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Articles © The
authors | Journal compilation © J Curr Surg and Elmer Press Inc™ | www.jcs.elmerpress.com This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited 179 Case Report J Curr Surg. 2015;5(2-3):179-181 ressElmer Adrenal Hematoma Simulating Malignancy in Idiopathic Thrombocytopenic Purpura Aleksandr A. Reznichenko Abstract Unilateral adrenal hemorrhages are unusual and have different etiolo- gies. Very rarely adrenal hematoma could be presented as an adrenal mass and may have features of neoplastic lesion. There have been a number of case reports on adrenal hematoma that presented as an adrenal tumor in the past; however, there were no reports where it was described in patients with idiopathic thrombocytopenic purpura (ITP). We present a patient with ITP and large adrenal mass suspicious for malignancy, which was surgically excised, and turned out to be an adrenal hematoma. Keywords: Adrenal hemorrhage; Adrenal hematoma; Adrenal mass; Idiopathic thrombocytopenic purpura; Adrenal carcinoma Introduction Adrenalhematomasoccurrarelyandhavemultiplecauses[1].They were reported in patients with trauma, long-term use of nonsteroi- dal anti-inflammatory drugs, neurofibromatosis, liver transplanta- tion, primary and metastatic adrenal tumors, heparin-associated thrombocytopenia, anticoagulation, sepsis, and anti-phospholipid antibodies [2-4]. Adrenal hemorrhage could be spontaneous [2], and sometimes has clinical and radiographic features of adrenal neoplasm [2, 3, 5-9]. So far there were no reports of adrenal hema- tomas in patients with idiopathic thrombocytopenic purpura (ITP). Adrenal masses require comprehensive workup to rule out hormone-producing tumor and malignancy. Lesions larger than 6 cm are rarely benign and should be excised [2]. We pre- sent a 65-year-old female with ITP and 7 × 9 × 6 cm adrenal hematoma, simulating malignancy. Case Report A 65-year-old female with history of hyperlipidemia, hyper- tension, diabetes mellitus, hypothyroidism, coronary artery disease, depression, tonsillectomy, cholecystectomy, tubal ligation, colonoscopy, and arthroscopy presented with lower gastrointestinal bleed, thrombocytopenia, weakness, petechi- al rash and easy bruising. She was diagnosed with ITP and was treated with steroids. Her platelets count improved from 50 to 120 and she had colonoscopy with polypectomy. Three months later, she started to have abdominal pain in left upper quadrant (LUQ) radiating to her back. Abdominal computer tomography (CT) showed 7 × 9 × 6 cm left adrenal mass with calcifications and central and peripheral enhancement with IV contrast, highly suspicious for primary adrenal carcinoma or metastatic disease (Fig. 1). Comprehensive endocrinology evaluation, including plasma and urine aldosterone, plasma renin activity, plasma and urine metanephrines, plasma and urine catecholamines, free cortisol and dehydroepiandroster- one (DHEAS) did not show hormonally active adrenal tumor. There was no evidence of adrenal insufficiency. Radiological evaluation with CT of the neck, chest, abdomen and pelvis was negative for any primary malignancy. Indication for surgery was large mass suspicious for primary left adrenal carcinoma. Patient underwent left adrenalectomy. Operation was started laparoscopically. During the procedure, significant desmoplas- tic reaction was noted around the mass with involvement of the splenic and left renal veins, and the case was converted to open. Final pathology evaluation of the excised mass showed Manuscript accepted for publication August 19, 2015 Department of Surgery, Tulare Regional Medical Center, Tulare, CA, USA. Email: areznik9@yahoo.com doi: http://dx.doi.org/10.14740/jcs278w Figure 1. Abdominal CT with contrast, axial view. Left adrenal mass enhancing with IV contrast.
2.
