Well-differentiated liposarcoma of spermatic cord presenting as recurrent inguinal hernia: a case report

Introduction

Liposarcomas are the most common type of soft tissue sarcoma, representing approximately 20% of all malignant neoplasms of mesenchymal origin. Well-differentiated liposarcoma and dedifferentiated liposarcoma are the two primary subgroups and make up approximately 40–45% of all liposarcomas (1,2). These tumors present as slow, enlarging masses and most commonly arise from the retroperitoneum and limbs. Well-differentiated liposarcoma arising from the paratesticular region is a rare occurrence with only a few hundred cases reported in the literature. Spermatic cord liposarcoma presenting as recurrence of a previously repaired inguinal hernia is exceedingly rare, with only a handful of documented case reports (3-13). We present the case for a patient who was incidentally found to have a well-differentiated liposarcoma of the spermatic cord after undergoing an operation to repair what was presumed to be a recurrent inguinal hernia. We present this case in accordance with the CARE reporting checklist (available at https://acr.amegroups.com/article/view/10.21037/acr-23-69/rc).

Case presentation

A 65-year-old man with a past medical history significant for hypertension, hyperlipidemia, cervicalgia, chronic low back/shoulder pain, active tobacco use (10 pack-year history), and prior robotic-assisted laparoscopic left inguinal hernia repair with mesh at an outside hospital. He presented to the Veterans Affair North Texas Health Care System general surgery clinic with a left groin bulge. He reported noticing a persistent bulge in the left inguinal region almost immediately after his index surgery, which was 2 years previously. However, he did not seek evaluation initially because he thought it was simply postoperative fluid accumulation. Initial patient interview was not concerning for acute incarceration or strangulation. At the time of presentation, the patient was tolerating a general diet without any issues. He also reported voiding and stooling patterns that were unchanged from his baseline. Review of systems was overall unremarkable. The patient stated that even though he was experiencing no major discomfort from his hernia recurrence, he wanted to have it repaired to avoid future complications. Physical exam on initial evaluation was only notable for a chronically incarcerated left inguinal hernia containing a large fat mass within the left hemiscrotum; there were no overlying skin changes and both testicles were easily palpable. The hernia contents descended into the scrotum and were firm, but non-tender. His laboratory tests were unremarkable. Preoperative computed tomography (CT) demonstrated a residual lipomatous lesion that extended from the hernia neck near the repair mesh to the left hemiscrotum, measuring 8 cm in its longest dimension (Figure 1). At that time, the decision was made to proceed with an elective, open repair of his suspected recurrent left inguinal hernia.

Figure 1 Preoperative CT depicting suspected recurrent left inguinal hernia. Top two panels represent axial view. Bottom left panel represents sagittal view. Bottom right panel represents coronal view. CT, computed tomography.

The patient arrived to the operating room on the day of his surgery without any major complaints or changes to his baseline state of health. The initial part of the operation proceeded normally and without complications. Upon identification and exploration of the spermatic cord structures, a hardened mass was palpated in the left inguinoscrotal area. No indirect hernia sac could be identified, and the transversalis fascia was intact. The mass was then isolated after careful dissection. Given the suspicious nature of this mass, intraoperative consultation from urology was obtained. After their evaluation, urology staff also shared the concern that this may represent a malignant neoplasm. Unable to obtain consent for formal oncologic resection, the decision was made to proceed with an excisional biopsy and have the patient follow up in clinic with urology. The surgical wound was closed. He had an uneventful recovery in the post-anesthesia care unit. He was discharged home in good condition. Histopathologic evaluation of the excised lipomatous mass (measuring 10 cm × 6.8 cm × 5.5 cm) was consistent with an atypical lipomatous tumor/well-differentiated liposarcoma. The patient did well postoperatively. He was evaluated by our urology colleagues in clinic with discussion and plans that included formal oncologic resection and a left orchiectomy. However, at this juncture, the patient elected to proceed with watchful waiting as he did not desire further surgical intervention.

