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| ID | Type | Description | Link |
|---|---|---|---|
| 2016-A01348-43 | Other Identifier | ID-RCB |
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The objective of this study is to collect data on tolerance and effects of early treatment with oxytocin in children with Prader Willi Syndrome aged from 3 to 4 years and to compare these infants with not treated age-matched infants with Prader Willi Syndrome.
In accordance with recommendations of regulatory authorities, we want to collect long term data of patients treated with oxytocin before the age of 6 months. Moreover clinical observations of these infants support long term effects on communication skills, global development and behaviour.
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| Label | Type | Description | Intervention Names |
|---|---|---|---|
| Oxytocin | Experimental | Groups of children with Prader Willi Syndrome treated by oxytocin for 7 days during their first 6 months of life. |
|
| Control | Experimental | Groups of children with Prader Willi Syndrome not treated by oxytocin for 7 days during their first 6 months of life. |
|
| Name | Type | Description | Arm Group Labels | Other Names |
|---|---|---|---|---|
| Oxytocin | Drug | Infant included in the ancient study (repeated administrations of oxytocin in infants with Prader Willi Syndrome aged from 0 to 6 months) |
|
| Measure | Description | Time Frame |
|---|---|---|
| Evaluation of communication skills. | Assessed by Vineland-II scale. | Day 1 |
| Measure | Description | Time Frame |
|---|---|---|
| Evaluation of adaptative behavior composite and 3 domains : "Daily living skills", "Socialization", "Motor skills". | Assessed by Vineland-II scale. | Day 1 |
| Evaluation of behavioral troubles. | Assessed by Child Behaviour Check List questionnaire. |
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Inclusion Criteria:
Exclusion Criteria:
If a patient has a contraindication to Magnetic resonance imaging, it may be included in the study but Magnetic resonance imaging will not be performed.
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| Name | Affiliation | Role |
|---|---|---|
| Maïthé Tauber, Pr | Centre de référence du syndrome de Prader-Willi- CHU Toulouse | Principal Investigator |
| Facility | Status | City | State | ZIP | Country | Contacts |
|---|---|---|---|---|---|---|
| Centre de référence du syndrome de Prader-Willi Hôpital des Enfants | Toulouse | 31059 | France |
| PubMed Identifier | Type | Citation | Retractions |
|---|---|---|---|
| 16150836 | Background | Amico JA, Vollmer RR, Cai HM, Miedlar JA, Rinaman L. Enhanced initial and sustained intake of sucrose solution in mice with an oxytocin gene deletion. Am J Physiol Regul Integr Comp Physiol. 2005 Dec;289(6):R1798-806. doi: 10.1152/ajpregu.00558.2005. Epub 2005 Sep 8. | |
| 2087513 | Background | Arletti R, Benelli A, Bertolini A. Oxytocin inhibits food and fluid intake in rats. Physiol Behav. 1990 Dec;48(6):825-30. doi: 10.1016/0031-9384(90)90234-u. |
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| Type | Date | Date Unknown |
|---|---|---|
| Release | Jun 6, 2023 | |
| Reset | Feb 9, 2024 | |
| Release | Mar 8, 2024 | |
| Reset | Aug 9, 2024 |
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| Release Date | Unrelease Date | Unrelease Date Unknown | Reset Date | MCP Release Number |
|---|---|---|---|---|
| Jun 6, 2023 | Feb 9, 2024 | |||
| Mar 8, 2024 |
| ID | Term |
|---|---|
| D011218 | Prader-Willi Syndrome |
