Letter to the Editor: Quality of life of patients affected by unruptured brain AVMs

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TO THE EDITOR: We read with great interest the study by Bervini et al.1 (Bervini D, Morgan MK, Ritson EA, et al: Surgery for unruptured arteriovenous malformations of the brain is better than conservative management for selected cases: a prospective cohort study. J Neurosurg 121:878–890, October 2014). In their study thef authors showed surgical treatment of Spetzler-Ponce Class A AVMs is superior to no treatment. We think this is a key paper for all surgeons involved in this fascinating field. Indeed, it can definitively clarify the role of surgery in the management of unruptured AVMs, “providing robust support for recommending treatment.”3 In addition, both the large size of the patient group and the rigorous method of data analysis made the study an objective reply to the results of ARUBA (A Randomized Trial of Unruptured Brain Arteriovenous Malformations).8 After this response, we believe that the field is ready for new questions. Unruptured brain AVMs are frequently symptomatic and sometimes disabling. Indeed, the quality of life (QOL) of a patient with an unruptured AVM can be limited by different factors, such as seizures, the need for antiepileptic medication, and neurological defects, which can have a substantial effect on daily activities, life plans, work ability, social life, and other aspects of the patient's life. The risk of bleeding, and the risks associated with AVMs in general, can be perceived in different ways by patients with these lesions, sometimes influencing the treatment decision. Bervini et al. reported that seizures and neurological deficits were present in 47.9% and 12.1% of patients with Spetzler-Ponce Class A, and in 57.9% and 17.8% of those with Spetzler-Ponce Class B unruptured AVMs, respectively. We are all aware that surgery can play a crucial role in treating these conditions and thus improving quality of life. For example, we know from literature that surgery can effectively eliminate seizures in AVM patients.4,5 QOL assessment is a widely used measurement of outcome of clinical trials and is increasingly recognized as a major end point for Phase III randomized controlled trials.2 The attention to QOL is a well established in neurooncological patient treatment,7 and it represents an emerging issue for patients undergoing cerebrovascular surgery.9 For example, some authors have shown that the preoperative QOL of patients with unruptured aneurysm is lower than for the normal population, and that surgery improves the QOL of such patients.6,10,11 We think that the next step, now, is to focus our attention on how we can improve the QOL of patients with unruptured brain AVMs. This new perspective might open a new and interesting scenario for treatment in general, and surgery, in particular, for these patients. In our department we recently started QOL assessment of AVM patients undergoing surgery, and we frequently registered both a lower preoperative score compared to normal population scores and an improvement of QOL after surgery. We wanted to share our observations with the neurosurgical community involved in this fascinating field and suggest some questions that have arisen as a result of our reading the paper by Bervini et al. Could QOL assessment be a new, modern, more appropriate way to compare different treatment options of patients affected by brain AVMs? Could surgery improve QOL of patients affected by Spetzler-Ponce Class B AVMs more than no treatment? Should we make the QOL of AVM patients central in our decision-making process? We thank Professor Morgan and coworkers for their remarkable work providing us data showing that surgery, in expert hands, is able not only to cure patients but probably also to improve the quality of their lives. We keep investigating.

References

  • 1

    Bervini DMorgan MKRitson EAHeller G: Surgery for unruptured arteriovenous malformations of the brain is better than conservative management for selected cases: a prospective cohort study. J Neurosurg 121:8788902014

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    • Export Citation
  • 2

    Editorial. Quality of life and clinical trials. Lancet 346:121995

  • 3

    Elhammady MSHeros RC: Editorial. Surgical management of unruptured cerebral arteriovenous malformations. J Neurosurg 121:8758772014

    • Search Google Scholar
    • Export Citation
  • 4

    Englot DJYoung WLHan SJMcCulloch CEChang EFLawton MT: Seizure predictors and control after microsurgical resection of supratentorial arteriovenous malformations in 440 patients. Neurosurgery 71:5725802012

    • Search Google Scholar
    • Export Citation
  • 5

    Hoh BLChapman PHLoeffler JSCarter BSOgilvy CS: Results of multimodality treatment for 141 patients with brain arteriovenous malformations and seizures: factors associated with seizure incidence and seizure outcomes. Neurosurgery 51:3033112002

    • Search Google Scholar
    • Export Citation
  • 6

    King JT JrHorowitz MBKassam ABYonas HRoberts MS: The short form-12 and the measurement of health status in patients with cerebral aneurysms: performance, validity, and reliability. J Neurosurg 102:4894942005

