Abstract
Primary liposarcoma of the spermatic cord (LSC) is a rare neoplasm; there are fewer than 100 cases reported in the English literature worldwide. A seventy-one year-old man, who had undergone radical retropubic prostatectomy (RRP) for localized prostate cancer in November 2004, noticed the enlargement of a mass in the left scrotum. Subsequently he underwent a biopsy of the lesion, which documented suspicion of leiomyosarcoma of the spermatic cord. Left radical orchiectomy was performed extending to the resection margin. The pathological examination showed a dedifferentiated liposarcoma of the left spermatic cord. To the best of our knowledge, this is the first report of LSC after RRP for prostate cancer in the English literature.
Radical retropubic prostatectomy (RRP) is a standard treatment option for clinically localized prostate cancer. Inguinal hernia is one of the major morbidities after RRP since its incidence is 6.7% to 21% (1, 2). From speculations in previous reports, certain kinds of manipulation during RRP that can weaken or distort the normal fascia structure at the internal inguinal ring, including incision and retraction of the transversalis fascia with a retractor, stretching of the vas deferens and incision of the endopelvic fascia, may have an important role (2). The pathogenesis of inguinal hernia after RRP is still largely unknown.
Liposarcomas of the spermatic cord (LSCs) are rare tumors. Liposarcomas are malignant neoplasms of adipose tissue arising from primitive mesenchymal cells. In almost 70% of cases they are located in the extremities and the retroperitoneum. The spermatic cord is a rare site of origin, accounting for about 3-7% of all liposarcoma (3). The first case of LSC after RRP for prostate cancer is reported here.
Case Report
A 71-year-old man noticed the enlargement of a mass in the left hemiscrotum for more than five months. He had undergone RRP for localized prostate cancer in November 2004. The pathological findings of the prostate had been Gleason sum 3+4=7, pT2a and negative margins. In February 2008, the serum prostate-specific antigen (PSA) level was 0.057 ng/ml and neither biochemical failure nor recurrence was confirmed. Computed tomography (CT) revealed a 35×25 mm low-density round mass in the left spermatic cord. Magnetic resonance imaging (MRI) showed a smooth margined low-intensity mass on T1- and T2-weighted image, which involved the epididymis and left spermatic cord (Figure 1). Subsequently the patient underwent a biopsy of the lesion. The pathology of the biopsy specimen was suspicious of leiomyosarcoma of the spermatic cord. In April 2008, left radical orchiectomy was performed extending to the resection margin. Macroscopically, the lesion was a capsulated mass (95×55 mm) composed mostly of adipose tissue infiltrating the spermatic structures (Figure 2). The pathological examination showed a well-differentiated liposarcoma which revealed fat cells and lipoblasts, and a dedifferentiated section which revealed spindle cells having atypical highly multiplied nuclei like a myxofibrosarcoma (Figure 3), and resulted in negative margins. There was no evidence of recurrence on CT and physical examination four months after surgery.
Discussion
The present case underwent conventional RRP and suffered from liposarcoma of the spermatic cord (LSC) beside the inguinal region, not an inguinal hernia, four years after prostatectomy. To date, the relationship between tumorigenesis of LSC and the RRP procedure which has some impact on the inguinal canal and inguinal region seems to be unclear, and no cases of LSC after RRP have been reported in the English literature.
LSC usually presents as a painless scrotal swelling, progressing in size slowly during a period ranging from months to years, as in the present case. Occasionally, a previously stable mass can rapidly increase in size. The average patient age at presentation is 55 years (range 16.5-85) with a slight right preponderance. Fewer than 6% of cases have a history of scrotal surgery or trauma (3). The clinical diagnosis in these conditions may be difficult, since the tumor can easily be mistaken for an inguinal hernia, hydrocele or spermatocele, as well as a tumor of the testis and epididymis. However, in the present case, a groin mass was palpable as a hard and fixed tumor. Therefore, the mass was suspected to be malignant.
Though liposarcomas have rarely been diagnosed preoperatively, ultrasonography, CT and MRI could provide useful information about the lipomatous nature of these masses.
The majority of spermatic cord tumors are benign (70-80%); lipoma is the most common. Malignant spermatic cord tumors are more frequently sarcomas (4). Rhabdomyo-sarcomas usually appear in young people and have a poor prognosis. Leiomyosarcomas, fibrosarcomas and liposarcomas are more commonly seen in older patients. LSC have been classified histologically into four categories, well-differentiated, dedifferentiated, myxoid and pleomorphic. Well-differentiated liposarcomas are further divided into adipocytic (lipoma-like), sclerosing liposarcoma, and into the two rare types of inflammatory and spindle cell liposarcoma. The sclerosing form is far more common in the retroperitoneum and spermatic cord than elsewhere (5).
Liposarcomas tend to spread primarily by local extension. When diagnosed or suspected preoperatively, radical orchiectomy with wide local excision is the recommended treatment for LSC. Adequate local resection provides the best chance of eradicating this disease. No therapeutic advantage has been attributed to superficial inguinal or retroperitoneal lymphadenectomy because liposarcomas tend to metastasize hematogenously rather than via the lymphatics.
Radiotherapy is controversial for local control of LSC. Liposarcomas are the most radiosensitive of all sarcomas and in some cases remission has been achieved with radiotherapy alone, although radiotherapy in eleven cases was reported to be associated with recurrence in five (3). In contrast, out of the 17 patients in some other studies treated with surgery and radiation, none had local recurrence (6-8). These retrospective reports provide no definitive evidence, but two prospective randomized trials on soft tissue sarcoma established that the addition of radiation to surgery substantially and significantly reduced the likelihood of local recurrence (9, 10). However, previously reported LSC series seemed not to be sufficiently large for a statistical analysis to demonstrate the necessity of additional radiotherapy. Our personal opinion is that radical surgical refinement with a negative margin is mandatory, whereas radiotherapy can be delayed if a close follow-up is performed. The role of chemotherapy for liposarcoma is not documented adequately.
In previous reports, the local recurrence rate after resection alone was 30-50% and local recurrence was the most common pattern of failure. Occasionally, local recurrence can be late. Thus, the customary 5-year landmark is not adequate to assess the therapeutic outcome. Regardless of initial therapy, the risk of local recurrence and subsequent increase in grade always necessitates long-term follow-up.
Footnotes
- Received July 28, 2008.
- Revision received December 9, 2008.
- Accepted December 15, 2008.
- Copyright© 2009 International Institute of Anticancer Research (Dr. John G. Delinassios), All rights reserved