Costa Régis Afonso, Seuánez Héctor N
Genetics Program, Instituto Nacional de Câncer, Rua André Cavalcanti 37, Rio de Janeiro, RJ, 20231-050, Brazil.
Department of Genetics, Universidade Federal do Rio de Janeiro, Rio de Janeiro, Brazil.
Mol Biol Rep. 2018 Jun;45(3):287-295. doi: 10.1007/s11033-018-4161-4. Epub 2018 Feb 17.
Neuroblastoma (NB) is the most common extracranial solid tumor in childhood. This malignancy shows a wide spectrum of clinical outcome and its prognosis is conditioned by manifold biological and genetic factors. We investigated the tumor genetic profile and clinical data of 29 patients with NB by multiplex ligation-dependent probe amplification (MLPA) to assess therapeutic risk. In 18 of these tumors, MYCN status was assessed by fluorescence in situ hybridization (FISH). Copy number variation was also determined for confirming MLPA findings in two 6p loci. We found 2p, 7q and 17q gains, and 1p and 11q losses as the most frequent chromosome alterations in this cohort. FISH confirmed all cases of MYCN amplification detected by MLPA. In view of unexpected 6p imbalance, copy number variation of two 6p loci was assessed for validating MLPA findings. Based on clinical data and genetic profiles, patients were stratified in pretreatment risk groups according to international consensus. MLPA proved to be effective for detecting multiple genetic alterations in all chromosome regions as requested by the International Neuroblastoma Risk Group (INRG) for therapeutic stratification. Moreover, this technique proved to be cost effective, reliable, only requiring standard PCR equipment, and attractive for routine analysis. However, the observed 6p imbalances made PKHD1 and DCDC2 inadequate for control loci. This must be considered when designing commercial MLPA kits for NB. Finally, four patients showed a normal MLPA profile, suggesting that NB might have a more complex genetic pattern than the one assessed by presently available MLPA kits.
神经母细胞瘤(NB)是儿童期最常见的颅外实体瘤。这种恶性肿瘤表现出广泛的临床结局,其预后受多种生物学和遗传因素的影响。我们通过多重连接依赖探针扩增(MLPA)研究了29例NB患者的肿瘤基因谱和临床数据,以评估治疗风险。在其中18例肿瘤中,通过荧光原位杂交(FISH)评估了MYCN状态。还确定了拷贝数变异,以确认两个6p位点的MLPA结果。我们发现2p、7q和17q增益以及1p和11q缺失是该队列中最常见的染色体改变。FISH证实了MLPA检测到的所有MYCN扩增病例。鉴于意外的6p失衡,评估了两个6p位点的拷贝数变异以验证MLPA结果。根据临床数据和基因谱,根据国际共识将患者分层为预处理风险组。MLPA被证明可有效检测国际神经母细胞瘤风险组(INRG)治疗分层要求的所有染色体区域的多种基因改变。此外,该技术被证明具有成本效益、可靠,仅需标准PCR设备,且对常规分析具有吸引力。然而,观察到的6p失衡使得PKHD1和DCDC2不适用于对照位点。在设计用于NB的商业MLPA试剂盒时必须考虑这一点。最后,四名患者的MLPA谱正常,这表明NB可能具有比目前可用的MLPA试剂盒评估的更为复杂的基因模式。