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Abstract
Background With greater survival rates, patient-reported outcome measures (PROMs) among survivors of ruptured cerebral aneurysm should be an increasing concern among neurointerventionalists. Prior studies were limited in scale and generalizability. Our study aims were to (1) evaluate the validity of cerebral aneurysm PROMs obtained from social media; (2) determine the persistence of PROMs over time; and (3) determine what PROMs still exist in those with no physical impairments.
Methods By engaging national brain aneurysm support groups and using an online questionnaire modeled after the generic EQ-5D instrument, we asked respondents to classify their health in five dimensions including mobility, self-care, usual activities, pain/discomfort, and anxiety/depression using a 3-point Likert scale.
Results In 2 months we received 604 responses from 46 states in the USA. Our cohort of ruptured aneurysm respondents reported PROMs similar to previously published series. Over time, headache and anxiety improved while depression, level of exercise, and return to work remained unchanged. We found that memory worsened after 2 years. Among those without any physical impairment, rates of 20.6%, 14.9%, 12.6%, and 23% were seen for significant headaches, significant memory loss, significant depression, and sense of life being negatively affected, respectively.
Conclusions Despite this novel study design, we obtained results comparable to prior studies. These results suggest that many patients with ruptured cerebral aneurysms, regardless of whether they are >2 years after the event and/or free of physical impairment, struggle with a poor quality of life. The latency, scale, and low cost of this study design may accelerate future cerebral aneurysm PROM research.
- Aneurysm
- Subarachnoid
- Technology
- Statistics
Statistics from Altmetric.com
Background
Increasing numbers of patients with ruptured cerebral aneurysms are surviving, with case fatality rates falling by 17% in absolute terms over the past 40 years.1 Neurosurgeons and neurointerventionalists, who primarily manage the anatomic and surgical aspects of patient care, are often regarded by the patient as the primary authority for the entire spectrum of brain aneurysm management. Patients are often very much concerned with any persisting symptoms and their overall quality of life. Large-scale quality of life data may therefore be of use to physicians to better counsel and manage their patients.
Numerous reports have described the severity, characteristics, and persistence of impaired health-related quality of life measures in patients with ruptured cerebral aneurysms. Limitations to the currently available studies include their finite scale and generalizability to the general population.
The aim of this study is to evaluate the validity of patient-reported outcome measures (PROMs) of cerebral aneurysm patients obtained from social media, which comprises data from a large sample of the entire country. We designed our questions to address two commonly raised health-related quality of life concerns. The first concern was to determine exactly what types of physical, cognitive, and emotional problems exist among patients with ruptured aneurysms, to what degree, and their persistence over time. The second question was to determine what type of cognitive and/or emotional problems still exist in patients with ruptured aneurysms with no physical impairments.
Methods
Our questionnaire consisted of two parts. The first section, designed for validation, explicitly confirmed the diagnosis of an intracranial aneurysm. The second section allowed respondents to classify their own health in five dimensions (mobility, self-care, usual activities, pain/discomfort, and anxiety/depression) in three severity levels: no problem, mild problem, severe problem. This questionnaire was modeled after the generic EQ-5D instrument developed by the EuroQol Group, and has been widely used to measure the quality of life for the purpose of quality of life years (QALY) and is supported by considerable clinometric evidence.2 We designed the instrument to capture generic, disease-specific, and utility measures.Based on a literature review and one round of pretesting on 460 focus group patients, an iterative revised list of quality of life topics was designed and chosen for the final round of data collection. Question design was again modeled on the EQ-5D test and designed to be disease-specific to patients with cerebral aneurysm (see table 1 for specific variables chosen for the questionnaire). The general categories included basic demographic, diagnostic, treatment, clinical course, and quality of life measures. An online survey was built, designed to preserve anonymity, and had preselected responses to facilitate data analysis. Twenty-seven questions were developed and designed to take approximately 5–10 min to complete.
Independent and dependent variables used
Sample population
No patients were contacted through our clinical practice. We engaged foundations dedicated to supporting patients with brain aneurysms such as the Niekro Foundation (3211 Facebook likes in September 2014) and the Brain Aneurysm Foundation (11 889 Facebook likes in September 2014) to facilitate participation through the use of social network news feeds that had the greatest potential to reach cerebral aneurysm survivors from around the country and world. Links were posted on their respective Facebook timelines and peak survey responses occurred at the time of these posts. Respondents were not compensated.
Data analyses
To validate our results we compared our overall symptom profile among patients with ruptured aneurysm with those previously published in the literature. We addressed two commonly raised cerebral aneurysm PROM concerns that were appropriate for our selected population of brain aneurysm survivors. The first concern was to determine exactly what symptoms were present, to what degree, and their persistence over time. The second concern was to identify symptoms that impair quality of life among patients without physical impairments. Statistical comparisons were performed with a χ2 test; p values <0.05 were considered statistically significant.
