Evidence map›Paper›PMID 37288425›Full record

ArticleFrontiers in oral health2023

Case report: a giant cell-rich gnathic bone lesion in a child with pycnodysostosis.

C Spencer, A Makka, S Singh, J McGuire, N Washaya, G Hein, M Zampoli, K Fieggen

Open access · goldAbstract readCase Reports
PubMed Full text at PMC Publisher
In one paragraph

Article in Frontiers in oral health, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
0.2field-weighted citation impact, top 42% 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

1 citing paper in PubMed, 1 citations in OpenAlex.

  1. 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

8 authors at 4 institutions in 2 countries.

C SpencerDivision of Human Genetics, University of Cape Town, Cape Town, South Africa.
A MakkaDepartment of Maxillo-Facial and Oral Surgery, University of the Western Cape, Cape Town, South Africa.
S SinghDivision of Anatomical Pathology, University of Cape Town and National Health Laboratory Services, Cape Town, South Africa.
J McGuireDivision of Otolaryngology, University of Cape Town, Cape Town, South Africa.
N WashayaDivision of Paediatric Pulmonology, Department of Paediatrics and Child Health, University of Cape Town, Cape Town, South Africa.
G HeinDepartment of Maxillo-Facial and Oral Surgery, University of the Western Cape, Cape Town, South Africa.
M ZampoliDivision of Paediatric Pulmonology, Department of Paediatrics and Child Health, University of Cape Town, Cape Town, South Africa.
K FieggenDivision of Human Genetics, University of Cape Town, Cape Town, South Africa.
University of Cape Town · ZAUniversity of the Western Cape · ZAGroote Schuur Hospital · ZANational Health Laboratory Service · ZA

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Pycnodysostosis is a skeletal dysplasia characterized by short stature, generalized osteosclerosis, acro-osteolysis, and recognizable facial features. Oral manifestations are commonly described and include a high-arched palate with dental crowding and malocclusion, hypoplastic enamel, and retained deciduous teeth with impacted permanent teeth, and there is an increased risk of developing osteomyelitis of the jaw. We report here the history of a 9-year-old male with the typical facial and skeletal phenotype of pycnodysostosis but novel oral features. He presented with bilateral progressive facial swelling, which caused functional impairment with chewing and contributed to his severe obstructive sleep apnea (OSA). The severity of his OSA required surgical intervention, and the lesions were resected. Extensive bone remodeling and replacement by fibrous tissue were noted on submucosal dissection, and bilateral subtotal maxillectomies were required. The histopathology of the biopsied lesion was consistent with a giant cell-rich lesion. Genetic testing identified a pathogenic homozygous variant in the

Indexed as

case reportgiant cell-richgnathic bonesobstructive sleep apneapycnodysostosis

Identifiers

PMID37288425
PMCPMC10242096
OpenAlexW4378084874

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.