Improving long-term outcomes in pediatric torcular dural sinus malformations with embolization and anticoagulation: a retrospective review of The Hospital for Sick Children experience

Jerry C. Ku MD1,2, Brian Hanak MD6, Prakash Muthusami MD3, Karl Narvacan MD1,2, Hidy Girgis MDCM, PharmD4, Karel terBrugge MD, FRCPC3,5, Timo Krings MD, PhD, FRCPC1,3,5, James T. Rutka MD, PhD, FRCSC1,2, and Peter Dirks MD, PhD, FRCSC1,2
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  • 1 Division of Neurosurgery, Department of Surgery, University of Toronto, Toronto;
  • | 2 Division of Neurosurgery, The Hospital for Sick Children, Toronto;
  • | 3 Department of Diagnostic Imaging, The Hospital for Sick Children, Toronto;
  • | 4 Division of Neurosurgery, Department of Surgery, University of Ottawa, Ottawa;
  • | 5 Division of Neuroradiology, Department of Medical Imaging, Toronto Western Hospital, University Health Network, Toronto, Ontario, Canada; and
  • | 6 Department of Neurosurgery, Loma Linda University Health, Loma Linda, California
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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.

ABBREVIATIONS

DAVF = dural arteriovenous fistula; DSM = dural sinus malformation; ICP = intracranial pressure; NBCA = N-butyl cyanoacrylate; SSS = superior sagittal sinus; SVD = spontaneous vaginal delivery; SWS = Sturge-Weber syndrome; tDSM = torcular DSM.

Supplementary Materials

    • Supplementary Table 1 (PDF 440 KB)

Image from Mavridis et al. (pp 404–415).

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