Adult ; Diagnostic Errors ; Histiocytosis, Sinus ; diagnosis ; Humans ; Male ; Rhinoscleroma ; diagnosis

Rhinoscleroma disease accompanied with laryngotracheal stenosis as a main clinical manifestation is seldom. Here one case is reported. At the beginning, the patient had pharyngalgia and his swallowing was not smooth. After that, he had difficulty in breathing. Surgical therapy, antibiotics treatment and nutritional support were performed. The patient was cured.
Constriction, Pathologic ; Deglutition ; Humans ; Larynx ; physiopathology ; Male ; Respiration ; Rhinoscleroma ; complications ; diagnosis ; therapy ; Trachea ; physiopathology

Scleroma of the larynx is a rare specific granulomatous disease and is usually presented as alaryngeal extension of a primary rhinoscleroma. We report an unusual case of laryngeal scleromain a 64-year-old female patient whose presented with progressive upper respiratory obstructivesymptoms lasted for 2 years. Symptoms were acutely exaggerated during the course of one month.On physical examination, the nasal mucosa was atrophic and crusted, and septal perforationwas noted. The fiberoptic laryngoscopy revealed a yellowish granulated epiglottic mass with adirty surface mimicking laryngeal cancer. Histological sectioning showed the characteristic featuresof scleroma including Mikulicz cells with scattered plasma cells and some neutrophils.Warthin-starry stains and electron microscopy examination showed numerous short rods insidethe cytoplasm of the Mikulicz cells. After the diagnosis, the patient underwent an emergency tracheostomyfollowed by long-term antibiotic treatment, and her symptoms have slowly improved.
Coloring Agents ; Cytoplasm ; Diagnosis ; Emergencies ; Female ; Humans ; Laryngeal Neoplasms ; Laryngoscopy ; Larynx* ; Microscopy, Electron ; Middle Aged ; Nasal Mucosa ; Physical Examination ; Plasma Cells ; Rhinoscleroma

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.
Endemic Diseases ; Fibrosis ; Human ; Klebsiella pneumoniae/*isolation & purification ; Korea ; Male ; Microscopy, Electron ; Middle Aged ; Nasal Mucosa/*microbiology/pathology/ultrastructure ; Rhinoscleroma/*pathology

Relapsing polychondritis (RP) is a rare autoimmune connective tissue disease of unclear pathogenesis and may present with the multisystem involvements and varied presentations, courses and responses to the therapy. The respiratory complaints or history such as dyspnea or snoring deserve to be in consideration, especially in the cases of no specific causes and must be ruled out the rare autoimmune airway diseases such as relapsing polychondritis, Wegener's granulomatosis, rhinoscleroma, or amyloidosis resulting in serious airway obstruction without adequate management. In this report we described a case of 15-year-old young male with the delayed diagnosis of relapsing polychondritis with intractable subglottic stenosis during the course of the management for several months despite of tonsillectomy for the relief of the airway obstruction.
Adolescent ; Airway Obstruction ; Amyloidosis ; Connective Tissue Diseases ; Constriction, Pathologic ; Delayed Diagnosis ; Dyspnea ; Humans ; Male ; Polychondritis, Relapsing ; Rhinoscleroma ; Snoring ; Tonsillectomy ; Wegener Granulomatosis

Antigens, CD20 ; metabolism ; Diagnosis, Differential ; Humans ; Infectious Mononucleosis ; metabolism ; pathology ; Leukocyte Common Antigens ; metabolism ; Lymphoma, Extranodal NK-T-Cell ; diagnosis ; metabolism ; pathology ; Nose Neoplasms ; diagnosis ; metabolism ; pathology ; Oral Ulcer ; metabolism ; pathology ; RNA, Viral ; metabolism ; Rhinoscleroma ; metabolism ; pathology

OBJECTIVETo study the prevalence of IgG4-positive plasma cells in inflammatory disease of nasal cavity and paranasal sinuses and its association with IgG4-related sclerosing disease (IgG4-SD).

METHODSThe expression of IgG4 and IgG in plasma cells of 103 cases diagnosed as inflammatory disease of nasal cavity and paranasal sinuses with dense lymphoplasmacytic infiltrate was studied by immunohistochemistry (EnVision) and quantitatively analyzed by medical image analysis system.