| ID | Term |
|---|---|
| D008607 | Intellectual Disability |
| D019954 | Neurobehavioral Manifestations |
| D009461 | Neurologic Manifestations |
| D009422 | Nervous System Diseases |
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| ID | Term |
|---|---|
| D010121 | Oxytocin |
| ID | Term |
|---|---|
| D010909 | Pituitary Hormones, Posterior |
| D010907 | Pituitary Hormones |
| D036361 | Peptide Hormones |
| D006728 | Hormones |
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| Control | Other | Not treated. |
|
| Day 1 |
| Evaluation of global development. | Assessed by Bayley Scales of Infant and Toddler Development. | Day 2 and 3 |
| Evaluation of orality and eating behaviour. | Assessed by:
| Day 2 |
| Evaluation of brain activity. | Assessed by a morphological Magnetic resonance imaging, a resting functional Magnetic resonance imaging. | Day 3 |
| Evaluation of plasma levels of ghrelin and other peptides involved in feeding behaviour or energy metabolism. | Circulating levels of acylated and non-acylated ghrelin and some peptides and neuropeptides involved in appetite regulation (leptin, cortisol, insulin, Glucagon like peptide-1, pancreatic polypeptide, orexin A, alpha-melanocyte stimulating hormone...). | Day 1 |
| 16677726 | Background | Billings LB, Spero JA, Vollmer RR, Amico JA. Oxytocin null mice ingest enhanced amounts of sweet solutions during light and dark cycles and during repeated shaker stress. Behav Brain Res. 2006 Jul 15;171(1):134-41. doi: 10.1016/j.bbr.2006.03.028. Epub 2006 May 4. |
| 17236194 | Background | Bittel DC, Kibiryeva N, Sell SM, Strong TV, Butler MG. Whole genome microarray analysis of gene expression in Prader-Willi syndrome. Am J Med Genet A. 2007 Mar 1;143A(5):430-42. doi: 10.1002/ajmg.a.31606. |
| 21858456 | Background | Dykens EM, Lee E, Roof E. Prader-Willi syndrome and autism spectrum disorders: an evolving story. J Neurodev Disord. 2011 Sep;3(3):225-37. doi: 10.1007/s11689-011-9092-5. Epub 2011 Aug 20. |
| 17636101 | Background | Dykens EM, Maxwell MA, Pantino E, Kossler R, Roof E. Assessment of hyperphagia in Prader-Willi syndrome. Obesity (Silver Spring). 2007 Jul;15(7):1816-26. doi: 10.1038/oby.2007.216. |
| 27481444 | Background | Dykens EM, Roof E, Hunt-Hawkins H. Cognitive and adaptive advantages of growth hormone treatment in children with Prader-Willi syndrome. J Child Psychol Psychiatry. 2017 Jan;58(1):64-74. doi: 10.1111/jcpp.12601. Epub 2016 Aug 2. |
| 22270375 | Background | Goldstone AP, Holland AJ, Butler JV, Whittington JE. Appetite hormones and the transition to hyperphagia in children with Prader-Willi syndrome. Int J Obes (Lond). 2012 Dec;36(12):1564-70. doi: 10.1038/ijo.2011.274. Epub 2012 Jan 24. |
| 19897177 | Background | Guastella AJ, Einfeld SL, Gray KM, Rinehart NJ, Tonge BJ, Lambert TJ, Hickie IB. Intranasal oxytocin improves emotion recognition for youth with autism spectrum disorders. Biol Psychiatry. 2010 Apr 1;67(7):692-4. doi: 10.1016/j.biopsych.2009.09.020. Epub 2009 Nov 7. |
| 21862226 | Background | Hall SS, Lightbody AA, McCarthy BE, Parker KJ, Reiss AL. Effects of intranasal oxytocin on social anxiety in males with fragile X syndrome. Psychoneuroendocrinology. 2012 Apr;37(4):509-18. doi: 10.1016/j.psyneuen.2011.07.020. Epub 2011 Aug 20. |
| 15931222 | Background | Kosfeld M, Heinrichs M, Zak PJ, Fischbacher U, Fehr E. Oxytocin increases trust in humans. Nature. 2005 Jun 2;435(7042):673-6. doi: 10.1038/nature03701. |
