(A204) Adalimumab: Aiding or Alarming in a Pediatric Chronic Recurrent Multifocal Osteomyelitis Patient with Sinusitis?

Introduction: Chronic recurrent multifocal osteomyelitis (CRMO) is an autoimmune disorder characterized by non-infectious osteolytic lesions and bone inflammation that predominantly presents in long bones of children. Rare cases of sinus osteomyelitis have been reported in patients taking adalimumab for CRMO.This case report and literature review describes a case of acute ethmoid sinusitis and osteomyelitis with intracranial extension in a pediatric CRMO patient with recent history of adalimumab use.

Methods: This is a case of sinonasal osteomyelitis presenting with headache and nasal bridge/lacrimal duct abscess observed within a 4-month period of a 13-year-old female discontinuing adalimumab treatment for CRMO. Interventions included functional endoscopic sinus surgery (FESS) and long term antibiotic therapy as operative cultures grew rare coagulase negative staphylococcus species and pathology results demonstrated viable bone. Outcomes were recorded based on resolution of infection at the time of discharge and clinical improvement monitored with serial examinations and radiologic imaging. A literature review was also performed which found a report of similar cases of CRMO treated with adalimumab in two 11-year-old female patients, both resulting in adalimumab-related sinonasal complications.

Results: The clinical improvement after treatment with FESS, antibiotics, as well as continued avoidance of adalimumab post-surgery, leads us to believe that our patient suffered from immunocompromised sinusitis related to adalimumab use, leading to an advanced presentation of our patient’s symptoms. These results are consistent with that of the cases noted in our literature review, as they similarly resolved with sinus surgery and antibiotics. Since cranial CRMO involvement is extremely rare, it is a strong possibility that adalimumab was a culprit in our patient’s case presentation.

Conclusions: Adalimumab therapy in the setting of CRMO can be a precipitating factor in the development of neurocranial osteomyelitis and its related complications, and otolaryngologists should be aware of this possibility.