Experiences of adolescents living with Silver-Russell syndrome
Experiences of adolescents living with Silver-Russell syndrome
Objective: the psychosocial impact of growing up with Silver-Russell syndrome (SRS), characterised by growth failure and short stature in adulthood, has been explored in adults; however, there are no accounts of contemporary lived experience in adolescents. Such data could inform current healthcare guidance and transition to adult services. We aimed to explore the lived experience of adolescents with SRS.
Design/setting/patients: in-depth, semi-structured interviews were conducted between January 2015 and October 2016 with a sample of eight adolescents aged 13–18 (five girls) with genetically confirmed SRS from the UK. Qualitative interviews were transcribed and coded to identify similarities and differences using thematic analysis; codes were then grouped to form overarching themes.
Results: we identified four themes from the interview data: (1) the psychosocial challenges of feeling and looking different; (2) pain, disability and fatigue; (3) anticipated stigma; and (4) building resilience and acceptance. Despite adolescents accepting SRS in their lives, they described ongoing psychosocial challenges and anticipated greater problems to come, such as stigma from prospective employers.
Conclusions: adolescents with SRS may experience psychosocial difficulties from as young as 10 years old related to feeling and looking different; pain, disability and fatigue; anticipated stigma; and future challenges around employment. We discuss these findings in relation to recommendations for the care of adolescents with SRS to prepare them for adult life.
adolescent health, genetics, growth, psychology, qualitative research
1195-1201
Ballard, Lisa
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Jenkinson, Elizabeth
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Byrne, Christopher
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Child, Jenny
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Inskip, Hazel
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Lokulo-Sodipe, Oluwakemi
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Mackay, Deborah
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Wakeling, Emma
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Davies, Justin
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Temple, Karen
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Fenwick, Angela J
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1 December 2021
Ballard, Lisa
48a7b1af-4d2b-4ec7-8927-84361a3c62a9
Jenkinson, Elizabeth
b1997162-8fa8-4d72-9b3a-a4b52e2dbef4
Byrne, Christopher
1370b997-cead-4229-83a7-53301ed2a43c
Child, Jenny
2d053263-ccc6-4876-a8a4-4cda40c3fc3a
Inskip, Hazel
5fb4470a-9379-49b2-a533-9da8e61058b7
Lokulo-Sodipe, Oluwakemi
d428f857-0e58-4964-b1f0-136af7432805
Mackay, Deborah
588a653e-9785-4a00-be71-4e547850ee4a
Wakeling, Emma
f7f73171-4d52-41bf-bd6c-61d202b98453
Davies, Justin
9f18fcad-f488-4c72-ac23-c154995443a9
Temple, Karen
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Fenwick, Angela J
95a1f4fa-7f6f-4c07-a93b-9ea39c231c31
Ballard, Lisa, Jenkinson, Elizabeth, Byrne, Christopher, Child, Jenny, Inskip, Hazel, Lokulo-Sodipe, Oluwakemi, Mackay, Deborah, Wakeling, Emma, Davies, Justin, Temple, Karen and Fenwick, Angela J
(2021)
Experiences of adolescents living with Silver-Russell syndrome.
Archives of Disease in Childhood, 106 (12), , [321376].
(doi:10.1136/archdischild-2020-321376).
Abstract
Objective: the psychosocial impact of growing up with Silver-Russell syndrome (SRS), characterised by growth failure and short stature in adulthood, has been explored in adults; however, there are no accounts of contemporary lived experience in adolescents. Such data could inform current healthcare guidance and transition to adult services. We aimed to explore the lived experience of adolescents with SRS.
Design/setting/patients: in-depth, semi-structured interviews were conducted between January 2015 and October 2016 with a sample of eight adolescents aged 13–18 (five girls) with genetically confirmed SRS from the UK. Qualitative interviews were transcribed and coded to identify similarities and differences using thematic analysis; codes were then grouped to form overarching themes.
Results: we identified four themes from the interview data: (1) the psychosocial challenges of feeling and looking different; (2) pain, disability and fatigue; (3) anticipated stigma; and (4) building resilience and acceptance. Despite adolescents accepting SRS in their lives, they described ongoing psychosocial challenges and anticipated greater problems to come, such as stigma from prospective employers.
Conclusions: adolescents with SRS may experience psychosocial difficulties from as young as 10 years old related to feeling and looking different; pain, disability and fatigue; anticipated stigma; and future challenges around employment. We discuss these findings in relation to recommendations for the care of adolescents with SRS to prepare them for adult life.
Text
archdischild-2020-321376.R2_Proof_hi
- Accepted Manuscript
More information
Accepted/In Press date: 8 March 2021
e-pub ahead of print date: 19 March 2021
Published date: 1 December 2021
Additional Information:
Funding Information:
Acknowledgements We would like to thank the Child Growth Foundation for its support (http://www.childgrowthf oundation.org/). CDB is funded in part by the Southampton National Institute for Health Research (NIHR) Biomedical Research Centre.
Funding Information:
Disclaimer The views expressed are those of the author(s) and not necessarily those of the NHS, the NIHR or the Department of Health and Social Care. Competing interests JHD has received travel bursaries from Novo Nordisk, SANDOZ, Ferring and Pfizer. HI reports grants from the UK NIHR and UK Medical Research Council (the latter for her salary) during the conduct of the study. OL-S reports grants from the NIHR during the conduct of the study.
Funding Information:
Funding This paper presents independent research funded by the NIHR under its Research for Patient Benefit (RfPB) Programme (Grant Reference Number PB-PG-1111–26003) with support from NIHR CRN: Wessex, NIHR Southampton BRC and NIHR Wellcome Trust Southampton Clinical Research Facility. The RfPB grant holders were IKT, JHD, JCC, CDB, AF and HI.
Publisher Copyright:
© Authors 2021
Keywords:
adolescent health, genetics, growth, psychology, qualitative research
Identifiers
Local EPrints ID: 447982
URI: http://eprints.soton.ac.uk/id/eprint/447982
ISSN: 0003-9888
PURE UUID: d43f221e-be1b-44d1-a0c9-326a2a06cb88
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Date deposited: 29 Mar 2021 16:37
Last modified: 17 Mar 2024 03:37
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Contributors
Author:
Elizabeth Jenkinson
Author:
Jenny Child
Author:
Oluwakemi Lokulo-Sodipe
Author:
Emma Wakeling
Author:
Angela J Fenwick
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