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We describe a rare case of leiomyosarcoma of the maxilla arising in a 71-year-old Japanese woman. The lesion was treated successfully by surgical resection with external irradiation of 70 Gy. No sign of recurrence or distant metastasis was present 3 years after operation. Histogenetically, this maxillary leiomyosarcoma apparently arose from the medial muscle of blood vessels or from primitive mesenchyme in the maxilla.
Cardiac leiomyosarcoma is a very rare entity that is found in less than 0.2 % of all cardiac tumors. At the time of primary diagnosis, it often shows advanced local invasion or may even be metastasized. Thus, complete resection can not easily be achieved. Cardiac transplantation has been reported as a therapeutic option. Here, we report on a case of a leiomyosarcoma reoccurrence arising 2 years after initial surgery. We performed a radical redo-extirpation without the necessity of transplantation.
Leiomyosarcoma of the inferior vena cava is rare and to date 145 cases have been reported. Here a new case of primary leiomyosarcoma of the inter-renal vena cava is reported. Surgical treatment consisted of excision of the vena cava on top of the renal arteries allowing radical resection. Reconstruction was original. The distal vena cava was sectioned and ligated just above the bifurcation and the free segment used to replace the excised part. In this the right venal vein was directly implanted. The left renal vein was reimplanted in the translocated segment via a venous (femoral vein) graft. Ligation of the distal vena cava was haemodynamically well-tolerated and the patient was free of symptoms at one year after operation. Control angiogram showed patency of the venous reconstruction. Early diagnosis is essential to improve patient survival, since it allows better radical resection, which is the only way of preventing the development of metastases.
To date, 12 cases of primary leiomyosarcomas of the pulmonary artery have been described in the literature. In only 3 instances was a surgical treatment attempted. We describe a patient with pulmonary artery leiomyosarcoma presenting in obstructive shock. Surgical excision under extracorporeal circulation allowed temporary relief, but the patient died in septic shock on the 10th postoperative day. Relevant clinical and pathological data are briefly reviewed.
Primary leiomyosarcoma of the heart is uncommon and we were able to find only 15 cases reported in the literature. Few of these cases were discovered during life and, as a result, even fewer have been treated surgically. We report a case of a right atrial leiomyosarcoma which was proven pathologically (histologically, immunohistochemically and ultrastructurally) after surgical resection. The clinical presentation, morphological, appearances and outcome are compared with those reported in the literature.
This is a report on a patient of 74 years of age presenting with ear pain and bleeding granulations on his tympanic membrane. X-ray of the mastoid revealed partial destruction of the bone structures and reduced pneumatisation. A tumour that was mainly located in the temporal bone was surgically removed and proved to be a leiomyosarcoma. This is the first case of a leiomyosarcoma of the temporal bone to be described. Differential diagnosis and therapy of this tumour are discussed.
The authors describe the occurrence of a confirmed leiomyosarcoma and a tumor suspected of being a leiomyosarcoma in 23-year-old twin sisters. In one of them the tumor had caused clinical symptoms; in the second it was detected at the presymptomatic stage as a result of increased attention after detection of the first tumor. In the first case treatment consisted in an extended abdominal hysterectomy, sparing one ovary, with bilateral interiliac lymph node extirpation; in the second case, in an extended abdominal hysterectomy with both adnexae and bilateral interiliac lymph node extirpation. So far - 13 and 10 months after the initial treatment, respectively - there are no signs of continued tumor growth in either case.
