Intravenous Leiomyomatosis with Uterine Leiomyoma and Adenomyosis: A Case Presentation and Brief Comment on the Histogenesis

Intravenous leiomyomatosis (IVL) is a rare benign neoplasm. Herein, we describe two cases of IVL at different levels of progression. The tumor in Case 1 was extensive, invading the right atrium after a hysterectomy for a uterine myoma. The tumor temporarily responded to hormonal treatment; however, tumor regrowth occurred. In contrast, the tumor in Case 2 extended only to the pelvic veins and was revealed preoperatively. Hysterectomy and bilateral salpingo-oophorectomy were performed, resulting in the complete surgical resection of the tumor. In Case 2, no recurrence has been observed. Tumor samples were evaluated for hyaluronan expression using Alcian blue staining (with and without hyaluronidase digestion). The tumor in Case 1 stained strongly positive for hyaluronan while the tumor in Case 2 stained weakly positive for hyaluronan. In contrast, a large non-IVL uterine leiomyoma (control) stained negative for hyaluronan. These results suggest a relationship between tumor hyaluronan expression and IVL progression, similar to that in other cancers.

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Oncological Properties of Intravenous Leiomyomatosis: Involvement of Mesenchymal Tumor Stem-Like Cells

Current Issues in Molecular Biology ◽

10.3390/cimb43020084 ◽

2021 ◽

Vol 43 (2) ◽

pp. 1188-1202

Author(s):

Saya Tamura ◽

Takuma Hayashi ◽

Hideki Tokunaga ◽

Nobuo Yaegashi ◽

Kaoru Abiko ◽

...

Keyword(s):

Stem Cells ◽

Cancer Stem Cells ◽

Malignant Tumor ◽

Uterine Leiomyoma ◽

Malignant Tumors ◽

Female Genital Tract ◽

Mesenchymal Tumor ◽

Uterine Leiomyosarcoma ◽

Intravenous Leiomyomatosis ◽

Ki 67

Uterine leiomyoma, also known as fibroids, is the most common benign neoplasm of the female genital tract. Leiomyoma is the most common uterine tumor. The leiomyoma subtypes account for approximately 10% of leiomyomas. Intravenous leiomyomatosis, a uterine leiomyoma subtype, is an intravascular growth of benign smooth muscle cells, occasionally with pelvic or extrapelvic extension. Uterine leiomyosarcoma, a malignant tumor, tends to metastasize hematogenously, and distant metastasis to the lungs and liver is common. Therefore, the oncological properties of this intravenous leiomyomatosis resemble those of the malignant tumor uterine leiomyosarcoma. Cancer stem cells migrate to distant organs via intravascular infiltration, leading to micrometastases. We examined the oncological properties of intravenous leiomyomatosis using molecular pathological techniques on tissue excised from patients with uterine leiomyoma. CD44-positive mesenchymal tumor stem-like cells were detected in both patients with intravenous leiomyomatosis and uterine leiomyosarcoma. The oncological properties of intravenous leiomyomatosis were found to be similar to those of uterine leiomyosarcoma. However, in intravenous leiomyomatosis, cyclin E and Ki-67-positive cells were rare and no pathological findings suspecting malignancy were observed. It is expected that establishing a treatment method targeting cancer stem cells will lead to the treatment of malignant tumors with a low risk of recurrence and metastasis.

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Intravenous leiomyomatosis with atypical histologic features: A case report

International Journal of Gynecological Cancer ◽

10.1136/ijgc-00009577-200301000-00015 ◽

2003 ◽

Vol 13 (1) ◽

pp. 83-87 ◽

Cited By ~ 5

Author(s):

P. M. Lam ◽

K. W. K. Lo ◽

M. M. Y. Yu ◽

T. K. Lau ◽

T. H. Cheung

Keyword(s):

Vessel Wall ◽

Uterine Leiomyoma ◽

Smooth Muscle Tumor ◽

Gonadotropin Releasing Hormone ◽

Intravenous Leiomyomatosis ◽

Venous Graft ◽

Prosthetic Reconstruction ◽

Surgical Preparation ◽

High Index Of Suspicion ◽

Muscle Tumor

Intravenous leiomyomatosis (IVL) is a rare smooth muscle tumor. We report a case of IVL with atypical histologic features, which did not respond to gonadotropin-releasing hormone agonists and could be only partially resected due to adherence to the vessel wall. Atypical histology may signify more aggressive behavior. IVL should always be considered when a patient presents with both uterine leiomyoma and venous thrombosis, and a high index of suspicion is crucial for early diagnosis. An adequate surgical preparation including venous graft or prosthetic reconstruction is essential as difficulty in removal may arise if the intravascular tumor adheres to the vessel wall.

