Evidence map›Paper›PMID 38515174›Full record

ArticleOrphanet journal of rare diseases2024

Exploring the support needs of Australian parents of young children with Usher syndrome: a qualitative thematic analysis.

L Johansen, F O'Hare, E R Shepard, L N Ayton, L J Pelentsov, L S Kearns, K L Galvin

Erratum issuedOpen access · goldAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Orphanet journal of rare diseases, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 5 papers.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed
3.3field-weighted citation impact, top 8% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

5 citing papers in PubMed, 9 citations in OpenAlex.

  1. Article
  2. Article
  3. Review
  4. Deaf/hard of hearing ecological assessment form-child (DEAF-C).Journal of deaf studies and deaf education · 2025
    Article
  5. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

7 authors at 3 institutions in 1 country.

L JohansenUsherKids Australia, Mordialloc, VIC, Australia.ORCID 0000-0003-1180-0428
F O'HareDepartment of Optometry and Vision Sciences, The University of Melbourne, Parkville, VIC, Australia.ORCID 0000-0002-0022-0527
E R ShepardUsherKids Australia, Mordialloc, VIC, Australia.ORCID 0000-0003-1602-4018
L N AytonDepartment of Optometry and Vision Sciences, The University of Melbourne, Parkville, VIC, Australia.ORCID 0000-0001-9907-084X
L J PelentsovClinical and Health Sciences, University of South Australia, Adelaide, SA, Australia.ORCID 0000-0001-8108-491X
L S KearnsCentre for Eye Research Australia, East Melbourne, VIC, Australia.ORCID 0000-0001-9432-4054
K L GalvinDepartment of Audiology and Speech Pathology, Faculty of Medicine, Dentistry and Health Sciences, The University of Melbourne, Parkville, VIC, Australia. kgalvin@unimelb.edu.au.ORCID 0000-0001-8095-8718
The University of Melbourne · AUThe Royal Victorian Eye & Ear Hospital · AUUniversity of South Australia · AU

Funding

National Health and Medical Research Council 1195713
6 · The paper itself

Abstract

backgroundAdvancements in genetic testing have led to Usher syndrome now being diagnosed at a much earlier age than in the past, enabling the provision of early intervention and support to children and families. Despite these developments, anecdotal reports suggest there are substantial gaps in the services and supports provided to parents of children with Usher syndrome. The current study investigated the support needs of parents of children with Usher syndrome Type 1 when their child was aged 0 to 5 years.

methodPurposive sampling was used, and six semi-structured interviews were conducted with Australian parents of children with Usher syndrome, Type 1. Data was analysed using modified reflexive thematic analysis.

resultsFour key themes were identified as being central to the support needs of parents of children with Usher syndrome aged 0 to 5 years. (1) Social Needs referred to parents' need for various sources of social support, (2) Informational Needs described the lack of information parents received regarding Usher syndrome from treating professionals, (3) Practical Needs included supports needed to assist parents in managing the day-to-day tasks of caring for a child with a disability, and (4) Emotional Needs represented the emotional support (both formal and informal) that parents needed to be a positive support to their child.

conclusionsFindings provide rich information for relevant support groups, policy makers, individual healthcare professionals, and professional governing bodies regarding the education of stakeholders and the development and implementation of best-practice treatment guidelines.

Indexed as

Usher SyndromesAustraliaChildChild, PreschoolHealth PersonnelHumansParentsQualitative ResearchSocial SupportDisease burdenInterview studyParentsRare diseaseSupport needsUsher syndrome

Identifiers

PMID38515174
PMCPMC10956185
OpenAlexW4393045268

What OpenQuestion holds

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LicenceCC BY
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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.