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Is ghrelin a biomarker of early-onset scoliosis in children with Prader-Willi syndrome?
Pacoricona Alfaro, Dibia Liz; Diene, Gwenaelle; Pinto, Graziella; Salles, Jean-Pierre; Gennero, Isabelle; Faye, Sandy; Molinas, Catherine; Valette, Marion; Arnaud, Catherine; Tauber, Maithé.
Afiliación
  • Pacoricona Alfaro DL; CERPOP, Toulouse University, Inserm - Paul Sabatier University, Toulouse, France.
  • Diene G; CERPOP, Toulouse University, Inserm - Paul Sabatier University, Toulouse, France.
  • Pinto G; Endocrinology, Obesity, Bone Diseases, Genetics and Gynecology Unit, Children's Hospital, University Hospital Center of Toulouse, Toulouse, France.
  • Salles JP; French National Reference Center for Prader-Willi Syndrome, Children's Hospital, University Hospital Center of Toulouse, 330, Avenue de Grande-Bretagne - TSA, 70034 - 31059, Toulouse Cedex 9, France.
  • Gennero I; Pediatric Endocrinology, Diabetology and Gynecology Department, Assistance-Publique Hôpitaux de Paris (AP-HP), Necker-Enfants Malades Hospital, Paris, France.
  • Faye S; Endocrinology, Obesity, Bone Diseases, Genetics and Gynecology Unit, Children's Hospital, University Hospital Center of Toulouse, Toulouse, France.
  • Molinas C; Toulouse Institute of Infectious and Inflammatory Diseases (Infinity), Inserm UMR1291 - CNRS UMR5051, University Toulouse III, Toulouse, France.
  • Valette M; French National Reference Center for Rare Diseases of Calcium and Phosphate Metabolism - ERN BOND, University Hospital Center of Toulouse, Toulouse, France.
  • Arnaud C; Toulouse Institute of Infectious and Inflammatory Diseases (Infinity), Inserm UMR1291 - CNRS UMR5051, University Toulouse III, Toulouse, France.
  • Tauber M; Clinical Biochemistry and Molecular Biology Department, Federative Institute of Biology, University Hospital Center of Toulouse, Toulouse, France.
Orphanet J Rare Dis ; 16(1): 305, 2021 07 08.
Article en En | MEDLINE | ID: mdl-34238321
BACKGROUND: Adolescents with idiopathic scoliosis display high ghrelin levels. As hyperghrelinemia is found in patients with PWS and early-onset scoliosis (EOS) is highly prevalent in these patients, our aims were to explore (1) whether ghrelin levels differ between those with and without EOS and correlate with scoliosis severity, and (2) whether ghrelin levels in the first year of life are associated with the later development of EOS. METHODS: We used a case control study design for the first question and a longitudinal design for the second. Patients with PWS having plasma ghrelin measurements recorded between 2013 and 2018 in our database were selected and 30 children < 10 years old with EOS and 30 age- and BMI-matched controls without EOS were included. The Cobb angle at diagnosis was recorded. In addition, 37 infants with a ghrelin measurement in the first year of life were followed until 4 years of age and assessed for EOS. Total ghrelin (TG), acylated (AG) and unacylated ghrelin (UAG), and the AG/UAG ratio were analyzed. RESULTS: EOS children had an AG/UAG ratio statistically significantly lower than controls. The Cobb angle was positively correlated with TG and UAG. TG and AG in the first year of life were higher in infants who later develop EOS without reaching a statistically significant difference. CONCLUSIONS: Our results suggest that ghrelin may play a role in the pathophysiology of EOS in PWS. Higher ghrelinemia in the first year of life required careful follow-up for EOS.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Síndrome de Prader-Willi / Escoliosis Tipo de estudio: Observational_studies / Risk_factors_studies Límite: Adolescent / Child / Humans / Infant Idioma: En Revista: Orphanet J Rare Dis Asunto de la revista: MEDICINA Año: 2021 Tipo del documento: Article País de afiliación: Francia Pais de publicación: Reino Unido
Texto completo
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Síndrome de Prader-Willi / Escoliosis Tipo de estudio: Observational_studies / Risk_factors_studies Límite: Adolescent / Child / Humans / Infant Idioma: En Revista: Orphanet J Rare Dis Asunto de la revista: MEDICINA Año: 2021 Tipo del documento: Article País de afiliación: Francia Pais de publicación: Reino Unido