A 64 year old male patient, farmer by profession, reported to outpatient department of Oral Medicine, Diagnosis and Radiology with a chief complaint of pain and pus discharge in relation to the left upper back tooth region since 4 months. Past history revealed that patient was asymptomatic 4 months ago, later he developed pain and pus discharge which was slow in onset, localized, dull in nature, intermittent with no aggravating or relieving factors. Further dialog history revealed that patient also experienced numbness on left side of the face since 2 months. Past medical history revealed that patient was hypertensive and diabetic since 5 and 4 years respectively and currently on medication. Past dental history and supportive investigations revealed that patient underwent check up 4 years ago at a ENT doctor where it was provisionally diagnosed as a case of Nasal myiasis, surgical debridement of the lesion was done and maggots were retrieved. Plain and contrast computed tomography of brain revealed a sharply defined, mildly hyperdense space occuping lesion of 24 x 19 mm in left temporal region and effaced sulcal spaces which showed mild enhancement on contrast with mild hyperostosis with widening of diploic spaces of greater wing of sphenoid bones which was suggestive of a meningioma. Following which patient was admitted at a Private dental hospital, treatment protocol was informed to the patient and after obtaining informed consent from the patient, he was posted for surgery under general anesthesia. Maxillectomy on the left side was done, after 2 months of regular debridements and obtaining sterile cultures from the surgical wound, acrylic palatal obturator was fabricated making alginate impressions. Extra oral examination revealed a gross facial asymmetry with sunken appearance with loss of normal nasolabial fold on the left side of the face (Figure 1). TMJ and lymph nodes examination showed no abnormality. Intra oral examination showed a surgical defect in relation to the left maxillary quadrant with well defined borders. The depth of the lesion showed a grayish black necrotic pseudo membranous slough with yellowish areas interspersed all over (Figure 2). Other intra oral findings were multiple missing teeth in left and right maxilla and periodontally compromised mandibular teeth. Based on the past history and intra oral examination, a provisional diagnosis of osteomyelitis of the surgical defect was given. Other lesions such as Tuberculosis, Squamous cell carcinoma, Tertiary syphilis, fungal infections such as Aspergillosis and Mucormycosis were considered.
Radiologic investigations of Panaromic radiograph and computed tomography were advised. Panaromic radiograph revealed multiple missing teeth (12 11 21 22 23 24 25 26 27 37) and huge surgical defect in favor of left maxillectomy (Figure 3). Paranasal sinus radiograph view showed radiolucency which is diffuse, with irregular borders involving the left maxillary antrum and deviated nasal spine and changes in favor of left maxillectomy (Figure 4). Computed axial tomography revealed in favor of post operative left maxillectomy, deviated nasal spine to right, left ethmoidal and sphenoidal sinusitis. Changes seen involving left pterygoid plates and posterior wall of maxillary antrum suggestive of residual changes of Osteomyelitis (Figure 5). Biochemical investigations such as complete blood picture, serum creatinine were within normal limits with increased Erythrocyte sedimentation rate and Glycoslyated hemoglobin was 9.2% suggestive of diabetes mellitus. Incisional biopsy was done and the specimen was obtained from the left maxillary antrum through endoscope under local anesthesia and was sent to Culture sensitivity tests which showed positivity for bacterial and fungal growth on KOH culture media. The specimen was further sent for Periodic acid Schiff staining which showed large non septate fungal hyphae branched at right angles (Figure 6). The specimen was sent for histopathological examination (Figure 7) which showed cellular connective tissue stroma composed of mixed inflammatory cell infiltrate predominantly neutrophils and lymphocytes, numerous large non septate fungal hyphae branching at right angle along with few areas of necrosis. There is presence of ciliated columnar epithelium along with few bony trabaculae and extensive areas of hemorrhage along with super added bacterial infection suggestive of "mucormycosis. Correlating clinically, radiologically and histologically a final diagnosis of CHRONIC OSTEOMYEILITIS WITH MUCORMYCOSIS was given. Our patient underwent extensive unilateral endoscopic nasosinusal surgery with debridement of left maxillary sinus, anterior and posterior ethmoidal cells and extensive removal of invaded mucosa under general anesthesia. Extensive fungal masses and necrotic slough were excised from left maxillary and sphenoidal sinus. Necrotic mucosa was removed till the healthy margins were visible which showed initial signs of bleeding. Liposomal Amphotericin B (5 mg/kg) was started immediately after the surgery and dosage was increased to (7.5 mg/kg) after 3 weeks. A week later patient underwent hyperbaric oxygen therapy for 21 dives, each session lasting for 150 min along with systemic delivery of antifungal drugs orally for time period of 4 weeks. Post operative visit of the patient showed negative cultures with no morbidity and patient was delivered an acrylic obturator with soft liners.