scholarly journals Case of de novo cerebral microbleeds in ischemic-type pediatric moyamoya disease

2021 ◽  
Vol 12 ◽  
pp. 284
Author(s):  
Kohei Inoue ◽  
Akihiko Momozaki ◽  
Takashi Furukawa ◽  
Fumitaka Yoshioka ◽  
Atsushi Ogata ◽  
...  

Background: Studies on pediatric patients with moyamoya disease who presented with de novo cerebral microbleeds (CMBs) are extremely rare. Case Description: Herein, we report a 7-year-old boy with moyamoya disease who had de novo CMBs during treatment. He presented with transient left-side motor weakness and was diagnosed with moyamoya disease. He underwent revascularization surgery on the right cerebral hemisphere. Six months after the surgery, he presented with transient right-side motor weakness and MRA revealed progression of stenosis in the left middle cerebral artery. After another 3 months, three de novo CMBs were identified. He underwent revascularization surgery on the left side. The symptom disappeared completely after surgery and no additional de novo CMBs were identified 1 year after surgery. Conclusion: This is the first report on de novo CMBs in pediatric patients. Although the significance of de novo CMBs in pediatric patients is completely unknown, attention should be paid to not only ischemic stroke but also hemorrhagic stroke. Although the short-term course is good in the current case, follow-up period is too short to assess for rebleeding and long-term follow-up is still important. Further, more cases should be collected.

2014 ◽  
Vol 13 (6) ◽  
pp. 647-649 ◽  
Author(s):  
Miki Fujimura ◽  
Naoto Kimura ◽  
Masayuki Ezura ◽  
Kuniyasu Niizuma ◽  
Hiroshi Uenohara ◽  
...  

The development of a de novo arteriovenous malformation (AVM) in patients with moyamoya disease is extremely rare. A 14-year-old girl developed an AVM in the right occipital lobe during the 4-year postoperative period following successful bilateral revascularization surgeries. She suffered a transient ischemic attack with hemodynamic compromise of the bilateral hemispheres at the age of 10 years. Results of an initial examination by 1.5-T MRI and MR angiography satisfied the diagnostic criteria of moyamoya disease but failed to detect any vascular malformation. Bilateral direct and indirect revascularization surgeries in the anterior circulation relieved her symptoms, and she underwent MRI and MR angiography follow-up every year after surgery. Serial T2-weighted MRI revealed the gradual appearance of flow voids in the right occipital lobe during the follow-up period. Magnetic resonance angiography ultimately indicated the development of an AVM 4 years after these surgeries when catheter angiography confirmed the diagnosis of an AVM in the right occipital lobe. The AVM remained asymptomatic, and the patient remained free of cerebrovascular events during the time she was observed by the authors. Acquired AVM in moyamoya disease is extremely rare, with only 3 pediatric cases including the present case being reported in the literature. The development of a de novo AVM in a postoperative patient with moyamoya disease appears to be unique, and this case may provide insight into the dynamic pathology of AVMs.


2017 ◽  
Vol 10 (2) ◽  
pp. 101
Author(s):  
Shohela Akhter ◽  
Rumana Riaaz ◽  
Roushan Jahan ◽  
Shahana A. Rahman

<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>


2018 ◽  
Vol 21 (6) ◽  
pp. 632-638 ◽  
Author(s):  
Tomomi Kimiwada ◽  
Toshiaki Hayashi ◽  
Reizo Shirane ◽  
Teiji Tominaga

OBJECTIVESome pediatric patients with moyamoya disease (MMD) present with posterior cerebral artery (PCA) stenosis before and after anterior circulation revascularization surgery and require posterior circulation revascularization surgery. This study evaluated the factors associated with PCA stenosis and assessed the efficacy of posterior circulation revascularization surgery, including occipital artery (OA)–PCA bypass, in pediatric patients with MMD.METHODSThe presence of PCA stenosis before and after anterior circulation revascularization surgery and its clinical characteristics were investigated in 62 pediatric patients (< 16 years of age) with MMD.RESULTSTwenty-three pediatric patients (37%) with MMD presented with PCA stenosis at the time of the initial diagnosis. A strong correlation between the presence of infarction and PCA stenosis before anterior revascularization was observed (p < 0.001). In addition, progressive PCA stenosis was observed in 12 patients (19.4%) after anterior revascularization. The presence of infarction and a younger age at the time of initial diagnosis were risk factors for progressive PCA stenosis after anterior revascularization (p < 0.001 and p = 0.002, respectively). Posterior circulation revascularization surgery, including OA-PCA bypass, was performed in 9 of the 12 patients with progressive PCA stenosis, all of whom showed symptomatic and/or radiological improvement.CONCLUSIONSPCA stenosis is an important clinical factor related to poor prognosis in pediatric MMD. One should be aware of the possibility of progressive PCA stenosis during the postoperative follow-up period and consider performing posterior circulation revascularization surgery.


