A case of incontinentia pigmenti.

Maeda, Reiko; Kasakura, Tatsuo; Sakamoto, Haruhiko; Otozura, Noriko; Yokokura, Yukihiro; Inose, Masasuke; Sato, Katsunori; Asakura, Akihito

doi:10.5794/jjoms.33.740

Cited by 1 publication

(2 citation statements)

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“…The name Bloch-Sulzberger syndrome, as it is known today, was proposed after it was proven that the accompanying symptoms were not restricted to those of the skin 3) . Maeda et al 7) examined 21 cases of BlochSulzberger syndrome reported in the field of dentistry and found that all these studies described tooth anomalies with a high incidence of comorbidity; in addition, hypodontia, abnormal tooth morphology and structure, and delayed eruption of deciduous teeth accounted for the three most common anomalies, similar to past reports. However, none of these studies had described cases of patients with cleft palate.…”

Section: Discussionsupporting

confidence: 55%

“…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) . Reports on orthodontic treatment for this syndrome when accompanied by cleft lip and palate are particularly limited, however, and, to our knowledge, there are none on the subsequent long-term course of such treatment.…”

Section: Introductionmentioning

confidence: 99%

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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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Section: Discussionsupporting

confidence: 55%