Aberrant uterine leiomyomas with extrauterine manifestation: intravenous leiomyomatosis and benign metastasizing leiomyomas
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Objective Intravenous leiomyomatosis (IVL) and benign metastasizing leiomyoma (BML) are uncommon variants of benignuterine leiomyomas with extrauterine manifestations. Categorizing the extent of disease allows clinicians to delineatethe clinical spectrum and the level of sophistication for complete surgical resection. Methods Twelve patients with IVL and BML were reviewed. They were divided into early versus late stage disease groups,and initial manifestation, clinical characteristics, laboratory values, surgical pathology, and follow up data weresummarized. Results Patients were mostly pre- or peri-menopausal and parous. Patients with late stage disease were more likely to presentwith cardiac symptoms or abnormal findings on chest X-ray, whereas those with early stage disease presented withclassical leiomyoma symptoms including heavy menstrual bleeding, increased myoma size, or lower abdominaldiscomfort. Tumor marker levels were within normal ranges. A trend of higher neutrophil to leukocyte ratio wasobserved in the late versus the early stage group (10.4 vs. 1.51, P=0.07); the platelet leukocyte ratio was statisticallyhigher in patients with late stage IVL (0.23 vs. 0.13, P=0.04). The overall recurrence rate was 25%. No recurrence wasobserved in stage I or stage III IVL groups, while 50% of the stage II IVL group showed recurrence in the pelvic cavity. Conclusion IVL and BML are benign myoma variants with paradoxically metastatic clinical presentation. Careful inquiry ofsystemic symptoms, the presence of underlying systemic inflammation, and a high index of suspicion are requiredfor preoperative diagnosis. Furthermore, a multidisciplinary approach is necessary to improve outcomes of surgicalresection.
Abstract
Objective Intravenous leiomyomatosis (IVL) and benign metastasizing leiomyoma (BML) are uncommon variants of benignuterine leiomyomas with extrauterine manifestations. Categorizing the extent of disease allows clinicians to delineatethe clinical spectrum and the level of sophistication for complete surgical resection. Methods Twelve patients with IVL and BML were reviewed. They were divided into early versus late stage disease groups,and initial manifestation, clinical characteristics, laboratory values, surgical pathology, and follow up data weresummarized. Results Patients were mostly pre- or peri-menopausal and parous. Patients with late stage disease were more likely to presentwith cardiac symptoms or abnormal findings on chest X-ray, whereas those with early stage disease presented withclassical leiomyoma symptoms including heavy menstrual bleeding, increased myoma size, or lower abdominaldiscomfort. Tumor marker levels were within normal ranges. A trend of higher neutrophil to leukocyte ratio wasobserved in the late versus the early stage group (10.4 vs. 1.51, P=0.07); the platelet leukocyte ratio was statisticallyhigher in patients with late stage IVL (0.23 vs. 0.13, P=0.04). The overall recurrence rate was 25%. No recurrence wasobserved in stage I or stage III IVL groups, while 50% of the stage II IVL group showed recurrence in the pelvic cavity. Conclusion IVL and BML are benign myoma variants with paradoxically metastatic clinical presentation. Careful inquiry ofsystemic symptoms, the presence of underlying systemic inflammation, and a high index of suspicion are requiredfor preoperative diagnosis. Furthermore, a multidisciplinary approach is necessary to improve outcomes of surgicalresection.
참고문헌 (17)
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