An Unusual Case of Disseminated Blastomycosis Presenting with Hoarseness and Odynophagia

Abstract

Introduction: Blastomycosis is a systemic infection that most commonly involves the lungs. Extrapulmonary dissemination usually affects the skin, bone, genitourinary tract, and central nervous system, and results from hematogenous spread in up to 40% of cases. Rarely, blastomycosis can involve the upper airways and is presumed to result from direct inoculation of the mucosa via inhalation. We report a case of disseminated blastomycosis in a patient presenting with hoarseness and odynophagia due to laryngeal infection, the most common site of head and neck involvement. Case Presentation: A 74-year-old man presented with a 1-month history of hoarseness and odynophagia. This was associated with a 20-pound weight loss due to anorexia. Medical comorbidities were significant for diabetes mellitus, ulcerative colitis, and ESRD, status post kidney transplant (8 years prior to presentation), on immunosuppression therapy consisting of mycophenolate mofetil, prednisone, and tacrolimus. Physical exam was remarkable for tenderness on palpation of the left side of the neck and crusted nodules on the left thigh and palm. Blood tests revealed leukocytosis with neutrophilic predominance and hypercalcemia. Chest roentgenogram showed pulmonary interstitial prominence without focal consolidations. Influenza, SARS-CoV-2 and Streptococcus group A PCRs were negative. CT scan of the neck revealed asymmetric soft tissue fullness near the laryngeal vestibule. CT scan of the chest revealed extensive milliary pattern of lung nodules and diffuse interstitial and groundglass opacities. Laryngoscopy demonstrated a mass arising from the left arytenoid and aryepiglottic fold. Biopsies revealed necrotizing acute inflammation with numerous uniform large yeast with broad-based budding, double contour wall and visible nuclei, consistent with Blastomyces. MRI of the brain was negative for involvement. Prior to the biopsies, diagnostic workup was notable for negative serum (1, 3) beta-D-glucan, QuantiFERON-TB Gold Plus, Cryptococcus antigen, and blastomyces and Histoplasma antibodies by immunodiffusion. Sputum samples were negative although the fungal cultures grew filamentous fungus. Histoplasma urine antigen was positive. The patient was initiated on liposomal amphotericin B and itraconazole with plan to convert to itraconazole monotherapy to complete a 12 month course. Discussion: Blastomycosis is an uncommon disease caused by the inhalation of the conidia of Blastomyces dermatitidis or Blastomyces gilchristii. Extrapulmonary dissemination is more common in immunocompromised patients with increased risk for severe pulmonary disease, including respiratory failure and ARDS, and a higher mortality rate. Lifelong suppressive antifungal therapy is generally not required following appropriately treated blastomycosis.