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PubMed1992

Superficial perineal leiomyosarcoma in an adolescent female and a review of the literature including vulvar leiomyosarcomas.

Superficial perineal leiomyosarcomas are rare, with only three previously reported examples. We encountered a superficial (deep subcutaneous) perineal leiomyosarcoma in a 17-year-old female. At follow-up two years after a wide excision, there were no signs of recurrence. The tumour was well differentiated and showed immunoreactivity for alpha-smooth muscle actin and desmin. A review of the literature on superficial leiomyosarcomas indicates that superficial perineal leiomyosarcomas may be more aggressive than superficial leiomyosarcomas in general. As the presented tumour occurred in a female, it was compared with vulvar leiomyosarcomas.

Adolescent↗

PubMed1992

Recurrent cutaneous leiomyosarcoma.

BACKGROUND: Soft tissue sarcomas comprise approximately 0.7% of all malignant neoplasms. Superficial leiomyosarcoma, a rare malignant lesion, constitutes 4.0-6.5% of all soft tissue sarcomas, an overall incidence of approximately 0.04% among all cancers. Currently, less than 125 cumulative cases of cutaneous and subcutaneous leiomyosarcoma have been reported in the English literature. METHODS: The authors report the case of a 70-year-old Japanese man with recurrent cutaneous leiomyosarcoma who was treated by wide local excision. A comprehensive literature survey is also presented. RESULTS: The patient is free from recurrence 1 year after wide local excision of a second recurrence of cutaneous leiomyosarcoma. Moreover, superficial leiomyosarcoma can be subdivided into cutaneous leiomyosarcoma and subcutaneous leiomyosarcoma, based on histopathologic and prognostic differences. CONCLUSIONS: The treatment of choice of superficial leiomyosarcoma is wide local excision. Cutaneous leiomyosarcoma is associated with local recurrence only, although subcutaneous leiomyosarcoma undergoes metastatic spread in 30-60% of cases, with a 30-40% mortality rate.

Aged↗

PubMed2026

Management of Soft Tissue and Visceral Leiomyosarcomas.

IMPORTANCE: Leiomyosarcoma is a rare and heterogeneous malignant mesenchymal neoplasm associated with substantial morbidity and mortality. Given recent advances in biologic understanding and the complexity of leiomyosarcoma, a consensus-driven approach is needed to harmonize management and address remaining clinical and research gaps. OBJECTIVE: To provide an evidence-based synthesis of current diagnostic and therapeutic approaches for leiomyosarcoma by an international panel of physicians, researchers, and patient advocates, focusing on site-specific management, systemic therapy strategies, and key areas of clinical uncertainty, while identifying unmet needs and research priorities. EVIDENCE REVIEW: This review is based on a comprehensive evaluation of the literature, including clinical trials, observational studies, and international consensus guidelines. Sources were identified through MEDLINE (via PubMed) and Embase database searches and reference screening, then supplemented by multidisciplinary expert consensus. Emphasis was placed on studies informing diagnosis, surgical management, radiotherapy, and systemic therapy in leiomyosarcoma. FINDINGS: The rarity and heterogeneity of leiomyosarcoma poses substantial challenges in its management. In localized disease, complete surgical resection remains the cornerstone of treatment, with evidence supporting the use of site-specific perioperative treatment strategies. Prospective data supporting neoadjuvant or adjuvant chemotherapy are lacking, and the role of radiotherapy differs across anatomic disease sites and institutions. In advanced disease, multiple systemic therapies demonstrate activity, including anthracycline-based and gemcitabine-based combinations, trabectedin, and tyrosine kinase inhibitors, although optimal sequencing after first-line therapy remains undefined. Emerging data suggest potential benefit from treatment continuation strategies and selected use of local therapies in oligometastatic settings. Molecular heterogeneity is increasingly recognized but has not yet translated into routine clinical implementation, and integration of molecular profiling into diagnostic pathways for predictive and therapeutic insights remains an unmet need. CONCLUSIONS AND RELEVANCE: This international consensus addresses the diagnosis and management of leiomyosarcoma. Management requires a multidisciplinary, site-specific approach informed by limited but evolving evidence. Key uncertainties persist, particularly regarding perioperative therapy, optimal sequencing and combination of systemic treatments, and integration of molecular data. Continued international collaboration and leiomyosarcoma-specific clinical trials are needed to refine treatment strategies and improve patient outcomes.

