Burkholderia Cepacia Causes Frontal Mucopyocele with Anterior Cranial Fossa Extension: A Novel Case Report

Introduction: Burkholderia cepacia complex (Bcc) is a group of gram-negative bacilli that have rarely been isolated in the ear, nose and throat region in immunocompetent patients. Bcc show resistance to most available antibacterial drugs.   Case Report: We present the case of an immunocompetent ۳۱-year-old male reporting a pulsating headache with right supraorbital swelling associated with exophthalmos. A brain CT scan showed an expansive giant cystic lesion occupying the right frontal sinus, extending to the anterior cranial fossa. Management and outcome: drainage with the resecting of the floor of the frontal sinus from the orbital plate of the ethmoid bone to the nasal septum (Draf IIb) was performed with wide marsupialization of the mucopyocele. Polymerase chain reaction-restriction fragment length polymorphism (PCR-RFLP) analysis was used to identify the isolate. MRI ۱ and ۱۲ months after surgery showed complete lesion removal. The patient was followed for ۱۲ months with complete recovery of symptoms.   Conclusion: Paranasal sinuses disease with cranial expansion and orbital complications constitutes an emergency. For the first time in the literature, Bcc was isolated in the frontal sinus, extending into the anterior cranial fossa, in an immunocompetent patient. An endoscopic surgical approach with microbiological identification and management by appropriate antibacterial drug treatment seems to be the key to success.

Keywords:

Burkholderia cepacia complex , mucocele , Frontal sinus , Surgical Endoscopy , Culture Media