Pulmonary benign metastasizing leiomyoma presenting as acute hypoxemic respiratory failure: a case report.

Pulmonary benign metastasizing leiomyoma is an uncommon condition, predominantly affecting women of childbearing age with a history of uterine smooth muscle tumors and uterine leiomyoma surgery for uterine leiomyoma. The progression of PBML is often unpredictable and depends on the extent of lung involvement. Generally, most patients remain asymptomatic, but a minority may experience coughing, wheezing, or shortness of breath, which are frequently misdiagnosed as pneumonia. consequently, this presents significant challenges in both treatment and nursing care before diagnosis. This paper reports the case of a 35-year-old woman primarily diagnosed with acute hypoxic respiratory failure who was transferred from the emergency room to the intensive care unit. The initial computed tomography scan of the patient's lungs indicated diffuse interstitial pneumonia, but the sequencing of the alveolar lavage fluid pathogen macro did not detect any bacteria, fungi, or viruses. Moreover, the patient remained in a persistent hypoxic state before the definitive diagnosis. Therefore, our focus was on maintaining the airway patency of the patient, using prone ventilation, inhaling nitric oxide, monitoring electrical impedance tomography, and preventing ventilator-associated pneumonia to improve oxygenation, while awaiting immunohistochemical staining of the patient's biopsied lung tissue. This would help us clarify the diagnosis and treat it based on etiology. After meticulous treatment and nursing care, the patient was weaned off the ventilator after 26 days and transferred to the respiratory ward after 40 days. This case study may serve as a reference for clinical practice and assist patients suffering from PBML.

Benign metastasizing uterine leiomyoma with lymphatic and pulmonary metastases: a case report and literature review.

BACKGROUND: Benign metastasizing leiomyoma (BML) is a rare disease usually observed in women of reproductive or premenopausal age with a history of uterine myomectomy or hysterectomy. The most common sites of metastases are the pulmonary, and other sites include heart, bones, liver, lymph nodes, bladder, skeletal muscles, and central nervous system. Here, we report a case of a 50 year-old woman with a history of hysterectomy who was initially suspected of uterine sarcoma but was finally confirmed to have BML with lung and lymph node metastases, and discuss the treatment and prognosis of BML. CASE PRESENTATION: A 50 year-old woman with a history of total abdominal hysterectomy presented with mild but persistent abdominal pain for more than 3 months. She was suspected of having uterine sarcoma before surgery and laparoscopic extensive debulking surgery including bilateral oophorectomy, pelvic and para-aortic lymph node dissection to the level of the left renal vein, and transcutaneous dissection of the right inguinal lymph nodes. Pathology confirmed a benign leiomyoma, and the patient was diagnosed with BML. No medication was administered after the surgery, and the follow-up was of no significance. CONCLUSION: Benign metastasizing leiomyoma (BML) is a rare disorder in which histologically benign smooth muscle tumors metastasize to extrauterine sites. Metastases are commonly observed in the lung, liver, lymph nodes, skin, bladder, esophagus, and skeletal muscles. BML is usually misdiagnosed as a malignant tumor before surgery until the pathology confirms its benign nature. However, this treatment remains controversial and undetermined. The prognosis is usually favorable owing to its benign nature.

Pulmonary benign metastasizing leiomyoma in patients aged 45 years and younger: clinical features and novelty in treatment.

