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      Extramedullary hematopoiesis in gastric wall under early gastric cancer in a man with a myeloproliferative disorder: a case report

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          Abstract

          Extramedullary hematopoiesis (EMH) is the proliferation of hematopoietic stem cells outside the bone marrow and often observed in the liver, spleen in association with myeloproliferative disorders. On the other hand, EMH in the gastric wall is extremely rare. We report a rare case of EMH foci coexisting with early gastric cancer, which resulted in severe gastrointestinal bleeding. A 70-year-old male was diagnosed with myelofibrosis 4 years ago and visited our emergency room with a complaint of hematemesis and tarry stools. Upper gastrointestinal endoscopy revealed three early-stage gastric cancers in the lower gastric body and antrum, and biopsy was performed. Persistent bleeding at the biopsy site of the hypogastric lesion led to the consideration of surgical intervention. An open distal gastrectomy was performed. Postoperative histopathological examination revealed the tumor of the lower gastric body had EMH foci associated with myelofibrosis.

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          Most cited references14

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          Nonhepatosplenic extramedullary hematopoiesis: associated diseases, pathology, clinical course, and treatment.

          To define associated clinical conditions, pathology, natural history, and treatment outcome of nonhepatosplenic extramedullary hematopoiesis (NHS-EMH). We retrospectively reviewed the medical charts of all patients identified as having NHS-EMH from 1975 to 2002. Diagnosis was made by tissue biopsy, fine-needle aspiration biopsy, or radionuclide bone marrow scanning. We identified 27 patients with antemortem diagnosis of NHS-EMH. The most common associated condition and disease site were myelofibrosis with myeloid metaplasia (MMM) (in 18 patients [67%]) and the vertebral column (in 7 patients [26%]; all involving the thoracic region), respectively. At the time of diagnosis of NHS-EMH, concurrent splenic EMH (in 22 patients [82%]; 15 [56%] had undergone splenectomy) and red blood cell transfusion dependency (in 12 patients [44%]) were prevalent. Of the 27 patients, 9 (33%) required no specific therapy. Specific therapy was radiation (in 7 patients with a 71% response rate) and surgical excision (in 6 patients with a 67% response). Treatment-associated complications were limited to surgery. Radiation therapy was not used in the non-MMM group, but low-dose radiation therapy was used in the MMM group for paraspinal or intraspinal EMH (median dose, 1 Gy; range, 1-10 Gy), pleural or pulmonary disease (median dose, 1.25 Gy; range, 1.00-1.50 Gy), and abdominal or pelvic disease (median dose, 2.02 Gy; range, 150-4.50 Gy). Median survival after the diagnosis of NHS-EMH was 13 months in the MMM group and 21 months in the non-MMM group. This retrospective study suggests that NHS-EMH is rare, is often associated with MMM, and preferentially affects the thoracic spinal region. Asymptomatic disease may require no specific treatment, whereas symptomatic disease is best managed with low-dose radiation therapy.
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            Intrathoracic extramedullary haematopoiesis manifested as a neoplastic lesion within anterior mediastinum.

            Intrathoracic extramedullary hematopoiesis (EMH), the formation of apparently normal blood cells outside the confines of the bone marrow, is an uncommon but well-defined entity. It is usually associated with hematologic disorders and located in the lower paravertebral sulci or rarely in the pleura. We report a case of EMH, which presented in a patient without hematologic disorders and was manifested as an anterior mediastinal mass. The first interesting aspect of our case was that EMH occurred in a patient with normal laboratory findings and no past medical history of anemia. The second remarkable characteristic was that EMH manifested as an anterior mediastinal mass, mimicking a neoplastic lesion. Definitive diagnosis of EMH was established by a video-assisted thoracoscopic surgical biopsy. In light of this diagnosis, no further surgical procedure was carried out.
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              Extramedullary hematopoiesis occurring as a nasal polyp in a man with a myeloproliferative disorder.

              We describe the case of a patient with a known myeloproliferative disorder who presented with epistaxis and what clinically appeared to be a nasal polyp. The mass was resected and proved to represent a focus of extramedullary hematopoiesis. The patient subsequently developed extramedullary hematopoiesis of the skin and the stomach wall. Following nasal polypectomy, he did well for a time, but he eventually died as a result of other complications of his disease. This unique case serves as a reminder that common rhinologic complaints can be a sign of significant and life-threatening pathology.
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                Author and article information

                Contributors
                Journal
                J Surg Case Rep
                J Surg Case Rep
                jscr
                Journal of Surgical Case Reports
                Oxford University Press
                2042-8812
                July 2022
                16 July 2022
                16 July 2022
                : 2022
                : 7
                : rjac337
                Affiliations
                Department of Surgery , North Alps Medical Center Azumi Hospital, Kita-Azumi-gun, Japan
                Department of Molecular Pathology , Shinshu University, Matsumoto, Japan
                Department of Surgery , North Alps Medical Center Azumi Hospital, Kita-Azumi-gun, Japan
                Department of Gastroenterology , North Alps Medical Center Azumi Hospital, Kita-Azumi-gun, Japan
                Department of Hematology , North Alps Medical Center Azumi Hospital, Kita-Azumi-gun, Japan
                Department of Gastroenterology , North Alps Medical Center Azumi Hospital, Kita-Azumi-gun, Japan
                Department of Hematology , North Alps Medical Center Azumi Hospital, Kita-Azumi-gun, Japan
                Department of Surgery , North Alps Medical Center Azumi Hospital, Kita-Azumi-gun, Japan
                Department of Surgery , North Alps Medical Center Azumi Hospital, Kita-Azumi-gun, Japan
                Department of Surgery , North Alps Medical Center Azumi Hospital, Kita-Azumi-gun, Japan
                Author notes
                Corresponding address. Department of Surgery, North Alps Medical Center Azumi Hospital, 3207-1, Ikeda-cho, Kita-Azumi-gun, Nagano 399-8695, Japan. Tel: +81-261623166; Fax: +81-261622711; E-mail: h.motoyama@ 123456azumi-ghp.jp
                Author information
                https://orcid.org/0000-0003-0882-2626
                Article
                rjac337
                10.1093/jscr/rjac337
                9291352
                35854821
                a4b4366d-4e10-446c-8097-e61e3dd3760c
                Published by Oxford University Press and JSCR Publishing Ltd. © The Author(s) 2022.

                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 non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com

                History
                : 4 May 2022
                : 22 June 2022
                Page count
                Pages: 4
                Categories
                AcademicSubjects/MED00910
                jscrep/0160
                Case Report

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