<p>Spinal cord cavernous malformations (SCCMs) are rare lesions believed to have moderate rates of debilitating hemorrhages, often requiring complex treatment. Previous studies have reported vastly different rates of hemorrhagic outcomes over time depending on treatments. In this meta-analysis, we aim to assess the long-term natural history of hemorrhagic events and risk in patients with a SCCM.&#xa0;Preferred reporting items for systematic reviews and meta-analysis guidelines (PRISMA) were utilized in this work. We extracted demographic data, follow-up lengths, lesion characteristics and locations, symptoms, and hemorrhagic complication rates. A random-effects meta-analysis was conducted to estimate the overall event rate per patient-year across studies. Individual patient-level data was extracted to reconstruct a Kaplan-Meier curve estimating cumulative incidence of hemorrhagic events over time.&#xa0;Eight studies were included, resulting in 769 patients, with a mean follow-up of 48.8 months. Mean age of patients was 40.2 ± 11.7 years and 337 were female. Mean lesion size was 9.57 ± 5.31&#xa0;mm and locations were 280 (36.4%) cervical, 397 (51.5%) thoracic, and 93 (12.1%) lumbar. Most common symptoms were pain (20.6%), weakness (27.3%), sensory deficits (33.6%), and bowel/bladder dysfunction (18.5%). A total of 118 hemorrhagic events occurred over 2390 person-years. The random-effects meta-analysis model gave a pooled event hemorrhage rate of 4.7% (95% CI 3.0–6.3) per year, and up to 21.9% in five years. The one-year cumulative risk of hemorrhage via Kaplan-Meier was calculated as 15.4% and the five-year cumulative risk of hemorrhage was calculated to be 58%.&#xa0;SCCMs report an annual hemorrhage risk of 4.7%, presenting mostly in thoracic regions with sensory deficits and weakness, with cumulative risk of hemorrhage reaching as high as 58% within five years on Kaplan-Meier curve analysis. Greater prospective cohorts of SCCM patients tracking hemorrhagic events and neurological deterioration from first onset of symptoms, with greater follow-up data, would be beneficial to further elucidating the natural course of SCCMs.</p>

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Natural history of hemorrhagic events in spinal cord cavernous malformation: an updated systematic review and Meta-Analysis

  • Aryan Wadhwa,
  • Samuel D. Pettersson,
  • Neal A. Shah,
  • Kyle Bui,
  • Naveen Arunachalam Sakthiyendran,
  • Shashvat Purohit,
  • Christopher S. Ogilvy

摘要

Spinal cord cavernous malformations (SCCMs) are rare lesions believed to have moderate rates of debilitating hemorrhages, often requiring complex treatment. Previous studies have reported vastly different rates of hemorrhagic outcomes over time depending on treatments. In this meta-analysis, we aim to assess the long-term natural history of hemorrhagic events and risk in patients with a SCCM. Preferred reporting items for systematic reviews and meta-analysis guidelines (PRISMA) were utilized in this work. We extracted demographic data, follow-up lengths, lesion characteristics and locations, symptoms, and hemorrhagic complication rates. A random-effects meta-analysis was conducted to estimate the overall event rate per patient-year across studies. Individual patient-level data was extracted to reconstruct a Kaplan-Meier curve estimating cumulative incidence of hemorrhagic events over time. Eight studies were included, resulting in 769 patients, with a mean follow-up of 48.8 months. Mean age of patients was 40.2 ± 11.7 years and 337 were female. Mean lesion size was 9.57 ± 5.31 mm and locations were 280 (36.4%) cervical, 397 (51.5%) thoracic, and 93 (12.1%) lumbar. Most common symptoms were pain (20.6%), weakness (27.3%), sensory deficits (33.6%), and bowel/bladder dysfunction (18.5%). A total of 118 hemorrhagic events occurred over 2390 person-years. The random-effects meta-analysis model gave a pooled event hemorrhage rate of 4.7% (95% CI 3.0–6.3) per year, and up to 21.9% in five years. The one-year cumulative risk of hemorrhage via Kaplan-Meier was calculated as 15.4% and the five-year cumulative risk of hemorrhage was calculated to be 58%. SCCMs report an annual hemorrhage risk of 4.7%, presenting mostly in thoracic regions with sensory deficits and weakness, with cumulative risk of hemorrhage reaching as high as 58% within five years on Kaplan-Meier curve analysis. Greater prospective cohorts of SCCM patients tracking hemorrhagic events and neurological deterioration from first onset of symptoms, with greater follow-up data, would be beneficial to further elucidating the natural course of SCCMs.