Literature DB >> 35388185

The natural history of adults with Rubinstein-Taybi syndrome: a families-reported experience.

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.
© 2022. The Author(s), under exclusive licence to European Society of Human Genetics.

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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


  30 in total

1.  Psychological and speech studies in Rubinstein-Taybi syndrome.

Authors:  R C Hennekam; A C Baselier; E Beyaert; A Bos; J B Blok; H B Jansma; V V Thorbecke-Nilsen; H Veerman
Journal:  Am J Ment Retard       Date:  1992-05

2.  Broad thumbs and toes and facial abnormalities. A possible mental retardation syndrome.

Authors:  J H RUBINSTEIN; H TAYBI
Journal:  Am J Dis Child       Date:  1963-06

3.  Repetitive behavior in Rubinstein-Taybi syndrome: parallels with autism spectrum phenomenology.

Authors:  Jane Waite; Joanna Moss; Sarah R Beck; Caroline Richards; Lisa Nelson; Kate Arron; Cheryl Burbidge; Katy Berg; Chris Oliver
Journal:  J Autism Dev Disord       Date:  2015-05

4.  Pilomatricomas in Rubinstein-Taybi syndrome.

Authors:  Despoina Papathemeli; Nicole Schulzendorff; Jürgen Kohlhase; Daniela Göppner; Ingolf Franke; Harald Gollnick
Journal:  J Dtsch Dermatol Ges       Date:  2015-03       Impact factor: 5.584

5.  Rubinstein-Taybi syndrome: an immune deficiency as a cause for recurrent infections.

Authors:  David R Naimi; Jose Munoz; Jack Rubinstein; Robert W Hostoffer
Journal:  Allergy Asthma Proc       Date:  2006 May-Jun       Impact factor: 2.587

Review 6.  Psychopathology, GABA, and the Rubinstein-Taybi syndrome: a review and case study.

Authors:  Jessica A Hellings; Shaheena Hossain; Judy K Martin; Ramil R Baratang
Journal:  Am J Med Genet       Date:  2002-03-08

7.  Behaviour across the lifespan in Cornelia de Lange syndrome.

Authors:  Laura Groves; Chris Oliver; Joanna Moss
Journal:  Curr Opin Psychiatry       Date:  2021-03-01       Impact factor: 4.741

Review 8.  Rubinstein-Taybi syndrome: clinical features, genetic basis, diagnosis, and management.

Authors:  Donatella Milani; Francesca Maria Paola Manzoni; Lidia Pezzani; Paola Ajmone; Cristina Gervasini; Francesca Menni; Susanna Esposito
Journal:  Ital J Pediatr       Date:  2015-01-20       Impact factor: 2.638

9.  Benign and malignant tumors in Rubinstein-Taybi syndrome.

Authors:  Max V Boot; Martine J van Belzen; Lucy I Overbeek; Nathalie Hijmering; Matias Mendeville; Quinten Waisfisz; Pieter Wesseling; Raoul C Hennekam; Daphne de Jong
Journal:  Am J Med Genet A       Date:  2018-01-23       Impact factor: 2.802

10.  Expanding the neurodevelopmental phenotypes of individuals with de novo KMT2A variants.

Authors:  Ada J S Chan; Cheryl Cytrynbaum; Ny Hoang; Patricia M Ambrozewicz; Rosanna Weksberg; Irene Drmic; Anne Ritzema; Russell Schachar; Susan Walker; Mohammed Uddin; Mehdi Zarrei; Ryan K C Yuen; Stephen W Scherer
Journal:  NPJ Genom Med       Date:  2019-04-26       Impact factor: 8.617

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  1 in total

1.  Clinical genomics testing: mainstreaming and globalising.

Authors:  Alisdair McNeill
Journal:  Eur J Hum Genet       Date:  2022-07       Impact factor: 5.351

  1 in total

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