The natural history of adults with Rubinstein-Taybi syndrome: a families-reported experience.
Sofia Douzgou HougeJanet Dell'OroCristina Rodriguez FonsecaAlessandra ReiJo MullinsIsabelle JusiewiczSylvia HuismanBrittany N SimpsonKlea VyshkaDonatella MilaniOliver BartschDidier LacombeSixto García-MiñaúrRaoul C M HennekamPublished in: European journal of human genetics : EJHG (2022)
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.
Keyphrases
- mental health
- healthcare
- quality improvement
- social media
- palliative care
- sleep quality
- end stage renal disease
- physical activity
- ejection fraction
- electronic health record
- emergency department
- newly diagnosed
- health information
- pain management
- intensive care unit
- depressive symptoms
- case report
- affordable care act
- peritoneal dialysis
- cross sectional
- prognostic factors
- deep brain stimulation
- acute coronary syndrome
- patient reported
- percutaneous coronary intervention