Journal Article↗

PubMed1979

Paratesticular rhabdomyosarcomas and leiomyosarcomas: a clinicopathological review.

One case of embryonal paratesticular sarcoma and 2 cases of leiomyosarcomas are reported. The 13-year-old boy with embryonal sarcoma is well after an orchiectomy and high ligation of the spermatic cord followed by radiotherapy and chemotherapy. A 66-year-old man has been doing well after orchiectomy and hemiscrotectomy for a paratesticular leiomyosarcoma. The third patient had a highly pleomorphic leiomyosarcoma and died 2 months postoperatively. The clinical and histological diagnosis of paratesticular rhabdomyosarcomas and leiomyosarcomas is reviewed and the various therapeutic approaches to these neoplasms are discussed. Retroperitoneal lymph node dissection, radiotherapy and chemotherapy are important adjuncts to orchiectomy in the management of rhabdomyosarcomas. On the contrary, retroperitoneal lymph node dissection and radiotherapy are not indicated in leiomyosarcomas, since these neoplasms tend to metastasize by the hematogenous route and are radioresistant. At the present time we are unable to evaluate chemotherapy in the management of paratesticular leiomyosarcomas.

Aged↗

PubMed1991

Intermediate and fine cytofilaments in cutaneous and subcutaneous leiomyosarcomas.

The expression of fine and intermediate cytofilaments in 10 cutaneous and seven subcutaneous leiomyosarcomas was studied immunohistochemically. All the tumors contained tumor cells which showed a positive immunoreactivity for desmin in formaldehyde-fixed and paraffin-embedded sections, but none of the seven anti-desmin antibodies used alone produced a distinct positive staining in all the tumors. A lack of correspondence in terms of immunoreactivity between tumor cells and the supposed muscle of origin was observed, especially in the subcutaneous leiomyosarcomas. In all cases, antibodies to muscle-specific and smooth muscle-specific actin were found to produce a positive staining in both the tumors and the supposed muscle of origin. Vimentin was detected in 8/10 cutaneous and 4/7 subcutaneous leiomyosarcomas, while the supposed muscle of origin was positive in 3/10 and 7/7 cases, respectively. Four of the cutaneous and three of the subcutaneous leiomyosarcomas contained tumor cells which stained positively for cytokeratins, while the supposed muscle of origin showed no positivity. It thus appears that a phenotypic shift in terms of vimentin and cytokeratin expression occurs in the tumor cells of cutaneous and subcutaneous leiomyosarcomas compared with the supposed muscle of origin. It is recommended that more than one monoclonal anti-desmin antibody is used to characterize these tumor entities. It is also concluded that the immunoreactivity for muscle-specific actins in superficial leiomyosarcomas is more constant, although less specific, than that of desmin and that the demonstration of the simultaneous expression of muscle-specific actins and desmin is helpful.

Actin Cytoskeleton↗

PubMed1992

Case report 747: Leiomyosarcoma of great saphenous vein.

Vena caval leiomyosarcomas are rare tumors; noncaval leiomyosarcomas are even less common and are rarely diagnosed preoperatively. We present a case of leiomyosarcoma of the great saphenous vein that was diagnosed by MRI based on its appearance and location. The presence of an intraluminal tumor in a vein on MRI should suggest the diagnosis of leiomyosarcoma of the vein, allowing definitive operative excision. With widespread use of MRI for indeterminate soft-tissue masses, we anticipate presurgical diagnosis of venous leiomyosarcoma will be made with increasing certainty and frequency.

Female↗

PubMed1975

Leiomyoma and leiomyosarcoma of the orbit.