1996 ◽  
Vol 85 (5) ◽  
pp. 772-776 ◽  
Author(s):  
Kiyohiro Houkin ◽  
Hiroshi Abe ◽  
Tetsuyuki Yoshimoto ◽  
Akihiro Takahashi

✓ Whether a diagnosis of moyamoya disease is justified in patients with typical angiographic evidence of moyamoya disease unilaterally and normal angiographic findings contralaterally remains controversial. In this study the authors analyzed longitudinal angiographic change, familial occurrence, and basic fibroblast growth factor (bFGF) concentration in the cerebrospinal fluid (CSF) of patients with “unilateral” moyamoya disease. Over a 10-year period, 10 cases of unilateral moyamoya disease were followed using conventional angiography or magnetic resonance angiography. Basic FGF in CSF, obtained from the subarachnoid space of the cerebral cortex during revascularization surgery, was measured in five cases. Among the 10 cases of unilateral moyamoya disease, only one pediatric case showed obvious signs of progression to typical bilateral disease. The other nine cases (including six adults and three children) remained stable throughout follow-up radiological examinations (magnetic resonance angiography) with a mean observation period of 3.5 years. There was no familial occurrence in these cases of unilateral moyamoya disease. Levels of bFGF, which are high in typical moyamoya disease, were low in these patients. The progression from unilateral moyamoya disease to the typical bilateral form of the disease appears to be infrequent. The low levels of bFGF in the CSF of these patients and the lack of familial occurrence strongly suggest that most cases of unilateral moyamoya disease, especially those found in adults, are distinct from typical bilateral moyamoya disease.


2017 ◽  
Vol 28 (04) ◽  
pp. 378-382 ◽  
Author(s):  
Sung Lee

Introduction Hydrocele of the canal of Nuck (HCN) causes an inguinal swelling in pediatric females similar to that observed in case of inguinal hernia. HCN should be considered in the differential diagnosis of hernia in such children. Although laparoscopic operations have been reported in several cases for the treatment of HCN in adults, they have not been reported for the treatment in pediatrics. This study aimed to evaluate the outcomes of laparoscopic intracorporeal hydrocelectomy with high ligation as a treatment for HCN in pediatrics. Materials and Methods I retrospectively reviewed the charts of 26 pediatric females aged ≤10 years who underwent laparoscopic hydrocelectomy with high ligation at Damsoyu Hospital, Seoul, Republic of Korea from September 2012 to December 2016. Results The mean age was 3.31 (1–10) years. HCN was on the right in 11 patients and on the left in 15. The presenting symptom in all cases was inguinal swelling. HCN was of the hourglass type inside the inguinal canal in 17 patients and in the abdominal cavity outside the inguinal canal in 9. All patients were treated with laparoscopic hydrocelectomy with high ligation. The mean operating time was 17.8 minute (15–30). The wounds were scarcely visible immediately after surgery. There were no postoperative complications and no recurrences on follow-up. Conclusion Laparoscopic intracorporeal hydrocelectomy with high ligation is effective for HCN in pediatrics owing to its short operating time, excellent cosmesis, and no recurrence.


2009 ◽  
Vol 111 (5) ◽  
pp. 927-935 ◽  
Author(s):  
Raphael Guzman ◽  
Marco Lee ◽  
Achal Achrol ◽  
Teresa Bell-Stephens ◽  
Michael Kelly ◽  
...  

Object Moyamoya disease (MMD) is a rare cerebrovascular disease mainly described in the Asian literature. To address a lack of data on clinical characteristics and long-term outcomes in the treatment of MMD in North America, the authors analyzed their experience at Stanford University Medical Center. They report on a consecutive series of patients treated for MMD and detail their demographics, clinical characteristics, and long-term surgical outcomes. Methods Data obtained in consecutive series of 329 patients with MMD treated microsurgically by the senior author (G.K.S.) between 1991 and 2008 were analyzed. Demographic, clinical, and surgical data were prospectively gathered and neurological outcomes assessed in postoperative follow-up using the modified Rankin Scale. Association of demographic, clinical, and surgical data with postoperative outcome was assessed by chi-square, uni- and multivariate logistic regression, and Kaplan-Meier survival analyses. Results The authors treated a total of 233 adult patients undergoing 389 procedures (mean age 39.5 years) and 96 pediatric patients undergoing 168 procedures (mean age 10.1 years). Direct revascularization technique was used in 95.1% of adults and 76.2% of pediatric patients. In 264 patients undergoing 450 procedures (mean follow-up 4.9 years), the surgical morbidity rate was 3.5% and the mortality rate was 0.7% per treated hemisphere. The cumulative 5-year risk of perioperative or subsequent stroke or death was 5.5%. Of the 171 patients presenting with a transient ischemic attack, 91.8% were free of transient ischemic attacks at 1 year or later. Overall, there was a significant improvement in quality of life in the cohort as measured using the modified Rankin Scale (p < 0.0001). Conclusions Revascularization surgery in patients with MMD carries a low risk, is effective at preventing future ischemic events, and improves quality of life. Patients in whom symptomatic MMD is diagnosed should be offered revascularization surgery.


2012 ◽  
Vol 18 (4) ◽  
pp. 484-489 ◽  
Author(s):  
C-H Yeh ◽  
Y-M Wu ◽  
Y-L Chen ◽  
H-F Wong

We describe our experience of the development of contralateral de novo intraosseous AVMs in a ten-year-old girl with AVMs of the mandible who underwent endovascular embolotherapy. She initially presented with intermittent oral bleeding. Computed tomography and digital subtraction angiography demonstrated intraosseous AVMs within the right mandible. The AVMs were treated by transosseous direct-puncture and transarterial embolization with Guglielmi detachable coils and n-butyl cyanoacrylate glue. However, de novo intraosseous AVMs developed within the previously healthy contralateral mandible and resulted in dangerous oral bleeding. Therefore, we suggest regular follow-up and prompt retreatment of any residual mandibular AVMs in patients undergoing endovascular or surgical treatment to prevent subsequent development of “secondary” AVMs and life-threatening oral bleeding.


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