lnu.sePublications
Change search
CiteExportLink to record
Permanent link

Direct link
Cite
Citation style
  • apa
  • harvard1
  • ieee
  • modern-language-association-8th-edition
  • vancouver
  • Other style
More styles
Language
  • de-DE
  • en-GB
  • en-US
  • fi-FI
  • nn-NO
  • nn-NB
  • sv-SE
  • Other locale
More languages
Output format
  • html
  • text
  • asciidoc
  • rtf
Living with recessive limb-girdle muscular dystrophy: affected young adults’ and parents’ perspectives, studied througha salutogenic framework
Linnaeus University, Faculty of Health and Life Sciences, Department of Health and Caring Sciences.ORCID iD: 0000-0002-8241-1094
2017 (English)Doctoral thesis, comprehensive summary (Other academic)
Abstract [en]

Aim: The overall aim of this thesis, using a salutogenic framework, was to develop knowledge about experiences and perceptions of living with recessive limb-girdle muscular dystrophy and its influences on health, from the affected young adults’ and their parents’ perspectives.

Methods: A qualitative explorative and descriptive study design was used. Semi-structured interviews were held with 14 young adults diagnosed with recessive limb-girdle muscular dystrophy, aged 20–0 years, and 19 parents. Data analyses were conducted using content analysis (I, II, III) and phenomenography (IV). In order to mirror the interview data, the participants also answered the 13-item sense of coherence questionnaire.

Findings: Recessive limb-girdle muscular dystrophy has a major impact on the affected young adults’ and their parents’ lives as the disease progresses. Health described in terms of well-being was thus perceived to be influenced, not only by physical, emotional and social consequences due to the disease and worry about disease progression but also by external factors, such as accessibility to support provided by society and other people’s attitudes. There was, however, a determination among the participants to try to make the best of the situation. The importance of being able to mobilize internal resources, having social support, meaningful daily activities, adapted environment, the young adult being seen as a person and having support from concerned professionals, including personal assistance when needed, was thereby described. Self-rated sense of coherence scores varied. Those who scored above or the same as median among the young adults (≥56) and the parents (≥68) expressed greater extent satisfaction regarding social relations, daily activities and external support than those who scored less than median.

Conclusion: This thesis highlights the importance of early identification of personal perceptions and needs to enable timely health-promoting interventions. Through dialogue, not only support needed for the person to comprehend, manage and find meaning in everyday life can be identified, but also internal and external resources available to enhance health and well-being, taking into account the person’s social context as well as medical aspects.

Place, publisher, year, edition, pages
Växjö: Linnaeus University Press , 2017. , p. 74
Series
Linnaeus University Dissertations ; 296/2017
Keywords [en]
chronic diseases, LGMD2, muscular dystrophy, parents, salutogenic, sense of coherence, young adults
National Category
Medical and Health Sciences
Research subject
Health and Caring Sciences, Caring Science
Identifiers
URN: urn:nbn:se:lnu:diva-68737ISBN: 978-91-88357-89-2 (print)OAI: oai:DiVA.org:lnu-68737DiVA, id: diva2:1156973
Public defence
2017-11-10, Wicksell, Hus K, Växjö, 10:30
Opponent
Supervisors
Available from: 2017-11-16 Created: 2017-11-14 Last updated: 2018-01-16Bibliographically approved
List of papers
1. Health perceptions of young adults living with recessive limb-girdle muscular dystrophy
Open this publication in new window or tab >>Health perceptions of young adults living with recessive limb-girdle muscular dystrophy
2016 (English)In: Journal of Advanced Nursing, ISSN 0309-2402, E-ISSN 1365-2648, Vol. 72, no 8, p. 1915-1925Article in journal (Refereed) Published
Abstract [en]

