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Year : 2013  |  Volume : 56  |  Issue : 3  |  Page : 319-321
Adenomatoid tumor of adrenal gland: A rare case report


Department of Pathology, The First Affiliated Hospital of Fujian Medical University, Fuzhou, China

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Date of Web Publication24-Oct-2013
 

How to cite this article:
Li S, Wang X, Zhang S. Adenomatoid tumor of adrenal gland: A rare case report . Indian J Pathol Microbiol 2013;56:319-21

How to cite this URL:
Li S, Wang X, Zhang S. Adenomatoid tumor of adrenal gland: A rare case report . Indian J Pathol Microbiol [serial online] 2013 [cited 2020 Feb 20];56:319-21. Available from: http://www.ijpmonline.org/text.asp?2013/56/3/319/120413


Sir,

Adenomatoid tumors are rare benign tumors of mesothelial origin, usually occurring in male and female genital tracts and rarely in other parts, such as pancreas, heart, pleura, omentum, lymph node, and adrenal gland. [1],[2],[3],[4],[5],[6],[7] The tumor is usually discovered accidentally and is easy to misdiagnose by clinical and imaging examination. However, there is no recurrence or metastases reported in the literature. We report a case of adenomatoid tumor arising from the left adrenal gland in an adult male.

A 32-year-old man had a left adrenal gland mass for the past 1 month, which was found accidentally by ultrasound. He was admitted to our hospital for evaluation. Physical examination was non-specific. Both clinical and blood analyses such as cortisol, whole blood, electrolyte, and urine tests were normal. Computed tomography (CT) scan demonstrated a left adrenal mass measuring about 4.9 × 3.6 cm in size. The lesion was of uneven density with a relatively smooth edge. Further, enhanced CT scan showed the majority of tumor had no significant enhancement, but a small part had mild to moderate enhancement [Figure 1]. Also, this tumor was totally resected by laparoscopic adrenalectomy.

On gross examination, the size of the removal tissue was 6.0 × 5.0 × 2.0 cm. Also, the cut surface had a tumor, which was measuring 4.0 × 2.0 × 2.0 cm with soft texture resembling jelly, and focal hemorrhage [Figure 2]. Microscopically, the boundary between the tumor and adrenal gland was unclear [Figure 3]. The tumor was composed of cystic and sinusoid-like channels, with lymphoid tissue or lymphoid follicular hyperplasia. A few of the cystic walls were adhered to membrane-like eosinophilic materials and the intracavity contained plasma-like substance. The shapes of the tumor cells were varied, such as flat, oval, and irregular. Signet-ring - like cells were also observed. No mitosis or necrosis was identified.

Immunohistochemically the tumor cells stained with cytokeratin 5/6, calretinin, D2-40, mesothelial cell (MC), and vimentin, and focally with epithelial membrane antigen (EMA). Lymphocytes showed positive immunoreactivity to lymphocyte antibody (LCA). Tumor cells were negative to chromogranin A (CgA), neuron-specific enolase (NSE), and cluster of differentiation 10 (CD10). Ki-67 index was 1%. After 2 1 / 2 years of follow-up, no recurrences or metastases were found.
Figure 1: Enhanced CT showed that the mass was wildly heterogeneously contrast enhancing

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Figure 2: Cross section of adrenalectomy specimen showing homogenous pinkish white

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Figure 3: Microscopic view of adenomatoid tumor. The mass has lymphoid follicular hyperplasia and wa s involving adjacent adrenal tissues (white arrow) (H and E, x40)