From 1960 through 1983 sixty-four so-called pure mesenchymal homologous sarcomas of the uterus were observed at the Women's University Hospital Zürich. A review of the histological slides revealed that only 40 cases represented true sarcomas according to the contemporary diagnostic criteria. These were 25 leiomyosarcomas and 15 endometrial stromal sarcomas. Leiomyosarcomas: The leading symptoms in these patients are uncharacteristic abdominal pain (15/25) as well as bleeding disturbances (11/25), the latter being present with equal frequency in the pre- and post-menopause. In 7 of the 11 patients with bleeding abnormalities the diagnosis was already made on examination of curettings, in all the other patients after laparotomy only. Sixteen patients had Stage I, four Stage III and five Stage IV tumors. The treatment consisted in surgery alone (9/25), surgery combined with radiotherapy (11/25) or with chemotherapy (2/25) as well as radiotherapy alone (1/25). In two cases no therapy was undertaken because of advanced cachexia of the patients. Only three patients are still alive without recurrence 4 to 12 years after the diagnosis, all had Stage I tumors. Endometrial stromal sarcomas: In these patients bleeding abnormalities represent the leading symptom (14/15), especially postmenopausal bleeding (13/14). In all cases with vaginal bloody discharge the diagnosis was made on examination of the curettings, in a single case after laparotomy only. Eight patients had Stage I, three Stage II, three Stage III and one Stage IV tumors. The treatment consisted of surgery alone (4/15) or in combination with radiotherapy (4/15), radiotherapy alone (6/15) and chemotherapy alone (1/15).(ABSTRACT TRUNCATED AT 250 WORDS)
In two men (69 and 65 years old) a gastric epithelioid leiomyosarcoma more than 25 cm in diameter first manifested itself by an increase in abdominal girth and ascites. In case 1, a cylindrical mass was palpable in the abdomen increasing in size over two years. Sonography and computed tomography showed a large cystic tumour which could not be related to any one organ. Cytology of the ascites fluid did not reveal any tumour cells. At operation a pedunculated tumour, 29 x 20 x 9 cm, was found, originating from the stomach. Six litres of ascites fluid were aspirated. There were no detectable abdominal metastases. The patient died nearly 4 years later after the tumour had been resected, three further laparotomies had to be performed because of extensive metastases to peritoneum and liver from the leiomyosarcoma. In case 2, no abdominal mass was palpable, but the patient's girth had increased over the last year. Sonography revealed a cystic tumour, about 30 cm in diameter. Examination of haemorrhagic ascites fluid (700 ml) did not show any tumour cells. The tumour, originating from the stomach wall, was excised (no metastases were seen). The patient remains symptom-free after 35 months.
The authors report a case of leiomyosarcoma of the cecum and review all the Italian reports of mesenchymal neoplasms registered in the RMS-Italy register. They emphasize the extreme rarity of intestinal leiomyosarcoma, especially with colon involvement: their case appears to be the only one registered in Italy in the last 12 years.
A patient passed a large tissue mass at home on postpartum day 1. Pathologic examination demonstrated leiomyosarcoma. A total abdominal hysterectomy was performed, which demonstrated residual disease. All tissue passed in the puerperium should be evaluated to ensure that significant pathologic conditions are not overlooked. Conservative management of leiomyosarcomas is controversial.
The case of primary leiomyosarcoma of the ovary in a 20-year-old Italian woman with nevoid basal cell carcinoma syndrome (NBCC) is described. This report first shows the association of ovarian leiomyosarcoma with NBCC, expanding the complex nature of this syndrome.
Vascular leiomyosarcomas are rare tumors, and only 21 have been described as developing in the systemic arteries. We present a case, the sixth in the literature, of a leiomyosarcoma originating in the popliteal artery of a 67-year-old woman treated with en bloc excision of the neoplasia and the artery followed by a popliteal tibioperoneal trunk bypass. The patient then was underwent adjuvant radiotherapy and chemotherapy. After 31 months of follow-up, the patient has mild claudication but does not have either recurrence or metastasis.
We present a case of an inflammatory leiomyosarcoma, a variant of smooth muscle cell tumor that could be mistaken for inflammatory malignant fibrous histiocytoma. The inflammatory cell infiltrates obscured the basic architecture of the tumor, and immunohistochemistry was required to arrive at the proper diagnosis. Since inflammatory leiomyosarcomas are rare and possibly misdiagnosed neoplasms, there are not enough data to indicate whether they deserve to be separated from other malignant smooth muscle cell neoplasms. By reporting this case we hope that more pathologists will become aware of this tumor type and that more cases will be collected for future clinicopathologic studies.
OBJECTIVE: To present our experience of leiomyosarcoma of the gastrointestinal tract during a 10 year period and review similar publications. DESIGN: Retrospective study. SETTING: Teaching hospital, Greece. SUBJECTS: 14 patients who were treated for leiomyosarcoma between 1983 and 1993. INTERVENTIONS: Tumours were diagnosed by endoscopy, computed tomography, and contrast examination. All patients were treated by resection and no adjuvant treatment was given. MAIN OUTCOME MEASURES: Morbidity, mortality, and outcome. RESULTS: The diagnosis was made preoperatively in 10/14 patients: 4 of the tumours were locally advanced at the time of laparotomy, and two had liver metastases. Resection was potentially curative (all macroscopic tumour removed) in 12 patients (86%). Three patients died of recurrent disease at 5, 8 and 63 months, respectively. The mean follow up in the 11 survivors was 44 months (range 24-83). CONCLUSION: Resection is the treatment of choice for these tumours. A multicentre trial would be required to assess the effect of adjuvant chemotherapy.