Results
The questionnaire was published online using a web-based program in June 2014. Social media links to this questionnaire were posted in July 2014 and the responses were analyzed 1 month later. We received a total of 604 responses. Responses originated from 46 different states in the USA (figure 1), with 15% of respondents from outside the USA including Canada, Europe, and Australia. Eighty-nine per cent of respondents were women. Overall, the majority (38.7%) were in the age range 46–55 years. Most had their aneurysm diagnosed more than 2 years previously (62.7%). Among those who underwent treatment, nearly equal numbers underwent clip placement with open surgery (38.5%) and endovascular treatment (36.7%). Four hundred and thirty-two (71.5%) reported that their aneurysms had ruptured while 172 (28.5%) had unruptured aneurysms. One hundred and eighty-three (42%) reported more than one aneurysm.
US geographical distribution of respondents from 46 states.
Among the 432 ruptured aneurysm survivors, the overall PROM profile was mostly similar to previously published series (table 2). We selected five published studies using PROMs in ruptured cerebral aneurysm patients.3–7 Their sample sizes ranged from 64 to 253 subjects with tools consisting of either the SF-36 or EQ-5D. Using weighted averages and combining categories, we did not see significant differences in anxiety/depression (p=0.22) or return to full mobility (p=0.37). However, we did see differences in headache (p=0.02) and return to usual activities (p<0.0001).
When comparing respondents with ruptured aneurysm more than 2 years previously (n=282) with those within the last 2 years (n=150), the data suggested a statistically significant improvement in headache and anxiety over time (figure 2); 26.7% of respondents with ruptured aneurysms within the last 2 years were headache-free, which improved to 36.6% for those with the diagnosis for more than two years. Similarly, 20% of the group with ruptured aneurysms within the last 2 years were free of anxiety, which improved to 28.8% for those who had the diagnosis for more than 2 years (p=0.04). Our sample was heavily weighted towards women, even more so than the known gender disparity in aneurysm incidence. Despite this, no differences were seen when controlling for gender.
Comparison of ruptured cerebral aneurysm patient-reported outcome measures with previous publications
Profile and persistence of ruptured cerebral aneurysm patient-reported outcome measures.
The majority of symptoms, however, appeared to persist even after 2 years. No statistically significant differences were found with regard to level of exercise, ability to return to work, overall sense of a return to normal life, or depression. Memory impairment was remarkably common and actually appeared to worsen, with 82% affected within the first 2 years, increasing to 90.1% of patients after 2 years; this was statistically significant (p=0.03).
Although 59% (356/604) of the survey participants categorized themselves as free of physical impairments, many still described symptoms that negatively affected their quality of life (figure 3). Of these respondents, 20.6% complained of severe, poorly controlled headaches; 14.9% complained of significant memory problems; and 12.6% admitted to significant depression. Also, 23.9% of participants in this cohort responded that they did not exercise and 23% concluded that their life had been negatively affected.
Patient-reported outcome measures in functionally independent patients.
Discussion
Numerous studies, either patient-reported or surveyed responses, have consistently shown that the majority of patients who survive an aneurysmal subarachnoid hemorrhage struggle with persisting physical, cognitive, and emotional challenges. Most prominently, cognitive dysfunction has been shown to exist with high frequency, with memory being affected in up to 60% and executive function and language in as many as 75%.8 Mood disturbances have been reported in up to half of patients in the second year after aneurysmal subarachnoid hemorrhage.9
Previous reports measuring health-related quality of life among patients with non-traumatic subarachnoid hemorrhage found no significant difference after 1, 2, 5 and 10 years.4 This study used a disease-specific modification of the SF-36/SF-12 questionnaire. They evaluated physical functioning, bodily pain, general health, social functioning, and mental health. In our series, although headache and anxiety appeared to improve, depression, level of exercise and ability to return to work was unchanged while memory worsened. Similar to prior reports looking at the persistence of symptoms, our results suggest that the majority of symptoms, particularly cognitive functioning, are chronic among patients with ruptured cerebral aneurysms.
When evaluating the quality of life profile among patients who are functionally independent, we see a persistence of deficits ranging between 15% and 20% of patients. This is of particular importance because the modified Rankin scale (mRS), which captures physical functional capacity, has been shown to improve over time. A Finnish study showed that, 5 years after aneurysmal subarachnoid hemorrhage, mRS scores improved from the scores at 4 months and these continued to improve between 5 and 12.5 years.6 Our results showed that, despite no physical impairment, rates of 20.6%, 14.9%, 12.6% and 23% were seen for significant headaches, significant memory loss, significant depression, and sense of life being negatively affected, respectively. Therefore, even though physical functioning improved among survivors of subarachnoid hemorrhage, psychosocial issues appear to be more refractory. Quinn et al10 evaluated 212 ruptured cerebral aneurysm survivors and reported that almost a third of those with a mRS score of 0 had been unable to return to work and one in six were classified as having a borderline clinical mood disturbance when evaluated by the Wimbledon Self-Report Scale (WSRS). Physicians may better serve these patients with intact physical functioning by administering screening questions and making the appropriate referrals for additional help if needed.
