Corvino Sergio, Somma Teresa, Certo Francesco, Bonomo Giulio, Grasso Erica, Esposito Felice, Berardinelli Jacopo, Barbagallo Giuseppe
Department of Neurosciences, Reproductive and Odontostomatological Sciences, Neurosurgical Clinic, School of Medicine, Università di Napoli "Federico II", 80131 Naples, Italy.
Department of Neurosciences, Division of Neurosurgery, Policlinico "G. Rodolico-S. Marco", University Hospital, 95123 Catania, Italy.
Cancers (Basel). 2024 Sep 11;16(18):3125. doi: 10.3390/cancers16183125.
Ollier disease (OD), acute myeloid leukemia (AML), and brain glioma (BG) are three apparently completely different neoplasms in terms of histopathology, clinic, natural history, and management, but they can affect the same patient. This study aimed to identify the common molecular pathways involved in the pathogenesis of all three diseases and discuss their current and potential role as therapeutic targets. A detailed and comprehensive systematic literature review according to PRISMA guidelines on OD patients harboring BG and/or AML was made. In addition, the unique case of a patient affected by all three considered diseases has been added to our case series. Demographic, pathological, treatment, and outcome data were analyzed and discussed, mainly focusing on the molecular findings. Twenty-eight studies reported thirty-three patients affected by OD and BG, and only one study reported one patient with OD and AML, while only our patient harbored all three pathologies. The IDH R132H mutation was the only genetic alteration shared by all three pathologies and was simultaneously detected in enchondromas and brain glioma in 100% (3/3) of OD patients with BG and also in the neoplastic blood cells of the single patient hosting all three diseases. The IDH1-R132H gene mutation is the etiopathogenetic common denominator among three apparently different tumors coexisting in the same patient. The adoption of mutant-specific IDH1 inhibitor molecules could represent a potential panacea for these conditions in the era of targeted therapies. Further studies with larger clinical series are needed to confirm our results and hypothesis.
骨软骨瘤病(OD)、急性髓系白血病(AML)和脑胶质瘤(BG)在组织病理学、临床、自然史和治疗方面显然是三种完全不同的肿瘤,但它们可能影响同一患者。本研究旨在确定这三种疾病发病机制中涉及的共同分子途径,并讨论它们作为治疗靶点的当前和潜在作用。根据PRISMA指南,对患有BG和/或AML的OD患者进行了详细而全面的系统文献综述。此外,我们的病例系列中增加了一名受所有这三种疾病影响的独特病例。对人口统计学、病理学、治疗和结局数据进行了分析和讨论,主要侧重于分子研究结果。28项研究报告了33例受OD和BG影响的患者,只有1项研究报告了1例患有OD和AML的患者,而只有我们的患者患有所有三种病理情况。IDH R132H突变是所有三种病理情况共有的唯一基因改变,在100%(3/3)患有BG的OD患者的内生软骨瘤和脑胶质瘤中同时检测到,在患有所有三种疾病的单一患者的肿瘤血细胞中也检测到。IDH1-R132H基因突变是同一患者中共存的三种明显不同肿瘤的病因学共同特征。在靶向治疗时代,采用突变特异性IDH1抑制剂分子可能是治疗这些疾病的潜在万能药。需要更多具有更大临床系列的研究来证实我们的结果和假设。