scholarly journals Oro-mandibular dystonia in pediatric moyamoya disease: Two cases report

2021 ◽  
Vol 12 ◽  
pp. 449
Author(s):  
Yuichiro Koga ◽  
Daina Kashiwazaki ◽  
Emiko Hori ◽  
Naoki Akioka ◽  
Satoshi Kuroda
Keyword(s):  
Blood Flow ◽  
Basal Ganglia ◽  
Moyamoya Disease ◽  
Bypass Surgery ◽  
Parietal Lobe ◽  
Underlying Mechanism ◽  
Case Description ◽  
Oromandibular Dystonia ◽  
Rare Symptom

Background: In this report, we describe rare two pediatric cases that developed oro-mandibular dystonia due to moyamoya disease. Case Description: A 7-year-old boy presented with oro-mandibular dystonia and transient weakness of the left extremities, and was diagnosed as moyamoya disease. Another 7-year-old boy developed oro-mandibular dystonia alone and was diagnosed as moyamoya disease. In both, cerebral blood flow (CBF) was markedly decreased in the involved hemispheres, including the basal ganglia and cerebral cortex. They successfully underwent combined bypass surgery and experienced no further attacks of oromandibular dystonia during follow-up periods. CBF almost normalized through surgical collaterals through direct and indirect bypass. Conclusion: When treating patients with oro-mandibular dystonia, moyamoya disease should be listed as one of the differential diseases. The underlying mechanism of oro-mandibular dystonia in moyamoya disease is still unclear, but persistent cerebral ischemia in the basal ganglia and/or parietal lobe may play a key role to induce this rare symptom.

scholarly journals Postoperative executive function in adult moyamoya disease: a preliminary study of its functional anatomy and behavioral correlates

2017 ◽  
Vol 126 (2) ◽  
pp. 527-536 ◽  
Author(s):  
Yu Lei ◽  
Yan-Jiang Li ◽  
Qi-Hao Guo ◽  
Xing-Dang Liu ◽  
Zhuang Liu ◽  
...  
Keyword(s):  
Executive Function ◽  
Moyamoya Disease ◽  
Parietal Lobe ◽  
Executive Dysfunction ◽  
Underlying Mechanism ◽  
Cognitive Improvement ◽  
Postoperative Changes ◽  
The Right ◽  
The Impact

OBJECTIVE Chronic frontal hemodynamic disturbances are associated with executive dysfunction in adult patients with moyamoya disease (MMD). However, the impact of surgical revascularization on executive dysfunction and its underlying mechanism remains unclear. The aim of the present study was to examine the postoperative radiological correlates of cognitive improvement and thereby explore its underlying mechanism. METHODS Fourteen patients who met the inclusion criteria were identified at Huashan Hospital, were operated on, and were successfully followed up for 6 months. Postoperative changes in cortical perfusion and regional amplitude of low-frequency fluctuations (ALFF) were examined by SPECT and resting-state functional MRI, respectively. Executive function was evaluated by 2 tests (Trail Making Test Part B and the summation of executive subtests of Memory and Executive Screening [MES-EX]). Follow-up neuropsychological outcomes were then correlated with radiological changes to identify nodes functioning as leading contributors to postoperative executive outcomes. RESULTS All patients underwent successful unilateral bypass procedures, with some operations performed on the left side and some on the right side. At the 6-month follow-up, the baseline and follow-up test scores for the different sides did not differ significantly. The group with good collaterals (Matsushima Grade A, 9 patients) exhibited significantly increased postoperative perfusion (change in [△] hemodynamics) in bilateral frontal (left, p = 0.009; right, p = 0.003) and left parietal lobe (p = 0.014). The Spearman's correlation test suggested that only the right frontal lobe exhibited significant positive postoperative radiological correlates with cognitive performance (△MES-EX vs △hemodynamics, r = 0.620, p = 0.018; △MES-EX vs △ALFF, r = 0.676, p = 0.008; △hemodynamics vs △ALFF, r = 0.547, p = 0.043). Subsequent regional ALFF analysis revealed that the right dorsolateral prefrontal cortex (DLPFC) was the only node in the responsible hemisphere to exhibit significant postoperative changes. CONCLUSIONS The results not only advance our understanding of pathological interactions of postoperative executive performance in adult MMD, but also indicate that the right DLPFC amplitude might be a quantitative predictor of postoperative executive control improvement.