AimThe aim of this study was to describe health perceptions related to sense of coherence among young adults living with recessive limb-girdle muscular dystrophy. BackgroundLimb-girdle muscular dystrophy refers to a group of progressive muscular disorders that may manifest in physical disability. The focus in health care is to optimize health, which requires knowledge about the content of health as described by the individual. DesignA descriptive study design with qualitative and quantitative data were used. MethodInterviews were conducted between June 2012-November 2013 with 14 participants aged 20-30years. The participants also answered the 13-item sense of coherence questionnaire. Qualitative data were analysed with content analysis and related to self-rated sense of coherence. FindingsHealth was viewed as intertwined physical and mental well-being. As the disease progressed, well-being was perceived to be influenced not only by physical impairment and mental strain caused by the disease but also by external factors, such as accessibility to support and attitudes in society. Factors perceived to promote health were having a balanced lifestyle, social relations and meaningful daily activities. Self-rated sense of coherence varied. The median score was 56 (range 37-77). Those who scored 56 described to a greater extent satisfaction regarding support received, daily pursuits and social life compared with those who scored <56. ConclusionCare should be person-centred. Caregivers, with their knowledge, should strive to assess how the person comprehends, manages and finds meaning in daily life. Through dialogue, not only physical, psychological and social needs but also health-promoting solutions can be highlighted.

Keywords
chronic diseases, disability, LGMD2, nursing, sense of coherence, SOC-13
National Category
Nursing
Research subject
Health and Caring Sciences
Identifiers
urn:nbn:se:lnu:diva-55771 (URN)10.1111/jan.12962 (DOI)000379932000017 ()27005675 (PubMedID)2-s2.0-85027936262 (Scopus ID)
External cooperation:
Available from: 2016-08-26 Created: 2016-08-26 Last updated: 2017-11-21Bibliographically approved
2. Young adults' experiences of living with recessive limb-girdle muscular dystrophy from a salutogenic orientation: an interview study
Open this publication in new window or tab >>Young adults' experiences of living with recessive limb-girdle muscular dystrophy from a salutogenic orientation: an interview study
2015 (English)In: Disability and Rehabilitation, ISSN 0963-8288, E-ISSN 1464-5165, Vol. 37, no 22, p. 2083-2091Article in journal (Refereed) Published
Abstract [en]

Purpose: To describe young adults' experiences of living with recessive limb-girdle muscular dystrophy (LGMD2) from a salutogenic orientation. Methods: A qualitative explorative interview study, including 14 participants aged 20-30 years, was performed focusing on comprehensibility, manageability and meaningfulness in daily life. Content analysis was used for data analysis. Result: Living with LGMD2 not only implies learning to live with the disease and the variations between good and bad periods but also means trying to make sense of a progressive disease that brings uncertainty about future health, by striving to make the best of the situation. Disease progression involves practical and mental struggle, trying to maintain control over one's life despite vanished physical functions that require continual adjustments to the body. Restrictions in a double sense were described, not only due to the disease but also due to poor comprehension of the disease in society. Lack of knowledge about LGMD2 among professionals often results in having to fight for the support needed. Conclusion: In order to manage daily life, it is important to be seen and understood as an individual in contacts with professionals and in society in general, to have informal social support and meaningful activities as well as access to personal assistance if necessary.Implications for RehabilitationRecessive limb-girdle muscular dystrophy (LGMD2) is a group of progressive disorders, which manifest in physical and psychological consequences for the individual.According to the salutogenic orientation, people need to find life comprehensible, manageable and meaningful, i.e. to achieve a sense of coherence (SOC), but living with LGMD2 may recurrently challenge the individual's SOC.Through the holistic view of the individual's situation that the salutogenic orientation provides, professionals may support the individual to strengthen SOC and thereby facilitate the movement towards health.

