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PubMed1998

[Leiomyosarcoma of the prostate: report of two cases].

We herein present two cases prostatic leiomyosarcoma. The first case was in a 45-year-old man who presented at our department with the chief complaints of pain on voiding and pollakisuria on November 13, 1996. Ultrasonography and computed tomographic (CT) scan revealed a prostatic tumor. A histological examination of biopsy specimens revealed leiomyosarcoma of the prostate. Total prostatectomy and partial cystectomy were performed. No adjuvant therapy was performed. He is still alive without disease 12 months after operation. The second case was in a 63-year-old man who was admitted to our hospital for treatment of a lung tumor and colon polyp on February 28, 1997. CT scans showed a large prostatic tumor and multiple tumors in the lung, liver and bilateral kidneys. He was referred to our department for evaluation of the prostatic tumor. A transrectal needle biopsy of the prostate for histological diagnosis revealed leiomyosarcoma. No treatment was performed and he died 3 months later. In addition, 57 cases of prostatic leiomyosarcoma collected from the Japanese literature are also reviewed.

Cystectomy↗

PubMed1999

Leiomyosarcoma of the colon: a second malignant neoplasm after treatment for a Wilms' tumor.

Leiomyosarcoma of the colon is a rare malignancy. We report the case of a 33-year-old woman with a leiomyosarcoma of the colon occurring as an intussusception 30 years after receiving abdominal irradiation for a Wilms' tumor. A review of the prior and current treatment for Wilms' tumor is discussed, as well as the association between second malignancies and abdominal irradiation. Leiomyosarcoma of the colon usually presents in the fifth and sixth decades of life and is more common in men. The most common symptom is pain. Ninety per cent are diagnosed at surgery for treatment of bleeding, perforation, or obstruction. Surgery remains the primary treatment. Leiomyosarcomas of the gastrointestinal tract are radioresistant, and adjuvant chemotherapy has shown no survival benefit. The overall prognosis is poor, with mean 5-year survival of 28 per cent. General surgeons need to be aware of the possibility of second malignant neoplasms after primary treatment of childhood cancers. Proper reporting is essential to study the long-term effects of early treatment of childhood cancers and in predicting the best treatment outcomes for these patients.

Adult↗

PubMed2005

A giant subcutaneous leiomyosarcoma arising in the inguinal region.

BACKGROUND: Subcutaneous leiomyosarcoma is a rare condition that accounts for 1% to 2% of all superficial soft tissue malignancies. Approximately 10% of cases arise in the trunk, although the extremities are the most commonly affected. CASE PRESENTATION: We report herein the case of a 31-year-old man with a subcutaneous leiomyosarcoma, measuring 124 x 105 mm, arising in the left inguinal region. A wide local excision (with a resection margin >/= 20 mm) was performed. Histological examination of the resected specimen revealed a leiomyosarcoma with high cellularity and two mitoses per 10 high-power fields. The patient remains well with no evidence of disease 5 years and 8 months after the operation. CONCLUSION: This is the first reported case of subcutaneous leiomyosarcoma arising in the inguinal region and also one of the largest tumors reported. The experience of this case and a review of the English-language literature (PubMed, National Library of Medicine, Bethesda, MD, USA) suggest that a resection margin of >/= 10 mm is recommended when excising this rare tumor.

Journal Article↗

PubMed2006

Radiation-induced leiomyosarcoma of the oropharynx.

Leiomyosarcoma is a malignant mesenchymal tumor originating from smooth muscle cells, which most frequently develops in the myometrium and in the gastro-intestinal tract. Reviewing the international literature, radiation-induced sarcoma arise in 0.035 to 0.2% of all irradiated patients. Especially in the head and neck region, radiation-induced leiomyosarcoma is an extremely rare lesion. The authors report a case of a radiation-induced leiomyosarcoma of the tonsillar region of the oropharynx in a 51-year-old male patient, who had undergone radiation therapy of this region 38 years before. The lesion was treated by radical surgery. Diagnostic steps, histological presentation and therapy are described in detail and the literature concerning radiation induced malignancies in general as well as radiation induced leiomyosarcoma in particular is reviewed. The highlights of this case are an extremely uncommon location and a rare pathological entity of radiation induced malignancies.

Journal Article↗

PubMedDate not supplied

Hypoglycemia attributable to insulin-like growth factor-II prohormone-producing metastatic leiomyosarcoma.

