Outcome after surgical or conservative management of cerebral cavernous malformations

Fiona Moultrie, Margaret A Horne, Colin B Josephson, Julie M Hall, Carl E Counsell, Jo J Bhattacharya, Vakis Papanastassiou, Robin J Sellar, Charles P Warlow, Gordon D Murray, Rustam Al-Shahi Salman, On behalf of the Scottish Audit of Intracranial Vascular Malformations (SAIVMs) steering committee and collaborators

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Abstract

OBJECTIVE: There have been few comparative studies of microsurgical excision vs conservative management of cerebral cavernous malformations (CCM) and none of them has reliably demonstrated a statistically and clinically significant difference.METHODS: We conducted a prospective, population-based study to identify and independently validate definite CCM diagnoses first made in 1999-2003 in Scottish adult residents. We used multiple sources of prospective follow-up to assess adults' dependence and to identify and independently validate outcome events. We used univariate and multivariable survival analyses to test the influence of CCM excision on outcome, adjusted for prognostic factors and baseline imbalances.RESULTS: Of 134 adults, 25 underwent CCM excision; these adults were younger (34 vs 43 years at diagnosis, p = 0.004) and more likely to present with symptomatic intracranial hemorrhage or focal neurologic deficit than adults managed conservatively (48% vs 26%; odds ratio 2.7, 95% confidence interval [CI] 1.1-6.5). During 5 years of follow-up, CCM excision was associated with a deterioration to an Oxford Handicap Scale score 2-6 sustained over at least 2 successive years (adjusted hazard ratio [HR] 2.2, 95% CI 1.1-4.3) and the occurrence of symptomatic intracranial hemorrhage or new focal neurologic deficit (adjusted HR 3.6, 95% CI 1.3-10.0).CONCLUSIONS: CCM excision was associated with worse outcomes over 5 years compared to conservative management. Long-term follow-up will determine whether this difference is sustained over patients' lifetimes. Meanwhile, a randomized controlled trial appears justified.CLASSIFICATION OF EVIDENCE: This study provides Class III evidence that CCM excision worsens short-term disability scores and increases the risk of symptomatic intracranial hemorrhage and new focal neurologic deficits.

Original languageEnglish
Pages (from-to)582-589
Number of pages8
JournalNeurology
Volume83
Issue number7
DOIs
Publication statusPublished - 3 Jul 2014

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Central Nervous System Cavernous Hemangioma
Intracranial Hemorrhages
Neurologic Manifestations
Confidence Intervals
Survival Analysis
Conservative Treatment
Young Adult
Randomized Controlled Trials
Odds Ratio

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Moultrie, F., Horne, M. A., Josephson, C. B., Hall, J. M., Counsell, C. E., Bhattacharya, J. J., ... On behalf of the Scottish Audit of Intracranial Vascular Malformations (SAIVMs) steering committee and collaborators (2014). Outcome after surgical or conservative management of cerebral cavernous malformations. Neurology, 83(7), 582-589. https://doi.org/10.1212/WNL.0000000000000684

Outcome after surgical or conservative management of cerebral cavernous malformations. / Moultrie, Fiona; Horne, Margaret A; Josephson, Colin B; Hall, Julie M; Counsell, Carl E; Bhattacharya, Jo J; Papanastassiou, Vakis; Sellar, Robin J; Warlow, Charles P; Murray, Gordon D; Al-Shahi Salman, Rustam; On behalf of the Scottish Audit of Intracranial Vascular Malformations (SAIVMs) steering committee and collaborators.

In: Neurology, Vol. 83, No. 7, 03.07.2014, p. 582-589.

Research output: Contribution to journalArticle

Moultrie, F, Horne, MA, Josephson, CB, Hall, JM, Counsell, CE, Bhattacharya, JJ, Papanastassiou, V, Sellar, RJ, Warlow, CP, Murray, GD, Al-Shahi Salman, R & On behalf of the Scottish Audit of Intracranial Vascular Malformations (SAIVMs) steering committee and collaborators 2014, 'Outcome after surgical or conservative management of cerebral cavernous malformations', Neurology, vol. 83, no. 7, pp. 582-589. https://doi.org/10.1212/WNL.0000000000000684
Moultrie, Fiona ; Horne, Margaret A ; Josephson, Colin B ; Hall, Julie M ; Counsell, Carl E ; Bhattacharya, Jo J ; Papanastassiou, Vakis ; Sellar, Robin J ; Warlow, Charles P ; Murray, Gordon D ; Al-Shahi Salman, Rustam ; On behalf of the Scottish Audit of Intracranial Vascular Malformations (SAIVMs) steering committee and collaborators. / Outcome after surgical or conservative management of cerebral cavernous malformations. In: Neurology. 2014 ; Vol. 83, No. 7. pp. 582-589.
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T1 - Outcome after surgical or conservative management of cerebral cavernous malformations

