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Ken Kazumata, Khin Khin Tha, Haruto Uchino, Tohru Shiga, Hideo Shichinohe, Masaki Ito, Naoki Nakayama and Takeo Abumiya

OBJECTIVE

After revascularization surgery, hyperperfusion and ischemia are associated with morbidity and mortality in adult moyamoya disease (MMD). However, structural changes within the brain following revascularization surgery, especially in the early postsurgical period, have not been thoroughly studied. Such knowledge may enable improved monitoring and clinical management of hyperperfusion and ischemia in MMD. Thus, the objective of this study was to investigate the topographic and temporal profiles of cerebral perfusion and related white matter microstructural changes following revascularization surgery in adult MMD.

METHODS

The authors analyzed 20 consecutive surgeries performed in 17 adults. Diffusion imaging in parallel with serial measurements of regional cerebral blood flow (rCBF) using SPECT was performed. Both voxel-based and region-of-interest analyses were performed, comparing neuroimaging parameters of postoperative hemispheres with those of preoperative hemispheres at 4 different time points within 2 weeks after surgery.

RESULTS

Voxel-based analysis showed a distinct topographic pattern of cerebral perfusion, characterized by increased rCBF in the basal ganglia for the first several days and gradually increased rCBF in the lateral prefrontal cortex over 1 week (p < 0.001). Decreased rCBF was also observed in the lateral prefrontal cortex, occipital lobe, and cerebellum contralateral to the surgical hemisphere (p < 0.001). Reduced fractional anisotropy (FA) and axial diffusivity (AD), as well as increased radial diffusivity (RD), were demonstrated in both the anterior and posterior limbs of the internal capsule (p < 0.001). Diffusion parameters demonstrated the greatest changes in both FA and RD on Days 1–2 and in AD on Days 3–6; FA, RD, and AD recovered to preoperative levels on Day 14. Patients with transient neurological deteriorations (TNDs), as compared with those without, demonstrated greater increases in rCBF in both the lateral prefrontal cortex and striatum as well as smaller FAs in the posterior limb of the internal capsule (p < 0.05).

CONCLUSIONS

The excessively increased rCBF and the recovery process were heterogeneous across brain regions, demonstrating a distinct topographic pattern during the initial 2 weeks following revascularization surgery in MMD. Temporary impairments in the deep white matter tract and immediate postoperative ischemia were also identified. The study results characterized postoperative brain perfusion as well as the impact of revascularization surgery on the brain microstructure. Notably, rCBF and white matter changes correlated to TNDs, suggesting that these changes represent potential neuroimaging markers for tracking tissue structural changes associated with hyperperfusion during the acute postoperative period following revascularization surgery for MMD.

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Yusuke Shimoda, Toshiya Osanai, Naoki Nakayama, Satoshi Ushikoshi, Masaaki Hokari, Hideo Shichinohe, Takeo Abumiya, Ken Kazumata and Kiyohiro Houkin

Hereditary hemorrhagic telangiectasia (HHT) is an autosomal dominant systemic disorder characterized by the enlargement of capillaries, recurrent nosebleeds, and multiple arteriovenous malformations (AVMs). Although cerebral AVMs are traditionally considered to be congenital lesions, some reports have described de novo AVMs, which suggests that the authors believed them to be dynamic conditions. In this article, the authors describe the case of a 5-year-old boy with HHT in whom a de novo cerebral AVM was detected after a negative MRI result at 5 months. To the authors’ knowledge, this is the first report of a de novo AVM in a patient with HHT. In patients with a family history of HHT, de novo AVMs are possible, even when no lesions are detected at the first screening. Therefore, regular screenings need to be performed, and the family should be informed that AVMs could still develop despite normal MRI results.