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International Journal of Clinical... 2023A rare dental developmental anomaly known as regional odontodysplasia (RO) manifests itself in hypoplastic and hypomineralized teeth with discolored yellow to brown...
UNLABELLED
A rare dental developmental anomaly known as regional odontodysplasia (RO) manifests itself in hypoplastic and hypomineralized teeth with discolored yellow to brown enamel. Radiographically, the affected tooth appears as a shell tooth with a large pulp chamber and a thin layer of teeth structure. The treatment plan is not well-established, and a multidisciplinary approach is often needed. However, a literature review revealed that most of the studies regarding RO are case reports, and more research, including epidemiological, genetic, and experimental studies, is needed to understand this condition better. Additionally, the cause of this anomaly is currently unknown. Removing the affected tooth is a controversial matter, and the treatment options should be chosen on an individual basis, considering the patient's goals for treatment. This report describes an uncommon case of an 8-year-old girl who has RO affecting the front teeth in the upper jaw and a long-standing infection caused by actinomycosis.
HOW TO CITE THIS ARTICLE
Helal N. Regional Odontodysplasia with Actinomycosis Infection: A Rare Case Report. Int J Clin Pediatr Dent 2023;16(1):190-197.
PubMed: 37020769
DOI: 10.5005/jp-journals-10005-2504 -
Head and Neck Pathology Jun 2012Segmental Odontomaxillary Dysplasia (SOD) is an uncommon developmental disorder of unknown etiology that causes a unilateral alteration of the maxilla associated with an... (Review)
Review
Segmental Odontomaxillary Dysplasia (SOD) is an uncommon developmental disorder of unknown etiology that causes a unilateral alteration of the maxilla associated with an abnormal growth and maturation of bone, lack of one or both premolars and delayed tooth eruption on the affected side, alteration of adjacent gingival tissue with or without facial cutaneous lesions. Radiographically is observed an irregular trabecular pattern and reduction of the maxillary sinus. There are 50 cases reported in the English-language literature. A literature review is herein presented emphasizing the clinical, radiographic, and histological features, and three additional cases of SOD affecting a 22 years-old woman, a 18 years-old man, and a 5 years-old boy, respectively are described.
Topics: Adolescent; Child, Preschool; Facial Asymmetry; Female; Humans; Male; Maxilla; Odontodysplasia; Young Adult
PubMed: 22139529
DOI: 10.1007/s12105-011-0315-6 -
American Journal of Human Genetics Feb 2015Singleton-Merten syndrome (SMS) is an infrequently described autosomal-dominant disorder characterized by early and extreme aortic and valvular calcification, dental...
Singleton-Merten syndrome (SMS) is an infrequently described autosomal-dominant disorder characterized by early and extreme aortic and valvular calcification, dental anomalies (early-onset periodontitis and root resorption), osteopenia, and acro-osteolysis. To determine the molecular etiology of this disease, we performed whole-exome sequencing and targeted Sanger sequencing. We identified a common missense mutation, c.2465G>A (p.Arg822Gln), in interferon induced with helicase C domain 1 (IFIH1, encoding melanoma differentiation-associated protein 5 [MDA5]) in four SMS subjects from two families and a simplex case. IFIH1 has been linked to a number of autoimmune disorders, including Aicardi-Goutières syndrome. Immunohistochemistry demonstrated the localization of MDA5 in all affected target tissues. In vitro functional analysis revealed that the IFIH1 c.2465G>A mutation enhanced MDA5 function in interferon beta induction. Interferon signature genes were upregulated in SMS individuals' blood and dental cells. Our data identify a gain-of-function IFIH1 mutation as causing SMS and leading to early arterial calcification and dental inflammation and resorption.
Topics: Amino Acid Sequence; Aortic Diseases; Arteries; Base Sequence; Calcinosis; DEAD-box RNA Helicases; Dental Enamel Hypoplasia; Exome; Genes, Dominant; Humans; Immunohistochemistry; Interferon-Induced Helicase, IFIH1; Interferon-beta; Metacarpus; Models, Molecular; Molecular Sequence Data; Muscular Diseases; Mutation, Missense; Odontodysplasia; Osteoporosis; Pedigree; Phenotype; Sequence Analysis, DNA; Tooth Abnormalities; Vascular Calcification
PubMed: 25620204
DOI: 10.1016/j.ajhg.2014.12.014 -
Schweizer Monatsschrift Fur Zahnmedizin... 2013
Topics: Comorbidity; Dental Enamel Hypoplasia; Dentin Dysplasia; Humans; Odontodysplasia
PubMed: 23836096
DOI: No ID Found -
European Oral Research May 2018The aim of this article was to review the literature and present a case of regional odontodysplasia (ROD) with special emphasis on clinical and radiographic features. A...
The aim of this article was to review the literature and present a case of regional odontodysplasia (ROD) with special emphasis on clinical and radiographic features. A 6-year-old girl was referred to our department with the chief complaint of missing her permanent maxillary left central incisor, lateral incisor, and both of her canines. The gingiva of the involved region was enlarged, fibrous, and tense. Radiographic examination showed abnormal tooth formation and shortened roots. After 3 years of follow up with temporary prosthetic rehabilitation, periodontal surgery was performed. Following forced eruption and levelling, abnormal tooth eruption and root development were observed. ProRoot MTA (Dentsply-Maillefer, Ballaigues, Switzerland) was used for root canal treatment. Intracanal fiber posts selected and access cavities were restored with composite resin. Prosthetic rehabilitation was completed with zirconia ceramic crowns. The time of diagnosis, characteristics of the present/existing symptoms, and functional and esthetic needs of the patient should be considered to determine the optimal treatment modality for ROD.
