Evidence map›Paper›PMID 40087658›Full record

ArticleOrphanet journal of rare diseases2025

Caregiver burden and familial impact in Down Syndrome Regression Disorder.

Katherine Chow, Panteha Hayati Rezvan, Lilia Kazerooni, Lina Nguyen, Natalie K Boyd, Benjamin N Vogel, Maeve C Lucas, Ruth Brown, Eileen A Quinn, Saba Jafarpour and 1 more

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PubMed Full text at PMC Publisher
In one paragraph

Article in Orphanet journal of rare diseases, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing 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

6 citing papers in PubMed.

  1. Observational
  2. Article
  3. Article
  4. Article
  5. Article
  6. Review
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

11 authors.

Katherine ChowKeck School of Medicine of the University of Southern California, Los Angeles, CA, USA.
Panteha Hayati RezvanBiostatistics and Data Management Core, The Saban Research Institute, Children's Hospital Los Angeles, Los Angeles, CA, USA.
Lilia KazerooniDivision of Neurology, Department of Pediatrics, Children's Hospital Los Angeles, Los Angeles, CA, USA.
Lina NguyenDivision of Neurology, Department of Pediatrics, Children's Hospital Los Angeles, Los Angeles, CA, USA.
Natalie K BoydDivision of Neurology, Department of Pediatrics, Children's Hospital Los Angeles, Los Angeles, CA, USA.
Benjamin N VogelDivision of Neurology, Department of Pediatrics, Children's Hospital Los Angeles, Los Angeles, CA, USA.
Maeve C LucasDivision of Neurology, Department of Pediatrics, Children's Hospital Los Angeles, Los Angeles, CA, USA.
Ruth BrownVirginia Institute for Psychiatric and Behavioral Genetics at Virginia Commonwealth University, Richmond, VA, USA.
Eileen A QuinnDivision of Developmental and Behavioral Pediatrics, University of Toledo College of Medicine and Life Sciences, Toledo, OH, USA.
Saba JafarpourDivision of Neurology, Department of Pediatrics, Children's Hospital Los Angeles, Los Angeles, CA, USA.
Jonathan D SantoroDivision of Neurology, Department of Pediatrics, Children's Hospital Los Angeles, Los Angeles, CA, USA. jdsantoro@chla.usc.edu.ORCID http://orcid.org/0000-0002-8350-8234

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundDown Syndrome Regression Disorder (DSRD) is an acute or subacute neurocognitive regression in individuals with Down syndrome (DS), characterized by a loss of previously acquired cognitive, adaptive, and social skills. DSRD profoundly affects individuals' ability to engage in activities of daily living, making them highly dependent on their caregivers who must provide significantly more support than before the DSRD diagnosis. This study aimed to examine caregiver burden, quality of life, and depression among caregivers of individuals with DSRD versus caregivers of those with DS and other neurological disorders (DSN). DESIGN/

methodsIn this cross-sectional study, caregivers of individuals with DSRD (n = 228) and DSN (n = 137) were recruited through Children's Hospital Los Angeles neurology clinic and a Facebook DSRD support group. Participants completed standardized questionnaires assessing quality of life (PedsQL Family Impact Module), caregiver burden (Zarit Caregiver Burden Assessment, ZCB), and depression (Glasgow Depression Scale, GDS), along with additional items addressing other factors of caregiver well-being. Data were analyzed using bivariate comparisons and univariate regression models to assess differences between groups.

resultsCaregivers of individuals with DSRD were more likely than caregivers of those with DSN to report increased financial burden (p = 0.003), housing changes (p = 0.02), disrupted sleep (p < 0.001), negative impacts on social networks (p < 0.001), and worsened mental health (p < 0.001). Furthermore, DSRD caregivers reported significantly higher levels of burden (mean difference [95% CI]: 8.3 [6.3, 9.7]) and depression symptoms (2 [0.7, 3.4]), reflecting greater perceived stress and burden. They also had lower quality of life scores (-27.9 [-30.2, -25.5]), indicating a more substantial impact on overall well-being and daily functioning compared to DSN caregivers. Additionally, caregivers in the DSRD group had higher odds (odds ratio [95% CI]: 4.7 [2.9, 7.7)] of meeting clinical depression criteria (GDS score ≥ 13) than caregivers in the DSN group.

conclusionsCaregivers of individuals with DSRD experience significantly greater distress and burden compared to those caring for individuals with DSN. The elevated risk of depression, combined with reduced quality of life and increased burden, underscores the multimodal nature of the challenges faced by this population.

Indexed as

CaregiversDown SyndromeAdolescentAdultChildChild, PreschoolCost of IllnessCross-Sectional StudiesDepressionFemaleHumansMaleMiddle AgedQuality of LifeSurveys and QuestionnairesYoung AdultBurdenCaregiverDepressionDown syndromeNeurologicQuality of lifeRegression

Identifiers

PMID40087658
PMCPMC11909950

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.