Prevalence and Patterns of Permanent Tooth Agenesis in Patients With Crouzon or Apert Syndrome : A Systematic Review and Meta‐Analysis

Article Article scientifique Revue de la littérature Prevalence and Patterns of Permanent Tooth Agenesis in Patients With Crouzon or Apert Syndrome : A Systematic Review and Meta‐Analysis eng 2025-10-14 author Becerril Santos Magdalena Cecilia 0009-0009-3332-3408 Division of Orthodontics, University Clinics of Dental Medicine, University of Geneva, Geneva, Switzerland. 406433 author Ongkosuwito Edwin M Section of Orthodontics and Craniofacial Biology, Department of Dentistry, Radboud University Medical Center, Nijmegen, the Netherlands. author Papadopoulou Alexandra 0000-0001-5981-0479 Division of Orthodontics, University Clinics of Dental Medicine, University of Geneva, Geneva, Switzerland. 974834 CUMD - Clinique d'orthodontie (CORT) author Antonarakis Gregory 0000-0001-6741-6922 Division of Orthodontics, University Clinics of Dental Medicine, University of Geneva, Geneva, Switzerland. 133276 CUMD - Division d'orthodontie - communs (DORT) ocr.70046 10.1111/ocr.70046 41085212 1601-6335 https://onlinelibrary.wiley.com/doi/10.1111/ocr.70046 Crouzon and Apert syndromes are rare syndromic craniosynostoses frequently associated with craniofacial and dental anomalies, including tooth agenesis. Although individual studies have reported tooth agenesis prevalence data in specific populations, no attempts have been made to systematically synthesise these data. This systematic review and meta-analysis evaluated the prevalence and patterns of permanent tooth agenesis in patients with Crouzon or Apert syndromes. The study followed a pre-registered protocol (PROSPERO CRD42024581856). Two independent investigators conducted a literature search in MEDLINE (via PubMed), Web of Science, Embase, ProQuest, and Google Scholar to identify studies on permanent tooth agenesis in individuals with Crouzon or Apert syndromes. Studies were deemed eligible if they reported on permanent tooth agenesis in patients diagnosed with Crouzon or Apert syndromes. Risk of bias was assessed using the Joanna Briggs Institute tool. Random-effects and inverse variance heterogeneity meta-analysis models were used for data synthesis. After database search, deduplication, and screening, seven studies were included, comprising a total of 89 individuals with Apert and 77 with Crouzon syndrome. The estimated overall prevalence of permanent tooth agenesis, excluding third molars, was 37% (95% CI: 28%-48%) in individuals with Apert and 31% (95% CI: 17%-46%) in those with Crouzon syndrome. In Apert syndrome, the most common tooth agenesis pattern was the bilateral absence of the mandibular second premolars, followed by the absence of the left maxillary lateral incisors and the bilateral absence of the maxillary lateral incisors. In Crouzon syndrome, the most frequent tooth agenesis pattern was the absence of the right mandibular second premolar, followed by the bilateral absence of the mandibular second premolars. Individuals with Crouzon or Apert syndrome present a high prevalence of tooth agenesis. Early diagnosis of missing permanent teeth is essential to optimise long-term treatment planning and improve functional and aesthetic outcomes. However, the evidence is limited by small sample sizes and heterogeneity across included studies. Apert syndrome Crouzon syndrome Hypodontia Meta‐analysis Tooth agenesis Dewey 617.6 209 Division d'orthodontie 24Q1 false 1448 Orthodontie 1065 Orthodontics & craniofacial research 150c39e0-702d-4ee7-ab72-c9272fe905dc Article (Published version) Public Orthod_Craniofacial_Res_-_2025_-_Be.pdf 1173599 application/pdf CC BY-4.0 357df6a6f554c9a6c0c9f5a56ca83a4b2721daddb2c1d742cb7e0d99cac3b205 38395a09-d6a2-423c-a226-5f53217a2a48 Supplemental data Public ocr70046-sup-0001-supplementary_mat.docx 113885 application/vnd.openxmlformats-officedocument.wordprocessingml.document f333f303457f7886c7ae8ea9f415559ee8cffa49fd2e8d270fa26eee6c229044 openAccess Faculté de médecine / Section de médecine dentaire / Département de réhabilitation oro-faciale / Division d'orthodontie

Title	Published in	Access level	OA Policy	Year	Views	Downloads

Prevalence and Patterns of Permanent Tooth Agenesis in Patients With Crouzon or Apert Syndrome : A Systematic Review and Meta‐Analysis	Orthodontics & craniofacial research			2025	9	108

Archive ouverte UNIGE

Becerril Santos, Magdalena Cecilia