BACKGROUND: Pulmonary benign metastasizing leiomyoma (PBML) is the most common extrauterine spread of uterine leiomyoma, and its biological behavior is traditionally thought to be hormone dependent. Studies on older PBML patients have been previously reported, but limited literature has been published regarding the clinical features and treatment of PBML in young women. METHODS: A total of 65 cases of PBML in women aged 45 years and younger were reviewed, including 56 cases selected from PubMed and 9 cases from our hospital. The clinical characteristics and management of these patients were analyzed. RESULTS: The median age of all the patients at diagnosis was 39.0 years. PBML most commonly presented as bilateral solid lesions (60.9%), with other rare imaging manifestations. The median interval time from a pertinent gynecologic procedure to diagnosis was 6.0 years. A total of 16.7% of patients received careful observation, and all achieved stable status in a median follow-up time of 18.0 months. A total of 71.4% of patients were administered anti-estrogen therapies, including surgical castration (33.3%), gonadotropin-releasing hormone analog (23.8%) and anti-estrogen drugs (14.3%). Eight of 42 patients underwent surgical resection of metastatic lesions. Patients who underwent curative surgery for the removal of pulmonary lesions combined with adjuvant anti-estrogen therapies had favorable outcomes compared with those who only underwent surgical resection. The disease control rates of surgical castration, gonadotropin-releasing hormone analog, and anti-estrogen drugs were 85.7%, 90.0%, and 50.0%, respectively. For two patients, sirolimus (rapamycin) achieved successful relief of symptoms and control of pulmonary lesions without lowering hormone levels and causing estrogen deficiency symptoms. CONCLUSIONS: In the absence of standard treatment guidelines for PBML, maintaining a low-estrogen environment using different kinds of antiestrogen therapies has been the mainstream strategy and has satisfying curative effects. A wait-and-see strategy might be an option, but therapeutic approaches must be contemplated when complications or symptoms progress. For PBML in young women, the negative effect on ovarian function of anti-estrogen treatment, especially surgical castration, should be considered. Sirolimus might be a new treatment option for young PBML patients, especially for those who want to preserve ovarian function.

Lung metastases and subsequent malignant transformation of a fumarate hydratase -deficient uterine leiomyoma.

Uterine leiomyomas, or fibroids, are very common smooth muscle tumors. Their potential to metastasize or transform into leiomyosarcomas is extremely low. Here, we report a patient who underwent hysterectomy due to a large leiomyoma and who was diagnosed with pulmonary tumors seven and nine years later. Histopathological re-evaluation confirmed the cellular leiomyoma diagnosis for the uterine tumor, whereas the pulmonary tumors met the diagnostic criteria of a leiomyosarcoma. Whole-exome sequencing revealed very similar mutational profiles in all three tumors, including a somatic homozygous deletion in a rare, but well-established leiomyoma driver gene FH. Tumor evolution analysis confirmed the clonal origin of all three tumors. In addition to mutations shared by all three tumors, pulmonary tumors harbored additional alterations affecting e.g. the cancer-associated genes NRG1 and MYOCD. The second pulmonary leiomyosarcoma harbored additional changes, including a mutation in FGFR1. In global gene expression profiling, the uterine tumor showed similar expression patterns as other FH-deficient leiomyomas. Taken together, this comprehensive molecular data supports the occasional metastatic capability and malignant transformation of uterine leiomyomas. Further studies are required to confirm whether FH-deficient tumors and/or tumors with cellular histopathology have higher malignant potential than other uterine leiomyomas.

The role of miRNA-221 and miRNA-126 in patients with benign metastasizing leiomyoma of the lung: an overview with new interesting scenarios.

Benign metastasizing leiomyoma (BML) is a rare disease characterized by extrauterine benign leiomyomatosis in patients with a previous or concomitant history of uterine leiomyoma. Currently, there are no specific criteria to predict the metastasizing ability of the uterine leiomyoma and the risk of malignant degeneration of pulmonary BML, and these are the aims of this study. We analyzed 10 uterine (three leiomyomas, four leiomyomas that gave rise to lung BML, three healthy tissues) and 11 pulmonary tissue samples (eight lung BML, three healthy tissues). Interestingly, one of the BML lesions exceptionally evolved into a leiomyosarcoma (case 2). Uterine leiomyoma microvascular density (MVD) was higher in the patients with uterine leiomyomas that gave rise to lung BML, reaching a peak in case 2. Strong positivity for the estrogen (ER) and progesterone (PR) receptors and a low proliferation index (Ki67 < 1%) were discovered both in patients with uterine leiomyoma and in patients with BML. Interestingly, in case 2, the last dedifferentiated leiomyosarcoma showed a weaker ER and PR positivity with a higher proliferation index (Ki67:30%). Regarding the uterine miRNA-126, a trend toward a hypo-expression between uterine leiomyoma and uterine leiomyoma that gave rise to lung BML was discovered, reaching the lowest level in case 2. Considering the pulmonary samples, we observed a higher miRNA-221 and a lower miRNA-126 expression in the leiomyosarcoma. We tried to better elucidate the biological behaviour of this rare disease. The analysis of the miRNA-221 and miRNA-126 could offer new diagnostic, prognostic and therapeutic perspectives.