Abstract WP171: Surgical Strategy And Results Of Additional Bypass For Patients With Moyamoya Disease Refractory To Previous Surgery

Stroke ◽  
2013 ◽  
Vol 44 (suppl_1) ◽  
Author(s):  
Satoshi Kuroda ◽  
Naoki Akioka ◽  
Daina Kashiwazaki ◽  
Hideo Hamada ◽  
Naoya Kuwayama ◽  
...  
Keyword(s):  
Disease Progression ◽  
Moyamoya Disease ◽  
Bypass Surgery ◽  
Posterior Cerebral Artery ◽  
Cerebral Hemodynamics ◽  
Surgical Revascularization ◽  
Surgical Strategies ◽  
Cerebrovascular Events ◽  
Before And After

Introduction —It is well known that surgical revascularization can improve cerebral hemodynamics and prevent further ischemic cerebrovascular events in moyamoya disease. However, a certain subgroup of patients repeats ischemic attacks even after surgery because of insufficient surgery or disease progression during follow-up periods. Hypothesis —Relevant designs and techniques in additional bypass surgery can resolve ischemic cerebrovascular events in patients with moyamoya disease refractory to previous bypass surgery. Methods —This study included totally 7 patients (9 hemispheres) with moyamoya disease refractory to previous bypass surgery. There were 5 children and 2 adults. They underwent previous bypass surgery in Japan and Europe 6 to 240 months before admission. Based on precise clinical and radiological analysis, cerebrovascular events were considered to occur because of insufficient bypass surgery in 5 patients and disease progression in the ipsilateral posterior cerebral artery in 2. Surgical strategies included wide craniotomy to cover the area where cerebral hemodynamics is still impaired and appropriate bypass procedures such as STA-MCA anastomosis, OA-PCA anastomosis, and indirect bypass. Using [123]I-IMP SPECT or [15]O-gas PET, cerebral hemodynamics was precisely examined before and after surgery Results —Postoperative course was uneventful and cerebral hemodynamics significantly improved in all 7 patients. Postoperative cerebral angiography revealed that additional bypass provided collateral blood flow to ischemic area before surgery. Ischemic cerebrovascular events rapidly resolved in 5 patients and gradually decreased in 2. Conclusion —This study strongly suggests adequate surgical design and procedures can resolve ischemic cerebrovascular events in patients with moyamoya disease refractory to previous bypass surgery.

Long-term natural history of hemorrhagic type moyamoya disease in 42 patients

2000 ◽  
Vol 93 (6) ◽  
pp. 976-980 ◽  
Author(s):  
Eiichi Kobayashi ◽  
Naokatsu Saeki ◽  
Hiromichi Oishi ◽  
Shinji Hirai ◽  
Akira Yamaura
Keyword(s):  
Risk Factors ◽  
Natural History ◽  
Moyamoya Disease ◽  
Bypass Surgery ◽  
Bleeding Episode ◽  
Content Type ◽  
History Of ◽  
Fine Print

