Multicentric Italian Case-control Study on 25OH Vitamin D Levels in Children and Adolescents with Prader-Willi Syndrome

Overview

Journal J Endocrinol Invest

Publisher Springer

Specialty Endocrinology

Date 2023 Jan 28

PMID 36708456

Authors

F M Panfili

A Convertino

G Grugni

L Mazzitelli

S Bocchini

A Crino

G Campana

M Cappa

M Delvecchio

M F Faienza

M R Licenziati

M Mariani

S Osimani

R Pajno

G Patti

I Rutigliano

M Sacco

E Scarano

D Fintini

Affiliations

Soon will be listed here.

Abstract

Purpose: 25OHD levels in patients with Prader-Willi Syndrome (PWS), the most frequent cause of genetic obesity with a peculiar fat mass distribution, are still debated. Insulin resistance (IR), Body Mass Index-SDS (BMI-SDS), Growth Hormone Therapy (GHT), and puberty onset seem to interact with 25OHD levels. The objectives of the study are: (1) To analyze 25OHD levels in pediatric PWS patients in comparison with a control group (CNT) (2) To evaluate a possible correlation between BMI-SDS, HOMA-IR, puberty, GHT, and 25OHD levels.

Methods: This is a retrospective case-control, multicenter study. Data were collected among 8 different Italian Hospitals (outpatient clinics), over a period of four years (2016-2020). We included 192 genetically confirmed PWS and 192 CNT patients, aged 3-18 years, matched 1:1 for age, gender, BMI-SDS, Tanner stage, sun exposure, and month of recruitment.

Results: No statistically significant differences in 25OHD levels were observed between the PWS population and the CNT (PWS 24.0 ng/mL vs CNT 22.5 ng/mL, p > 0.05), OR = 0.89 (95% CI 0.58-1.35). We observed a slight, although non-significant, reduction in 25OHD levels comparing NW and OB populations. HOMA-IR, puberty onset, genotype and GHT (previous or ongoing) did not show statistically significant correlation with 25OHD levels.

Conclusions: Our findings could be useful for clinicians to optimize the therapeutic management as well as to increase awareness of PWS.

Citing Articles

Endocrine features of Prader-Willi syndrome: a narrative review focusing on genotype-phenotype correlation.

Madeo S, Zagaroli L, Vandelli S, Calcaterra V, Crino A, de Sanctis L Front Endocrinol (Lausanne). 2024; 15:1382583.

PMID: 38737552 PMC: 11082343. DOI: 10.3389/fendo.2024.1382583.

References

Butler M, Hartin S, Hossain W, Manzardo A, Kimonis V, Dykens E . Molecular genetic classification in Prader-Willi syndrome: a multisite cohort study. J Med Genet. 2018; 56(3):149-153. PMC: 7387113. DOI: 10.1136/jmedgenet-2018-105301. View

Cassidy S, Schwartz S, Miller J, Driscoll D . Prader-Willi syndrome. Genet Med. 2012; 14(1):10-26. DOI: 10.1038/gim.0b013e31822bead0. View

Angulo M, Butler M, Cataletto M . Prader-Willi syndrome: a review of clinical, genetic, and endocrine findings. J Endocrinol Invest. 2015; 38(12):1249-63. PMC: 4630255. DOI: 10.1007/s40618-015-0312-9. View

Bochukova E, Lawler K, Croizier S, Keogh J, Patel N, Strohbehn G . A Transcriptomic Signature of the Hypothalamic Response to Fasting and BDNF Deficiency in Prader-Willi Syndrome. Cell Rep. 2018; 22(13):3401-3408. PMC: 5896230. DOI: 10.1016/j.celrep.2018.03.018. View

Longhi S, Grugni G, Gatti D, Spinozzi E, Sartorio A, Adami S . Adults with Prader-Willi syndrome have weaker bones: effect of treatment with GH and sex steroids. Calcif Tissue Int. 2015; 96(2):160-6. DOI: 10.1007/s00223-014-9949-1. View

Brunetti G, Grugni G, Piacente L, Delvecchio M, Ventura A, Giordano P . Analysis of Circulating Mediators of Bone Remodeling in Prader-Willi Syndrome. Calcif Tissue Int. 2018; 102(6):635-643. DOI: 10.1007/s00223-017-0376-y. View

