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PubMed2002

Thoracic paravertebral leiomyosarcoma: rare but it does occur.

STUDY DESIGN: Marginal resection of a paraspinal tumor in the thoracic vertebra was performed. OBJECTIVE: To document a very rare pathology for a paraspinal tumor. SUMMARY OF BACKGROUND DATA: Primary leiomyosarcoma of the spine or paravertebral space is extremely rare. A case of a patient who was operated on for a mass in the spinal canal and whose pathology was reported to be leiomyosarcoma is presented. METHODS: Marginal resection of the paravertebral mass was performed. RESULTS: The pathology of the tumor was reported as leiomyosarcoma. CONCLUSION: Leiomyosarcomas may develop at any site where smooth muscle cells are present. However, primary leiomyosarcoma of the spine or paravertebral space is extremely rare. Leiomyosarcoma, although rare, should be kept in mind as one of the possible diagnoses when a patient with a paraspinal tumor is presented.

Actins↗

PubMed2007

HMB-45 reactivity in conventional uterine leiomyosarcomas.

We studied the human melanoma black-45 (HMB-45) reactivity in 25 uterine leiomyosarcomas including 23 conventional and 2 myxoid variants. Eleven tumors were poorly differentiated, and 14 were well to moderately differentiated. Nine uterine leiomyosarcomas labeled with HMB-45 in 10% or less of the tumor cells. Six were poorly differentiated and 3 were well differentiated. Our study indicates that 36% of conventional leiomyosarcomas focally express HMB-45. HMB-45 reactivity was more common in the poorly differentiated than in the well-differentiated group of leiomyosarcomas. In light of our findings and of those recently reported in the literature, we believe that the term PEComa should not be used for uterine leiomyosarcomas with clear cells or for conventional leiomyosarcomas that stain positively with HMB-45.

Adult↗

PubMedDate not supplied

Metastatic uterine leiomyosarcoma coexisting with papillary carcinoma of the thyroid gland.

Metastatic leiomyosarcoma to the thyroid gland has rarely been described. We report a 54-year-old postmenopausal woman with uterine leiomyosarcoma, who presented with a single "cold" nodule in the right thyroid lobe 3 months after hysterectomy. The lesion was identified as a papillary thyroid carcinoma. In a separate area of the thyroid, a 1.2-mm area of a malignant mesenchymal neoplasm with morphologic and immunohistochemical features of leiomyosarcoma existed. Seven months after thyroidectomy the patient developed micronodular lung disease visible on successive chest computed tomography scans, consistent with metastatic disease from the primary uterine leiomyosarcoma that showed very good and prolonged response to chemotherapy. The thyroid papillary carcinoma was likely the recipient of an early and possibly the first metastasis of the patient's uterine leiomyosarcoma. This is the first report of metastatic leiomyosarcoma coexisting with a primary thyroid carcinoma and supports the possibility of a common pathway connecting thyroid gland neoplasms and sarcomas.

Biopsy, Needle↗

PubMed1991

Intermediate and fine cytofilaments in cutaneous and subcutaneous leiomyosarcomas.

The expression of fine and intermediate cytofilaments in 10 cutaneous and seven subcutaneous leiomyosarcomas was studied immunohistochemically. All the tumors contained tumor cells which showed a positive immunoreactivity for desmin in formaldehyde-fixed and paraffin-embedded sections, but none of the seven anti-desmin antibodies used alone produced a distinct positive staining in all the tumors. A lack of correspondence in terms of immunoreactivity between tumor cells and the supposed muscle of origin was observed, especially in the subcutaneous leiomyosarcomas. In all cases, antibodies to muscle-specific and smooth muscle-specific actin were found to produce a positive staining in both the tumors and the supposed muscle of origin. Vimentin was detected in 8/10 cutaneous and 4/7 subcutaneous leiomyosarcomas, while the supposed muscle of origin was positive in 3/10 and 7/7 cases, respectively. Four of the cutaneous and three of the subcutaneous leiomyosarcomas contained tumor cells which stained positively for cytokeratins, while the supposed muscle of origin showed no positivity. It thus appears that a phenotypic shift in terms of vimentin and cytokeratin expression occurs in the tumor cells of cutaneous and subcutaneous leiomyosarcomas compared with the supposed muscle of origin. It is recommended that more than one monoclonal anti-desmin antibody is used to characterize these tumor entities. It is also concluded that the immunoreactivity for muscle-specific actins in superficial leiomyosarcomas is more constant, although less specific, than that of desmin and that the demonstration of the simultaneous expression of muscle-specific actins and desmin is helpful.