The advantages of acquiring PROMs via social media and web-based questionnaires are obvious with regard to scale, low cost, and short latency in data acquisition. The primary disadvantages to data being collected in this fashion concern sampling bias and construct validity. One might think that support groups and online social networks may not capture data from patients who are older and not as familiar with technology, or those who are doing extremely well and may have little reason to participate. However, as social media improves its user interface, greater numbers of older users are being engaged. Patients who make very favorable recoveries may still be concerned about de novo aneurysms or the risk posed to their families and might also have reason to be participate.
With regard to construct validity, we were not able to independently confirm via history and/or imaging whether these patients did in fact have a brain aneurysm and whether or not the aneurysms did in fact rupture. Users of targeted social network groups are likely to be a self-selected motivated group of individuals. The likelihood that individuals without a diagnosis of brain aneurysm would be a part of this targeted social network and complete the questionnaire seems unlikely. We hope that the relatively larger size of the dataset relative to previously published single-center series can overcome the effect any erroneous outliers may have on biasing the data.
Privacy concerns are also relevant with data acquired in this fashion. Institutional review boards often hesitate on the interpretation of studies that analyze people’s behavior on social media websites because it is ethically unclear whether Facebook, Twitter or other types of postings count as public or private behavior, and therefore require their authors’ consent to be used in research. Data used for this study were explicity requested via the online questionnaire and not obtained indirectly via tracking users’ online activity.
We did try to minimize the degree of inaccurate responders by targeting social media sites that specifically supported brain aneurysm survivors, asking screening questions in the beginning of the study, not providing any compensation for the study, modeling questions based on the EQ-5D, and having a sufficiently large volume of responses.
Despite this novel method of data acquisition for PROMs, our results were comparable to previously published studies with confirmed diagnoses of ruptured brain aneurysms. Furthermore, our data were not restricted by geography, and included subjects from around the country and even outside the USA. These results in and of themselves may provide some reassurance that there may be a reasonable degree of construct validity in obtaining patient-reported data from targeted social media sites.
These results suggest that a significant number of patients with previously ruptured cerebral aneurysms, regardless of whether it is more than 2 years since their hemorrhagic event or they are functionally independent, continue to struggle with a poor quality of life. The notion that aneurysmal subarachnoid hemorrhage is a single event that can be cured by occlusion of the aneurysm is obsolete. They have the potential to regrow and new or de novo aneurysms may develop over time. The psychological trauma that results from a ruptured aneurysm has been demonstrated to contribute to chronic post-traumatic stress disorder (PTSD). Our results provide further evidence that neurosurgeons and neurointerventionalists should be sensitized to the symptoms of PTSD and refer patients to specialists so that PTSD may be diagnosed and treated as early as possible after subarachnoid hemorrhage.11 Patients who have experienced a ruptured aneurysm may benefit from survivorship care plans similar to those used for long-term cancer survivors. A survivorship care plan aims to inform the patient of long-term effects, to identify psychosocial resources in their community, and to provide guidance on follow-up care, prevention, and health maintenance.7 They call for further research on how best to provide follow-up and support for survivors of subarachnoid hemorrhage, including studies to explore patients’ expectations for recovery.
Conclusion
Impaired quality of life after a ruptured cerebral aneurysm remains a challenging problem for patients. Our understanding of the extent and causes of these symptoms are finite. Multiple mechanisms have been proposed as determinants of these cognitive deficits, including the mode of treatment, secondary ischemia or the initial effect of the aneurysmal subarachnoid hemorrhage.12 Hopefully these questions can be better addressed with the application of technology—with social media as an example—to shorten the latency for data acquisition and improve its relevance by obtaining data from a much broader geographic representation and accomplish this with negligible cost. However, there are concerns for sampling bias and construct validity with this methodology that should be addressed in future similar endeavors.
Acknowledgments
The authors acknowledge Kimberly Chapman and Natalie Niekro from the Joe Niekro Foundation and Christine Buckley from the Brain Aneurysm Foundation.
References
Footnotes
Contributors MC and EZM contributed to the design, data collection, analysis and drafting of the manuscript. BO contributed to the data analysis.
Competing interests None.
Patient consent Obtained.
Ethics approval Code of Federal Regulations (45 CFR 46.101 b) Exemption: Research involving the collection or study of existing data, documents, records, pathological specimens, or diagnostic specimens, if these sources are publicly available or if the information is recorded by the investigator in such a manner that subjects cannot be identified, directly or through identifiers linked to the subjects.
Provenance and peer review Not commissioned; externally peer reviewed.
Data sharing statement The authors agree to share any data on request.