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PubMed1999

Leiomyosarcoma of the heart and its pulmonary metastasis, both with prominent osteoclast-like multinucleated giant cells expressing tartrate-resistant acid phosphatase activity.

An autopsy case of cardiac leiomyosarcoma and its pulmonary metastasis, both with osteoclast-like multinucleated giant cells (OMGC) mimicking the so-called giant cell variant of malignant fibrous histiocytoma (MFH), is reported. The patient, a 70-year-old male, was admitted for sudden dyspnea. Extensive work-up established only a left atrial tumor mass. Three months after admission, the patient developed multiple intracranial and pulmonary metastases, followed by a worsening clinical course characterized by semicoma and dyspnea, and subsequently died 6 months after the onset of his symptoms. At subsequent autopsy, the left atrial polypoid tumor was found to have invaded destructively to the left half of the cardiac wall. Histology of the cardiac tumor revealed a bimorphic sarcoma in which a poorly differentiated leiomyosarcoma comfirmed by histologic and immunohistochemical findings was juxtaposed to a small nodule with features closely mimicking giant cell MFH. The pulmonary metastatic nodules exhibited features that were entirely indistinguishable from giant cell MFH except for the fact that a minority of polymorphic cells manifested myogenic differentiation. We believe that such a MFH-like pattern represents a pleomorphic form of leiomyosarcoma rather than a dedifferentiated one. The OMGC within the MFH-like component coexpressed CD68 and tartrate-resistant acid phosphatase activity.

Acid Phosphatase↗

PubMed2000

Ovarian leiomyosarcoma: an autopsy case report.

Primary non-specific sarcoma of the ovary is extremely rare, and only 22 reported cases of pure leiomyosarcoma (LMS) are known to the authors. We present an autopsy case of a primary ovarian leiomyosarcoma in a 73-year-old woman. She had noticed an abdominal mass after difficulty in defecating for several months. The excision of tumor with bilateral salpingo-oophorectomy and hysterectomy was carried out. A diagnosis of pure leiomyosarcoma of the left ovary was made on pathological examination with immunohistochemistry. Adjuvant radio-chemotherapy was not given. At 18 months' follow up, abdomino-pelvic sonography revealed an abdominal tumor and hepatic metastasis. The patient died 3.5 years after the initial surgery. The post-mortem examination revealed a peritoneal recurrent tumor and extensive distant metastases of the liver, lungs, pancreas, gastric mucosa, muscle and skin. The prognosis of the ovarian LMS is poor from the pertinent literature. Several prognostic indicators on histology including mitotic activity, proliferative activity and p53 status of the tumor are discussed.

Aged↗

PubMed2000

Leiomyosarcoma of the pulmonary vein.

A case of a 74-year-old man with leiomyosarcoma of the pulmonary vein is reported. The patient felt transient chest oppression while playing golf 1 week before he visited a clinic with a common cold. He underwent an ultrasonographic examination of the heart, which showed a mass lesion in the left atrium. The preoperative clinical diagnosis was myxoma of the left atrium. Cardiac surgery revealed the mass to be a leiomyosarcoma, probably extending from the left inferior pulmonary vein. The patient underwent a left lower lobectomy of the lung, and the tumor was confirmed to have originated from the wall of the left inferior pulmonary vein. Although the patient had a metastatic lesion in the right axillary lymph node 11 months later, which was excised, he remained free of disease 14 months after the initial operation. Histologically, the tumors were composed of pleomorphic cells with bizarre nuclei and spindle cells with blunt-ended nuclei with 1-4 mitotic figures in 10 high power fields. Immunohistologically, the tumor cells were positive for alpha-smooth muscle actin and desmin. We reviewed 17 cases of leiomyosarcoma of the pulmonary vein (six males and 11 females with a mean age of 50 years in each group). The present case was the oldest in age and to our knowledge was the first reported case with metastasis in a distant lymph node.