Actin Cytoskeleton↗

PubMed1996

Immunohistochemistry (Ki-67 and p53) as a tool in determining malignancy in smooth muscle neoplasms (exemplified by a myxoid leiomyosarcoma of the uterus).

Smooth muscle tumours of the uterus at times represent a problem as it may be difficult to distinguish between benign and malignant tumours. The myxoid leiomyosarcoma (a rare type of neoplasm) reported here is an example of this. We present a case history and examine the suitability of Ki-67 and p53 as indicators of malignancy. The two antibodies were tested on seven leiomyomas, three atypical (borderline) leiomyomas, seven leiomyosarcomas and the myxoid leiomyosarcoma using microwave oven antigen retrieval. The leiomyomas had the lowest and the leiomyosarcomas the highest proliferation rate. The leiomyomas had no expression of p53, the atypical leiomyomas had a few scattered positive nuclei, and 5/7 of the leiomyosarcomas had overexpression of p53. The myxoid leiomyosarcoma had a positive reaction for p53 in clusters. The results suggest that Ki-67 and p53 might be useful as indicators of malignancy.

Biomarkers, Tumor↗

PubMed2004

c-Kit expression in patients with uterine leiomyosarcomas: a potential alternative therapeutic treatment.

PURPOSE: Uterine leiomyosarcomas are rare tumors characterized by their resistance to chemotherapy and radiation treatment. Surgery is the primary method of treatment, but for patients with unresectable disease, alternate therapeutic options are clearly warranted. According to initial observations of c-KIT expression, correlation with a bad prognosis, and the successful therapeutic possibility of STI571 in gastrointestinal stromal tumors, the data have encouraged us to study c-KIT expression in these tumors. EXPERIMENTAL DESIGN: We analyzed the expression of c-KIT and genetic assessment of exon 11 of c-kit gene in 32 uterine leiomyosarcomas. RESULTS: In 17 cases (53.1%), we observed a c-KIT expression in tumor cells. Of the 17 patients with distinct c-KIT-positive immunoreactivity, eight had I or II stage disease and nine had III or IV stage disease. Molecular genetic analysis of exon 11, analyzed by direct DNA sequencing, was performed for all of the c-KIT-positive uterine leiomyosarcomas. No mutations were found. CONCLUSION: The conventional chemotherapy in leiomyosarcomas appears to be ineffective for patients with metastatic or unresectable disease, and the management of these patients poses a special problem. In these women, new therapeutic strategies are warranted. The treatment with STI571 in leiomyosarcoma patients might be hypothesized, because uterine leiomyosarcomas also express c-KIT.

Adult↗

PubMed2006

Oncogenic property of acrogranin in human uterine leiomyosarcoma: direct evidence of genetic contribution in in vivo tumorigenesis.

