A rare case of recurrent intravenous leiomyomatosis: Role of GnRH analogues?

Kaur, Supreet; Tongaonkar, HB; Maheshwari, Ana; Menon, Santosh

doi:10.4103/0019-509x.175577

Cited by 7 publications

(10 citation statements)

References 4 publications

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“…In another case, the unresectable IVLM shrank significantly by 9 months of GnRHa therapy, which could be completely removal thereafter [43]. GnRHa given pre or postoperatively in premenopausal women was shown to be able to reduce preoperative tumors size, and suppress the progression of the residual tumor [9,15,19,33,34,36,37]. Moreover, the intraoperative blood loss and medical costs was 7.5 and 7 times higher in patients with ICLM when compared with patients with IVLM but not extended beyond the abdominal cavity [18].…”

Section: Discussionmentioning

confidence: 97%

“…However, complete resection cannot be performed in many cases for extension of tumors [5,24,27,37], densely adherent to the luminal wall [4, 5, 7, 13-16, 19-22, 41, 42], and patients' will [27,28], or constrained medical resources. In such conditions, recurrence occurred postoperatively [4,9,10,12,15,21,22,24], lethal complications such as uncontrolled hemorrhage [20,42], heart failure, and sudden death happened [3,7]. The complete resection rate was significantly lower in patients with firm/extensive adhesion than that in those with no/slight adhesion of IVC or above (Table 2).…”

Section: Discussionmentioning

confidence: 99%

“…The residual tumor was partly reduced in 3 patients with preoperative GnRHa administration initially [19,33,34], but the effects were limited and the tumor increased in size later soon. 3 patients had ovarian preservation and administration of GnRHa postoperatively (15 to 24 months), and the tumors were effectively suppressed or disappeared during the treatment [9,15,35]. The other 2 patients had received incomplete resection with BSO and the administration of GnRHa postoperatively, the residual tumors remained stable [36,37].…”

Section: Pre/postoperative Gnrha and Iclmmentioning

confidence: 99%

“…Under a rare condition, it reaches the right atrium by an extensive spread beyond the inferior vena cava (IVC), which is known as intracardiac leiomyomatosis (ICLM) [3]. ICLM is histologically benign but clinically aggressive, which can cause fatalities by acute heart or other organs' failure [3][4][5][6], surgical complications such as uncontrolled hemorrhage [7,8], and recurrence [9][10][11].…”

Section: Introductionmentioning

confidence: 99%

“…Treatment for ICLM varied because of its rarity and complexity, which included surgeries [12][13][14], anti-estrogen hormone therapies [9,12,15], and even a wait-andsee strategy [1,16]. Complete tumor resection was the most recommended [17,18], but sometimes it cannot be achieved due to the high risks of surgery [5,7,13,15,16,[19][20][21][22], patient's poor conditions [4,12,23,24], lack of medical resources [25], financial constraints [2,26], or patients' refusal [27,28].…”

Section: Introductionmentioning

confidence: 99%

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The role of estrogen deprivation therapy in premenopausal women with primary unresectable intracardiac leiomyomatosis: a systematic review and meta-analysis

Liang

Lei

Xie

et al. 2021

Orphanet J Rare Dis

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Background Intracardiac leiomyomatosis (ICLM) is a rare life-threatening form of intravenous leiomyomatosis (IVLM). The incomplete resection and recurrence are associated with high morbidity and mortality. The objective of this study is to identify that whether estrogen deprivation therapies, including bilateral salpingo-oophorectomy (BSO)-based surgery and gonadotrophin releasing hormone agonists (GnRHa) administration, could bring benefits to patients with primary unresectable ICLM. Methods PubMed/MEDLINE (Ovid) was searched (up to May 2021) for studies reporting individual patient data on demographics, clinicopathological features, treatment, and follow-up information. Exclusion criteria were patients who may have been included in two or more publications. This study was performed in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Results A total of 114 patients from 70 papers were included. Several reports showed that the tumor in the right atrium and inferior vena cava shrank dramatically after BSO-based surgery, or GnRHa administrated preoperatively in premenopausal women. The rate of complete resection was 64.04% in patients with ICLM, which was 85.25% in no/slight adhesion and no pulmonary nodules group, while 22.22% in firm/extensive adhesion and/or pulmonary nodules group (p < 0.0001). Meanwhile, the recurrence rates in patients with complete resection and incomplete resection were 4.29% and 37.84% respectively (p < 0.0001). Furthermore, complete resection with BSO had the lowest recurrence rate of 3.13%, incomplete resection with BSO had a progression rate of 45.45%, while incomplete resection with ovarian preservation had the highest progression rate of 75.00%. Conclusions The recurrence rate of ICLM was closely related to firm/extensive adhesion in IVC or above, and/or pulmonary nodules. BSO-based surgery might reduce the recurrence rate no matter ICLM could be completely resected or not. In addition, estrogen deprivation therapies could decrease tumor burden as a primary treatment, and further make a secondary complete resection feasible in premenopausal women with initially unresectable ICLM.