OBJECTIVE: To review the causes of nonpancreatic tumor-associated hypoglycemia and report the first case of hypoglycemia attributable to a leiomyosarcoma, which did not cause hypoglycemia in its primary site but only after metastasizing. METHODS: A case report is presented of a 62-year-old man with a gastric leiomyosarcoma diagnosed and surgically treated 8 years previously, who was found to have 14 large, rounded masses in his liver and a blood glucose level of 19 mg/dL. Biopsy of the largest mass revealed a leiomyosarcoma. RESULTS: Evaluation of the cause of the hypoglycemia revealed that circulating insulin, connecting peptide, proinsulin, insulin-like growth factor-I (somatomedin C), and insulin-like growth factor-II levels were below normal, whereas the insulin-like growth factor-II prohormone concentration was increased twofold. Basal and corticotropin-stimulated serum cortisol values were normal. CONCLUSION: This is the first case report of hypoglycemia occurring only after metastasis of a leiomyosarcoma. A possible causal relationship between the hypoglycemia and the increased circulating insulin-like growth factor-II prohormone is suggested, and alternative explanations and treatment are discussed.

Journal Article↗

PubMed2007

Clinical course of nonvisceral soft tissue leiomyosarcoma in 225 patients from the Scandinavian Sarcoma Group.

BACKGROUND: Leiomyosarcoma of nonvisceral soft tissues is an uncommon malignant tumor; thus, only small numbers of cases have been reported. This study was based on a large series of patients from the Scandinavian Sarcoma Group Register acquired during a 15-year period (from 1986 to 2001). Follow-up information was available for all patients. METHODS: The authors analyzed the clinical features of 225 patients with cutaneous, subcutaneous, or deep-seated leiomyosarcoma of the extremities, trunk wall, and superficial parts of the head and neck region to determine the natural course of the disease. Only patients who received their treatment at a specialist sarcoma center were included. Re-evaluation of histopathology was performed. RESULTS: The age of the patients (121 women and 104 men) ranged from 20 years to 98 years (median, 70 years), and the tumors ranged in size from 0.6 cm to 35 cm (median, 4.0 cm). Eighty-two percent of the tumors were classified as high grade. The median follow-up for survivors was 5.5 years. The local treatment was adequate in 154 of 206 patients (75%) who were without metastasis at presentation. At 10 years, 84% of the 206 patients with localized disease at presentation were free from local recurrence, 66% remained metastasis free, and 49% were alive. Multivariate analysis showed that higher malignancy grade (P = .006), larger tumor size (P = .003), and deeper tumor location (P = .002) were correlated significantly with decreased metastasis-free survival, inadequate local treatment was correlated with local recurrence (P = .007), and high malignancy grade was correlated with decreased overall survival (P = .007). CONCLUSIONS: The long-term prognosis for patients with subcutaneous and deep-seated soft tissue leiomyosarcoma remains poor despite the ability to achieve adequate local control through nonmutilating surgery with or without radiotherapy.

Adult↗

PubMed2006

Prosthetic replacement of the infrahepatic inferior vena cava for leiomyosarcoma.

HYPOTHESIS: Resection of the infrahepatic inferior vena cava associated with prosthetic graft replacement for caval leiomyosarcoma is an acceptable procedure to obtain prolonged and good-quality survival. DESIGN: A consecutive sample clinical study with a mean follow-up of 40 months. SETTING: The surgical department of an academic tertiary center and an affiliated secondary care center. PATIENTS: Eleven patients, with a mean age of 51 years, who have primary leiomyosarcoma of the infrahepatic inferior vena cava. INTERVENTIONS: All of the patients underwent radical resection of the tumor en bloc with the affected segment of the vena cava. Reconstruction consisted of 10 cavocaval polytetrafluoroethylene grafts and 1 cavobiliac graft. An associated right nephrectomy was performed in 2 cases. The left renal vein was reimplanted in the graft in 3 cases. MAIN OUTCOME MEASURES: Cumulative disease-specific survival, disease-free survival, and graft patency rates expressed by standard life-table analysis. RESULTS: No patients died in the postoperative period. The cumulative (SE) disease-specific survival rate was 53% (21%) at 5 years. The cumulative (SE) disease-free survival rate was 44% (19%) at 5 years. The cumulative (SE) graft patency rate was 67% (22%) at 5 years. CONCLUSION: Radical resection followed by prosthetic graft reconstruction is a valuable method for treating primary leiomyosarcoma of the infrahepatic inferior vena cava.

