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PubMed1996

[Uterine leiomyosarcomas: a surprising pathology. Review of the literature. Six case reports].

OBJECTIVE: The aim of this study was to determine the clinical features and laboratory tests which define a population with a high risk of leiomyosarcoma. METHOD: A retrospective analysis of 660 hysterectomies performed between 1989 and 1994 was conducted. There were 414 cases of leiomyoma and 6 leiomyosarcomas (1.4%). RESULTS: The indication for hysterectomy was complication of fibromyomatous uterus in all cases. Diagnosis of leiomyosarcoma was never suspected before surgery and the pathology examination. Complimentary radiotherapy was given in 2 patients among the 6 with leiomyosarcoma. Mean survival was 35 months. There were no cases of recurrence or metastasis. None of the laboratory tests performed provided clues to diagnosis. CONCLUSION: Physicians should be aware of possible misdiagnosis of a leiomyosarcoma when proposing medical treatment of uterine fibroma.

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PubMed1997

[Leiomyosarcoma of the small intestine].

Leiomyosarcomas of the small bowel are quite rare. These tumors are mostly located in the jejunum distal of the duodeno-jejunal flexure. The non-metastasizing leiomyosarcomas have a good prognosis, according to the literature. The adequate surgical treatment is the resection of the small bowel. Chemotherapy is discussed if there is evidence of metastatic disease. For the diagnosis of leiomyosarcomas tumor markers have no significance. We report the case of a 79 years old patient with a leiomyosarcoma of the jejunum who was admitted as an emergency with an acute abdomen and underlying chronic anaemia. This case demonstrates the often unspecific symptoms and characteristics of leiomyosarcomas.

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PubMed1999

Retroperitoneal leiomyosarcomas unassociated with the gastrointestinal tract: a clinicopathologic analysis of 17 cases.

Data are limited on leiomyosarcomas within the abdomen and retroperitoneum, particularly if one discounts those associated with the gastrointestinal (GI) tract. Recently, some authors proposed that certain tumors in this location are more appropriately termed extra-GI stromal tumors, given their histologic resemblance to GI stromal tumors as opposed to conventional soft tissue leiomyosarcomas. We evaluated the clinical and pathologic features of 17 cases of leiomyosarcoma (16 retroperitoneal, one intra-abdominal) and recorded the tumor size, predominant cell type, tumor cellularity, nuclear pleomorphism, extent of tumor cell necrosis, and number of mitotic figures per 10 high power fields (MFs/10HPFs). Cases were only included for study if the excised tumor did not arise from abdominal or pelvic viscera or major blood vessels, if adequate clinical follow-up was available, and if the tumor unequivocally resembled conventional soft tissue leiomyosarcoma, both by light microscopic and immunohistochemical examination. The cohort included 16 women and 1 man, and ages ranged from 44 to 72 years (median, 60 yr). Tumors ranged in size from 6.5 to 29.5 cm (median, 13.5 cm). Fifteen tumors were composed predominantly of spindled cells, one tumor was composed predominantly of epithelioid cells, and one tumor was composed of an admixture of spindled and epithelioid cells. Follow-up intervals ranged from 4 to 169 months (median, 47 mo). Fifteen (88%) of seventeen patients developed an adverse outcome, defined as the development of metastatic disease or death due to tumor. Patients whose tumors had greater than 10 MFs/10HPFs had significantly shorter intervals to either metastasis or death than did those whose tumors had 10 or fewer MFs/10HPFs (8.4 mo vs. 42 mo; P = .003). No other features correlated with time to adverse outcome. In conclusion, the majority of patients with leiomyosarcomas located within the abdomen or retroperitoneum progress to metastatic disease or die from their tumor. The only feature that is significantly associated with a shorter interval to either metastasis or death is more than 10 MFs/10HPFs.

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PubMed2001

A huge abdominal mass mimicking ovarian cancer: p53-negative but aneuploid myxoid leiomyosarcoma of the uterus.