Aged↗

PubMed2003

Cytogenetic and clinicopathological analysis of soft-tissue leiomyosarcomas.

To identify a characteristic cytogenetic aberration and cytogenetic-morphological correlation in soft-tissue leiomyosarcomas, a karyotypic and clinicopathological analysis of 15 cases of leiomyosarcoma was performed. The histological type was classical in nine cases, pleomorphic in three cases and myxoid in three cases. The histological grade was 1 in three cases, 2 in 10 cases and 3 in two cases. Nine of 15 tumors displayed an abnormal karyotype, whereas the other six tumors displayed a normal karyotype. The relative consistency of involvement of 3p, 3q, 6q, 7p, 7q, 9p, 10p, 11p, 11q, 12p, 16q, 17p and 19q was recognized, although characteristic chromosomal rearrangements were not detected. All six tumors that had a normal karyotype were of the classical type, whereas those displaying an abnormal karyotype contained another morphological type along with the classical type. The results of the present study suggest that chromosomal aberrations contribute to morphological changes in soft-tissue leiomyosarcomas.

Adult↗

PubMed1999

Leiomyosarcoma of the spermatic cord.

BACKGROUND: A rare case of leiomyosarcoma from the spermatic cord is described. A 72-year-old man complained of a hard, golf-ball sized mass in the right inguinal canal. METHODS: Transinguinal radical orchiectomy was performed and histologic examination revealed leiomyosarcoma originating from the spermatic cord. Distant metastases were not observed by further examinations. Radiation as adjuvant therapy was carried out in order to prevent the local recurrence. RESULTS/DISCUSSION: The patient has been alive for 16 months with no evidence of disease. In leiomyosarcoma of the spermatic cord, locoregional recurrence is common. In addition to transinguinal radical orchiectomy, local radiation therapy should be carried out as an adjuvant.

Aged↗

PubMed1999

Case of spontaneous regression of metastatic lesions of leiomyosarcoma of the esophagus.

A rare case of spontaneous regression of esophageal leiomyosarcoma is reported. A 63-year-old woman underwent esophagectomy and reconstruction at our hospital after diagnosis of leiomyosarcoma. Nineteen months after esophagectomy, a coin lesion was detected in the right lung and right thoracotomy revealed pleural dissemination. The lesion in the right lung was resected and was confirmed histologically as recurrence of leiomyosarcoma. During further surgery, metastatic lesions were found in the spleen and pleura. However, all metastatic lesions were found to decrease without any anti-cancer therapy.

Biopsy, Needle↗

PubMed2001

Simultaneous leiomyosarcoma and squamous cell carcinoma of the esophagus: report of a new case.

Leiomyosarcoma of the esophagus is an uncommon neoplasm of mesenchymal origin. The simultaneous occurrence of leiomyosarcoma and squamous cell carcinoma in the esophagus as separate tumors is a very rare event. We present a case leiomyosarcoma associated with squamous cell carcinoma that was diagnosed after surgical resection. The clinical, radiologic, endoscopic and pathologic findings of this rare entity, and four additional cases previously reported in the literature are reviewed.

Biopsy, Needle↗

PubMed2003

Leiomyosarcoma of the esophagus.

Leiomyosarcomas of the esophagus are rare, malignant, smooth-muscle tumors. The presenting symptoms are indistinguishable from other esophageal neoplasms, though the history may be longer due to the slow growth of these tumors. Barium studies may show large intramural masses with ulceration or tracking, expansile intraluminal masses or areas of luminal narrowing. Endoscopic biopsies may give a high false negative rate especially in cases where the mucosa is intact. The treatment of choice is surgical excision. Synchronous and metachronous metastases do not preclude surgery, provided the metastases are also resectable. Prognosis is better than in patients with squamous esophageal cancer. The role of adjuvant radiotherapy and chemotherapy is controversial. We report a 40-year-old man who presented to us with dysphagia and was found to have a leiomyosarcoma of the esophagus. He was treated successfully with esophagectomy and is disease-free after 7 years. We review the literature on esophageal leiomyosarcomas and their management.

