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PubMed2003

Clinical presentation and outcome of high-grade urinary bladder leiomyosarcoma in adults.

OBJECTIVES: To determine the clinical presentation and outcome of patients with high-grade bladder leiomyosarcoma. METHODS: Between July 1986 and April 1998, 36 adult patients (mean follow-up 56 months) with a diagnosis of urinary bladder leiomyosarcoma were evaluated at the University of Texas M. D. Anderson Cancer Center. We retrospectively reviewed the records of these patients for information on clinical features, treatment, and outcome. RESULTS: The mean age of the patients was 63 years. Twenty-six patients were white men in their seventh decade. The most common symptom at presentation was gross hematuria (81% of patients), followed by increased urinary frequency (28%), and dysuria (19%). Thirty-five patients were treated surgically for bladder leiomyosarcoma; of these, 12 (34%) developed recurrent disease (5 with local recurrence and 7 with distant metastasis), with a median time to diagnosis of recurrent disease of 8.3 months. The most common site of distant failure was the retroperitoneum. The disease-specific survival rate at 1, 3, and 5 years was 88.6%, 62.0%, and 62.0%, respectively. Multivariate analyses demonstrated that only the Memorial Sloan-Kettering Cancer Center disease stage system was a significant predictor of survival of patients with bladder leiomyosarcoma (P = 0.018). CONCLUSIONS: In patients with high-grade leiomyosarcoma of the bladder, radical cystectomy in the face of localized disease, whether at the initial presentation or after neoadjuvant chemotherapy, can result in a 5-year, disease-specific survival rate of 62.0%.

Adult↗

PubMed2003

The proto-oncogene c-kit is expressed in leiomyosarcomas of the uterus.

OBJECTIVE: The proto-oncogene c-kit encodes for a 145-kDa transmembrane tyrosine kinase receptor. Interaction with its ligand, stem cell factor, is essential in the development of hematopoietic stem cells, mast cells, gametocytes, melanocytes, and interstitial cells of Cajal. C-kit expression has been identified in a number of different neoplasms that includes mastocytosis/mast cell leukemia, acute myeloblastic leukemia, seminoma/dysgerminoma, and gastrointestinal stromal tumors. This study examines c-kit expression in uterine endometrial stromal sarcomas, leiomyomas, and leiomyosarcomas using immunohistochemistry. METHODS: Archival tissue from 38 patients with the uterine mesenchymal tumors (16 leiomyosarcomas, 8 leiomyomas, 11 low-grade endometrial stromal sarcomas, and 3 high-grade endometrial stromal sarcomas) was stained with polyclonal antibody for c-kit. Modified avidin biotin (ABC) immunoperoxidase method was employed for antibody detection. Individual tumors were considered positive if more than 10% of the cells comprising the neoplasm displayed immunoreactive staining. Staining intensity was graded 1+ to 3+ and distribution graded as focal (10-30% of the cells), intermediate (30-60% of the cells), or diffuse (>60% of the cells). RESULTS: C-kit was positive in 12 (75%) of the 16 leiomyosarcomas. The staining was 3+ and diffuse in the majority of the positive tumors. C-kit expression was not detected in any of the 8 leiomyomas. Two of the 3 high-grade endometrial stromal sarcomas displayed c-kit positivity. Staining was diffuse and 3+ in both of these tumors. Expression of c-kit was observed in only 3 of the 11 low-grade endometrial stromal sarcomas. CONCLUSIONS: C-kit is expressed in uterine leiomyosarcomas and endometrial stromal sarcomas. Adjunctive diagnostic studies using c-kit may be useful in distinguishing leiomyosarcomas from benign leiomyomas in uterine tumors that offer uncharacteristic features. Furthermore, studies should investigate the prospect of treating these malignant tumors with tyrosine kinase inhibitors.

Adult↗

PubMed2006

[Leiomyosarcoma of the lip].

