Current Literature In Clinical Science
Long-Term Outcomes in Psychogenic Syncope: A Fallout for Neurologists
Long-Term Follow-Up of Psychogenic Pseudosyncope. Saal DP, Overdijk MJ, Thijs RD, van Vliet IM, van Dijk. Abstract Neurol 2016;87(21):2214–2219. Epub 2016 Oct 26.
OBJECTIVE: To determine the outcome of patients with psychogenic pseudosyncope (PPS) after communication of the diagnosis. METHODS: This was a retrospective cohort study of patients with PPS referred in 2007 to 2015 to a tertiary referral center for syncope. We reviewed patient records and studied attack frequency, factors affecting attack frequency, health care use, and quality of life using a questionnaire. We explored influences on attack freedom and attack frequency in the 6 months before follow-up for age, sex, education level, duration until diagnosis, probability of diagnosis, additional syncope, and acceptance of diagnosis. RESULTS: Forty-seven of 57 patients with PPS could be traced, of whom 35 (74%) participated. Twelve (34%) were attack-free for at least 6 months. The median time from diagnosis to follow-up was 50 months (range 6-103 months). Communicating and explaining the diagnosis resulted in immediate reduction of attack frequency (p = 0.007) from the month before diagnosis (median one attack, range 0-156) to the month after (median one attack, range 0-16). In the 6 months before follow-up, the number of admissions decreased from 19 of 35 to 0 of 35 (p = 0.002). The use of somatic and mental health care shifted toward the latter (p < 0.0001). Quality of life at follow-up (Short Form Health Survey 36) showed lower scores for 7 of 8 domains compared to matched Dutch control values; quality of life was not influenced by attack freedom. CONCLUSIONS: After communication of the diagnosis in PPS, attack frequency decreased and health care use shifted toward mental care. Low quality of life underlines that PPS is a serious condition.
Commentary Functional neurologic disorders (FNDs) are quite frequent in medical and neurologic practice and among the most common diagnoses referred to neurologists. On first glance, it might seem puzzling as to why there is a relatively small literature on follow-up for these patients. On closer look, however, patients are sent away from neurologists to mental health and other specialists, often with no clinician taking full responsibility for following their neurologic symptoms. The lack of follow-up with neurology may carry the implication that there is no real disease for neurologists to focus on. Adherence with psychiatric follow-up is poor, and patients continue to seek medical opinions not understanding or believing their diagnosis. Nevertheless, there are some data on outcomes for psychogenic nonepileptic seizures (PNESs), the most common of the FNDs. The results are disappointing in that only about one third of patients stop having seizures, and even most of those who are better do not return to work (1), suggesting a complex relationship with the underlying psychiatric comorbidities. Studies are limited and biased as they derive from heterogeneous, retrospective studies in patients referred to Epilepsy Currents, Vol. 17, No. 3 (May/June) 2017 pp. 163–164 © American Epilepsy Society
tertiary epilepsy centers. Those who decide on follow-up are likely different from those who do not. Short-term outcome (immediately upon communicating the diagnosis) is better, but there is rapid falloff over months (2). While communicating the diagnosis of PNES may halt seizures early on for a minority of patients (3), the majority continue to recur or develop other functional symptoms even though they decrease their health care utilization (4). Long-term outcome studies are far fewer and have variable results, though one large study showed sustained reduction of anticonvulsant use but poor return to employment and poor psychiatric outcomes (5). Better understanding of long-term outcomes for PNESs and other FNDs will help to inform treatment for these complex disorders. Psychogenic syncope (or psychogenic pseudosyncope, PPS), a less common and much less studied cause of loss of consciousness, has similarities and overlap with PNESs and may be more likely be missed as it often presents to cardiologists or internists who are less likely to refer to psychiatrists even when the etiology is unexplained (6). Until recently, there have been no studies on outcomes in PPS, in part because of the extreme difficulty in gathering a reliable case series. Saal and colleagues (7) were able to gather retrospective data on long-term outcomes for PPS, identifying 57 patients followed over a period of 50 months to 8 years. They required positive features for diagnosis: eyes closed during the attack, high frequency of spells, long duration, and an episode captured