Comparison of the clinical and pathologic features of two orbital leiomyomas, two leiomysarcomas, and one embryonal rhabdomyosarcoma showed the leiomyomas occurred in young individuals and the leiomyosarcomas in older patients. The histopathologic diagnosis rested on the intense cytoplasmic eosinophilia and nostriated longitudinal cytoplasmic filaments demonstrated by means of the trichrome stain. The leiomyosarcomas disseminated 15 months and seven years after their orbital presentations. The treatment of both leiomyoma and leiomyosarcoma is surgical. Leiomyomas are encapsulated growths that may have small satellite nodules projecting from the main tumor mass; thus, a margin of normal tissue should also be excised, lest a small lobulation be left behind to serve as the seed for a late recurrence. Once the diagnosis of leiomyosarcoma has been made, and no evidence of metastasis has been found after a thorough systemic evaluation, the orbit should be exenterated, because the tumor is unencapsulated and liable to widespread dissemination. Rhabdomyosarcoma has a much more fulminant course than leiomyosarcoma, and especially more so than that of leiomyoma of childhood. The histopathologic diagnosis of a malignant smooth muscle tumor in a child should always be questioned, since embryonal rhabdomyosarcoma is a much more likely diagnosis.

Adolescent↗

PubMedDate not supplied

Leiomyosarcoma: a rare pedal finding.

Leiomyosarcoma of the skin and subcutaneous tissue is a rare soft tissue neoplasm comprised of spindle-shaped smooth muscle cells. The statistical incidence of superficial leiomyosarcomas varies from 2.3% to 5.3% of malignant soft tissue tumors and from 4.0% to 6.5% of soft tissue sarcomas. Its occurrence in the foot is even more unusual. In a literature review extending from 1936-present, the authors have been able to locate only 12 known cases of leiomyosarcoma involving the foot and ankle. A presentation of the incidence, demographics of distribution, clinical characteristics, histopathologic features, prognosis, and treatment of leiomyosarcoma will be presented along with a unique case history involving a recalcitrant heel ulcer complicated by leiomyosarcoma.

Aged↗

PubMed1978

[Primary leiomyosarcoma of the liver (author's transl)].

The occurrence of leiomyosarcoma was demonstrated in a tissue cylinder obtained by liver biopsy in a 68-year-old woman with unclear hepatomegaly. The patient died 8 months after she had experienced first signs of illness. Autopsy revealed a primary leiomyosarcoma of the liver with metastases in the lungs, bilaterally in the pleura, in the kidneys, and in the periportal and spleno-pancreatic lymph nodes. The occurrence of a primary tumour in the urogenital system or in the gastrointestinal-tract could be excluded. The diagnosis of leiomyosarcoma was based on the microscopical demonstration of smooth muscle fibres with enlarged red-like and partially atypical cell nuclei and atypical mitoses. Furthermore, we observed extensive necroses and haemorrhagia in the tumour tissue. Preexisting tumours of the liver as, e.g., teratoma or hepatoblastoma were not found. No cirrhosis of the liver could be detected. Obviously, the leiomyosarcoma had its origin in the smooth muscle fibres of the liver vascular system.--According to the literature primary leiomyosarcomas in the liver are only rarely found.

Aged↗

PubMed1979

Primary leiomyosarcoma of the bone and its comparison with fibrosarcoma.

Two cases of primary leiomyosarcoma of the bone are recorded, one in the distal fibula of a 66-year-old man, the other in the proximal tibia of a 61-year-old woman. The cytological, histological, and ultrastructural features of leiomyosarcoma of bone are described and compared with those of fibrosarcoma. These features are sufficiently characteristic to enable a confident diagnostic distinction between leiomyosarcoma and fibrosarcoma. Nevertheless, certain basic similarities exist between these two tumors, manifested at the ultrastructural level by the presence of myofilaments in fibrosarcoma; it would seem that the observed differences relate to the degree of development of the myofilamentous structures. It is postulated that primary leiomyosarcoma of the bone need not necessarily always arise from the media of blood vessels; it might also conceivably develop through advanced myogenic metaplasia of a sarcoma originating from fibroblastic tissue.

Aged↗

PubMed1979

Vascular leiomyosarcoma: the malignant counterpart of vascular leiomyoma.

