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Benign Metastasizing Leiomyoma
Abstract
Benign metastasizing leiomyoma is a rare condition usually seen in reproductive age women which presented with multiple benign smooth muscle tumors in extra uterine sites, which display unusual growth pattern. In this report, a case of benign metastasizing leiomyoma is discussed.
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Introduction
Benign metastasizing leiomyoma (BML) is a rare disorder that affects women with a history of uterine leiomyoma, which is found to metastasise within extrauterine sites. The aetiology of BML remains unexplained. Because BML is rare, and most publications contain descriptions of single cases, no statistically determined time relations were found between the primary and secondary surgeries, which may have aetiological implications.
Objectives
To determine age before BML surgery, age during diagnosis of BML, type of prior surgery, and location of metastasis based on the literature.
Methods
A systematic review of four databases (Medline/PubMed, Embase, Web of Science, and Cochrane) covering articles published from 1 January 1965 to 10 April 2016. The inclusion criteria were full-text articles in English and articles containing case reports. Articles in languages other than English (39), articles containing incomplete data (14), i.e. no information regarding the time of surgery and/or the site of metastasis, articles bereft of case studies (25), and articles with access only to summaries, without access to the complete text (10) were excluded. Of 321 titles identified, only 126 articles met the aforementioned criteria.
Results and conclusions
The mean age during primary surgery and BML diagnosis was 38.5 years and 47.3 years, respectively. The most common surgery was total hysterectomy. The most frequent site of metastasis was the lungs; other organs were affected less frequently.The site of metastases and their number were not related to the longer time span between the patient’s initial surgery and occurrence of metastasis. The analysed data, such as the age during primary surgery, age during BML diagnosis, site and type of metastasis, do not provide us a clear answer. Thus, BML pathogenesis is most probably complex in nature and requires further multidirectional research.
We report a case of pulmonary benign metastasizing leiomyoma (BML) from the uterus in a 77-year-old woman. The patient presented for investigation of multiple pulmonary nodules on a routine chest roentgenogram. Because a preoperative diagnosis could not be made, the largest tumor, 3.5 cm in diameter, was resected from the right lower lobe, and histological examination confirmed BML. She had undergone hysterectomy with oophorectomy for uterine leiomyomas 12 years earlier, at the age of 65. The microscopic findings of the lung tumor were similar to those of the uterine leiomyoma, and both lesions were histologically benign. Although this disease is considered to be hormone-dependent, metastasis was found in this elderly postmenopausal woman whose tumor was negative for estrogen receptor.
We present a case of leiomyomatosis peritonealis disseminata (LPD) after myoma morcellation and review the literature using the keywords leiomyomatosis peritonealis disseminata and disseminated peritoneal leiomyomatosis. The search was conducted in Medline, EMBASE, and Cochrane Database of systematic reviews. We encountered 132 cases of LPD in the English literature; 113 in the reproductive age group, 7 in postmenopausal women, 6 in males, and another in a horse. The possible causes could be divided into hormonal, subperitoneal mesenchymal stem cells, metaplasia, genetic, or iatrogenic after morcellation of myoma during laparoscopic surgery. Our case and 4 others reported in the literature support the contribution of the iatrogenic theory. It appears that LPD could be due to metaplasia of mesenchymal cells of the peritoneum and, in susceptible women, leaving fragments of myoma in the abdominal cavity might contribute to the development of LPD. Accordingly, one should avoid leaving fragments of the uterus or myoma tissue in the abdominal cavity after morcellation.
Leiomyomatosis peritonealis disseminata is a pseudomalignant condition which mimics disseminated abdominal carcinoma. A new case is reported and compared to the 11 well-documented cases in the literature. All reported patients have experienced a benign clinical course. The peritoneal nodules are composed of histologically benign-appearing smooth-muscle cells, substantiated by electron-microscopic study. The histogenesis of this entity is discussed in the light of our ultrastructural findings and the observation of concurrent endometriosis. The theory of metaplasia of the subcoelomic mesenchyme in the pathogenesis of the leiomyomatous lesions is favored. The serum estrogen levels determined in our patient did not reveal any abnormal change. The inducing factor(s) remain to be identified. Also, an unusual sensitivity of the coelomic tissues in these patients, in response to metaplastic stimuli, is postulated as a possible contributing factor.
A case of histologically benign cardiac and pulmonary metastases from a uterine leiomyoma in a 45-year-old woman is reported. The solitary cardiac tumor consisted of five lobules with a stalk attached to the anterior papillary muscle of the right ventricle, which occupied the right ventricular cavity and almost completely obstructed the pulmonary main trunk in the systolic cycle. Multiple small nodules were found throughout both lungs. The cardiac tumor was resected at open heart surgery and open lung biopsy of the pulmonary lesion was simultaneously performed. Both of them were histologically identical to the apparently histologically benign uterine leiomyoma which had been resected five years previously. Based on the clinical and laboratory findings, it is postulated that antegrade metastases via the venous system resulted in the cardiac metastasis from the uterine leiomyoma and the secondary pulmonary metastasis from the cardiac tumor. In contrast to the light microscopic findings, ultrastructural examination suggested the possibility of malignancy associated with the presence of immature smooth muscle cells. This is the first reported case of a so-called benign metastasizing leiomyoma occurring in the heart.