163
Long-Term Outcomes in Psychogenic Syncope
on tilt testing with continual blood pressure measurements to be considered definite PPS; otherwise they were considered to have probable PPS. They enrolled 35 patients with PPS (33 definite, 2 probable), of whom 57% also had comorbid syncope, a higher percentage than the 10–25% of comorbid epilepsy reported in PNES studies. However, similar to PNES, nearly 70% of participant were women who had a mean age of 36 years at diagnosis and had low quality of life. A clear explanation of the psychological nature of the diagnosis reduced PPS immediately. Whether patients accepted the psychological nature of the disorder did not seem to affect their outcome. Also, at delayed follow-up, one third of participants were still without episodes of PPS. This study is the first to gather a series of patients with PPS, and their methods are sound. The size of this series is also good. However, there are some limitations to this study. First, similar to other outcomes studies for FNDs, this study is a retrospective and biased sample obtained through referrals to a tertiary center. Second, there is no control group in the study to compare PPS to other FNDs or syncope outcomes. Instead, subjects were compared with normative data on quality-oflife measures. In addition, this study did not utilize a standard treatment. Most importantly though, there are likely a large number of patients with PPS who remain undiagnosed and therefore are not directed to mental health treatment. Sending patients to subspecialty clinicians based on different somatic symptoms and different practice styles is problematic for patients with FNDs, including PPS, and can readily lead to disintegration of their care. Providers have variable levels of comfort with these patients, and access to knowledgeable and caring providers is inconsistent and may interfere with outcomes. This has not been studied. Many patients are prescribed unhelpful, expensive, and potentially dangerous medications over years and lack continuity of care as they reverberate through the health system circuit without a central clinician. If we begin to insist that one clinician
164
take responsibility as leader of the team of care providers for communicating clearly, treating these patients with dignity and respect, and tracking what happens to them, we are more likely to measure the true results of our interventions and find out what works for patients. We will not be successful if we do not stop the fallout with FNDs. by Barbara A. Dworetzky, MD References 1. Arain AM, Hamadani AM, Islam, S, Abou-Khalil BW. Predictors of early seizure remission after diagnosis of psychogenic nonepileptic seizures. Epilepsy Behav, 2007:11:409–412. 2. Duncan R, Razvi S, Mulhern S. Newly presenting psychogenic nonepileptic seizures: Incidence, population characteristics, and early outcome from a prospective audit of a first seizure clinic. Epilepsy Behav. 2011;20:308–311. 3. Mayor R, Brown RJ, Cock H, House A, Howlett S, Singhal S, Smith P, Reuber M. Short-term outcome of psychogenic non-epileptic seizures after communication of the diagnosis. Epilepsy Behav. 2012 Dec;25(4):676–681. 4. McKenzie P, Oto M, Russell A, Pelosi A, Duncan R. Early outcomes and predictors in 260 patients with psychogenic nonepileptic attacks. Neurology. 2010 Jan 5;74(1):64–69. 5. Duncan R, Graham CD, Oto M, Russell A, McKernan L, Copstick S. Primary and secondary care attendance, anticonvulsant and antidepressant use and psychiatric contact 5-10 years after diagnosis in 188 patients with psychogenic non-epileptic seizures. J Neurol Neurosurg Psychiatry. 2014;85:954–958. 6. Heyer GL, Albert DV, Weber A, Gedela S, Vidaurre J. Comparison of semiologies between tilt-induced psychogenic nonsyncopal collapse and psychogenic nonepileptic seizures Epilepsy Behav. 2016;62:171–175. 7. Saal DP, Overdijk MJ, Thijs RD, van Vliet IM, van Dijk JG. Long-term follow-up of psychogenic pseudosyncope. Neurology. 2016;87:2214– 2219.
