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      Eosinophilic sialoadenitis in a patient with severe asthma: a case report

      case-report

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          Abstract

          Activated eosinophils can infiltrate various tissues and cause inflammatory tissue damage. Asthma is a typical type of eosinophilic inflammatory disease that occurs in the respiratory system. Eosinophilic sialodochitis and sialoadenitis of the salivary gland are rare diseases clinically characterized by painful swelling. In this report, we present a 68-year-old woman with asthma who presented to our hospital with mandibular swelling. Her asthma had been well controlled with an inhaled combination of a corticosteroid and a long-acting β2 agonist, although she reported a past history of frequent asthma attacks and hospitalization. Laboratory investigation on admission revealed blood eosinophilia (2,673/μL), high levels of total immunoglobulin E (390 U/mL) and immunoglobulin G4 (183 mg/dL). Bone marrow examination showed no evidence of eosinophilic neoplasia. Histological examination of her minor salivary glands disclosed an infiltration of mixed lymphocytes and eosinophils. Chromatolytic eosinophils with Charcot-Leyden crystals were also observed within the edematous dermis and fibrous tissues surrounding the minor salivary gland. The patient was diagnosed with eosinophilic sialoadenitis. Treatment with oral corticosteroids (0.5 mg/kg/day) was initiated. Thereafter, the mandibular swelling improved. This report describes a rare case of eosinophilic sialoadenitis in a patient with severe eosinophilic asthma, for which histopathological and immunefluorescence microscopic analyses were performed.

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          Most cited references14

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          Functions of tissue-resident eosinophils

          Tissue-resident eosinophils selectively secrete cytokines and other mediators that have diverse functions in health and disease.
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            Eosinophil extracellular DNA trap cell death mediates lytic release of free secretion-competent eosinophil granules in humans.

            Eosinophils release their granule proteins extracellularly through exocytosis, piecemeal degranulation, or cytolytic degranulation. Findings in diverse human eosinophilic diseases of intact extracellular eosinophil granules, either free or clustered, indicate that eosinophil cytolysis occurs in vivo, but the mechanisms and consequences of lytic eosinophil degranulation are poorly understood. We demonstrate that activated human eosinophils can undergo extracellular DNA trap cell death (ETosis) that cytolytically releases free eosinophil granules. Eosinophil ETosis (EETosis), in response to immobilized immunoglobulins (IgG, IgA), cytokines with platelet activating factor, calcium ionophore, or phorbol myristate acetate, develops within 120 minutes in a reduced NADP (NADPH) oxidase-dependent manner. Initially, nuclear lobular formation is lost and some granules are released by budding off from the cell as plasma membrane-enveloped clusters. Following nuclear chromatolysis, plasma membrane lysis liberates DNA that forms weblike extracellular DNA nets and releases free intact granules. EETosis-released eosinophil granules, still retaining eosinophil cationic granule proteins, can be activated to secrete when stimulated with CC chemokine ligand 11 (eotaxin-1). Our results indicate that an active NADPH oxidase-dependent mechanism of cytolytic, nonapoptotic eosinophil death initiates nuclear chromatolysis that eventuates in the release of intact secretion-competent granules and the formation of extracellular DNA nets.
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              Eosinophil extracellular trap cell death-derived DNA traps: Their presence in secretions and functional attributes.

              Activated human eosinophils, as well as neutrophils, can release extracellular chromatin to form DNA traps through cytolytic extracellular trap cell death (ETosis). Although formations of neutrophil DNA traps are recognized in patients with various inflammatory conditions, neither the presence of ETosis-derived eosinophil DNA traps in human allergic diseases nor the characteristics of these DNA traps have been studied.
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                Author and article information

                Journal
                Asia Pac Allergy
                Asia Pac Allergy
                APA
                Asia Pacific Allergy
                Asia Pacific Association of Allergy, Asthma and Clinical Immunology
                2233-8276
                2233-8268
                July 2021
                13 July 2021
                : 11
                : 3
                : e29
                Affiliations
                [1 ]Division of Infectious Diseases and Respiratory Medicine, Department of Internal Medicine, National Defense Medical College, Saitama, Japan.
                [2 ]Department of General Medicine, National Defense Medical College, Saitama, Japan.
                [3 ]Department of Pathology and Laboratory Medicine, National Defense Medical College, Saitama, Japan.
                [4 ]Department of General Medical Practice and Laboratory Diagnostic Medicine, Akita University Graduate School of Medicine, Akita, Japan.
                Author notes
                Correspondence to Jun Miyata. Division of Infectious Diseases and Respiratory Medicine, Department of Internal Medicine, National Defense Medical College, 3-2, Namiki, Tokorozawa-shi, Saitama 359-8513, Japan. Tel: +81-4-2995-1211, Fax: +81-4-2996-5225, junmiyata@ 123456ndmc.ac.jp
                Author information
                https://orcid.org/0000-0002-6359-5676
                https://orcid.org/0000-0002-3189-1702
                https://orcid.org/0000-0003-0409-7527
                https://orcid.org/0000-0002-3537-7735
                Article
                10.5415/apallergy.2021.11.e29
                8331254
                34386405
                d62655ac-63b0-4677-9c7b-66861bc9a87d
                Copyright © 2021. Asia Pacific Association of Allergy, Asthma and Clinical Immunology.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( https://creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 29 January 2021
                : 08 July 2021
                Categories
                Educational & Teaching Material

                Immunology
                sialoadenitis,asthma,eosinophil,degranulation,charcot-leyden crystal
                Immunology
                sialoadenitis, asthma, eosinophil, degranulation, charcot-leyden crystal

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