RESULTSImmunohistochemical study showed marked infiltration by IgG4-positive plasma cells (>50 per high-power field) in 28 cases, moderate infiltration (30 to 50 per high-power field) in 23 cases, mild (10 to 29 per high-power field) in 30 cases and negative (<10 per high-power field) in 22 cases (P < 0.05). Twenty-two cases studied fulfilled the diagnostic criteria of IgG4-SD (>50 IgG4-positive plasma cells per high-power field and IgG4-to-IgG ratio > 40%), including 3 cases of chronic sinusitis (3/20), 3 cases of nasal polyps (3/18), 3 cases of inflammatory pseudotumor (3/17), 4 cases of fungal sinusitis (4/20), 1 case of rhinoscleroma (1/12), 7 cases of Wegener's granulomatosis (7/11) and 1 case of Rosai-Dorfman disease (1/2).

CONCLUSIONInflammatory disease of nasal cavity and paranasal sinuses fulfilling the diagnostic criteria IgG4-SD is not uncommon. Definitive diagnosis of IgG4-SD requires correlation with other clinical and laboratory findings. Some cases of unexplained inflammatory disease of nasal cavity and paranasal sinus may represent a member of the IgG4-SD spectrum. IgG4 carries diagnostic value in differential diagnosis of inflammatory disease occurring in nasal cavity and paranasal sinuses.

Adolescent ; Adult ; Aged ; Aged, 80 and over ; Diagnosis, Differential ; Female ; Granuloma, Plasma Cell ; metabolism ; pathology ; Granulomatosis with Polyangiitis ; metabolism ; pathology ; Histiocytosis, Sinus ; metabolism ; pathology ; Humans ; Immunoglobulin G ; metabolism ; Immunohistochemistry ; Male ; Middle Aged ; Nasal Cavity ; immunology ; pathology ; Nasal Polyps ; metabolism ; pathology ; Nose Diseases ; immunology ; pathology ; Paranasal Sinus Diseases ; immunology ; pathology ; Paranasal Sinuses ; immunology ; pathology ; Plasma Cells ; immunology ; Rhinoscleroma ; metabolism ; pathology ; Sinusitis ; metabolism ; pathology ; Young Adult

OBJECTIVETo find a fast and simple method for detection of specific pathogens in upper aerodigestive tract.

METHODSSixty-one cases of specific infections in upper aerodigestive tract encountered during a 10-year period in Beijing Tongren Hospital were retrospectively studied. Six histochemical stains, including PAS, Giemsa, Gram, methylene blue, modified Warthin-Starry and acid-fast stains were applied. The morphology of different pathogens was studied and the staining patterns were compared.

RESULTSThere were 23 cases of pharyngeal treponemal infection, 10 cases of short treponemal infection, 4 cases of mycobacterial infection, 4 cases of infection by rhinoscleroma bacilli, 1 case of sinonasal fungal infection, 1 case of combined infection of bacteria and Oidium albicans, 2 cases of tonsillar Actinomycetes and 16 cases of non-specific bacterial infections. Both pharyngeal treponemal infection and infection by rhinoscleroma bacilli could be detected by modified Warthin-Starry stain. As for sinonasal fungal infection, PAS, Giemsa and modified Warthin-Starry stains were useful in differentiating different types of fungi. Mycobacteria were best demonstrated by conventional acid-fast stain.

CONCLUSIONSSpecial histochemical stains performed on histologic sections are useful for diagnosing specific infections in upper aerodigestive tract.

Actinomycosis ; microbiology ; pathology ; Adolescent ; Adult ; Aged ; Aged, 80 and over ; Child ; Female ; Humans ; Male ; Middle Aged ; Mycobacterium ; isolation & purification ; Mycobacterium Infections ; microbiology ; pathology ; Palatine Tonsil ; microbiology ; pathology ; Pharyngeal Diseases ; microbiology ; pathology ; Pharynx ; microbiology ; pathology ; Retrospective Studies ; Rhinoscleroma ; microbiology ; pathology ; Staining and Labeling ; Treponema ; isolation & purification ; Treponemal Infections ; microbiology ; pathology ; Young Adult

Adolescent ; Adult ; Aged ; Aged, 80 and over ; Antigens, CD ; metabolism ; Antigens, Differentiation, Myelomonocytic ; metabolism ; Child ; Diagnosis, Differential ; Female ; Follow-Up Studies ; Histiocytosis, Sinus ; diagnostic imaging ; metabolism ; pathology ; surgery ; Humans ; Male ; Middle Aged ; Nose Diseases ; diagnostic imaging ; metabolism ; pathology ; surgery ; Nose Neoplasms ; metabolism ; pathology ; Plasmacytoma ; metabolism ; pathology ; Recurrence ; Rhinoscleroma ; metabolism ; pathology ; S100 Proteins ; metabolism ; Tomography, X-Ray Computed ; Young Adult