OBJECTIVE: Less than 20 myxoid leiomyosarcoma cases were reported in literature. Since, these tumors are very rare and may exhibit highly malignant behavior despite their low mitotic index, clinical course and optimum type of therapy of myxoid variant of leiomyosarcoma were not well understood. The goal of this report is to contribute the better understanding of this rare type of tumor. METHODS: A 39-year-old woman presented with a huge abdominal cystic mass. Laparotomy was performed and frozen section diagnosis was low-grade uterine leiomyosarcoma. TAH-BSO, omentectomy, pelvic lymph node and peritoneal samplings were carried out. No chemotherapy was performed after surgical therapy. RESULTS: Final histopathological diagnosis was uterine myxoid leiomyosarcoma. The tumor was p53-negative and had aneuploid DNA content. The patient tolerated well the operation and she is alive and free of disease after 24 months of primary surgical treatment. CONCLUSION: Uterine myxoid leiomyosarcoma may present a huge abdominal cystic mass and can be treated successfully with surgery alone.

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PubMed1999

Leiomyosarcoma of the parotid gland: a case report and review of the literature.

BACKGROUND: A primary leiomyosarcoma of the parotid gland has been reported only three times in the English literature. This type of tumor represents an extremely rare group of salivary gland neoplasm. METHODS: A 44 year old man was initially seen with a 5 cm right parotid mass which was resected with a total parotidectomy, preserving the facial nerve. The patient had no palpable cervical lymph nodes. RESULTS: The majority of the specimen was made up of a relatively well demarcated tumor 5 cm in diameter. The tumor was noted to contain moderate to poorly differentiated primary leiomyosarcoma of the parotid. CONCLUSIONS: A primary leiomyosarcoma of the parotid gland is an extremely rare existing entity. A review of the literature reveals that primary leiomyosarcoma and other sarcomas of the major salivary glands may share similar histogenesis and biologic behavior with their soft tissue counterparts.

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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.

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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.

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PubMed1995

Epstein-Barr virus-associated leiomyosarcomas in liver transplantation recipients. Origin from either donor or recipient tissue.

BACKGROUND: Leiomyosarcoma, a mesenchymal malignancy with smooth muscle differentiation, is extremely rare in children. Immunosuppression, due to either antirejection medication in organ transplantation recipients or human immunodeficiency virus infection (HIV), appears to constitute a predisposition. METHODS: Two cases of leiomyosarcoma in pediatric liver transplantation recipients were investigated and compared clinically with respect to site of origin and course of the disease and pathologically by routine histology and electron microscopy, by forensic DNA methodology for origin from donor or recipient tissue, and by EBER-1 in situ hybridization for evidence of latent Epstein-Barr virus (EBV) infection. RESULTS: A 9-year-old male developed a high grade, poorly differentiated leiomyosarcoma in his allografted liver 2 years after transplantation, and despite antineoplastic chemotherapy, he died of metastatic disease. The genotype of his tumor indicated an origin from allografted tissue. A 12-year-old female had a low grade retroperitoneal leiomyosarcoma involving the superior mesenteric vein. After resection, she remained disease free without chemotherapy. The genotype of her tumor indicated an origin from native tissue. In both tumors, latent EBV infection was documented. CONCLUSIONS: Neoplastic smooth muscle proliferation in immunosuppressed liver transplantation recipients is analogous to the more common posttransplantation lymphoproliferative disorder in involving transformation of either engrafted donor tissue or recipient tissue elsewhere in the body, in displaying a wide spectrum of histologic differentiation, grade and clinical behavior, and in exhibiting evidence of latent EBV infection.

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PubMed2000

Enhancement of antitumor effect of bleomycin by low-voltage in vivo electroporation: a study of human uterine leiomyosarcomas in nude mice.

