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PubMed1992

Leiomyosarcoma of the orbit: diagnosis of its recurrence by fine-needle aspiration cytology.

An 8 1/2-yr-old boy with proptosis was diagnosed to have low-grade leiomyosarcoma of the orbit following lateral orbitotomy and histology of an incompletely excised intraconal mass. He received chemotherapy but had recurrence of proptosis at the age of 12 yr. Ultrasonography revealed a solid mass and its fine-needle aspiration cytology features were consistent with recurrence of leiomyosarcoma. A year later, the cytodiagnosis was confirmed by histology of the excised tumor from a second lateral orbitotomy. Masson's trichrome stain confirmed the smooth muscle nature of the neoplasm and ultrastructural features were in favor of leiomyosarcoma. The patient received intermittent chemotherapy, and is clinically free from disease at the age of 17 yr.

Antineoplastic Combined Chemotherapy Protocols↗

PubMed2001

Characterization of chromosome aberrations associated with soft-tissue leiomyosarcomas by twenty-four-color karyotyping and comparative genomic hybridization analysis.

Data on the chromosome aberrations associated with leiomyosarcomas of soft tissues are limited, complex, and incomplete. The aim of this study was to characterize genetic aberrations associated with this tumor group, to identify consistent regions of involvement and to determine correlations with clinical outcome. Chromosomes were prepared from 10 primary soft-tissue leiomyosarcoma samples, and preparations from four of them, plus the cell line SK-LMS-1, were suitable for analysis using 24-color karyotyping by multifluor fluorescence in situ hybridization. This method allowed rearranged chromosomes to be characterized, which would not have been possible by banding analysis alone. The remaining six chromosome preparations were analyzed using standard Giemsa banding. The chromosome imbalances associated with all the samples were determined by comparative genomic hybridization analysis. Taken together, the results show both intra- and intertumor heterogeneity and considerable complexity. Although no highly consistent rearrangements were found, some regions of the genome frequently were involved, including 1q21, 5p14-pter, and 20q13, which likely harbor genes that play a role in the pathogenesis of soft-tissue leiomyosarcomas. There were no obvious correlations between the chromosomal changes identified and available clinical details.

Adult↗

PubMed2005

Alterations of cancer-related genes in soft tissue sarcomas: hypermethylation of RASSF1A is frequently detected in leiomyosarcoma and associated with poor prognosis in sarcoma.

Aberrant methylation is a main mechanism of tumor suppressor gene inactivation in carcinogenesis. In this study, the methylation status of RASSF1A, p16, MLH1, MSH2 and ERalpha was investigated in 84 primary soft tissue sarcomas (STSs), including 22 liposarcomas, 18 malignant fibrous histiocytomas (MFHs), 18 leiomyosarcomas, 6 rhabdomyosarcomas, 6 neurogenic sarcomas and several other sarcoma entities. RASSF1A hypermethylation was detected in 17 of 84 (20%) STSs; however, methylation was more frequent in leiomyosarcomas (39%) compared to MFHs (6%; p < 0.015) and liposarcomas (18%). The p16 CpG island was methylated in 22 out of 82 (27%) cases. In 7 out of 81 (9%) STS samples, the promoter of MLH1 was methylated and in liposarcoma the methylation frequency was higher (14%). For MSH2, no hypermethylation was detected. Methylation of ERalpha was detected in 48 of 63 (76%) STSs, but also in 4 of 8 (50%) normal tissue samples. Furthermore, we analyzed mutational activation of K-ras and BRAF. In 4 out of 84 (5%) of STSs, a substitution at codon 599 of BRAF was found; however, no alteration of K-ras was detected. In an univariate Cox proportional-hazards regression model, we found that the risk of a tumor-related death for STS patients with methylated RASSF1A was significantly increased (RR = 2.9; p = 0.037). In summary, our data indicate that inactivation of RASSF1A is a common event in STS, especially in leiomyosarcoma. Thus, the methylation status of cancer-related genes was distinct in different STS and methylation of RASSF1A promoter can serve as prognostic marker in STSs.

