This study was performed to investigate the association of dental dysplasia with childhood cancer. We examined the occurrence of agenesis, microdontia, and enamel changes of permanent teeth in pediatric cancer survivors. Seventy-six patients with pediatric cancer and hematologic diseases were referred to our department for the first time from October 2005 to December 2019. Of these, 17 patients who presented for a checkup in our department fulfilled the study criteria and were analyzed. Clinical examinations and panoramic radiographs were performed to investigate dental dysplasia of permanent teeth. A total of 34 developmental abnormalities (18 missing teeth, 16 cases of microdontia) occurred in 5 patients. The patients' medical records were also analyzed to assess the relationships of specific types of dysplasia with the treatment duration and cumulative drug dose administered. All five pediatric cancer survivors had dental dysplasia; all five had tooth agenesis, and four had microdontia. All five patients were < 4 years of age and had undergone high-dose chemotherapy. In this study, childhood cancer survivors who received high-dose chemotherapy before age 4 years experienced the dental dysplasia, including tooth agenesis, microdontia. These findings highlight the need for careful dental monitoring and early intervention in pediatric patients undergoing high-dose chemotherapy. This research was conducted in accordance with the Declaration of Helsinki and approved by the Medical Ethics Committee of the Medical University of Kobe (protocol code No. B230066, 1 September 2023).
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