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PubMed1999

Leiomyosarcoma of the gastric cardia and fundus.

BACKGROUND: Gastric leiomyosarcoma is rare among gastric malignancies, and only 20% of the cases are located in the gastric cardia or fundus. It has clinical manifestations different from tumors in other sites of the stomach. We reviewed cases of leiomyosarcoma of the gastric cardia or fundus to evaluate their clinicopathologic characters and patient survival. METHODS: From May, 1981, to June, 1998, a total of 15 patients who underwent curative resection of leiomyosarcoma of the gastric cardia or fundus were retrospectively identified and studied. All the clinical and pathologic data were reviewed and recorded. RESULTS: There were 11 men and four women. Their mean age was 59.1 years (range, 37-73 years). Tarry stools and epigastric fullness and pain were the most common symptoms, followed by body weight loss. All 15 patients had submucosal tumors in the gastric cardia or fundus, as shown by endoscopy, barium contrast radiography and computerized tomography. The incidence of esophageal involvement by the tumors was quite low. The mean tumor size was 10 cm, ranging from 4 cm to 20 cm. Locoregional organs were involved in nine patients. The median and mean survivals were 17.8 months and 31.7 months, respectively (range, 10.1-80.1 months) after curative resection. The three-year and five-year survival rates were 53% and 22%, respectively. CONCLUSIONS: The definitive preoperative diagnosis of the tumor is difficult before surgery, even though imaging studies show positive findings. Surgical resection with an adequate safe margin of normal tissue is the treatment of choice. Tumor size, serosal invasion with locoregional organ involvement and high tumor grade were the prognostic factors in the study.

Adult↗

PubMed1999

Pulmonary artery leiomyosarcoma.

Pulmonary artery leiomyosarcoma is a rare but highly lethal disease, and can be mistaken for pulmonary thromboembolism. We report a case of pulmonary artery leiomyosarcoma managed with surgical resection, chemotherapy, and radiotherapy. A 57-year-old woman was admitted with complaints of aggravated dyspnea. She was initially treated with oxygen therapy and heparinization for a suspected pulmonary embolism. Echocardiography revealed a dilated right atrium and ventricle and severe tricuspid regurgitation, with an estimated systolic right ventricular pressure of 95 mm Hg; a shadow of a mass in the main pulmonary artery was also noted. Right ventriculography revealed a filling defect, and to-and-fro motion of the mass in the main pulmonary artery. The left pulmonary artery was almost totally occluded by the mass. The patient's condition improved dramatically after palliative excision of the mass and patch reconstruction of the outflow tract of the right ventricle with a bicuspid xenograft. Pathologic examination of the mass revealed leiomyosarcoma. Chemotherapy and radiotherapy were subsequently administered and follow-up imaging studies 3 months postoperatively revealed no recurrence of the tumor. The patient remains well, more than 1 year after treatment. This report emphasises that pulmonary artery sarcoma should be considered in the differential diagnosis in cases of suspected pulmonary thromboembolism.

Diagnosis, Differential↗

PubMed1999

Comparative immunohistochemical and molecular analysis of uterine and extrauterine leiomyosarcomas.

Histologic criteria defining malignancy in smooth muscle tumors are currently site specific. This study was undertaken to determine whether, in leiomyosarcomas (LMS) occurring in different anatomic locations, there were differences in patterns of expression of molecules that have been demonstrated to be associated with biologically aggressive behavior in malignant neoplasms, and also to determine their diagnostic utility. Formalin-fixed paraffin-embedded tissue blocks were selected from 16 extrauterine leiomyosarcomas (EULMS), 14 cases of uterine leiomyosarcomas (ULMS) and from five cases each of uterine and extrauterine leiomyomas (LM). Utilizing immunohistochemical (ABC) techniques with antigen retrieval, we assessed serial sections of each tumor for immunoreactivity with Glut1, CD44s, bcl2, cyclin D1, and estrogen receptor. Molecular genotyping for detecting k-ras-2 point mutation, p53 gene loss, and mdm2 gene amplification was performed on microdissected tumor samples from the same histologic sections. All of the uterine and extrauterine LM were diffusely positive for CD44s, bcl2, and cyclin D1, and uniformly negative for Glut1. In contrast, 50% of the ULMS and 25% of EULMS were Glutl positive. Moreover, Glut1 positivity closely correlated with aggressive biologic behavior reflected by distant metastatic spread. Eighty-percent of LM and 70% of the ULMS were estrogen receptor positive, whereas only one retroperitoneal tumor had focal weak positivity. Over 80% of the extrauterine and 50% of the uterine sarcomas showed absence of CD44s immunoreactivity. Percentage of cyclin D1 immunoreactivity was independent of tumor grade and inversely proportional to the percent of bcl2 positivity. An LMS of the male breast contained k-ras-2 exon 1 point mutation (codon 12 aspartate substitution of glycine). P53 allelic imbalance was present in 29% of ULMS and 57% EULMS. Mdm2 amplification was present in three of six EULMS but not in ULMS. In addition to clinical staging, Glut1 positivity together with patterns of immunoreactivity of CD44 and bcl2 may be helpful in identifying aggressive smooth muscle tumors of the uterus and some EULMS. The presence of estrogen receptor staining may be helpful in identifying uterine versus nonuterine LMS. Although sample numbers are too small for definite conclusions, this study suggests that there are differences in glucose transport, expression of adhesion molecules, and estrogen receptors in ULMS and EULMS, which in part may be due to the estrogen dependency of the ULMS. P53 mutations and mdm2 amplifications appear to be more frequent in EULMS.

