Innovative multimodal management in an aneurysmal bone cyst of the temporal bone: our experience in a 3-month-old infant and literature review.

[PURPOSE] Aneurysmal bone cysts (ABCs) are benign but locally aggressive osteolytic lesions. In pediatric patients, cranial involvement is uncommon, and the temporal bone (TB) represents an exceptionally rare site of disease. Owing to their behaviour and proximity to critical neurovascular structures, the diagnosis and management of TB-ABCs remain particularly challenging for both neurosurgeons and otolaryngologists.

[METHODS] We report a case of primary TB-ABC in a 3-month-old infant, managed using a stepwise multimodal strategy including preoperative embolization and Denosumab administration to facilitate subsequent definitive surgical resection. To the best of our knowledge, this patient represents the youngest reported case of TB-ABC and the first pediatric TB-ABC managed using a multimodal approach including Denosumab. In addition, a systematic literature review was conducted on this intriguing topic.

[RESULTS] The literature review identified ten pediatric TB-ABCs treated surgically. Preoperative embolization was performed in four patients, while no adjuvant medical therapy was reported. No recurrences were observed over a mean follow-up period of 12 months. Persistent hearing loss due to inner-ear fistulation was reported in two patients. In contrast, our patient underwent a multimodal treatment strategy combining preoperative embolization and Denosumab therapy, which resulted in a marked reduction in lesion volume. This approach facilitated a safer subsequent gross total resection while preserving hearing and facial nerve function. Postoperative radiological follow-up showed no evidence of recurrence.

[CONCLUSION] Pediatric TB-ABCs are exceedingly rare and potentially dangerous lesions. Preoperative embolization combined with targeted medical therapy may represent a useful adjunct to reduce intraoperative bleeding and facilitate gross total resection in selected cases. Given the rarity of this condition, a multidisciplinary team comprising otolaryngologists, neurosurgeons, pediatric oncologists, and neuroradiologists is essential to optimize clinical outcomes.