AU - Moultrie, Fiona

AU - Horne, Margaret A

AU - Josephson, Colin B

AU - Hall, Julie M

AU - Counsell, Carl E

AU - Bhattacharya, Jo J

AU - Papanastassiou, Vakis

AU - Sellar, Robin J

AU - Warlow, Charles P

AU - Murray, Gordon D

AU - Al-Shahi Salman, Rustam

AU - On behalf of the Scottish Audit of Intracranial Vascular Malformations (SAIVMs) steering committee and collaborators

N1 - © 2014 American Academy of Neurology. Affiliations: From the Division of Clinical Neurosciences, Centre for Clinical Brain Sciences (F.M., C.B.J., J.M.H., R.J.S., C.P.W., R.A.-S.S.), and Centre for Population Health Sciences (M.A.H., G.D.M.), University of Edinburgh; the Division of Applied Health Sciences (C.E.C.), University of Aberdeen; and the Institute of Neurological Sciences (J.J.B., V.P.), Southern General Hospital, Glasgow, UK.

PY - 2014/7/3

Y1 - 2014/7/3

N2 - OBJECTIVE: There have been few comparative studies of microsurgical excision vs conservative management of cerebral cavernous malformations (CCM) and none of them has reliably demonstrated a statistically and clinically significant difference.METHODS: We conducted a prospective, population-based study to identify and independently validate definite CCM diagnoses first made in 1999-2003 in Scottish adult residents. We used multiple sources of prospective follow-up to assess adults' dependence and to identify and independently validate outcome events. We used univariate and multivariable survival analyses to test the influence of CCM excision on outcome, adjusted for prognostic factors and baseline imbalances.RESULTS: Of 134 adults, 25 underwent CCM excision; these adults were younger (34 vs 43 years at diagnosis, p = 0.004) and more likely to present with symptomatic intracranial hemorrhage or focal neurologic deficit than adults managed conservatively (48% vs 26%; odds ratio 2.7, 95% confidence interval [CI] 1.1-6.5). During 5 years of follow-up, CCM excision was associated with a deterioration to an Oxford Handicap Scale score 2-6 sustained over at least 2 successive years (adjusted hazard ratio [HR] 2.2, 95% CI 1.1-4.3) and the occurrence of symptomatic intracranial hemorrhage or new focal neurologic deficit (adjusted HR 3.6, 95% CI 1.3-10.0).CONCLUSIONS: CCM excision was associated with worse outcomes over 5 years compared to conservative management. Long-term follow-up will determine whether this difference is sustained over patients' lifetimes. Meanwhile, a randomized controlled trial appears justified.CLASSIFICATION OF EVIDENCE: This study provides Class III evidence that CCM excision worsens short-term disability scores and increases the risk of symptomatic intracranial hemorrhage and new focal neurologic deficits.

AB - OBJECTIVE: There have been few comparative studies of microsurgical excision vs conservative management of cerebral cavernous malformations (CCM) and none of them has reliably demonstrated a statistically and clinically significant difference.METHODS: We conducted a prospective, population-based study to identify and independently validate definite CCM diagnoses first made in 1999-2003 in Scottish adult residents. We used multiple sources of prospective follow-up to assess adults' dependence and to identify and independently validate outcome events. We used univariate and multivariable survival analyses to test the influence of CCM excision on outcome, adjusted for prognostic factors and baseline imbalances.RESULTS: Of 134 adults, 25 underwent CCM excision; these adults were younger (34 vs 43 years at diagnosis, p = 0.004) and more likely to present with symptomatic intracranial hemorrhage or focal neurologic deficit than adults managed conservatively (48% vs 26%; odds ratio 2.7, 95% confidence interval [CI] 1.1-6.5). During 5 years of follow-up, CCM excision was associated with a deterioration to an Oxford Handicap Scale score 2-6 sustained over at least 2 successive years (adjusted hazard ratio [HR] 2.2, 95% CI 1.1-4.3) and the occurrence of symptomatic intracranial hemorrhage or new focal neurologic deficit (adjusted HR 3.6, 95% CI 1.3-10.0).CONCLUSIONS: CCM excision was associated with worse outcomes over 5 years compared to conservative management. Long-term follow-up will determine whether this difference is sustained over patients' lifetimes. Meanwhile, a randomized controlled trial appears justified.CLASSIFICATION OF EVIDENCE: This study provides Class III evidence that CCM excision worsens short-term disability scores and increases the risk of symptomatic intracranial hemorrhage and new focal neurologic deficits.

U2 - 10.1212/WNL.0000000000000684

DO - 10.1212/WNL.0000000000000684

M3 - Article

VL - 83

SP - 582

EP - 589

JO - Neurology

JF - Neurology

SN - 0028-3878

IS - 7

ER -