Adult↗

PubMed2000

[Primary leiomyosarcoma of bone. Report of 5 anatomo-clinical cases and review of the literature].

INTRODUCTION: Leiomyosarcoma is a malignant smooth muscle tumor occurring most frequently in uterus or soft tissues and more rarely in bone. MATERIALS AND METHODS: We report the clinicopathologic, immunohistochemical and ultrastructural findings of five cases of primary leiomyosarcoma of bone treated in our Department between 1991 and 1994. The pertinent medical literature is discussed. RESULTS: The tumors were located respectively in the distal tibia (n=2), the distal femur, the sternum and the ilium (n=1). Four lesions were high-grade and one low-grade. All patients (3 women and 2 men) underwent wide surgical resection associated with polychemotherapy in four cases. Two patients died of metastatic disease, two had local recurrence and one is alive with no evidence of disease at the last follow-up. DISCUSSION: Excluding cases which involve the facial skeleton, there are to our knowledge 95 cases of primary leiomyosarcoma of bone reported in the literature. This tumor arises more commonly in adults (mean age: 49 years) with an equal gender distribution and involves predominantly the long bones near the knee. In the majority of cases, plain X-rays exhibit an osteolytic lesion with cortical penetration and indistinct margins. The diagnosis is based on microscopic features demonstrating fusiform tumor cells arranged in interwoven bundles, and the immunohistochemical results of widespread cytoplasmic positivity for smooth muscle actin. The best pronostic parameter is the histologic grade correlated with both the recurrence and metastatic rates as well as the survival rate. Surgery constitutes the main treatment since chemotherapy or radiotherapy did not provide an improved prognosis over a wide resection.

Adult↗

PubMed2000

[A case of epithelial membrane antigen-positive leiomyosarcoma of the urinary bladder].

A 41-year-old woman was admitted with gross hematuria and pain on urination. Cystoscopy showed a huge and lobulated submucosal non-papillary bladder tumor. Pelvic computed tomography demonstrated a heterogeneous and enhanced lobulated mass, 8 cm in diameter, with extravesical invasion but there appeared to be no metastatic lesions. Transurethral biopsy revealed leiomyosarcoma pathologically. Total cystectomy and construction of an ileal conduit were performed. The tumor was histologically diagnosed as leiomyosarcoma. Immunohistochemical studies revealed the tumor to be positive for epithelial membrane antigen (EMA) and muscle actin but negative for desmin, and S-100. We reviewed 102 cases of vesical leiomyosarcoma reported in Japan. Among these 102 cases, there were no EMA-positive cases. Immunohistochemical and electromicroscopic evaluation should be performed to evaluate this disease.

Adult↗

PubMed1998

[Leiomyosarcoma of the stomach].

Gastric leiomyosarcoma (GLMS) is a malignant, smooth muscle neoplasm accounting for not more than 0.45%-3.5% of primary gastric malignancies and 17%-20% of all smooth muscle tumors of the stomach. A well-characterized variant has been variously referred to as leiomyoblastoma and epithelioid leiomyosarcoma. Because of the rarity of GLMS, few authors have tried to correlate clinical presentation, pathological findings, and treatment. There is no uniform therapeutic approach for leiomyosarcoma of the stomach and reported survival rates vary widely. We present a 56-year-old man whom we treated for this condition.

Humans↗

PubMedDate not supplied

Orbital leiomyosarcoma after retinoblastoma.

Patients with the inherited, bilateral form of retinoblastoma have an increased incidence of osteogenic sarcoma such that the mortality from the secondary tumor exceeds that of the initial bilateral retinoblastoma. We report a 29-year-old male survivor of bilateral retinoblastomas originally diagnosed at 8 months of age, whose treatment eventually included bilateral enucleation, bilateral orbital radiation, and systemic chemotherapy. At age 26, a tumor removed from his right maxillary sinus was diagnosed as fibroma. At age 29, he developed an inferior orbital mass that extended into the right maxillary sinus. A biopsy and comparison with the previous maxillary sinus mass revealed both lesions to be leiomyosarcoma. Both light and electron microscopy supported the diagnosis. The patient has survived treatment with orbital exenteration and maxillectomy combined with postoperative radiation to the right orbital-maxillary area. This appears to be the fourth case of leiomyosarcoma in the third decade of life in a male patient with a previously irradiated orbit after enucleation for bilateral retinoblastoma. Leiomyosarcoma appears to be another orbital tumor associated with bilateral retinoblastoma.