BACKGROUND: Superficial leiomyosarcomas are rare malignant tumors that may be subdivided into cutaneous and subcutaneous cases. CASE-REPORT: A 58 year-old man was seen for a lower lip tumor that appeared 3 years earlier. Histopathologic and immunohistochemical examinations showed features of cutaneous leiomyosarcoma. DISCUSSION: Cutaneous leiomyosarcoma may derive from the arrector pili, smooth muscle of sudoral glands or genital dartoic muscle. The subcutaneous form arises from smooth muscle wall of blood vessels. Superficial leiomyosarcomas occur frequently in the extensor surface of the lower extremities and involve the mucosal area in rare cases. Only five cases arising in the lips have been described. Lip cutaneous leiomyosarcoma may derive from ectopic sweat glands of the lips or from a hypodermic tumor that extends to the lip.

Humans↗

PubMed1999

Prognostic significance of bcl-2 expression in leiomyosarcoma of the uterus.

We examined bcl-2 expression as well as p53 expression and mutation in human uterine smooth muscle tumours to determine the influence of bcl-2 expression on prognosis in patients with uterine leiomyosarcomas. bcl-2 protein was expressed in nearly all benign smooth muscle tumours but in only 57% of leiomyosarcomas. Benign smooth muscle tumours were usually negative for p53 protein, but 16 out of 21 (76%) leiomyosarcomas were positive. A p53 gene mutation was detected in nine of the 16 leiomyosarcomas that showed p53-positive staining. A significant positive correlation was observed between p53 mutation and p53 expression, between the number of mitoses and the Ki-67 labelling index, and between clinical stage and p53 mutation. A significant negative correlation was observed between bcl-2 expression and p53 mutation, and between bcl-2 expression and p53 overexpression. Univariate survival analysis revealed that bcl-2 expression, p53 mutation and clinical stage (stage 1 vs stages 2-4) all showed a significant correlation with prognosis. In a multivariate stepwise regression analysis, positive bcl-2 expression and stage 1 disease were the independent predictors of a favourable prognosis. Our results suggest that bcl-2 is frequently expressed in human uterine smooth muscle tumours, and that its expression may correlate with a favourable prognosis in patients with uterine leiomyosarcoma.

Adult↗

PubMed2000

Leiomyosarcomas of the oral cavity: an unusual topographic subset easily mistaken for nonmesenchymal tumours.

AIMS: Oral leiomyosarcoma is rare and poorly documented. We aimed to characterize these lesions clinicopathologically in order to facilitate their distinction from other spindle cell neoplasms in the oral cavity. METHODS AND RESULTS: Ten cases of oral leiomyosarcoma were retrieved and studied histologically and immunohistochemically. Clinical data were obtained from referring pathologists and prior literature concerning 46 comparable cases was reviewed. Nine out of 10 cases occurred in adults; 50% arose in the jaws and four showed bone involvement. Histological appearances were similar to leiomyosarcomas elsewhere. In addition to myogenic markers, two cases were also keratin-positive. Four patients developed local recurrence or metastatic disease and three died of tumour (median follow-up 37 months). CONCLUSIONS: Leiomyosarcoma is under-recognized in the mouth, often being mistaken for a spindle-celled epithelial neoplasm. Aside from an unusual but infrequent tendency to spread to lymph nodes and a location-specific differential diagnosis, its clinicopathological features are comparable to leiomyosarcomas at other locations.

Adult↗

PubMed2002

Clinicopathological prognostic factors in soft tissue leiomyosarcoma: a multivariate analysis.

AIMS: Prognostic factors affecting survival in cases of leiomyosarcoma of soft parts were investigated in this study. METHODS AND RESULTS: A retrospective study of 267 patients was carried out. This group comprised 142 females (53%) and 125 males (47%), whose ages ranged from 7 to 95 years (median 58 years). One hundred and five cases were superficially situated (arising from the skin or subcutis), while the remaining 162 cases were deeply situated (subfacial). Nineteen were cases of pleomorphic leiomyosarcoma where the diagnosis had been amended from malignant fibrous histiocytoma to leiomyosarcoma whilst under review. Of the 167 patients with follow-up data, 83 died of leiomyosarcoma. In univariate analysis, depth, tumour size (>or=50 mm), mitotic rate of >20 per 10 high-power fields (HPF), tumour necrosis of >50% and a high stage according to the most recent American Joint Committee on Cancer (AJCC) staging for soft tissue sarcoma were found to lessen significantly the rate of survival (log rank test; P < 0.05). However, in multivariate analysis (Cox's proportional hazards model), tumour size and high AJCC stage were the only factors that were correlated independently with decreased survival. CONCLUSIONS: This study indicates that the most reliable prognostic parameters are tumour size and AJCC stage in leiomyosarcoma.

