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PubMedDate not supplied

Acute heart failure as manifestation of metastatic uterine leiomyosarcoma to the heart and lung.

Uterine leiomyosarcoma is an uncommon malignant neoplasm that frequently metastasizes through the bloodstream to distant organs such as the lung, bone, and brain. We report on and discuss a patient who had sudden onset of acute heart failure with pulmonary edema and bilateral pleural effusions, resulting from a high-grade uterine leiomyosarcoma metastatic to the left atrium. Surgical removal of the tumor terminated the acute heart failure and pulmonary edema. Other unusual features of this case include the metastatic leiomyosarcoma to the left atrium with expression of acute heart failure and the pulmonary tumor lesion obscured by the shadows of pleural effusion, pulmonary edema, and the breast.

Acute Disease↗

PubMed2004

Expression of the mitogen-inducible gene-2 (mig-2) is elevated in human uterine leiomyomas but not in leiomyosarcomas.

To investigate the role of a recently cloned cell proliferation-related gene, mig-2 (mitogen-inducible gene-2), in the growth of uterine leiomyomas, this gene's expression at mRNA and protein levels was examined in normal myometrium, leiomyomas, and leiomyosarcomas of the uterus. Northern blotting, Western blotting, and immunohistochemical staining demonstrated that mig-2 expression was increased in leiomyomas compared with normal myometrium, especially during the secretory phase of the menstrual cycle. In contrast, the mig-2 expression was greatly decreased in leiomyosarcomas. Direct sequencing of the whole coding region of mig-2 cDNA and Southern blotting revealed that mig-2 alterations, such as mutations, rearrangement, and amplification, were not present in either leiomyomas or leiomyosarcomas. These findings suggest that mig-2 expression is transcriptionally elevated in leiomyomas and could be involved in its hormone-mediated growth of leiomyomas of the uterus.

Basic Helix-Loop-Helix Proteins↗

PubMed2005

A peculiar site of leiomyosarcoma: the tongue tip--report of a case.

Primary leiomyosarcoma of the tongue is an exceedingly rare malignant mesenchymal tumour, which is hardly ever addressed in the differential diagnosis of a tongue lesion. Only 7 cases of this tumour have been documented in English-language medical literature. This study reports an additional case of primary leiomyosarcoma of the tongue in a 54-year-old woman, presenting with a foreign body sensation at the tip of the tongue with a rapidly and progressively enlarging ulcerative nodule. The patient received tumour excision with a 1-year follow-up without evidence of local recurrence or distant metastasis. Leiomyosarcoma in this unusual site seems to have better prognosis than elsewhere, although the number of cases are too small to draw a conclusion. This study further reviews the literature review and discusses the clinico-pathological presentation of this peculiar rare tongue lesion.

Female↗

PubMed2004

Infantile intestinal leiomyosarcoma is prognostically favorable despite histologic aggressiveness: case report and literature review.

Intestinal leiomyosarcomas in children are extremely rare. The authors present a case of infantile intestinal leiomyosarcoma of the colon that was resected and was classified histologically as grade 2 because of abundant mitoses. The tumor cells infiltrated the submucosa and the serosal surface. Immunohistochemically, these cells were positive for alpha-smooth muscle actin but not for CD34 or CD117, by which the tumor was differentiated from gastrointestinal stromal tumor (GIST). The patient underwent follow-up without adjuvant therapy and is now doing well with no evidence of the disease for 42 months. Based on the current case and a review of 18 reported cases, it is concluded that infantile intestinal leiomyosarcoma, which is differentiated from GIST, has a favorable prognosis even when the histologic appearance is suggestive of aggressive biological characteristics. Adjuvant therapy is unnecessary when tumors are completely resected.

Actins↗

PubMed2005

Myxoid leiomyosarcoma of the liver.

This is the first report of a myxoid leiomyosarcoma arising in a cirrhotic liver. The tumor was resected from a 64-year-old man. On gross examination, it was soft and hemorrhagic. The tumor was composed of deceptively benign-looking smooth muscle cells with clear cytoplasm suspended in a myxoid stroma with foci of hemorrhage. Immunohistochemistry and electron microscopy confirmed that this was a smooth muscle cell neoplasm. The abundance of glycogen and ultrastructural signs of smooth muscle differentiation were considered consistent with an immature smooth muscle cell phenotype consistent with the diagnosis of myxoid leiomyosarcoma. Since myxoid leiomyosarcomas are aggressive tumors, it is important to recognize them histologically and also bear in mind that these tumors can occur even in unusual extrauterine locations such as a cirrhotic liver.