Uterine leiomyosarcoma is an extremely malignant neoplasm with high rates of distant metastasis, and systemic chemotherapy is not particularly effective. Thus, the introduction of more active anticancer agents, or of a new drug delivery system, is urgently needed. Recently, electrochemotherapy has been introduced as a way of enhancing the cytotoxic effects of chemotherapeutic agents. This involves administering the drug in combination with electric pulses (which permeabilize tumor cell membranes and allow the drug to enter the cells). In particular, bleomycin (BLM) cannot cross the plasma membrane efficiently, but its cytotoxicity can be enhanced by electropermeabilization. The aim of the present study was to investigate the effect of low-voltage electroporation (EP) in combination with local BLM injection on the growth of uterine leiomyosarcoma in nude mice. Human uterine leiomyosarcoma cells (SK-LMS-1) were implanted subcutaneously into nude mice. Tumor growth in mice treated with EP (100 V/cm) plus BLM was compared with that in mice receiving BLM alone, EP alone, or no treatment (controls). Tissue BLM concentrations and histological analysis (including mitotic counts) were evaluated in tumor tissues. There was a significant reduction in tumor growth in mice that received EP with BLM. One hour after the treatment, the local BLM concentration was 10 times higher in the tumors that received EP with BLM than in those receiving only BLM. Moreover, the mitotic count was lower in the tumors that received EP plus BLM than in the controls. These results demonstrate the possible therapeutic value of low-voltage EP with BLM in human uterine leiomyosarcoma.

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PubMed2003

Leiomyosarcoma of the breast: a difficult diagnosis on fine-needle aspiration biopsy.

Leiomyosarcoma of the breast is rarely encountered in fine-needle aspiration (FNA) cytologic material. We report a case of primary leiomyosarcoma of the breast in a 52-yr-old female. Aspiration cytology showed large, dissociated round to spindle cells with abundant vacuolated cytoplasm, pleomorphic nuclei, prominent nucleoli, and occasional intranuclear cytoplasmic invaginations. Mitotic figures, osteoclast-like giant cells, and stromal fragments were identified. A diagnosis of malignant neoplasm representing either a sarcoma, a poorly differentiated carcinoma, or a metaplastic carcinoma was made. The patient underwent a wide excision of the lesion after negative work-up. Histologic examination and immunohistochemical studies established the diagnosis of leiomyosarcoma. This case is presented here because we feel that, although FNA cytology with eventual ancillary studies is a valuable diagnostic tool to evaluate any breast mass, malignant spindle cell neoplasms of the breast still represent a diagnostic challenge for the cytopathologist. Recognition of all cytologic features of leiomyosarcoma may help to formulate a correct diagnosis.

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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.

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PubMed1987

Primary leiomyosarcoma of the inferior vena cava: a case report and review of the literature.

Sixty-six cases of leiomyosarcoma of the inferior vena cava (IVC) have been reported. We present another case and review the characteristics and treatment of this rare tumor. IVC leiomyosarcomas spread by extension into adjacent tissue planes. Direct extension into the heart is known, but not into the kidneys, adrenals, or bowel. IVC leiomyosarcomas arising below the renal vein cause pain in the right-lower quadrant, back, and flank, and varying degrees of lower extremity edema; those arising in the middle caval segment cause right-upper-quadrant pain and sometimes renovascular hypertension; those arising above the hepatic vein cause varying manifestations of the Budd-Chiari syndrome. A reasonable approach to early diagnosis involves oral and intravenous contrast studies followed by computed tomography, angiography, and vena cavography. Aggressive surgical treatment is optimal in light of the tumor's slow growth pattern and relatively low malignant potential. (Metastases have been reported in fewer than 50% of cases). Tumors involving the lower IVC are most amenable to surgery; extensive collateral venous drainage of the left kidney preserves renal function during resection of middle caval tumors. Upper caval leiomyosarcomas are the least amenable to complete removal. Postoperative survival of all patients has ranged from a few weeks to eight years.

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PubMed2006

A second leiomyosarcoma in the urinary bladder of a child with a history of retinoblastoma 12 years following partial cystectomy.

This case describes a twin with bilateral retinoblastoma who developed leiomyosarcoma of the bladder at age 17 and again at 39. At 17-years of age she was diagnosed with a leiomyosarcoma of the bladder after presenting with recurrent urinary tract infections, hematuria, and dysuria. She was treated with partial cystectomy. After a 12-year disease-free interval, she was diagnosed with a second leiomyosarcoma of the bladder. This case supports the relationship between the genetic form of retinoblastoma and leiomyosarcoma and illustrates the necessity for extensive follow-up and well-defined treatment of secondary neoplasms.