| 22365820 | Background | Lischke A, Gamer M, Berger C, Grossmann A, Hauenstein K, Heinrichs M, Herpertz SC, Domes G. Oxytocin increases amygdala reactivity to threatening scenes in females. Psychoneuroendocrinology. 2012 Sep;37(9):1431-8. doi: 10.1016/j.psyneuen.2012.01.011. Epub 2012 Feb 23. |
| 18655878 | Background | McDonald NA, Kuzmiski JB, Naderi N, Schwab Y, Pittman QJ. Endogenous modulators of synaptic transmission: cannabinoid regulation in the supraoptic nucleus. Prog Brain Res. 2008;170:129-36. doi: 10.1016/S0079-6123(08)00412-3. |
| 25599930 | Background | Meziane H, Schaller F, Bauer S, Villard C, Matarazzo V, Riet F, Guillon G, Lafitte D, Desarmenien MG, Tauber M, Muscatelli F. An Early Postnatal Oxytocin Treatment Prevents Social and Learning Deficits in Adult Mice Deficient for Magel2, a Gene Involved in Prader-Willi Syndrome and Autism. Biol Psychiatry. 2015 Jul 15;78(2):85-94. doi: 10.1016/j.biopsych.2014.11.010. Epub 2014 Nov 20. |
| 21465655 | Background | Miller JL, Lynn CH, Driscoll DC, Goldstone AP, Gold JA, Kimonis V, Dykens E, Butler MG, Shuster JJ, Driscoll DJ. Nutritional phases in Prader-Willi syndrome. Am J Med Genet A. 2011 May;155A(5):1040-9. doi: 10.1002/ajmg.a.33951. Epub 2011 Apr 4. |
| 26994593 | Background | Salles J, Strelnikov K, Carine M, Denise T, Laurier V, Molinas C, Tauber M, Barone P. Deficits in voice and multisensory processing in patients with Prader-Willi syndrome. Neuropsychologia. 2016 May;85:137-47. doi: 10.1016/j.neuropsychologia.2016.03.015. Epub 2016 Mar 16. |
| 20876615 | Background | Schaller F, Watrin F, Sturny R, Massacrier A, Szepetowski P, Muscatelli F. A single postnatal injection of oxytocin rescues the lethal feeding behaviour in mouse newborns deficient for the imprinted Magel2 gene. Hum Mol Genet. 2010 Dec 15;19(24):4895-905. doi: 10.1093/hmg/ddq424. Epub 2010 Sep 28. |
| 22876265 | Background | Skokauskas N, Sweeny E, Meehan J, Gallagher L. Mental health problems in children with prader-willi syndrome. J Can Acad Child Adolesc Psychiatry. 2012 Aug;21(3):194-203. |
| 7852523 | Background | Swaab DF, Purba JS, Hofman MA. Alterations in the hypothalamic paraventricular nucleus and its oxytocin neurons (putative satiety cells) in Prader-Willi syndrome: a study of five cases. J Clin Endocrinol Metab. 1995 Feb;80(2):573-9. doi: 10.1210/jcem.80.2.7852523. |
| 9585637 | Background | van Lieshout CF, de Meyer RE, Curfs LM, Koot HM, Fryns JP. Problem behaviors and personality of children and adolescents with Prader-Willi syndrome. J Pediatr Psychol. 1998 Apr;23(2):111-20. doi: 10.1093/jpepsy/23.2.111. |
| 40025514 | Derived | Valette M, Diene G, Glattard M, Cortadellas J, Molinas C, Faye S, Benvegnu G, Boulanouar K, Payoux P, Salles JP, Arnaud C, Cabal S, Tauber M. Early oxytocin treatment in infants with Prader-Willi syndrome is safe and is associated with better endocrine, metabolic and behavioral outcomes. Orphanet J Rare Dis. 2025 Mar 1;20(1):96. doi: 10.1186/s13023-025-03560-3. |
| Aug 9, 2024 |
| D000015 | Abnormalities, Multiple |
| D000013 | Congenital Abnormalities |
| D009358 | Congenital, Hereditary, and Neonatal Diseases and Abnormalities |
| D025063 | Chromosome Disorders |
| D030342 | Genetic Diseases, Inborn |
| D000096803 | Imprinting Disorders |
| D009765 | Obesity |
| D050177 | Overweight |
| D044343 | Overnutrition |
| D009748 | Nutrition Disorders |
| D009750 | Nutritional and Metabolic Diseases |
| D006730 |
| Hormones, Hormone Substitutes, and Hormone Antagonists |
| D010455 | Peptides |
| D000602 | Amino Acids, Peptides, and Proteins |