Adult↗

PubMed1981

Sonographic features of gastrointestinal leiomyosarcoma.

Five cases of gastrointestinal leiomyosarcoma were studied by ultrasound. Sonographic features of a huge mass with a thick echogenic rim and central sonolucent cavitation were seen in four cases with presenting symptoms of an abdominal mass. In a fifth case necrotic metastasis was seen in the liver after removal of a leiomyosarcoma. Although not pathognomonic, such features should strongly suggest the diagnosis of leiomyosarcoma.

Aged↗

PubMed2004

Smooth muscle tumors of uncertain malignant potential and leiomyosarcomas of the uterus: MR findings.

PURPOSE: To study the MR characteristics of nonbenign uterine smooth muscle tumors. MATERIALS AND METHODS: Nine patients with pathologically proven leiomyosarcomas, and three patients with smooth muscle tumors of uncertain malignant potential (SMTUMP) were included in this study. Twelve cases of benign uterine leiomyomas and variants, in which gynecologists suspected leiomyosarcomas, were also analyzed. In each case we studied the size, location, signal intensity, and contrast enhancement of the tumors. RESULTS: Nine of the 12 nonbenign characters had more than 50% of high-intensity areas on T2-weighted images (T2WI), and some hyperintense foci on T1-weighted images (T1WI). In the contrast study, nine of 12 nonbenign characters had well-demarcated unenhanced areas. On the other hand, only two of 12 benign characters showed such a signal intensity pattern, and none of them had a pocket-like unenhanced area. CONCLUSION: Although there were some exceptions, more than 50% of high signal on T2WI, and the presence of any small high-signal areas on T1WI with unenhanced pockets were considered MR-suggestive for SMTUMPs and leiomyosarcomas.

Adult↗

PubMed1978

Leiomyosarcomas of the extremities: angiography as a diagnostic aid.

Thirteen cases of leiomyosarcomas of the extremities are reported. Most of the cases were operated on without information concerning benignancy or malignancy. Plain radiographs showed amorphous calcification in two cases. Three cases had angiographic study and all revealed malignant neovascularity. In no literature that we reviewed was angiography mentioned in the cases of leiomyosarcomas of the extremities. Venography alone has been done in many reported cases. The leiomyosarcoma of the extremities looks often benign but is notorious for local recurrences and distant metastases.

Adult↗

PubMed1980

Leiomyosarcoma of the gastrointestinal tract: should we consider metastasis to regional lymph nodes?

About one third of leiomyosarcomas of the gastrointestinal tract do develop metastasis; and nearly 90% of the metastasis are intra-abdominal. It is said that leiomyosarcoma rarely involves regional nodes, although two series reported 8% of leiomyosarcomas of the stomach and 7% of the small intestines had nodal metastasis at initial operations (mostly of limited resections). It is suggested that wider resection of primary tumor with adjacent mesentery may reveal higher frequency of occult nodal metastasis and might prevent some of the loco-regional relapse and sarcomatosis.

Gastrointestinal Neoplasms↗

PubMed1986

Leiomyosarcoma of the esophagus: report of a case and review of the literature.

Leiomyosarcoma of the esophagus is a very rare neoplasm, the total number of reported cases in the English literature being 44 patients. We describe another case of the esophageal leiomyosarcoma to add to the recorded experience of this rare tumor. The therapeutic and prognostic implications of the tumor are discussed, and the related literature is reviewed. In spite of the progress in the thoracic surgical care of patients and the progress in the field of oncology, overall prognosis of patients with leiomyosarcoma of the esophagus remains unchanged. Patients with polypoid lesions and well-differentiated histologic features appear to show better chance of survival. Simple excision of the lesion can be as effective as radical excision, especially in well-localized lesions.

Esophageal Neoplasms↗

PubMed1994

Uterine leiomyosarcomas coexistent with cellular and atypical leiomyomata in a young woman during the treatment with luteinizing hormone-releasing hormone agonist.

