Generalized odontodysplasia in a 5-year-old patient with Hallermann-Streiff syndrome: clinical aspects, cone beam computed tomography findings, and conservative clinical approach

Damasceno, Juliana Ximenes; Couto, José Luciano Pimenta; Alves, Karla Shangela da Silva; Chaves, Cauby Maia; Costa, Fábio Wildson Gurgel; Pimenta, Alynne de Menezes Vieira; Fonteles, Cristiane Sá Roriz

doi:10.1016/j.oooo.2014.04.013

Cited by 15 publications

(15 citation statements)

References 22 publications

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“…Among syndromes featuring RO, there has been published only one case revealing a mandibular bone pattern abnormality to date, which was published by some of the present authors. 8 Damasceno et al 8 showed an altered maxillomandibular trabecular pattern associated with a generalized odontodysplasia on CBCT of a 5-year-old girl diagnosed with Hallermann-Streiff syndrome. In both situations (syndromic and nonsyn-dromic RO), a combination of some aspects appears to explain the altered radiopacity in areas surrounding the altered teeth: (1) since teeth displaying RO features do not have a normal development, it may be reasonable disturbance on the alveolar ridge formation process followed by an alteration in the alveolar bone density; 19 (2) an abnormal periosteal appositional bone formation or a defective endosteal resorption could alter the mandibular bone trabeculae organization.…”

Section: Discussionmentioning

confidence: 99%

“… 3 Although teeth alterations without other extraoral findings are the usual clinical presentation of RO, it has been reported RO teeth simultaneously with epidermal nevus syndrome, 4 5 hypo-phosphatasia, 6 hydrocephalus, 7 and Hallermann-Streiff syndrome. 8 These associations with systemic conditions reinforce the uncertain etiology of the RO, which has been related to other factors, such as a viral infection, 9 use of inappropriate medication during pregnancy, 10 trauma, nutritional deficiency, infection, and metabolic abnormalities. 11 …”

Section: Introductionmentioning

confidence: 92%

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Mandibular Regional Odontodysplasia in an 8-year-old Boy showing Teeth Disorders, Gubernaculum Tracts, and Altered Bone Fractal Pattern

Cavalcante

Fonteles

Ribeiro

et al. 2018

International Journal of Clinical Pediatric Dentistry

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Regional odontodysplasia (RO) is a rare nonhereditary developmental condition usually affecting maxilla and female patients. A few number of articles have used the cone beam computed tomography (CBCT) to describe imaging findings in RO. Thus, this article aims to report unusual imaging findings in a Brazilian child who presented with mandibular RO. A healthy 8-year-old boy was referred for evaluation of an anterior mandibular gingival enlargement. Radiographically, all right mandibular teeth showed wide pulp chambers and shortened roots with thin dentinal walls and open apices, featuring a “ghost teeth“ appearance, which indicated a diagnosis of RO. Missing teeth and altered radiopacity were observed. Cone beam computed tomography provided great insight into RO, allowing to identify gubernaculum tracts (GTs) associated with RO teeth. Fractal analysis of the bone surrounding RO teeth revealed an altered trabecular pattern.Clinical significanceThis case describes a rare developmental teeth-related pathology in a boy who showed unusual features on imaging exams. The CBCT provided the observation of RO tooth-related GTs, probably a new finding added to the international literature regarding RO, as well as the pulp chamber volume characterization of an affected tooth not published yet. In addition, it was observed an altered fractal pattern of the mandibular bone adjacent to RO teeth, which has not been described to date.How to cite this article: de Sa Cavalcante D, Fonteles CSR, Ribeiro TR, Kurita LM, Pimenta AVM, Carvalho FSR, Costa FWG. Mandibular Regional Odontodysplasia in an 8-year-old Boy showing Teeth Disorders, Gubernaculum Tracts, and Altered Bone Fractal Pattern. Int J Clin Pediatr Dent 2018;11(2):128-134.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 92%

Mandibular Regional Odontodysplasia in an 8-year-old Boy showing Teeth Disorders, Gubernaculum Tracts, and Altered Bone Fractal Pattern

Cavalcante

Fonteles

Ribeiro

et al. 2018

International Journal of Clinical Pediatric Dentistry

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“…Hallermann-Streiff syndrome is a rare congenital disease, first described by Aubry in 1893, then by Hallermann in 1948, and Streiff in 1950 (Cohen, 1991). About 250 cases have been reported to date (Damasceno et al, 2014). The only available data about prevalence come from Higurashi et al (1990), who assessed a prevalence of 0.04/1000 births with 1 case of HSS among 27 472 newborns during a 15-year study.…”

Section: Discussionmentioning

confidence: 99%

“…Dental abnormalities occur in 80% to 85% of cases (Cohen, 1991). These include natal teeth, impacted deciduous teeth, hypodontia, teeth malformations, odontodysplasia, and short-root abnormalities (Sharp and Kopel, 1970; Hutchinson, 1971; Cohen, 1991; Damasceno et al, 2014; Gungor et al, 2015).…”

Section: Discussionmentioning

confidence: 99%

“…Nonsurgical treatment consisted in conservation of deciduous teeth with intensive oral hygiene program and removable prosthodontics to cope with the permanent missing teeth. Orthodontics was also used through multiattached appliances in order to grant teeth alignment (Sharp and Kopel, 1970; Vadiakas et al, 1995; Defraia et al, 2003; Limeres et al, 2004; Kirzioğlu and Ceyhan, 2009; Chee and Lee, 2011; Robotta and Schafer, 2011; Parikh and Gupta, 2012; Thomas et al, 2013; Damasceno et al, 2014; Gungor et al, 2015).…”

Section: Discussionmentioning

confidence: 99%

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Diagnosis and Innovative Multidisciplinary Management of Hallermann-Streiff Syndrome

Dulong

Bornert

Gros

et al. 2018

The Cleft Palate Craniofacial Journal

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Hallermann-Streiff syndrome (HSS) is a rare congenital disorder that mainly affects head and face development. We described the different patterns of the disease throughout the whole growth period and provided innovative treatment steps. Indeed, early genioplasty and dental implantation before growth completion were performed. These steps allowed to improve facial growth and to provide orthodontic anchorage, respectively. Complementary orthognathic surgery achieved satisfactory occlusion and refined aesthetics. We believe such an approach could be considered as a relevant treatment modality to complete multidisciplinary care in patients with HSS.

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Cone‐beam computed tomography in the diagnosis of regional odontodysplasia

Mabrouk

Chouchène

Maatouk

et al. 2022

Clinical Case Reports

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Generalized odontodysplasia in a 5-year-old patient with Hallermann-Streiff syndrome: clinical aspects, cone beam computed tomography findings, and conservative clinical approach

Cited by 15 publications

References 22 publications

Mandibular Regional Odontodysplasia in an 8-year-old Boy showing Teeth Disorders, Gubernaculum Tracts, and Altered Bone Fractal Pattern

Mandibular Regional Odontodysplasia in an 8-year-old Boy showing Teeth Disorders, Gubernaculum Tracts, and Altered Bone Fractal Pattern

Diagnosis and Innovative Multidisciplinary Management of Hallermann-Streiff Syndrome

Cone‐beam computed tomography in the diagnosis of regional odontodysplasia

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