Dental and oral anomalies in incontinentia pigmenti: a systematic review

Minić, Snežana; Trpinac, Dušan; Gabriel, Heinz; Genčík, Martin; Obradović, Miljana

doi:10.1007/s00784-012-0721-5

Cited by 50 publications

(48 citation statements)

References 29 publications

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“…Regarding the increased frequency of high hard palate in our sample (71.4%), in a systematic review conducted in the period between 1993-2012, with 1,286 patients diagnosed with IP, where only 513 provided sufficient information to evaluate possible dental and oral anomalies, aged from 1 year, it was observed that 54.4% had some dental and/or oral anomaly, and high hard palate was one of the most frequent oral abnormalities. Despite the relative small absolute number of palate abnormalities, the number was 25 times higher than that found in general population (16) , indicating that high hard palate may also be a distinctive and useful finding for the diagnosis, as a minor criterion of IP. One advantage is that it is visible since birth, in contrast to dental anomalies that are detectable only after 1 year (16) .…”

Section: Discussionmentioning

confidence: 82%

“…Despite the relative small absolute number of palate abnormalities, the number was 25 times higher than that found in general population (16) , indicating that high hard palate may also be a distinctive and useful finding for the diagnosis, as a minor criterion of IP. One advantage is that it is visible since birth, in contrast to dental anomalies that are detectable only after 1 year (16) . In the evaluation of the stomatognathic system, there was a high prevalence of tongue mobility change (n=6), and oral (n=2) or oronasal breathing (n=1).…”

Section: Discussionmentioning

confidence: 82%

“…The prognosis of female patients diagnosed with IP is generally good and depends not only on skin manifestations, but also of extracutaneous features, which may affect the quality of life (16) . The presence of other alterations, such as dental and/or oral abnormalities, are included as minor IP diagnostic criteria (17) .…”

Section: Discussionmentioning

confidence: 99%

See 2 more Smart Citations

Alterações estomatognáticas e de fala são comuns entre crianças com incontinência pigmentar

Reis

Maria

Rosa

et al. 2015

Audiol., Commun. Res.

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Section: Discussionmentioning

confidence: 82%

Section: Discussionmentioning

confidence: 82%

See 1 more Smart Citation

Alterações estomatognáticas e de fala são comuns entre crianças com incontinência pigmentar

Reis

Maria

Rosa

et al. 2015

Audiol., Commun. Res.

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“…However, none of these studies had described cases of patients with cleft palate. Moreover, Minic et al 8) conducted a systematic review of dental findings in Bloch-Sulzberger syndrome between 1993 and 2010 and reported a high incidence of dental shape anomalies, hypodontia, and delayed dentition, together with cleft and high-arched palates. Furthermore, approximately 1 in 3 of cases were reported to involve a cleft palate.…”

Section: Discussionmentioning

confidence: 99%

“…Thereafter, the pigmented spots gradually disappear, and the prognosis is good 11) . Tooth abnormalities include hypodontia, delayed eruption, pegshaped teeth, impacted teeth, morphological anomalies, and malocclusion 6,8) . In addition to reports on skin symptoms associated with this syndrome 11) , a number of studies have also described associated dental features 2,4,6,7,9,13) and corresponding orthodontic treatment 1,3,10,14,16) .…”

Section: Introductionmentioning

confidence: 99%

Orthodontic Treatment for Bloch-Sulzberger Syndrome in Patient with Cleft Lip and Palate

Nojima

Onoda

Nishii

et al. 2017

Bull. Tokyo Dent. Coll.

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We performed orthodontic treatment, fitted prostheses, and provided restorative treatment in a patient with Bloch-Sulzberger syndrome and cleft lip and palate during the early mixed dentition period. We report the case after a subsequent 6-year retention phase including the period of pubertal growth. A girl aged 8 years 4 months visited our hospital with the chief complaint of crowding of the anterior teeth and anterior crossbite. She had bilateral cleft lip, alveolus, and palate; a Class II molar relationship; winging of both the maxillary bilateral central incisors; and spacing in the mandibular anterior teeth arches. Anterior crossbite comprised 0 mm overbite and −1 mm overjet. The crown diameter was at least one standard deviation smaller than normal in both the deciduous and permanent teeth, and the crowns were slightly peg-shaped. Panoramic radiograph confirmed congenital absence of 21 permanent teeth. Cephalometric analysis revealed poor growth of the maxilla, downward growth of the mandible, and lingual inclination of the maxillary central incisors. The diagnosis was skeletal anterior crossbite with cleft lip, alveolus, and palate, accompanied by hypodontia. Orthodontic treatment comprised an edgewise appliance and an expansion arch to improve crowding and anterior crossbite. The appliance was removed 2 years after treatment initiation, followed by crown restorations of the maxillary central incisors and mandibular deciduous anterior teeth. A metal retainer was then fitted to the maxillary dentition. She was subsequently placed in a 6-year retention phase including pubertal growth, during which occlusal stability and esthetics were maintained.

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Orodental manifestations in ectodermal dysplasia—A review

Bergendal¹

2014

American J of Med Genetics Pt A

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Oral signs and symptoms are present in most ectodermal dysplasias (EDs). The aim of this article is to summarize some of the literature on current knowledge of oral manifestations and orofacial function in EDs. The review will focus on the most common forms where dental manifestations can be crucial for a differential diagnosis of ED among individuals with hypodontia and oligodontia, and preferably where the investigations included persons who had a genetically verified diagnosis. Disturbances in tooth development are common and can appear as tooth agenesis, variations in size and shape of teeth, defects in the mineralized tissues, and problems in tooth eruption. Abnormalities in number, size, and shape of teeth, and reduced salivary secretion, present in isolated oligodontia as well as in hypohidrotic ED and incontinentia pigmenti. In some more rare EDs these symptoms appear in combination with clefts of lip and/or palate in some affected individuals. Leukokeratosis in the oral mucosa presents in 70% of genetically confirmed cases of pachyonychia congenita. Also, orofacial function is often affected in ED, due to malformations, an incomplete dentition, and low salivary secretion which can compromise chewing, swallowing, and speech. In conclusion, there is a clinical overlap in oral signs and symptoms between isolated oligodontia and the most common EDs. Studies with genetically confirmed diagnoses and larger cohorts, as well as multicenter collaboration and the establishing of international registries, would create a basis for refined diagnostics, where oral examinations should be an integrated part of clinical assessment.

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Dental and oral anomalies in incontinentia pigmenti: a systematic review

Abstract: 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.

Cited by 50 publications

References 29 publications

Alterações estomatognáticas e de fala são comuns entre crianças com incontinência pigmentar

Alterações estomatognáticas e de fala são comuns entre crianças com incontinência pigmentar

Orthodontic Treatment for Bloch-Sulzberger Syndrome in Patient with Cleft Lip and Palate

Orodental manifestations in ectodermal dysplasia—A review

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