Adult↗

PubMedDate not supplied

Leiomyosarcoma of the sinonasal tract: a case report and literature review.

Leiomyosarcoma of the paranasal sinuses is uncommon and has an aggressive clinical behavior. Only 28 cases have been described in the literature, and, of these, only 3 patients treated with surgery had a disease-free survival. In this report, we describe the clinical history of a 57-year-old woman with a leiomyosarcoma of the paranasal sinuses successfully treated with a combination of surgery, radiation therapy, and chemotherapy. We obtained a total response with ifosfamide, doxorubicin, dacarbazine, and epirubicin. This case report emphasizes the possibility of treating this aggressive tumor with chemotherapy in accordance with the same therapy used in the treatment of uterine leiomyosarcomas.

Antineoplastic Combined Chemotherapy Protocols↗

PubMedDate not supplied

[Epitheloid leiomyosarcoma of the mouth in childhood].

A six-year-old girl is presented with an increasing mass involving the anterior vestibule and the floor of the mouth. Histologic and immunohistochemical examination revealed a poorly differentiated epitheloid leiomyosarcoma with destruction of the mandible. According to the CWS-96-study the patient underwent preoperative chemotherapy followed by complete resection of the mandibular body with the surrounding soft tissues. Recurrent tumor became evident only three months later on. The further treatment consisted of tumor resection, oral chemotherapy and irradiation. Nevertheless tumor control could not be achieved. The patient died of progressive disease 16 months after diagnosis. Leiomyosarcoma is extremely uncommon in childhood, especially with localisation in the oral cavity. Diagnosis is based on histologic examination and immunohistochemistry. In the presented case the lack of smooth muscle actin expression made diagnosis difficult. The preoperative chemotherapy could not achieve reduction of tumor size, so that extensive surgery became necessary. Estimation of the prognosis of the oral leiomyosarcomas in childhood is difficult. High grade tumours and involvement of bone seem to be associated with bad clinical outcome. The presented case is confirming that.

Adolescent↗

PubMed1992

Leiomyosarcoma of the pulmonary hilar vessels.

Leiomyosarcomas are uncommon tumors. They have a predilection for deep soft tissues, with a rare group arising in medium-sized or large veins, far less frequently in arteries. We report a case of a 63-year-old man, with an enlarging mass located in the left pulmonary hilar region which was discovered on a routine chest radiograph. Cytology of the sputum and bronchoscopic biopsies did not reveal any malignant cells. Over 5 months observation, there was a clear progression of the mass, and the patient finally underwent a left pneumonectomy which allowed the diagnosis of an intravascular leiomyosarcoma partially destroying major arteries and veins in the hilar region. A review of the literature reveals 67 cases of leiomyosarcoma involving the pulmonary artery. None of these cases had an associated venous pathology. The disease is found mainly in adult women. Clinical diagnosis is very difficult because most of the cases have no specific clinical signs; the major differential diagnosis is that of pulmonary thrombo-embolism. Prognosis depends on histological degree of the tumor and extent of the disease, metastases occur mainly in the liver, lung, and brain and less frequently in regional lymph nodes. Treatment is always surgical, chemotherapy is ineffective and the effectiveness of radiotherapy depends on the total dose of irradiation. Sarcomas of hilar blood vessels have a very poor prognosis with a one year survival estimated, from the onset of symptoms, at 20%.

Humans↗

PubMed1997

[Leiomyosarcoma of the inferior vena cava].

