Why did a healthy non-smoker in his 60s have worsening hoarseness and shortness of breath over the past year? This is the diagnostic challenge faced by Kenneth W. Altman, MD, of Geisinger Medical Center, Danville, PA, and colleagues in a case reported by JAMA Otolaryngology Head and Neck Surgery .
At presentation, the patient stated that he did not have any symptoms of sore throat, cough, dysphagia, or reflux, nor did he have a fever or unexplained weight loss. He told clinicians that he had never smoked his entire life, had not undergone any head or neck surgery, and had not been exposed to radiation. His medical records show that his primary care physician had prescribed inhaled corticosteroids for patients with suspected asthma, but the treatment did not improve the patient’s symptoms.
Physical examination revealed that the patient was hoarse and hoarse. However, examination of his neck did not reveal anything unusual. The clinician performed a video stroboscopic examination and found that he had severe swelling of the laryngeal mucosa, mainly affecting the false vocal cords, interaryngeal and posterior annular areas. The patient’s right true vocal cord was immobile, with compensatory extrusion of the false vocal cord, and no mucosal waves.
Enhanced CT scan of the neck showed irregularity of the free edge of the true vocal cords. However, there were no signs of a tumor or swollen lymph nodes in the neck.
They performed urgent direct laryngoscopy, biopsy and modified barium swallow study, which showed aspiration. Microscopically, the patient’s throat mucosa was swollen, irregular, and fragile, and the airway was significantly constricted. Given the risk of airway obstruction, clinicians performed a tracheostomy during direct laryngoscopy and placed a gastrostomy tube to address the risk of aspiration.
Hematoxylin-eosin staining of biopsy specimen showed numerous epithelioid histiocytes and giant cells with a background of lymphocytes and necrosis, findings consistent with necrotizing granulomatous inflammation . Staining for acid-fast bacteria returned negative results, and Grocott methylamine-silver staining showed extensive phagocytosis of budding yeast by multinucleated giant cells. Based on these findings, clinicians initially diagnosed blastomycosis. PCR testing confirmed the presence of this rare fungal infection.
Patient received long-term treatment with itraconazole. At the 2-month follow-up, the clinician noted that the patient’s voice was less hoarse and that he was able to tolerate the tracheostomy capping procedure. A follow-up modified barium swallow study showed normal swallowing function to return, so his gastrostomy tube was removed.
At 5-month follow-up, repeat video stroboscopic examination showed a decrease in supraglottic edema with increased mobility of the true vocal cords, and eventually, the tracheostomy tube was removed .
report this rarity Clinicians of the cases of laryngeal blastomycosis noted that “diagnosis requires a high degree of suspicion, as symptoms and laryngoscopy findings may resemble many different pathologies, such as carcinoma, tuberculosis, and sarcoidosis.” The team cautioned that the data suggest that, This invasive fungal infection of the throat “is often misdiagnosed, leading to delayed care and unnecessary treatments such as laryngectomy, radiation therapy, steroids and anti-TB drugs”.
First described by Gilchrist in 1894, the case authors explained that when aerosolized conidia from soil destruction are inhaled, “Blasmoderma dermatitidis is a dimorphic fungus that Grows as mold in the environment and as yeast in the human body”. The fungus is most likely to grow in the Great Lakes and Mississippi-Ohio Valley areas.
According to the CDC, in five states where blastomycosis can be reported — Arkansas, Louisiana, Michigan, Minnesota and Wisconsin — The annual incidence is approximately 1 to 2 cases per 100,000 people. Of those states, Wisconsin is thought to have the highest infection rate, ranging from 10 to 40 cases per 100,000 people per year in some northern counties.
Most commonly affected, blastobacterial infections may also involve the skin, bone, genitourinary and central nervous systems, they wrote, adding that the first report Cases of laryngeal blastomycosis date back to 1918.
Typically, blastomycosis initially presents as a “hidden hoarseness,” followed by symptoms such as coughing, shortness of breath, pain, or difficulty swallowing, they noted. However, infection is asymptomatic in about 50% of cases.
Few data have been published to guide the management of this condition, but treatment usually consists of laryngoscopy and tissue biopsy stains and cultures, the case authors wrote. “Appropriate tissue sampling is important because if biopsy is inadequate, fungal burden may be low,” they noted, and histological studies showed “nonspecific inflammatory infiltrates and reactive surface epithelial changes.”
Clinically, infection may manifest as inflammatory changes including granulomas, polymorphonuclear leukocyte aggregates, epithelioid histiocytes, and multinucleated giant cells.
Regarding diagnostic histopathology, the case authors explain that “Blastobacteria are better visualized on periodate-Schiff and Grocott silver stains.”
Isolation of blastocysts in culture can provide a definitive diagnosis within a few weeks, although the real-time PCR authors note that assays provide a faster diagnosis. The infection is usually controlled with 6 to 12 months of itraconazole treatment and, in most cases, resolves symptoms.
Kate Kneisel is a freelance medical journalist based in Belleville, Ontario.
Altman reports relationships with Merck, Vindico, AXDEV and Lyra Site PI.
No other disclosures reported.