Primary Leiomyosarcoma of Adrenal Gland with Tissue Eosinophilic Infiltration

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/ by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. Eosinophils are associated with numerous disorders, including helminthic parasitic infections, allergic diseases, and some tumors. 1 Although eosinophils are commonly encountered in human solid tumors, their functional role in neoplasm remains a matter of controversy. 1 The correlation of tumor-associated tissue eosinophilia (TATE) with prognosis has shown variable results in several malignancies. Primary adrenal leiomyosar-coma is a very rare malignant mesenchymal tumor with smooth muscle differentiation. Twenty-two cases have been reported in the English 5,6 and Korean 7 literature. The association of tissue eosinophilia with leiomyosarcoma is very rare, with only one case of uterine leiomyosarcoma reported in the literature. 4 We experienced a very rare case of primary adrenal leiomyosarcoma with marked tissue eosinophilia, it is the first case reported in the literature. A 28-year-old man presented with a three-month history of right flank pain, associated with persistent and stabbing pain and weight loss. Computed tomography showed a well-circum-scribed heterogeneously enhancing 13.8-cm mass located at the right suprarenal area (Fig. 1). No metastatic lesion was noted. White blood cell count and eosinophil differential count were within the normal range. Biochemical examination revealed no functional tumor of the adrenal gland. The patient had no signs or symptoms of human immunodeficiency virus or Epstein-Barr virus infection. The patient underwent a right open adrenalec-tomy. The resected adrenal gland mass showed a large lobulated mass (15×13×6 cm, 792 g) almost replacing the adrenal gland. The mass was a well-circumscribed and partially encapsulated solid tumor. On histologic evaluation, the spindle cell tumor showed a rim of fibrous tissue with entrapped atrophic adrenal cortical cells, geographic coagulation necrosis with surrounding fibrosis and frequent mitotic figures (25/10 high-power field) (Fig. 2A). The surgical resection margin was free of tumor. We used Fédération Nationale des Centres de Lutte Contre le Cancer (FNCLCC) or the tumor grading and the tumor grade was 3. The tumor cells were strongly cytoplasmic positive for smooth muscle actin (Fig. 3A) and desmin (Fig. 3B). However, pan-cy-tokeratin, CD117, S100-protein, and human melanoma black 45 were all negative. On the bases of clinical data, histomor-phological features and immunohistochemical studies, we diagnosed primary adrenal leiomyosarcoma. Interestingly, there was intense tissue eosinophilia (Fig. 2B). The eosinophilic infiltration was mainly …