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PubMed1996

Uterine leiomyosarcoma: analysis of treatment failures and survival.

The objective of this retrospective multicenter study was to assess the rates, times, and sites of recurrences of 126 patients with uterine leiomyosarcomas. Surgery was the initial therapy for all patients. Median follow-up of survivors was 50 months (range, 3-168 months). Of the 90 patients with stage I-II disease, 26 received postoperative irradiation and/or chemotherapy. Thirty-five (38.9%) patients developed recurrent disease after a median time of 16 months (range, 2-102 months). Recurrence was pelvic in 5 (14.3%) patients, distant in 23 (65.7%), and pelvic plus distant in 7 (20.0%). The overall recurrence rate was similar in patients who received adjuvant treatment and in those who did not. None of the 15 patients who underwent pelvic irradiation developed local recurrences, but 5 of them failed in distant sites. Of the 16 patients with stage III leiomyosarcomas, 2 died of intercurrent disease within 1 month from surgery and 11 received postoperative irradiation and/or chemotherapy. Thirteen patients developed recurrent tumor after a median time of 8 months (range, 1-21 months). Recurrence was pelvic in 3, distant in 4, and pelvic plus distant in 6 patients. Of the 20 patients with stage IV leiomyosarcomas, after surgery 6 were clinically free of disease (group A) and 14 had clinically evaluable residual disease (group B). With regard to group A, 3 patients received postoperative irradiation and/or chemotherapy. Five patients developed recurrent disease after a median time of 11 months (range, 8-16 months). Recurrence was distant in 3 patients and pelvic plus distant in 2. With regard to group B, 11 patients underwent postoperative chemotherapy. Eleven patients died after a median time of 6 months (range, 1-15 months), and 3 are still alive with clinical evidence of disease after 4, 5, and 8 months, respectively, from surgery. Cox model showed that stage (P = 0.0001), mitotic count (P = 0.0002), and age (P = 0.0048) were independent prognostic variables for disease-free survival. In conclusion, uterine leiomyosarcomas have an aggressive clinical behavior, with a propensity to recur both locally and moreover at distant sites. Tumor stage is the strongest prognostic variable. Only patients with early-stage disease have a chance of surviving, whereas the treatment of patients with advanced or recurrent disease is palliative.

Adult↗

PubMed1999

Leiomyosarcoma of the uterus: a clinicopathologic multicenter study of 71 cases.

OBJECTIVE: The aim of this study was to evaluate the behavior of uterine leiomyosarcomas in relation to their clinical and pathologic features and to identify possible prognostic factors. METHODS: Seventy-one patients with histologically proven uterine leiomyosarcoma were included in the analysis. Leiomyosarcomas were defined as uterine smooth-muscle tumors with five or more mitoses per 10 high-power fields and cytologic atypia. Cox proportional hazards regression model was used to identify independent prognostic factors. RESULTS: The median follow-up time was 108 months; 5-year overall survival rate was 65%. Evaluating the correlation between several clinicopathologic parameters, tumors with vascular space involvement had a statistically significantly higher stage than tumors without vasular space involvement (P = 0.015). In a univariate Cox model early tumor stage (P < 0.0001), age <50 years (P < 0.0001), low mitotic count (P = 0.05), and the absence of vascular space involvement (P < 0.0005) were associated with good prognosis. In a multivariate analysis age (P = 0.002), tumor stage (P = 0.004), vascular space involvement (P = 0.003), and mitotic count in stage I tumors (P = 0.002) were found to be independent parameters for good prognosis in patients with uterine leiomyosarcoma. CONCLUSION: Early tumor stage, age <50 years, and absence of vascular space involvement were independently associated with good prognosis. Mitotic count was detected to be a strong prognostic parameter in early tumor stage, but failed to act as an independent prognostic parameter in patients with tumor stage II-IV disease.

