Article (Scientific journals)
The use of oxytocin to improve feeding and social skills in infants with Prader-Willi syndrome
Tauber, Maïthé; Boulanouar, Kader; Gwenaelle, Diene et al.
2017In Pediatrics, 139 (2)
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Keywords :
Prader-Willi syndrome; Oxytocin
Abstract :
[en] BACKGROUND AND OBJECTIVES: Patients with Prader–Willi syndrome (PWS) display poor feeding and social skills as infants and fewer hypothalamic oxytocin (OXT)-producing neurons were documented in adults. Animal data demonstrated that early treatment with OXT restores sucking after birth. Our aim is to reproduce these data in infants with PWS. METHODS: We conducted a phase 2 escalating dose study of a short course (7 days) of intranasal OXT administration. We enrolled 18 infants with PWS under 6 months old (6 infants in each step) who received 4 IU of OXT either every other day, daily, or twice daily. We investigated the tolerance and the effects on feeding and social skills and changes in circulating ghrelin and brain connectivity by functional MRI. RESULTS: No adverse events were reported. No dose effect was observed. Sucking assessed by the Neonatal Oral-Motor Scale was abnormal in all infants at baseline and normalized in 88% after treatment. The scores of Neonatal Oral-Motor Scale and videofluoroscopy of swallowing significantly decreased from 16 to 9 (P < .001) and from 18 to 12.5 (P < .001), respectively. Significant improvements in Clinical Global Impression scale scores, social withdrawal behavior, and mother–infant interactions were observed. We documented a significant increase in acylated ghrelin and connectivity of the right superior orbitofrontal network that correlated with changes in sucking and behavior. CONCLUSIONS: OXT is well tolerated in infants with PWS and improves feeding and social skills. These results open perspectives for early treatment in neurodevelopment diseases with feeding problems.
Disciplines :
Pediatrics
Author, co-author :
Tauber, Maïthé;  Hôpital des Enfants - Toulouse > Endocrinologie
Boulanouar, Kader;  Université de Toulouse > Toulouse Neuroimaging Center
Gwenaelle, Diene;  Hôpital des Enfants - Toulouse > INSERM UMR1027 > Endocrinologie
Cabal-Berthoumieu, Sophie;  Hôpital des Enfants - Toulouse > Endocrinologie
Ehlinger, Virginie;  Université Paul Sabatier - Toulouse 3 - UPS > INSERM URM1027
Fichaux-Bourin, Pascale;  Hôpital des Enfants - Toulouse > Endocrinologie
Molinas, Catherine;  Université Paul Sabatier - Toulouse 3 - UPS > INSERM U1043
Faye, Sandy;  Hôpital des Enfants - Toulouse > Endocrinologie & INSERM
Valette, Marion;  Hôpital des Enfants - Toulouse > Endocrinologie & INSERM
Pourrinet, Jeanne;  Hôpital des Enfants - Toulouse > Endocrinologie
Cessans, Catie;  Hôpital des Enfants - Toulouse > Endocrinologie
Viaux-Sauvelon, Sylvie;  Groupe Hospitalier Pitié-Salpêtrière > Psychiatrie de l'Enfant
Bascoul, Céline;  Hôpital des Enfants - Toulouse > Psychiatrie de l'Enfant
Guedeney, Antoine;  Hôpital Bichat Claude Bernard > Psychiatrie de l'Enfant
Delhanty, Patrick;  Erasmus Universiteit Rotterdam - EUR > Internal Medicine
Geenen, Vincent ;  Université de Liège > Centre d'immunologie
Martens, Henri ;  Université de Liège > Centre d'immunologie
Muscatelli, Françoise;  INSERM U191 > Institut de Neurobiologie de la Méditerranée
Cohen, David;  Centre National de la Recherche Scientifique - CNRS > Unité mixte 7222
Consoli, Angèle;  Université Pierre et Marie Currie - Paris 6 - UPMC > Psychiatrie de l'Enfant
Payoux, Pierre;  Université Paul Sabatier - Toulouse 3 - UPS > INSERM & Toulouse NeuroImaging Center
Arnaud, Catherine;  Université Paul Sabatier - Toulouse 3 - UPS > Soutien Méthodologique à la Recherche
Salles, Jean-Pierre;  Hôpital des Enfants > Endocrinologie
More authors (13 more) Less
Language :
English
Title :
The use of oxytocin to improve feeding and social skills in infants with Prader-Willi syndrome
Publication date :
2017
Journal title :
Pediatrics
ISSN :
0031-4005
eISSN :
1098-4275
Publisher :
American Academy of Pediatrics, Elk Grove Village, United States - Illinois
Volume :
139
Issue :
2
Peer reviewed :
Peer Reviewed verified by ORBi
Funders :
Hospital University of Toulouse
Available on ORBi :
since 19 January 2017

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