Adult↗

PubMedDate not supplied

A mesocolic leiomyosarcoma metastatic to the breast: case report and review of the literature.

This report describes a patient with mesocolic leiomyosarcoma, initially treated by excisional surgery. One year later the patient developed breast and hepatic metastases. To our knowledge, metastasis of leiomyosarcoma to the breast has not been previously reported in the literature. The discussion covers unusual sites for metastases of primary leiomyosarcomas and those tumors that most commonly give rise to breast metastases. Certain rare primary tumors of the breast related to the family of sarcomas are also brought to the attention of the reader. These may or may not have malignant characteristics. In the light of the differences in prognosis and treatment, recognition and precise diagnosis is of importance.

Breast Neoplasms↗

PubMed2001

Leiomyosarcoma of the vulva. A case report.

BACKGROUND: Leiomyosarcoma of the vulva is a rare mesenchymal tumor. Biologic features of a low grade tumor were investigated by an immunohistochemical workup. CASE: A 38-year-old woman presented with a slowly growing vulvar mass. Surgical treatment was performed, and a low grade leiomyosarcoma of the vulva was diagnosed. Immunohistochemical reactions were performed with monoclonal antibodies against desmin, vimentin, smooth muscle actin, cytokeratin, S-100 protein, estrogen, progesterone and androgen receptor, p53 protein, Ki-67 antigen, leukocyte common antigen and polyclonal antibodies to factor VIII-related antigen. Expression of estrogen, progesterone and androgen receptor was present in addition to a moderate number of Ki-67-positive cells and absence of p53 protein overexpression and lymphatic cell infiltration besides adequate microvessel density for smooth muscle tumors. Since the immunohistochemical markers indicated a less aggressive tumor, any further adjuvant therapy was rejected. The patient was without recurrence 24 months later. CONCLUSION: The immunohistologic profile proved the low histologic grade of vulvar leiomyosarcoma. The findings helped to estimate prognosis and plan therapy.

Adult↗

PubMed2001

Leiomyosarcoma of the ethmoidal cells.

Leiomyosarcomas of the paranasal sinuses are rare malignant tumors. A case of a 68-year-old female with leiomyosarcoma of the ethmoidal cells is presented. Since half a year she had a stuffed nose on both sides. Preoperatively, several attacks of epistaxis on the right side occurred. CT scans showed a tumor of the ethmoidal cells on the right side. The tumor was completely removed via a functional endoscopic endonasal approach and right sphenoethmoidectomy and maxillary sinus surgery. The operation was followed by a radiotherapy with 72 Gy. Up to 29 months after the operation local recurrence could not be observed. Endonasal tumor resection followed by radiotherapy in a case of leiomyosarcoma without invasion of orbit and skull base can allow tumor control.

Aged↗

PubMed2001

[Primary osseous leiomyosarcoma of the jaw. Apropos of a case].

Primary leiomyosarcoma of bone is a malignant smooth muscle tumour uncommonly found in the jaws. We report a case of primary leiomyosarcoma of the mandible in a 31-year-old man treated with wide resection and radiotherapy then secondary reconstruction with a fibula free flap. No recurrence or distant metastasis has developed four years after the initial surgery. We review the literature on primary leiomyosarcoma of the jaws.

Adult↗

PubMed2001

A case of primary leiomyosarcoma of the chest wall successfully resected under the video-assisted thoracoscopic approach.

We report a case of a 62-year-old woman with primary leiomyosarcoma of the chest wall which was successfully resected under the video-assisted thoracoscopic approach. The disease was found during the treatment for a malignant melanoma of the left heel. On the preoperative CT images, the lesion was suspected to be a metastasis of the malignant melanoma. The thoracoscopic surgery revealed that the tumor originated from the parietal pleura, and it was resected with a 5-mm margin of normal pleura. Histopathologically, the tumor was diagnosed as low-grade leiomyosarcoma. Since no residual tumor cells were proven in the resected margins histologically, further resection was not performed. At present, she is alive and well with no sign of recurrence of leiomyosarcoma two years and one month after operation. Thoracoscopic surgery is worth trying for accurate diagnosis of and effective treatment for a chest lesion without apparent invasion of the chest wall on the preoperative CT images.

Biopsy↗

PubMed2002

[A case of leiomyosarcoma of the spermatic cord].

