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Cytokine profiles contribute to understanding the pathogenic difference between Good syndrome and oral lichen planus: two case reports and literature review.

Maehara T, Moriyama M, Kawano S, Hayashida JN, Furukawa S, Ohta M, Tanaka A, Yamauchi M, Ohyama Y, Kiyoshima T, Nakamura S - Medicine (Baltimore) (2015)

Bottom Line: However, IL-4 and IL-17 were detected in OLP patients only.These results suggest that the pathogenesis of GS is different from that of OLP.GS is a unique inflammatory disorder characterized by dysfunction of Th2 and Th17 immune reactions via abnormal T-B cell interaction.

View Article: PubMed Central - PubMed

Affiliation: From the Section of Oral and Maxillofacial Oncology (TM, MM, SK, J-NH, SF, MO, AT, MY, SN); Section of Oral and Maxillofacial Surgery (YO); and Division of Maxillofacial Diagnostic and Surgical Sciences, Laboratory of Oral Pathology, Faculty of Dental Science, Kyushu University, Fukuoka, Japan (YK).

ABSTRACT
We described and analyzed the pathogenic difference between Good syndrome (GS) and oral lichen planus (OLP) in oral mucosa. Good syndrome (GS) is a rare disease characterized by B and T cell immunodeficiency associated with hypogammaglobulinemia and thymoma. GS patients frequently develop oral lichenoid lesions with lymphocytic infiltration beneath the basal layer. Oral lichen planus (OLP) is a chronic inflammatory disease of the oral mucosa characterized by destruction of basal cells by Langerhans cells, macrophages, and T lymphocytes. Although the histological features of the lesions of both diseases are very similar, the pathogenesis of GS in the oral mucosa remains unknown. In this study, we thus investigated the expression of infiltrating lymphocyte subsets (CD3, CD20, CD4, and CD8) and T helper (Th) cytokines including interferon (IFN)-γ (Th1 type), interleukin (IL)-4 (Th2 type), IL-17 (Th17 type), and IL-10 (regulatory T cell type) by immunohistochemistry in buccal mucosa specimens from 2 GS patients compared with 15 OLP patients. All patients showed a predominance of CD3 T cells over CD20 B cells, and CD4 Th cells over CD8 cytotoxic T cells. This polarization was especially prominent in GS. IFN-γ and IL-10 were strongly detected in the infiltrating lymphocytes of all patients. However, IL-4 and IL-17 were detected in OLP patients only. These results suggest that the pathogenesis of GS is different from that of OLP. GS is a unique inflammatory disorder characterized by dysfunction of Th2 and Th17 immune reactions via abnormal T-B cell interaction.

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Related in: MedlinePlus

Clinical findings in oral cavity from patient with Good syndrome (GC) (Case 1). (A) Oral mucosa and tongue at the initial visit. (B) Oral mucosa and tongue after thymectomy.
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Figure 1: Clinical findings in oral cavity from patient with Good syndrome (GC) (Case 1). (A) Oral mucosa and tongue at the initial visit. (B) Oral mucosa and tongue after thymectomy.

Mentions: A 48-year-old woman was referred to the Department of Oral and Maxillofacial Surgery, Kyushu University Hospital, in August 2008, for erosive and hyperkeratotic lesions of the oral mucosa and tongue recurring for over a year (Fig. 1A). She had no remarkable medical history except for hypoferric anemia. She had low hemoglobin concentration (8.9 g/dL), decreased hematocrit (29.1%), low erythrocyte indices (mean corpuscular volume, 71.0 fl; mean corpuscular hemoglobin, 21.7 pg), and white blood cell count of 4760 cells/mm3 (neutrophils, 70.3%; lymphocytes, 24.4%; eosinophils, 5.3%; basophils, 0.0%). All serum chemistry data were within normal limits. Her C-reactive protein concentration was 0.03 mg/dL. Serum antibody tests were negative for Dsg1, Dsg3, HIV, hepatitis B virus (HBV), and hepatitis C virus (HCV) antibody. Serum levels of IgG, IgA, IgM, and IgE were less than normal limits (516, 6, 14, and 20 mg/dL, respectively). Computed tomography (CT) and magnetic resonance imaging (MRI) findings raised suspicions of thymoma, and thymectomy and intravenous immunoglobulin were performed in January 2009. Pathological findings revealed World Health Organization (WHO) classification type AB thymoma (Fig. 2), which, combined with clinical features, resulted in a definitive diagnosis of GS. The erosive and hyperkeratotic lesions of the oral mucosa and tongue were significantly improved by the thymectomy and intravenous immunoglobulin (Fig. 1B).