Keywords
Chronic diseases, LGMD2, muscular weakness, rare diseases
National Category
Nursing
Research subject
Health and Caring Sciences
Identifiers
urn:nbn:se:lnu:diva-51124 (URN)10.3109/09638288.2014.998782 (DOI)000369748600007 ()25582030 (PubMedID)2-s2.0-84942898801 (Scopus ID)
Available from: 2016-03-21 Created: 2016-03-21 Last updated: 2017-11-30Bibliographically approved
3. Experiences of being parents of young adults living with recessive limb-girdle muscular dystrophy from a salutogenic perspective
Open this publication in new window or tab >>Experiences of being parents of young adults living with recessive limb-girdle muscular dystrophy from a salutogenic perspective
2017 (English)In: Neuromuscular Disorders, ISSN 0960-8966, E-ISSN 1873-2364, Vol. 27, no 6, p. 585-595Article in journal (Refereed) Published
Abstract [en]

Recessive limb-girdle muscular dystrophies (LGMD2) involve progressive muscle weakness. Parental support is important for young adults living with LGMD2, but no study has been identified focusing on the parents' experiences. The salutogenic perspective concentrates on how daily life is comprehended, managed and found meaningful, i.e. the person's sense of coherence. The aim of this study was to describe, from a salutogenic perspective, experiences of being parents of young adults living with LGMD2. Nineteen participants were included. Data were collected by semi-structured interviews and the self-administrated 13-item sense of coherence questionnaire. Interview data were analysed with content analysis and related to self-rated sense of coherence. The result shows experiences of being influenced, not only by thoughts and emotions connected to the disease but also by caregiving duties and the young adults' well-being. Simultaneously, difficulty in fully grasping the disease was expressed and uncertainty about progression created worries about future management. Trying their best to support their young adults to experience well-being and to live fulfilled lives, the importance of having a social network, support from concerned professionals and eventually access to personal assistance was emphasized. The need to have meaningful pursuits of one's own was also described. The median sense of coherence score was 68 (range 53–86). Those who scored high (≥68) described satisfaction with social network, external support provided, work and leisure activities to a greater extent than those who scored low (<68). The result shows that the young adults' disease has a major impact on the parents' lives. Assessment of how the parents comprehend, manage and find meaning in everyday life may highlight support needed to promote their health.

Place, publisher, year, edition, pages
Elsevier, 2017
Keywords
Limb-girdle muscular dystrophy, Parents, Sense of coherence
National Category
Neurology
Research subject
Health and Caring Sciences
Identifiers
urn:nbn:se:lnu:diva-64640 (URN)10.1016/j.nmd.2017.01.024 (DOI)000405881300013 ()28314630 (PubMedID)2-s2.0-85019188869 (Scopus ID)
Available from: 2017-06-02 Created: 2017-06-02 Last updated: 2017-11-14Bibliographically approved

Open Access in DiVA

Living with muscular dystrophy(3864 kB)105 downloads
File information
File name FULLTEXT01.pdfFile size 3864 kBChecksum SHA-512
56543a0d7f784e4cc35542b96fbf2b92d1e8b34ec2a3a22674e1ee51d6c51fa7d03993a19e6ae37a52b6307bd236dc76e76d2de1ef9dadc498069bd9572789c7
Type fulltextMimetype application/pdf

Other links

Buy Book (SEK 160 + VAT and postage) lnupress@lnu.se

Search in DiVA

By author/editor
Aho, Anna Carin
By organisation
Department of Health and Caring Sciences
Medical and Health Sciences

Search outside of DiVA

GoogleGoogle Scholar
Total: 105 downloads
The number of downloads is the sum of all downloads of full texts. It may include eg previous versions that are now no longer available

isbn
urn-nbn

Altmetric score

isbn
urn-nbn
Total: 192 hits
CiteExportLink to record
Permanent link

Direct link
Cite
Citation style
  • apa
  • harvard1
  • ieee
  • modern-language-association-8th-edition
  • vancouver
  • Other style
More styles
Language
  • de-DE
  • en-GB
  • en-US
  • fi-FI
  • nn-NO
  • nn-NB
  • sv-SE
  • Other locale
More languages
Output format
  • html
  • text
  • asciidoc
  • rtf