Adult↗

PubMed1999

Intratesticular leiomyosarcoma in a young man after high dose doping with Oral-Turinabol: a case report.

BACKGROUND: Androgenic anabolic steroids have been suspected of activity as carcinogens in the development of carcinoma and angiosarcoma of the liver and adenocarcinoma of the prostate. Although the proliferation of smooth muscle cells is stimulated by sexual steroids, to the authors' knowledge a possible relation between androgenic anabolic steroids and the development of leiomyosarcoma has not previously been reported in humans. METHODS: A 32-year-old man underwent right radical orchiectomy for a tumor of the upper pole of the right testicle. Routine histopathologic examination and immunohistochemical staining were performed. RESULTS: The tumor was identified as an intratesticular leiomyosarcoma based on its typical growth pattern and the characteristic immunohistochemical staining profile. The patient reported a 5-year history of systematic use of high dose Oral-Turinabol (4-chloro-1-dehydro-17alpha-methylteststerone) that began at age 18 years and stopped approximately 9 years before presentation. CONCLUSIONS: The rarity of intratesticular leiomyosarcoma, the experimental induction of similar tumors in animals by androgens and estrogens, and the unusually young age at presentation of the patient in the current study support the hypothesis that high dose doping with androgenic anabolic steroids could have played a cocarcinogenic role in the development of the tumor in this case.

Actins↗

PubMed1998

Epithelioid leiomyosarcoma of the larynx.

BACKGROUND: Tumors of smooth muscle origin are rare in the upper aerodigestive tract, due to the paucity of smooth muscle in the area. A review of the literature revealed 34 reported cases of leiomyoma, 9 cases of leiomyosarcoma, and only 2 cases of epithelioid leiomyoma arising in the larynx. METHODS: A case report of an epithelioid leiomyosarcoma arising in the right piriform fossa of a 38-year-old man is presented. RESULTS: The tumor was treated with conservative surgical resection and postoperative radiotherapy. There was no evidence of locoregional recurrence 2 years postsurgery, and laryngeal function was preserved. CONCLUSIONS: To our knowledge, this is the third case of a laryngeal epithelioid smooth muscle tumor to be reported in the world literature and the only documentation of an epithelioid leiomyosarcoma arising in the larynx.

Adult↗

PubMed1976

Gastric epithelioid leiomyoma and leiomyosarcoma (leiomyoblastoma).

A series of 127 surgical specimens of epithelioid leiomyomatous tumors (leiomyoblastomas) of the gastric wall from the files of the Armed Forces Institute of Pathology (AFIP) were studied as to biologic behavior, morphogenesis, and histologic features of value in distinguishing benign and malignant variants. These tumors affect middle-aged men primarily and usually present with upper gastrointestinal bleeding or peptic ulcer-like symptoms. They are composed of a mixture of round epithelioid and spindle cells, many of which have clear cytoplasm. The cells are ensheathed by delicate reticular fibers. The presence of a perithelial or glomoid pattern in some tumors suggests a possible relationship to angiomyoma, glomus tumors, and "pericytoma." The epithelioid leiomyoma, the benign form, often arises in the mid- and distal stomach, especially on the anterior wall. Microscopically, it is recognized by the presence of large epithelioid cells and infrequent mitotic figures. Of 103 epithelioid leiomyomas, only one metastasized and thus was biologically malignant. The epithelioid leiomyosarcoma often arises in the proximal stomach and also distally, especially on the posterior wall. Two histologic types of epithelioid leiomyosarcoma are distinguished from the benign epithelioid leiomyoma by the small size of the cells and occasional higher mitotic counts. One sarcoma variant is a small cell caricature of the leiomyoma. The other is more anaplastic, assoicated with a loss of reticular fibers surrounding the cells and an alveolar arrangement. Epithelioid leiomyosarcomas are the most common type of gastric sarcoma. They are aggressive neoplasms; 63% metastasized, usually within 2 years after diagnosis.

Adolescent↗

PubMed1980

Primary leiomyosarcoma of bone: a study of five cases.

