| Literature DB >> 35388185 |
Sofia Douzgou1,2, Janet Dell'Oro3, Cristina Rodriguez Fonseca4, Alessandra Rei3, Jo Mullins5, Isabelle Jusiewicz6, Sylvia Huisman7,8, Brittany N Simpson9,10, Klea Vyshka11, Donatella Milani12, Oliver Bartsch13, Didier Lacombe14, Sixto García-Miñaúr15,16, Raoul C M Hennekam7.
Abstract
The existing knowledge about morbidity in adults with Rubinstein-Taybi syndrome (RTS) is limited and detailed data on their natural history and response to management are needed for optimal care in later life. We formed an international, multidisciplinary working group that developed an accessible questionnaire including key issues about adults with RTS and disseminated this to all known RTS support groups via social media. We report the observations from a cohort of 87 adult individuals of whom 43 had a molecularly confirmed diagnosis. The adult natural history of RTS is defined by prevalent behavioural/psychiatric problems (83%), gastrointestinal problems (73%) that are represented mainly by constipation; and sleep problems (62%) that manifest in a consistent pattern of sleep apnoea, difficulty staying asleep and an increased need for sleep. Furthermore, over than half of the RTS individuals (65%) had skin and adnexa-related problems. Half of the individuals receive multidisciplinary follow-up and required surgery at least once, and most frequently more than once, during adulthood. Our data confirm that adults with RTS enjoy both social and occupational possibilities, show a variegated experience of everyday life but experience a significant morbidity and ongoing medical issues which do not appear to be as coordinated and multidisciplinary managed as in paediatric patients. We highlight the need for optimal care in a multidisciplinary setting including the pivotal role of specialists for adult care.Entities:
Mesh:
Year: 2022 PMID: 35388185 PMCID: PMC9259744 DOI: 10.1038/s41431-022-01097-8
Source DB: PubMed Journal: Eur J Hum Genet ISSN: 1018-4813 Impact factor: 5.351