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      Unmasked immune reconstitution inflammatory syndrome towards B-cell non-Hodgkin lymphoma during treatment of esophageal actinomycosis in a patient with advanced HIV: a case report

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          Abstract

          Background

          Actinomycosis is an unusual chronic bacterial infection, even rarer in people living with HIV. It is not considered an AIDS-defining disease. However, the role in co-presentation or overlap with other opportunistic conditions of advanced HIV is unknown.

          Case presentation

          A 49-year-old Peruvian male presented with a 4-month history of dysphagia, odynophagia, hyporexia and wasting. He underwent an upper digestive endoscopy, in which ulcers with a necrotic center were observed, therefore, the initial diagnostic assumption was esophageal cancer. Subsequent pathology report excluded neoplasms and confirmed the diagnosis of actinomycosis. Serology for human immunodeficiency virus was requested, yielding a positive result. Antimicrobial treatment with amoxicillin and antiretroviral therapy were indicated, with slow clinical improvement. After 4 months, epigastric discomfort presented, for which a new upper digestive endoscopy was performed, revealing a deep gastric ulcer, which was compatible with diffuse large B-cell non-Hodgkin lymphoma.

          Conclusion

          Esophageal actinomycosis in people living with HIV is very rare. We suggest HIV-associated immunosuppression is not enough to allow for actinomycosis to develop, and masked underlying entities should be sought. The existence of such entities in people living with HIV should raise awareness of the possibility of unmasked immune reconstitution inflammatory syndrome once treatment has started.

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          Most cited references17

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          Actinomycosis: etiology, clinical features, diagnosis, treatment, and management

          Actinomycosis is a rare chronic disease caused by Actinomyces spp., anaerobic Gram-positive bacteria that normally colonize the human mouth and digestive and genital tracts. Physicians must be aware of typical clinical presentations (such as cervicofacial actinomycosis following dental focus of infection, pelvic actinomycosis in women with an intrauterine device, and pulmonary actinomycosis in smokers with poor dental hygiene), but also that actinomycosis may mimic the malignancy process in various anatomical sites. Bacterial cultures and pathology are the cornerstone of diagnosis, but particular conditions are required in order to get the correct diagnosis. Prolonged bacterial cultures in anaerobic conditions are necessary to identify the bacterium and typical microscopic findings include necrosis with yellowish sulfur granules and filamentous Gram-positive fungal-like pathogens. Patients with actinomycosis require prolonged (6- to 12-month) high doses (to facilitate the drug penetration in abscess and in infected tissues) of penicillin G or amoxicillin, but the duration of antimicrobial therapy could probably be shortened to 3 months in patients in whom optimal surgical resection of infected tissues has been performed. Preventive measures, such as reduction of alcohol abuse and improvement of dental hygiene, may limit occurrence of pulmonary, cervicofacial, and central nervous system actinomycosis. In women, intrauterine devices must be changed every 5 years in order to limit the occurrence of pelvic actinomycosis.
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            Actinomyces and related organisms in human infections.

            Actinomyces israelii has long been recognized as a causative agent of actinomycosis. During the past 3 decades, a large number of novel Actinomyces species have been described. Their detection and identification in clinical microbiology laboratories and recognition as pathogens in clinical settings can be challenging. With the introduction of advanced molecular methods, knowledge about their clinical relevance is gradually increasing, and the spectrum of diseases associated with Actinomyces and Actinomyces-like organisms is widening accordingly; for example, Actinomyces meyeri, Actinomyces neuii, and Actinomyces turicensis as well as Actinotignum (formerly Actinobaculum) schaalii are emerging as important causes of specific infections at various body sites. In the present review, we have gathered this information to provide a comprehensive and microbiologically consistent overview of the significance of Actinomyces and some closely related taxa in human infections.
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              Atypical presentations of actinomycosis.

              Actinomycotic infections of the cervicofacial region are uncommon. Most major medical centers report approximately one case per year. Presenting clinical manifestations are confusing because they often mimic other disease processes. Diagnosis may be difficult due to a general lack of familiarity with the disease and the fastidious nature of the organism in culture. The cervicofacial manifestations of actinomycosis are varied, and a high index of suspicion is required to make an accurate and timely diagnosis. Retrospective chart review with the presentation of four unusual cases of actinomycosis were performed. Two patients were initially seen with dysphagia from a tongue base mass. The third patient was initially seen with a 3-week history of worsening hoarseness and stridor. Examination revealed an ulcerative lesion of the left hemilarynx and pyriform sinus. All three patients were thought to have a neoplastic process. Diagnosis was made on histologic examination of a tissue biopsy. The fourth patient was initially seen with a buccal space mass that was draining externally. Culture of the purulent drainage revealed Actinomyces. In all four cases, symptoms resolved after appropriate antimicrobial therapy. Actinomycosis of the head and neck, although rare, is an important entity to the otolaryngologist. A confusing clinical presentation combined with the fastidious nature of the organism make for a difficult diagnosis. A high index of suspicion is required to make an accurate diagnosis and institute the appropriate antibiotic therapy.
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                Author and article information

                Contributors
                elsa_vargas11@hotmail.com
                pcmerfer@gmail.com
                gonza_cornejo@icloud.com
                jmontenegroi@cientifica.edu.pe
                juanochirinos@hotmail.com
                alfredochiappe911@hotmail.com
                Journal
                AIDS Res Ther
                AIDS Res Ther
                AIDS Research and Therapy
                BioMed Central (London )
                1742-6405
                14 July 2023
                14 July 2023
                2023
                : 20
                : 48
                Affiliations
                [1 ]GRID grid.441917.e, ISNI 0000 0001 2196 144X, Universidad Peruana de Ciencias Aplicadas, ; Avenida Alameda San Marcos cuadra 2 S/N, Chorrillos, Lima, 15023 Peru
                [2 ]GRID grid.414887.6, ISNI 0000 0004 6095 1668, Hospital Nacional Dos de Mayo. Parque “Historia de la Medicina Peruana” S/N, ; Lima, 15003 Peru
                [3 ]GRID grid.430666.1, ISNI 0000 0000 9972 9272, Facultad de Ciencias de la Salud, , Universidad Científica del Sur. Carretera Panamericana, ; Sur km19, Villa El Salvador, Lima, 15067 Peru
                [4 ]GRID grid.10800.39, ISNI 0000 0001 2107 4576, Centro de Investigaciones Tecnológicas, Biomédicas y Medioambientales, , Universidad Nacional Mayor de San Marcos, ; Lima, 070102 Peru
                [5 ]GRID grid.441904.c, ISNI 0000 0001 2192 9458, Instituto de Investigaciones en Ciencias Biomédicas, , Universidad Ricardo Palma, ; Lima, Peru
                [6 ]Clínica Angloamericana, Calle Alfredo Salazar 350, Lima, San Isidro 15073 Peru
                Article
                526
                10.1186/s12981-023-00526-y
                10347741
                107140c1-9c52-4bfa-af95-30a01577caab
                © The Author(s) 2023

                Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

                History
                : 27 April 2023
                : 13 May 2023
                Categories
                Case Report
                Custom metadata
                © BioMed Central Ltd., part of Springer Nature 2023

                Infectious disease & Microbiology
                human immunodeficiency virus,actinomycosis,immune reconstitution inflammatory syndrome,non-hodgkin lymphoma

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