Efficacy and Toxicity of Calcitonin Treatment in Children with Cherubism: A Single-Center Cohort Study.

Cherubism is a rare autosomal dominant disease characterized by expansile osteolytic jawbone lesions. The effect and safety of off-label calcitonin treatment during the progressive phase of the disease are not well described. In this retrospective study, we present data on the radiological response and adverse effects of subcutaneously administered calcitonin in a cohort of nine cherubism children (three female, six male). Two of the nine patients underwent two separate treatment courses with a significant off-treatment interval in between; therefore, a total of 11 treatment courses with a mean duration of 17.9 months (range <1 to 35, SD 10.8) were studied. To measure the response, the cumulative volume of cherubism lesions was calculated from available three-dimensional imaging. The primary outcome was the change in the volume of lesions during calcitonin treatment and only assessed for the eight treatment courses with a minimal duration of 6 months. A statistically significant reduction in the mean cumulative volume of lesions was seen regardless of treatment duration. Average volume reduction was highest in the first half year of treatment, with a gradual, ongoing reduction thereafter. For the secondary outcome, the change in the cumulative volume of lesions after treatment cessation was assessed for the seven treatment courses with follow-up imaging available. After six of these seven treatment courses, the cumulative volume increased again but remained undoubtedly smaller than the initial volume at the start of therapy. Adverse effects were assessed for all 11 treatment courses and occurred in 73% of them. Most adverse effects were mild and low grade, with the most severe being one grade 3 symptomatic hypocalcemia requiring hospitalization and early treatment termination. Calcitonin treatment seems effective and tolerable in treating actively progressing cherubism in children. However, further research is required to better understand the pharmacological treatment of cherubism, including also other drugs, dosing, and protocols. © 2023 The Authors. Journal of Bone and Mineral Research published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research (ASBMR).

Subjective alveolar nerve function after bilateral sagittal split osteotomy or distraction osteogenesis of mandible.

PURPOSE: The present retrospective cohort study compared the subjective inferior alveolar nerve (IAN) function after distraction osteogenesis (DOG) and bilateral sagittal split osteotomy (BSSO) in mandibular advancement surgery. MATERIALS AND METHODS: Treatment consisted of correction of a retrognathic mandible using DOG (30 patients) or BSSO (35 patients). Subjective IAN function was recorded using a questionnaire 1 year after surgery. A total of 130 IANs were evaluated. RESULTS: In 37 nerves (28.5%), an IAN disturbance was observed. In this group of 37 nerves, BSSO had been performed in 26 (70.3%) and DOG in 11 (29.7%). After eliminating confounders (eg, age, amount of advancement, gender), no significant difference (odds ratio 0.652, 95% confidence interval 0.221 to 1.920) was found. Age was significantly related to subjective IAN disturbances for women but not for men. This was seen in women older than 22 years and increased for women older than 36 years to an odds ratio of 22.8 (95% confidence interval 2.580 to 201.488). Satisfaction also correlated with age, independent of gender. CONCLUSION: No difference was found in subjective IAN disturbances after 1 year between DOG and BSSO for lengthening the mandible.