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Section: Discussionmentioning

confidence: 97%

Section: Discussionmentioning

confidence: 99%

Section: Pre/postoperative Gnrha and Iclmmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

The role of estrogen deprivation therapy in premenopausal women with primary unresectable intracardiac leiomyomatosis: a systematic review and meta-analysis

Liang

Lei

Xie

et al. 2021

Orphanet J Rare Dis

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Unraveling the challenges of intravenous leiomyomatosis: a retrospective analysis of 11 cases

Wang,

Liu,

Feng

2023

Arch Gynecol Obstet

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Objective This study provides a concise overview of diagnostic and treatment strategies for intravenous leiomyomatosis (IVL), a rare disease with nonspecific clinical manifestations, based on cases from a tertiary referral hospital in China. Methods We retrospectively analyzed 11 premenopausal patients with confirmed IVL between 2018 and 2022. Clinical data from Ultrasound, Enhanced CT, and MRI were studied, along with surgical details, postoperative pathology, and follow-up information. Results Premenopausal patients showed no disease-specific symptoms, with 90.9% having a history of gynecological or obstetric surgery, and 72.7% having prior uterine fibroids. Cardiac involvement was evident in two cases, with echocardiography detecting abnormal floating masses from the inferior vena cava. Pelvic ultrasound indicated leiomyoma in 90.9% of cases, with ≥ 50 mm size. Surgery was the primary treatment, and lesions above the internal iliac vein resulted in significantly higher intraoperative blood loss (median 1300 ml vs. 50 ml, p = 0.005) and longer hospital stays (median 10 days vs. 4 days, p = 0.026). Three patients with lesions above the inferior vena cava required combined surgery with cardiac specialists. Recurrence occurred in 2 out of 11 patients with incomplete lesion resection. Conclusions IVL mainly affects premenopausal women with uterine masses, primarily in the pelvic cavity (Stage I). Pelvic ultrasound aids early screening, while Enhanced CT or MR assists in diagnosing and assessing venous lesions. Complete resection is crucial to prevent recurrence. Lesions invading the internal iliac vein and above pose higher risks during surgery. A multidisciplinary team approach is essential for patients with lesions above the inferior vena cava, with simultaneous surgery as a potential treatment option.

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Clinical analysis of uterine intravenous leiomyomatosis: A retrospective study of 260 cases

Peng

Zhong

Zhu

et al. 2021

J of Obstet and Gynaecol

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Methods:We collected the clinical data of 260 patients admitted to the hospital from April 2003 to September 2019 with pathologically confirmed intravenous leiomyomatosis (IVL) and followed up with these patients regularly. Univariate and multivariate logistic regression analyses were carried out on the relevant recurrence factors. Results: A total of 166 patients were regularly followed up, the median follow-up time was 36 (range 2-168) months, 14 (5.4%) patients eventually relapsed, and the median recurrence time was 8.5 (range 2-42) months. The univariate analysis showed that age (p = 0.003) and surgical type (p < 0.001) were associated with recurrence, and multivariate regression analysis demonstrated that surgical type was the only factor associated with recurrence (p < 0.001, OR 20.01). Conclusions: The use of gonadotrophin releasing hormone agonist (GnRHa) cannot reduce the postsurgical recurrence rate of patients with UIVL. Compared to total hysterectomy and bilateral salpingo-oophorectomy (TH-BSO), total hysterectomy (TH) does not increase the odds of recurrence, but the chance of recurrence with tumorectomy (TE) is 20 times higher than that of TH-BSO.

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A rare case of recurrent intravenous leiomyomatosis: Role of GnRH analogues?

Cited by 7 publications

References 4 publications

The role of estrogen deprivation therapy in premenopausal women with primary unresectable intracardiac leiomyomatosis: a systematic review and meta-analysis

The role of estrogen deprivation therapy in premenopausal women with primary unresectable intracardiac leiomyomatosis: a systematic review and meta-analysis

Unraveling the challenges of intravenous leiomyomatosis: a retrospective analysis of 11 cases

Clinical analysis of uterine intravenous leiomyomatosis: A retrospective study of 260 cases

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