The clinical and pathological findings of six cases of leiomyosarcoma arising from blood vessels of different caliber are described. The term vascular leiomyosarcoma, having both a topographic and morphologic significance, is proposed for these tumors. The histologic pattern is characterized by a proliferation of atypical smooth muscle cells with a large number of intermingled blood vessels. Mitoses were counted per 10 high power field (hpf) and tumors were divided in three groups I, 10 to 20 mitoses, group II, 20 to 35 mitoses, and group III, more than 35 mitoses per 10 hpf. The mitotic index seems to be the most important pathological feature on which a prognostic evaluation for vascular leiomyosarcoma can be based. Tumors in group I had neither local recurrences nor metastases; the one tumor in group II had one local recurrence, but the patient is free of disease 6 years after surgical treatment; the three tumors in group III developed distant metastases and constitutional symptoms. Vascular leiomyoma, bizarre leiomyoma, and hemangiopericytoma are included in the differential diagnosis of vascular leimyosarcoma. The possibility that vascular leiomyosarcoma arising from small vessels represents the malignant counterpart of vascular leiomyoma is proposed.

Adult↗

PubMed1992

Establishment and characterization of human uterine leiomyosarcoma heterotransplanted into nude mice.

Human uterine leiomyosarcoma is a rare gynecological malignancy with a generally poor prognosis. We have established a human uterine leiomyosarcoma tumor line in nude mice, designated UTS-1, and describe the characteristics of this tumor. The UTS-1 tumor doubled in 12.1 days and retained the histological characteristics of leiomyosarcoma, even after 14 serial generations. Ultrastructurally, the tumor is characterized by nuclear pleomorphism typical of smooth muscle, intracytoplasmic filaments with dense bodies, a relative paucity of micropinocytotic vesicles, and an incomplete external lamina. Immunohistochemically, the UTS-1 cells reacted with antibodies against vimentin, desmin, smooth-muscle actin and myosin, but not with antibodies against keratin, CEA and S-100 protein. Serum levels of AFP, CA125, CEA and SCC ranged within normal limits in tumor-bearing mice. The serum level of immunosuppressive acidic protein correlated well with an activity of the tumor. Estrogen and progesterone receptors were not detected in the tumor. Chromosomal analysis showed a human karyotype with some marker chromosomes and a modal number of 85 chromosomes. The UTS-1 tumor should prove a useful model to explore the biological characteristics and treatment of human uterine leiomyosarcoma.

Animals↗

PubMed1992

Computed tomography appearance of bone metastases of leiomyosarcoma.

Bone lesions in leiomyosarcoma are uncommon. When encountered, they are usually a late manifestation of disease seen in the setting of widespread systemic metastases. We present five cases of metastatic leiomyosarcoma, including one in which a bone lesion predated detection of a primary gastric leiomyosarcoma and one in which bone metastases signaled disease recurrence. Computed tomography in 14 of 16 osseous leiomyosarcomas demonstrated a lytic, non-expansile pattern.

Adult↗

PubMed1992

Leiomyosarcoma of the uterus: ultrasonography and serum lactate dehydrogenase level.

Between January 1, 1979, and September 30, 1990, a total of 1,886 patients in the National Defense Medical College Hospital, a self-referred population, had a hysterectomy because of signs and symptoms presumably resulting from uterine myomas. After hysterectomy with presumed benign disease, a histologic diagnosis of leiomyosarcoma was made in 7 patients (0.37%). Preoperative diagnosis of leiomyosarcoma was not made in any of the 7 patients. However, serum lactate dehydrogenase levels were abnormally elevated in 3 of them, and degenerative changes were found within the tumor by ultrasonography in 5 of them. Furthermore, increased lactate dehydrogenase levels and degenerative changes within the tumor were found in 3 of the patients whose tumors had 10 or more mitoses per 10 high-power fields. The prognosis for the leiomyosarcomas with increased mitotic rates is very poor. Therefore, a degenerative change within the uterine mass and an increased lactate dehydrogenase level, when present, should suggest the diagnosis of leiomyosarcoma.

Adult↗

PubMed1978

Angiography of abdominal leiomyosarcoma.

