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

Aged ; Antigens, CD ; metabolism ; Antigens, Differentiation, Myelomonocytic ; metabolism ; Diagnosis, Differential ; Female ; Follow-Up Studies ; Histiocytosis, Sinus ; pathology ; surgery ; Humans ; Immunoglobulin G ; blood ; Paranasal Sinus Diseases ; pathology ; surgery ; Paranasal Sinuses ; pathology ; S100 Proteins ; metabolism ; Sclerosis ; pathology

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

OBJECTIVETo study the clinicopathologic features and differential diagnosis of extranodal Rosai-Dorfman disease (RDD) of the upper respiratory tract.

METHODSThe clinical, pathologic and immunohistochemical features of 10 cases of RDD were evaluated.

RESULTSAmong the 10 cases studied, there were 3 males and 7 females. The age of patients ranged from 20 to 61 years old (mean 38 years). The lesion arose in the nasal cavity (7 cases), nasopharynx (2 cases) or hard palate to trachea (1 case). Most of the patients presented with nasal obstruction, rhinorrhagia or tumor mass in the nasal/nasopharyngeal regions. CT scan often showed the presence of soft tissue lesion without bone destruction. Histologically, extranodal RDD was characterized by light-staining bands alternating with dark-staining bands. The light-staining bands were formed by aggregates of large round or polygonal histiocytes with emperipoiesis. The dark-staining bands were formed by abundant lymphoplasmacytic infiltrates. Immunohistochemical study showed that the histiocytes strongly expressed S-100 protein and partially expressed CD68. Six patients had no recurrence after surgical resection.

CONCLUSIONSExtranodal RDD of the upper respiratory tract is a rare disorder of histiocytic proliferation, which usually involves the nasal cavity and paranasal sinuses. RDD can easily mimic rhinoscleroma, mainly due to the overlapping morphologic appearance. Immunohistochemical study is helpful in the differential diagnosis.

Adult ; Antigens, CD ; metabolism ; Antigens, Differentiation, Myelomonocytic ; metabolism ; Diagnosis, Differential ; Female ; Follow-Up Studies ; Histiocytic Sarcoma ; metabolism ; pathology ; Histiocytosis, Langerhans-Cell ; metabolism ; pathology ; Histiocytosis, Sinus ; diagnostic imaging ; metabolism ; pathology ; surgery ; Humans ; Male ; Middle Aged ; Nasal Cavity ; pathology ; Nasopharyngeal Diseases ; diagnostic imaging ; metabolism ; pathology ; surgery ; Neoplasms, Muscle Tissue ; metabolism ; pathology ; Nose Diseases ; diagnostic imaging ; metabolism ; pathology ; surgery ; S100 Proteins ; metabolism ; Tomography, X-Ray Computed ; Young Adult

Sinus histiocytosis with massive lymphadenopathy (SHML) is a rare disorder characterized by a nonneoplastic proliferation of distinctive histiocyte cells within lymph node sinuses and lymphatics in extranodal sites. SHML occurs worldwide and is primarily a disease of childhood and early adulthood. A 26-yr-old man presented with painless palpable lymph node in cervical area. Radiographic studies revealed pleural effusion with lymphadenopathy and calcification in mediastinum. The cervical lymph node biopsy showed dilated sinuses filled with histiocytes with clear cytoplasm. The cells stained positive with CD68 and S-100. These cytologic and immunohistochemical findings were considered consistent with the diagnosis of SHML.
Adult ; Antigens, CD/metabolism ; Antigens, Differentiation, Myelomonocytic/metabolism ; Histiocytes/pathology ; Histiocytosis, Sinus/*diagnosis/metabolism/pathology ; Humans ; Lymph Nodes/pathology ; Male ; Neck ; Pleural Effusion/*radiography ; S100 Proteins/metabolism ; Tomography, X-Ray Computed

Adult ; Antigens, CD ; metabolism ; Antigens, Differentiation, Myelomonocytic ; metabolism ; Carcinoma, Giant Cell ; metabolism ; pathology ; Diagnosis, Differential ; Granuloma, Plasma Cell ; metabolism ; pathology ; Histiocytosis, Sinus ; metabolism ; pathology ; surgery ; Humans ; Immunohistochemistry ; Lung Diseases ; metabolism ; pathology ; surgery ; Lung Neoplasms ; metabolism ; pathology ; Male ; Pneumonectomy ; methods ; S100 Proteins ; metabolism ; Vimentin ; metabolism

Diagnosis, Differential ; Histiocytoma, Benign Fibrous ; metabolism ; pathology ; Histiocytosis, Langerhans-Cell ; metabolism ; pathology ; Histiocytosis, Sinus ; metabolism ; pathology ; surgery ; Humans ; Infant ; Male ; S100 Proteins ; metabolism ; Xanthogranuloma, Juvenile ; metabolism ; pathology

Antigens, CD ; metabolism ; Antigens, CD1 ; metabolism ; Antigens, Differentiation, Myelomonocytic ; metabolism ; Diagnosis, Differential ; Histiocytosis, Langerhans-Cell ; metabolism ; pathology ; Histiocytosis, Sinus ; pathology ; Humans ; Infant ; Langerhans Cells ; pathology ; Lymph Nodes ; pathology ; Lymphohistiocytosis, Hemophagocytic ; pathology ; Male ; S100 Proteins ; metabolism

Adult ; Diagnosis, Differential ; Female ; Histiocytoma, Malignant Fibrous ; immunology ; pathology ; Histiocytosis, Langerhans-Cell ; immunology ; pathology ; Histiocytosis, Sinus ; immunology ; pathology ; Hodgkin Disease ; immunology ; pathology ; Humans ; Ki-1 Antigen ; metabolism ; Lewis X Antigen ; metabolism ; Lung ; immunology ; pathology ; Lung Neoplasms ; immunology ; pathology ; Lymph Nodes ; immunology ; pathology

OBJECTIVETo investigate the diagnosis and differential diagnosis of extranodal Rosai-Dorfman disease.

METHODSTwo cases of extranodal Rosai-Dorfman disease were studied using hematoxylin-eosin, and immunohistochemical staining, along with a literature review.

RESULTSThe lesions of RDD were characterized by the presence of large histiocytes with emperipolesis, accompanied by infiltration of lymphocytes, plasma cells and other inflammatory cells. The large histiocytes had an abundant cytoplasm, pale to eosinophilic in appearance, positive for S-100 protein staining, with a vesicular nucleus and a small basophilic nucleolus in each cell.

CONCLUSIONSExtranodal Rosai-Dorfman disease is known as an idiopathic proliferative disease of histiocytes with a distinct morphologic feature and is very rare. Differential diagnosis from other types of fibrohistiocytic proliferation lesions is recommended.

Antigens, CD ; metabolism ; Antigens, Differentiation, Myelomonocytic ; metabolism ; Brain ; pathology ; surgery ; Brain Diseases ; metabolism ; pathology ; surgery ; Dermatologic Surgical Procedures ; Diagnosis, Differential ; Histiocytosis, Sinus ; metabolism ; pathology ; surgery ; Humans ; Male ; Middle Aged ; S100 Proteins ; metabolism ; Skin ; pathology ; Skin Diseases ; metabolism ; pathology ; surgery