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Multimodality management and outcomes of brain arterio-venous malformations (AVMs) in children: personal experience and review of the literature, with specific emphasis on age at first AVM bleed.

Abstract The purpose of this paper is to study the presentation and analyse the results of multimodality treatment of brain arterio-venous malformations (AVMs) in children at our centre and review age at first AVM rupture in the literature.
PMID
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Authors

Mayor MeshTerms

Disease Management

Treatment Outcome

Keywords

DSA

Embolisation

Microsurgery

Outcome

Radiosurgery

Ruptured AVMs

Journal Title child's nervous system : chns : official journal of the international society for pediatric neurosurgery
Publication Year Start




PMID- 28324183
OWN - NLM
STAT- MEDLINE
DA  - 20170321
DCOM- 20170417
LR  - 20170421
IS  - 1433-0350 (Electronic)
IS  - 0256-7040 (Linking)
VI  - 33
IP  - 4
DP  - 2017 Apr
TI  - Multimodality management and outcomes of brain arterio-venous malformations
      (AVMs) in children: personal experience and review of the literature, with
      specific emphasis on age at first AVM bleed.
PG  - 573-581
LID - 10.1007/s00381-017-3383-4 [doi]
AB  - PURPOSE: The purpose of this paper is to study the presentation and analyse the
      results of multimodality treatment of brain arterio-venous malformations (AVMs)
      in children at our centre and review age at first AVM rupture in the literature. 
      METHODS: Of 52 patients aged <18 years, 47 with brain AVMs (27 males and 20
      females) aged 4-17 years (mean 12.2) were retrospectively reviewed. PubMed search
      revealed five additional studies including 267 patients where the prevalence of
      age-related AVMs rupture was analysed. RESULTS: In our study, 37 patients had
      bled, 9 were symptomatic without haemorrhage and 1 was incidental.
      Spetzler-Martin score distribution was 5 cases grade I, 18 grade II, 21 grade III
      and 3 grade IV. Appropriate imaging was performed, either CT/MRI angiogram only
      (in emergency cases) or catheter angiogram, prior to definitive treatment. There 
      were 40 supratentorial and 7 infratentorial AVMs. Twenty-nine patients had
      microsurgery alone and 9 patients were treated by radiosurgery only. Three
      patients were embolised, all followed by radiosurgery, with one requiring surgery
      too, while 4 patients had combined surgery and radiosurgery. One patient is
      awaiting radiosurgery while another was not treated. Good outcomes, classified as
      modified Rankin score (mRS) 0-2 improved significantly after intervention to
      89.4% from 38.3% pre-treatment (p value <0.0001). Angiography confirmed 96.6%
      obliteration after first planned operation. Repeat cerebral angiogram around age 
      18 was negative in all previously cured patients. Reviewing the literature, 82.0%
      (95% CI = [77-87]; N = 267) of children diagnosed with brain AVMs (mean age 11.4 
      +/- 0.4) presented with a bleed in the last 22 years. Males significantly
      outnumbered females (136 vs 84) (p < 0.001). Ninety-five patients underwent
      surgical intervention alone when compared to other treatment modalities (p <
      0.001). CONCLUSIONS: Microsurgical excision of surgically accessible intracranial
      AVMs remains the primary treatment option with very good outcomes. A significant 
      number of patients' AVMs ruptured around puberty; therefore, understanding the
      pathophysiology of AVM instability at this age may aid future therapy.
FAU - Shtaya, Anan
AU  - Shtaya A
AD  - Wessex Neurological Centre, University Hospital Southampton, Southampton, UK.
      [email protected]
AD  - Atkinson Morley Neurosurgery Centre, Academic Neurosurgery Unit, St George's,
      University of London, London, SW17 0RE, UK. [email protected]
FAU - Millar, John
AU  - Millar J
AD  - Wessex Neurological Centre, University Hospital Southampton, Southampton, UK.
FAU - Sparrow, Owen
AU  - Sparrow O
AD  - Wessex Neurological Centre, University Hospital Southampton, Southampton, UK.
LA  - eng
PT  - Journal Article
PT  - Review
DEP - 20170321
PL  - Germany
TA  - Childs Nerv Syst
JT  - Child's nervous system : ChNS : official journal of the International Society for
      Pediatric Neurosurgery
JID - 8503227
SB  - IM
MH  - Adolescent
MH  - Arteriovenous Fistula/diagnostic imaging/*therapy
MH  - Brain/diagnostic imaging
MH  - Child
MH  - Child, Preschool
MH  - *Disease Management
MH  - Female
MH  - Humans
MH  - Intracranial Arteriovenous Malformations/diagnostic imaging/*therapy
MH  - Male
MH  - Microsurgery/methods
MH  - Neuroimaging
MH  - PubMed/statistics & numerical data
MH  - *Treatment Outcome
PMC - PMC5382178
OTO - NOTNLM
OT  - DSA
OT  - Embolisation
OT  - Microsurgery
OT  - Outcome
OT  - Radiosurgery
OT  - Ruptured AVMs
EDAT- 2017/03/23 06:00
MHDA- 2017/04/18 06:00
CRDT- 2017/03/22 06:00
PHST- 2016/12/16 [received]
PHST- 2017/03/10 [accepted]
AID - 10.1007/s00381-017-3383-4 [doi]
AID - 10.1007/s00381-017-3383-4 [pii]
PST - ppublish
SO  - Childs Nerv Syst. 2017 Apr;33(4):573-581. doi: 10.1007/s00381-017-3383-4. Epub
      2017 Mar 21.

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