Five cases of leiomyosarcoma of bone are described. The patients, 3 men and 2 women, ranged in age from 24 to 74 years. Four of the five tumors were located in the distal half of the femur. Radiographically, all tumors presented as purely osteolytic lesions. Angiography, performed in 3 cases, revealed hypervascularity and the presence of irregular, tortuous vessels at the site of the lesion in 1, but not in 2 others. Histologically, the five tumors showed features characteristic of leiomyosarcomas. The most prominent features were blunt-ended, cigar-like, chromatin-rich nuclei in elongated acidophilic cells arranged in bundles which intersected each other at wide angles. The importance of trichrome stains, such as van Gieson stain, for th recognition of leiomyosarcomas is emphasized. Electron microscopic examination, performed in 3 cases, showed as the most prominent features spindle-shaped cells in parallel arrangement containing abundant myofilaments which elongated densities, pinocytotic vesicles, and basal lamina structures.

Adult↗

PubMed1981

Leiomyosarcoma of the breast: a case of long survival and late hepatic metastasis.

A case of leiomyosarcoma of the breast that metastasized to the liver 15 years after mastectomy is reported. Leiomyosarcoma appears to have less aggressive biologic behavior than other types of breast sarcomas. Analysis of the reported cases of smooth muscle tumors of the breast revealed some guidelines for the histologic diagnosis of these tumors. Tumors with three or more mitoses per 10 HPF are leiomyosarcomas, and those with no mitotic activity, necrosis, and significant cellular atypia are leiomyomas. The borderline cases are best considered smooth muscle tumors with uncertain malignant potential.

Breast Neoplasms↗

PubMed1986

Leiomyosarcoma of the external soft tissues. A clinicopathologic, immunohistochemical, and electron microscopic study.

This clinicopathologic study concerns 25 cases of leiomyosarcoma of the external soft tissues, including an immunohistochemical survey of 19 cases and an electron microscopic examination of six. There was a female preponderance in a ratio of 16:9. The most common site of tumors was the thigh (nine cases), followed by the knee (five cases). Three tumors were located in the dermis, 9 were confined down to the subcutis, 2 involved the deep fascia, and 11 involved the skeletal muscle. Superficially located tumors were smaller than deeply located ones. According to the predominant histologic features, the tumors were classified into well (6), moderately (14), and poorly (5) differentiated. In addition to the classical microscopic features, dedifferentiated areas resembling malignant fibrous histiocytoma were noted in three primary tumors and one recurrent tumor. In six tumors, the anatomical location and histologic features were highly suggestive of leiomyosarcoma arising in the venous wall. Many tumor cells immunoreactive for actin were seen in 13 and for desmin in 9 of the 19 cases. Four tumors contained a few tumor giant cells positive for alpha-1-antichymotrypsin. Ultrastructurally, the tumors were characterized by smooth muscle tumor cells showing a spectrum of differentiation of varying degrees. In two cases, histiocyte-like tumor cells were detected. The most reliable prognostic parameter was the depth of the initial tumors; all patients with a leiomyosarcoma confined down to the subcutis are living, most of them more than 5 years after excision, whereas seven of the nine patients with a tumor involving the muscle have died within 5 years after excision.

Actins↗

PubMed1987

Radiation-induced leiomyosarcoma of the great vessels presenting as superior vena cava syndrome.

A patient with a pleomorphic intravascular leiomyosarcoma of the great vessels of the neck and mediastinum presented clinically with a superior vena cava syndrome. A latent period of 29 years elapsed between receiving orthovoltage radiation to the neck and right side of chest to treat recurrent ganglioneuroblastoma, and the appearance of a leiomyosarcoma and subsequent recurrences. The patient underwent partial resection of the tumor, received adjunct chemotherapy, and was shown to be free of disease by clinical tests and by magnetic resonance imaging (MRI) 17 months after completion of chemotherapy. The criteria for the diagnosis of radiation-induced sarcomas are reviewed in relation to the present case. The critical role of magnetic resonance imaging in both the diagnosis and continued follow-up of the patient is described. This would appear to be the first reported case of radiation-induced intravascular leiomyosarcoma of the great vessels of the neck and mediastinum presenting as a superior vena cava syndrome.

Adult↗

PubMed1995

Gastrointestinal leiomyosarcoma metastatic to the liver. Durable tumor regression by hepatic chemoembolization infusion with cisplatin and vinblastine.