Long-Term Outcomes in Psychogenic Syncope: A Fallout for Neurologists
Long-Term Follow-Up of Psychogenic Pseudosyncope. Saal DP, Overdijk MJ, Thijs RD, van Vliet IM, van Dijk. Abstract Neurol 2016;87(21):2214–2219. Epub 2016 Oct 26.
OBJECTIVE: To determine the outcome of patients with psychogenic pseudosyncope (PPS) after communication of the diagnosis. METHODS: This was a retrospective cohort study of patients with PPS referred in 2007 to 2015 to a tertiary referral center for syncope. We reviewed patient records and studied attack frequency, factors affecting attack frequency, health care use, and quality of life using a questionnaire. We explored influences on attack freedom and attack frequency in the 6 months before follow-up for age, sex, education level, duration until diagnosis, probability of diagnosis, additional syncope, and acceptance of diagnosis. RESULTS: Forty-seven of 57 patients with PPS could be traced, of whom 35 (74%) participated. Twelve (34%) were attack-free for at least 6 months. The median time from diagnosis to follow-up was 50 months (range 6-103 months). Communicating and explaining the diagnosis resulted in immediate reduction of attack frequency (p = 0.007) from the month before diagnosis (median one attack, range 0-156) to the month after (median one attack, range 0-16). In the 6 months before follow-up, the number of admissions decreased from 19 of 35 to 0 of 35 (p = 0.002). The use of somatic and mental health care shifted toward the latter (p < 0.0001). Quality of life at follow-up (Short Form Health Survey 36) showed lower scores for 7 of 8 domains compared to matched Dutch control values; quality of life was not influenced by attack freedom. CONCLUSIONS: After communication of the diagnosis in PPS, attack frequency decreased and health care use shifted toward mental care. Low quality of life underlines that PPS is a serious condition.
Commentary Functional neurologic disorders (FNDs) are quite frequent in medical and neurologic practice and among the most common diagnoses referred to neurologists. On first glance, it might seem puzzling as to why there is a relatively small literature on follow-up for these patients. On closer look, however, patients are sent away from neurologists to mental health and other specialists, often with no clinician taking full responsibility for following their neurologic symptoms. The lack of follow-up with neurology may carry the implication that there is no real disease for neurologists to focus on. Adherence with psychiatric follow-up is poor, and patients continue to seek medical opinions not understanding or believing their diagnosis. Nevertheless, there are some data on outcomes for psychogenic nonepileptic seizures (PNESs), the most common of the FNDs. The results are disappointing in that only about one third of patients stop having seizures, and even most of those who are better do not return to work (1), suggesting a complex relationship with the underlying psychiatric comorbidities. Studies are limited and biased as they derive from heterogeneous, retrospective studies in patients referred to Epilepsy Currents, Vol. 17, No. 3 (May/June) 2017 pp. 163–164 © American Epilepsy Society
tertiary epilepsy centers. Those who decide on follow-up are likely different from those who do not. Short-term outcome (immediately upon communicating the diagnosis) is better, but there is rapid falloff over months (2). While communicating the diagnosis of PNES may halt seizures early on for a minority of patients (3), the majority continue to recur or develop other functional symptoms even though they decrease their health care utilization (4). Long-term outcome studies are far fewer and have variable results, though one large study showed sustained reduction of anticonvulsant use but poor return to employment and poor psychiatric outcomes (5). Better understanding of long-term outcomes for PNESs and other FNDs will help to inform treatment for these complex disorders. Psychogenic syncope (or psychogenic pseudosyncope, PPS), a less common and much less studied cause of loss of consciousness, has similarities and overlap with PNESs and may be more likely be missed as it often presents to cardiologists or internists who are less likely to refer to psychiatrists even when the etiology is unexplained (6). Until recently, there have been no studies on outcomes in PPS, in part because of the extreme difficulty in gathering a reliable case series. Saal and colleagues (7) were able to gather retrospective data on long-term outcomes for PPS, identifying 57 patients followed over a period of 50 months to 8 years. They required positive features for diagnosis: eyes closed during the attack, high frequency of spells, long duration, and an episode captured
