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Primary pulmonary artery leiomyosarcoma.

Primary pulmonary artery sarcomas are uncommon neoplasms with up to 20% of cases being leiomyosarcomas. In many instances, the diagnosis is difficult and delayed. We report the case of a 75-year-old male who presented with symptoms suggestive of recurrent pulmonary thromboemboli, in which a primary leiomyosarcoma of the pulmonary artery was successfully removed surgically. Three years after surgery, the patient is well and with no evidence of recurrence or metastatic disease.

Aged↗
PubMed2003

MMP-1 and MMP-2 expression in uterine leiomyosarcoma and correlation with different clinicopathologic parameters.

OBJECTIVE: Matrix metalloproteinases (MMPs) have been suggested to play an important role in tumor invasion and metastasis because they degrade a wide range of components of the extracellular matrix. In the present study, we analyzed the expression of MMP-1 and MMP-2 proteins in patients with uterine leiomyosarcoma. METHODS: MMP-1 and MMP-2 expression was investigated by immunohistochemistry from paraffin-embedded tissue sections in 21 patients with uterine leiomyosarcoma (LMS). The immunohistochemical findings were correlated with different clinicopathologic characteristics of the patients. RESULTS: MMP-1 was expressed in 86% and MMP-2 was expressed in 48% of uterine LMS. There was a statistically significant positive correlation between vascular space involvement and MMP-2 expression (P =.05) and between age and MMP-2 expression, with patients over 50 years old having significantly more frequent MMP-2-positive tumors than patients younger than 50 years (P =.006). The relationship between MMP-2 expression and tumor stage and recurrence disease did not reach statistical significance. A trend towards prolonged disease-free survival was observed in women with MMP-2-negative LMS compared with patients with MMP-2-positive LMS (P =.09). Furthermore, a univariate analysis revealed that early tumor stage (P =.0001), age at diagnosis less than 50 years (P =.02), and the absence of vascular space involvement (P =.04) were associated with longer overall survival. CONCLUSION: The statistically significant positive correlation between MMP-2 expression and vascular space involvement as well as the prolonged disease-free survival rate in patients with MMP-2 negative uterine LMS suggest that MMP-2 plays an important role in tumor invasion and metastasis. Further clinical studies with larger numbers of cases need to be performed to verify these findings.

Adult↗
PubMed1995

Primary leiomyosarcoma of the maxilla with regional lymph node metastasis. Report of a case and review of the literature.

A rare case of oral leiomyosarcoma diagnosed with the aid of immunohistochemical and electron microscopic examinations together with a review of the literature are reported. The patient was a 70-year-old Japanese man. The primary tumor involved the maxillary gingiva and bone and metastasized to the cervical lymph nodes. On histologic examination the tumor showed invasive growth into the maxillary bone. It was composed of interlacing fascicles of spindle-shaped cells with eosinophilic cytoplasm and elongated, blunt-ended nuclei. The tumor formed extensive metastatic foci in the cervical lymph nodes. On immunohistochemical examination most of the tumor cells were positive for desmin, smooth muscle-specific actin, and myosin. The ultrastructural characteristics of the tumor cells were abundant microfilaments, pinocytotic vesicles, and basement membrane formation. The findings were indicative of a tumor demonstrating myogenic differentiation. A review of the literature during the past 50 years disclosed a total of 60 oral leiomyosarcomas, including our case.

Actin Cytoskeleton↗
PubMed2003

[Epstein-Barr virus associated central nervous system leiomyosarcoma occurring after renal transplantation: case report and review of the literature].

Central nervous system leiomyosarcomas are extremely rare, however, they became more frequent among immunodeficient patients, either in a patients infected with human immunodeficiency virus (HIV), or after organ transplantation. The data of the literature indicate that the infection by Epstein-Barr virus (EBV) plays a causal role in the development of these tumours but its precise role in the oncogenesis remains unresolved. We report a new case of EBV associated leiomyosarcoma of the left cavernous sinus occurring after renal transplantation. The epidemiological, clinical, pathological and therapeutic characteristics of these tumours are discussed.

