Primary leiomyosarcoma of the spine: an analysis of imaging manifestations and clinicopathological findings

Zhang, Jiahui; Chen, Yongye; Xing, Xiaoying; Wang, Qizheng; Liu, Ke; Zhang, Enlong; Lang, Nada

doi:10.1186/s13244-022-01336-y

Cited by 5 publications

(3 citation statements)

References 15 publications

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“…[ 3 ] Recently, published studies documented several primary cervical spinal LMS lesions [ Table 1 ] following both surgery and oncologic treatment in which the longest recurrence-free survival was 38 months. [ 7 , 9 , 10 ]…”

Section: Discussionmentioning

confidence: 99%

A rare presentation of leiomyosarcoma metastasis to the cervical spine: A case report and a brief review

Koruga,

Rončević,

Koruga

et al. 2024

Surgical Neurology International

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Background: Leiomyosarcoma (LMS) is a rare malignancy that originates from smooth muscle. The most common sites of metastases include the lungs, liver, kidney, and skin. Notably, metastases of LMS to the central nervous system/or spine are extremely rare. When a cervical spinal LMS lesion was encountered, the patient successfully underwent gross total tumor resection with negative margins. Case Description: A 63-year-old female had undergone an anterior cervical C5–C7 diskectomy and fusion 18 years ago and resection of a retroperitoneal LMS 3 years ago. She newly presented with right-sided numbness and pain of 2 months duration that correlated with a focal right-sided C5-level hemiparesis (i.e., 4/5 motor strength). When the cervical magnetic resonance demonstrated a right-sided C5 intralaminar mass with extension into the C5–C6 foramen, she underwent posterior tumor resection; pathologically, this proved to be an LMS metastasis. Respectively, 1- and six months postoperatively, follow-up magnetic resonance imaging scans showed no tumor recurrence; she tolerated adjuvant oncological treatment accompanied by physical therapy. However, in one postoperative year, the lesion recurred, and she is presently under consideration for additional surgical management. Conclusion: Gross total surgical resection is the first line of treatment for patients with metastatic LMS. Here, a patient with a C5 laminar/C5–C6 foraminal bony LMS metastasis underwent posterior tumor resection accompanied by adjuvant oncological treatment but exhibited disease recurrence within one postoperative year.

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Section: Discussionmentioning

confidence: 99%

A rare presentation of leiomyosarcoma metastasis to the cervical spine: A case report and a brief review

Koruga,

Rončević,

Koruga

et al. 2024

Surgical Neurology International

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“…Due to the lack of pathognomonic radiologic features, it may be challenging to make a definitive diagnosis. Hence a multimodal investigative approach is needed [71]. Using a well-rounded approach comprised of MRI, CT, and PET seems to be the most beneficial as it can help investigate the extent and site of the primary tumor, in addition to proper diagnoses, catering to individual patients.…”

Section: Discussionmentioning

confidence: 99%

Leiomyogenic Tumor of the Spine: A Systematic Review

Kharbat,

Balasubramanian,

Sankarappan

et al. 2024

Cancers

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The study cohort consisted of 83 patients with a mean age of 49.55 (SD 13.72) with a female preponderance (60 patients). Here, 32.14% of patients had primary LTS; the remaining were metastases. Clinical presentation included nonspecific back pain (57.83%), weakness (21.69%) and radicular pain (18.07%). History of uterine neoplasia was found in 33.73% of patients. LTS preferentially affected the thoracic spine (51.81%), followed by the lumbar (21.67%) spine. MRI alone was the most common imaging modality (33.33%); in other cases, it was used with CT (22.92%) or X-ray (16.67%); 19.23% of patients had Resection/Fixation, 15.38% had Total en bloc spondylectomy, and 10.26% had Corpectomy. A minority of patients had laminectomy and decompression. Among those with resection, 45.83% had a gross total resection, 29.17% had a subtotal resection, and 16.67% had a near total resection. Immunohistochemistry demonstrated positivity for actin (43.37%), desmin (31.33%), and Ki67 (25.30). At a follow-up of 19.3 months, 61.97% of patients were alive; 26.25% of 80 patients received no additional treatment, 23.75% received combination radiotherapy and chemotherapy, only chemotherapy was given to 20%, and radiotherapy was given to 17.5%. Few (2.5%) had further resection. For an average of 12.50 months, 42.31% had no symptoms, while others had residual (19.23%), other metastasis (15.38%), and pain (7.69%). On follow-up of 29 patients, most (68.97%) had resolved symptoms; 61.97% of the 71 patients followed were alive.

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“…When possible, surgery with negative surgical margins remained the primary treatment modality, independent of origin sites (Wang and Lucas, 2019). No clear survival benefit has been demonstrated with the use of adjuvant chemotherapy or radiotherapy (Zhang et al, 2022). However, due to possible differences in biological behavior, chemotherapy and/or radiotherapy are still recommended, especially for cases that failed to completely resect the lesion (Sun et al, 2022).…”

Section: Discussionmentioning

confidence: 99%

Case report: the dissociated response and clinical benefit of primary leiomyosarcoma of the bone treated with penpulimab plus lenvatinib after failed multi-line therapy

Wang,

Han,

Liu

et al. 2023

Front. Pharmacol.

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Leiomyosarcoma occurring in the bone as primary tumor localization is extremely scarce with limited cases described in the literature, accounting for less than 0.7% of all primary bone malignancies. Once distant metastasis occurs, patients have limited treatments and often a somber prognosis, which underscore the need for innovative and effective treatment approaches. The emerging evidence suggests that anti-angiogenic therapy could inhibit angiogenesis and normalize vascular permeability in the tumor microenvironment, which, in turn, would increase immune effector cell infiltration into tumors. Immunotherapy depends on the accumulation and activity of immune effector cells within the tumor microenvironment, and immune responses and vascular normalization seem to be reciprocally regulated. Immunotherapy combined with anti-angiogenic therapy has recently made great progress in the treatment of various types of tumors. However, the effectiveness of the combination treatment in metastatic leiomyosarcoma is undetermined. In this study, we presented a rare case of primary leiomyosarcoma of the bone located in the trochanteric region of the femur, accompanied by multiple distant metastases. After the failure of multi-line therapies including AI regiments as the adjuvant chemotherapy, anlotinib as the first-line therapy, GT regiment as the second-line therapy, and eribulin as the third-line therapy, the patient received combinational therapy with penpulimab plus lenvatinib. The best efficacy for this regimen was a partial response, with a progression-free survival of 8.4 months according to the iRECIST criteria. After a dissociated response was detected without severe toxicities, the patient received local radiotherapy and continued treatment on penpulimab plus lenvatinib and eventually achieved long-term survival benefits with a total of over 60 months of overall survival with good quality of life and ongoing treatment. As our previous retrospective study found that one-third of advanced STS patients could still achieve clinical benefits from rechallenge with multi-targeted tyrosine kinase inhibitors (TKIs), after the failure of previous TKI therapy, this case provided the potential clinical activity of immunotherapy combined with anti-angiogenic TKI rechallenge in metastatic leiomyosarcoma.

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Primary leiomyosarcoma of the spine: an analysis of imaging manifestations and clinicopathological findings

Cited by 5 publications

References 15 publications

A rare presentation of leiomyosarcoma metastasis to the cervical spine: A case report and a brief review

A rare presentation of leiomyosarcoma metastasis to the cervical spine: A case report and a brief review

Leiomyogenic Tumor of the Spine: A Systematic Review

Case report: the dissociated response and clinical benefit of primary leiomyosarcoma of the bone treated with penpulimab plus lenvatinib after failed multi-line therapy

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