A 44-year-old male was referred to our hospital for further treatment of lung and adrenal metastases from leiomyosarcoma occurring in the left spermatic cord. He had undergone high orchiectomy 5 months before, but no adjuvant therapy was done. Although systemic CYVADIC therapy (cyclophosphamide, vincristine, doxorubicin hydrochloride, dacarbazine) was performed in our hospital, he died of metastatic disease 10 months after the initial therapy. Leiomyosarcoma arising in the spermatic cord is a rare entity. We present a case of leiomyosarcoma of the spermatic cord, and to our knowledge, this is the 23rd case reported in Japan.

Adrenal Gland Neoplasms↗

PubMed2002

[Epithelial leiomyosarcoma of gingiva: a case report and review of literature].

Epithelioid leiomyosarcoma (EL) is a rare malignant tumor of mesenchymal origin. The Authors review the literature and report a case of gingival epithelioid leiomyosarcoma in a 40-year-old patient. In this case the leiomyosarcoma was located in the lower front dental group and invaded the symphysis menti. A segmentary mandibolectomy was performed with reconstruction using a non-revascularized autologous iliac bone graft. The differential diagnosis of primary EL is quite complex and it is grouped with other sarcomas, sarcomatoid carcinoma, myoepithelioma, amelanotic melanoma and metastases from gastrointestinal EL. Anatomopathological examination and immunohistochemical study enabled a definitive diagnosis of primary EL of gingiva. The follow-up calls for clinical-radiological check-ups every three months for the first year and every six months thereafter. One year after surgery there were no signs of recurrence.

Adult↗

PubMed2002

[Leiomyosarcoma of the larynx. One case report].

The leiomyosarcoma of the larynx is a neoplasm extremely odd, only 46 cases are reported in the literature. We present a case seen in our Department. A male patient, 39-year-old, smoker, snoring and respiratory insufficient, who showed a whitish mass, 2.5 cm, round, steady on the right vocal fold. Urgent tracheostomy was performed but afterwards the patient refused new surgery. Biopsy revealed a malignant tumour of fusiform cells, validating as diagnostic through immunohistochemical analysis, a leiomyosarcoma case. Five months later the patient was seen again and a total laryngectomy performed. Eleven months after surgery the patient is alive and no signs of recurrence detected. In this paper diagnosis and treatment of leiomyosarcoma is discussed.

Adult↗

PubMed2002

Leiomyosarcomas: analysis of clinical presentations in 6 patients.

OBJECTIVE: To identify characteristic features of leiomyosarcomas in an attempt to diagnose them preoperatively. STUDY DESIGN: The 3 years experience with 6 patients with uterine leiomyosarcomas at Hayatabad Medical Complex, Peshawar was retrospectively reviewed. RESULTS: The mean age of occurrence was 50 years with abnormal uterine bleeding the commonest symptom. Leiomyosarcomas had no preferential uterine location and were of variable sizes. Only 1 case was diagnosed preoperatively. CONCLUSION: We have observed that physical examination and imaging techniques are rarely helpful in establishing the diagnosis preoperatively. We recommend larger prospective studies to evaluate the efficacy of preoperative monitoring of enlarged uteri.

Adult↗

PubMed2003

[Primary ureteral leiomyosarcoma: a rare cause of obstructive uropathy].

OBJECTIVES: Transitional cell carcinoma is the most frequent primary malignant neoplasia obstructive ureteral pathology. Nevertheless, obstructive primary ureteral leiomyosarcoma (stromal tumour) is an unusual disease, diagnosed by histopathological examination of nephroureterectomy surgical specimens. METHODS/RESULTS: We report a case of ureteral obstructive uropathy in a 38-year-old woman, with non functional left kidney. Intravenous and retrograde pyelography revealed pyelocalyceal and ureteral dilatation and a large polypoid intraluminal lobulated mass in the pelvic ureter. Pathologic and immunohistochemical techniques showed a nonepithelial, stromal malignant tumour with appearance of smooth muscle cell differentiation: leiomyosarcoma. CONCLUSIONS: Primary leiomyosarcoma is an unusual ureteral obstructive etiology and should be considered in this condition. Correct preoperative diagnosis is rarely made and this tumour is diagnosed by pathological examination. Abdomino-pelvic computerized tomography scan and ultrasound should be performed before diagnosing a lesion with these features as a primitive ureteral tumour.

Adult↗

PubMedDate not supplied

Leiomyosarcoma of the ovarian vein: report of a case.

Venous leiomyosarcomas are rare tumors, generally originating from the inferior vena cava. Only two cases of ovarian vein leiomyosarcoma have been reported thus far. Herein we report the case of a 50-year-old woman with a leiomyosarcoma of the right ovarian vein. The patient underwent radical en bloc extirpation of the tumor followed by radiation therapy. The clinicopathologic features and treatment are discussed.

Female↗