Object. The purpose of this study was to delineate the long-term natural history of hemorrhagic moyamoya disease (MMD).Methods. A retrospective review was conducted among 42 patients suffering from hemorrhagic MMD who had been treated conservatively without bypass surgery. The group included four patients who had undergone indirect bypass surgery after an episode of rebleeding. The follow-up period averaged 80.6 months. The clinical features of the first bleeding episode and repeated bleeding episodes were analyzed to determine the risk factors of rebleeding and poor outcome.Intraventricular hemorrhage with or without intracerebral hemorrhage was a dominant finding on computerized tomography scans during the first bleeding episode in 29 cases (69%). During the follow-up period, 14 patients experienced a second episode of bleeding, which occurred 10 years or longer after the original hemorrhage in five cases (35.7%). The annual rebleeding rate was 7.09%/person/year. The second bleeding episode was characterized by a change in which hemisphere bleeding occurred in three cases (21.4%) and by the type of bleeding in seven cases (50%). After rebleeding the rate of good recovery fell from 45.5% to 21.4% and the mortality rate rose from 6.8% to 28.6%. Rebleeding and patient age were statistically significant risk factors of poor outcome. All four patients in whom there was indirect revascularization after the second bleeding episode experienced a repeated bleeding episode within 8 years.Conclusions. The occurrence of rebleeding a long time after the first hemorrhagic episode was not uncommon. Furthermore, the change in which hemisphere and the type of bleeding that occurred after the first episode suggested the difficulty encountered in the prevention of repeated hemorrhage.

Occipital artery to middle cerebral artery bypass in pediatric moyamoya disease: rescue therapy after failed revascularization

2021 ◽  
pp. 1-8
Author(s):  
Akikazu Nakamura ◽  
Akitsugu Kawashima ◽  
Hugo Andrade-Barazarte ◽  
Takayuki Funatsu ◽  
Juha Hernesniemi ◽  
...  
Keyword(s):  
Blood Flow ◽  
Cerebral Blood Flow ◽  
Middle Cerebral Artery ◽  
Moyamoya Disease ◽  
Cerebral Artery ◽  
Clinical Condition ◽  
Rescue Therapy ◽  
Occipital Artery ◽  
Revascularization Procedure

OBJECTIVEPatients with pediatric moyamoya disease (PMMD) showing recurrent symptoms or decreased cerebral blood flow after initial revascularization therapy may require additional revascularization to improve their clinical condition. The authors evaluated the clinical and hemodynamic benefits of an occipital artery (OA)–middle cerebral artery (MCA) bypass for patients with PMMD who have undergone an initial revascularization procedure.METHODSThe authors retrospectively identified 9 patients with PMMD who had undergone OA-MCA bypass between March 2013 and December 2017, and who had received a previous superficial temporal artery–MCA bypass. The following clinical data were collected: initial revascularization procedure, symptoms (presence or recurrence), pre- and postoperative cerebral blood flow and cerebrovascular reactivity (CVR) changes, posterior cerebral artery (PCA) stenosis, PCA-related and nonrelated symptoms, and latest follow-up.RESULTSPreoperatively, all patients (n = 9) suffered non–PCA-related recurrent symptoms, and 4 had PCA-related symptoms. At 1-year follow-up, all patients with PCA-related symptoms showed complete recovery. Additionally, 8 (89%) patients with non-PCA symptoms experienced improvement. Only 1 (11%) patient showed no improvement after the surgical procedure. The mean pre- and postoperative CVR values of the MCA territory were 14.8% and 31.3%, respectively, whereas the respective mean CVR values of the PCA territory were 22.8% and 40.0%.CONCLUSIONSThe OA-MCA bypass is an effective rescue therapy to improve the clinical condition and hemodynamic changes caused by PMMD in patients who experience recurrent symptoms after initial revascularization.

scholarly journals Moyamoya disease in a child

2017 ◽  
Vol 10 (2) ◽  
pp. 101
Author(s):  
Shohela Akhter ◽  
Rumana Riaaz ◽  
Roushan Jahan ◽  
Shahana A. Rahman
Keyword(s):  
Moyamoya Disease ◽  
Collateral Circulation ◽  
Bypass Surgery ◽  
Pediatric Patients ◽  
Cerebral Stroke ◽  
Radiological Evaluation ◽  
One Year ◽  
The Right