Benign metastasizing leiomyoma in femur and thigh with a history of uterine leiomyoma: A case report and literature review.

Benign metastasizing leiomyoma (BML) is a rare disease that is characterized by well-differentiated smooth muscle tumors occurring extrauterine site in women with a history of uterine leiomyoma. The lung is the most common metastatic site for BML. A 48-year-old woman, who had histories of laparoscopic myomectomy and transabdominal total hysterectomy, visited an orthopedics complaining of a mass in her left thigh and difficulty in walking. Magnetic resonance imaging (MRI) and fluorine 18 fluorodeoxyglucose positron emission tomography/computed tomography revealed multiple mass lesions in her both thighs and left femur as well as both lungs. She was referred to our hospital for further examination. We diagnosed her tumors as BML according to histopathological analysis of tumor specimen. The left thigh tumor was resected and the treatment with gonadotropin releasing hormone agonist regressed the size of the residual tumors by approximately 30%. BML should be considered when multiple soft tissue tumors are found in women with a history of leiomyomas.

[Benign metastasizing uterine leiomyoma with lung metastasis: problems of diagnosis and treatment]. / Dobrokachestvennaya metastaziruyushchaya leiomioma matki s porazheniem legkikh: problemy diagnostiki i lecheniya.

Uterine leiomyoma (LM) is a smooth muscle tumor occurring in 30% of women of reproductive age. This neoplasm is the most common benign tumor of female genitals. Uterus is the most frequent localization of tumor. However, LM can be detected in external genitalia, ovaries, bladder and urethra. Despite the benign nature, LM is characterized by metastasizing as one of the main properties of malignant neoplasms. For the first time, Steiner described this case in 1939. The author found similar histological structure of pulmonary and uterine neoplasms. LM metastases in bone tissue, skin, mediastinum, muscles, lymph nodes, omentum and retroperitoneal space are rarer. In this manuscript, we report complex treatment of benign metastasizing uterine leiomyoma followed by lung metastasis.

Loss of ATRX/DAXX expression and alternative lengthening of telomeres in uterine leiomyomas.

BACKGROUND: Uterine leiomyomas (ULs) are the most common gynecologic tumors and affect 3 of every 4 women by the age of 50 years. The majority of ULs are classified as conventional tumors, whereas 10% represent various histopathological subtypes with features that mimic malignancy. These subtypes include cellular and mitotically active ULs and ULs with bizarre nuclei. Uterine leiomyosarcoma (ULMS), the malignant counterpart of UL, is an aggressive cancer with poor overall survival. The early diagnosis and preoperative differentiation of ULMS from UL are often challenging because their symptoms and morphology resemble one another. Recent studies have shown frequent loss of alpha-thalassemia/mental retardation syndrome X-linked (ATRX) or death domain-associated protein (DAXX) expression in ULMS, and this is often associated with an alternative lengthening of telomeres (ALT) phenotype. METHODS: To investigate ATRX and DAXX expression and the presence of ALT in UL subtypes, immunohistochemical and telomere-specific fluorescence in situ hybridization analyses were performed. The study material consisted of 142 formalin-fixed, paraffin-embedded tissue samples representing various UL subtypes and 64 conventional ULs. RESULTS: A loss of ATRX or DAXX and/or ALT was detected in 6.3% of the histopathological UL subtype samples (9 of 142). Two patients whose ULs showed either ATRX loss or ALT were later diagnosed with a pulmonary smooth muscle tumor. Pulmonary tumors displayed molecular alterations found in the corresponding uterine tumors, which indicated metastasis to the lungs. All conventional ULs displayed normal ATRX, DAXX, and telomeres. CONCLUSIONS: These results highlight the differences between conventional and histopathologically atypical ULs and indicate that some UL subtype tumors may harbor long-term malignant potential.