HISTORY AND CLINICAL FINDINGS: In a 34-year-old woman who underwent ultrasound investigation because of recurrent pyelonephritis a space-occupying lesion in the liver was an incidental finding. Computed tomography (CT) and magnetic resonance imaging (MRI) confirmed a tumour in segment I of the liver. The patient had been on contraceptives for 12 years. She had no symptoms. INVESTIGATIONS: Laboratory tests, including tumour markers, were unremarkable. Diagnostic tests to exclude malignant tumour with metastases were negative (thyroid scintigraphy, mammography, coloscopy and gastroscopy). Further tests (ultrasound, coeliaco-mesentericography, hydro-CT and spiral CT, MRI) revealed a tumour, 5 x 4 x 4 cm, in segment I of the liver, most likely an adenoma. Surgical intervention was indicated by the size and questionable malignancy of the tumor. TREATMENT AND COURSE: At operation a tumour was found which originated from the inferior vena cava (IVC) and displaced segment I of the liver. The tumour was resected and the venous wall reconstructed with a Goretex patch. Histological examination indicated a poorly differentiated leiomyosarcoma of the IVC. Adjuvant radiotherapy was undertaken postoperatively. There has been no evidence of recurrence after 10 months. CONCLUSIONS: Leiomyosarcoma of the IVC can be mistaken, both by ultrasound and tomographic diagnostic procedures, for a tumour in segment I of the liver, especially an adenoma. A leiomyosarcoma of the IVC should be included in the differential diagnosis of an hepatic tumour that lies close to segment I of the liver.

Adenoma↗

PubMed1988

Synthesis and secretion of insulin-like growth factor II by a leiomyosarcoma with associated hypoglycemia.

We describe a case of recurrent hypoglycemia apparently caused by secretion of insulin-like growth factor II (IGF-II) by a leiomyosarcoma. A 67-year-old woman presented with recurrent severe hypoglycemia and a large mass in the thorax. During hypoglycemia, plasma cortisol was elevated, but insulin and growth hormone levels were low. After resection of a large leiomyosarcoma, the hypoglycemia resolved. After an eight-year remission, both the tumor and symptomatic hypoglycemia recurred. During a second operation a second large tumor was removed, with relief of the patient's hypoglycemia. The tumor contained high concentrations of IGF-II mRNA and 2100 ng of IGF-II immunoreactive peptide per gram. Filtration through a BioGel P-60 gel column established that 77 percent of the IGF-II was present as a larger molecule, demonstrating incomplete processing of the pro-IGF-II peptides. A similar fraction of high-molecular-weight IGF-II was present in the serum, indicating that the tumor was the chief source of IGF-II. The high-molecular-weight IGF-II found in both the tumor and serum was fully reactive with the IGF-II receptor. Radioimmunoassay showed that the concentrations of insulin-like growth factor I (IGF-I) in tumor and serum were low, suggesting feedback inhibition of growth hormone secretion by IGF-II. Eight months after reoperation, plasma concentrations of IGF-I and IGF-II were normal, and high-molecular-weight IGF-II was virtually undetectable. We conclude that the most likely cause of this patient's recurrent hypoglycemia was IGF-II produced by the leiomyosarcoma.

Aged↗

PubMed2003

Dermal leiomyosarcoma.

Leiomyosarcoma of the skin is a rare tumor that cannot be diagnosed clinically because its gross appearance is nonspecific. The primary skin tumor usually appears as a solitary nodule. It may arise de novo in the dermis or in the subcutaneous tissue, or as a metastatic lesion from an internal neoplasm. Dermal leiomyosarcomas rarely metastasize, unlike the subcutaneous tumors. We describe a case of the dermal type of leiomyosarcoma.

Adult↗

PubMed2001

Synchronous oral leiomyosarcoma and squamous cell carcinoma.

An unusual case of synchronous squamous cell carcinoma and leiomyosarcoma of the oral cavity is reported in a patient without any identified environmental risk or predisposing factors. The invasive squamous cell carcinoma involved the tongue, whereas the leiomyosarcoma was located in the soft palate. No immunostaining was found for human papillomavirus or Epstein-Barr virus, and in situ hybridization showed negativity for human papillomavirus DNA within the tumor cells. Alterations of bcl -2, c-erb -b2 and Rb oncoproteins were not found immunohistochemically. Overexpression of p53 was detected by immunohistochemistry in both tumors, but p53 gene mutations were not found by polymerase chain reaction. Neither loss of heterozygosity of p53 nor microsatellite instability was detected in this patient. The smooth muscle nature of the leiomyosarcoma was confirmed by immunohistochemical methods. To our knowledge, synchronous smooth muscle and epithelial oral tumors have not previously been reported.