PubMed: 30775712
DOI: 10.26650/eor.2018.495 -
The Journal of Clinical Pediatric... Sep 2023Regional odontodysplasia (RO) is a rare developmental abnormality of epithelial and mesenchymal dental tissues. Due to its poorly understood etiology, assessing and...
Regional odontodysplasia (RO) is a rare developmental abnormality of epithelial and mesenchymal dental tissues. Due to its poorly understood etiology, assessing and discussing related clinical cases of this dental anomaly is crucial to guide professionals in improving its treatment and outcomes. This article aimed to report the case of a 9-year-old male patient who presented to our department with the main complaint of absent eruption of permanent left mandibular quadrant teeth. This is the first case reported in China from a patient with multiple cutaneous nevi on the face and neck, and based on the retrieved clinical and radiographic features, we described and discussed the treatment and etiology of RO.
Topics: Male; Humans; Child; Odontodysplasia; Neck; Mandible; Tooth Eruption
PubMed: 37732452
DOI: 10.22514/jocpd.2023.068 -
European Journal of Paediatric Dentistry Sep 2020Segmental odontomaxillary dysplasia is an uncommon nonhereditary growth disorder that affects the maxilla, gums and ipsilateral dentition. The disorder is diagnosed...
BACKGROUND
Segmental odontomaxillary dysplasia is an uncommon nonhereditary growth disorder that affects the maxilla, gums and ipsilateral dentition. The disorder is diagnosed mainly based on dental (over-retention of primary teeth, dental agenesis and diastemas) and bone findings (bone sclerosis, irregular trabeculation of immature bone and reduced maxillary sinus). This paper provides a case report.
CASE REPORT
A 5-year-old child with skin manifestations including hypertrichosis, facial erythema and pigmented nevus was diagnosed with type II segmental odontomaxillary dysplasia based on clinical, radiographic and histopathological analysis.
CONCLUSION
The skin findings can help with the suspicion of segmental odontomaxillary dysplasia, although the definitive diagnosis is typically established by a paediatric dentist based on clinical and radiological findings.
Topics: Child, Preschool; Diastema; Humans; Maxilla; Odontodysplasia; Skin Diseases; Tooth, Deciduous
PubMed: 32893658
DOI: 10.23804/ejpd.2020.21.03.14 -
Contemporary Clinical Dentistry 2015Regional odontodysplasia (RO) is a rare developmental anomaly involving both mesodermal and ectodermal components in primary or permanent dentition. It affects the...
Regional odontodysplasia (RO) is a rare developmental anomaly involving both mesodermal and ectodermal components in primary or permanent dentition. It affects the maxilla and the mandible or both; however, maxilla is more commonly involved. This article reports the case of 33-month-old boy who came with the chief complaint of delayed eruption of mandibular teeth. Findings of clinical and radiographic examination were consistent with those of RO. Maxillary dentition was unaffected. Clinical and radiographic features and treatment options are discussed.
PubMed: 26097362
DOI: 10.4103/0976-237X.156054 -
Journal of Oral and Maxillofacial... Jul 2009Regional odontodysplasia is an uncommon developmental anomaly affecting a localized area of dentition, with distinctive clinical, radiographic, and histological...
Regional odontodysplasia is an uncommon developmental anomaly affecting a localized area of dentition, with distinctive clinical, radiographic, and histological findings. This article reviews a case of a 14-year-old female who reported with unerupted maxillary anterior teeth. This case was rare in that it involved maxillary dentition with unerupted maxillary anterior teeth on the right side, but the left central incisor was missing. The molars on the right side were showing some amount of abnormality. Radiographically, the affected teeth had a 'ghostly' appearance, showing a marked reduction in radiodensity. Both enamel and dentin appeared to be very thin, the pulp chamber was exceedingly large, and the roots were short with wide open apices. All the characteristics were consistent with the diagnosis of regional odontodysplasia. The care and treatment of this patient required a multidisciplinary approach. The unerupted maxillary anterior teeth were surgically removed, following which temporary prosthetic restoration was provided to improve esthetics and to restore the function.
PubMed: 21887003
DOI: 10.4103/0973-029X.57671 -
Contemporary Clinical Dentistry 2022Regional odontodysplasia (RO), also called ghost teeth, is a rare nonhereditary developmental dental anomaly affecting the epidermal and mesenchymal tissues associated...
Regional odontodysplasia (RO), also called ghost teeth, is a rare nonhereditary developmental dental anomaly affecting the epidermal and mesenchymal tissues associated with the development of tooth which can affect both primary and permanent dentition. It can affect the child's overall quality of life and sometimes may lead to skeletal malocclusion. Management of such patients requires a multidisciplinary approach. Essix retainers are being widely used as retention appliances. Various modifications of this appliance are also being attempted. Thus, this article aims to focus on the use of Essix retainer as an interim prosthesis by modifying it with the incorporation of pontics to manage partial edentulousness and mild orthodontic corrections in a 7-year-old child diagnosed with bilateral RO.
PubMed: 36213853
DOI: 10.4103/ccd.ccd_434_21