To identify potential oncogenes that contribute to the development of uterine leiomyosarcoma, we conducted a cDNA microarray analysis between normal uterine smooth muscle and uterine leiomyosarcoma. We found that acrogranin (also named PCDGF or progranulin) is overexpressed in uterine leiomyosarcoma. With immunohistochemical staining of 12 leiomyosarcoma cases, we verified acrogranin expression in tumor cells. Furthermore, the intensity of acrogranin expression correlated with high histologic grade and poor prognosis. To directly analyze the oncogenic properties of acrogranin, we established an immortalized uterine smooth muscle cell line by transfection of human telomerase reverse transcriptase into primary culture. This cell line retained the original characteristics of uterine smooth muscle cells, including spindle-shaped extension as well as expression of vimentin, estrogen receptor alpha, progesterone receptor, and alpha smooth muscle actin. Transfection of acrogranin into the immortalized uterine smooth muscle cells resulted in colony formation in soft agar, but the diameter of the colonies did not exceed 100 mum. Transfection of both acrogranin and SV40 early region (SV40ER) into the immortalized uterine smooth muscle cells resulted in an increased number of colonies and increased colony size in soft agar versus transfection of SV40ER alone. We show that only immortalized uterine smooth muscle cells expressing both acrogranin and SV40ER are capable of tumor formation in nude mice. Thus, acrogranin is overexpressed in uterine leiomyosarcoma cells, particularly in high-grade cases, and forced expression of acrogranin in immortalized uterine smooth muscle cells contributes to malignant transformation, which suggest that acrogranin plays an important role in the pathogenesis of uterine leiomyosarcoma.

Adult↗

PubMedDate not supplied

Oral leiomyosarcoma in childhood. Report of a case with fine needle aspiration cytology.

BACKGROUND: Leiomyosarcomas are rare tumors in the pediatric age group, and occurrence of this neoplasm in the oral cavity is exceedingly rare. This article highlights the fine needle aspiration (FNA) cytology diagnosis of a case of recurrent oral leiomyosarcoma in childhood. CASE: An 11-year-old male noticed a swelling in the oral cavity near the left lower jaw. It was excised and diagnosed as leiomyosarcoma on histopathology. Four months later the patient presented with a progressive swelling in the oral cavity that extended to the lower jaw. The recurrent swelling was subjected to FNA, and its cytologic features were consistent with leiomyosarcoma. There was a very good initial response to chemotherapy and radiation therapy. However, because of noncompliance with advice for further therapy, the patient had a second local recurrence and dissemination of the disease to the skeletal system, abdomen and thorax. FNA cytology diagnosis of the second locally recurrent lesion and abdominal mass were consistent with leiomyosarcoma. Immunocytochemical staining revealed a positive reaction in the cytoplasm of tumor cells for vimentin and desmin in the FNA smear and paraffin section, respectively. CONCLUSION: Fine needle aspiration cytology is a useful technique for detection of recurrence and metastasis during follow-up of childhood oral leiomyosarcoma.

Biopsy, Needle↗

PubMed2003

Mineralization in musculoskeletal leiomyosarcoma: radiologic-pathologic correlation.

OBJECTIVE: Mineralization in leiomyosarcoma, a malignant tumor of smooth muscle, has not been widely recognized. In this article, we report our experience with four cases of primary leiomyosarcoma of soft tissue or bone in which mineralization was visible on either radiography or CT. In none of the cases was the diagnosis of leiomyosarcoma considered before biopsy. In one case of a soft-tissue leiomyosarcoma, the presence of mineralization was a factor that led to the misinterpretation of the needle biopsy specimen as soft-tissue osteosarcoma. CONCLUSION: Histologically, mineralization in leiomyosarcoma appears to be caused by either nonneoplastic ossification or dystrophic mineralization in the tumor. This feature can cause leiomyosarcomas to be confused with other neoplasms.

Adult↗

PubMed2001

A high-grade primary leiomyosarcoma of the bladder in a survivor of retinoblastoma.