Adolescent↗

PubMed2000

Immunohistochemical detection of cytokeratin and epithelial membrane antigen in leiomyosarcoma: a systematic study of 100 cases.

Although 'aberrant' expression of the epithelial markers, cytokeratin (CK) and epithelial membrane antigen (EMA), in leiomyosarcoma has been described previously, there has not been a study of this phenomenon with clinicopathological correlation in a large series of lesions at different anatomical sites. We investigated systematically the immunohistochemical reactivity for CK and EMA in 100 cases of leiomyosarcoma. CK and EMA were positive in 38% and 44% of the cases, respectively. Although staining was usually focal, extensive immunoreactivity was observed in 11% with CK and 6% with EMA. There was no correlation between immunoreactivity for CK and EMA in leiomyosarcomas and non-neoplastic smooth muscle at the same location. Immunoreactivity for CK and EMA was not correlated with the location, age, sex, histological grade, or histological features, except for more frequent EMA positivity in vascular and uterine tumors than in soft tissue cases. These results indicate that CK and/or EMA-positive leiomyosarcomas do not have distinctive clinicopathological features differing from those of negative cases. However, the considerable frequency of immunoreactivity for these epithelial markers in leiomyosarcoma, occasionally with diffuse and strong immunopositivity, should be recognized as a potentially serious diagnostic pitfall in the differential diagnosis of other malignant spindle cell neoplasms.

Abdominal Neoplasms↗

PubMedDate not supplied

Inflammatory leiomyosarcoma of the ankle: a case report and review of the literature.

Inflammatory leiomyosarcoma is a rare morphologic expression of smooth muscle tumor, which is more often found in the trunk, extremities, and retroperitoneoum. We present a case of inflammatory leiomyosarcoma arising near the ankle joint, superior to the medial malleolus, in a 55-year-old man. Immunohistochemistry, electron microscopy, and cytogenetics were used in the differentiation of this rare morphologic expression of leiomyosarcoma. As in all soft tissue leiomyosarcomas, the accurate prediction of the clinical behavior of this morphologic expression often proves to be difficult. Age, vascular invasion, depth, grade, tumor disruption, size, and stage have prognostic value. Nevertheless, prognosis of inflammatory leiomyosarcoma seems to be favorable when treated early with wide excision of the tumor as in the case presented. Sixteen months after excision of the tumor the patient is doing well with no signs of recurrence.

Ankle Joint↗

PubMed2005

SECONDARY LEIOMYOSARCOMAS: A Report of 4 Cases.

Survivors of childhood cancer risk developing second malignant neoplasms (SMN). a small proportion of SMNs is represented by soft tissue sarcoma (STS), including leiomyosarcoma. The files of the STS Italian Cooperative Group (ICG) were reviewed and 4 patients with secondary leiomyosarcoma were identified, only two of them still alive. In 3 cases, the leiomyosarcoma occurred in the radiation field. The authors found no clear differences between primary and secondary leiomyosarcomas, but leiomyosarcoma proved to be the most frequent SMN registered by the ICG, confirming the need for further studies to identify any distinctive features or syndromes.

Adolescent↗

PubMed2001

Leiomyosarcoma arising in a remnant esophagus after esophagectomy: a case report.

We report an extremely rare case of leiomyosarcoma arising from a remnant esophagus. A 52-year-old Japanese man was referred to our hospital for treatment of a tumor arising from the remnant esophagus. Four years earlier, he underwent a subtotal esophagectomy for esophageal squamous cell carcinoma (well differentiated squamous cell carcinoma, T1N0M0 Stage I) located in the lower esophagus. After preoperative studies, partial esophagectomy with laryngeal preservation and reconstruction using a free graft from the jejunum were performed. Histopathological and immunohistochemical examination revealed leiomyosarcoma without metastasis. Immunohistochemical examination showed that most tumor cells were positive for smooth muscle actin and vimentin, but were negative for cytokeratin and S100. The deeply biopsied specimens are helpful for preoperative histological diagnosis. Mitotic activity has been considered an important criterion of malignancy. However, some cases with minimal mitosis in the tumor grow rapidly and were associated with poor prognosis. Therefore, we advocate that the clinical behavior is the only true indication of malignancy. We also provide a review of 64 cases of esophageal leiomyosarcoma reported in the Japanese literature with available data between 1969 and 1999, including the present case, and discuss their clinicopathological features. Asynchronous occurrence of leiomyosarcoma and squamous cell carcinoma in the esophagus is most unusual and has never been reported. Patients with infiltrating type leiomyosarcoma measuring more than 5 cm in diameter tend to have a poor prognosis. Chemotherapy did not exhibit any survival benefits. In the present patient, no recurrence has been noted for 23 months after surgery.