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Intravenous leiomyomatosis: the first reported case of intraoperative intracaval embolisation of tumour to the right atrium

BMJ Case Reports ◽

10.1136/bcr-2019-233341 ◽

2020 ◽

Vol 13 (3) ◽

pp. e233341

Author(s):

Gillian A Corbett ◽

Catherine O'Gorman ◽

Waseem Kamran

Keyword(s):

Right Atrium ◽

Uterine Leiomyoma ◽

Histopathological Examination ◽

Vena Cava ◽

Abdominal Distension ◽

Preoperative Imaging ◽

Intravenous Leiomyomatosis ◽

Midline Laparotomy ◽

First Case ◽

The Right

Intravenous leiomyomatosis is extremely rare. This case describes a 42-year-old woman who presented with abdominal distension, cyclical bloating and urinary retention. Preoperative imaging showed a multilobulated uterine mass. Following multidisciplinary team discussion, a complete staging surgery consisting of midline laparotomy, total hysterectomy and bilateral salpingo-oophrectomy was performed. Intraoperatively, a large multilobulated uterine mass was noted with engorgement of the infundibulopelvic ligaments due to intravascular extension of tumour. On removal of the uterus, the patient desaturated and became hypotensive. Intraoperative transoesophageal echocardiography revealed mass extending from the inferior vena cava (IVC) into the right atrium (RA). The cardiothoracic surgical team retrieved a worm-like mass extending from the IVC into the RA. Histopathological examination diagnosed a large uterine leiomyoma with intravenous leiomyomatosis. The mass from the RA was a bland spindle cell tumour which matched the uterine mass histopathologically. Intravenous leiomyomatosis is a rare variant of uterine leiomyoma. Although intracardiac extension has been described, this is the first case of intraoperative embolisation of pelvic tumour to the RA at hysterectomy.

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Uterine Leiomyoma with Intravenous Leiomyomatosis

EJVES Extra ◽

10.1016/j.ejvsextra.2004.10.001 ◽

2005 ◽

Vol 9 (1) ◽

pp. 4-6 ◽

Cited By ~ 4

Author(s):

M.A. Murphy ◽

A. Kothari ◽

S. Westaby ◽

S. Kehoe ◽

A. Handa

Keyword(s):

Uterine Leiomyoma ◽

Intravenous Leiomyomatosis

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Intravenous Leiomyomatosis with Intracardiac Extension: Echocardiographic Study and Literature Review

Texas Heart Institute Journal ◽

10.14503/thij-13-3533 ◽

2014 ◽

Vol 41 (5) ◽

pp. 502-506 ◽

Cited By ~ 18

Author(s):

Rongjuan Li ◽

Yanguang Shen ◽

Yan Sun ◽

Chuanchen Zhang ◽

Ya Yang ◽

...

Keyword(s):

Inferior Vena Cava ◽

Right Atrium ◽

Uterine Leiomyoma ◽

Inferior Vena ◽

Vena Cava ◽

Common Disease ◽

Intravenous Leiomyomatosis ◽

Endothelial Surface ◽

The Right ◽

The Masses

Uterine leiomyomatosis is a common disease in women; however, intravenous leiomyomatosis with intracaval and intracardiac tumor extension is rare. We sought to analyze the clinical and echocardiographic features of intracardiac leiomyomatosis. From January 2003 through July 2012, 7 women (age range, 24–59 yr) underwent surgical resection of histopathologically diagnosed intracardiac leiomyomas at our hospital. Most of the patients had histories of hysterectomy or uterine leiomyoma. We retrospectively analyzed their preoperative echocardiograms. We found that the tumors had no stalks, did not adhere to the wall of the right side of the heart, were highly mobile, and moved back and forth in the right atrium near the tricuspid orifice. All tumors originated from the inferior vena cava and had borders well demarcated from that structure's wall. Most of the masses extended into the inferior vena cava and right atrium through the right internal and common iliac veins. Computed tomograms revealed pelvic tumors and contiguous filling defects in 6 patients. When echocardiograms reveal a right-sided cardiac mass that originates from the inferior vena cava, particularly in women who have a history of hysterectomy or uterine leiomyoma, intracardiac leiomyomatosis should be suspected. If the mass has no stalk and freely moves within the inferior vena cava and right-sided cardiac chambers without attachment to the endothelial surface or endocardium, intracardiac leiomyomatosis should be diagnosed. We discuss our findings and briefly review the relevant medical literature.

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Primary Ovarian Leiomyoma: A common tumor in rare location and report of eight cases

10.21203/rs.2.17330/v1 ◽

2019 ◽

Author(s):

wu zaigui ◽

Dong minyue

Keyword(s):

Uterine Leiomyoma ◽

Pelvic Mass ◽

Case Presentation ◽

Surgical Decision ◽

Pelvic Tumors ◽

Pathologic Findings ◽

Rare Location ◽

The Mean ◽

Benign Ovarian Tumors ◽

Fertile Women

Abstract Background: Leiomyomas are the most common pelvic tumors in fertile women and rare conditions of them have extrauterine locations. Ovarian Leiomyoma accounts only for 0.5 to 1% of all benign ovarian tumors with fewer than 60 cases reported in a manner of case report. Here we present a series of eight patients over a six-year period(2012-2018). The clinico-pathological features, diagnosis and management were discussed. Case presentation: We had experienced eight cases of ovarian leiomyoma.The clinical features,pathologic findings,diagnosis, treatment were reviewed. The mean age of these patients was 38.5 years. The majority of these patients may be asymptomatic and usually diagnosed incidentally during pelvic examination or pathologic examination after surgery. Six cases presented only with pelvic mass sized from 2 to 20 cm and even some patients persisted for more than twenty years while the other two were diagnosed due to four years of primary infertile or three months of irregular vagina bleeding. Three patients coincident with uterine leiomyoma while the others not. Three had ovarian leiomyoma degenerated and one case had a evidence of atypical 4/10. Conclusions : ovarian leiomyoma was very rare but it should be considered in the diagnosis of pelvic or ovarian solid masses. Magnetic resonance imaging was provital in its different diagnose while fast frozen pathology during operation was very necessary for its surgical decision. A proper surgical decision should be made according to patients age.

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