{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T17:21:29Z","timestamp":1787764889263,"version":"build-2784847793"},"reference-count":7,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"2","license":[{"start":{"date-parts":[[2026,4,1]],"date-time":"2026-04-01T00:00:00Z","timestamp":1775001600000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0"}],"content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2026,4]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Cleidocranial dysplasia (CCD) is a rare autosomal dominant skeletal disorder characterized by multiple dental and skeletal abnormalities. While isolated case reports exist, this case is unique due to its clear familial inheritance pattern and the need for urgent surgical dental intervention in a symptomatic young adult, thereby contributing new clinical insights to the limited literature on CCD. A female patient in her early 20s presented with pain and pus discharge in the mandibular left posterior region for 3 months. Extraoral findings included short stature, frontal bossing, hypertelorism, midfacial hypoplasia, and hypoplastic clavicles. Intraoral examination revealed generalized microdontia, malalignment, multiple carious teeth, and a partially erupted mandibular left third molar with pericoronitis. A positive family history of similar skeletal features in her father and sibling raised suspicion of a syndromic diagnosis. Imaging studies showed open cranial sutures, underdeveloped maxilla, and an impacted mandibular molar with surrounding bone resorption. Differential diagnoses such as Down\u2019s syndrome, Gardner\u2019s syndrome, pycnodysostosis, and mandibuloacral dysplasia were ruled out, and a final diagnosis of CCD was established. The patient underwent surgical extraction of the third molar, excision of the associated follicular tissue, and socket curettage. Histopathology confirmed chronic nonspecific inflammation. Healing was uneventful with complete resolution of symptoms. The present case report underscores the importance of early diagnosis and timely dental intervention in patients with CCD. A multidisciplinary approach that includes dental, surgical, and genetic considerations is essential for improving both functional outcomes and quality of life in affected individuals.<\/jats:p>","DOI":"10.4103\/ccd.ccd_4_25","type":"journal-article","created":{"date-parts":[[2026,6,15]],"date-time":"2026-06-15T15:00:08Z","timestamp":1781535608000},"page":"142-146","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":0,"title":["Two-generation Cleidocranial Dysplasia Involving Three Individuals of a Family: A Rare Case Report"],"prefix":"10.4103","volume":"17","author":[{"given":"Maroti B.","family":"Wadewale","sequence":"first","affiliation":[{"name":"Department of Head and Neck Surgical Oncology, Malabar Cancer Centre, Thalassery, Kerala, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sanpreet Singh","family":"Sachdev","sequence":"additional","affiliation":[{"name":"Department of Oral Pathology and Microbiology, Bharati Vidyapeeth (Deemed to be University) Dental College and Hospital, Navi Mumbai, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Suleka","family":"Ranganath","sequence":"additional","affiliation":[{"name":"Department of Cleft and Craniofacial Surgery, Ashwini Trauma Centre, Cuttack, Odisha, India"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2026,6,15]]},"reference":[{"key":"R1-20260803","first-page":"173","article-title":"New genetic variants of RUNX2 in mexican families cause cleidocranial dysplasia","volume":"13","author":"Toral L\u00f3pez","year":"2024","journal-title":"Biology (Basel)"},{"key":"R2-20260803","first-page":"103611","article-title":"Demographic, clinical, and radiological characteristics of cleidocranial dysplasia: A systematic review of cases reported in South America","volume":"77","author":"Cano-P\u00e9rez","year":"2022","journal-title":"Ann Med Surg (Lond)"},{"key":"R3-20260803","doi-asserted-by":"crossref","first-page":"572","DOI":"10.1186\/s12903-024-04353-z","article-title":"Three-dimensional evaluation of dental characteristics in patients with cleidocranial dysplasia","volume":"24","author":"Lu","year":"2024","journal-title":"BMC Oral Health"},{"key":"R4-20260803","doi-asserted-by":"crossref","first-page":"606","DOI":"10.32677\/IJCR.2020.v06.i11.001","article-title":"Clinico-radiographic spectrum of cleidocranial dysplasia: A case series","volume":"6","author":"Nayak","year":"2020","journal-title":"Indian J Case Rep"},{"key":"R6-20260803","doi-asserted-by":"crossref","first-page":"1350","DOI":"10.3390\/medicina57121350","article-title":"Genetic pattern, orthodontic and surgical management of multiple supplementary impacted teeth in a rare, cleidocranial dysplasia patient: A case report","volume":"57","author":"Inchingolo","year":"2021","journal-title":"Medicina (Kaunas)"},{"key":"R7-20260803","first-page":"14","article-title":"Assessment of different patterns of impacted mandibular third molars and their associated pathologies","volume":"2","author":"Nazir","year":"2014","journal-title":"J Adv Med Dent Sci Res"},{"issue":"Suppl 1","key":"R8-20260803","doi-asserted-by":"crossref","first-page":"72","DOI":"10.1111\/eos.12418","article-title":"Tooth formation and eruption \u2013 Lessons learnt from cleidocranial dysplasia","volume":"126","author":"Kreiborg","year":"2018","journal-title":"Eur J Oral Sci"}],"container-title":["Contemporary Clinical Dentistry"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.ovid.com\/10.4103\/ccd.ccd_4_25","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T16:50:11Z","timestamp":1787763011000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ovid.com\/10.4103\/ccd.ccd_4_25"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,4]]},"references-count":7,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2026]]}},"URL":"https:\/\/doi.org\/10.4103\/ccd.ccd_4_25","relation":{},"ISSN":["0976-237X","0976-2361"],"issn-type":[{"value":"0976-237X","type":"print"},{"value":"0976-2361","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,4]]},"assertion":[{"value":"2025-01-04","name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-03-23","name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}}]}}