163
Long-Term Outcomes in Psychogenic Syncope
on tilt testing with continual blood pressure measurements to be considered definite PPS; otherwise they were considered to have probable PPS. They enrolled 35 patients with PPS (33 definite, 2 probable), of whom 57% also had comorbid syncope, a higher percentage than the 10–25% of comorbid epilepsy reported in PNES studies. However, similar to PNES, nearly 70% of participant were women who had a mean age of 36 years at diagnosis and had low quality of life. A clear explanation of the psychological nature of the diagnosis reduced PPS immediately. Whether patients accepted the psychological nature of the disorder did not seem to affect their outcome. Also, at delayed follow-up, one third of participants were still without episodes of PPS. This study is the first to gather a series of patients with PPS, and their methods are sound. The size of this series is also good. However, there are some limitations to this study. First, similar to other outcomes studies for FNDs, this study is a retrospective and biased sample obtained through referrals to a tertiary center. Second, there is no control group in the study to compare PPS to other FNDs or syncope outcomes. Instead, subjects were compared with normative data on quality-oflife measures. In addition, this study did not utilize a standard treatment. Most importantly though, there are likely a large number of patients with PPS who remain undiagnosed and therefore are not directed to mental health treatment. Sending patients to subspecialty clinicians based on different somatic symptoms and different practice styles is problematic for patients with FNDs, including PPS, and can readily lead to disintegration of their care. Providers have variable levels of comfort with these patients, and access to knowledgeable and caring providers is inconsistent and may interfere with outcomes. This has not been studied. Many patients are prescribed unhelpful, expensive, and potentially dangerous medications over years and lack continuity of care as they reverberate through the health system circuit without a central clinician. If we begin to insist that one clinician
164
take responsibility as leader of the team of care providers for communicating clearly, treating these patients with dignity and respect, and tracking what happens to them, we are more likely to measure the true results of our interventions and find out what works for patients. We will not be successful if we do not stop the fallout with FNDs. by Barbara A. Dworetzky, MD References 1. Arain AM, Hamadani AM, Islam, S, Abou-Khalil BW. Predictors of early seizure remission after diagnosis of psychogenic nonepileptic seizures. Epilepsy Behav, 2007:11:409–412. 2. Duncan R, Razvi S, Mulhern S. Newly presenting psychogenic nonepileptic seizures: Incidence, population characteristics, and early outcome from a prospective audit of a first seizure clinic. Epilepsy Behav. 2011;20:308–311. 3. Mayor R, Brown RJ, Cock H, House A, Howlett S, Singhal S, Smith P, Reuber M. Short-term outcome of psychogenic non-epileptic seizures after communication of the diagnosis. Epilepsy Behav. 2012 Dec;25(4):676–681. 4. McKenzie P, Oto M, Russell A, Pelosi A, Duncan R. Early outcomes and predictors in 260 patients with psychogenic nonepileptic attacks. Neurology. 2010 Jan 5;74(1):64–69. 5. Duncan R, Graham CD, Oto M, Russell A, McKernan L, Copstick S. Primary and secondary care attendance, anticonvulsant and antidepressant use and psychiatric contact 5-10 years after diagnosis in 188 patients with psychogenic non-epileptic seizures. J Neurol Neurosurg Psychiatry. 2014;85:954–958. 6. Heyer GL, Albert DV, Weber A, Gedela S, Vidaurre J. Comparison of semiologies between tilt-induced psychogenic nonsyncopal collapse and psychogenic nonepileptic seizures Epilepsy Behav. 2016;62:171–175. 7. Saal DP, Overdijk MJ, Thijs RD, van Vliet IM, van Dijk JG. Long-term follow-up of psychogenic pseudosyncope. Neurology. 2016;87:2214– 2219.
