Dental and oral anomalies in incontinentia pigmenti: a systematic review

@article{Mini2012DentalAO,
  title={Dental and oral anomalies in incontinentia pigmenti: a systematic review},
  author={Sne{\vz}ana Mini{\'c} and Du{\vs}an Trpinac and Heinz Gabriel and Martin Gen{\vc}{\'i}k and Miljana Obradovi{\'c}},
  journal={Clinical Oral Investigations},
  year={2012},
  volume={17},
  pages={1-8}
}
ObjectivesIncontinentia 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 methodsWe analyzed the literature data from 1,286 IP cases from the period 1993–2010.ResultsDental and/or oral anomalies were diagnosed for 54.38% of the investigated… 
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In the diagnosis of IP, the presence of IKBKG mutation typical for IP, and existence of family relatives with diagnosed IP are taken into account and proposed as major criteria one of the stages of IP skin lesions.
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References

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TLDR
This is the first time that the presence of gothic palate in patients with IP has been documented and, in two female subjects and one male subject, in which nonrandomed X inactivation did not occur, gothsic palate could be supposed as characteristic of IP.
Ocular anomalies in incontinentia pigmenti: literature review and meta-analysis.
TLDR
This study suggests that IP is far more frequent than anyone could estimate and early detection and treatment of ophthalmological, neurological etc. findings may prevent severe consequences that IP may cause.
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TLDR
A 12-year-old female with incontinentia pigmenti associated with enamel hypocalcification and hypoplasia of the permanent teeth is reported, one of the lesser known ectodermal dysplasias with fewer than 800 cases reported in the world literature.
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TLDR
The distribution of manifestations in a pediatric cohort and guidelines for follow-up of incontinentia pigmenti are analyzed to better understand the pathological mechanisms of IP and develop new therapies.
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TLDR
The oral and dental manifestations of an IP case in a Venezuelan pediatric patient showed macular pigmented lesions in her face, trunk, back, legs, and fingers as well as abnormal hair distribution, alopecia on the vertex, and hypoplasia of eyebrows.
Incontinentia pigmenti in a surviving male is accompanied by hypohidrotic ectodermal dysplasia and recurrent infection.
TLDR
A NEMO stop codon mutation has been identified that has arisen de novo in his affected mother and may explain why this child survived for two years and 7 months with familial Incontinentia pigmenti.
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TLDR
A large case series of seven patients, all female, who presented to the Department of Paediatric Dentistry at the Eastman Dental Hospital over the last 16 years is reported and the dental features of these cases were typical and included missing teeth, microdontia and delayed eruption.
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TLDR
It is found that, while the frequency of the first three cutaneous stages of IP was comparable with previous studies, none of the secondary cases manifested any serious neurological complications but all displayed stage IV pale anhidrotic reticulate lines on their posterior calves.
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TLDR
The mechanism by which NEMO/IKKgamma deficiency causes, via the NF-kappaB pathway, the phenotypical expression of the disease has recently been elucidated, and the newest research findings on eosinophil recruitment through eotaxin release by activated keratinocytes are described in the review.
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