The spectrum of angiographic findings of abdominal leiomyosarcoma in 33 cases is presented, and the value of angiography in the diagnosis and management of such patients is discussed. Angiographic features of abdominal leiomyosarcomas are different depending upon the site of origin. Small bowel lesions are hypervascular, and those in the stomach and colon moderately vascular. Well circumscribed masses with enlarged feeding arteries and draining veins form the predominant presentation of small bowel leiomyosarcoma. In the retroperitoneum these neoplasms are usually hypovascular to moderately vascular, and displacement of major vessels, particularly the inferior vena cava, is the most common angiographic finding. Bladder leiomyosarcoma can be either moderately vascular or hypervascular. Vascularity of metastases is usually similar to that of the primary lesion.

Abdominal Neoplasms↗

PubMed1975

Preoperative cytological diagnosis of gastric leiomyosarcoma. A report of three cases.

Gastric leiomyosarcoma has rarely been diagnosed prior to operation. Three cases are presented in which a cytological diagnosis compatible with leiomyosarcoma could be made preoperatively by exfoliative cytology using a brushing technique under direct vision. Data which favor the diagnosis of leiomyosarcoma are: (1) spindle-shaped cells with malignant looking nuclei; (2) cells with extremely long, fiber-like cytoplasms and hyperchromatic nuclei; (3) large, anaplastic malignant cells. Smears from 3 patients showed a mixture of these different cell types. This cytological presentation is probably unique and diagnostic for leiomyosarcoma.

Cell Nucleus↗

PubMed1992

Leiomyosarcoma in dogs: 44 cases (1983-1988).

During a 5-year period, leiomyosarcoma was diagnosed in 57 dogs. Forty-four dogs were included in the study on the basis of completeness of medical records. All dogs underwent exploratory laparotomy, and dogs were allotted to 4 groups according to primary site of tumor: spleen (16 dogs, median age 10.3 years), stomach/small intestine (13 dogs, median age 10.3 years), cecum (10 dogs, median age 11.8 years), and liver (5 dogs, median age 9 years). All dogs with leiomyosarcoma of the liver had visible metastasis and were euthanatized at surgery. In the other 3 groups, 79% of the dogs had no gross evidence of metastasis at surgery, and 64% survived greater than 2 weeks. Median survival in these 3 groups was 10 months (range, 1 month to 7 years); 48% died of metastasis, 32% died of unrelated causes, and 16% died of unknown causes. The prognosis in dogs with leiomyosarcoma of the spleen, stomach, small intestine, and especially the cecum is good to excellent if surgery is performed. In dogs with leiomyosarcoma of the liver, the prognosis is poor.

Animals↗

PubMed1992

Primary venous leiomyosarcoma: a rare but lethal disease.

Primary venous leiomyosarcoma is rare, only 197 patients have been reported. To examine progress in diagnosis, treatment, and clinical outcome, we retrospectively reviewed data of 13 patients, 12 women and one man (mean age, 55 years; range, 19 to 75 years), who in the last 35 years underwent surgical treatment for primary venous leiomyosarcoma at our institution. The tumor arose from the inferior vena cava in eight, iliac vein in two, ovarian vein in one, and greater saphenous vein in two patients. Primary venous leiomyosarcoma was detected by physical examination in nine patients, symptoms were present in six. The 13 patients underwent 16 surgical procedures to resect primary (12), recurrent (2), or metastatic (2) tumors. Local excision alone was performed in seven, and local excision with reconstruction of the inferior vena cava or iliac vein was performed in six patients. The tumor was greater than 10 cm in eight patients. The perioperative mortality rate was 15% (2 of 13). Median survival of the 11 early survivors was 3.5 years (range, 6 months to 17 years). Currently five patients are alive (four of them free of known tumor), with a median survival of 3 years (6 months to 17 years) after surgery. Of the eight deceased patients, primary venous leiomyosarcoma recurred after resection in six. Tumor recurrence was not affected by tumor grade, size, or adjuvant treatment. Although early detection with modern imaging techniques could potentially be of benefit, wide local excision with selective venous reconstruction affords the only hope for prolonged survival.

Adult↗