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Adenomatoid tumors were first reported in 1945. [8] They are common in male and female genital tract, and rare in the other parts. Immunohistochemical and ultrastructural features were consistent with mesothelial differentiation. [1] To our knowledge, adrenal surface is not covered by mesothelial cells. Some researchers believe that adrenal adenomatoid tumor derived from mesothelial rests.[9] A persuasive hypothesis was proposed in view of embryologic occurrence that the original mesothelial retraction is relevant to the process of Mόllerian tube of gonadal development. [6] So far, there is no report about the genetics of adrenal adenomatoid tumor, except for succinate dehydrogenase complex subunit D (SDHD) mutation.[10] The age of occurrence ranges from 24 to 65 years, and majority of those affected are men. The left side appears to be more commonly occurred than the right side. Most cases are asymptomatic and discovered accidentally. In rare cases, adrenal adenomatoid tumors accompany other diseases and symptoms such as acquired immune deficiency syndrome (AIDS), hematuria, cysts, and kidney stones. It is difficult to distinguish adenomatoid tumor from the other tumors of adrenal glands by ultrasound, CT, and magnetic resonance imaging (MRI) examination. The tumor sizes of adrenal adenomatoid tumors are variable, ranging from 0.5 to 19.0 cm. Also, the sections mostly present as solid peach pink or gray, with or without cystic areas. Microscopically, tubulocystic pattern is often seen, lined by epithelioid to flat cells. Lymphoid follicles and malnutrition calcification can occur. [1] Sometimes, signet-ring -like cells are present, which lead to misdiagnosis as metastatic adenocarcinoma. Immunohistochemistry shows positive staining of calretinin, cytokeratin 5/6, MC, D2-40, and ultrastructure by electron microscopy indicates that the tumor cells have long and dense microvillus. The mesothelial origin of adenomatoid tumor is supported by both of them. [1]

Adrenal adenomatoid tumors can be safely removed by laparoscopy. Tumor tissue involving adjacent adrenal tissues does not affect the prognosis. Metastasis or recurrences have not been reported, though occasional ones display an infiltrative growth pattern. [9]

 
   References Top

1.Garg K, Lee P, Ro JY, Qu Z, Troncoso P, Ayala AG. Adenomatoid tumor of theadrenal gland: A clinicopathologic study of 3 cases. Ann Diagn Pathol 2005;9:11-5.  Back to cited text no. 1
    
2.Overstreet K, Wixom C, Shabaik A, Bouvet M, Herndier B. Adenomatoid tumor of the pancreas: A case report with comparison of histology and aspiration cytology. Mod Pathol 2003;16:613-7.  Back to cited text no. 2
    
3.Natarajan S, Luthringer DJ, Fishbein MC. Adenomatoid tumor of the heart: Report of a case. Am J Surg Pathol 1997;21:1378-80.  Back to cited text no. 3
    
4.Minato H, Nojima T, Kurose N, Kinoshita E. Adenomatoid tumor of the pleura. Pathol Int 2009;59:567-71.  Back to cited text no. 4
    
5.Yeh CJ, Chuang WY, Chou HH, Jung SM, Hsueh S. Multiple extragenital adenomatoid tumors in the mesocolon and omentum. APMIS 2008;116:1016-9.  Back to cited text no. 5
    
6.Isotalo PA, Nascimento AG, Trastek VF, Wold LE, Cheville JC. Extragenital adenomatoid tumor of a mediastinal lymph node. Mayo Clin Proc 2003;78:350-4.  Back to cited text no. 6
    
7.Timonera ER, Paiva ME, Lopes JM, Eloy C, van der Kwast T, Asa SL. Composite adenomatoid tumor and myelolipoma of adrenal gland: Report of 2 cases. Arch Pathol Lab Med 2008;132:265-7.  Back to cited text no. 7
    
8.Golden A, Ash JE. Adenomatoid tumors of the genital tract. Am J Pathol 1945;21:63-79.  Back to cited text no. 8
    
9.El-Daly H, Rao P, Palazzo F, Gudi M. A rare entity of an unusual site: Adenomatoid tumour of the adrenal gland: A case report and review of the literature. Patholog Res Int 2010;2010:702472.  Back to cited text no. 9
    
10.Limbach AL, Ni Y, Huang J, Eng C, Magi-Galluzzi C. Adenomatoid tumour ofthe adrenal gland in a patient with germline SDHD mutation: A case report andreview of the literature. Pathology 2011;43:495-8.  Back to cited text no. 10
    

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Correspondence Address:
Sheng Zhang
Department of Pathology, The First Affiliated Hospital of Fujian Medical University, Fuzhou
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.120413

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