Abstract Disclosure: J. Udayasankar: None. R. Vaezeafshar: None. N. Patel: None. A 61 year old female with uncontrolled type 2 Diabetes, A1c over 11% for over 3 years was seen in Diabetes clinic for follow up after a 2 year hiatus. She happened to mention an isolated 1 inch area of tingling over Right (R) cheek. She was previously seen in ED and discharged home on Benadryl. She had also seen an acupuncturist, a dentist and family physician, latter 2 providers treated her with 2 different antibiotics without change in symptoms. She denied fever, headaches, visual acuity changes, diplopia or nasal discharge. Exam in clinic revealed hypoesthesia below eye and towards nasal dorsum, no changes in skin warmth or color noted, eye movements were intact and mild tenderness was elicited on pressure over R maxillary sinus. Atypical presentation of Invasive fungal sinusitis (IFS) was suspected clinically. Seen by ENT and CT and MRI of Sinuses and Orbits showed R maxillary sinusitis, mild enhancement and enlargement of the inferior rectus muscle and infraorbital nerve / foramen present with stranding within the extraconal fat of the R inferior orbit thought to be related to extension of inflammation. Endoscopic sinus surgery showed R maxillary sinusitis with necrotic mucosa along orbital floor and orbital floor dehiscence concerning for invasive infection. Frozen section showed necrotic, inflammatory tissue. Discharged home from ED. Surgical pathology showed necrotic tissue and fungal hyphae with acute angle branching on GMs and PAS stains. Presence of crystals and pigmentation raised possibility of Aspergillus Niger infection. She was readmitted to hospital for repeat surgery - R medial maxillectomy and R pterygopalatine fossa dissection - biopsy of R maxillary sinus walls, right periorbital area and pterygopalatine fossa showed presence of fungal organism. Mold PCR panel showed Aspergillus niger. She received therapy with IV Amphotericin B and was discharged home on Oral Voriconazole. She is doing well and her diabetes control improved on insulin therapy. Patients with uncontrolled Diabetes are immunocompromised and vulnerable to atypical infections that can be potentially lethal if not diagnosed and treated timely. Patient presented with isolated tingling over R cheek for months. Lack of symptom improvement despite two courses of antibiotics and A1c over 11% raised clinical suspicion of IFS . Interestingly frozen path did not reveal hyphae that led to her first discharge from hospital. Surgical pathology and PCR of biopsied material made the diagnosis of IFS - Aspergillus sinusitis and treatment with antifungal medication prevented orbital and cerebral complications of IFS and death. Therefore, awareness of this disease entity in ambulatory patients is important and could be life saving for patients. Presentation: Thursday, June 15, 2023
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