WNT10A mutations are associated with tooth agenesis. This study aimed to assess the clinical outcomes of dental implants in patients carrying WNT10A mutations with different molecular statuses and phenotypes over a long-term follow-up period. Patients with tooth agenesis were screened by whole-exome sequencing (WES) from January 2010 to September 2023. Carriers of pathogenic WNT10A mutations who underwent subsequent dental implant therapy were evaluated. WES identified 66 patients with tooth agenesis carrying WNT10A mutations. Of these, 27 patients (11 males/16 females; mean age 19.6 years) met the inclusion criteria. Homozygotes had significantly more missing teeth (24.4 ± 3.8) than heterozygotes (10.9 ± 5.1, p < 0.0001) and compound heterozygotes (13.8 ± 3.5, p = 0.001). A total of 160 conventional implants were placed. Seven implants were lost in four patients before final prosthesis placement, resulting in overall failure rates of 14.8% at the subject level and 4.4% at the implant level. The 5-year cumulative survival rate (range: 0.23-12.74 years) was 85.2% and 95.6% at the subject and implant levels, respectively. The mean marginal bone loss (MBL) was 0.80 mm (95% CI: 0.59-1.02). Subgroup analysis revealed higher MBL in homozygotes (p = 0.002), sites requiring extensive bone augmentation (p = 0.016), anterior regions (p = 0.031), and full-arch bridge cases (p < 0.0001). Patient satisfaction and OHIP-C49 scores were favorable. Implant rehabilitation is predictable for patients with WNT10A mutations. However, increased MBL observed in homozygotes with severe tooth agenesis and in cases requiring extensive alveolar bone augmentation highlights the importance of genetic diagnosis prior to implant therapy in patients with severe tooth agenesis.
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