Second nonocular malignancies develop with increased incidence in patients with hereditary retinoblastoma. Osteosarcoma is by far the most common type with an incidence of up to 50%, followed by soft tissue sarcomas. Visceral leiomyosarcoma is extremely rare and only 2 cases have been reported in the past 2 decades, one in the liver and another one in the urinary bladder, both of which developed after cyclophosphamide therapy. Here we report a case of vesical leiomyosarcoma that was diagnosed in a 49-year-old woman 47 years after the diagnosis of a hereditary retinoblastoma. The patient's retinoblastoma was treated with unilateral enucleation without adjuvant radiation or chemotherapy. We believe that this is the first report of vesical leiomyosarcoma occurring in a patient with retinoblastoma without a prior history of radiation or chemotherapy. This report is significant not only because of the rarity of vesical leiomyosarcoma as a second nonocular tumor in retinoblastoma patients, but also because of the infrequency of vesical leiomyosarcoma in general. We also investigated the potential molecular pathogenesis of the leiomyosarcoma.

Actins↗

PubMed1991

[Primary cutaneous leiomyosarcoma].

Primary leiomyosarcomas of the superficial soft tissues are very rare neoplasms. Because of the different biological behaviour, they should be divided into cutaneous and subcutaneous tumours. Differential diagnosis includes leiomyomas, skin metastases from extracutaneous leiomyosarcomas, and other spindle-cell tumours of the skin. Ultrastructural and immunohistochemical investigations are useful when light microscopic features do not allow an unequivocal diagnosis. Although local recurrences develop in almost half the patients with cutaneous leiomyosarcomas, the prognosis is predominantly good. In contrast, metastatic spread is more common in patients with subcutaneous leiomyosarcomas. Treatment for superficial leiomyosarcomas consists in wide excision. We report on a 76-year-old women with a local recurrence of a primary cutaneous leiomyosarcoma.

Aged↗

PubMed1981

Leiomyosarcoma of the soft tissue. A correlative cytological and histological study of 11 cases.

Leiomyosarcoma of the soft tissues is a well-defined and characteristic entity histologically but correlative cytomorphological studies are lacking. A correlative histological and cytological study of 11 cases is presented. The leiomyosarcomas were characterized histologically by elongated tumour cells arranged in bundles intersecting each other at wide angles. The nuclei were elongated and often blunt-ended, tending to be aligned in tandem-position or in rows. The smears from leiomyosarcoma were poor in tumour cells and most cells were arranged in clusters of strands. The characteristic blunt-ended cells and nuclei of leiomyosarcoma could be identified in the smears and the nuclei were focally arranged in rows and sometimes in parallel. Nuclear atypia permits diagnosis of malignancy, although the atypia was generally not very pronounced. We consider that a knowledge of the corresponding histological features is mandatory for differentiating cytologically between various soft tissue sarcomas, including leiomyosarcomas. Further correlative cytological and histological studies of other soft tissue sarcomas are necessary for a more valid interpretation of the smears from leiomyosarcomas.

Adult↗

PubMed1996

Leiomyosarcoma of the rectum: a series of twelve cases.

BACKGROUND: Leiomyosarcoma of the rectum is a rare disease without well-documented report on its management and prognosis in Taiwan. To evaluate the effectiveness and prognosis of different treatments for rectal leiomyosarcoma, we reviewed a series of 12 patients with rectal leiomyosarcoma treated at our hospital. METHODS: This study is a retrospective review of 12 rectal leiomyosarcomas treated at VGH-Taipei from 1976 to 1995, with emphasis on symptoms, therapeutic procedures and prognosis. RESULTS: The majority of tumors occurred in men (n = 10) and the average age at presentation was 54.4 years. Bloody stool and perianal pain were the two most common symptoms. Nine tumors were found in the lower rectum, within 4 cm above the dentate line. Five patients received abdominoperineal resection, four received wide local excision, and three had diversion colostomy. The effect of adjuvant therapy, either chemo- or radiotherapy, was poor. The overall one-year and five-year survival rates were 83% and 46%, respectively. Liver was the principal metastatic site. The local recurrence rate was much higher in patients receiving wide local excision than in those receiving abdominoperineal resection (100% vs. 20%). CONCLUSIONS: Abdominoperineal resection is an appropriate choice of treatment for patients with operable rectal leiomyosarcoma due to its lower local recurrence rate. Wide local excision could be considered when the lesion is less than 2 cm in diameter or when patients are not suitable for radical surgery. The effect of adjuvant therapy, either radiotherapy or chemotherapy, is poor for rectal leiomyosarcoma.