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    • Export Citation
  • 7

    Klein EAltshuler DHallock ASzerlip N: Quality of life research in neuro-oncology: a quantitative comparison. J Neurooncol 116:3333402014

    • Search Google Scholar
    • Export Citation
  • 8

    Mohr JPParides MKStapf CMoquete EMoy CSOverbey JR: Medical management with or without interventional therapy for unruptured brain arteriovenous malformations (ARUBA): a multicentre, non-blinded, randomised trial. Lancet 383:6146212014

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    • Export Citation
  • 9

    Sanchez CEOgilvy CSCarter BS: Outcomes studies in cerebrovascular neurosurgery. Neurosurg Focus 22:3E112007

  • 10

    Yamashiro SNishi TKoga KGoto TKaji MMuta D: Improvement of quality of life in patients surgically treated for asymptomatic unruptured intracranial aneurysms. J Neurol Neurosurg Psychiatry 78:4975002007

    • Search Google Scholar
    • Export Citation
  • 11

    Yamashiro SNishi TKoga KGoto TMuta DKuratsu J: Postoperative quality of life of patients treated for asymptomatic unruptured intracranial aneurysms. J Neurosurg 107:108610912007

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    • Export Citation
Keywords:

Response

We believe that Drs. Della Puppa, Rustemi, and Scienza have made an excellent point regarding the importance of studying brain AVM treatment from the point of view of the patient with regard to QOL. Developing instruments to measure this (e.g., the SF-36 [36-Item Short-Form Health Survey]) is important and commendable. There is no doubt that with time and progress in surgery, the outcome measures employed have evolved to increase our understanding of outcomes from “good,” “bad,” and “dead” to a greater outcome range used today. The future will see a shift to more sophisticated outcome measure scores. Hopefully, this will be matched by innovations and improvement in surgical care. We do measure SF-36 and neuropsychological outcomes at our institutions for our more recent patients to improve our understanding of the impact of management decisions (e.g., neuropsychological outcomes1). We are fortunate to have dedicated personnel that can be devoted to this task.

However, moving to more personalized and complex evaluation tools has significant challenges. The more we move away from the binary outcome system of alive or dead, which can be easily assessed and validated by many, the greater the challenges for accuracy and compliance. Patient-centered QOL instruments (e.g., SF-36) can be very difficult tools to apply with 100% compliance because of the greater commitment to time and effort required of both the patient and the treating team, the uncertainty as to the way individuals may assess their QOL from time to time, and translating the interpretation of these QOL outcomes to the informed consent process for future patients. As an example, on evaluating neuropsychological outcomes with dedicated staff for this purpose, we were successful in recruiting fewer than 60% of eligible patients.1

In addition to QOL, there is also the interpretation of what an individual is willing to trade. For example, parents may consider surgery worthwhile if it will result in a slight hemiparesis or homonymous hemianopia of their child presenting with a hemorrhage from a Spetzler-Ponce Class C brain AVM in order to reduce the chance of their child's dying before they do, whereas many adults will not accept this outcome. Furthermore, the perceived QOL may not be fixed over time. Someone believing that they have a good QOL at one point in time may not have a fixed view of it at another. Therefore, judging QOL is a difficult measure to make and interpret and results in an extraordinarily difficult measure to apply to guide future management.

It would be a mistake to assume that the simple-to-apply modified Rankin Scale (mRS) reflects fully patients' perception of their QOL. However, we believe that the mRS is useful as a measure that allows rough benchmarking between units for each Spetzler-Ponce class or Spetzler-Martin grade. Having said this, we did find correlation between mRS outcome scores and more detailed outcome evaluation, reassuring us that the mRS remains useful.1

Drs. Della Puppa, Rustemi, and Scienza point out the importance of seizure management with regard to the impact upon QOL. Our preliminary results for Kaplan-Meier analysis of first seizure following surgery for supratentorial brain AVMs (both ruptured and unruptured) is given in Fig. 1, with the number at risk reported in Table 1. These results suggest that in our hands, the potential to “effectively cure seizures” (as commented upon by Drs. Della Puppa, Rustemi, and Scienza) is uncertain and less dramatic than previously reported.

FIG. 1.
FIG. 1.

