Nonthrombotic pulmonary embolism caused by intravenous leiomyomatosis

Wu, Yi-Hsiao; Lee, Yung-Tsai; Lee, Chuin-I; Tzeng, Yun-Hsuan; Wei, Jeng

doi:10.1097/md.0000000000014118

Cited by 3 publications

(4 citation statements)

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“…Pulmonary embolism is a potentially life-threatening medical condition; most commonly caused by a deep vein thrombosis [ 1 ]. However, fat embolism, amniotic fluid embolism, septic embolism, tumoral embolism, and nonthrombotic pulmonary artery embolism should be considered, especially when anticoagulation treatment fails [ 2 ].…”

Section: Discussionmentioning

confidence: 99%

“…It is usually caused by a deep vein thrombosis [ 1 ]. Other causes of intravascular obstruction are described [ 2 ] but are extremely rare. This causes substantial delay in correct diagnosis and treatment, which is often only obtained by a multidisciplinary approach.…”

Section: Introductionmentioning

confidence: 99%

“…CT with contrast medium enhancement typically shows a hypoattenuating intravascular filling defect. In combination with a fibromatous uterus on MRI, this may suggest IVL [2], although the presence of a normal uterus on imaging does not exclude it [3]. Echocardiography is used to evaluate intracardiac extension, repercussions on the right heart function, and differential diagnosis of other intracardiac masses [9].…”

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confidence: 99%

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Intravascular Leiomyomatosis as a Rare Cause of Nonthrombotic Pulmonary Embolism

Maercke

Rompuy

Poppe

et al. 2020

Case Reports in Vascular Medicine

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Intravascular leiomyomatosis (IVL) is a very rare condition. It is characterized by the proliferation of benign smooth muscle cells within vascular structures without invasion of these tissues. Symptoms depend on the site of origin and the extent of invasion. Rarely, this neoplasm is located in the inferior vena cava or in the pulmonary vasculature potentially causing symptoms of dyspnea, chest pain, or syncope. We report the case of a 53-year-old woman who was referred to our hospital with extensive pulmonary embolism comprising of a subtotal occlusion of the right pulmonary artery with extension into the left pulmonary artery. Due to persistent dyspnea (New York Heart Association class II) despite anticoagulation, after a six-week period, imaging was repeated and showed stable findings. As she was not responding to adequate anticoagulant therapy, intima sarcoma of the pulmonary artery was suspected, and a pulmonary endarterectomy (PEA) was performed. A smooth, white, intravascular mass was easily and completely removed. Analysis demonstrated a lesion consisting of cells without atypia, showing expression of alpha-smooth muscle actin (alpha SMA) and desmin with partial expression of estrogen receptor (ER) and progesterone receptor (PR), leading to the diagnosis of intravascular leiomyomatosis. The patient fully recovered. Complete surgical removal of the intravascular tumor is recommended to relieve symptoms and prevent possible complications. Clinicians have to be aware that in unresolved pulmonary embolism, nonthrombotic and rare causes, like an intima sarcoma or intravascular leiomyomatosis, should be considered.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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confidence: 99%

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Intravascular Leiomyomatosis as a Rare Cause of Nonthrombotic Pulmonary Embolism

Maercke

Rompuy

Poppe

et al. 2020

Case Reports in Vascular Medicine

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“…The differential diagnosis of IVL should include benign myoma, thrombus, leiomyosarcoma, soft tissue sarcoma, lymphoma, cardiac myxoma, tumor thrombosis of Wilms tumor or metastasis [2] , [25] , [26] , [27] .…”

Section: Ivl: Review Of Literaturementioning

confidence: 99%

Intravenous leiomyomatosis: Case series and review of the literature

Mathey

Huber

2021

International Journal of Surgery Case Reports

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Introduction Intravenous leiomyomatosis (ILV) is a rare pathology, part of leiomyoma beyond the uterus (LBU), characterized by benign smooth muscle cell tumor outside of the uterus and mainly affecting premenopausal woman with a medical history of leiomyoma or gynecologic surgical treatment. The treatment depends on the localization of the tumor, age of the patient, initial size, symptoms and the suitability for surgery but should always aims in toto surgical resection. Case presentation Retrospective case series and review of literature. Clinical discussion Symptoms presented by the patient were aspecific and only localized in the pelvic area. All cases were fortuitous histopathological diagnosis. No relapse was. Two out of 5 patients have pulmonary nodules, only one was biopsied and diagnosed with PBML (pulmonary benign metastasizing leiomyoma). Conclusion IVL and BML are rare disease that can co-exist. Because of tumoral hormonal receptors, hormonotherapy could be an optional treatment but to date no clear efficacy is demonstrated. In case of high recurrence risk such as voluminous initial mass, impairment of broad ligament, failure of total surgical resection, adjuvant hormonotherapy could be useful. Recurrence rate is about 16.6-30% and can occur even dozen years later and even after radical surgery, justifying a regular follow up.

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Clinical features and prognostic factors analysis of intravenous leiomyomatosis

Chen

Zhang

et al. 2023

Front. Surg.

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BackgroundThe treatment and prognostic factors of intravenous leiomyomatosis (IVL) remain lacking systematic evidence.MethodsA retrospective study was conducted on IVL patients from the Qilu Hospital of Shandong University, and IVL cases were published in PubMed, MEDLINE, Embase and Cochrane Library databases. Descriptive statistics were used for the basic characteristics of patients. The Cox proportional hazards regression analysis was used to assess the high-risk factors related to the progression-free survival (PFS). The comparison of survival curves was performed by Kaplan–Meier analysis.ResultsA total of 361 IVL patients were included in this study, 38 patients from Qilu Hospital of Shandong University, and 323 patients from the published literature. Age ≤45 years was observed in 173 (47.9%) patients. According to the clinical staging criteria, stage I/II was observed in 125 (34.6%) patients, and stage III/IV was observed in 221 (61.2%) patients. Dyspnea, orthopnea, and cough were observed in 108 (29.9%) patients. Completed tumor resection was observed in 216 (59.8%) patients, and uncompleted tumor resection was observed in 58 (16.1%) patients. Median follow-up period was 12 months (range 0–194 months), and 68 (18.8%) recurrences or deaths were identified. The adjusted multivariable Cox proportional hazard analysis showed age ≤45 years (vs. >45) (hazard ratio [HR] = 2.09, 95% confidence interval [CI] 1.15–3.80, p = 0.016), and uncompleted tumor resection (vs. completed tumor resection) (HR = 22.03, 95% CI 8.31–58.36, p < 0.001) were high-risk factors related to the PFS.ConclusionPatients with IVL have a high probability of recurrence after surgery and a poor prognosis. Patients younger than 45 years and with uncompleted tumor resection are at higher risk of postoperative recurrence or death.

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Nonthrombotic pulmonary embolism caused by intravenous leiomyomatosis

Cited by 3 publications

References 9 publications

Intravascular Leiomyomatosis as a Rare Cause of Nonthrombotic Pulmonary Embolism

Intravascular Leiomyomatosis as a Rare Cause of Nonthrombotic Pulmonary Embolism

Intravenous leiomyomatosis: Case series and review of the literature

Clinical features and prognostic factors analysis of intravenous leiomyomatosis

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