Articles © The
authors | Journal compilation © J Curr Surg and Elmer Press Inc™ | www.jcs.elmerpress.com180 Adrenal Hematoma Simulating Malignancy J Curr Surg. 2015;5(2-3):179-181 organized adrenal hemorrhage with necrosis (Fig. 2). Patient had successful recovery and had no symptoms or recurrence for 2 years. Abdominal CT performed 24 months after surgery showed no evidence of recurrence (Fig. 3). Discussion Adrenal hematomas are rare entities and only few series were published [3, 4]. These lesions were most commonly described as a consequence of trauma, but also in patients taking non-ste- roidal anti-inflammatory drugs (NSAIDs), with neurofibroma- tosis, after liver transplantation, with primary and metastatic adrenal tumors, with heparin-associated thrombocytopenia, on anticoagulation, on hormonal therapy, with severe sepsis and anti-phospholipid antibodies [2-4]. Spontaneous idiopathic ad- renal hematomas were also reported [2, 5, 7, 8]. Clinical presentation of adrenal hematomas varies, and may include flank or abdominal pain (like in our patient), fe- ver, tachycardia, weakness, hypotension, nausea, vomiting, changes in mental status, weight loss, abdominal distention and palpable abdominal mass [2]. ITP is a clinical syndrome in which a decreased number of circulating platelets manifest as a bleeding tendency. The differential diagnosis includes such other causes of thrombo- cytopenia as leukemia, myelodysplasia, aplastic anemia, and adverse drug reactions. In ITP, an abnormal autoantibody, usually immunoglobulin G binds to circulating platelet mem- branes. Hemorrhages are the only clinical manifestations and they may be mild, such as petechiae, easy bruising, epistaxis, and gingival bleeding, although severe complications such as hemoptysis, gastrointestinal and genitourinary tract bleeding are also observed and are generally associated with marked thrombocytopenia. The most frequent cause of death in asso- ciation with ITP is intracranial bleeding [10, 11]. There was no adrenal hemorrhage reported in patients with ITP. We present a unique patient with ITP and adrenal hematoma. Imaging studies are important in the diagnosis of adre- nal masses. Both CT and magnetic resonance imaging (MRI) were successfully used to diagnose adrenal hemorrhage. Ac- cording to the literature, adrenal hematomas usually appeared as rounded or oval hypodense heterogeneous masses with or without calcifications, and absence of contrast enhancement. Benign adrenal lesions bigger than 6 cm are extremely rare and always should be surgically excised, after evaluation for pos- sible secreting adrenal tumor [2, 3]. Adrenal hematomas could masquerade malignant neoplasm like in our patient, who had 7 × 9 × 6 cm adrenal mass, enhancing with IV contrast. Hormonal testing to distinguish functional and nonfunc- tional adrenal mass is extremely important and should include evaluation for pheochromocytoma, Cushing’s disease, primary and secondary hyperaldosteronism, and virilizing tumors. All biochemical studies in our patient were negative for hormone- producing neoplasm. Adrenal hematomas were previously described in patients with bleeding diathesis, such as heparin-induced thrombocy- topenia and anticoagulation therapy; however, there were no reports in patients with ITP. This is a first case of adrenal mass simulating neoplasm in patient with ITP. Conclusion Adrenal hematomas are rare and could simulate malignancy. This is a first report of adrenal hematoma in ITP, presenting as a mass. Lesions larger than 6 cm should be surgically excised after ruling out hormonally producing tumor. Competing Interests The author declares that he does not have any competing in- terests. Abbreviations CT: computer tomography; MRI: magnetic resonance imag- ing; ITP: idiopathic thrombocytopenic purpura; DHEAS: de- Figure 3. Abdominal CT with contrast 2 years after surgery, axial view. No evidence of recurrence. Figure 2. Pathology. Adrenal hemorrhage with necrosis.
3.
Articles © The
authors | Journal compilation © J Curr Surg and Elmer Press Inc™ | www.jcs.elmerpress.com 181 Reznichenko J Curr Surg. 2015;5(2-3):179-181 hydroepiandrosterone; NSAIDs: non-steroidal anti-inflamma- tory drugs; LUQ: left upper quadrant References 1. Greendyke RM. Adrenal Hemorrhage. Am J Clin Pathol. 1965;43:210-215. 2. Christoforides C, PetrouA, Loizou M. Idiopathic unilateral adrenal haemorrhage and adrenal mass: a case report and review of the literature. Case Rep Surg. 2013;2013:567186. 3. Fedala NS, Haddam AM, Chentli F, Youcef RS, Kalafat N. Adrenal Hematoma: A Report of Three Cases. Journal of endocrinology and diabetes mellitus. 2014;2:117-121 4. Vella A, Nippoldt TB, Morris JC, 3rd. Adrenal hemor- rhage: a 25-year experience at the Mayo Clinic. Mayo Clin Proc. 2001;76(2):161-168. 5. Kobayashi T, Uenoyama S, Miura K, Takehara Y. Idi- opathic unilateral adrenal hematoma: report of a case. Surg Today. 2004;34(3):279-282. 6. Abdullah KG, Stitzlein RN, Tallman TA. Isolated adrenal hematoma presenting as acute right upper quadrant pain. J Emerg Med. 2012;43(4):e215-217. 7. Imachi H, Murao K, Yoshimoto T, Sugimoto M, Kake- hi Y, Hayashi T, Kushida Y, et al. Idiopathic unilateral adrenal hemorrhage in an elderly patient. Endocrine. 2010;37(2):249-252. 8. el Khader K, el Ghorfi MH, Ouali M, Ibnattya A, Hachimi M, Lakrissa A. [Spontaneous hematoma of the adrenal glands]. Prog Urol. 2001;11(3):517-519. 9. Marti JL, Millet J, Sosa JA, Roman SA, Carling T, Udels- man R. Spontaneous adrenal hemorrhage with associated masses: etiology and management in 6 cases and a review of 133 reported cases. World J Surg. 2012;36(1):75-82. 10. Kessler CM, Sandler SG. Immune Thrombocytopenic Pur- pura- Medscape Reference. April 21, 2014. 11. Cortelazzo S, Finazzi G, Buelli M, Molteni A, Viero P, Barbui T. High risk of severe bleeding in aged patients with chronic idiopathic thrombocytopenic purpura. Blood. 1991;77(1):31-33.
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