Staged embolisation of a giant torcular dural sinus malformation in a neonate

2021 ◽  
Vol 14 (11) ◽  
pp. e244216
Author(s):  
Evan M Luther ◽  
Aria Jamshidi ◽  
Hunter King ◽  
Robert Starke

Torcular dural sinus malformations (tDSMs) represent a rare subset of paediatric cerebrovascular malformations and are often diagnosed antenatally via ultrasound. The management of these in utero lesions remains controversial as previous studies suggested elective termination of the pregnancy because of their presumably high mortality and severe long-term morbidity. However, more recent evaluations have suggested that the overall prognosis for infants harbouring these lesions may be much better than previously believed. As such, we present the case of a neonate with a giant tDSM, diagnosed in utero, who was treated postnatally via staged transarterial and transvenous embolisation to alleviate worsening obstructive hydrocephalus and brainstem compression. We provide details regarding the surgical approach and long-term neurological outcomes for this patient. To the best of our knowledge, this is one of the largest reported tDSM presented in the literature.

Author(s):  
Matthew McInnes ◽  
Katherine Fong ◽  
Andrea Grin ◽  
Karel ter Brugge ◽  
Susan Blaser ◽  
...  

Background:Dural sinus malformation (DSM) is a term used to describe congenital vascular malformations characterized by massive dilation of one or more dural sinuses: these dilatations are typically associated with arteriovenous shunts. Such malformations can present antenatally but their early natural history and anatomy is poorly defined.Methods:We reviewed five years of autopsy experience and retrieved three primary vascular malformations of the fetal dural sinuses with ultrasound, magnetic resonance imaging (MRI) and post-mortem correlation.Results:Fetal ultrasound and MRI obtained between 19 and 23 weeks gestational age demonstrated in all cases dilation of the dural sinuses. In two cases vascular thromboses were present in either the dilated dural sinus (one of three) or the associated arteriovenous fistula (one of three). All cases were autopsied at 22-23 weeks gestational age. In one there was imaging and autopsy evidence of remodeling of the dural sinuses associated with a pial arteriovenous fistula. In two cases, no arteriovenous malformation was identified on initial imaging, but only became evident at autopsy. One case showed morphological overlap with vein of Galen aneurysmal malformation, with a midline arteriovenous shunt and vein of Galen ectasia. The other demonstrated a perisylvian dural arteriovenous fistula.Conclusion:In utero thrombosis of feeding vascular malformations or of the dural sinus malformation may be prominent. The early in utero developmental trajectory of dural sinus malformation (DSM) is poorly defined and deserves further study.


2019 ◽  
Vol 12 (3) ◽  
pp. e227808
Author(s):  
Zena Tansley-Ahmed ◽  
Kit Wu ◽  
Loucas Christodoulou

Acute cerebellitis (AC) is a principal cause of acute cerebellar dysfunction in previously well children. Although the condition is usually benign, fatal complications include obstructive hydrocephalus and brainstem compression; therefore, prompt accurate diagnosis is vital.1There are various pathogens reported in the literature as aetiological agents of AC; however, adenovirus is very rarely mentioned, with only one previous case report in the literature to the best of our knowledge.2This case demonstrates the importance of recognising adenovirus as a cause of AC, particularly when preceded by a respiratory tract infection in the paediatric age group. Furthermore, we highlight the role of early neuroimaging in differentiating AC from other causes of acute cerebellar dysfunction, which require different management. Our patient made a full recovery with no long-term deficits demonstrating that comprehensive investigation and consideration of atypical pathogens in the context of AC is vital in securing a favourable outcome.


2017 ◽  
Vol 10 (7) ◽  
pp. 669-674 ◽  
Author(s):  
Nicholas K Cheung ◽  
Albert HY Chiu ◽  
Andrew K Cheung ◽  
Jason D Wenderoth

BackgroundPreliminary short-term results for stent-assisted coil embolization (SACE) using woven/braided stents have been promising. However, evidence supporting mid- to long-term efficacy and durability is lacking.ObjectiveTo report the long-term results for the durability of elective intracranial aneurysms treated with woven stents.Materials and methodsBetween May 2012 and May 2015, 98 consecutive patients with 103 aneurysms underwent elective woven SACE across three Australian neurovascular centres. All patients had immediate, 6- and 18-month clinical and radiological follow-up. Radiological assessment was performed with modified Raymond–Roy occlusion scores based on angiography results, while clinical assessment was based on the modified Rankin Scale.ResultsSix-month follow-up was available in 100 aneurysms, and an 18-month follow-up in 97 aneurysms. Total occlusion rates of 82% were achieved at inception, 82% at 6 months, and 90% at 18 months. Satisfactory occlusion with small neck remnants was present in 17% at inception, 16% at 6 months, and 9% at 18 months. Good neurological outcomes were achieved in 95% at 18 months. Intraprocedural thromboembolic events were recorded in 3% and delayed events in 1% (all in patients taking clopidogrel). Aneurysm recurrence occurred in one patient (1%). Technical complications occurred in 5%. The total complication rate was 10%.ConclusionsWoven SACE is safe, efficacious, and durable at long-term 18-month follow-up, with very low recurrence and re-treatment rates. Preliminary results appear better than those for traditional laser-cut stents.