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PubMed1999

Frequent occurrence of loss of heterozygosity among tumor suppressor genes in uterine leiomyosarcoma.

OBJECTIVE: Leiomyosarcoma of the uterus is a rare smooth muscle tumor; it is extremely malignant and the rates of local recurrence and metastasis are high. Since tumor suppressor genes are commonly altered in malignant tumors, it is possible that mutations in such genes are involved in the development of uterine leiomyosarcoma. METHODS: Fifty-five patients (37-70 years of age) diagnosed as having smooth muscle tumors of the uterus were selected. DNA was extracted from four or five 8-microm-thick consecutive tissue sections of each smooth muscle tumor from the paraffin-embedded blocks. Loss of heterozygosity (LOH) was investigated at nine loci within or close to tumor suppressor genes (TP53, RB1, DCC, NM23, WT1, D14S267, P16, DPC4, PTCH). RESULTS: Nineteen of twenty leiomyosarcomas revealed at least one instance of LOH among eight of the nine markers tested (one locus showed no LOH at all). In fact, 11 of the 20 cases exhibited two or more instances of LOH and, of the remaining 9 cases, 4 showed a point mutation of p53 in addition to an alteration in one of the 9 markers, while one exhibited a p53 mutation only. CONCLUSION: An accumulation of genetic alterations among tumor suppressor genes may play a key role in the tumorigenesis and progression of uterine leiomyosarcoma.

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PubMed2001

Epithelioid leiomyosarcoma of the uterine cervix.

BACKGROUND: Leiomyosarcomas with an epithelioid appearance arising in the uterine cervix are extremely rare neoplasms. We present a case of triple uterine cancer containing cervical epithelioid leiomyosarcoma. CASE: A 72-year-old Japanese woman, gravida 1, para 1, 55 years in menopause, visited her local doctor due to atypical bleeding and the endometrial smear was suspect for malignancy. She was referred to our hospital. Endometrial curettage revealed endometrioid adenocarcinoma, G1, with a fragment of sarcomatous tumor. A total hysterectomy with bilateral salpingo-oophorectomy was performed. The histological findings revealed triple uterine cancer consisting of endometrial adenocarcinoma, cervical epithelioid leiomyosarcoma, and cervical squamous cell carcinoma. CONCLUSION: This extremely rare case of triple uterine cancer containing cervical epithelioid leiomyosarcoma is the first report, to the best of our knowledge.

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PubMed2002

Treatment of uterine leiomyosarcoma in a xenograft nude mouse model using high-intensity focused ultrasound: a potential treatment modality for recurrent pelvic disease.

OBJECTIVE: The objective was to test the efficacy of high-intensity focused ultrasound (HIFU) for treatment of uterine leiomyosarcoma in a Xenograft nude mouse model. METHODS: A total of 65 athymic nude mice were inoculated subcutaneously with 5 to 7 x 10(6) ELT-5B cells, a uterine leiomyosarcoma cell line derived from the Eker rat. Thirty animals showed tumor growth. The tumor volume was measured transcutaneously once a week. Animals were randomly assigned to three groups: HIFU treatment (n = 17), sham treatment (n = 7), and control (n = 6). A HIFU device, operating at a frequency of 2.0 MHz and an intensity of 2000 W/cm(2), was used for treatment. RESULTS: Within 3 weeks of a single HIFU treatment, 100% reduction in tumor volume was observed in all animals, except one. A second HIFU treatment was applied to that animal, resulting in 100% reduction in tumor volume. The tumors in the sham-treated animals continued to grow at a similar rate to that of the control group to approximately 500% of the tumor volume at the time of treatment. All animals were monitored for a maximum of 3 months. No metastasis was observed in the HIFU-treated animals. Histological examination confirmed a complete tumor disappearance after HIFU treatment. CONCLUSION: We have shown that HIFU can effectively treat uterine leiomyosarcoma tumors inoculated in Xenograft nude mice, demonstrating HIFU's potential use for treatment of recurrent uterine leiomyosarcoma.

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