Whole exome sequencing of benign pulmonary metastasizing leiomyoma reveals mutation in the BMP8B gene.

BACKGROUND: Benign metastasizing leiomyoma (BML) is an orphan neoplasm commonly characterized by pulmonary metastases consisting of smooth muscle cells. Patients with BML have usually a current or previous uterine leiomyoma, which is therefore suggested to be the most probable source of this tumour. The purpose of this case report was to determine the possible genetic grounds for pulmonary BML. CASE PRESENTATION: We present a case report in an asymptomatic 44-year-old female patient, who has developed uterine leiomyoma with subsequent pulmonary BML. Whole exome sequencing (WES) was used to detect somatic mutations in BML lesion. Somatic single nucleotide mutations were identified by comparing the WES data between the pulmonary metastasis and blood sample of the same BML patient. One heterozygous somatic mutation was selected for validation by Sanger sequencing. Clonality of the pulmonary metastasis and uterine leiomyoma was assessed by X-chromosome inactivation assay. CONCLUSIONS: We describe a potentially deleterious somatic heterozygous mutation in bone morphogenetic protein 8B (BMP8B) gene (c.1139A > G, Tyr380Cys) that was identified in the pulmonary metastasis and was absent from blood and uterine leiomyoma, and may play a facilitating role in the metastasizing of BML. The clonality assay confirmed a skewed pattern of X-chromosome inactivation, suggesting monoclonal origin of the pulmonary metastases.

[The clinical characteristics of pulmonary benign metastasizing leiomyoma].

Objective: Pulmonary benign metastasizing leiomyoma (PBML) is a rare entity that leiomyoma of uterus metastasized to the lung. The clinical characteristics of this rare disease were analyzed in this article. Methods: The detailed clinical records of 7 patients diagnosed as PBML at Peking Union Medical College Hospital between January 2001 and June 2015 were reviewed. Results: All patients were women with median age of 44 years (range 28-62). Symptoms included dyspnea (2/7), chest pain (1/7), cyanosis (1/7), cough (1/7) and bloody sputum (1/7), while 4/7 cases were asymptomatic. Six patients had the past-history of leiomyoma of uterus 20 months to 14 years ago among whom 5 patients received hysterectomy. Chest CT showed bilateral, random-distributed multiple round solid nodules, or diffuse-distributed miliary nodules, or single solid nodule, even some small cavities. Extra-pulmonary metastasis was found in left superclavicular lymph node (1 case) and right heart (1 case). Histological tissues were obtained by video-assisted thoracic surgery lung biopsy (4/7), mass resection on tricuspid valve (1/7), transbronchil lung biopsy (1/7), and CT-guided percutaneous lung biopsy (1/7). Pathology showed an interlacing pattern by spindle cells having elongated nuclei without cellular atypia. Ki-67 index was less than 1%. Molecules such as smooth muscle antibody, estrogen receptor (ER) and progestrone receptor (PR) were positive in immunohistochemistry staining. Neither letrozole nor zoladex was effective. Two patients responded to bilateral adnexectomy, presenting as shrunk nodules. No relapsed disease was seen in one patient with single nodule after resection. There was only one patient with disease-related mortality, whose chest CT showed milliary nodules. Conclusion: Although CT findings of PBML are similar to malignancies, the clinical outcome is good. Despite the positive expression of ER and PR, the effectiveness of hormone related treatment is limited. And periodical follow up is suggested even to those uneventful patients.

Benign metastasizing leiomyoma in triple location: lungs, parametria and appendix.