Adult↗

PubMed1997

Leiomyosarcoma metastatic to the orbit: diagnosis of fine-needle aspiration.

A 47-yr-old man with history of metastatic low-grade rectal leiomyosarcoma presented with progressive protrusion of his left eye due to an enlarging orbital mass. The differential diagnosis included tumor metastasis or orbital infection due to an unknown infectious agent. Diagnostic fine-needle aspiration (FNA) of the orbit was performed on an urgent basis to institute proper therapy and to save the patient's eyesight. Cytomorphologic examination of the material demonstrated a spindle-cell neoplasm consistent with metastatic leiomyosarcoma. It is a rare event for leiomyosarcoma to occur in the orbit. On our review of the literature, the cytology of primary orbital leiomyosarcoma on FNA has only been reported once. To our knowledge, this is the first report of the FNA cytomorphology of metastatic leiomyosarcoma to the orbit.

Biopsy, Needle↗

PubMed1981

Ten-year survival after synchronous resection of liver metastasis from intestinal leiomyosarcoma.

This paper reports on a case of intestinal leiomyosarcoma with metastases to the liver, peritoneum, and omentum, which has been successfully treated with four operations. The initial operation was performed for a primary intestinal leiomyosarcoma and intraperitoneal seeding. Three months later "shelling out" of a liver metastasis was carried out. The patient has subsequently been operated upon twice for abdominal recurrence at six years and nine months, and nine years and six months, respectively. The patient is free from symptoms and is working now, ten years after resection of metastasic leiomyosarcoma of the liver. A review of the literatures has revealed that our patient is the first ten-year survivor after resection of hepatic metastasis of leiomyosarcoma. An aggressive surgery is acceptable in selected patients who have spreading leiomyosarcoma of the bowel.

Follow-Up Studies↗

PubMed1995

Leiomyosarcoma of the prostate. Report of 23 cases.

BACKGROUND: Leiomyosarcoma of the prostate is a rare neoplasm that accounts for less than 0.1% of prostate malignancies. Previous reports of this neoplasm consisted of single case studies or small series, often combined with cases of rhabdomyosarcoma. The relationship of prognosis with histologic and immunohistochemical findings has not, to the authors' knowledge, been described in a large series of cases, and the efficacy of various treatments is uncertain. METHODS: The authors undertook a clinicopathologic study of all cases of prostate leiomyosarcoma observed at their institution from 1929 to 1994. Twenty-three cases were retrieved from the files of the Department of Pathology, Mayo Clinic (Rochester, MN), and clinical follow-up was available for 14. Immunohistochemical studies, including actin, desmin, S-100 protein, keratin, and vimentin were performed for 18 cases. RESULTS: Patients ranged in age from 41 to 78 years, with a mean of 61 years. Presenting symptoms included urinary obstruction (100%), perineal pain (25%), burning on ejaculation (7%), and weight loss (7%). The neoplasms ranged from 3.3 to 21 cm (mean, 9 cm) in greatest dimension and were often associated with necrosis. Seven tumors were Grade 2, 10 Grade 3 and 6 Grade 4 (Broders' grading system; scale, 1-4). Mitotic figure counts varied from 2 to 24 per 10 high power fields. Fifteen of 15 (100%) cases were immunoreactive for vimentin, 10 of 16 (63%) were immunoreactive for actin, and 3 of 15 (20%) were weakly reactive for desmin. Keratin expression was observed in 4 of 15 cases (27%), and S-100 protein was negative in all cases. Treatment varied, and usually included a combination of radiation therapy, chemotherapy, and radical prostatectomy or cystoprostatecomy. Follow-up ranged from 2 to 72 months, with a mean of 19 months. Ten patients died from tumor 3 to 72 months (mean, 22 months) after diagnosis. Four patients were alive, including three with residual tumor and one without evidence of tumor at 2, 4, 30, and 4.5 months, respectively. Local recurrence occurred in 10 of 11 patients, including 5 who had gross residual tumor present after surgery. Metastases developed up to 40 months after surgery (mean, 10.3 months), and most frequently involved the lungs. CONCLUSIONS: These findings indicate that prostate leiomyosarcoma has a varied histologic appearance ranging from spindled cell neoplasm reminiscent of smooth muscle to pleomorphic sarcoma. Epithelioid features may be present. Most tumors are immunoreactive with antibodies to vimentin and actin, and reactivity with antikeratin antibodies does not exclude the diagnosis of leiomyosarcoma. Prostate leiomyosarcoma has a poor prognosis, although the length of survival is variable. Radical surgery was the treatment of choice in the current series, but complete excision was difficult in most cases and did not result in cure.

