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Keywords:

  • osteosarcoma;
  • sarcoma;
  • testis

Abstract

  1. Top of page
  2. Abstract
  3. Introduction
  4. Case report
  5. Discussion
  6. References

Abstract  This report describes a case of primary osteosarcoma of the testis in a 60-year-old man. Treatment consisted of an inguinal orchiectomy with no adjuvant therapy. The patient is alive and doing well without recurrent disease at 18 months after diagnosis. Only three reports have been published on primary osteosarcoma of the testis. The origin of this tumor from undifferentiated mesenchymal cells or from a malignant transformation of pre-existing teratomatous elements is still unclear. Management guidelines are difficult to establish due to the rarity of such tumors, but inguinal orchiectomy with careful follow up appears to be sufficient treatment.


Introduction

  1. Top of page
  2. Abstract
  3. Introduction
  4. Case report
  5. Discussion
  6. References

Genitourinary sarcomas are uncommon in adults, estimated at less than 2.7% of all sarcomas.1 We present a case of primary and pure osteosarcoma of the testis. As known from the literature, these cases are extremely rare; histogenesis, treatment and outcome at long-term follow up are still to be defined.

Case report

  1. Top of page
  2. Abstract
  3. Introduction
  4. Case report
  5. Discussion
  6. References

A 60-year-old white man presented with a 4-month history of a left scrotal mass, which had been rapidly growing in the last month. No history of prior urinary tract infections, scrotal trauma, or genitourinary surgery was reported. Physical examination revealed a hard left testicular tumor that did not fluctuate or transmit light. The right testis was normal. An ultrasound confirmed a left heterogeneous testicular mass. A computed tomography (CT) scan of the abdomen, pelvis and scrotum showed a 12 cm × 9.5 cm left testicular mass almost entirely calcified (Fig. 1), with no retroperitoneal or inguinal adenopathy. A chest radiograph and a CT scan, as well as alpha-fetoprotein (AFP), beta-human chorionic gonadotropin (β-HCG) and lactate dehydrogenase (LDH) were normal. Subsequently, a left inguinal orchiectomy was performed.

image

Figure 1. Computed tomography showed a 12 cm × 9.5 cm left testicular mass almost entirely calcified.

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Gross examination showed an encapsulated tumor contained within the tunica albuginea, weighing 700 g. It was firm to bony-hard on palpation, and attached to a spermatic cord 8 cm long. The longitudinal opening of the testis showed an off-white colored tumor which measured 12 cm × 9.5 cm. It was about 90% calcified (Fig. 2).

image

Figure 2. Longitudinal opening of the testis showed an off-white colored tumor. It was approximately 90% calcified.

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Microscopic examination revealed a tumorous formation of mesenchymal nature, displaying obvious malignant characteristics, such as a proliferation of fusiform cells with frank cytonuclear atypical features, along with a few abnormal mitoses. A marked bony differentiation represented by lines of variable thickness exists, often well calcified, with areas of cartilaginous differentiation (Fig. 3). No testicular tissue was seen, but the epididymis was identified. The final diagnosis was a well-differentiated osteosarcoma.

image

Figure 3. Microscopic examination revealed a sarcomatous cell proliferation with a marked bony differentiation (HE, ×200).

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The patient had an uneventful postoperative course. Quarterly follow-up CT of the chest, abdomen and pelvis, as well as tumor markers, remained negative 18 months postoperatively.

Discussion

  1. Top of page
  2. Abstract
  3. Introduction
  4. Case report
  5. Discussion
  6. References

Primary intratesticular sarcoma that is neither associated with germ cell elements nor paratesticular elements is a unique subset of intrascrotal sarcoma. To our knowledge, the tumor in our patient represents the fourth report of an intratesticular osteosarcoma.2–4

The three cases already reported were described in 30, 63 and 78-year-old patients, respectively. All the tumors were completely contained within the tunica vaginalis.

Little information exists concerning the histogenesis of an intratesticular osteosarcoma. Two hypotheses were suggested. The first one proposes a neoplastic transformation of sequestered embryonic remains of osteogenic tissue or primitive mesenchymal cells. The second one suggests a transformation of pre-existing teratomatous elements that acquired a potential for a preferential mesenchymal malignancy.5 It has been also suggested that rearrangement of the p53 gene, localized at 17p13, is probably important in the oncogenesis of osteosarcoma.6

Due to the rarity of primary testicular sarcomas, management recommendations are difficult to establish. The four patients, including ours, underwent inguinal orchiectomy with no adjuvant treatment. Careful surveillance of β-HCG and AFP which, although not specific for sarcoma, is useful to exclude serologic relapse from undetected germ cell tumor metastases,5 was carried out.

The prognosis of osteosarcoma of the testis is unclear. Follow up with no evidence of recurrence or metastasis was short, with only 9 weeks and 6 months in two patients.2,3 Nevertheless, Lee concluded that primary pure testicular osteosarcoma may be associated with a favorable prognosis; his patient remained well without evidence of disease 44 months after operation.4 Our patient is alive and well without recurrent disease at 18 months after diagnosis. At present, inguinal orchiectomy with strict and careful follow-up evaluations appears to be sufficient treatment.

References

  1. Top of page
  2. Abstract
  3. Introduction
  4. Case report
  5. Discussion
  6. References