案例報告

罕見睪丸旁侵襲性血管黏液瘤擬副睪腫瘤之病例報告

陳祺方^{1, 2}、王道遠^2,3、陳建志^{1, 3}、林永杰⁴

¹馬偕紀念醫院泌尿科, ²馬偕醫學院³馬偕紀念醫院淡水院區⁴馬偕紀念醫院新竹分院

Rare Paratesticular Aggressive Angiomyxoma Mimicking an Epididymal Tumor in an 82-year-old Man: One Case report

Chi-Fang, Chen ^{1, 2}. Li-Chen, Chen^{1, 2}

¹ Department of Urology, Mackay Memorial Hospital, Taipei, Taiwan

² Department of Pathology, Tamsui branch, MacKay Memorial Hospital, No.45, Minsheng Rd., Tamsui Dist., New Taipei City, Taiwan

³ Department of Medicine, Mackay Medical College, Taipei, Taiwan

⁴ Department of Urology, Hsinchu branch, MacKay Memorial Hospital, No. 690, Sec. 2, Guangfu Rd., East Dist., Hsinchu City, Taiwan

Background:

Aggressive angiomyxoma (AAM) is a rare mesenchymal tumor. In 1983, Steeper et al. described nine cases of female pelvis and perineum tumors which they called AAMs.[1] In men, less then 50 cases have been reported, involving the scrotum, pelvic region, perineum and inguinal area. [2]AAM is difficult to diagnose without pathological proof, and misdiagnosis as hydrocele, spermatocele, testicular and paratesticular neoplasia is common.[1, 3] In the literature review, only four cases were identified. Herein, we report on a 82-year-old male patient who had paratesticular AAM, and to the best of our knowledge is the oldest case in the literature.

Case presentation:

A 82-year-old man recently diagnosed with prostate cancer, presented to our outpatient department with a right palpable painless paratesticular mass. The physical examination revealed a round paratesticular mass that was firm in consistency without tenderness. It was fixed on the lower pole of the right testis and mimicked an epididymal or testicular neoplasm. Scrotal sonography showed a heterogenous round mass over the tail of epididymis (Fig 1a, b). Rich blood supply was noted under color Doppler sonography (Fig 1c). The patient denied history of cryptorchidism or previous trauma. Further imaging, including whole abdominal computer tomography scan and bone scan showed no lymphadenopathy or distant tumor metastasis. The tumor markers LDH (Lactate dehydrogenase), beta-HCG (Human Chorionic Gonadotropin), alpha-fetal protein (AFP), and CEA were negative.

Epididymal tumor excision by a scrotal approach was arranged, however the operation was shifted to right orchiectomy because of severe tumor adhesion onto the testis. Gross examination showed a gray, round, and solid tumor with clear boundaries (Fig 2). Histologically, the testicular adnexal mass was a deep angiomyxoma composed of bland spindle cells in a myxoid matrix containing delicate vessels. The lesion was immunoreactive for CD34, smooth muscle actin and desmin, and negative for S100, estrogen and progesterone receptors. The patient has been followed-up for 4 months without disease recurrence.

Conclusion:

In men, AAMs should be considered in the differential diagnosis of paratesticular tumors. To the best of our knowledge, four paratesticular AAMs have been reported. It is a slowly growing benign tumor, but its locally aggressive and infiltrative nature makes it difficult to resect alone without orchiectomy. Although approximately 70% of cases locally recurred within the first 3 years[3], our review showed a lower rate of local recurrence of AAMs in males. Due to its atypical presentation and rarity in males, we presented our clinical images and typical histologic findings for future references