Benign metastasizing leiomyoma (BML) usually are situated in one organ, most often in lungs. BML patients typically have a history of uterine leiomyoma treated with hysterectomy, myomectomy or subtotal hysterectomy. The aim of the study was to present the case of a 53-year-old woman with triple location in the lungs, parametria and appendix. She had undergone a myomectomy 26 years earlier. In 2015, she was admitted to the surgical department because of abdominal pain, whereupon a cholecystectomy was performed. CT scans showed pelvic mass with pulmonary metastasis. Upon discharge the patient was referred to the Gynecology Clinic, where a laparotomy was performed. The intraoperative findings were: 1) uterus with multiple leiomyomas, 2) four tumors in the parametria, 3) tumor connected to the appendix. A subtotal hysterectomy, with a bilateral salpingo-oophorectomy, removal of the tumors from the parametria and appendectomy was performed. Pathology confirmed the diagnosis based on morphology and immunohistochemical staining (strongly positive for estrogen receptors and SMA, while Ki67 was very low, below 1%). Upon postoperative recovery, the patient was referred to the Thoracic Surgery Department. During the thoracotomy, multiple nodes, surrounded by lung parenchyma, were revealed. Wedge resection was performed, for localized pulmonary lesions, and sent for pathological examination. The final pathological diagnosis was benign metastasizing leiomyomatosis. In conclusion, the triple location of BML could possibly be a result of a parallel different metastasizing mechanism, although it is impossible to exclude one mechanism, which may be the cause of the metastases in three locations.

Benign Metastasizing Leiomyoma: New insights into a rare disease with an obscure etiopathogenesis.

BACKGROUND: Benign metastasizing leiomyoma (BML) is a rare disease with an unknown etiopathogenesis that mostly affects middle-aged women with uterine leiomyoma. Many metastatic nodules outside the uterus characterize the condition. The metastases are smooth muscle lesions without malignancy. Morphologically and immunohistochemically, they resemble uterine leiomyomas, indicating a shared clonal origin. The lungs are the most prevalent site for incidental metastasis detection. BML has a relatively slow progression and good prognosis, and historically, there has been a lack of established guidelines for its treatment. CASE PRESENTATION: Herein, we report a case of BML in a patient with multiple metastases. Through extensive histological and immunohistochemical analyses, this complex case enabled not only the definitive diagnosis of BML, but also shed light on its complex etiopathogenesis. CONCLUSION: This study presents novel histology evidence suggesting a potential causal relationship between metaplasia and the development of BML.

Multiple benign metastasizing leiomyoma in the abdominal wall: a case report and literature review.

Benign metastasizing leiomyoma (BML) is a rare disease that results from metastasis of uterine leiomyoma to distant sites with benign pathologic features. The lung is the most common metastatic site for BML. This report describes the case of a 49-year-old woman who presented with a mass in the abdominal wall with a surgical history of uterine myomectomy. Ultrasound and Magnetic resonance imaging (MRI) revealed multiple mass lesions. The histopathology of the mass specimen indicated BML. The imaging and clinical features of BML are discussed based on the characteristics of this case and related literature reports.

Intravenous leiomyomatosis in the inferior vena cava and right atrium with pulmonary benign metastasizing leiomyoma secondary to a pelvic arteriovenous fistula: a case report and literature review.

BACKGROUND: To report the diagnosis and treatment of a rare disease of intravenous leiomyomatosis (IVL) originating from the uterus, growing in the inferior vena cava (IVC) and extending into the right atrium (RA) associated with a pelvic arteriovenous fistula (AVF). This is the first reported case of IVL in the IVC and RA with pulmonary benign metastasizing leiomyoma (PBML) secondary to a pelvic AVF despite the use of GnRH agonists in a non-menopausal woman. CASE PRESENTATION: The patient was a 50-year-old premenopausal woman with a history of surgical resection for and anti-estrogen conservative drug for pulmonary benign metastasizing leiomyoma (PBML) 5 years. The patient nevertheless developed IVL in the IVC, internal iliac vein and RA accompanied by AVF. Vaginal ultrasound combined with echocardiography and Computerized tomographic venography imaging assists in the diagnosis of IVL combined with AVF, with histopathology and immunohistochemistry ultimately confirming the diagnosis. the patient ultimately was performed with a combination of hysterectomy, bilateral adnexectomy, and resection of tumors in the IVC and RA without cardiopulmonary bypass and sternotomy. CONCLUSION: BML may be difficult to control with incomplete removal of the uterus and ovaries even with the use of anti-estrogenic medications, and medically induced AVF resulting from fibroid surgery may accelerate this process and the development of IVL.

