Dental anomalies in 14 patients with IP: clinical and radiological analysis and review

Santa-Maria, Fernanda D; Mariath, Luiza Monteavaro; Poziomczyk, Cláudia Schermann; Maahs, Márcia Angélica Peter; Rosa, Rafael Fabiano Machado; Zen, Paulo Ricardo Gazzola; Schüller-Faccini, Lavínia; Kiszewski, Ana Elisa

doi:10.1007/s00784-016-1977-y

Cited by 10 publications

(7 citation statements)

References 27 publications

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“…12 The phenotypic array of odontological anomalies is broad and complex. [31][32][33] Dental anomalies are constant in IP, and careful tooth investigation is mandatory. Particular dental abnormalities are considered as major diagnostic criterion for IP 34 (Table 1).…”

Section: Major Criteriamentioning

confidence: 99%

“…Particular dental abnormalities are considered as major diagnostic criterion for IP 34 (Table 1). Recent data from a systematic review included palate anomalies in the oral findings of IP patients 32 and in an IP diagnostic criteria update. The main anomalies are the following: multiple dental agenesis in temporary and permanent dentition (prevalence of approximately 60% and 90%, respectively), coronary morphological abnormalities (in approximately 70% of cases), delayed dentition and various types of dento-maxillary dysmorphia (class II in 40% of cases, class III in 30% of cases and/or transverse maxillo-mandibular hypodevelopment) and arched palate or an orofacial cleft.…”

Section: Major Criteriamentioning

confidence: 99%

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Multidisciplinary consensus recommendations from a European network for the diagnosis and practical management of patients with incontinentia pigmenti

Bodemer

Diociaiuti

Hadj‐Rabia

et al. 2020

Acad Dermatol Venereol

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Background Incontinentia pigmenti (IP) is a rare multisystemic X-linked dominant genetic disorder characterized by highly diagnostic skin lesions. The disease can be misdiagnosed in infants, and complications affecting the eyes and/or the brain can be severe. Our objective was to highlight the urgency of an appropriate diagnosis and management strategy, as soon as the first symptoms appear, and the need for a well-codified monitoring strategy for each child.Methods An in-depth literature review using a large number of databases was conducted. The selection criteria for articles were literature review articles on the disease, case series and retrospective studies based on the disease, clinical studies (randomized or not) on treatment, articles discussing patient care and management (treatment, diagnosis, care pathways), and recommendations. The research period was from 2000 until 2018. A group of multidisciplinary experts in IP management was involved, issued from different healthcare providers of the European Network for Rare Skin Diseases (ERN-Skin). The final recommendations have been submitted to two patient representative associations and to a general practitioner and a neonatal specialist prior to their finalization. Results and conclusionThe diagnosis of IP must be promptly performed to detect potential extracutaneous manifestations, thus allowing the timely implementation of specific therapeutic and monitoring strategies. Eye involvement can be a therapeutic urgency, and central nervous system (CNS) involvement requires a very rigorous long-term follow-up.Assessments and patient support should take into account the possible co-occurrence of various symptoms (including motor, visual and cognitive symptoms).

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Section: Major Criteriamentioning

confidence: 99%

Section: Major Criteriamentioning

confidence: 99%

Multidisciplinary consensus recommendations from a European network for the diagnosis and practical management of patients with incontinentia pigmenti

Bodemer

Diociaiuti

Hadj‐Rabia

et al. 2020

Acad Dermatol Venereol

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“…The appearance of conoid teeth in patients with IP is often reported (Cao et al, 2017;Al-Abdallah et al, 2015;Santa-Maria et al, 2017). A harmonic smile has a great influence on the life and self-esteem of children and conoid teeth can cause diastemas and aesthetic dissatisfaction, which was a patient's complaint of this report.…”

Section: Discussionmentioning

confidence: 77%

“…The reported patient presents agenesis of 7 permanent teeth, which are the most common dental findings found in IP patients, according to Al-Abdallahet et al (2015). However, in the case series presented by Santa-Maria et al (2017) , anomalies in dental crowns were the most frequent oral features identified, occurring in 71.4% of cases, with the central incisor being the most affected.…”

Section: Discussionmentioning

confidence: 89%

Dental anomalies of a child with incontinentia pigmenti: Case report

Regis

Neves

Lima

et al. 2021

RSD

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Incontinentia Pigmenti is a rare multi-system dominant genetic disorder caused by a mutation of the IKBKG/NEMO gene, localized on the X-chromosome, locus Xq28, characterized by dermatological, ocular, neurological, and dental alterations. This case report shows Incontinentia Pigmenti dental findings and emphasizes the importance of the dentist's knowledge about these anomalies so that the patient can be regularly monitored throughout the development of the dentition. Keywords: Child, Incontinentia Pigmenti, Tooth Abnormalities.

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“…Among these, the dental abnormalities (e.g. delayed dentition, partial anodontia, hypodontia, abnormally shaped teeth) are the most commonly reported, occurring in more than 80% of all patients [ 16 , 17 ].…”

Section: Discussionmentioning

confidence: 99%