Kroonen L, Herman M, Pizzutillo P, MacEwen G . Prader-Willi Syndrome: clinical concerns for the orthopaedic surgeon. J Pediatr Orthop. 2006; 26(5):673-9. DOI: 10.1097/01.bpo.0000226282.01202.4f. View

Pellikaan K, Rosenberg A, Kattentidt-Mouravieva A, Kersseboom R, Bos-Roubos A, Veen-Roelofs J . Missed Diagnoses and Health Problems in Adults With Prader-Willi Syndrome: Recommendations for Screening and Treatment. J Clin Endocrinol Metab. 2020; 105(12). PMC: 7553248. DOI: 10.1210/clinem/dgaa621. View

Grugni G, Sartorio A, Crino A . Growth hormone therapy for Prader-willi syndrome: challenges and solutions. Ther Clin Risk Manag. 2016; 12:873-81. PMC: 4898426. DOI: 10.2147/TCRM.S70068. View

10.

Waterbury S . Implications of vitamin D toxicity & deficiency. Nurse Pract. 2018; 43(5):22-30. DOI: 10.1097/01.NPR.0000531916.07387.d4. View

11.

Munns C, Shaw N, Kiely M, Specker B, Thacher T, Ozono K . Global Consensus Recommendations on Prevention and Management of Nutritional Rickets. J Clin Endocrinol Metab. 2016; 101(2):394-415. PMC: 4880117. DOI: 10.1210/jc.2015-2175. View

12.

Cediel G, Corvalan C, Aguirre C, de Romana D, Uauy R . Serum 25-Hydroxyvitamin D associated with indicators of body fat and insulin resistance in prepubertal chilean children. Int J Obes (Lond). 2015; 40(1):147-52. DOI: 10.1038/ijo.2015.148. View

13.

Saneei P, Salehi-Abargouei A, Esmaillzadeh A . Serum 25-hydroxy vitamin D levels in relation to body mass index: a systematic review and meta-analysis. Obes Rev. 2013; 14(5):393-404. DOI: 10.1111/obr.12016. View

14.

Orces C . The Association between Body Mass Index and Vitamin D Supplement Use among Adults in the United States. Cureus. 2019; 11(9):e5721. PMC: 6823089. DOI: 10.7759/cureus.5721. View

15.

Matar M, Al-Shaar L, Maalouf J, Nabulsi M, Arabi A, Choucair M . The Relationship Between Calciotropic Hormones, IGF-1, and Bone Mass Across Pubertal Stages. J Clin Endocrinol Metab. 2016; 101(12):4860-4870. DOI: 10.1210/jc.2016-3071. View

16.

Geserick M, Vogel M, Eckelt F, Schlingmann M, Hiemisch A, Baber R . Children and adolescents with obesity have reduced serum bone turnover markers and 25-hydroxyvitamin D but increased parathyroid hormone concentrations - Results derived from new pediatric reference ranges. Bone. 2019; 132:115124. DOI: 10.1016/j.bone.2019.115124. View

17.

Purtell L, Viardot A, Campbell L . Vitamin D levels in primary growth hormone deficiency disorder Prader-Willi syndrome. Endocrine. 2016; 53(2):619-20. DOI: 10.1007/s12020-016-0889-6. View

18.

Fintini D, Pedicelli S, Bocchini S, Bizzarri C, Grugni G, Cappa M . 25OH vitamin D levels in pediatric patients affected by Prader-Willi syndrome. J Endocrinol Invest. 2017; 41(6):739-742. DOI: 10.1007/s40618-017-0781-0. View

19.

Barrea L, Muscogiuri G, Pugliese G, Aprano S, de Alteriis G, Di Somma C . The Sun's Vitamin in Adult Patients Affected by Prader-Willi Syndrome. Nutrients. 2020; 12(4). PMC: 7230761. DOI: 10.3390/nu12041132. View

21.

Sode-Carlsen R, Farholt S, Rabben K, Bollerslev J, Schreiner T, Jurik A . Body composition, endocrine and metabolic profiles in adults with Prader-Willi syndrome. Growth Horm IGF Res. 2010; 20(3):179-84. DOI: 10.1016/j.ghir.2009.12.004. View