Dataset Information

Potential of unilateral combined bypass surgery to accelerate contralateral radiological progression in pediatric moyamoya disease.

ABSTRACT:

Background

Surgical cerebral revascularization is recommended for treating pediatric moyamoya disease (MMD). However, whether unilateral combined bypass surgery can cause disease progression on the contralateral side is uncertain. The study aimed to investigate the vascular architecture and regional cerebral blood flow (rCBF) status of patients with pediatric MMD after successful unilateral combined bypass surgery and to identify the possible risk factors.

Methods

Pediatric patients diagnosed with MMD and admitted to Xuanwu Hospital who underwent combined bypass surgery between 2019 and 2021 were enrolled. Digital subtraction angiography (DSA) and magnetic resonance imaging (MRI) with arterial spin labeling (ASL) were performed to investigate the vascular architecture and rCBF during surgery and at short-term follow-up. Suzuki's angiographic staging and moyamoya vessel grading system were both used. Progression was defined as an increase in either Suzuki stage or moyamoya vessel grade detected after unilateral surgery. All analyses were performed with conventional statistic methods.

Results

A total of 27 successive patients with a median age of 8 [interquartile range (IQR), 5-14] years old were identified. On the non-operated (non-OP) side, 11 (40.7%) patients demonstrated progression, all of whom showed an increase in the moyamoya vessel grade, and 5 also displayed Suzuki stage progression during the median 4.7 (IQR, 3.7-5.7) months follow-up. However, rCBF barely changed on the non-OP side compared to preoperation [preoperation: median, 49.6, (IQR, 42.9-61.1) mL/100 g/min; postoperation: median, 50.2, (IQR, 43.5-59.3) mL/100 g/min; P=0.445].

Conclusions

Combined bypass surgery might accelerate the radiological progression on the contralateral side, which occurs before the decline of rCBF. Those with earlier Suzuki stage MMD of the non-OP side are prone to rapid progression after unilateral combined revascularization.

SUBMITTER: Luo J

PROVIDER: S-EPMC10585540 | biostudies-literature | 2023 Oct

REPOSITORIES: biostudies-literature

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Potential of unilateral combined bypass surgery to accelerate contralateral radiological progression in pediatric moyamoya disease.

Luo Jichang J Feng Yao Y Lu Xia X Fang Shiyuan S Zheng Shasha S Zeng Gao G Yan Lin L Yang Bin B Wang Tao T Jiao Liqun L Luo Yumin Y Han Ziping Z Ma Yan Y

Quantitative imaging in medicine and surgery 20230904 10

<h4>Background</h4>Surgical cerebral revascularization is recommended for treating pediatric moyamoya disease (MMD). However, whether unilateral combined bypass surgery can cause disease progression on the contralateral side is uncertain. The study aimed to investigate the vascular architecture and regional cerebral blood flow (rCBF) status of patients with pediatric MMD after successful unilateral combined bypass surgery and to identify the possible risk factors.<h4>Methods</h4>Pediatric patien ...[more]

PMID: 37869311

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Project description:Background Early-stage unilateral moyamoya disease (MMD) is difficult to discriminate from isolated intracranial atherosclerotic stenosis, and identification of contralateral progression may aid in the diagnosis of MMD. The RNF213 (ring finger protein 213) R4810K variant is a strong genetic susceptibility factor for MMD; however, the role of contralateral progression in unilateral MMD is unknown. Methods and Results Patients who had undergone RNF213 R4810K genotyping with suspected unilateral MMD between January 2017 and August 2021 from 2 tertiary university hospitals were retrospectively reviewed. We compared the clinical features and radiographic outcomes of patients with and without this variant. The risk factors of contralateral progression in patients with suspected unilateral MMD were evaluated. The RNF213 R4810K variant was observed in 72 of 123 patients with suspected unilateral MMD, all of which were heterozygous. The allele frequency of the R4810K variant was significantly higher in the suspected unilateral MMD group compared with the historical control group (29.3% versus 1.2%; P<0.0001). Family history of MMD was significantly more common in patients with the variant than in those without (17% versus 4%; P=0.003). Eleven of 72 patients with the variant developed contralateral progression, whereas only 1 of 51 patients without the variant developed contralateral progression during a median follow-up period of 28 months (log-rank test; P=0.03). The presence of the RNF213 R4810K variant significantly correlated with contralateral progression (adjusted odds ratio, 6.39 [95% CI, 1.11-36.63]; P=0.04). Conclusions Contralateral progression is more likely to occur in patients with suspected unilateral MMD with the RNF213 R4810K variant than in those without the variant. However, because our study used a small sample size, this finding should be carefully interpreted and requires further studies with more patients and longer follow-up periods.