Benign Metastasizing Leiomyoma: Is "Wait and Watch" Strategy Feasible?

Benign metastasizing leiomyoma (BML) is a rare disease that affects women with a history of uterine leiomyoma. The aim of this study is to investigate the clinical characteristics and outcome patterns. We collected 385 cases from previous reports indexed in PubMed and Google Scholar and made a thorough review. All relevant clinical parameters were carefully reviewed, including age at diagnosis, clinical presentations, course of disease, medical history, imaging, molecular tests, treatment, and outcomes. Univariate survival analysis was performed to investigate the effects of treatment strategies on outcomes. The mean age at diagnosis was 46.2 years. The most common site for the metastasis was lung, and followed by abdomen/pelvis/retroperitoneum, bone, lymph nodes, and heart. The medical histories and molecular alterations were non-specific, and the pathogenesis was still unclear. Due to its unresectable nature, hormone deprivation treatment, including oophorectomy and hormone drugs, is the most effective strategy to reduce or delay tumor progression. The present study may provide a useful consultation for diagnosing and managing BMLs.

Benign Metastasizing Leiomyoma Causing Severe Tricuspid Regurgitation and Heart Failure.

We present the case of a 51-year-old woman with severe tricuspid valve regurgitation due to aseptic tricuspid valve vegetation. She presented with bilateral lower extremity edema and a tricuspid valve vegetation was found on echocardiography. Initially, infectious and autoimmune causes of valve vegetation were considered; however, on biopsy, the mass was ultimately found to be a benign metastasizing leiomyoma (BML). Additional history revealed clinical features consistent with uterine leiomyomas, which metastasized to all leaflets the tricuspid valve, causing symptoms of heart failure. Benign metastasizing leiomyoma is rare itself, but when found, typically presents as asymptomatic pulmonary nodules. Mechanism of spread is unknown. Diagnosis is typically made long after a hysterectomy or fibroidectomy, but in our case, the BML was found prior to an actual fibroid diagnosis. By comparison, metastasis to the heart is extremely rare and has a higher potential for morbidity. Our patient required open heart surgery and tricuspid valve replacement for management of her symptoms, but her risk of further or recurrent metastasis going forward is unknown. Management strategy to prevent metastases in such cases of aggressive disease is not an established protocol and needs to be further studied.

The panorama of pulmonary leiomyoma: A tale of two tumours.

Pulmonary leiomyoma has a diverse clinical spectrum of disease. Here we describe two patients with vastly different presentations. The first case is a 23-year-old female with a chronic cough and no visible tracheal lesion on computer tomography (CT) chest imaging. Flexible bronchoscopy revealed a small tracheal nodule, with histopathology confirming pulmonary leiomyoma. The second case is a 57-year-old female with a painless abdominal mass. CT imaging revealed widespread lung 'cannonball' nodules. Percutaneous biopsy confirmed metastatic leiomyoma. After progression on surveillance and endocrine therapy, she was commenced on doxorubicin with interval radiological improvement. These cases highlight the clinical heterogeneity in this disease, and thus, complexity in devising standardized diagnostic and therapeutic protocols.

Pulmonary benign metastasizing uterine leiomyoma (PBML): a case report and review of the literature.