Adult↗
PubMed2000

Primary intraoral leiomyosarcoma of the tongue: an immunohistochemical study and review of the literature.

Leiomyosarcoma is a relatively uncommon mesenchymal tumor that exhibits smooth-muscle differentiation. We report a new case of leiomyosarcoma involving the tongue of a 67-year-old male. Histologically, the tumor was composed of variably oriented fascicles of spindle-shaped cells with 'cigar-shaped' nuclei and eosinophilic cytoplasm, containing occasional PAS-positive granules. Atypical mitotic figures and necrotic foci were frequently detected. Consistent desmin, alpha-smooth muscle-specific and sarcomeric actin, and vimentin immunoreactivity was demonstrated in the tumor cells, whereas cytokeratins, CD 30, CD 31, CD 34, CD 45, CD 68, EMA, GFAP, HMB 45 and S-100 protein were negative. The patient underwent wide surgical excision of the tumor and is alive and disease-free at a 5-year follow-up. This report emphasizes the difficulties in the differential diagnosis of these uncommon tumors in an intra-oral location.

Aged↗
PubMed1978

Leiomyosarcoma of paranasal sinuses.

A leiomyosarcoma of the upper respiratory and food passages is a rarity. Two rare cases of leiomyosarcoma of the paranasal sinuses, of which one developed recurrence are reported.

Adult↗
PubMed1986

Leiomyosarcoma of the trachea.

A case of primary leiomyosarcoma in the trachea of a 63-year-old woman is presented. Owing to the rare occurrence of this tumour in this site, the clinical diagnosis and pathological classification are difficult. In the English literature, only six cases of primary leiomyosarcoma in the trachea are on record.

Female↗
PubMed2001

Childhood leiomyosarcoma: a report from the soft tissue sarcoma Italian Cooperative Group.

BACKGROUND: Only a few reports on the clinical features and management of childhood leiomyosarcoma are available. To contribute additional information on the management of this rare tumor, we report on a series of 16 pediatric patients treated from 1982 to 1998 by the Soft Tissue Sarcoma Italian Cooperative Group. PATIENTS AND METHODS: Primary surgery was conservative in all but two patients, and consisted of biopsy--three cases, non-radical excision--four, and radical resection--nine (involving a primary re-excision in 4 of 9). In two cases secondary radical surgery was performed after primary chemotherapy. Chemotherapy was administered to 9 of 16 patients, radiotherapy to three. RESULTS: After a median follow-up of seven years (range 3-18), the five-year event-free survival (EFS) and overall survival were 56.3% and 72.9%, respectively; 12 of 16 patients were alive (nine of them in continuos complete remission). Univariate analysis was performed to compare EFS according to different subgroups: size represented the most significant prognostic factor. CONCLUSIONS: Complete surgical resection is the mainstay of treatment for leiomyosarcoma. The role of both adjuvant chemotherapy and radiotherapy has yet to be established, and awaits cooperative multicentric studies.

Adolescent↗
PubMed2003

Leiomyosarcoma of the uterus: a clinicopathologic study of 21 cases.

BACKGROUND: The purpose of the present study was to identify the prognostic factors of and to determine the most appropriate mode of treatment for uterine leiomyosarcoma. METHODS: We reviewed the hospital records, including surgical notes and pathologic reports, of 21 patients with uterine leiomyosarcoma treated at Chang Gung Memorial Hospital, Kaohsiung, Taiwan, between 1987 and 1997. Univariate analysis was performed using the log-rank test. Cox regression was used to identify independent prognostic factors. RESULTS: The mean follow-up time was 30 months, and the 5-year disease-free survival rate was 55%. Evaluating the correlation between clinicopathologic parameters and survival, early stage (p = 0.0002), tumor cells without necrosis (p = 0.0026), low-grade tumor (p = 0.015), absence of vascular space involvement (p = 0.006), and tumor without atypia (p = 0.016) were associated with good prognosis. However, in a multivariate analysis using the Cox model, only advanced stage (p = 0.032) and tumor necrosis (p = 0.032) were found to be independent poor prognostic factors. The 5-year disease-free survival was only 21% in patients with the presence of any one or both of these two factors. Five of 11 patients in this group had received aggressive adjuvant therapy after surgery, and none of them survived more than 19 months. CONCLUSIONS: We found that patients with advanced stage or presence of tumor necrosis had an extremely poor prognosis. Adjuvant therapy seemed to play a limited role, and provided no survival benefit. Treatment for these patients should be palliative until effective therapeutic modalities prove otherwise.

