Literature DB >> 23696535

Healthcare transition in patients with rare genetic disorders with and without developmental disability: neurofibromatosis 1 and Williams-Beuren syndrome.

Andrea Van Lierde1, Francesca Menni, Maria Francesca Bedeschi, Federica Natacci, Sophie Guez, Paola Vizziello, Maria Antonella Costantino, Faustina Lalatta, Susanna Esposito.   

Abstract

There are between 5,000 and 8,000 distinct rare diseases (RDs) affecting 6-8% of the population, most of which are caused by genetic defects. Many are highly complex, childhood-onset, multi-system disorders that are often associated with developmental disability, and require lifelong, highly specialized care and support. As larger numbers of children with previously fatal RDs survive into adulthood, they encounter significant challenges in transitioning from family-centered, developmentally focused, multidisciplinary pediatric care to a less supportive adult healthcare system that is often unfamiliar with these conditions. This paper discusses the challenges of the transition from pediatric to adult health care in two groups of patients with multisystem genetic RDs (neurofibromatosis 1 [NF1] and Williams-Beuren syndrome [WBS]), and analyzes strategies for making the process easier for patients with and without developmental disabilities. Our findings show that there are still no guidelines in national healthcare programs on how to transition RD adolescents with and without developmental disabilities, and only a few pediatric centers have implemented the elements of transition in their general practice. Evidence regarding programs to facilitate transition is inconclusive and the transition from pediatric medicine to adult medicine for RDs remains a major challenge. However, transition requires both time and personnel, which are difficult to find in periods of fiscal austerity. Nevertheless, we should strongly advocate for governments investing more into transition infrastructure or they will face increased long-term social and economic costs due to poor treatment compliance, disengagement from services, increased genetic risks, and higher rates of disease-related complications.
Copyright © 2013 Wiley Periodicals, Inc.

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Year:  2013        PMID: 23696535     DOI: 10.1002/ajmg.a.35982

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  11 in total

1.  Spinal cord issues in adult patients with MPS: transition of care survey.

Authors:  Kemel A Ghotme; Fernando Alvarado-Gomez; Christina Lampe; Klane K White; Martha Solano-Villareal; Roberto Giugliani; Paul R Harmatz
Journal:  Childs Nerv Syst       Date:  2018-05-27       Impact factor: 1.475

Review 2.  The Transition to Adulthood for Youth Living with Rare Diseases.

Authors:  Melanie Sandquist; TjaMeika Davenport; Jana Monaco; Maureen E Lyon
Journal:  Children (Basel)       Date:  2022-05-12

Review 3.  Management of adolescents with congenital adrenal hyperplasia.

Authors:  Deborah P Merke; Dix P Poppas
Journal:  Lancet Diabetes Endocrinol       Date:  2013-11-15       Impact factor: 32.069

4.  Lifespan Development: Symptoms Experienced by Individuals with Neurofibromatosis Type 1 Associated Plexiform Neurofibromas from Childhood into Adulthood.

Authors:  Sally E Jensen; Zabin S Patel; Robert Listernick; Joel Charrow; Jin-Shei Lai
Journal:  J Clin Psychol Med Settings       Date:  2019-09

5.  Worries and needs of adults and parents of adults with neurofibromatosis type 1.

Authors:  Andre B Rietman; Hanneke van Helden; Pauline H Both; Walter Taal; Jeroen S Legerstee; AnneLoes van Staa; Henriette A Moll; Rianne Oostenbrink; Agnies M van Eeghen
Journal:  Am J Med Genet A       Date:  2018-05       Impact factor: 2.802

6.  Growing up with Fragile X Syndrome: Concerns and Care Needs of Young Adult Patients and Their Parents.

Authors:  M C Van Remmerden; L Hoogland; S E Mous; B Dierckx; M Coesmans; H A Moll; K Lubbers; C R Lincken; A M Van Eeghen
Journal:  J Autism Dev Disord       Date:  2020-06

7.  Transition to adult care of young patients with neurofibromatosis type 1 and cognitive deficits: a single-centre study.

Authors:  S Lausdahl; M M Handrup; S L Rubak; M D Jensen; C Ejerskov
Journal:  Orphanet J Rare Dis       Date:  2022-05-21       Impact factor: 4.123

Review 8.  Transitional Care in Pediatric Brain Tumor Patients: A Systematic Literature Review.

Authors:  Florian Ebel; Ladina Greuter; Raphael Guzman; Jehuda Soleman
Journal:  Children (Basel)       Date:  2022-04-02

9.  Care of adults with neurofibromatosis type 1: a clinical practice resource of the American College of Medical Genetics and Genomics (ACMG).

Authors:  Douglas R Stewart; Bruce R Korf; Katherine L Nathanson; David A Stevenson; Kaleb Yohay
Journal:  Genet Med       Date:  2018-04-26       Impact factor: 8.822

Review 10.  Healthcare transition from childhood to adulthood in Tuberous Sclerosis Complex.

Authors:  Angela Peron; Maria Paola Canevini; Filippo Ghelma; Fabiano Di Marco; Aglaia Vignoli
Journal:  Am J Med Genet C Semin Med Genet       Date:  2018-09-25       Impact factor: 3.908

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