Benign metastasizing leiomyoma (BML) is a rare disorder characterized by the presence of benign smooth muscle tumours in extrauterine sites, typically the lungs. It classically involves perimenopausal women with a history of uterine surgery. The condition follows an indolent course but may cause clinical symptoms with large or widespread lesions. Case presentation: The authors report the case of a 47-year-old female who presented with a 6-month history of irregular vaginal bleeding and severe hot flushes. The patient had no previous history of gynaecological surgery. Ultrasonography and subsequent MRI identified a suspicious 105×65 mm mass involving the right uterine cornu and broad ligament. Computed tomography identified bilateral lung nodules, suspicious for metastases. Histological assessment of the final uterine surgical specimen identified a benign dissecting leiomyoma involving the broad ligament and cervix. BML was diagnosed after thoracoscopic resection of a lung lesion which revealed a histologically identical tumour with entrapped normal lung alveoli. Clinical discussion: This case shows that there is a minority of patients without previous uterine surgery who still go on to develop pulmonary BML. In our case, a combined treatment approach was adopted, involving substitution of hormone replacement therapy to a non-hormonal alternative, thoracoscopic resection of lung lesions and interval surveillance imaging of the chest. Conclusions: BML is a rare condition but should be considered as a differential in women with pulmonary nodules and a history of uterine leiomyomata. Its diagnosis and subsequent counselling can be challenging; therefore cases should be treated by multidisciplinary teams in tertiary specialized centres.

A Rare Case of Benign Metastasizing Leiomyoma Causing T11 Spinal Cord Compression: A Report and Literature Review.

Diagnosis of uterine smooth muscle tumors depends upon histologic characteristics as both benign and malignant share clinical features such as metastases. A benign metastasizing leiomyoma is a rare benign smooth muscle tumor that metastasizes to extrauterine sites with simultaneous uterine leiomyoma or previously biopsy-proven leiomyoma during myomectomy or hysterectomy. Benign metastasizing leiomyoma metastasizes outside the uterus, predominantly to the lungs and lymph nodes. However, the involvement of other organs, such as the heart, liver, spine, and soft tissue, is also reported. Here, we present a case of a 42-year-old woman with a history of uterine leiomyoma with prior myomectomy and hysterectomy, who presented with worsening back pain and lower extremity weakness and was found to have an acute cord compression, a serious complication caused by mass effect and a medical emergency that requires prompt attention to prevent permanent spinal cord damage. Sacral soft tissue biopsy and T11 spinal bone biopsy both demonstrated leiomyoma with immunostains positive for desmin, smooth muscle actin, and positive estrogen and progesterone receptors. No atypia, necrosis, and mitosis were identified. The patient had hepatic and pulmonary metastasis on imaging. The final diagnosis was benign metastasizing leiomyoma. There is no standard treatment for benign metastasizing leiomyoma. Both surgical and pharmacological approaches are employed. Although most cases are benign, there is a possibility for life-threatening complications. Benign metastasizing leiomyomas can be considered when multiple soft tissue tumors are found in premenopausal women with a history of uterine leiomyomas. Multidisciplinary discussion between oncologists, gynecologists, and relevant specialists is crucial in the optimal evaluation and treatment of benign metastasizing leiomyoma.

Pancreatic metastasis of leiomyoma found 27 years after uterine fibroid surgery.

A 59-year-old female was referred to our hospital for pancreatic tumor. She had undergone resection of uterine fibroids 27 years ago, and leiomyoma in the left thigh 8 years ago. Tumor markers were normal. A well-defined hypoechoic tumor measuring 26 mm × 22 mm was detected at the pancreatic tail using ultrasound. Inside the tumor, a small cystic structure was observed. High-sensitivity Doppler imaging revealed faint signals indicating blood flow from the periphery to the interior. Contrast-enhanced ultrasonography in the arterial phase revealed slow, heterogenous enhancement from the periphery to the center of the mass that occurred later than that in the pancreatic parenchyma. The tumor then exhibited homogeneous enhancement except for the periphery, with subsequent washout. Endoscopic ultrasound-fine-needle aspiration was performed. Histopathological examination diagnosed a leiomyoma. The left thigh tumor resected 8 years ago had been diagnosed as benign metastatic leiomyoma (BML). We considered that the current pancreatic tumor was also BML clinically. We report this case because pancreatic metastasis from uterine fibroids is extremely rare. Uterine fibroids are benign tumors, but there are rare cases of metastasis to various organs, which should be considered when performing ultrasound.