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Dental and oral anomalies in incontinentia pigmenti: a systematic review
Abstract
Objectives Incontinentia pigmenti (IP) is an X-linked genodermatosis caused by a mutation of the IKBKG gene. The objective of this study was to present a systematic review of the dental and oral types of anomalies, to determine the total number and sex distribution of the anomalies, and to analyze possible therapies. Materials and methods We analyzed the literature data from 1,286 IP cases from the period 1993-2010. Results Dental and/or oral anomalies were diagnosed for 54.38% of the investigated IP patients. Most of the anomaly types were dental, and the most frequent of these were dental shape anomalies, hypodontia, and delayed dentition. The most frequent oral anomaly types were cleft palate and high arched palate. IKBKG exon 4-10 deletion was present in 86.36% of genetically confirmed IP patients. Conclusions According to the frequency, dental and/or oral anomalies represent the most frequent and important IP minor criteria. The most frequent mutation was IKBKG exon 4-10 deletion. The majority of dental anomalies and some of the oral anomalies could be corrected. Clinical relevance Because of the presence of cleft palate and high arched palate in IP patients, these two anomalies may be considered as diagnostic IP minor criteria as well.
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Dental and oral anomalies like delayed dentition, pegged or conical teeth, anodontia or hypodontia, and cleft or high-arched palate occur in almost all patients with incontinentia pigmenti.
“Dental and oral anomalies like delayed dentition, pegged or conical teeth, anodontia or hypodontia, and cleft or high-arched palate occur in almost all patients.[32]”
From Ectodermal Dysplasia – An Overview and Update (2024) · cites this paper as [32]
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Metadata
- Authors
- Minić, S., Trpinac, D., Gabriel, H., Gencik, M., & Obradović, M.
- Affiliations
- University of Belgrade (RS)
- Corresponding author
- Minić, Snežana
- Venue
- Clinical Oral Investigations
- Published
- 2012-03-28
- Volume
- 17
- Issue
- 1
- Pages
- 1-8
- ISSN
- 1432-6981 (ISSN-L)
- 1436-3771
- OpenAlex
- W2116728509
- Cited by
- 64
- References
- 0
- Citations by year
- 2026: 3 · 2025: 2 · 2024: 5 · 2023: 3 · 2022: 6 · 2021: 3 · 2020: 2 · 2019: 3 · 2018: 4 · 2017: 9 · 2016: 4 · 2015: 9 · 2014: 6 · 2013: 4
- FWCI
- 0.74
- Type
- journal article
- Language
- en
- Open access
- closed
- Licence
- www.springer.com/tdm (tdm)
- Locations
- Clinical Oral Investigations (publishedVersion)
- PubMed (publishedVersion)
- Topics
- Health Sciences › Medicine › Dermatology › Genetic and rare skin diseases.
- Health Sciences › Dentistry › General Dentistry › dental development and anomalies
- Health Sciences › Dentistry › Oral Surgery › Cleft Lip and Palate Research
- Keywords
- incontinentia pigmenti, dental anomalies, oral anomalies, genodermatosis, tooth agenesis, cleft palate, high-arched palate, sex distribution
- MeSH terms
- Anodontia, Cleft Palate, Exons, Humans, Incontinentia Pigmenti, Mouth Abnormalities, Palate, Tooth Abnormalities, Tooth Eruption, Gene Deletion, I-kappa B Kinase
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
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