Aged↗

PubMed1991

Leiomyosarcoma of the inferior vena cava: analysis and search of world literature on 141 patients and report of three new cases.

Leiomyosarcoma of the inferior vena cava is a rare and potentially curable tumor. Uncertainty about the results of treatment derives from lack of a large series in the same center and of a long-term follow-up of the published cases. A review of the world literature from 1871 to 1989 allowed us to collect information on 141 patients with inferior vena cava leiomyosarcoma to which our three cases have to be added. The tumor arose from the lower segment of the inferior vena cava (infrarenal portion) in 49 patients, from the middle segment (from the renal veins to the hepatic veins) in 59, and from the upper segment (from the hepatic veins to the right atrium) in 34 patients. Complete clinical, pathologic, and therapeutic data and up-to-date follow-up have been obtained through personal correspondence with several authors. All data have been examined with both univariate and multivariate analyses as predictive factors for outcome. Variables, associated with an increased risk of death from disease, included the involvement of inferior vena cava upper segment and a high-grade tumor. Patients who underwent a radical resection of the tumor (82 patients, 56.9%) had a significantly better survival (27.9% and 14.2%, 5- and 10-year survival rates, respectively). Of these patients, those with tumor of the inferior vena cava middle segment fared better than those with lower segment tumor (5- and 10-year survival rates were 48.3% and 34.4%, respectively, for middle segment tumor and 9.3% and 0.0% for lower segment tumor). Variables associated with a good outcome and longer survival were the presence of abdominal pain and the absence of a palpable abdominal mass. Despite the high rate of recurrence (52.4% of patients undergoing radical operation; median time, 25 months), radical resection of inferior vena cava leiomyosarcoma is the only chance for a long-term cure. An earlier and more accurate preoperative diagnosis, by means of modern diagnostic techniques (echography, CT scanning, magnetic resonance imaging) will allow a higher rate of radical resection to be performed with an increase in patient survival.

Adult↗

PubMed2002

Pararenal vena cava leiomyosarcoma versus leiomyomatosis: difficult diagnosis.

Inferior vena cava (IVC) leiomyosarcoma is a rare cancer treated with wide surgical resection. IVC leiomyomatosis is a benign tumor treated with open caval excision. A patient with an IVC tumor with iliocaval thrombosis was treated with thrombolysis and iliac stents. The patient was sent 1 year later to our institution with an IVC mass. Transvenous biopsy was consistent with leiomyomatosis. At surgery, the lesion was adherent, resected and the IVC reconstructed. Pathologic evaluation documented well-differentiated leiomyosarcoma not leiomyomatosis. Thorough evaluation of iliocaval thrombosis is recommended before endovascular management. Diagnostic modalities may be inadequate to differentiate leiomyosarcoma from leiomyomatosis.

Adult↗

PubMed2003

Epithelioid leiomyosarcoma of the gastrocolic ligament.

We present the case of a 56-year old man who presented with a large, well circumscribed cystic mass in the gastrocolic ligament. The histopathological study revealed an epithelioid leiomyosarcoma. Epithelioid leiomyosarcomas are generally solid tumours of the stomach. It is uncommon in the gastrocolic ligament and in a cystic formation. Preoperative diagnosis with ultrasound, computerized tomography and endoscopy was not made. Complete surgical resection of the mass was performed. Histopathological study confirmed the diagnosis of epithelial leiomyosarcoma. Two years after surgery, clinical and radiological examinations of the patient were normal.

Digestive System Surgical Procedures↗