We report a case of a 28-year-old woman who had received 2 months of intranasal buserelin (a luteinizing hormone-releasing hormone agonist; LH-RH agonist) therapy for presumed uterine leiomyomata. In addition to no reduction of the tumor size evaluated by sonography, heavy vaginal bleeding and abdominal pain recurred and worsened during the therapy. Pathological examination of the myomectomy and hysterectomy specimens revealed leiomyosarcomas coexistent with cellular, atypical, and classical leiomyomata. We suggest that this is a case of leiomyosarcomas arising in preexisting leiomyomata rather than de novo from the smooth muscle fibers of the myometrium. Furthermore, the potential of LH-RH, agonist therapy to delay the surgical treatment of an unsuspected leiomyosarcoma is high-lighted. Close monitoring of the treatment response by improvement of clinical symptoms and sonographic assessment of tumor size may be helpful in early diagnosis of an underlying malignant tumor.

Adult↗

PubMed1995

Leiomyosarcoma of the broad ligament: a case report and literature review.

Solitary tumors of the broad ligament are rare. The most frequent solid tumor of the broad ligament is leiomyoma. Leiomyosarcoma of the broad ligament (LBL) is an extremely rare malignant tumor. Nine cases of leiomyosarcoma of the broad ligament had been reported previously. A 59-year-old woman with LBL is reported. Treatment consisted of resection and total abdominal hysterectomy with bilateral salpingo-oophorectomy. No evidence of metastasis is noted after 12 months follow-up. We conclude that the management of LBL is excision of the leiomyosarcoma as completely as possible and postoperative adjuvant chemotherapy.

Broad Ligament↗

PubMed1996

Combination chemotherapy with hydroxyurea, dacarbazine (DTIC), and etoposide in the treatment of uterine leiomyosarcoma: a Gynecologic Oncology Group study.

Advanced or recurrent uterine leiomyosarcomas have traditionally been resistant to most chemotherapeutic regimens. Preliminary reports suggested the combination of hydroxyurea, dacarbazine (DTIC), and etoposide (VP-16) was sufficiently effective to warrant larger trials. In a Phase II trial undertaken by the Gynecologic Oncology Group, 39 patients with advanced or recurrent leiomyosarcoma were treated with 2 g of hydroxyurea, 700 mg/m2 of DTIC, and 300 mg/m2 of VP-16 in divided doses every 4 weeks. Thirty-eight patients were evaluable for response. Two patients experienced complete responses and five had partial responses for a total objective response rate of 18.4% (95% confidence interval: 7.7-34.3%). In general, therapy was well tolerated with moderate toxicity. Six of the seven responders had disease outside the pelvis. The combination of hydroxyurea, DTIC, and VP-16 exhibits moderate activity against uterine leiomyosarcoma.

Adult↗

PubMed1997

Stage IIIC ovarian leiomyosarcoma in a premenopausal woman with multiple recurrences: prolonged survival with surgical therapy.

A case of Stage IIIC primary ovarian leiomyosarcoma in a premenopausal woman with multiple recurrences alive and well 7 years after diagnosis is presented. In addition to the typical light microscopic, immunohistochemical, and electron microscopic features of ovarian leiomyosarcoma, the tumor was progesterone receptor positive. This is 28th report of primary ovarian leiomyosarcoma and the first report of progesterone receptor in this tumor. This is the longest reported survival in a woman with this disease.

Adult↗

PubMed1998

Prolonged oral etoposide in recurrent or advanced leiomyosarcoma of the uterus: a gynecologic oncology group study.

OBJECTIVE: In a previous study by the Gynecologic Oncology Group only modest activity was seen with bolus etoposide in leiomyosarcoma of the uterus (an 11% response rate). To exploit the schedule dependency of etoposide, which favors longer exposure, a Phase II trial of prolonged oral etoposide was conducted in this tumor. METHODS: Eligibility included leiomyosarcoma of the uterus, measurable disease, one prior chemotherapy regimen which did not include etoposide, WBC >/= 3000/microliter, platelet count >/=100, 000/microliter, serum creatinine </=2 mg%, and adequate hepatic function. The starting etoposide dose was 50 mg/m2/day (30-40 mg/m2/day for prior radiotherapy) as a single dose for 21 days, every 28 days. Based on toxicity, a dose escalation to a maximum etoposide dose of 60 mg/m2/day was prescribed. RESULTS: Thirty-six patients were entered on this study; 34 were evaluable for toxicity and 29 were evaluable for response. A median of 2 courses were given (range 1-14). Grade 4 neutropenia occurred in 20.6% and grade 4 thrombocytopenia occurred in 5.8%. One patient developed acute myeloid leukemia 10 months after completing 7 cycles of therapy. Among the patients evaluable for response, 27 had received prior chemotherapy and 6 had received prior radiation therapy. Two partial responses (6.9%) were observed. CONCLUSION: This regimen has minimal activity as second-line chemotherapy in leiomyosarcoma of the uterus. No benefit to the chronic oral schedule is evident.

