound and the development of metabolic syndrome. Pediatrics 2014;133:e114–9.10.1542/peds.2013-0966Suche in Google Scholar PubMed
4. Peneau S, Gonzalez-Carrascosa R, Gusto G, Goxe D, Lantieri O, et al. Age at adiposity rebound: determinants and association with nutritional status and the metabolic syndrome at adulthood. Int J Obes (Lond) 2016;40:1150–6.10.1038/ijo.2016.39Suche in Google Scholar PubMed
5. Arisaka O, Sairenchi T, Ichikawa G, Koyama S. Increase of body mass index (BMI) from 1.5 to 3 years of age augments the degree of insulin resistance corresponding to BMI at 12 years of age. J Pediatr Endocrinol Metab 2017;30:455–7.10.1515/jpem-2016-0227Suche in Google Scholar PubMed
6. Takishima S, Nakajima K, Nomura R, Tsuji-Hosokawa A, Matsuda N, et al. Lower body weight and BMI at birth were associated with early adiposity rebound in 21-hydroxylase deficiency patients. Endocr J 2016;63:983–90.10.1507/endocrj.EJ16-0194Suche in Google Scholar PubMed
7. Chen SY, Lin SJ, Lin SH, Chou YY. Early adiposity rebound and obesity in children with congenital hypothyroidism. Pediatr Neonatol 2013;54:107–12.10.1016/j.pedneo.2012.10.008Suche in Google Scholar PubMed
8. Drosty AR, Emmett PM, Cowin S, Reilly JJ. Factors associated with early adiposity rebound. Pediatrics 2000;105:1115–8.10.1542/peds.105.5.1115Suche in Google Scholar PubMed
9. Miller JL, Lynn CH, Driscoll DC, Goldstone AP, June-Anne Gold, et al. Nutritional phases in Prader-Willi syndrome. Am J Med Genet A 2011;155A:1040–9.10.1002/ajmg.a.33951Suche in Google Scholar PubMed PubMed Central
10. Coupaye M, Lorenzini F, Lloret-Linares C, Molinas C, Pinto G, et al. Growth hormone therapy for children and adolescents with Prader-Willi syndrome is associated with improved body composition and metabolic status in adulthood. J Clin Endocrinol Metab 2013;98:E328–35.10.1210/jc.2012-2881Suche in Google Scholar PubMed
11. Butler MG. Prader-Willi syndrome: obesity due to genomic imprinting. Curr Genomics 2011;12:204–15.10.2174/138920211795677877Suche in Google Scholar PubMed PubMed Central
12. Brambilla P, Crinò A, Bedogni G, Bosio L, Cappa M, et al. Metabolic syndrome in children with Prader-Willi syndrome: the effect of obesity. Nutr Metab Cardiovasc Dis 2011;21:269–76.10.1016/j.numecd.2009.10.004Suche in Google Scholar PubMed
13. Grugni G, Crino A, Bedogni G, Cappa M, Sartorio A, et al. Metabolic syndrome in adult patients with Prader-Willi syndrome. Nutr Metab Cardiovasc Dis 2013;23;11:1134–40.10.1016/j.numecd.2012.11.006Suche in Google Scholar PubMed
14. Fintini D, Grugni G, Bocchini S, Brufani C, Di Candia S, et al. Disorders of glucose metabolism in Prader-Willi syndrome: results of a multicenter Italian cohort study. Nutr Metab Cardiovasc Dis 2016;26;9:842–47.10.1016/j.numecd.2016.05.010Suche in Google Scholar PubMed
©2018 Walter de Gruyter GmbH, Berlin/Boston
Artikel in diesem Heft
- Frontmatter
- Original Articles
- Targeted next generation sequencing in patients with maturity-onset diabetes of the young (MODY)
- Assessment of dental caries and gingival status among a group of type 1 diabetes mellitus and healthy children of South India – a comparative study
- Early adiposity rebound in patients with Prader-Willi syndrome
- Arterial stiffness as a measure of cardiovascular risk in obese adolescents and adolescents with diabetes type 1
- Dysregulated glucose homeostasis in congenital central hypoventilation syndrome
- Do parents of children with metabolic diseases benefit from the Triple P – Positive Parenting Program? A pilot study
- Early higher dosage of alglucosidase alpha in classic Pompe disease
- Insulin resistance in children with familial hyperlipidemia
- Insulin resistance linked to subtle myocardial dysfunction in normotensive Turner syndrome young patients without structural heart diseases
- Acetylcholinesterase activity and bone biochemical markers in premature and full-term neonates
- Baseline characteristics of gender dysphoric youth
- Case Reports
- Multi-disciplinary evaluation of a 5-month-old with hypertrophic cardiomyopathy related to a functional adrenocortical tumor
- A case of an infant with extremely low birth weight and hypothyroidism associated with massive cutaneous infantile hemangioma
- Possible hints and pitfalls in diagnosing Peutz-Jeghers syndrome
Artikel in diesem Heft
- Frontmatter
- Original Articles
- Targeted next generation sequencing in patients with maturity-onset diabetes of the young (MODY)
- Assessment of dental caries and gingival status among a group of type 1 diabetes mellitus and healthy children of South India – a comparative study
- Early adiposity rebound in patients with Prader-Willi syndrome
- Arterial stiffness as a measure of cardiovascular risk in obese adolescents and adolescents with diabetes type 1
- Dysregulated glucose homeostasis in congenital central hypoventilation syndrome
- Do parents of children with metabolic diseases benefit from the Triple P – Positive Parenting Program? A pilot study
- Early higher dosage of alglucosidase alpha in classic Pompe disease
- Insulin resistance in children with familial hyperlipidemia
- Insulin resistance linked to subtle myocardial dysfunction in normotensive Turner syndrome young patients without structural heart diseases
- Acetylcholinesterase activity and bone biochemical markers in premature and full-term neonates
- Baseline characteristics of gender dysphoric youth
- Case Reports
- Multi-disciplinary evaluation of a 5-month-old with hypertrophic cardiomyopathy related to a functional adrenocortical tumor
- A case of an infant with extremely low birth weight and hypothyroidism associated with massive cutaneous infantile hemangioma
- Possible hints and pitfalls in diagnosing Peutz-Jeghers syndrome