Pajavand Amir Mohammad, Sharifi Guive, Anvari Amir, Bidari-Zerehpoosh Farahnaz, Shamsi Mohammad A, Nateghinia Saeedeh, Meybodi Tohid Emami
Institute for Cognitive and Brain Sciences, Shahid Beheshti University Government College University, Tehran, Iran.
Skull Base Research Center, Loghman Hakim Hospital, Shahid Beheshti University of Medical Sciences, Tehran, Iran.
Front Hum Neurosci. 2021 Apr 6;15:620439. doi: 10.3389/fnhum.2021.620439. eCollection 2021.
Neurofibromatosis type 1 (NF1) is associated with peripheral and central nervous system tumors. It is noteworthy that the regions in which these tumors frequently arise are the optic pathways (OPs) and the brainstem. Thus, we decided to trace the procedure of diffusion Magnetic Resonance Imaging (dMRI) alterations along with Short-Wavelength Automated Perimetry (SWAP) examinations of the OPs after surgery and chemotherapy over 1 year, which enabled us to evaluate chemotherapy's efficacy in an NF1 patient with an OP tumor. In this study, a 25-year-old woman with NF1 and left optic radiation (OR) glioma underwent surgery to remove the glioma. Immunohistochemistry (IHC) revealed a Pilocytic Astrocytoma (PA) WHO grade I. Post-operation chemotherapy done using nine treatment cycles of administering Temozolomide (TMZ) for 5 days every 4 weeks. Applying the region of interest (ROI) differential tractography method and SWAP four times every 3 months allowed us to follow the patient's visual acuity alterations longitudinally. The differential deterministic tractography method and statistical analyses enabled us to discover the white matter (WM) tracts anisotropy alterations over time. Furthermore, statistical analyses on the SWAP results along time illustrated possible alterations in visual acuity. Then, we could compare and associate the findings with the SWAP examinations and patient symptoms longitudinally. Statistical analyses of SWAP tests revealed a significant improvement in visual fields, and longitudinal differential tractography showed myelination and dense axonal packing in the left OR after 1 year of treatment. In this study, we examined an old hypothesis suggesting that chemotherapy is more effective than radiotherapy for NF1 patients with OP gliomas (OPGs) because of the radiation side effects on the visual field, cognition, and cerebrovascular complications. Our longitudinal clinical case study involving dMRI and SWAP on a single NF1-OPG patient showed that chemotherapy did not suppress the OP myelination over time. However, it should be noted that this is a clinical case study, and, therefore, the generalization of results is limited. Future investigations might focus on genetic-based imaging, particularly in more cases. Further, meta-analyses are recommended for giving a proper Field Of View (FOV) to researchers as a subtle clue regarding precision medicine.
1型神经纤维瘤病（NF1）与外周和中枢神经系统肿瘤相关。值得注意的是，这些肿瘤经常发生的区域是视路（OPs）和脑干。因此，我们决定在1年的手术和化疗后，通过短波长自动视野计（SWAP）对视路进行检查，同时追踪扩散磁共振成像（dMRI）的变化过程，这使我们能够评估化疗对一名患有视路肿瘤的NF1患者的疗效。在本研究中，一名患有NF1和左侧视辐射（OR）胶质瘤的25岁女性接受了手术切除胶质瘤。免疫组织化学（IHC）显示为世界卫生组织I级毛细胞型星形细胞瘤（PA）。术后化疗采用替莫唑胺（TMZ）每4周给药5天，共进行9个治疗周期。每3个月应用感兴趣区域（ROI）差异纤维束成像方法和SWAP四次，使我们能够纵向跟踪患者的视力变化。差异确定性纤维束成像方法和统计分析使我们能够发现白质（WM）束各向异性随时间的变化。此外，对SWAP结果随时间的统计分析表明视力可能发生了变化。然后，我们可以纵向比较这些发现，并将其与SWAP检查和患者症状联系起来。SWAP测试的统计分析显示视野有显著改善，纵向差异纤维束成像显示治疗1年后左侧OR有髓鞘形成和密集的轴突堆积。在本研究中，我们检验了一个旧的假设，即由于放疗对视野、认知和脑血管并发症的副作用，化疗对患有视路胶质瘤（OPGs）的NF1患者比放疗更有效。我们对一名NF1-OPG患者进行的涉及dMRI和SWAP的纵向临床病例研究表明，随着时间的推移，化疗并没有抑制视路的髓鞘形成。然而，应该注意的是，这是一个临床病例研究，因此，结果的推广是有限的。未来的研究可能会集中在基于基因的成像上，特别是在更多的病例中。此外，建议进行荟萃分析，以便为研究人员提供一个适当的视野（FOV），作为关于精准医学的一个微妙线索。
