Editor-in-Chief
Prof. Yi-Fei WANG,
Asian J Androl 2007; 9 (5): 723-725 |
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This web only provides the extract of this article. If you want to read the figures and tables, please reference the PDF full text on Blackwell Synergy. Thank you. - Letters to the Editor - Scrotal aggressive angiomyxoma mimicking inguinal hernia Chia-Chang Wu1,4, Stephen Shei-Dei Yang1,3, Daniel T. H. Chin2, Cheng-Hsing Hsieh1, Yu-Mei Hsueh4, Yao-Chou Tsai1,3 1Department of Urology, 2
Department of Pathology, Buddhist Tzu Chi General Hospital, Taipei Branch, Taibei 231, Taiwan,
China
Correspondence to: Dr Yao-Chou Tsai, Department of Urology, Buddhist Tzu Chi General Hospital, Taipei Branch, 289 Jianguo Road,
Xindian City, Taipei 231, Taiwan, China.
DOI: 10.1111/j.1745-7262.2007.00286.x Dear Sir, I am Dr Yao-Chou Tsai, from Department of Urology, Buddhist Tzu Chi General Hospital, Taipei Branch, Taipei, Taiwan, China. I write to you to present a case report of scrotal aggressive angiomyxoma (AAM). AAM is a rare, locally infiltrative mesenchymal benign neoplasm with a predilection for involving the female pelvis, perineum, vagina and cervix. Its occurrence in men is much less frequent and only scattered case reports have been reported. To our knowledge, only 43 cases of AAM occurring in men have been reported in the English-language literature [1]. AAM in the male inguinal area can present as an inguinal mass that is usually diagnosed and treated as inguinal hernia [2, 3]. Herein, we report an unusual case of a 40-year-old man with an AAM involving the scrotum, who underwent a herniorrhaphy at a prior hospital. The AAM was not removed. To our knowledge, this is the first documented case in which scrotal AAM was missed during surgical exploration for inguinal hernia. A 40-year-old man complained of a right scrotal mass, which had grown slowly over the previous10 years. No local pain or urinary tract symptoms were noted. The scrotal mass was initially diagnosed as an inguinal hernia and a herniorrhaphy was performed at a prior hospital. However, the scrotal mass still remained in situ after the operation. Therefore, he came to our hospital for further management. On physical examination, the right scrotal mass was elastic, non-tender, non-transilluminated and not reducible.
Scrotal ultrasonography revealed a homogeneous, hypoechoic, extratesticular, extraepididymal, well-demarcated mass
lesion. No calcification was present. Pelvic computed tomography (CT) demonstrated a 5.0 × 5.1 × In surgery, an incision through a previous herniorrhaphy wound was done and a large tumor was found within the spermatic fascia (Figure 1). The boundary of the tumor was well circumscribed and away from the right testicle and epididymis (Figure 2). The cut surface of the tumor was yellowish and gelatinous, and no necrosis or cystic changes were present. The frozen section of the tumor showed a loose myxoid and fibrous background containing blood vessels of variable caliber. The evenly distributed stromal cells were wavy, spindle or stellate in shape and had delicate cytoplasmic processes. Mitoses and cellular pleomorphism were lacking (Figure 3). The findings are consistent with AAM. The tumor was excised completely and the patient was discharged on the first postoperative day. The patient has remained free of local recurrence for 12 months since the operation. Since Steeper and Rosai [4] first described nine cases of aggressive angiomyxoma in female adults in 1983, its incidence has increased year by year. Male AAM tends to be an asymptomatic round mass lesion that is sometimes initially diagnosed as an inguinal hernia. Previously reported AAM that have been found incidentally during herniorrhaphy have been resected smoothly with satisfactory postoperative results [2, 3]. However, in clinical practice, it is possible that a scrotal AAM is missed if a concurrent inguinal hernia exists. To avoid this, detailed preoperative physical examination and confirmative tumor removal is necessary. Preoperative imaging studies do not always provide a correct diagnosis because the tumor usually shows an invasive and hypervascular picture that makes the tumor seem malignant [5]. Gray-scale ultrasonography demonstrates a hypoechoic, homogeneous and well-demarcated mass without significant flow using color Doppler ultrasonography. On CT, AAM is hypodense relative to muscle and hypoattenuating with specific swirling internal architecture on enhanced scans. The tumor has high signal intensity using T2-weighted magnetic resonance imaging. The swirling internal architecture of lower intensity after intravenous contrast enhancement is also seen within the high signal tumor. Grossly, AAM is yellowish in color and usually well demarcated and encapsulated. The cut surface demonstrates gelatinous and glistening material with delicate, white, fibrous strands. Microscopically, AAM demonstrates stellate and spindle-shaped cells in a loose myxoid stroma without mitosis or nuclear atypia. Vascularization is abundant, and small to large thick-walled vessels together with collagen fibrils are scattered in the hypocellular background. Aggressive angiomyxoma should be distinguished from other benign and low local recurrent potential lesions such as intramuscular myxoma, myxoid neurofibroma, myxoid or spindle cell lipoma, superficial angiomyxoma and angiomyofibroblastoma. The malignant tumors with widespread metastasis potential should also be considered a differential diagnosis (the myxoid variants of liposarcoma, malignant fibrous histiocystoma and embryonal/botryoid rhabdomyosarcoma) [6]. Characteristically, AAM is locally aggressive and infiltrative without metastasizing potential, with the exception of two cases of death reported by Blandamura et al. [7] and Siassi et al. [8]. Its local recurrence rate is approximately 37% and average time of recurrence is 9 months to 14 years postoperatively [2, 4]. In conclusion, AAM should be considered when an usually soft tissue mass is found during physical examination or surgical correction for inguinal hernia. Because of its highly recurrent characteristics, wide excision of the tumor and strict postoperative follow up is recommended. References 1 Idrees MT, Hoch BL, Wang BY, Unger PD. Aggressive angiomyxoma of male genital region. Report of 4 cases with immunohistochemical evaluation including hormone receptor status. Ann Diagn Pathol 2006; 10: 197_204. 2 Murakami S, Ishiga N, Sho T, Sakai K, Fujii Y, Yamamoto Y, et al. Aggressive angiomyxoma occurring in the scrotum: report of a case. Surg Today 1996; 26: 289_91. 3 Clatch RJ, Drake WK, Gonzalez JG. Aggressive angiomyxoma in men. A report of two cases associated with inguinal hernias. Arch Pathol Lab Med 1993; 117: 911_3. 4 Steeper TA, Rosai J. Aggressive angiomyxoma of the female pelvis and perineum. Report of nine cases of a distinctive type of gynecologic soft-tissue neoplasm. Am J Surg Pathol 1983; 7: 463_75. 5 Chihara Y, Fujimoto K, Takada S, Hirayama A, Cho M, Yoshida K, et al. Aggressive angiomyxoma in the scrotum expressing androgen and progesterone receptors. Int J Urol 2003; 10: 672_5. 6 Iezzoni JC, Fechner RE, Wong LS, Rosai J. Aggressive angiomyxoma in males. A report of four cases. Am J Clin Pathol 1995; 104: 391_6. 7 Blandamura S, Cruz J, Faure Vergara L, Machado Puerto I, Ninfo V. Aggressive angiomyxoma: a second case of metastasis with patient's death. Hum Pathol 2003; 34: 1072_4. 8 Siassi RM, Papadopoulos T, Matzel KE, Siassi RM, Papadopoulos T, Matzel KE. Metastasizing aggressive angiomyxoma. N Eng J Med 1999; 341: 1772. |
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