Biomarkers, Tumor↗

PubMed2003

Clinicopathological analysis of c-kit expression in carcinosarcomas and leiomyosarcomas of the uterine corpus.

OBJECTIVE: The purpose of this study was to evaluate the expression of the protooncogene, c-kit, in carcinosarcomas and leiomyosarcomas of the uterine corpus and determine the associations between c-kit expression and clinicopathologic factors, including clinical outcome. METHODS AND MATERIALS: Using a polyclonal anti-KIT-antibody, immunohistochemical staining was performed on formalin-fixed paraffin-embedded tissue blocks from 21 carcinosarcomas, 17 leiomyosarcomas, and 1 endometrial stromal sarcoma. KIT-positive tumors were defined as those tumors demonstrating immunopositivity in > or =30% of tumor cells examined. KIT-negative lesions demonstrated immunopositivity in <30% of tumor cells. Two authors independently scored the slides as positive or negative. Staining was repeated on all specimens and independently scored, and in the occasion of a mismatch, a third staining was performed. The carcinosarcomas were catalogued as to whether the sarcomatous and/or carcinomatous elements expressed c-kit. Clinical data were abstracted for those patients with uterine carcinosarcomas. The associations between clinicopathologic characteristics and c-kit expression were compared using univariate and multivariate analyses. Kaplan-Meier curves based on c-kit expression were plotted for progression-free and overall survival and compared using the log-rank test. RESULTS: Nine of 21 (43%) carcinosarcomas demonstrated immunopositivity for the KIT receptor, although staining was relatively weak. In contrast, only 1/17 (6%) leiomyosarcomas demonstrated KIT immunopositivity (P = 0.029). The solitary endometrial stromal sarcoma evaluated did not demonstrate significant KIT positivity. The majority of KIT-positive carcinosarcomas (6/9 (67%)) demonstrated KIT presence in the sarcomatous portion as compared to the carcinomatous portion (4/9 (44%)). No clinical factor had a statistically significant association with c-kit expression. The lack of c-kit expression was the only factor that was significantly associated with disease recurrence in univariate and multivariate analyses (P < 0.05), although there appeared to be a trend toward a low stage associated with kit positivity. The median progression-free interval for the KIT-negative cohort was 8 months, while it had not been reached for the KIT-positive cohort after median follow-up of 15 months (P = 0.0462). CONCLUSIONS: A significant proportion of carcinosarcomas of the uterine corpus display immunoreactivity for c-kit. Patients with KIT-positive carcinosarcomas may have an improved progression-free survival compared to KIT-negative tumors; however, further data are needed to determine whether this finding is confounded by stage.

Aged↗

PubMed2005

Myxoid leiomyosarcoma of the vulva.

BACKGROUND: Myxoid leiomyosarcoma (MLMS) of the vulva is a mesenchymal tumor with only five reported cases in the literature. CASE: We report an 85-year-old woman with a unilateral nonulcerating, painless vulvar mass. According to the patient, the mass slowly enlarged. Initial biopsies were benign. However, clinically, this lesion was suspicious for a soft tissue tumor. Therefore, the mass was removed by a wide local excision. Definitive histology revealed a myxoid leiomyosarcoma of the vulva. At present, 25 months after the operation, the patient is well with no sign of recurrence. CONCLUSIONS: Vulvar myxoid leiomyosarcomas are rare and can be confused with other benign or malignant tumors. It is important to be aware of this rare tumor variant, in order to plan appropriate treatment.

Aged↗

PubMed2005

Phase II evaluation of liposomal doxorubicin (Doxil) in recurrent or advanced leiomyosarcoma of the uterus: a Gynecologic Oncology Group study.