All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Discussion

Paratesticular liposarcomas are rare clinical entities that typically present as a slowly-enlarging mass. Review of the literature demonstrates that these neoplasms are often mistaken for hydroceles, inguinal hernias, or cord lipomas. Patients presenting with paratesticular liposarcoma are typically in the 5^th through 7^th decades of life; though a patient as young as 5 years of age has been reported (14). Risk factors for soft tissue sarcomas, including paratesticular liposarcomas, include genetic mutations, exposure to external beam radiation, and exposure to certain chemicals (15). Previous literature includes only a few series describing patients with spermatic cord liposarcoma who presented as suspected recurrent inguinal hernias failing initial repair. For our patient, the operative report from the initial robotic-associated laparoscopic inguinal hernia repair was not available to clarify presence of a hernia defect and/or abnormal intraoperative findings. Though, from an intraperitoneal view, there may have been no sign of a spermatic cord mesenchymal lesion or any other lesion suspicious for malignancy. Upon presentation to our institution, preoperative CT was ordered to determine if this was a postoperative chronic fluid collection or a recurrence and to guide surgical planning. The radiographic findings appeared consistent with lipomatous tissue (e.g., similar density as surrounding subcutaneous fat, no invasion of surrounding structures, and well-demarcated borders) and thus did not raise suspicion for neoplasm in the preoperative setting. The relatively shortened interval from first repair to re-presentation should have perhaps raised suspicion that what was presumed to be a fat containing hernia may have actually been a more insidious process. However, because the patient did not undergo surgical intervention within the same institution, the details of the index operation were not clear and the CT scan findings raised the suspicion of a recurrence.

Though a standardized treatment pathway for patients with paratesticular liposarcoma has not been established, most cases are treated with wide local excision plus possible orchiectomy. Data from a retrospective review of 265 cases (all subtypes included) noted improved recurrence-free survival for those who underwent complete tumor resection with orchiectomy versus those who underwent tumor resection only (16). For six of the published case reports focusing on individuals with prior hernia repairs, patients also received radiation therapy in addition to their tumor resection (5,8-10,12,17). Reasons for adjuvant radiation were described as the presence of lesions with poorly differentiated components, incomplete resection, and/or recurrence. As is the case for any malignant tumor, serial follow-up and surveillance has been recommended for these patients after tumor resection. Published follow-up varies widely; ranging from a few months to 8 years. Although there is limited data on mortality and recurrence after resection for all-comers with paratesticular liposarcoma, the majority of patients for whom follow-up data is available do not have a recurrence. More specifically, for patients who had prior inguinal hernia repairs and then re-presented with a paratesticular liposarcoma (11 case reports), at last follow-up, five had no tumor recurrence, two had tumor recurrence, and four had no information regarding tumor recurrence (3-13). Most of these cases were patients with well-differentiated liposarcoma or myxoid subtype which likely contributes to their improved outcomes. This differs for patients with dedifferentiated paratesticular liposarcomas, for whom recurrence is higher, outcomes are poorer, and thus, the use of chemotherapy and radiation is more frequent (18-20).

Conclusions

While paratesticular liposarcoma is a rare entity, clinicians should maintain this pathology in their differential diagnosis. Our case highlights the added challenge of diagnosing paratesticular liposarcoma in cases where patients present with early recurrence of a previously repaired inguinal hernia. Timely diagnosis and management of paratesticular liposarcoma that presents as early hernia recurrence requires appropriate cross-sectional imaging, surgical exploration, and histopathologic evaluation. This thorough approach matters because recurrent hernias can sometimes take months, if not years, to be addressed; especially if they are asymptomatic. In patients who present with a groin bulge after a prior inguinal hernia repair, it is important that malignancy be considered and that reoperation not be delayed until the time symptoms develop. The mainstay of treatment for liposarcoma of the groin remains wide local excision with orchiectomy and/or radiation as potential adjuncts.

Acknowledgments

Funding: None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://acr.amegroups.com/article/view/10.21037/acr-23-69/rc

Peer Review File: Available at https://acr.amegroups.com/article/view/10.21037/acr-23-69/prf

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://acr.amegroups.com/article/view/10.21037/acr-23-69/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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