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Benign leiomyoma with multiple metastases to vertebrae and calvarium: An index case with comprehensive review of endocrine targets
Neurosurgical Review ( IF 2.5 ) Pub Date : 2020-02-20 , DOI: 10.1007/s10143-020-01268-5
İlhan Elmaci 1 , Meric A Altinoz 2 , Burcu Eylem Kahraman Ozlu 1 , Ramazan Sari 1 , Ozlem Er 3 , Ayca Ersen Danyeli 4 , Ercan Karaarslan 5
Affiliation  

“Benign” metastatic leiomyomas (BML) are indolently growing metastatic tumors which mostly associate with uterine leiomyomas in women in reproductive ages. The reason to define these lesions as “benign” despite metastasis is their pathological features with low mitotic counts, lack of or minimal nuclear atypia, pseudocyst formation, and coagulative necrosis unlike leiomyosarcomas. Despite lack of pathological malignant features, they may cause significant morbidity and even mortality. Here, we describe a BML case with metastases to vertebrae and skull bones. Vertebral and skull metastases of BMLs were very rarely reported. In treatment of these tumors, hysterectomy and GnRH modifier treatments are widely employed. GnRH agonists act by desensitization and downregulation of the GnRH receptors, while GnRH antagonists act via the canonical competitive blockage. These treatments reduce FSH and LH levels, thereby reducing the systemic levels of sex steroids which stimulate leiomyoma growth. However, leiomyomas inherently harbor aromatase activity and synthesize their own estrogen; hence, treatment with systemic estrogen antagonists may provide better tumor control. Another important factor in BML pathogenesis is progesterone, and both progesterone receptor antagonists and high-dose progesterone receptor agonists may reduce BML growth. Following surgical treatment of the calvarial mass and radiotherapy of the vertebral metastatic foci, our BML case was successfully managed with hysterectomy and anastrozole treatment. Higher awareness of BML cases and their molecular endocrinological features in the neurosurgical community may pave to develop better strategies for treatment of these tumors causing high morbidity.



中文翻译:

良性平滑肌瘤,多处转移至椎骨和颅骨:全面回顾内分泌指标的索引病例

“良性”转移性平滑肌瘤(BML)是生长缓慢的转移性肿瘤,在育龄妇女中主要与子宫平滑肌瘤有关。尽管有转移,但仍将这些病变定义为“良性”的原因是其病理特征是有丝分裂计数低,核异型性缺失或缺乏或极少,假性囊肿形成和凝固性坏死不像平滑肌肉瘤。尽管缺乏病理学上的恶性特征,但它们可能会导致明显的发病率甚至死亡。在这里,我们描述了一个BML病例,其转移至椎骨和颅骨。很少报道BML的椎骨和颅骨转移。在这些肿瘤的治疗中,子宫切除术和GnRH修饰剂治疗被广泛采用。GnRH激动剂通过脱敏和下调GnRH受体发挥作用,而GnRH拮抗剂则通过典型的竞争性阻断起作用。这些治疗降低了FSH和LH水平,从而降低了刺激平滑肌瘤生长的性类固醇的全身水平。然而,平滑肌瘤固有地具有芳香酶活性并合成其自身的雌激素。因此,使用全身性雌激素拮抗剂可以更好地控制肿瘤。BML发病机理中的另一个重要因素是孕激素,孕激素受体拮抗剂和大剂量孕激素受体激动剂均可降低BML的生长。颅骨肿块的外科手术治疗和椎骨转移灶的放疗后,我们的BML病例通过子宫切除术和阿那曲唑治疗获得了成功。

更新日期:2020-02-20
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