OBJECTIVE: This is a phase II group-wide study of liposomal doxorubicin chemotherapy in patients with advanced or recurrent uterine leiomyosarcomas. The aim was to evaluate clinical response and toxicity. METHODS: Patients with histologically confirmed persistent or recurrent leiomyosarcomas of the uterus with documented disease progression after appropriate local therapy were invited to participate in this study. Bidimensionally measurable disease, GOG performance status of 0, 1, or 2 (Karnofsky 80-100) was required; all patients must have failed local therapeutic measures and be considered incurable. Other eligibility criteria included adequate hepatic, renal, and hematologic function. Patients were ineligible if they had received previous chemotherapy or had other noncutaneous malignancies. Patients received liposomal doxorubicin 50 mg/m2 IV over 1 h. Courses were repeated every 4 weeks until disease progression or adverse side effects supervened. RESULTS: Thirty-five patients were entered into this study between May 2000 and June 2001. Three patients were determined ineligible because of wrong pathological cell type or inadequate pathology information and one was inevaluable for lack of data. Median age was 52 years (range 36-78 years). GOG performance status was 2 in 1 instance, 1 in 15 cases, and 0 in 15 others. Eleven patients (35.5%) had received radiotherapy. A median of 2.0 courses was given (range 1-8). Five patients (16.1%) experienced grade 3 or 4 neutropenia, and seven (22.6%) had grade 3 or 4 anemia. Two patients developed grade 3 and one patient developed grade 4 cardiovascular adverse events, not necessarily drug related. There were seven cases of grade 3 or 4 gastrointestinal toxicity and two patients developed grade 3 dermatologic toxicity. One complete (3.2%) and four partial (12.9%) responses were reported. Ten patients (32.3%) had stable disease, 15 (48.4%) had increasing disease, and response could not be assessed in 1 (3.2%). CONCLUSION: The dose and schedule of liposomal doxorubicin employed in this trial showed no advantage over historical results with doxorubicin in the treatment of uterine leiomyosarcoma.

Adult↗

PubMed2006

Reclassification of a tubal leiomyosarcoma as an eGIST by molecular evaluation of c-KIT.

BACKGROUND: Extragastrointestinal stromal tumors (eGISTs) are rare mesenchymal-derived tumors arising outside of the GI tract. eGISTs are often histologically confused with leiomyosarcoma. Distinction between eGIST and leiomyosarcoma is critical because of the unique responsiveness of eGISTs to the molecularly targeted agent imatinib. CASE: A woman presented with a history of tubal spindle cell tumor that was initially diagnosed and treated as a leiomyosarcoma. Because of minimal response to sarcoma directed chemotherapy, the possibility that the tumor was in fact an eGIST was investigated and supported by immunohistochemical and mutational analyses of the c-Kit receptor tyrosine kinase. The patient currently has stable disease control on imatinib for the last 18 months. CONCLUSIONS: The possibility of eGIST should be considered in the differential diagnosis of tumors with a spindle cell morphology in the gynecologic tract especially when involving the ovary, fallopian tube, or uterine serosa.

Base Sequence↗

PubMed2006

Activity of trabectedin (ET-743, Yondelis) in metastatic uterine leiomyosarcoma.

BACKGROUND: The ongoing search for more effective agents in the treatment of uterine leiomyosarcomas is warranted because of the poor prognosis related to these tumors. CASE: A case of advanced, recurrent and refractory uterine leiomyosarcoma is presented that responded to trabectedin (ET-743) 1.2 mg/m2 intravenously over 24 h every 3 weeks after failing four prior regimens. A durable objective response lasting at least 8 months was documented. CONCLUSION: Trabectedin (ET-743) has activity in uterine leiomyosarcoma and warrants further investigation.

Adult↗

PubMed2003

Primary orbital leiomyosarcoma: a case report with MRI findings.

PURPOSE: To present a case of a primary orbital leiomyosarcoma and the corresponding magnetic resonance imaging (MRI) appearance. DESIGN: Observational case report. METHODS: Correlation of MRI with clinicopathologic findings. RESULTS: A 56-year-old woman presented with a 10-month history of diplopia in left gaze and left exophthalmos. An enhanced MRI of the left orbit revealed an extraconal, peripheral-enhancing mass measuring 2 cm in maximal diameter with displacement of the medial rectus muscle. The mass was excised, and the diagnosis of leiomyosarcoma was made by pathologic examination. CONCLUSIONS: Although computed tomography scan and ultrasound have been the imaging modalities used previously to evaluate cases of primary orbital leiomyosarcoma, MRI can provide important additional information regarding tumor characterization that is useful in the diagnosis and treatment of this rare malignancy.

