Evidence map›Paper›PMID 39436578›Full record

ArticleQuality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation2025

Health-related quality of life in 205 children with arthrogryposis multiplex congenita.

Shahrzad Nematollahi, Emmanouil Rampakakis, Michael Amara, Reggie C Hamdy, Frank Rauch, Lauren C Hyer, Michelle A James, Haluk Altiok, Ellen Raney, Jonathan Pellett and 5 more

Abstract read
PubMed Publisher
In one paragraph

Article in Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing 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

3 citing papers in PubMed.

  1. Article
  2. Article
  3. Article
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

15 authors.

Shahrzad NematollahiSchool of Physical and Occupational Therapy, McGill University, Montreal, Québec, Canada. shahrzad.nematollahi@mail.mcgill.ca.ORCID http://orcid.org/0000-0003-1248-7306
Emmanouil RampakakisFaculty of Medicine and Health Sciences, McGill University, Montreal, Québec, Canada.
Michael AmaraSchool of Physical and Occupational Therapy, McGill University, Montreal, Québec, Canada.
Reggie C HamdyDepartment of Clinical research, Shriners Children's-Canada, Montreal, Québec, Canada.
Frank RauchDepartment of Clinical research, Shriners Children's-Canada, Montreal, Québec, Canada.
Lauren C HyerOrthopaedic Surgery, Shriners Children's Greenville, Greenville, SC, USA.
Michelle A JamesOrthopaedic Surgery, Shriners Children's Northern California, Sacramento, CA, USA.
Haluk AltiokOrthopaedic Surgery Shriners Children's Chicago, Illinois, IL, USA.
Ellen RaneyOrthopaedic Surgery, Shriners Children's Portland, Portland, OR, USA.
Jonathan PellettOrthopaedic Surgery, Shriners Children's Honolulu, Honolulu, HI, USA.
Cary MielkeOrthopedic Surgery, Shriners Children's Shreveport, Louisiana, LA, USA.
Sarah B NossovOrthopedic Surgery, Shriners Children's Philadelphia, Philadelphia, PA, USA.
Sena TavukcuDepartment of Clinical research, Shriners Children's-Canada, Montreal, Québec, Canada.
Philip F GiampietroDepartment of Pediatrics, Division of Medical Genetics, University of Illinois, Chicago, IL, USA.
Noémi Dahan-OlielSchool of Physical and Occupational Therapy, McGill University, Montreal, Québec, Canada.ORCID http://orcid.org/0000-0001-8567-7173

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

purposeArthrogryposis multiplex congenita (AMC) describes a heterogeneous group of rare congenital conditions. Health-related quality of life (HRQL) may be reduced in AMC due to broadly heterogeneous physical impairments and participation limitations. This study described HRQL in children and youth with AMC, compared HRQL between child self- and parent-proxy reports, and identified factors associated with better/worse HRQL.

methodsData on 205 children with AMC (age 8-21 years) from a North American AMC registry across eight hospital sites was used. HRQL was assessed cross-sectionally using the Patient Reported Outcome Measurement Information System (PROMIS) and European Quality of Life-5 Dimensions-Youth-3 Levels (EQ-5D-Y-3 L) by self-report, parent proxy-report or both.

resultsMean child-reported PROMIS T-scores were significantly lower than the normal mean for the Upper Extremity (mean = 33.0) and Mobility (mean = 37.2) but in the normal range for Pain Interference (mean = 46.6) and Peer Relationships (mean = 51.7). A lot of problems in EQ-5D-Y-3 L was reported by 37% in Feeling Worried/ Sad/ Unhappy, 46% in Having Pain/Discomfort, 50% in Doing Usual Activities, 56% in Mobility, and 57% in Looking After Myself. Compared to child-report, parents reported significantly worse PROMIS T-scores and higher problems in EQ-5D domains. Wheelchair use, being small for gestational age, prolonged hospitalization after birth, increased number of orthopedic surgeries, and caregiver's stress were associated with lower HRQL scores.

conclusionFindings indicate the importance of considering both the child's and parents' reports of HRQL, and to provide multimodal interventions that focus on the effect of childhood and parental characteristics to promote HRQL among children with AMC.

Indexed as

ArthrogryposisHealth StatusQuality of LifeAdolescentChildCross-Sectional StudiesFemaleHumansMaleParentsPatient Reported Outcome MeasuresSelf ReportSurveys and QuestionnairesYoung AdultArthrogryposis multiplex congenitaHealth-related quality of lifePatient reported outcome measuresRare diseasesRegistries

Identifiers

PMID39436578

What OpenQuestion holds

Textmetadata
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.