Increased prevalence of overweight and obesity in children with X-linked hypophosphatemia

Background/aim: X-linked hypophosphatemia (XLH) is a rare disease characterized by low phosphate levels. Scientific evidence points to a link between h ypophosphatemia and obesity in general population. The aim of our longitudinal observational study was to investigate the prevalence of obesity and...

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Main Authors: Volha V Zhukouskaya, Anya Rothenbuhler, Annamaria Colao, Carolina Di Somma, Peter Kamenický, Séverine Trabado, Dominique Prié, Christelle Audrain, Anna Barosi, Christèle Kyheng, Anne-Sophie Lambert, Agnès Linglart
Format: Article
Language:English
Published: Bioscientifica 2020-02-01
Series:Endocrine Connections
Subjects:
Online Access:https://ec.bioscientifica.com/view/journals/ec/9/2/EC-19-0481.xml
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author Volha V Zhukouskaya
Anya Rothenbuhler
Annamaria Colao
Carolina Di Somma
Peter Kamenický
Séverine Trabado
Dominique Prié
Christelle Audrain
Anna Barosi
Christèle Kyheng
Anne-Sophie Lambert
Agnès Linglart
spellingShingle Volha V Zhukouskaya
Anya Rothenbuhler
Annamaria Colao
Carolina Di Somma
Peter Kamenický
Séverine Trabado
Dominique Prié
Christelle Audrain
Anna Barosi
Christèle Kyheng
Anne-Sophie Lambert
Agnès Linglart
Increased prevalence of overweight and obesity in children with X-linked hypophosphatemia
Endocrine Connections
x-linked hypophosphatemia
rickets
phosphorus
overweight
obesity
author_facet Volha V Zhukouskaya
Anya Rothenbuhler
Annamaria Colao
Carolina Di Somma
Peter Kamenický
Séverine Trabado
Dominique Prié
Christelle Audrain
Anna Barosi
Christèle Kyheng
Anne-Sophie Lambert
Agnès Linglart
author_sort Volha V Zhukouskaya
title Increased prevalence of overweight and obesity in children with X-linked hypophosphatemia
title_short Increased prevalence of overweight and obesity in children with X-linked hypophosphatemia
title_full Increased prevalence of overweight and obesity in children with X-linked hypophosphatemia
title_fullStr Increased prevalence of overweight and obesity in children with X-linked hypophosphatemia
title_full_unstemmed Increased prevalence of overweight and obesity in children with X-linked hypophosphatemia
title_sort increased prevalence of overweight and obesity in children with x-linked hypophosphatemia
publisher Bioscientifica
series Endocrine Connections
issn 2049-3614
2049-3614
publishDate 2020-02-01
description Background/aim: X-linked hypophosphatemia (XLH) is a rare disease characterized by low phosphate levels. Scientific evidence points to a link between h ypophosphatemia and obesity in general population. The aim of our longitudinal observational study was to investigate the prevalence of obesity and associated factors in a large cohort of children with XLH. Patients/methods: We studied 172 XLH-children 5–20 years of age (113 girls/59 boys). Anthropometric parameters (weight, height, and BMI) were collected at birth and during follow-up at mean ages of 5.3, 8.2, 11.3, and 15.9 years (groups 1, 2, 3, and 4, respectively). In each group, subjects were classified based on International Obesity Taskforce (IOTF) cut off values of BMI for age and sex as overwe ight or obese (IOTF 25–30 or ≥30 kg/m2, respectively). Results: In each age-group, almost 1/3 of XLH-patients were classified a s overweight or obese (29.4, 28.7, 27.5, and 36.7% in groups 1, 2, 3, and 4, respectively). Children without a XLH-family history had higher BMI-IOTF at every point of follow-up, compared to those with positive XLH-family history. Similarly, higher BMI-IOTF wa s significantly associated with treatment duration (23.3 ± 4.4 vs 23.8 ± 3.8 vs 25.2 ± 4.5 kg/m2, for subjects with treatment duration of <5, 5–10 and >10 years, respectively, P for trend = 0.025). Multiple regression analysis confirmed an association of treatment durati on and lack of XLH-family history with higher BMI-IOTF. Conclusion: One out of three of XLH-children have phenotypically unfavourable metabolic profile expressed as increased prevalence of overweight or obesi ty in comparison to general population. Both the lack of XLH family history and the duration of treatment increase the risk of higher BMI-IOTF. BMI should be carefully monitored in children, and later in adults, with XLH.