Carcinoma, Squamous Cell↗

PubMed2002

Leiomyosarcoma of somatic soft tissues: a tumor of vascular origin with multivariate analysis of outcome in 42 cases.

Leiomyosarcomas of the somatic soft tissues (SST) are rare compared with their retroperitoneal and cutaneous counterparts and, therefore, have not been extensively studied. We have analyzed the clinicopathologic features of 42 SST leiomyosarcomas referred in consultation to determine what factors affect outcome. Cutaneous, visceral, retroperitoneal, uterine, gastrointestinal, and major vessel leiomyosarcomas were excluded. By definition all lesions possessed at least focal cytologic atypia and mitotic activity, although the latter varied from <1/10 high power fields to 66/10 high power fields. The patients (21 females and 21 males) ranged in age from 26 to 86 years (mean 60 years); tumors developed in the lower (n = 28) or upper extremity (n = 11) and trunk (n = 3). Most arose in deep (n = 27) as opposed to superficial (n = 15) soft tissue; 39 arose from a small vein. During the follow-up period (mean 47 months, range 9-162 months), 3 of 38 (8%) patients developed local recurrence and 17 of 38 metastasized (45%) mostly to the lungs. In a univariate analysis age >62 years, size >4 cm, extensive necrosis, modified updated French Federation of Cancer Centers (FFCC) grade, and whether the tumor had been "disrupted" by a previous incisional biopsy or incomplete excision were significantly correlated with metastasis. AJCC stage also approached significance (p = 0.096) but could not be reliably tested because of the sparseness of the data. In multivariate analyses the logistic regression model that best predicted metastasis at 36 months incorporated the effects of age, FFCC grade, and disruption and had a sensitivity of 94.1% and a specificity of 95.2%. Disruption was the only significant risk factor for metastasis in a multivariate analysis (relative risk 2.70; p = 0.0001) but was strongly correlated with large size and deep location. Other parameters did not improve the predictive power of the model significantly. We concluded that the majority of SST leiomyosarcomas are actually of vascular origin, an observation that has clinical and possibly biologic ramifications. Our histologic definition of leiomyosarcoma to include atypia and any level of mitotic activity appears warranted by the biologic outcome in our cases. The risk of metastasis can be calculated from a model incorporating age, FFCC grade, and disruption. Because disruption correlates with size and depth, it could represent a surrogate as opposed to causal marker for metastasis. Nevertheless, in view of their vascular origin, the possibility that tumor disruption may facilitate or promote access to the bloodstream merits further study.

Adult↗

PubMed2000

Hepatic metastases from leiomyosarcoma: A single-center experience with 34 liver resections during a 15-year period.