Administration, Oral↗

PubMed2002

Phase II study of mitomycin, doxorubicin, and cisplatin in the treatment of advanced uterine leiomyosarcoma: a Gynecologic Oncology Group study.

OBJECTIVE: Because of preliminary observations favoring the use of mitomycin, doxorubicin, and cisplatin (MAP) chemotherapy in leiomyosarcomas, the Gynecologic Oncology Group (GOG) decided to conduct a phase II clinical trial of this combination regimen in patients with advanced disease. METHODS: Patients with histologically confirmed uterine leiomyosarcoma who had not previously received cytotoxic drugs were considered for participation in this clinical trial. Eligible patients had measurable disease, GOG performance status 0-2, and adequate bone marrow, renal, and hepatic function according to standard criteria. Mitomycin 8 mg/m(2) and doxorubicin 40 mg/m(2) were each given by iv injection followed immediately by cisplatin 60 mg/m(2) in 1 liter of 0.45% saline plus mannitol 25 g. Patients who remained free from tumor progression or intolerable toxicity received at least three, to a maximum of six, cycles of MAP. RESULTS: Forty-one patients were registered, of whom 4 were determined ineligible (wrong cell type, 2; wrong site of origin, 1; inadequate pathology material, 1). Thirty-five of the 37 were evaluable for response after receiving from one to six (median three) cycles of MAP. Three patients (9%) achieved a complete response and 5 (14%) exhibited a partial response. The most common adverse effects were leukopenia (33 patients) and thrombocytopenia (30 patients). Pulmonary toxicity was seen in 10 patients and was a factor in the clinical deterioration and death of 2. CONCLUSION: MAP is active against advanced uterine leiomyosarcomas, but not remarkably so. Despite its low therapeutic index, this novel, possibly interactive, combination may serve as a forerunner to regimens that more efficiently exploit the enhancement of sarcoma cell kill under hypoxic conditions.

Antineoplastic Combined Chemotherapy Protocols↗

PubMed2002

Surgical resection of pulmonary and extrapulmonary recurrences of uterine leiomyosarcoma.

OBJECTIVE: The objective was to determine long-term survival and predictors of outcome in a retrospective cohort of patients who underwent surgical resection of recurrent uterine leiomyosarcoma (LMS). METHODS: Between January 1991 and March 2001, 41 patients who underwent surgical resection for recurrent uterine leiomyosarcoma were identified. The records of these patients were reviewed and abstracted data included patient age, date of initial diagnosis, tumor histology and grade, residual tumor after all operations, the use of adjuvant therapy, dates and sites of all recurrences, and disease status at last follow-up. Survival was determined from the time of first recurrence to last follow-up. Survival curves were estimated using the Kaplan-Meier method and P values were generated using the likelihood ratio test from the Cox proportional hazards model and chi(2) analysis. RESULTS: Forty-one patients with recurrent uterine LMS (17 local pelvic, 18 distant, 6 both) underwent surgical resection at time of first recurrence. A thoracic procedure alone was performed in 13 cases. Information on residual disease was available for 37 patients. The disease-specific 2-year survival for all 41 patients was 71.2% (95% CI: 58.1, 87.3). In univariate analysis, time to first recurrence and optimal resection were significantly associated with longer overall survival. CONCLUSION: Optimal surgical resection for recurrent uterine leiomyosarcoma may provide an opportunity for long-term survival in a select patient population. Time to first recurrence and optimal surgical resection were predictors of improved outcome in this study.

