Primary non-Hodgkin′s lymphoma of the salivary gland: A spectrum of lymphoepithelial sialadenitis, low-grade B-cell lymphoma of mucosa-associated lymphoid tissue with transformation to high-grade lymphoma

Agale, Shubhangi Vinayak; D'Costa, Grace; Hastak, Meenal; Shedge, Rakesh

doi:10.4103/0377-4929.64351

Cited by 8 publications

(4 citation statements)

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“…One unique feature of SjS is the formation of leukocyte aggregates in the salivary and lacrimal glands, referred to as either lymphoepithelial sialadenitis or LF [56,57]. At times, these LF appear histologically to be germinal-like centers and may be indicative of a more pronounced and severe hypergammaglobulinemia [58].…”

Section: Discussionmentioning

confidence: 99%

Gene therapy using IL-27 ameliorates Sjögren's syndrome-like autoimmune exocrinopathy

et al. 2012

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IntroductionSjögren's syndrome (SjS) is a systemic autoimmune disease characterized by decreased salivary and lacrimal gland secretions, resulting in severe dry mouth and dry eyes. Recent studies have suggested that TH17 cells and its signature cytokine IL-17 are involved in the underlying pathogenic mechanisms leading to destructive inflammation and autoimmunity. In the present study, we examined whether IL-27, a natural inhibitor of TH17 activity, could down-regulate or reverse SjS in C57BL/6.NOD-Aec1Aec2 mice, a model of primary-SjS.MethodsRecombinant serotype 2 adeno-associated viral (AAV2) vectors expressing either IL-27 (rAAV2-IL27) or LacZ (rAAV2-LacZ) were injected into 6 or 14 week-old C57BL/6.NOD-Aec1Aec2 mice. Changes in IL-27, IL-17, and IL-10 cytokine levels in peripheral blood were determined by ELISAs, while flow cytometry analyses were used to quantify cytokine-positive splenocytes. Histological assessment of salivary glands, anti-nuclear autoantibody (ANA) staining, and stimulated saliva flow rates were used to profile SjS disease severity.ResultsMice systemically treated with intravenous rAAV2-IL27 injections at either 6 or 14 weeks of age exhibited long-term elevated levels of serum IL-27 with concomitantly reduced levels of IL-17 compared with sera from mice injected with rAAV2-LacZ or saline out to 20 weeks post-inoculation. Most importantly, disease profiles revealed that rAAV2-IL27 treatment had little effect on lymphocytic focus (LF) scores, but resulted in structural changes in LF, lower titers of ANAs with changes in staining patterns, and a less severe clinical disease as determined by saliva flow rates.ConclusionsThese data support the concept that IL-27, when provided exogenously, can induce a suppressive effect on SjS development and thus may be an effective therapeutic agent for regulating TH17 pro-inflammatory activity in autoimmune diseases where the TH17 system has been shown to play an important role in their pathogenesis.

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Section: Discussionmentioning

confidence: 99%

Gene therapy using IL-27 ameliorates Sjögren's syndrome-like autoimmune exocrinopathy

et al. 2012

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“…Although no residual MALT lymphoma component was present in this case, the possibility of a high-grade transformation of thymic MALT lymphoma has been reported previously [8]. Primary salivary gland high-grade B-cell lymphomas occurring with or without a preceding MALT lymphoma have also been observed in Sjögren syndrome [11,12].…”

Section: Discussionmentioning

confidence: 49%

Thymic Hyperplasia with Lymphoepithelial Sialadenitis (LESA)-Like Features: Strong Association with Lymphomas and Non-Myasthenic Autoimmune Diseases

Porubský

Popović

Badve

et al. 2021

Cancers

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Thymic hyperplasia (TH) with lymphoepithelial sialadenitis (LESA)-like features (LESA-like TH) has been described as a tumor-like, benign proliferation of thymic epithelial cells and lymphoid follicles. We aimed to determine the frequency of lymphoma and autoimmunity in LESA-like TH and performed retrospective analysis of cases with LESA-like TH and/or thymic MALT-lymphoma. Among 36 patients (21 males) with LESA-like TH (age 52 years, 32–80; lesion diameter 7.0 cm, 1–14.5; median, range), five (14%) showed associated lymphomas, including four (11%) thymic MALT lymphomas and one (3%) diffuse large B-cell lymphoma. One additional case showed a clonal B-cell-receptor rearrangement without evidence of lymphoma. Twelve (33%) patients (7 women) suffered from partially overlapping autoimmune diseases: systemic lupus erythematosus (n = 4, 11%), rheumatoid arthritis (n = 3, 8%), myasthenia gravis (n = 2, 6%), asthma (n = 2, 6%), scleroderma, Sjögren syndrome, pure red cell aplasia, Grave’s disease and anti-IgLON5 syndrome (each n = 1, 3%). Among 11 primary thymic MALT lymphomas, remnants of LESA-like TH were found in two cases (18%). In summary, LESA-like TH shows a striking association with autoimmunity and predisposes to lymphomas. Thus, a hematologic and rheumatologic workup should become standard in patients diagnosed with LESA-like TH. Radiologists and clinicians should be aware of LESA-like TH as a differential diagnosis for mediastinal mass lesions in patients with autoimmune diseases.

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“…Salivary gland lymphomas have a better survival rate compared with nodal and other extranodal lymphomas, and the prognosis for a well‐evaluated and appropriately treated patient with lymphoma presenting in the salivary glands is excellent 9 …”

Section: Discussionmentioning

confidence: 99%

DLBCL of bilateral submandibular glands and MALToma of thyroid—A rare coexistence

et al. 2021

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Involvement of the bilateral submandibular glands and thyroid by the same lymphoma simultaneously has been reported in the literature. However, two different types of lymphomas presenting simultaneously at different sites have never been reported. This case report highlights this rare occurrence. A 65‐year‐old female, a known case of Hashimoto’s thyroiditis with raised anti‐TPO antibodies, presented with thyroid swelling for 1 year and bilateral submandibular swelling for 3 years. FNAC and flow cytometry showed features of mucosa‐associated lymphoid tissue lymphoma in the thyroid gland, whereas the bilateral submandibular glands showed features of diffuse large B cell lymphoma. Histopathology and immunohistochemistry from the submandibular swelling led to similar diagnoses as the flow cytometry.

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Primary non-Hodgkin′s lymphoma of the salivary gland: A spectrum of lymphoepithelial sialadenitis, low-grade B-cell lymphoma of mucosa-associated lymphoid tissue with transformation to high-grade lymphoma

Cited by 8 publications

References 0 publications

Gene therapy using IL-27 ameliorates Sjögren's syndrome-like autoimmune exocrinopathy

Gene therapy using IL-27 ameliorates Sjögren's syndrome-like autoimmune exocrinopathy

Thymic Hyperplasia with Lymphoepithelial Sialadenitis (LESA)-Like Features: Strong Association with Lymphomas and Non-Myasthenic Autoimmune Diseases

DLBCL of bilateral submandibular glands and MALToma of thyroid—A rare coexistence

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