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      Gastrointestinal Alpha Heavy Chain Disease With Persistent Campylobacter Jejuni Colonization and Refractory Giardiasis

      case-report

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          ABSTRACT

          Alpha heavy chain disease (αHCD) is a rare variant of the mucosa-associated lymphoid tissue lymphoma characterized by expression of a monotypic truncated immunoglobulin α heavy chain. αHCD frequently involves the gastrointestinal (GI) tract, and its pathogenesis has been linked to clonal B-cell expansion from chronic immune stimulation by infectious agents. We report a rare case of GI αHCD with 5 concomitant pathogens identified on a GI multiplex real-time polymerase chain reaction panel, featured by persistent Campylobacter jejuni colonization and refractory giardiasis.

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          The 2016 revision of the World Health Organization classification of lymphoid neoplasms.

          Steven H. Swerdlow, Elias Campo, Stefano Pileri … (2016)
          A revision of the nearly 8-year-old World Health Organization classification of the lymphoid neoplasms and the accompanying monograph is being published. It reflects a consensus among hematopathologists, geneticists, and clinicians regarding both updates to current entities as well as the addition of a limited number of new provisional entities. The revision clarifies the diagnosis and management of lesions at the very early stages of lymphomagenesis, refines the diagnostic criteria for some entities, details the expanding genetic/molecular landscape of numerous lymphoid neoplasms and their clinical correlates, and refers to investigations leading to more targeted therapeutic strategies. The major changes are reviewed with an emphasis on the most important advances in our understanding that impact our diagnostic approach, clinical expectations, and therapeutic strategies for the lymphoid neoplasms.
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            Immunoproliferative small intestinal disease associated with Campylobacter jejuni.

            Marc Lecuit, Antoine Martin, Claire Poyart … (2004)
            Immunoproliferative small intestinal disease (also known as alpha chain disease) is a form of lymphoma that arises in small intestinal mucosa-associated lymphoid tissue (MALT) and is associated with the expression of a monotypic truncated immunoglobulin alpha heavy chain without an associated light chain. Early-stage disease responds to antibiotics, suggesting a bacterial origin. We attempted to identify a causative agent. We performed polymerase chain reaction (PCR), DNA sequencing, fluorescence in situ hybridization, and immunohistochemical studies on intestinal-biopsy specimens from a series of patients with immunoproliferative small intestinal disease. Analysis of frozen intestinal tissue obtained from an index patient with immunoproliferative small intestinal disease who had a dramatic response to antibiotics revealed the presence of Campylobacter jejuni. A follow-up retrospective analysis of archival intestinal-biopsy specimens disclosed campylobacter species in four of six additional patients with immunoproliferative small intestinal disease. These results indicate that campylobacter and immunoproliferative small intestinal disease are associated and that C. jejuni should be added to the growing list of human pathogens responsible for immunoproliferative states. Copyright 2004 Massachusetts Medical Society
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              Emerging role of infectious etiologies in the pathogenesis of marginal zone B-cell lymphomas.

              Emanuele Zucca, Francesco Bertoni, Barbara Vannata … (2014)
              Extranodal marginal zone B-cell lymphomas of the mucosa-associated lymphoid tissue (MALT) arise from lymphoid populations that are induced by chronic inflammation in extranodal sites. The most frequently affected organ is the stomach, where MALT lymphoma is incontrovertibly associated with a chronic gastritis induced by a microbial pathogen, Helicobacter pylori. Gastric MALT lymphoma therefore represents a paradigm for evaluating inflammation-associated lymphomagenesis, which may lead to a deeper understanding of a possible etiologic association between other microorganisms and nongastric marginal zone lymphomas. Besides infectious etiology, chronic inflammation caused by autoimmune diseases, such as Sjögren syndrome or Hashimoto thyroiditis, can also carry a significant risk factor for the development of marginal zone lymphoma. In addition to the continuous antigenic drive, additional oncogenic events play a relevant role in lymphoma growth and progression to the point at which the lymphoproliferative process may eventually become independent of antigenic stimulation. Recent studies on MALT lymphomas have in fact demonstrated genetic alterations affecting the NF-κB) pathway, a major signaling pathway involved in many cancers. This review aims to present marginal zone lymphoma as an example of the close pathogenetic link between chronic inflammation and tumor development, with particular attention to the role of infectious agents and the integration of these observations into everyday clinical practice. See all articles in this CCR Focus section, "Paradigm Shifts in Lymphoma."
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                Author and article information

                Contributors
                Zilan Lin
                Pritika Sharma
                Gabriel Heering
                Brad Dworkin
                Amir Steinberg
                Fouzia Shakil
                Beth Schorr-Lesnick
                Journal
                Journal ID (nlm-ta): ACG Case Rep J
                Journal ID (iso-abbrev): ACG Case Rep J
                Journal ID (coden): ACGCRJ
                Journal ID (publisher-id): AC9
                Title: ACG Case Reports Journal
                Publisher: Wolters Kluwer (Maryland, MD )
                ISSN (Electronic): 2326-3253
                Publication date Collection: August 2024
                Publication date (Electronic): 22 August 2024
                Volume: 11
                Issue: 8
                Electronic Location Identifier: e01467
                Affiliations
                [1 ]Department of Internal Medicine, New York Medical College, Valhalla, NY
                [2 ]Department of Gastroenterology and Hepatobiliary Diseases, New York Medical College, Valhalla, NY
                [3 ]Department of Hematology and Oncology, New York Medical College, Valhalla, NY
                [4 ]Department of Pathology, New York Medical College, Valhalla, NY
                Author notes
                Correspondence: Wei Tang, MD. E-mail: wei.tang@ 123456wmchealth.org .
                Author information
                https://orcid.org/0000-0002-6599-980X
                Article
                Publisher ID: ACGCR-24-0240 Accession ID: 00019
                DOI: 10.14309/crj.0000000000001467
                PMC ID: 11340919
                PubMed ID: 39176219
                SO-VID: 3fda98a0-a966-421b-9030-a9cea7c6044c
                Copyright © © 2024 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of The American College of Gastroenterology.
                License:

                This is an open access article distributed under the terms of the Creative Commons Attribution Licence 4.0 (CC BY), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 14 March 2024
                Date accepted : 11 July 2024
                Categories
                Subject: Case Report
                Subject: Small Bowel
                Custom metadata
                OPEN-ACCESS TRUE

                Keywords: heavy chain disease,extranodal marginal zone lymphoma,mucosa associated lymphoid tissue lymphoma,campylobacter jejuni,giardia lamblia,helicobacter pylori
                Data availability:
                Keywords: heavy chain disease, extranodal marginal zone lymphoma, mucosa associated lymphoid tissue lymphoma, campylobacter jejuni, giardia lamblia, helicobacter pylori

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