Adult↗

PubMed1994

Fine-needle aspiration cytology of hepatic leiomyosarcoma.

Hepatic leiomyosarcoma is a rare malignancy to involve the liver, occurring as a primary liver sarcoma in patients without an underlying medical disorder, as a metastatic malignancy, and with increasing incidence, as a primary tumor in AIDS patients. A series of hepatic leiomyosarcomas diagnosed by FNA biopsy, including the first reported case in an adult AIDS patient, were reviewed with respect to cytomorphologic features. The series consisted of five men and two women ranging from 24 to 72 years of age. One case was a primary hepatic lesion in a 24-yr-old man with AIDS and six were metastatic from a number of sites (stomach, small bowel, retroperitoneum, vena cava, and seminal vesicle). Two cytologic patterns were identified. Aspirates of spindle-cell leiomyosarcomas (six cases) generally produced hypocellular smears composed primarily of small irregular clusters of cells with blunt-ended, uniform, overlapping nuclei. The differential diagnosis included benign reactive processes and other benign and malignant spindle-cell neoplasms. The aspirate of an epithelioid leiomyosarcoma (one case) revealed a second pattern characterized by highly cellular smears with many single polygonal cells having eccentric, malignant nuclei and a characteristic clear quality to the cytoplasm in Papanicolaou-stained material. This epithelial appearance generated a differential diagnosis that included hepatocellular carcinoma, metastatic carcinoma, and melanoma. Careful integration of clinical information, cytomorphologic features, and ancillary studies will allow specific FNA diagnosis of hepatic leiomyosarcoma in most cases.

Acquired Immunodeficiency Syndrome↗

PubMed2001

Genomic alterations in uterine leiomyosarcomas: potential markers for clinical diagnosis and prognosis.

Genomic alterations were analyzed in 21 uterine leiomyosarcomas (ULMSs) by comparative genomic hybridization. DNA copy number changes were detected in all 21 tumors. The most frequent losses were 13q (16/21 = 76%), 10q (13/21 = 62%), 16q (8/21 = 38%), 12p (7/21 = 33%), and 2p (9/21 = 43%). The most common gains were 17p (8/21 = 38%), Xp (7/21 = 33%), and 1q (7/21 = 33%). High-copy-number gains (ratio > 1.5) were identified in Xp, 1q, and 17p. Loss of 13q was identified in both low-grade and high-grade tumors. Inactivation of a tumor suppressor gene in 13q may be an early event in the development of leiomyosarcomas. Loss of 10q, 2p, and 12p and gains of 1q as well as 17p were frequently found in high-grade tumors and recurrent tumors. Inactivation of tumor suppressor genes and activation of oncogenes in these regions may be associated with a more aggressive behavior of ULMS. Patients with only loss of 13q and without the other alterations listed above had longer survival times. Gains of Xp, 17p, and 1q and losses of 13q, 10q, 16q, 12p, and 2p have been reported in extra-uterine leiomyosarcomas. Our findings indicate that the pathogenesis of uterine leiomyosarcomas and extra-uterine leiomyosarcomas follows the same genetic pathways.

Chromosome Aberrations↗