Evidence map›Paper›PMID 33446226›Full record

ArticleOrphanet journal of rare diseases2021

Best practice guidelines in managing the craniofacial aspects of skeletal dysplasia.

Ravi Savarirayan, David E Tunkel, Laura M Sterni, Michael B Bober, Tae-Joon Cho, Michael J Goldberg, Julie Hoover-Fong, Melita Irving, Shawn E Kamps, William G Mackenzie and 5 more

Abstract read
In one paragraph

Article in Orphanet journal of rare diseases, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
11citing papers in PubMed, 1 pooled it
–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

11 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Guideline
  2. Diagnosis, treatment, and management recommendations for cleidocranial dysplasia: A modified Delphi panel.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2026
    Article
  3. Article
  4. Article
  5. Article
  6. Article
  7. Review
  8. Article
  9. Review
  10. Hearing loss in Norwegian adults with achondroplasia.Orphanet journal of rare diseases · 2021
    Observational
  11. Changes in skeletal dysplasia nosology.Romanian journal of morphology and embryology = Revue roumaine de morphologie et embryologie
    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.

Ravi SavarirayanVictorian Clinical Genetics Services, Murdoch Children's Research Institute, University of Melbourne, Parkville, VIC, 3052, Australia. ravi.savarirayan@vcgs.org.au.ORCID 0000-0002-5105-8427
David E TunkelDepartment of Otolaryngology-Head and Neck Surgery, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
Laura M SterniEudowwod Division of Pediatric Respiratory Sciences, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
Michael B BoberDivision of Orthogenetics - Nemours/ A.I. duPont Hospital for Children, Wilmington, DE, USA.
Tae-Joon ChoDivision of Pediatric Orthopaedics, Seoul National University Children's Hospital, Seoul, South Korea.
Michael J GoldbergDepartment of Orthopedics and Sports Medicine, Seattle Children's Hospital, Department of Orthopaedics and Sports Medicine, University of Washington, Seattle, WA, USA.
Julie Hoover-FongMcKusick-Nathans Department of Genetic Medicine, Johns Hopkins University, Baltimore, MD, USA.
Melita IrvingDepartment of Clinical Genetics Guy's, St Thomas NHS, London, UK.
Shawn E KampsDepartment of Orthopedics and Sports Medicine, Seattle Children's Hospital, Department of Orthopaedics and Sports Medicine, University of Washington, Seattle, WA, USA.
William G MackenzieDepartment of Orthopedic Surgery - Nemours/ A.I. duPont Hospital for Children, Wilmington, DE, USA.
Cathleen RaggioDepartment of Orthopedic Surgery, Hospital for Special Surgery, New York, NY, USA.
Samantha A SpencerDepartment of Orthopedic Surgery, Boston Children's Hospital, Boston, MA, USA.
Viviana BompadreDepartment of Orthopedics and Sports Medicine, Seattle Children's Hospital, Department of Orthopaedics and Sports Medicine, University of Washington, Seattle, WA, USA.
Klane K WhiteDepartment of Orthopedics and Sports Medicine, Seattle Children's Hospital, Department of Orthopaedics and Sports Medicine, University of Washington, Seattle, WA, USA.
on behalfof the Skeletal Dysplasia Management Consortium

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundRecognition and appropriate management of the craniofacial manifestations of patients with skeletal dysplasia are challenging, due to the rarity of these conditions, and dearth of literature to support evidence-based clinical decision making.

methodsUsing the Delphi method, an international, multi-disciplinary group of individuals, with significant experience in the care of patients with skeletal dysplasia, convened to develop multi-disciplinary, best practice guidelines in the management of craniofacial aspects of these patients.

resultsAfter a comprehensive literature review, 23 initial statements were generated and critically discussed, with subsequent development of a list of 22 best practice guidelines after a second round voting.

conclusionsThe guidelines are presented and discussed to provide context and assistance for clinicians in their decision making in this important and challenging component of care for patients with skeletal dysplasia, in order standardize care and improve outcomes.

Indexed as

OsteochondrodysplasiasHumansBest practiceCraniofacialOtolaryngologySkeletal dysplasia

Identifiers

PMID33446226
PMCPMC7809733

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.