Project description:Objective: Extracranial-intracranial bypass is currently recognized as the optimal treatment for hemorrhagic-type moyamoya disease (MMD) which reduces incidence of rebleeding. Recent studies have reported the advantage of combined bypass over direct bypass for the general MMD patients. However, the effect of direct bypass and combined bypass surgery specifically for hemorrhagic-type MMD had not been investigated yet. Methods: Hemorrhagic-type MMD patients who underwent direct and combined bypass surgery with complete clinical and radiological documentation from a multicenter cohort between 2009 and 2017 were retrospectively included. Surgical methods included superficial temporal artery-middle cerebral artery (STA-MCA) anastomosis (direct bypass), combined STA-MCA bypass with encephalodurosynangiosis (EDS), and combined STA-MCA bypass with encephaloduroarteriosynangiosis (EDAS). Matsushima standard on follow-up catheter angiography was used to assess surgical outcome. Modified Rankin Scale, incidence of rebleeding and ischemia during follow-up were recorded. Rebleeding-free survival rates between direct and combined bypass were compared by Kaplan-Meier analysis. Results: Sixty eight hemorrhagic-onset MMD patients were included in this study, among which 71 hemispheres were treated with surgery (direct bypass: 17; bypass+EDS: 24; bypass+EDAS: 30). Forty six (64.8%) hemispheres had satisfactory revascularization (Matsushima level 2-3) and 26 (36.6%) had poor neoangiogenesis. Matsushima level was not significantly different between surgical groups (P = 0.258). Good neoangiogenesis from dural grafts was achieved in 26 (36.6%) hemispheres, and good neoangiogenesis from STA grafts was only seen in 4 (out of 30, 12.5%) hemispheres. Multivariate analysis showed bypass patency [P < 0.001, OR (95%CI): 13.41 (3.28-54.80)] and dural neoangiogenesis [P < 0.001, OR (95%CI): 13.18 (3.26-53.36)] both independently contributed to good angiographic outcome. During follow-up, incidences of rebleeding or ischemic events, and re-bleeding free survival rate were not significantly different between surgical groups (P = 0.433, P = 0.559, and P = 0.997). However, patients who underwent combined bypass surgery had significantly lower mRS at follow-up comparing to patients who underwent direct bypass (P = 0.006). Conclusion: Combined bypass surgery and direct bypass surgery offered similar revascularization for hemorrhagic MMD. Bypass patency and dural angiogenesis both contributed to revascularization independently. The potential of indirect bypass to grow new vessels in hemorrhagic-MMD patients was generally limited, but dural leaflets offered better neoangiogenesis than STA grafts and was therefore recommended for surgical revascularization of hemorrhagic MMD.

