Temporal bone histiocytosis in children: a systematic review.

OBJECTIVE

To analyze the diagnosis, treatment, and outcomes of temporal bone histiocytosis (TBH) in children.

METHODS

A systematic review was conducted based on PRISMA guidelines. A literature review on the databases PubMed and Embase was conducted. Risk of bias was evaluated using the MINOR Score.

RESULTS

Eight retrospective studies with 84 subjects were included. The mean age was 3 years with a prevalence of male. Clinical features comprehend otorrhea, otalgia, recurrent external and media otitis, postauricular swelling, dermatitis, and external ear canal polyps/mass. 52/84 (62 %) children had single system involvement, and most extra-temporal bone involvement was pituitary gland with diabetes insipidus. CT scan revealed osteolytic lesions without surrounding sclerosis and punched-out lesions centered in the squamous part. The subsites most involved were: mastoid, external and middle ears and temporal squama, less frequently optic capsule and petrous apex. One-third of cases had bilateral involvement. MRI was helpful for detecting intracranial lesions. Hearing loss was conductive (20 cases), sensorineural (4) and mixed (5). Treatment strategies included: CT and RT, CT alone, surgery and RT, and surgery and CT. The overall recurrence rate was 20.2 % (17/84). The duration of follow-up ranged from 6 weeks to 18 years. Temporal bone complications were facial palsy, cholesteatoma, and chronic dermatitis.

CONCLUSION

Management of temporal bone histiocytosis in children requires a multidisciplinary approach. The role of otolaryngologist is crucial for the early diagnosis and surgical treatment of selected cases. A long-term follow-up is necessary to detect recurrences and otological complications.