Cytokine profiles contribute to understanding the pathogenic difference between Good syndrome and oral lichen planus: two case reports and literature review.

Maehara T, Moriyama M, Kawano S, Hayashida JN, Furukawa S, Ohta M, Tanaka A, Yamauchi M, Ohyama Y, Kiyoshima T, Nakamura S - Medicine (Baltimore) (2015)

Clinical findings in oral cavity from patient with Good syndrome (GC) (Case 1). (A) Oral mucosa and tongue at the initial visit. (B) Oral mucosa and tongue after thymectomy.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4554038&req=5

Figure 1: Clinical findings in oral cavity from patient with Good syndrome (GC) (Case 1). (A) Oral mucosa and tongue at the initial visit. (B) Oral mucosa and tongue after thymectomy.
Mentions: A 48-year-old woman was referred to the Department of Oral and Maxillofacial Surgery, Kyushu University Hospital, in August 2008, for erosive and hyperkeratotic lesions of the oral mucosa and tongue recurring for over a year (Fig. 1A). She had no remarkable medical history except for hypoferric anemia. She had low hemoglobin concentration (8.9 g/dL), decreased hematocrit (29.1%), low erythrocyte indices (mean corpuscular volume, 71.0 fl; mean corpuscular hemoglobin, 21.7 pg), and white blood cell count of 4760 cells/mm3 (neutrophils, 70.3%; lymphocytes, 24.4%; eosinophils, 5.3%; basophils, 0.0%). All serum chemistry data were within normal limits. Her C-reactive protein concentration was 0.03 mg/dL. Serum antibody tests were negative for Dsg1, Dsg3, HIV, hepatitis B virus (HBV), and hepatitis C virus (HCV) antibody. Serum levels of IgG, IgA, IgM, and IgE were less than normal limits (516, 6, 14, and 20 mg/dL, respectively). Computed tomography (CT) and magnetic resonance imaging (MRI) findings raised suspicions of thymoma, and thymectomy and intravenous immunoglobulin were performed in January 2009. Pathological findings revealed World Health Organization (WHO) classification type AB thymoma (Fig. 2), which, combined with clinical features, resulted in a definitive diagnosis of GS. The erosive and hyperkeratotic lesions of the oral mucosa and tongue were significantly improved by the thymectomy and intravenous immunoglobulin (Fig. 1B).

Bottom Line: However, IL-4 and IL-17 were detected in OLP patients only.These results suggest that the pathogenesis of GS is different from that of OLP.GS is a unique inflammatory disorder characterized by dysfunction of Th2 and Th17 immune reactions via abnormal T-B cell interaction.

View Article: PubMed Central - PubMed

Affiliation: From the Section of Oral and Maxillofacial Oncology (TM, MM, SK, J-NH, SF, MO, AT, MY, SN); Section of Oral and Maxillofacial Surgery (YO); and Division of Maxillofacial Diagnostic and Surgical Sciences, Laboratory of Oral Pathology, Faculty of Dental Science, Kyushu University, Fukuoka, Japan (YK).

ABSTRACT
We described and analyzed the pathogenic difference between Good syndrome (GS) and oral lichen planus (OLP) in oral mucosa. Good syndrome (GS) is a rare disease characterized by B and T cell immunodeficiency associated with hypogammaglobulinemia and thymoma. GS patients frequently develop oral lichenoid lesions with lymphocytic infiltration beneath the basal layer. Oral lichen planus (OLP) is a chronic inflammatory disease of the oral mucosa characterized by destruction of basal cells by Langerhans cells, macrophages, and T lymphocytes. Although the histological features of the lesions of both diseases are very similar, the pathogenesis of GS in the oral mucosa remains unknown. In this study, we thus investigated the expression of infiltrating lymphocyte subsets (CD3, CD20, CD4, and CD8) and T helper (Th) cytokines including interferon (IFN)-γ (Th1 type), interleukin (IL)-4 (Th2 type), IL-17 (Th17 type), and IL-10 (regulatory T cell type) by immunohistochemistry in buccal mucosa specimens from 2 GS patients compared with 15 OLP patients. All patients showed a predominance of CD3 T cells over CD20 B cells, and CD4 Th cells over CD8 cytotoxic T cells. This polarization was especially prominent in GS. IFN-γ and IL-10 were strongly detected in the infiltrating lymphocytes of all patients. However, IL-4 and IL-17 were detected in OLP patients only. These results suggest that the pathogenesis of GS is different from that of OLP. GS is a unique inflammatory disorder characterized by dysfunction of Th2 and Th17 immune reactions via abnormal T-B cell interaction.

Show MeSH
Related in: MedlinePlus