OBJECTIVE: To describe a large single-center experience with hepatic resection for metastatic leiomyosarcoma. SUMMARY BACKGROUND DATA: Liver resection is the treatment of choice for hepatic metastases from colorectal carcinoma. In contrast, the role of liver resection for hepatic metastases from leiomyosarcoma has not been defined. METHODS: The records of 26 patients who between 1982 and 1996 underwent a total of 34 liver resections for hepatic metastases from leiomyosarcoma were reviewed. There were 23 first, 9 second, and 2 third liver resections. The records were analyzed with regard to survival and predictive factors. RESULTS: In the 23 first liver resections, there were 15 R0, 3 R1, and 5 R2 resections. Median survival was 32 months after R0 resection and 20.5 months after R1/2 resection. The 5-year survival rate was 13% for all patients and 20% after R0 resection. In 10 patients with extrahepatic tumor at the time of the first liver resection, 6 R0 and 4 R2 resections were achieved. After R0 resection, the median survival was 40 months (range 5-84 months), with a 5-year survival rate of 33%. After repeat liver resection, the median survival was 31 months (range 5-51 months); after R0 resection, median survival was 31 months and after R1/2 resection it was 28 months. There was no 5-year survivor in the overall group after repeat liver resection. CONCLUSIONS: Despite frequent tumor recurrence, the long-term outcome after liver resection for hepatic metastases from leiomyosarcoma is superior to that after chemotherapy and chemoembolization. Although survival after tumor debulking also seems to be more favorable than after nonoperative therapy, these data indicate that only an R0 resection offers the chance of long-term survival. The presence of extrahepatic tumor should not be considered a contraindication to liver resection if complete removal of all tumorous masses appears possible. In selected cases of intrahepatic tumor recurrence, even repeated liver resection might be worthwhile. In view of the poor results of chemoembolization and chemotherapy in hepatic metastases from leiomyosarcoma, liver resection should be attempted whenever possible.

Adult↗

PubMed2001

Specific but variable expression of h-caldesmon in leiomyosarcomas: an immunohistochemical reassessment of a novel myogenic marker.

h-Caldesmon is considered a novel specific marker for tumors with smooth muscle differentiation. To reassess its diagnostic use, the authors evaluated the immunohistochemical expression of h-caldesmon and other myogenic markers (calponin, alpha-smooth muscle actin, HHF35, and desmin) in 30 leiomyosarcomas (external soft tissues [15], retroperitoneum [8], uterus [5], other sites [2]), 26 myofibroblastic lesions, and 26 fibrohistiocytic tumors of varying biologic potential and histology. In contrast with previous data, h-caldesmon was expressed only in 11 (36%) of the 30 leiomyosarcomas analyzed, whereas they consistently expressed actins and frequently expressed calponin (86%) and desmin (76%). Leiomyosarcomas with the expression of h-caldesmon were well or moderately differentiated and primarily confined to the retroperitoneum or uterus. All but one leiomyosarcomas in the external soft tissues examined were negative for h-caldesmon, and the h-caldesmon-negative tumors showed moderately to poorly differentiated morphology. All myofibroblastic lesions examined were negative for h-caldesmon despite their constant expressions of at least one of the other markers. h-Caldesmon was not expressed in fibrohistiocytic tumors either, although focal positivity for the other markers was seen in subsets of the tumors. Thus, h-caldesmon can be regarded as a specific myogenic marker. However, one should be aware that the expression of h-caldesmon in leiomyosarcomas can be more variable according to their locations and/or extent of smooth muscle differentiation than considered previously.

Adult↗

PubMed2005

Management of superficial leiomyosarcoma: a retrospective study of 10 cases.

BACKGROUND: Superficial leiomyosarcoma is a rare soft-tissue tumor. Management of this tumor, including pathological evaluations and therapies, is not clearly defined in the existing literature. METHODS: Ten patients with superficial leiomyosarcoma treated in our institutes were recalled and scheduled for examination. Assessments were carried out according to clinical characteristics, therapies used, histological grade (based on the grading system of the Fédération Nationale des Centres de Lutte Contre Le Cancer), tumor-node-metastasis stage (using the American Joint Committee on Cancer staging system), recurrences rates, state of metastases, and the current condition of each patient. RESULTS: No local recurrences or distant metastases were seen in eight patients with low-grade and early-stage disease, whereas two patients with high-grade and advanced-stage disease had recurrence and one of these two patients died of the disease. CONCLUSIONS: This study indicates that evaluations using the Fédération Nationale des Centres de Lutte Contre Le Cancer grading system and the American Joint Committee on Cancer tumor staging system were useful. The authors advocate that surgical treatment of low-grade cutaneous leiomyosarcoma by complete excision with a narrow margin is adequate. For low-grade and early-state subcutaneous or soft-tissue leiomyosarcoma, wide excision with a minimum 2-cm lateral margin and one-tissue barrier deep margin is recommended. Management of high-grade leiomyosarcomas is still difficult; adequate wide excision may be the only option.

Adult↗

PubMedDate not supplied

Rapid growth of myxoid leiomyosarcoma of the vulva during pregnancy: a case report.