Adult↗
PubMed1986

Accumulation of globotriaosylceramide in a case of leiomyosarcoma.

Analysis of the glycosphingolipid composition in one case of uterine leiomyosarcoma metastasized to the liver showed an accumulation of globotriaosylceramide as compared with normal liver and uterus from which the tumour originated. The structure and the amount of glycosphingolipids were established by using specific glycosidases, permethylation analysis and h.p.l.c. The reason for the accumulation of globotriaosylceramide in leiomyosarcoma remains to be answered.

Chromatography, High Pressure Liquid↗
PubMed2003

Leiomyosarcoma of the kidney in a dog.

A leiomyosarcoma that occurred in the right kidney of a dog (7-year-old female Labrador retriever) was studied pathologically. On histological examination, tumour cells were spindle-shaped, the cell density was high, and complex fasciculated tumour cells showing longitudinal and transverse cross-sections were observed. Both ends of the nuclei were rounded, the nucleoli were clear and many images of nuclear division were observed. Immunohistochemically, tumour cells reacted intensely with anti-vimentin antibody and anti-actin smooth muscle antibody, and mildly with anti-desmin antibody. On electron microscopy, the nuclei of tumour cells were rounded at both ends and elongated elliptically, and a large number of microfibrils with focal density and dense patches were observed in the cytoplasm. This is a case report of a very rare leiomyosarcoma of the dog kidney.

Animals↗
PubMed1999

Leiomyosarcoma of the rectum: what role does adjuvant therapy play?

Leiomyosarcoma of the rectum is an exceedingly rare malignancy and for this reason the literature fails to provide definitive management guidelines with regard to the place of adjuvant therapies. The role of radiotherapy (RT) is often downplayed on the basis of articles written at a time when state-of-the art RT equipment was unavailable. A case of leiomyosarcoma of the rectum is presented and the literature is reviewed. Because the rarity of this tumour type virtually precludes a prospective randomized trial of adjuvant therapies, the authors recommend (in otherwise fit patients) postoperative pelvic RT because its morbidity is minimal. Smaller tumours may benefit to a greater extent than those lesions that are large at presentation and thereby run a worse clinical course.

Humans↗
PubMed2001

Primary leiomyosarcoma of inferior vena cava, a rare entity: Imaging features.

Leiomyosarcoma of the inferior vena cava (IVC) is an uncommon neoplasm, most frequently seen in the sixth decade with a female predominance. Imaging modalities allow an early and accurate preoperative diagnosis resulting in a higher rate of surgical resection and improved survival. Imaging findings in a 65-year-old woman with leiomyosarcoma of IVC are described. Computed tomography and MRI typically showed a non-fatty, necrotic intraluminal IVC mass with extension to retroperitoneal compartments. Cavography was useful to evaluate the collateral circulation associated with the extensive intraluminal mass. The lesion was biopsied under ultrasound guidance.

Aged↗
PubMed2000

Primary leiomyosarcoma of the cervical spine causing spontaneous compression fracture: report of an autopsy case.

An extremely rare case of the primary leiomyosarcoma of the cervical spine presenting as a compression fracture is reported. A 69-year-old man complained of dysesthesia of both hands. A plain radiograph, computed tomography and magnetic resonance imaging of the cervical spine showed a compression fracture of the vertebral body at C7 without the formation of an obvious mass. From radiological findings, the fracture was considered to be caused by an infiltrative vertebral bone tumor. Biopsy of the tumor revealed a leiomyosarcoma, a metastatic tumor being ruled out by a systemic investigation. The patient died of pleural metastasis 6 months later, and an autopsy confirmed the primary site of the tumor as the cervical spine.