BACKGROUND: Gastrointestinal leiomyosarcoma metastatic to the liver is considered most resistant to any combination of systemic chemotherapy containing doxorubicin and/or ifosphamide. METHODS: Fourteen patients with gastrointestinal leiomyosarcoma metastatic to the liver were treated with hepatic chemoembolization infusion consisting of polyvinyl alcohol sponge particles mixed with cisplatin powder (150 mg) followed by an intrahepatic arterial infusion of vinblastine (10 mg/m2). RESULTS: Ten major (> 50% regression) tumor responses were observed (70%) in patients lasting from 8 to 31+ months (median, 12 months) after an average of two hepatic chemoembolization procedures, usually 4 weeks apart. Transient side effects included right upper quadrant pain requiring narcotics, significant hepatic enzyme elevation, particularly of lactic dehydrogenase with a minimal increase in bilirubin, paralytic ileus requiring nasogastric suction up to 72 hours, urinary electrolyte losses (potassium+, magnesium++, sodium+) requiring supplements, and occasionally mild but transient leukopenia and thrombocytopenia. CONCLUSIONS: Hepatic chemoembolization infusion appears to induce a high rate of durable tumor response in patients with notoriously chemoresistant gastrointestinal leiomyosarcoma metastatic to the liver.

Adult↗

PubMed2002

Tumor specific gene expression profiles in human leiomyosarcoma: an evaluation of intratumor heterogeneity.

Leiomyosarcomas are malignant smooth muscle tumors characterized by a spectrum of histopathologic features and clinical behavior. Gene expression profiling of leiomyosarcomas may identify differential gene signatures that may allow for the clinical stratification of the tumors. Typically, surgical specimens from these tumors are large and manifest a variegated macroscopic appearance. Because of their large size at the time of diagnosis, sufficient tissue is available for regional and clonal heterogeneity assessment. However, if the gene expression profiles of samples taken from different locations in the tumors are drastically different, biologic classification on the basis of random sample analysis may not be adequate. Therefore, to assess intertumor and intratumor heterogeneity, the authors performed a gene expression study using leiomyosarcoma specimens from three excised sarcomas from an equal number of different patients. Comparisons between tumor and normal samples from the three patients as well as between carefully mapped peripheral and core specimens from the same tumor (excised from one of the patients), were performed. Analysis of the expression profiles demonstrated minimal intratumor variations compared with intertissue variations, indicating homogeneous tumor specific gene expression profiles. The authors also identified genes that are expressed differentially in tumor and normal tissue.

Biomarkers, Tumor↗

PubMed2002

Peritoneal washing cytology findings of disseminated myxoid leiomyosarcoma of uterus: report of a case with emphasis on possible differential diagnosis.

We report the cytology findings of a rare case of myxoid leiomyosarcoma of the uterus with intraabdominal dissemination. The cytology showed uniformly dispersed spindly to polygonal "epithelioid" tumor cells focally linked by background matrix. Spindly tumor cells attaching to and radiating from branching capillary structures were also present. Occasionally, scattered "signet-ring" cells were found, mimicking metastatic poorly differentiated adenocarcinoma. The background mucoid substance was more conspicuous in the cell block sections. Gross and histologic examination of the surgical specimen revealed a large uterine leiomyosarcoma with prominent myxoid change. Ultrastructural study showed that the "signet-ring" appearance was mainly due to degenerative cytoplasmic change with ballooning of mitochondria, dilatation of endoplasmic reticulum, and elevation of outer nuclear membrane. In contrast to other spindle cell malignancies such as sarcomatoid mesothelioma, sarcomatoid carcinoma, or malignant melanoma, true sarcoma cells in general carry a distinctive cytologic appearance in washing/effusion fluid. In a correct clinical setting, the peculiar association with mucoid matrix and absence of classic lipoblasts should also raise the suspicion of metastatic myxoid leiomyosarcoma.

Aged↗

PubMed2005

A case of uterine adenomyoma with bizarre smooth muscle cells mimicking leiomyosarcoma.

We report a case of adenomyoma of the uterus that was cytologically difficult to distinguish from leiomyosarcoma. Examination of a uterine cervical smear revealed numerous spindle cells that were present in cell clusters or as isolated cells. These cells contained nuclei that were oval-shaped/elongated with nucleoli and delicate wispy cytoplasm. Large and bizarre nuclei were also identified. Based on these cytological findings leiomyosarcoma was considered: however, this diagnosis remained uncertain because of the absence of mitosis and/or necrotic substance. Histologically, we recognized leiomyomatous smooth muscle cells growing in a solid pattern and intermingled with endometrial-type glands. Moreover, bizarre smooth muscle cells were observed in the surface layer of the tumor. These observations suggest that for a diagnosis of uterine leiomyosarcoma the presence of mitosis and/or necrosis is important in addition to nuclear atypia.

Adenomyoma↗