Imaging Sci Dent.  2019 Dec;49(4):307-315. 10.5624/isd.2019.49.4.307.

Case series of cleidocranial dysplasia: Radiographic follow-up study of delayed eruption of impacted permanent teeth

• Yeom HG
• Park WJ
• Choi EJ
• Kang KH
• Lee BD

• KMID: 2466554
• DOI: http://doi.org/10.5624/isd.2019.49.4.307

Abstract

This report describes 3 cases of cleidocranial dysplasia (CCD) and presents relevant findings on long-term follow-up radiographic images of impacted permanent teeth with delayed eruption. Radiographic images of 3 CCD patients were reviewed retrospectively. These images were mainly composed of panoramic and skull radiographs, and the follow-up periods were 3, 13, and 13 years, respectively. The distinct features revealed by the images were described, and the eruption state of impacted permanent teeth was evaluated. The features common to the 3 cases were multiple supernumerary teeth, the presence of Wormian bone, underdevelopment of the maxilla and the maxillary sinus, and clavicular hypoplasia. The eruption of impacted permanent teeth was not observed without proper dental treatment in adult CCD cases, even after long time periods had elapsed. When proper orthodontic force was applied, tooth movement was observed in a manner not significantly different from the general population.

Keyword

Cleidocranial Dysplasia; Jaw; Radiography

MeSH Terms

Adult
Cleidocranial Dysplasia*
Follow-Up Studies*
Humans
Jaw
Maxilla
Maxillary Sinus
Radiography
Retrospective Studies
Skull
Tooth Movement
Tooth*
Tooth, Supernumerary

Figure

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  Fig. 1 Case 1. Chest radiograph of a 1-year-old patient shows a focal defect of the right clavicle and congenital pseudarthrosis of the clavicle.

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  Fig. 2 Case 1. A. Panoramic radiograph of a 7-year-old girl shows delayed eruption of permanent teeth and an impacted supernumerary tooth above the impacted right lower canine. The right lower primary lateral incisor and canine are fused. Skull lateral (B) and posteroanterior (C) radiographs show underdevelopment of the maxilla, Wormian bone in the cranial suture, and parietal bossing.

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  Fig. 3 Case 1. Panoramic radiograph of a 10-year-old patient reveals 3 newly formed supernumerary teeth. New supernumerary teeth are found distal to the upper right and left canines and the lower left canine. The supernumerary tooth above the right lower canine has already been extracted.

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  Fig. 4 Case 2. A. Panoramic radiograph of a 23-year-old male patient shows multiple impacted permanent teeth and prolonged retention of numerous deciduous teeth. Supernumerary teeth are seen at the left upper third molar region and above the right lower first premolar. B. Skull lateral radiograph shows maxillary hypoplasia. C. The sagittal cone-beam computed tomographic image shows Wormian bone in the rhomboidal suture. D. Waters' view shows a hypoplastic zygomatic arch, a small maxillary sinus, parietal bossing, and a large anterior fontanelle.

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  Fig. 5 Case 2. A. Panoramic radiograph of a 35-year-old man shows a similar appearance to a panoramic radiograph obtained of the same patient when he was 23 years old. Dense alveolar crestal bone is seen around the upper right first premolar and both lower premolars. B. The coronal cone-beam computed tomographic image shows a definite sclerotic appearance of both mandibular premolar regions.

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  Fig. 6 Case 2. Chest radiograph of a 35-year-old patient shows a congenital ossification defect in both distal third clavicles, which are displaced. Another visible skeletal deformity is spina bifida in the lower cervical spine and the upper thoracic spine.

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  Fig. 7 Case 2. Panoramic radiographs of post-orthodontic treatment. A. At 35 years old. B. At 36 years old. Two panoramic radiographs show additional eruptive patterns of permanent teeth.

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  Fig. 8 Case 3. A. Panoramic radiograph of a woman at 46 years of age reveals multiple impacted permanent teeth. B and C. Skull lateral and posteroanterior radiographs show underdevelopment of the maxilla, edgeto-edge bite, and wormian bone in the cranial suture and anterior fontanelle.

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  Fig. 9 Case 3. Chest radiograph of a 46-year-old woman shows underdevelopment of the right and left clavicles.

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  Fig. 10 Case 3. A. Panoramic radiograph of a 59-year-old woman shows a similar appearance to a panoramic radiograph obtained of the same patient when she was 46 years old. The supernumerary teeth are not clearly visible in the panoramic image. B–D. The axial cone-beam computed tomographic images reveal multiple supernumerary teeth. Five supernumerary teeth are located in the regions of the right upper canine, the left upper central incisor (where a dentigerous cyst was also present), the lower left first and second premolars, and the lower right central incisor.

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