Adult↗

PubMed1994

Endorectal ultrasound and leiomyosarcoma of the rectum.

Leiomyosarcomas of the rectum are uncommon neoplasms accounting for fewer than 1% of all malignancies of the colon and rectum. Approximately 215 cases have been described in the literature. Distinction from leiomyoma is often difficult, but regarding dignity is important. The case reported is that of a 68-year-old man in whom a 2.2-cm rectal mass covered by mucosa was diagnosed by rectoscopy and endorectal ultrasound. After treatment by wide local excision the histological specimen revealed a low-grade, highly differentiated leiomyosarcoma. Due to the lack of a large series of this disease, there is disagreement over the therapeutic strategy. At the moment a selective treatment approach seems to be the most frequently advocated. Large tumors and those extending beyond the rectal wall are treated by radical surgical resection. Leiomyosarcomas less than 2.5 cm in size and confined to the bowel wall can be treated by wide local excision. Endosonography can provide exact assessment of tumor size and expansion and is of great value in selecting the appropriate treatment.

Aged↗

PubMed1992

Dedifferentiated leiomyosarcoma of the intestinal tract: histological, ultrastructural and immunohistochemical examinations.

Six cases of dedifferentiated leiomyosarcoma of the small and large bowel are presented with histological, ultrastructural and immunohistochemical examination. One case arose in the jejunum, two in the ileum, and the other three in the large intestine. The tumours were submucosal in four cases with large areas of ulceration; two were polypoid. Four tumours showed typical leiomyosarcomatous appearance with dedifferentiated components and two were typical leiomyosarcomas at the primary site with differentiated components only in metastatic foci. By immunohistochemistry, typical leiomyosarcomatous areas showed a positive reaction for muscle-specific actin (MSA), MB1, MB2 and myosin. In contrast, desmin-positive cells were scattered throughout the tumour or were not present. Tumour cells in dedifferentiated components were positive for alpha-1-antitrypsin and alpha-1-antichymotrypsin in all cases but one; neuron specific enolase, MB1, MB2 and myosin were positive with variety. MSA was faintly positive in only a few tumour cells of two cases and desmin was not detected in any of the cases studied. Ultrastructurally, tumour cells in typical leiomyosarcomatous areas demonstrated evident smooth muscle features, although in dedifferentiated areas they lacked such features except in one case. Our results indicate that dedifferentiated elements may derive from ordinary leiomyosarcoma and loose muscle features due to dedifferentiation.

Aged↗

PubMedDate not supplied

Leiomyosarcoma in childhood and adolescence.

BACKGROUND: Few series of leiomyosarcoma in patients < 21 years of age have been reported. We reviewed our institutional experience with this neoplasm to learn disease characteristics, patterns of relapse, and outcome. METHODS: The records of 21 patients with leiomyosarcoma admitted to our institution were reviewed retrospectively; 18 of these were diagnosed after 1970. Overall survival was estimated using the Kaplan-Meier method. RESULTS: Ninety-five percent (20 of 21) were initially treated with a wide local excision that was complete with a negative microscopic margin in 10 (48%). There also was a strong correlation between grade and surgical margins. High-grade tumors were associated with a lower rate of complete resection. The majority underwent additional therapy. Radiation was used to treat both initial and recurrent disease in nine patients, with four of these undergoing brachytherapy. Thirteen patients were treated with adjuvant chemotherapy, most commonly doxorubicin (seven patients) and cisplatin (six patients). The median length of survival was 9.3 years, and there were nine disease-related deaths (43%). Of interest was the progressive decrease in survival with time. The 5-year overall survival rate was 79%; the 10-year rate was 49%. Three patients died of progressive disease > 10 years after initial diagnosis. CONCLUSIONS: We conclude that leiomyosarcomas arising in childhood and adolescence are associated with a good initial chance of survival that decreases progressively over time. Known prognostic factors from larger adult series are consistent with the present data, but they are not provable because of the small number of patients. In particular, the grade was correlated with surgical margins.

Adolescent↗