Female↗

PubMed1999

Mutations in exon 11 of c-Kit occur preferentially in malignant versus benign gastrointestinal stromal tumors and do not occur in leiomyomas or leiomyosarcomas.

Gastrointestinal stromal tumors (GISTs) comprise the largest subset of mesenchymal tumors of the gastrointestinal tract. These neoplasms differ histologically and immunohistochemically from typical leiomyomas and leiomyosarcomas. Most GISTs express CD34 and CD117 (c-kit protein) but not desmin. Recently, gain-of-function mutations of c-kit proto-oncogene have been shown in five solitary GISTs and in tumors and leukocytes from a family with multiple GISTs. An in-frame deletion or a point mutation in exon 11 of c-kit was detected in these cases. Stable transfection of the mutant c-kit complementary DNA was also shown to induce malignant transformation of murine lymphoid cells, suggesting that the c-kit mutations contribute to tumor development. In this study, we evaluated 43 GISTs and 14 smooth muscle tumors for mutations in the exon 11 of c-kit by a PCR-assay. Half of the malignant GISTs (12/24) and only one benign GIST (1/19) revealed mutant bands. No mutant bands were found in 3 leiomyomas and 11 leiomyosarcomas. Sequence analysis confirmed the presence of an in-frame deletion of 3-21 bp in all 13 GISTs with mutant bands. Wild-type bands from 8 malignant and 11 benign GISTs and 7 smooth muscle tumors without mutant bands were cloned and sequenced. Additional mutations were found in 3 malignant and 2 benign GISTs. There were no mutations in 3 leiomyomas and 4 leiomyosarcomas. The mutation status of exon 11 did not correlate with immunohistochemically detectable expression of the CD117, as virtually all GISTs with or without such mutations showed CD117 immunoreactivity. The c-kit mutations occur preferentially in malignant GISTs and might be a clinically useful adjunct marker in the evaluation of GISTs. The conservation of the c-kit mutation pattern, observed in consecutive lesions from the same patients, suggests that these mutations might be useful tumor markers in monitoring recurrence or minimal residual disease.

Adult↗

PubMed2001

Leiomyosarcoma of the prostate: a study of two cases.

Leiomyosarcoma of the prostate is rare neoplasm that accounts for less than 0.1% of prostate malignancies. A number of treatment approaches have been adopted including radical surgery, radiotherapy and chemotherapy, but in no instance has a successful outcome been obtained. Prostate leiomyosarcoma has a poor prognosis, although survival time can vary. For these reasons, it is important to correctly identify this disease and report its occurrence, type of treatment, and the response to therapy of each patient diagnosed as having leiomyosarcoma of the prostate in an attempt to improve our understanding of the natural history of these lesions.

Humans↗

PubMed2004

Surgical management of leiomyosarcoma of the mediastinum.

Leiomyosarcoma of the mediastinum is sufficiently rare; therefore therapeutic approaches are not well defined. Current therapy primarily rests with the thoracic surgeon. We report a single case of a large leiomyosarcoma of the mediastinum invading the heart and right lung. Our diagnostic and therapeutic approach to this patient is presented and the pertinent literature is reviewed. We conclude that little advancement has been made with regard to adjuvant therapies for leiomyosarcoma of the mediastinum, and the only opportunity for cure remains with full surgical extirpation.

Adult↗

PubMed1998

Leiomyosarcoma of the esophagus: results of surgical treatment.