Leiomyosarcoma is a rare malignant tumor originating from the smooth muscular tissue in any part of the organism. The only therapy is its complete removal. We describe herein the operative treatment of a retroperitoneal leiomyosarcoma with gasless laparoscopic complete removal. The procedure was successfully performed in a consenting woman with an abdominal mass. Gasless laparoscopic removal was performed with a mechanical retractor (Laparolift, Origin Medsystem Inc.), obviating the creation of the pneumoperitoneum and of the sealed environment. The technique is a simple, safe, and effective surgical method. Gasless technique guarantees a clear vision, makes possible continuous suction of smoke and fluids, and allows the use of conventional instruments and easy management of suturing. The present case has proved to be another abdominal procedure that can be carried out with all the advantages of gasless miniinvasive surgery.
Syrian hamsters treated with estrogen and androgen for 8 months develop leiomyosarcomas in the vas deferens. Metabolism of estrogen by cytochrome P450s (CYPs) produces catechols and reactive oxygen species, and may contribute to tumor formation. To examine this issue, male hamsters were treated with 17 beta-estradiol (E2), testosterone propionate (TP) or both hormones. Reproductive tract tissues from control and treated animals were immunostained with antibodies specific for four CYP enzymes (1A1, 1A2, 1B1 and 3A1/2). Immunoreactive CYP1A1 was not found in the reproductive tract of control or treated animals. In untreated hamsters, CYP1A2 was detected only in principal cells of the caput epididymis. TP alone had no effect, but treatment with E2 induced expression of CYP1A2 in columnar epithelial cells throughout the epididymis and lining of the vas deferens. Treatment with E2 + TP blocked the induction of CYP1A2 seen in surface epithelial cells treated with E2 alone, but not the constitutive expression of this enzyme. Instead, simultaneous exposure to both hormones induced CYP1A2 in basal cells of the epididymis and vas deferens. CYP3A1/2 was not detected in the reproductive tract of control or TP-treated males, but immunostaining was induced in the inner layer of vas deferens smooth muscle by E2, and in all smooth muscle layers by dual hormone treatment. In controls, CYP1B1 was present in smooth muscle lining the epididymis and surrounding the vas deferens and dual hormone treatment increased staining intensity for CYP1B1 in these cells. Immunoreactive CYP1A2 was not detectable in leiomyosarcomas but the enzyme was present in both columnar and basal cells of the vas deferens epithelium adjacent to the tumors. In contrast, tumor cells showed heterogeneous expression of both CYP1B1 and CYP3A1/2. The relationships between hormone treatment, differential CYP expression and tumor formation strengthen our hypothesis that metabolism of estrogen is an important element in this model of hormonal carcinogenesis.
We report a rare case demonstrating the relationship between secondary amyloidosis and uterine leiomyosarcoma. A 59-year-old female with high fever was referred to our hospital. Laboratory tests revealed increased white blood cells, eosinophilia and an accelerated erythrocyte sedimentation rate. Endoscopic examination of the stomach and colon revealed amyloid deposits in their mucosa. The patient showed no symptoms that suggested amyloidosis. No other organs or tissues were surveyed for amyloid deposition. Ga scintigraphy, computed tomography and magnetic resonance imaging suggested necrotic infectious leiomyoma of the uterus, which was considered to be the cause of the fever. The patient underwent total hysterectomy. The histological diagnosis of the mass revealed a low-grade uterine leiomyosarcoma with necrosis. Amyloid deposits in the gastric mucosa disappeared 1 year after the operation. In this case, amyloid deposition was detected by endoscopic biopsy before clinical manifestations. The deposition was reversible and was successfully treated. Thus, it is logical and useful to undertake endoscopic mucosa biopsy to check for amyloid deposition in patients with systemic inflammation, whose serum amyloid A protein level has been high for several months. In addition, peripheral eosinophilia was also detected in this case. Although eosinophilia associated with malignant tumor has been recognized, it is an uncommon occurrence.