Project description:BackgroundMoyamoya disease is a rare chronic steno-occlusive cerebrovascular disease. It may have variable clinical symptoms associated with cerebral stroke, including motor paralysis, sensory disturbances, seizures, or headaches. However, patients with moyamoya disease rarely present with involuntary movement disorders, including limb-shaking syndrome, with no previous reports of limb-shaking syndrome occurring after revascularization procedures for this disease. Although watershed shifts can elicit transient neurological deterioration after revascularisation, symptoms originating from the contralateral hemisphere following the revascularization procedure are rare. Here, we report the case of moyamoya disease wherein the patient developed limb-shaking syndrome derived from the contralateral hemisphere after unilateral revascularisation.Case descriptionA 16-year-old girl presented with transient left upper and lower limb numbness and headache. Based on digital subtraction angiography, she was diagnosed with symptomatic moyamoya disease. Single-photon emission computed tomography (SPECT) showed decreased cerebral blood flow (CBF) on the right side, and she underwent direct and indirect bypasses on this side. Involuntary movements appeared in her right upper limb immediately postoperatively. SPECT showed decreased CBF to the bilateral frontal lobes. Subsequently, the patient was diagnosed with limb-shaking syndrome. After performing left-hemispheric revascularisation, the patient's symptoms resolved, and SPECT imaging confirmed improvements in CBF to the bilateral frontal lobes.ConclusionRevascularization for moyamoya disease can lead to watershed shifts, which can induce limb-shaking syndrome derived from abnormalities in the contralateral hemisphere of the revascularized side. For patients with new-onset limb-shaking syndrome after moyamoya revascularisation procedures, additional revascularization may be warranted for treatment of low perfusion areas.

Project description:ObjectiveTo analyze the clinical efficacy of superficial temporal artery-middle cerebral artery (STA-MCA) bypass grafting surgery combined with temporal muscle patch and STA-MCA bypass grafting surgery alone on patients with moyamoya disease.MethodsTotally 73 patients confirmed with moyamoya disease in our hospital between January 2019 and December 2021 were enrolled. Among them, 43 patients treated with STA-MCA bypass grafting surgery combined with temporal muscle patch were assigned to the experiment group, whereas 30 patients treated with STA-MCA bypass grafting surgery alone to the control group. The following items of the 2 groups were compared: clinical efficacy, total effective rate, and disease control rate 6 months after surgery, the changes of modified Rankin Scale (mRS) and Karnofsky performance scale (KPS) scores before and on the seventh day and 6 months after surgery, and changes of Glasgow coma scale scores before and 24 hours after surgery. In addition, the incidences of cerebral ischemia and cerebral hemorrhage within 1 year after surgery were counted. The cerebral perfusion-associated indexes including relative mean transit time (rMTT), relative time-to-peak, relative cerebral blood flow (rCBF), and relative cerebral blood volume (rCBV) on the seventh day and 6 months after surgery were compared between the 2 groups, and the predictive value of cerebral perfusion-associated indexes before surgery for clinical efficacy on patients was analyzed.ResultsThe Glasgow coma scale score after surgery (P>0.05) was similar between the 2 groups, but the clinical efficacy and total effective rate of the 2 groups were notably different (both P<0.05). Compared with those before surgery, mRS scores of both groups declined, whereas KPS scores increased (both P<0.05) on the seventh day after surgery. In addition, compared with those before surgery and on the seventh day after surgery, mRS scores of both groups decreased 6 months after surgery, whereas KPS scores increased (both P<0.05). Both the groups showed decreased rMTT and rTPP, and increased rCBF and rCBV on the seventh day after surgery than those before surgery (all P<0.05). In addition, both the groups still showed decreased rMTT and rTPP, and increased rCBF and rCBV 6 months after surgery than those before surgery and on the seventh day after surgery (all P<0.05). Most notably, the experimental group displayed improved cerebral perfusion-associated indexes than the control group 6 months after surgery (all P<0.05). The relief group showed notably higher rCBF and rCBV levels than the nonrelief group (both P<0.05). According to ROC analysis, the areas under the curves of rCBF and rCBV in forecasting the clinical efficacy on patients were 0.842 and 0.823, respectively.ConclusionSuperficial temporal artery-middle cerebral artery bypass grafting surgery combined with temporal muscle patch can deliver a higher total clinical curative rate for patients with moyamoya disease and can alleviate their coma.

Dataset Information

Potential of unilateral combined bypass surgery to accelerate contralateral radiological progression in pediatric moyamoya disease.

Background

Methods

Results

Conclusions

Publications

Potential of unilateral combined bypass surgery to accelerate contralateral radiological progression in pediatric moyamoya disease.

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