<p class="Abstract">A 6 year old boy presented with sign symptoms of cerebral stroke. After clinical and radiological evaluation, a diagnosis of bilateral moyamoya disease was made. An indirect bypass surgery encephaloduroateriosynangiosis was done on the right side at first then on the left side.  Patient was partially improved. At one year follow-up, there was the evidence of occlusion of collateral circulation on the right side and the patient became symptomatic again. It is noted that, although the early indirect bypass surgery is indicated in the pediatric patients for preventing the stroke but the outcome is difficult to judge.</p>

scholarly journals Progression of unilateral moyamoya disease resulted in spontaneous occlusion of ipsilateral cavernous dural arteriovenous fistula: Case report

2016 ◽  
Vol 22 (3) ◽  
pp. 362-364 ◽  
Author(s):  
Peng Liu ◽  
Ya Xu ◽  
Xianli Lv ◽  
Huijian Ge ◽  
Ming Lv ◽  
...  
Keyword(s):  
Case Report ◽  
Arteriovenous Fistula ◽  
Moyamoya Disease ◽  
Dural Arteriovenous Fistula ◽  
Unusual Case ◽  
Clinical Symptoms ◽  
Underlying Mechanism ◽  
Facial Vein ◽  
Unilateral Moyamoya Disease

The pathogenic association between cavernous dural arteriovenous fistula (CDAVF) and moyamoya disease remains unclear. This unusual case is the first report of a progression of unilateral moyamoya disease resulting in the spontaneous occlusion of ipsilateral CDAVF. A 52-year-old woman presented with two-week spontaneous exophthalmos, chemosis and tinnitus, and cerebral angiography showed a right CDAVF coexisting with ipsilateral moyamoya disease. Transvenous approaches through the inferior petrosal sinus and facial vein were attempted but failed. However, a progression of the moyamoya disease and disappearance of the CDAVF were observed on one month follow-up angiogram in accordance with the resolution of clinical symptoms. This extremely rare coincidental presentation may have deeper pathogenic implications. This case report may give a clue to the underlying mechanism of the progression of moyamoya disease and occlusion of the CDAVF.

133Xe-DSPECT: Bedeutung der zerebrovaskulären Reservekapazität für Diagnostik und Therapie der chronischen zerebralen Ischämie

1988 ◽  
Vol 27 (04) ◽  
pp. 127-134 ◽  
Author(s):  
P. Schmiedek ◽  
T. Kreisig ◽  
K. Einhäupl ◽  
W. Bauer ◽  
E. Moser ◽  
...  
Keyword(s):  
Blood Flow ◽  
Bypass Surgery ◽  
Reserve Capacity ◽  
Non Invasive ◽  
Flow Reserve ◽  
Diagnostik Und Therapie ◽  
Severe Reduction ◽  
Before And After ◽  
Invasive Method

Using 133Xe-DSPECT, measurements of regional cerebral blood flow (rCBF) were performed before and after stimulation with 1 g of Diamox® in 55 patients with symptomatic occlusion of the internal carotid artery (ICA) and 26 patients without evidence of brain disease (normals). In normals, a negative correlation (r = -0,74) of the Diamox-induced increase of hemispheric blood flow (reserve capacity) and rCBF at rest was observed. In 48 of the 55 patients (87%) with ICA-occlusion a reduced reserve capacity was found. The extent of reduction, however, did not correlate with the findings of transmission CT, because 15 of 21 patients (71 %) with normal CT-scans presented a moderate to severe reduction of reserve capacity. In contrast, reserve capacity corresponded to the extent of arterial collateralisation as documented by cerebral panangiography. In 31 patients EC/IC-bypass surgery was performed. In follow-up studies reserve capacity increased, especially in patients who had a severe reduction preoperatively. Therefore, the combined measurement of rCBF at rest and of reserve capacity is a sensitive, non-invasive method for the diagnosis of (even bilateral) cerebrovascular disease and can be helpful to identify patients with hemodynamic ischemia, who may benefit from EC/IC-bypass surgery.

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