Evidence map›Paper›PMID 35196159›Full record

ArticleJournal of child neurology2022

The Lived Experience of Parents' Receiving the Diagnosis of CDKL5 Deficiency Disorder for Their Child.

Scott Demarest, Rebekah Marsh, Lauren Treat, Michael P Fisher, Amanda Dempsey, Mohammed Junaid, Jenny Downs, Helen Leonard, Tim Benke, Megan A Morris

Open access · greenAbstract read
In one paragraph

Article in Journal of child neurology, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.

0numbers the graph read from it
0cells of the map it votes in
9citing papers in PubMed
2.4field-weighted citation impact, top 11% 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

9 citing papers in PubMed, 14 citations in OpenAlex.

  1. Article
  2. Development of a patient-centered conceptual disease model in Ring 14 syndrome: a patient-centered model of lived experience.Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation · 2026
    Article
  3. Article
  4. Article
  5. Identifying the Impacts, Obstacles and Information Barriers for Parents of Children Living With Genetic Neurodevelopmental Disorders: A Qualitative Study.Health expectations : an international journal of public participation in health care and health policy · 2025
    Observational
  6. Article
  7. Article
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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

10 authors at 4 institutions in 2 countries.

Scott DemarestAdult and Child Consortium for Health Outcomes Research Science, University of Colorado.ORCID 0000-0002-4544-3724
Rebekah MarshAdult and Child Consortium for Health Outcomes Research Science, University of Colorado.
Lauren TreatDepartment of Pediatrics, University of Colorado.ORCID 0000-0002-1846-9558
Michael P FisherDepartment of Health Sciences, 1492Towson University, Towson, MD, USA.
Amanda DempseyAdult and Child Consortium for Health Outcomes Research Science, University of Colorado.
Mohammed Junaid117610Telethon Kids Institute, Nedlands, Western Australia, Australia.
Jenny Downs117610Telethon Kids Institute, Nedlands, Western Australia, Australia.ORCID 0000-0001-7358-9037
Helen Leonard117610Telethon Kids Institute, Nedlands, Western Australia, Australia.
Tim BenkeAdult and Child Consortium for Health Outcomes Research Science, University of Colorado.
Megan A MorrisAdult and Child Consortium for Health Outcomes Research Science, University of Colorado.
Outcomes Research Consortium · USThe Kids Research Institute Australia · AUChildren's Hospital Colorado · USTowson University · US

Funding

Multi-Site Validation of Biomarkers and Core Clinical Outcome Measures for Clinical Trials Readiness in CDKL5 Deficiency DisorderU01NS114312 · NINDS · UNIVERSITY OF COLORADO DENVER · PI BENKE, TIMOTHY A, DOWNS, JENNY · 2021 to 2025
$4.7M
Colorado Neurological Sciences Academic Development Award (NSADA)K12NS089417 · NINDS · UNIVERSITY OF COLORADO DENVER · PI BENKE, TIMOTHY A, BROOKS-KAYAL, AMY R. · 2015 to 2019
$1.3M
NINDS NIH HHS U01 NS114312
6 · The paper itself

Abstract

CDKL5 deficiency disorder (CDD), a severe developmental and epileptic encephalopathy, is being diagnosed earlier with improved access to genetic testing, but this may also have unanticipated impacts on parents' experience receiving the diagnosis. This study explores the lived experience of parents receiving a diagnosis of CDD for their child using mixed methods. Thirty-seven semistructured interviews were conducted with parents of children with a diagnosis of CDD, which were coded and analyzed to identify themes. Grief was a nearly universal theme expressed among participants. Parents of younger children discussed grief in the context of receiving the diagnosis, whereas parents of older children indicated they were at different stages along the grieving journey when they received the diagnosis. Parents with less understanding of their child's prognosis (poorer prognostic awareness) connected their grief to receiving the diagnosis as this brought a clear understanding of the prognosis. Several themes suggested what providers did well to improve the diagnostic experience for parents, much of which aligns with existing literature around how to provide serious news. Additionally, parents identified long-term benefits of having a diagnosis for their child's medical problems. Although interview data were concordant with a survey of parents' diagnostic experience from a large international cohort, most participants in this study were relatively affluent, white mothers and further research is needed to better understand if other groups of parents have a different diagnostic experience. This study gives context of parental experience that providers should be aware of when conveying new genetic diagnoses to families.

Indexed as

Epileptic SyndromesSpasms, InfantileAdolescentChildFamilyHumansParentsProtein Serine-Threonine KinasesCDKL5 protein, humanProtein Serine-Threonine Kinaseschildrenepileptic encephalopathygeneticsneurodevelopmentnext-generation sequencingquality of liferisk factors

Identifiers

PMID35196159
PMCPMC10231496
OpenAlexW4213456881

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

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.