Author(s):  
Jerry C. Ku ◽  
Brian Hanak ◽  
Prakash Muthusami ◽  
Karl Narvacan ◽  
Hidy Girgis ◽  
...  

OBJECTIVE Torcular dural sinus malformations (tDSMs) are rare pediatric cerebrovascular malformations characterized by giant venous lakes localized to the midline confluence of sinuses. Historical clinical outcomes of patients with these lesions were poor, though better prognoses have been reported in the more recent literature. Long-term outcomes in children with tDSMs are uncertain and require further characterization. The goal of this study was to review a cohort of tDSM patients with an emphasis on long-term outcomes and to describe the treatment strategy. METHODS This study is a single-center retrospective review of a prospectively maintained data bank including patients referred to and cared for at The Hospital for Sick Children for tDSM from January 1996 to March 2019. Each patient’s clinical, radiological, and demographic information, as well as their mother’s demographic information, was collected for review. RESULTS Ten patients with tDSM, with a mean follow-up of 58 months, were included in the study. Diagnoses were made antenatally in 8 patients, and among those cases, 4 families opted for either elective termination (n = 1) or no further care following delivery (n = 3). Of the 6 patients treated, 5 had a favorable long-term neurological outcome, and follow-up imaging demonstrated a decrease or stability in the size of the tDSM over time. Staged embolization was performed in 3 patients, and anticoagulation was utilized in 5 treated patients. CONCLUSIONS The authors add to a growing body of literature indicating that clinical outcomes in tDSM may not be as poor as initially perceived. Greater awareness of the lesion’s natural history and pathophysiology, advancing endovascular techniques, and individualized anticoagulation regimens may lead to continued improvement in outcomes.


Author(s):  
U Schneider ◽  
A Fiedler ◽  
A Lesser ◽  
G Kasprian ◽  
J Intek ◽  
...  

2019 ◽  
Author(s):  
WH Becker ◽  
P Kunkel ◽  
F Brassel

2019 ◽  
Vol 24 (5) ◽  
pp. 558-571 ◽  
Author(s):  
Kartik Bhatia ◽  
Hans Kortman ◽  
Christopher Blair ◽  
Geoffrey Parker ◽  
David Brunacci ◽  
...  

OBJECTIVEThe role of mechanical thrombectomy in pediatric acute ischemic stroke is uncertain, despite extensive evidence of benefit in adults. The existing literature consists of several recent small single-arm cohort studies, as well as multiple prior small case series and case reports. Published reports of pediatric cases have increased markedly since 2015, after the publication of the positive trials in adults. The recent AHA/ASA Scientific Statement on this issue was informed predominantly by pre-2015 case reports and identified several knowledge gaps, including how young a child may undergo thrombectomy. A repeat systematic review and meta-analysis is warranted to help guide therapeutic decisions and address gaps in knowledge.METHODSUsing PRISMA-IPD guidelines, the authors performed a systematic review of the literature from 1999 to April 2019 and individual patient data meta-analysis, with 2 independent reviewers. An additional series of 3 cases in adolescent males from one of the authors’ centers was also included. The primary outcomes were the rate of good long-term (mRS score 0–2 at final follow-up) and short-term (reduction in NIHSS score by ≥ 8 points or NIHSS score 0–1 at up to 24 hours post-thrombectomy) neurological outcomes following mechanical thrombectomy for acute ischemic stroke in patients < 18 years of age. The secondary outcome was the rate of successful angiographic recanalization (mTICI score 2b/3).RESULTSThe authors’ review yielded 113 cases of mechanical thrombectomy in 110 pediatric patients. Although complete follow-up data are not available for all patients, 87 of 96 (90.6%) had good long-term neurological outcomes (mRS score 0–2), 55 of 79 (69.6%) had good short-term neurological outcomes, and 86 of 98 (87.8%) had successful angiographic recanalization (mTICI score 2b/3). Death occurred in 2 patients and symptomatic intracranial hemorrhage in 1 patient. Sixteen published thrombectomy cases were identified in children < 5 years of age.CONCLUSIONSMechanical thrombectomy may be considered for acute ischemic stroke due to large vessel occlusion (ICA terminus, M1, basilar artery) in patients aged 1–18 years (Level C evidence; Class IIb recommendation). The existing evidence base is likely affected by selection and publication bias. A prospective multinational registry is recommended as the next investigative step.


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