topic x-linked hypophosphatemia
rickets
phosphorus
overweight
obesity
url https://ec.bioscientifica.com/view/journals/ec/9/2/EC-19-0481.xml
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spelling doaj-96d17c4323c4493fab48a127761405cb2020-11-25T00:01:31ZengBioscientificaEndocrine Connections2049-36142049-36142020-02-0192144153https://doi.org/10.1530/EC-19-0481Increased prevalence of overweight and obesity in children with X-linked hypophosphatemiaVolha V Zhukouskaya0Anya Rothenbuhler1Annamaria Colao2Carolina Di Somma3Peter Kamenický4Séverine Trabado5Dominique Prié6Christelle Audrain7Anna Barosi8Christèle Kyheng9Anne-Sophie Lambert10Agnès Linglart11APHP, Reference Center for Rare Disorders of the Calcium and Phosphate Metabolism, FilièreOSCAR and Platform of Expertise for Rare Diseases Paris-Saclay, Bicêtre Paris-Saclay Hospital, Le Kremlin-Bicêtre, France; Division of Endocrinology, Department of Clinical Medicine and Surgery, University of Naples Federico II, Naples, ItalyAPHP, Reference Center for Rare Disorders of the Calcium and Phosphate Metabolism, FilièreOSCAR and Platform of Expertise for Rare Diseases Paris-Saclay, Bicêtre Paris-Saclay Hospital, Le Kremlin-Bicêtre, France; APHP, Department of Endocrinology and Diabetology for Children, Bicêtre Paris Saclay Hospital, Le Kremlin-Bicêtre, FranceDivision of Endocrinology, Department of Clinical Medicine and Surgery, University of Naples Federico II, Naples, ItalyDivision of Endocrinology, Department of Clinical Medicine and Surgery, University of Naples Federico II, Naples, Italy; IRCCS SDN, Naples, ItalyAPHP, Reference Center for Rare Disorders of the Calcium and Phosphate Metabolism, FilièreOSCAR and Platform of Expertise for Rare Diseases Paris-Saclay, Bicêtre Paris-Saclay Hospital, Le Kremlin-Bicêtre, France; APHP, Department of Endocrinology and Reproductive Diseases, Bicêtre Paris-Saclay Hospital, Le Kremlin-Bicêtre, France; Paris Sud – Paris Saclay University, Faculté de Médecine, Le Kremlin-Bicêtre, FranceParis Sud – Paris Saclay University, Faculté de Médecine, Le Kremlin-Bicêtre, France; APHP, Department of Molecular Genetics, Pharmacogenetics and Hormonology, Bicêtre Paris-Saclay Hospital, Le Kremlin-Bicêtre, FranceUniversité Paris V, Faculté de Médecine, Paris, France; Hôpital Necker EnfantsMalades APHP, INSERM U1151, Paris, FranceAPHP, Reference Center for Rare Disorders of the Calcium and Phosphate Metabolism, FilièreOSCAR and Platform of Expertise for Rare Diseases Paris-Saclay, Bicêtre Paris-Saclay Hospital, Le Kremlin-Bicêtre, FranceAPHP, Reference Center for Rare Disorders of the Calcium and Phosphate Metabolism, FilièreOSCAR and Platform of Expertise for Rare Diseases Paris-Saclay, Bicêtre Paris-Saclay Hospital, Le Kremlin-Bicêtre, FranceAPHP, Department of Adolescent Medicine, Bicêtre Paris Saclay Hospital, Le Kremlin-Bicêtre, FranceAPHP, Reference Center for Rare Disorders of the Calcium and Phosphate Metabolism, FilièreOSCAR and Platform of Expertise for Rare Diseases Paris-Saclay, Bicêtre Paris-Saclay Hospital, Le Kremlin-Bicêtre, France; APHP, Department of Endocrinology and Diabetology for Children, Bicêtre Paris Saclay Hospital, Le Kremlin-Bicêtre, France; APHP, Department of Adolescent Medicine, Bicêtre Paris Saclay Hospital, Le Kremlin-Bicêtre, FranceAPHP, Reference Center for Rare Disorders of the Calcium and Phosphate Metabolism, FilièreOSCAR and Platform of Expertise for Rare Diseases Paris-Saclay, Bicêtre Paris-Saclay Hospital, Le Kremlin-Bicêtre, France; APHP, Department of Endocrinology and Diabetology for Children, Bicêtre Paris Saclay Hospital, Le Kremlin-Bicêtre, France; Paris Sud – Paris Saclay University, Faculté de Médecine, Le Kremlin-Bicêtre, FranceBackground/aim: X-linked hypophosphatemia (XLH) is a rare disease characterized by low phosphate levels. Scientific evidence points to a link between h ypophosphatemia and obesity in general population. The aim of our longitudinal observational study was to investigate the prevalence of obesity and associated factors in a large cohort of children with XLH. Patients/methods: We studied 172 XLH-children 5–20 years of age (113 girls/59 boys). Anthropometric parameters (weight, height, and BMI) were collected at birth and during follow-up at mean ages of 5.3, 8.2, 11.3, and 15.9 years (groups 1, 2, 3, and 4, respectively). In each group, subjects were classified based on International Obesity Taskforce (IOTF) cut off values of BMI for age and sex as overwe ight or obese (IOTF 25–30 or ≥30 kg/m2, respectively). Results: In each age-group, almost 1/3 of XLH-patients were classified a s overweight or obese (29.4, 28.7, 27.5, and 36.7% in groups 1, 2, 3, and 4, respectively). Children without a XLH-family history had higher BMI-IOTF at every point of follow-up, compared to those with positive XLH-family history. Similarly, higher BMI-IOTF wa s significantly associated with treatment duration (23.3 ± 4.4 vs 23.8 ± 3.8 vs 25.2 ± 4.5 kg/m2, for subjects with treatment duration of <5, 5–10 and >10 years, respectively, P for trend = 0.025). Multiple regression analysis confirmed an association of treatment durati on and lack of XLH-family history with higher BMI-IOTF. Conclusion: One out of three of XLH-children have phenotypically unfavourable metabolic profile expressed as increased prevalence of overweight or obesi ty in comparison to general population. Both the lack of XLH family history and the duration of treatment increase the risk of higher BMI-IOTF. BMI should be carefully monitored in children, and later in adults, with XLH. https://ec.bioscientifica.com/view/journals/ec/9/2/EC-19-0481.xmlx-linked hypophosphatemiaricketsphosphorusoverweightobesity