Chronic Laryngotracheal Granulomatous Disease Secondary to Sporothrix schenckii in an Immunocompromised Patient

Abstract

Objective: To describe a rare presentation of laryngotracheal granulomatous disease secondary to sporotrichosis. Methods: The authors report a case of laryngeal sporotrichosis in an immunocompromised patient, with accompanying endoscopic images and pathology. Results/case: A 72-year-old immunocompromised female with a history of rose-handling presented with a year of hoarseness and breathy voice. Flexible nasolaryngoscopy showed diffuse nodularity; biopsy of the lesions demonstrated granulomatous inflammatory changes, and fungal culture grew Sporothrix schenkii. Long-term itraconazole treatment was initiated, with improvement in dysphonia and few residual granulomas on follow-up examination. Conclusion: When evaluating granulomatous disease of the airway, a broad differential including infectious or inflammatory etiologies should be considered, especially in immunocompromised patients. Adequate tissue samples should be collected to facilitate special staining. The current recommendations for laryngeal sporotrichosis include treatment with a prolonged course of itraconazole.

Epidemiology of Clinical Sporotrichosis in the Americas in the Last Ten Years

“…A total of 48 publications related to sporotrichosis were found in North America [ 5 , 6 , 7 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 ]. There were 1460 patients in total associated with infection caused by species of the genus Sporothrix .…”

“…According to the previous classification, it was found that in Canada, only two case reports were found, one from Ontario and the other from Toronto [ 5 , 6 ]. In the US, 27 reports containing 1 clinical case were found (81.5% S. schenckii , 18.5% Sporothrix spp., S. schenckii complex, and S. schenckii sensu lato ) [ 7 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 ]. Of these, seven cases came from California, three from Oklahoma, two cases from Kansas, Texas, Arizona, Minnesota, and Florida, one case from Michigan, Nebraska, Oregon, Pennsylvania, and finally, one case without a specific city or region.…”

“…In the US, it was reported that Sporothrix spp. (85.19%) and S. schenckii (14.81%) were responsible for this pathology [ 7 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 ]. Finally, in Mexico, 85.05% were due to Sporothrix spp., 14.33% S. schenckii , 0.55% S. globosa, and 0.07% S. mexicana [ 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 ].…”

“…The most frequent variety reported was lymphocutaneous with 956 cases, followed by fixed cutaneous with 388 cases, and the disseminated form with 83 [ 5 , 6 , 7 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 ]. A lymphocutaneous presentation evolved into a disseminated after 10 months.…”

“…The most common reported etiological agent with the new taxonomical classification was Sporothrix spp. with 85.00% (1241/1460), followed by S. schenckii with 14.38% (210/1460), S. globosa with 0.54% (8/1460), and S. mexicana with 0.068% (1/1460) [ 5 , 6 , 7 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 ].…”