Evidence map›Paper›PMID 24663682›Full record

ArticleJournal of dental research2014

Dental Anomalies Associated with Craniometaphyseal Dysplasia.

I-P Chen, A Tadinada, E H Dutra, A Utreja, F Uribe, E J Reichenberger

Registry-linked trialAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Journal of dental research, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT01630460 (Identification of Mutations That Lead to Craniometaphyseal Dysplasia in Families and Isolated Cases and Studies of Cellular and Molecular Mechanisms), which is not on this map. Cited by 11 papers.

0numbers the graph read from it
0cells of the map it votes in
11citing 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.

NCT01630460 recruitingnot on this map

Identification of Mutations That Lead to Craniometaphyseal Dysplasia in Families and Isolated Cases and Studies of Cellular and Molecular Mechanisms

TypeobservationalSponsorUConn HealthRan2009 to 2030Enrolled600ConditionsCraniometaphyseal Dysplasia
3 · Its place in the literature

Who cites it

11 citing papers in PubMed.

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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

6 authors.

I-P ChenDepartment of Oral Health and Diagnostic Sciences, School of Dental Medicine, University of Connecticut Health Center, Farmington, CT, USA.
A TadinadaDepartment of Oral Health and Diagnostic Sciences, School of Dental Medicine, University of Connecticut Health Center, Farmington, CT, USA.
E H DutraDepartment of Craniofacial Sciences, School of Dental Medicine, University of Connecticut Health Center, Farmington, CT, USA.
A UtrejaDepartment of Craniofacial Sciences, School of Dental Medicine, University of Connecticut Health Center, Farmington, CT, USA.
F UribeDepartment of Craniofacial Sciences, School of Dental Medicine, University of Connecticut Health Center, Farmington, CT, USA.
E J ReichenbergerDepartment of Reconstructive Sciences, Center for Regenerative Medicine and Skeletal Development, University of Connecticut Health Center, Farmington, CT, USA reichenberger@uchc.edu.

Funding

ZONISAMIDE VERSUS PLACEBOM01RR006192 · NCRR · UNIVERSITY OF CONNECTICUT SCH OF MED/DNT · PI DRAZINIC, CAROLYN M · 1994 to 2008
$22.1M
SKELETAL, CRANOFACIAL AND ORAL BIOLOGY TRAIINING GRANTT32DE007302 · NIDCR · UNIVERSITY OF CONNECTICUT SCH OF MED/DNT · PI LURIE, ALAN GORDON · 1996 to 2010
$6.1M
Control of Bone Formation in Craniometaphyseal DysplasiaR01DE019458 · NIDCR · UNIVERSITY OF CONNECTICUT SCH OF MED/DNT · PI REICHENBERGER, ERNST J · 2011 to 2015
$1.8M
NCRR NIH HHS M01 RR006192NCRR NIH HHS M01RR006192NIDCR NIH HHS R01 DE019458NIDCR NIH HHS R01-DE019458NIDCR NIH HHS T32 DE007302NIDCR NIH HHS T32-DE007302
6 · The paper itself

Abstract

Craniometaphyseal dysplasia (CMD) is a rare genetic disorder encompassing hyperostosis of craniofacial bones and metaphyseal widening of tubular bones. Dental abnormalities are features of CMD that have been little discussed in the literature. We performed dentofacial examination of patients with CMD and evaluated consequences of orthodontic movement in a mouse model carrying a CMD knock-in (KI) mutation (Phe377del) in the Ank gene. All patients have a history of delayed eruption of permanent teeth. Analysis of data obtained by cone-beam computed tomography showed significant bucco-lingual expansion of jawbones, more pronounced in mandibles than in maxillae. There was no measurable increase in bone density compared with that in unaffected individuals. Orthodontic cephalometric analysis showed that patients with CMD tend to have a short anterior cranial base, short upper facial height, and short maxillary length. Microcomputed tomography (micro-CT) analysis in homozygous Ank (KI/KI) mice, a model for CMD, showed that molars can be moved by orthodontic force without ankylosis, however, at a slower rate compared with those in wild-type Ank (+/+) mice (p < .05). Histological analysis of molars in Ank (KI/KI) mice revealed decreased numbers of TRAP(+) osteoclasts on the bone surface of pressure sides. Based on these findings, recommendations for the dental treatment of patients with CMD are provided.

Indexed as

Acid PhosphataseAnimalsBone DensityBone Diseases, DevelopmentalCephalometryCone-Beam Computed TomographyCraniofacial AbnormalitiesDisease Models, AnimalGene Knock-In TechniquesHumansHyperostosisHypertelorismIsoenzymesMandibleMaxillaMiceAcid PhosphataseAcp5 protein, mouseank protein, mouseIsoenzymesPhenylalaninePhosphate Transport ProteinsTartrate-Resistant Acid PhosphataseANKHcone-beam computed tomographycraniofacial abnormalitiesmouse modelorthodonticstooth eruption

Identifiers

PMID24663682
PMCPMC4023465

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Registered trials

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.