Evidence map›Paper›PMID 27927250›Full record

ArticleOrphanet journal of rare diseases2016

Experiences of caregivers of children with inherited metabolic diseases: a qualitative study.

Shabnaz Siddiq, Brenda J Wilson, Ian D Graham, Monica Lamoureux, Sara D Khangura, Kylie Tingley, Laure Tessier, Pranesh Chakraborty, Doug Coyle, Sarah Dyack and 20 more

Abstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Orphanet journal of rare diseases, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 38 papers.

0numbers the graph read from it
0cells of the map it votes in
38citing papers in PubMed
–field-weighted citation impact
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

38 citing papers in PubMed.

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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

30 authors.

Shabnaz SiddiqFaculty of Medicine, School of Epidemiology, Public Health and Preventive Medicine, University of Ottawa, 451 Smyth Road, Ottawa, ON, K1H 8M5, Canada.
Brenda J WilsonFaculty of Medicine, School of Epidemiology, Public Health and Preventive Medicine, University of Ottawa, 451 Smyth Road, Ottawa, ON, K1H 8M5, Canada.
Ian D GrahamFaculty of Medicine, School of Epidemiology, Public Health and Preventive Medicine, University of Ottawa, 451 Smyth Road, Ottawa, ON, K1H 8M5, Canada.
Monica LamoureuxNewborn Screening Ontario, Children's Hospital of Eastern Ontario (CHEO), Ottawa, ON, Canada.
Sara D KhanguraFaculty of Medicine, School of Epidemiology, Public Health and Preventive Medicine, University of Ottawa, 451 Smyth Road, Ottawa, ON, K1H 8M5, Canada.
Kylie TingleyFaculty of Medicine, School of Epidemiology, Public Health and Preventive Medicine, University of Ottawa, 451 Smyth Road, Ottawa, ON, K1H 8M5, Canada.
Laure TessierNewborn Screening Ontario, Children's Hospital of Eastern Ontario (CHEO), Ottawa, ON, Canada.
Pranesh ChakrabortyNewborn Screening Ontario, Children's Hospital of Eastern Ontario (CHEO), Ottawa, ON, Canada.
Doug CoyleFaculty of Medicine, School of Epidemiology, Public Health and Preventive Medicine, University of Ottawa, 451 Smyth Road, Ottawa, ON, K1H 8M5, Canada.
Sarah DyackDepartment of Pediatrics, Dalhousie University and IWK Health Centre, Halifax, NS, Canada.
Jane GillisUniversity of British Columbia, BC Children's Hospital, Vancouver, BC, Canada.
Cheryl GreenbergDepartment of Pediatrics and Child Health, University of Manitoba and Children's Hospital Research Institute of Manitoba, Winnipeg, MB, Canada.
Robin Z HayeemsInstitute of Health Policy Management and Evaluation, University of Toronto, Toronto, ON, Canada.
Shailly Jain-GhaiUniversity of Alberta, Stollery Children's Hospital, Edmonton, AB, Canada.
Jonathan B KronickDivision of Clinical and Metabolic Genetics, University of Toronto and The Hospital for Sick Children, Toronto, ON, Canada.
Anne-Marie LabergeMedical Genetics, Department of Pediatrics, CHU Sainte-Justine, Montréal, QC, Canada.
Julian LittleFaculty of Medicine, School of Epidemiology, Public Health and Preventive Medicine, University of Ottawa, 451 Smyth Road, Ottawa, ON, K1H 8M5, Canada.
John J MitchellMcGill University Health Centre, The Montreal Children's Hospital, Montréal, QC, Canada.
Chitra PrasadGenetics, Metabolism and Paediatrics, London Health Sciences Centre, Western University, London, ON, Canada.
Komudi SiriwardenaUniversity of Alberta, Stollery Children's Hospital, Edmonton, AB, Canada.
Rebecca SparkesUniversity of Calgary, Alberta Children's Hospital, Calgary, AB, Canada.
Kathy N SpeechleyDepartment of Paediatrics, Western University, London, ON, Canada.
Sylvia StocklerUniversity of British Columbia, BC Children's Hospital, Vancouver, BC, Canada.
Yannis TrakadisMcGill University Health Centre, The Montreal Children's Hospital, Montréal, QC, Canada.
Sarah WafaMcGill University Health Centre, The Montreal Children's Hospital, Montréal, QC, Canada.
Jagdeep WaliaQueen's University, Kingston General Hospital, Kingston, ON, Canada.
Kumanan WilsonFaculty of Medicine, School of Epidemiology, Public Health and Preventive Medicine, University of Ottawa, 451 Smyth Road, Ottawa, ON, K1H 8M5, Canada.
Nataliya YuskivUniversity of British Columbia, BC Children's Hospital, Vancouver, BC, Canada.
Beth K PotterFaculty of Medicine, School of Epidemiology, Public Health and Preventive Medicine, University of Ottawa, 451 Smyth Road, Ottawa, ON, K1H 8M5, Canada. bpotter@uottawa.ca.
Canadian Inherited Metabolic Diseases Research Network (CIMDRN)

Funding

CIHR TR3-119195
6 · The paper itself

Abstract

backgroundWe sought to understand the experiences of parents/caregivers of children with inherited metabolic diseases (IMD) in order to inform strategies for supporting patients and their families. We investigated their experiences regarding the management of disease, its impact on child and family life, and interactions with the health care system.

methodsFrom four Canadian centres, we conducted semi-structured telephone interviews with parents/caregivers of children with an IMD who were born between 2006 and 2015 and who were participating in a larger cohort study. Participants were selected with the aim of achieving a diverse sample with respect to treatment centre, IMD, and age of the child. Interviews emphasized the impacts of the disease and its treatment on the child and family and explicitly queried perceptions of interactions with the health care system. We identified emergent themes from the interview data.

resultsWe completed interviews with 21 parents/caregivers. The 21 children were aged <1 to 7 years old with IMD that included amino acid disorders, urea cycle disorders, fatty acid oxidation disorders, and organic acid disorders or 'other' IMD. Most parents reported that they and their families had adapted well to their child's diagnosis. Parents used proactive coping strategies to integrate complex disease management protocols into routine family life. An important source of stress was concern about the social challenges faced by their children. Participants reported positive interactions with their most involved health care providers within the metabolic clinic. However, they reported challenges associated with the health care system outside of disease-specific metabolic care, when encountering systems and providers unfamiliar with the child's disease.

conclusionsThe successful use of proactive coping strategies among parents of children with IMD in this study suggests the potential value of promoting positive coping and is an important direction for future study. Parents' social concerns for their children were important stressors that warrant consideration by health care providers positioned to support families. Our results with respect to experiences with care highlight the important role of specialized metabolic clinics and point to a need for better coordination of the care that takes place outside the disease-specific management of IMD.

Indexed as

CaregiversMetabolism, Inborn ErrorsAdaptation, PsychologicalChildFamily HealthHome Care ServicesHumansStress, PsychologicalCopingExperiences with careFamily supportInherited metabolic diseasesQualitative research

Identifiers

PMID27927250
PMCPMC5142351

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.