Kaplan-Meier analysis of the first seizure following surgery for supratentorial brain AVM. SPC = Spetzler-Ponce Class.

TABLE 1

Number at risk for Fig. 1

DescriptionYear
012345678910111213141516
No. at risk: supratentorial SPC A317201129846548352923191813106531
No. at risk: supratentorial SPC B & C, no preop seizures14285543728252115973221111
No. at risk: supratentorial SPC B & C, preop seizure1025536241915119775442000
Total56134121914511288675339332619169642
SPC = Spetzler-Ponce Class.

We welcome Drs. Della Puppa, Rustemi, and Scienza's plans to encourage the application of more sophisticated outcome measures. The more lenses that we can apply, the more that we will understand. We are sure that the experience of surgery changes patients, and measuring these changes is important. We also need to understand how to interpret and apply the outcomes measured and translate these for intended patients.

Reference

1

Marshall GAJonker BPMorgan MKTaylor AJ: Prospective study of neuropsychological and psychosocial outcome following surgical excision of intracerebral arteriovenous malformations. J Clin Neurosci 10:42472003

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Article Information

Contributor Notes

INCLUDE WHEN CITING Published online February 13, 2015; DOI: 10.3171/2014.9.JNS141969.DISCLOSURE The authors report no conflict of interests.

© AANS, except where prohibited by US copyright law.

Headings
Figures
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    Kaplan-Meier analysis of the first seizure following surgery for supratentorial brain AVM. SPC = Spetzler-Ponce Class.

References
  • 1

    Bervini DMorgan MKRitson EAHeller G: Surgery for unruptured arteriovenous malformations of the brain is better than conservative management for selected cases: a prospective cohort study. J Neurosurg 121:8788902014

    • Search Google Scholar
    • Export Citation
  • 2

    Editorial. Quality of life and clinical trials. Lancet 346:121995

  • 3

    Elhammady MSHeros RC: Editorial. Surgical management of unruptured cerebral arteriovenous malformations. J Neurosurg 121:8758772014

    • Search Google Scholar
    • Export Citation
  • 4

    Englot DJYoung WLHan SJMcCulloch CEChang EFLawton MT: Seizure predictors and control after microsurgical resection of supratentorial arteriovenous malformations in 440 patients. Neurosurgery 71:5725802012

    • Search Google Scholar
    • Export Citation
  • 5

    Hoh BLChapman PHLoeffler JSCarter BSOgilvy CS: Results of multimodality treatment for 141 patients with brain arteriovenous malformations and seizures: factors associated with seizure incidence and seizure outcomes. Neurosurgery 51:3033112002

    • Search Google Scholar
    • Export Citation
  • 6

    King JT JrHorowitz MBKassam ABYonas HRoberts MS: The short form-12 and the measurement of health status in patients with cerebral aneurysms: performance, validity, and reliability. J Neurosurg 102:4894942005

    • Search Google Scholar
    • Export Citation
  • 7

    Klein EAltshuler DHallock ASzerlip N: Quality of life research in neuro-oncology: a quantitative comparison. J Neurooncol 116:3333402014

    • Search Google Scholar
    • Export Citation
  • 8

    Mohr JPParides MKStapf CMoquete EMoy CSOverbey JR: Medical management with or without interventional therapy for unruptured brain arteriovenous malformations (ARUBA): a multicentre, non-blinded, randomised trial. Lancet 383:6146212014

    • Search Google Scholar
    • Export Citation
  • 9

    Sanchez CEOgilvy CSCarter BS: Outcomes studies in cerebrovascular neurosurgery. Neurosurg Focus 22:3E112007

  • 10

    Yamashiro SNishi TKoga KGoto TKaji MMuta D: Improvement of quality of life in patients surgically treated for asymptomatic unruptured intracranial aneurysms. J Neurol Neurosurg Psychiatry 78:4975002007

    • Search Google Scholar
    • Export Citation
  • 11

    Yamashiro SNishi TKoga KGoto TMuta DKuratsu J: Postoperative quality of life of patients treated for asymptomatic unruptured intracranial aneurysms. J Neurosurg 107:108610912007

    • Search Google Scholar
    • Export Citation
  • 1

    Marshall GAJonker BPMorgan MKTaylor AJ: Prospective study of neuropsychological and psychosocial outcome following surgical excision of intracerebral arteriovenous malformations. J Clin Neurosci 10:42472003

    • Search Google Scholar
    • Export Citation
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