Smooth muscle tumors arising in the vulva are rare. Leiomyosarcoma is the most common variant of vulvar sarcoma, and very few cases have been reported during pregnancy. A 36-year-old woman presented with a progressively enlarging vulvar mass during pregnancy, diagnosed as a Bartholin's gland cyst. The lesion was resected at 38 weeks of gestation during cesarean section and diagnosis of myxoid leiomyosarcoma of the vulva was made. Six weeks later the patients were referred to our center and submitted to wide vulvar excision with groin lymph node dissection that revealed the presence of a small residual focus of leiomyosarcoma. At 30 months of follow-up the patient was well without any sign of recurrent disease. Leiomyosarcoma should be included in the differential diagnosis of vulvar masses; progressively enlarging vulvar lesion should be biopsied even during pregnancy. Leiomyosarcoma should be considered in the differential diagnosis of vulvar mass.

Adult↗

PubMedDate not supplied

The use of Palma's procedure in the salvage therapy for a leiomyosarcoma of the right pelvic sidewall: an intraoperative multidisciplinary approach.

Leiomyosarcomas of the ovary and broad ligament are relatively rare. Less than 50 cases of primary ovarian and broad ligament leiomyosarcomas have been reported. The prognosis is poor, with late-stage disease being a problem in the majority of cases. It is difficult to determine the exact role of surgery, chemotherapy, and radiotherapy in the management of these tumors. So far, no cases of ovarian or broad ligament leiomyosarcomas have been reported where primary surgery for the sarcoma had to be combined with a Palma's procedure. We report a case of a right pelvic sidewall leiomyosarcoma with involvement of 10 cm of the right external iliac vein. A review of the imaging preoperatively did not suggest involvement of the venous system. Resection of the pelvic mass was carried out and a Palma's procedure was performed by the vascular surgeon. Histology reported an incompletely excised high-grade leiomyosarcoma. Chemotherapy was given postoperatively. Imaging at 12 months after surgery showed a patent vascular graft and no evidence of recurrence. There was no clinical evidence of recurrence at 21 months postsurgery.

Combined Modality Therapy↗

PubMed1997

Expression of myogenic marker proteins in human leiomyosarcoma.

A series of formalin-fixed, paraffin-embedded leiomyosarcomas (n = 11) was studied immunohistochemically for their expression of various myogenic marker proteins. According to their predominant histological appearance, the tumors were classified as well (n = 4), moderately (n = 5), or poorly (n = 2) differentiated. Using monoclonal anti-muscle specific actin antibodies from clone HHF35 all examined tumors were positively stained. Desmin was not always found in leiomyosarcomas, since positive staining could be demonstrated only in eight cases. As revealed by staining with anti-vinculin antibodies from clone hVIN-1 using the APAAP technique, all leiomyosarcomas with the exception of one expressed vinculin. Typically, the vinculin immunoreactivity was detected diffusely throughout the majority of neoplastic cells as well as in vascular smooth muscle cells of blood vessels. Nine leiomyosarcomas displayed a positive staining for calponin, an actin-binding protein expressed in smooth muscle cells and their precursors. The distribution of calponin resembled that of vinculin in decorating myofibrils of nearly all tumor cells. Actinin immunoreactivity was present in tumor cells of all cases, but was expressed also in nontumor cells such as epithelia. These results suggest that the monoclonal antibodies against vinculin and calponin may serve as additional diagnostic markers for myogenic differentiation in leiomyosarcomas and related tumors.

Actins↗

PubMed1998

Intestinal leiomyosarcoma as the cause of severe hemoperitoneum.

Leiomyosarcomas represent 10-20% of malignant tumors in the small bowel and 1% of those located in the large bowel. These tumors have aspecific symptoms depending on their size, location and histology. Intestinal leiomyosarcomas may be the cause of numerous types of surgical emergency. We report 2 cases of intestinal leiomyosarcoma in the jejunum and transverse colon. Both were diagnosed during an emergency exploratory celiotomy. The clinical features on admission were similar to those of acute abdomen. The main cause of surgical emergency was an extremely rare complication of gastrointestinal leiomyosarcoma: a severe hemoperitoneum. We briefly describe the surgical emergencies caused by intestinal leiomyosarcomas as well as the surgical methods employed in these cases.

Colonic Neoplasms↗