A 40-year-old woman had multiple smooth muscle tumors in the left inguinal region, the bilateral thighs, the omentum, the peritoneum, and the right infundibulum pelvic ligament associated with uterine leiomyomas. She had a history of uterine leiomyomas, which were resected 13 years ago. Histopathologic evaluation revealed tumor masses composed of smooth muscle cells with relatively low cellularity, which were consistent with a diagnosis of leiomyoma. Tumor necrosis and nuclear atypia were absent. Mitotic figures were very scarce (less than 1 mitotic figure per 10 high-power fields). Immunohistochemical evaluation revealed a positive reaction of the tumor cells to muscle markers, estrogen receptors, and progesterone receptors. No pulmonary lesion was found. Similar instances of uterine leiomyomas with histologically benign extrauterine smooth muscle tumors have been reported. This curious condition has been referred to as "benign metastasizing leiomyoma," in which most of the reported cases involve the lungs. The distribution of extrauterine tumors in our case is very unusual and may be the first case with multiple leiomyomas in deep soft tissue of the limbs. Consideration was given to the concept that these may be of multifocal origin, rather than metastases.
Leiomyomatosis peritonealis disseminata (LPD) is a rare smooth muscle tumor. In the literature more than 100 cases have been described. LPD is characterized by multiple small nodules on the peritoneal surface, mimicking a malignant process with metastases, but generally demonstrates benign histologic features. Exposure to estrogen seems to play an etiologic role. Many patients have uterine leiomyomas as well. The diagnosis of LPD is easily made on biopsy. Reduction of estrogen exposure is generally sufficient to cause regression of LPD. Surgical castration or gonadotrophin releasing hormone agonists seem good alternatives in the case of progression or recurrence of LPD. In six patients a malignant leiomyosarcoma has been described shortly after the diagnosis of LPD was made. Five of these patients did not have uterine leiomyomas or exposure to exogenous or increased endogenous estrogen. The relationship with pregnancy in the sixth patient may be coincidental. Whether malignant transformation of LPD occurs remains uncertain. Characteristics of these patients differ from those of LPD patients and may indicate a high malignant potential, necessitating a different approach.
Leiomyomatosis peritonealis disseminata has been attributed to estrogen stimulation and is seen only rarely in postmenopausal women. In such cases, pathogenesis is uncertain.
Leiomyomatosis peritonealis disseminata tumors were resected from a postmenopausal woman. She was receiving tamoxifen therapy for breast cancer and had bilateral ovarian Brenner tumors. Estrogen and progesterone receptors were detected. Immunohistochemical analysis indicated that LH receptors were present.
Luteinizing hormone receptors were identified in leiomyomatosis peritonealis disseminata in one woman. Levels of FSH and LH increase after menopause, and immunohistochemical analysis showed the presence of LH receptors, so gonadotropin rather than estrogen stimulation might have contributed to development of leiomyomatosis peritonealis disseminata in this uncommon case.
We present an unusual case of benign metastasizing leiomyoma in association with intracaval leiomyomatosis. To our knowledge, this is the first reported case of metastasizing leiomyoma with coexistent intravenous leiomyomatosis (IVL). Magnetic resonance imaging is useful for the diagnosis of pelvic and caval IVL.
We report two patients with benign uterine leiomyoma metastasizing to the nervous system, respectively, to the skull base and to the spine. Although primary cranial and metastatic spinal leiomyomas have been rarely described, to our knowledge no case of benign leiomyoma metastasizing to the skull base has been reported before.
Two female patients with history of hysterectomy for benign leiomyoma, subsequently metastasizing to the lungs and smooth muscles of the skin, presented with a focal neurologic deficit. Magnetic resonance imaging revealed a sacral mass in one case and a skull base tumor in the other.
Both patients underwent surgery for resection of the lesions. Good postoperative results were obtained. Histologic examination of the surgical specimen revealed a benign metastasizing leiomyoma.
Benign metastasizing leiomyoma should be considered in the differential diagnosis of mass lesions in the sacral spine and skull base in patients who have a history of uterine leiomyoma or benign metastases of the same disease in organs outside the nervous system.
Benign metastasizing leiomyomas (BMLs) occur predominantly in women during reproductive years. The condition is characterized by uterine leiomyomas associated with the development, typically years later, of slow-growing metastatic lesions. The most commonly affected organs are the lungs, but BMLs have been reported in lymph nodes, deep soft tissues, mesentery, bones, the central nervous system, and the heart. In many cases, these lesions have an indolent course and are discovered rather incidentally. However, occasionally they can present with debilitating symptoms or even life-threatening complications. The presence of estrogen and progesterone receptors in these tumors supports their origin from uterine smooth muscle and, more importantly, makes them amenable to hormonal manipulation. Radical interventions, such as extensive tumor debulking and oophorectomy for hormonal control, although effective in many cases, are not always possible or desirable and carry significant morbidity. Here we present two cases of BMLs to illustrate the role of newer therapeutic agents, the estrogen receptor modulators and the aromatase inhibitors, in the hormonal manipulation of these tumors.
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