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      Adrenal extramedullary hematopoiesis associated with beta-thalassemia trait in an adult woman: a case report and review of literature

      case-report

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          Highlights

          • •

            Adrenal myelolipoma with extramedullary hematopoiesis (EMH) is a rare condition.

          • •

            EMH could be presented with several clinical hematological disorders.

          • •

            Surgical management becomes inevitable in certain adrenal EMH cases especially in the presence of large adrenal mass.

          Abstract

          Introduction

          Adrenal myelolipoma with extramedullary hematopoiesis (EMH) is a rare condition.

          Case presentation

          We report a case of 48-year-old woman suffering from recurrent right hypochondrial pain for two years. Laboratory work-up revealed chronic microcytic iron deficiency anemia with beta thalassemia trait, right adrenal myelolipoma with histopathological evidence of hematopoietic cells (EMH). The patient underwent open resection of the adrenal gland and the post-operative course was uneventful.

          Conclusions

          EMH could be presented with several clinical hematological disorders. Surgical management becomes inevitable in certain adrenal EMH cases especially in the presence of a large adrenal mass.

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          Most cited references30

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          Myelolipoma: CT and pathologic features.

          Laurence Kenney, C Heffess, B. Wagner … (1998)
          To correlate clinical and pathologic patterns of myelolipoma with the computed tomographic (CT) appearance. All cases of myelolipoma accessioned by the Armed Forces Institute of Pathology from 1981 through 1997 were reviewed. CT images were scored for the location and size of each myelolipoma and the presence of calcification, hemorrhage, fat, and pseudocapsule. Pathologic findings for the pattern of fat and bone marrow elements were correlated with CT findings. In 74 patients, 86 myelolipomas were found, of which 72 were in an adrenal gland (eight were bilateral), and 14 extra-adrenal masses were found in 10 patients. Four clinicopathologic patterns emerged: (a) isolated adrenal myelolipoma in 37 patients (fat evident at CT, no other disorders present); (b) myelolipoma with hemorrhage in nine patients (imaging features similar to those of isolated adrenal myelolipomas but larger [mean diameter, 14.2 vs 9.9 cm; P = .01]; (c) extra-adrenal myelolipoma in 10 patients (imaging findings similar to those of adrenal myelolipomas, found most often in the retroperitoneum); and (d) myelolipomatous foci within other adrenal pathologic conditions in 18 patients (smaller, lower fat content, more heavily calcified). Myelolipomas are adrenal or extra-adrenal masses, with hemorrhage more common in larger lesions (diameter, > 10 cm). The CT appearance of myelolipomatous foci, which can be found within other pathologic adrenal conditions, is different from that of isolated adrenal myelolipomas.
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            Paraspinal extramedullary hematopoiesis in patients with thalassemia intermedia.

            Ali Taher, Hani Mhaidli, Rachid K Haidar (2010)
            Ineffective erythropoiesis in patients with thalassemia intermedia drives extramedullary hematopoietic tumor formation in several parts of the body. Paraspinal involvement has received increasing attention due to the associated morbidity secondary to spinal cord compression. Although the history and physical examination may help narrow the differential diagnosis, radiographic imaging remains essential to confirm the existence of hematopoietic tissue. Characteristic appearance has been observed mainly on magnetic resonance imaging. Several treatment options have been described, including transfusion therapy, laminectomy, radiotherapy, and the use of fetal hemoglobin inducing agents that decrease the hematopoietic drive. However, the ideal management scheme remains controversial. Until large prospective trials evaluate the efficacy and safety of the available treatment options, both in single and in combination therapy, an individualized approach should be entertained.
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              Extra-medullary haematopoiesis: a pictorial review of its typical and atypical locations.

              Dharamraj Sohawon, Kenneth Lau, Theodore Lau … (2012)
              Extra-medullary haematopoiesis (EMH) occurs in many haematological disorders and is secondary to insufficient bone marrow function or ineffective erythropoiesis. It usually presents as haematopoietic masses in several typical and atypical body locations. This pictorial review briefly discusses the common EMH locations encountered in clinical practice, including the liver, spleen, lymph nodes and paravertebral regions. Unusual presentation as focal hepatic and splenic masses is also discussed. Some atypical EMH locations are then presented together with their pathophysiology and relevant illustrations. These include EMH in the intra-spinal canal, pre-sacral region, nasopharynx and paranasal sinuses. Intra-spinal EMH can cause cord compression and present with neurological symptoms. In these cases, urgent treatment may be required. Haematopoietic masses in the other atypical locations can present with obstructive symptoms or may be diagnosed incidentally on imaging. EMH in unusual locations need to be monitored with follow-up imaging to ensure their stability. In some circumstances, tissue biopsy is appropriate to confirm the diagnosis and exclude other sinister pathology (e.g. malignancy). Treatment is only required where symptoms are present. Management options include blood transfusion, radiotherapy, hydroxyurea or surgical resection in selected cases.
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                Author and article information

                Contributors
                Ayman El-Menyar
                Journal
                Journal ID (nlm-ta): Int J Surg Case Rep
                Journal ID (iso-abbrev): Int J Surg Case Rep
                Title: International Journal of Surgery Case Reports
                Publisher: Elsevier
                ISSN (Electronic): 2210-2612
                Publication date PMC-release: 19 May 2016
                Publication date Collection: 2016
                Publication date (Electronic): 19 May 2016
                Volume: 24
                Pages: 83-87
                Affiliations
                [a ]Department of Surgery, Hamad General Hospital, Doha, Qatar
                [b ]Clinical Research, Trauma Surgery, Hamad General Hospital, Doha, Qatar
                [c ]Internal Medicine, Weill Cornell Medical College, Doha, Qatar
                Author notes
                [* ]Corresponding author at: Department of Surgery, Trauma Surgery, Clinical Research, Hamad General Hospital, P.O Box 3050, Doha, Qatar.Clinical ResearchTrauma SurgeryHamad General HospitalDohaQatar aymanco65@ 123456yahoo.com
                Article
                Publisher ID: S2210-2612(16)30145-6
                DOI: 10.1016/j.ijscr.2016.05.024
                PMC ID: 4885136
                PubMed ID: 27232291
                SO-VID: 7f2887d1-760a-474e-b992-4c66b8a0223f
                Copyright © © 2016 The Author(s)
                License:

                This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                Date received : 20 March 2016
                Date revision received : 2 May 2016
                Date accepted : 15 May 2016
                Categories
                Subject: Case Report

                Keywords: extra medullary hematopoiesis,adrenal,incidentiloma,thalassemia,case report
                Data availability:
                Keywords: extra medullary hematopoiesis, adrenal, incidentiloma, thalassemia, case report

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