Coping with Wolf-Hirschhorn syndrome: quality of life and psychosocial features of family carers

dc.contributor.authorBerrocoso Cascallana, Sarah
dc.contributor.authorAmayra Caro, Imanol
dc.contributor.authorLázaro Pérez, Esther
dc.contributor.authorMartínez, Óscar
dc.contributor.authorLópez Paz, Juan Francisco
dc.contributor.authorGarcía Martín, Maitane
dc.contributor.authorPérez Álvarez, Manuel
dc.contributor.authorAl-Rashaida, Mohammad Ali Hussein
dc.contributor.authorRodríguez Bermejo, Alicia Aurora
dc.contributor.authorLuna Ovalle, Paula
dc.contributor.authorPérez Núñez, Paula
dc.contributor.authorBlanco, Raquel
dc.contributor.authorNevado, Julián
dc.date.accessioned2025-10-03T11:07:19Z
dc.date.available2025-10-03T11:07:19Z
dc.date.issued2020-10-19
dc.date.updated2025-10-03T11:07:18Z
dc.description.abstractBackground: Wolf-Hirschhorn Syndrome (WHS) is a rare, congenital disease characterized by a distinctive facial phenotype, seizures, intellectual disability and developmental delay, and pre and postnatal growth requiring lifelong care. The psychosocial status of the family caregivers of children diagnosed with WHS is unknown. This study aims to characterize the sociodemographic and psychosocial profile of WHS caregivers and analyze how these variables impact their quality of life (QoL) and well-being. Results: The sociodemographic and clinical profile of 22 Spanish caregivers of children with WHS and the characteristics of those affected have been described. Significant relationships were found between sociodemographic and psychosocial characteristics among caregivers. The impact on the parents’ QoL and negative relationship with the symptomatology were assessed. The use of engagement strategies such as problem focused coping was associated with improved psychological QoL and social support. Conclusions: WHS caregivers share similarities in their profile and needs with caregivers of children with other rare diseases. Pychosocial support groups involving parents caring for children with the same disease could improve caregivers’ well-being and QoL by strengthening their social support network and using positive coping styles.en
dc.description.sponsorshipThis research was supported by the Education Department of the Basque Government (BOPV, 18th August 2015), grant number: PRE_2015_1_0169 to Sarah Berrocosoen
dc.identifier.citationBerrocoso, S., Amayra, I., Lázaro, E., Martínez, O., López-Paz, J. F., García, M., Pérez, M., Al-Rashaida, M., Rodríguez, A. A., Luna, P. M., Pérez-Núñez, P., Blanco, R., & Nevado, J. (2020). Coping with Wolf-Hirschhorn syndrome: quality of life and psychosocial features of family carers. Orphanet Journal of Rare Diseases, 15(1). https://doi.org/10.1186/S13023-020-01476-8
dc.identifier.doi10.1186/S13023-020-01476-8
dc.identifier.eissn1750-1172
dc.identifier.urihttps://hdl.handle.net/20.500.14454/3848
dc.language.isoeng
dc.publisherBioMed Central Ltd
dc.rights© The Author(s). 2020
dc.subject.other4p deletion
dc.subject.otherCaregivers
dc.subject.otherCoping
dc.subject.otherDepression
dc.subject.otherQuality of life
dc.subject.otherSocial support
dc.subject.otherSpirituality
dc.subject.otherWolf-Hirschhorn syndrome
dc.titleCoping with Wolf-Hirschhorn syndrome: quality of life and psychosocial features of family carersen
dc.typejournal article
dcterms.accessRightsopen access
oaire.citation.issue1
oaire.citation.titleOrphanet Journal of Rare Diseases
oaire.citation.volume15
oaire.licenseConditionhttps://creativecommons.org/licenses/by/4.0/
oaire.versionVoR
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