Nasal Rhinoscleroma in a Nonendemic Area: A Case Report

Rhinoscleroma is a chronic, granulomatous infectious lesion most frequently affecting the respiratory tract mucosa. This disease is endemic to Africa, Central and South America, South Central and Eastern Europe, the Middle East, and China. We report an extremely rare disease of nasal rhinoscleroma in Korea. The patient was a 63-yr-old man, who suffered from chronic nasal obstruction and septal deviation. Characteristic histology from a nasal biopsy was proven and special stains for bacteria were employed: periodic acid-Schiff, Warthin-Starry silver, Giemsa, and Gram stains. Isolation of Klebsiella rhinoscleromatis from a culture of nasal discharge was failed, but ultrastructural examination of numerous phagocyzed bacilli in the Mikulicz cells confirmed the diagnosis. The histiocytic nature of the Mikulicz cells was confirmed, using CD 68 and alpha-1 antitrypsin, and by the ultrastructural features of Mikulicz cells. Here, we emphasize the recognition of this rare entity in nonendemic regions, frequently leading to delayed diagnosis.

Rhinoscleroma: A case report

Rhinoscleroma, or scleroma, is a chronic, slowly progressing granulomatous disease involving the upper respiratory tract, especially the nasal vestibules, choanae, pharynx, and larynx. Almost certainly the causative agent is Klebsiella rhinoscleromatis. The disease occurs frequently in Eastern Europe, the Middle East, and parts of Latin America, but it is hard to find such case in Korea. We prisent a case of rhinoscleroma involving the nasal vestibule in a 18-year-old male who was admitted due to nasal obstruction for 5 years and epistaxis for 2 months. The resected specimen was an irregular polypoid mass with relatively firm consistency and measured 3 cm in the largest diameter. Microscopically, the lesion was characterized by extensive fibrosis and inflammatory cell infiltration. The infiltrates consisted of predominantly lumphocytes, plasma cells, foamy or granular histiocytes which were singly scattered or grouped in clusters. In the cytoplasm of the histiocytes, round slightly basophilic bodies were noted. Warthin-Starry satin showed short positive rods within the cytoplasm of the cell (Mikulicz cell). Electron microscopically, the cytoplasm of Mikulicz cells contained large, round or irregular shaped clear vacuoles in which numerous Klebsiella bacilli attached to the boundaries of the vaculoes were noted. With higher magnifications, the bacilli were seen as roung or rod-shaped organisms.