Aged↗
PubMed2000

Primary hepatic leiomyosarcoma in a patient with hepatitis C virus-related liver cirrhosis.

We describe an autopsy case of primary hepatic leiomyosarcoma in a 68-year-old man with hepatitis C virus-related liver cirrhosis. The patient, who had a history of acute hepatitis 20 years previously, died of a ruptured hepatic tumor. At autopsy, a well-circumscribed 14 x 16 x 15 cm tumor replaced the medial site of the right hepatic lobe with multiple intrahepatic and distant metastases. Histologically the tumor, which had extensive central necrosis, consisted predominantly of well or moderately differentiated spindle-shaped cells, which were positive for smooth muscle actin and vimentin on immunohistochemical staining. In addition, clusters of markedly atypical cells and myxoid change of the matrix were discretely found in the focal and small areas of the tumor. These findings indicated that many sections were necessary for the histologically accurate estimation of primary hepatic smooth muscle tumor. The histological examination of a non-tumorous lesion showed liver cirrhosis. Hepatitis C virus was detected in the cytoplasm of cirrhotic hepatocytes by immunohistochemistry and reverse transcriptase-polymerase chain reaction, but not in the tumor cells. This suggested that the virus was not directly involved in the development of primary hepatic leiomyosarcoma.

Actins↗
PubMed2003

Unicentric Castleman's disease with leiomyosarcoma: a rare association.

A 54-year-old asymptomatic woman was first diagnosed with unicentric Castleman's disease at 51 years of age. The disease was identified incidentally behind the inferior vena cava after a computed tomography (CT) scan. She was followed conservatively and no enlargement of the mass was observed. However, a follow-up CT scan revealed another solid mass in the left retroperitoneal area. Both tumors were surgically removed in the same session. The retrocaval mass was histologically confirmed to be Castleman's disease of the hyaline vascular type. Histological examination of the left peritoneal mass revealed low-grade leiomyosarcoma. We present here a case of leiomyosarcoma associated with unicentric Castleman's disease, both of which were localized in the retroperitoneum.

Castleman Disease↗
PubMed2005

A uterine leiomyosarcoma that became positive for HMB45 in the metastasis.

Uterine smooth muscle tumors are usually spindle cell lesions, but a minority is composed of epithelioid cells. Foci of clear cells can be found in these latter tumors. Recently, it has been shown that some of these tumors can be positive for HMB45, and some authors have advocated calling these lesions perivascular epithelioid cell (PEC) tumors or PEComas. The case we describe here clearly shows that the so called PEC is just a smooth muscle cell capable of changing its immunophenotype. The patient involved is a 29-year-old black woman who was found to have an epithelioid leiomyosarcoma of the uterus in November 1995. She was treated with a simple hysterectomy and bilateral salpingo-oophorectomy. A metastatic lesion was found in her liver. She, therefore, also received chemotherapy and was free of disease until October 2002, when a recurrent tumor was detected in her spine. After undergoing resection of the lesion at 2 different times, in 2002 and 2003, the patient was treated with radiotherapy and is currently receiving chemotherapy. On microscopic examination, the tumor in the uterus and liver both proved to be an epithelioid leiomyosarcomas that was diffusely positive for smooth muscle actin. Approximately 15% of the cells had clear cytoplasm, but sections from 2 different blocks were completely negative for HMB45. However, although the tumors resected from the spine in 2002 and 2003 showed features similar to those of the uterine neoplasm, but with a lower percentage of cells positive for smooth muscle actin and more clear cells, several of the clear cells were positive for HMB45. To the best of our knowledge, this is the first case of a uterine smooth muscle cell tumor that became positive for HMB45 when it metastasized.

Actins↗