BACKGROUND: This study examined the results of surgical treatment of leiomyosarcoma of the esophagus. METHODS: Between January 1920 and December 1996, 17 patients (9 men and 8 women) with leiomyosarcoma of the esophagus were treated surgically at the Mayo Clinic. Median age was 58 years and ranged from 26 to 76 years. Symptoms included dysphagia in 11 patients (64.7%) and odynophagia in 6 (35.3%). The tumor was located in the middle third of the esophagus in 10 patients (58.8%) and in the cervical esophagus in 7 (41.2%). Procedures performed included esophagogastrectomy in 9 patients (Ivor Lewis in 5, left thoracoabdominal in 3, and transhiatal in 1), enucleation in 3, transgastric excision in 1, and exploration without resection in 4. RESULTS: The procedure was considered curative in 11 patients (64.7%). There was one operative death (mortality, 5.9%). Complications occurred in 3 patients (17.6%) and included anastomotic leak in 2 and bleeding requiring reoperation in 1. Growth pattern was infiltrating in 7, polypoid in 5, and intramural in 5. Histologically, the tumor was grade 1 in 6 patients, grade 2 in 2, grade 3 in 7, and grade 4 in 2. The tumor was postsurgically classified as stage I in 2 patients, stage IIA in 7, stage IIB in 1, stage IIIA in 5, stage IV in 1, and unknown in 1. Six patients (35.3%) received adjuvant treatment. Follow-up was complete in 16 patients (94.1%) and ranged from 1 to 182 months (median, 48 months). Five- and 10-year actuarial survivals were 47.0% and 31.0%, respectively. Seven patients (41.2%) are currently alive (median survival, 72 months); all underwent curative resection. Factors affecting survival included completeness of resection, growth pattern, postsurgical stage, tumor grade, and tumor location (p < 0.05). CONCLUSIONS: We conclude that leiomyosarcoma of the esophagus is rare. Complete resection provides long-term survival.

Adult↗

PubMed1992

Leiomyosarcoma of the stomach: determinants of long-term survival.

To determine factors associated with a favorable long-term prognosis in gastric leiomyosarcoma, we retrospectively reviewed the medical records of 93 Mayo Clinic patients with this biopsy-proven tumor diagnosed during the 25-year period from 1964 through 1988. Six patients who had Carney's triad (gastric epithelioid leiomyosarcoma, pulmonary chondroma, and functioning extra-adrenal paraganglioma) were excluded from data analysis. The other 87 patients participated in follow-up until death or for a median duration of 5.8 years for those who were alive at the conclusion of the study. The most common symptoms at the time of initial assessment were abdominal pain (51%), melena (36%), and weight loss (16%). Most often, the tumor was located in the greater curvature (25%), fundus (20%), or lesser curvature (16%) of the stomach. Two percent of patients had tumors at multiple sites. All 87 gastric leiomyosarcomas were histologically confirmed; 38% were grade 1, 37% were grade 2, and 25% were either grade 3 or grade 4. Metastatic involvement was noted in 15% of patients at the time of diagnosis. The 5- and 10-year survival rates were 45% and 34%, respectively, and the 5- and 10-year tumor recurrence rates were 57% and 65%, respectively. Variables that were associated with long-term survival were low histologic grade of the tumor, absence of metastatic lesions, and small tumor size (P < 0.01); variables such as site of the tumor, initial symptoms, age, and sex provided no significant additional prognostic information.

Adult↗

PubMed2002

Uterine leiomyosarcoma metastatic to the thyroid.

BACKGROUND: Uterine leiomyosarcoma is an aggressive tumor that has a propensity for distant metastasis. Distant sites of spread include the lung, liver, brain, and bone. CASE: A woman in her 5th decade was diagnosed with stage IV leiomyosarcoma of the uterus. Distant metastasis in the chest was confirmed by thoracotomy at the time of her original diagnosis. She subsequently experienced multiple episodes of metastasis including metastatic disease to her thyroid. She survived 71 months after diagnosis of stage IV disease. CONCLUSION: Our case represents the first reported experience with uterine leiomyosarcoma metastatic to the thyroid. Additionally the case is notable because the patient experienced multiple episodes of asymptomatic stable disease after repetitive chemotherapeutic regimens.

Bone Neoplasms↗

PubMed1998

Metastatic uterine leiomyosarcoma presenting as a primary sarcoma of the parotid gland.

BACKGROUND: Leiomyosarcoma of the uterus has a high metastatic potential to distant sites due to its tendency for hematogenous spread. CASE: A 49-year-old woman presented with an enlarging parotid mass, diagnosed originally as a primary fibrosarcoma. Six years later, she developed pulmonary metastases and heavy, abnormal uterine bleeding. At hysterectomy, a uterine leiomyosarcoma, identical morphologically to the previous lesions, was identified. All tumors showed similar immunohistochemical staining, suggesting the metastatic nature of the original parotid tumor. CONCLUSION: This rare case of uterine leiomyosarcoma, presenting as a primary parotid sarcoma, underscores the importance of considering the possibility of a uterine primary tumor when a sarcoma arises in an organ in which these tumors are unusual.

Female↗