- Email: [email protected]
Provocative techniques should not be used for the diagnosis of psychogenic nonepileptic seizures
Epilepsy & Behavior 15 (2009) 110–114
Contents lists available at ScienceDirect
Epilepsy & Behavior journal homepage: www.elsevier.com/locate/yebeh
Controversies in Epilepsy and Behavior
Provocative techniques should not be used for the diagnosis of psychogenic nonepileptic seizures Beth A. Leeman * Departments of Neurology, Beth Israel Deaconess Medical Center, Massachusetts General Hospital, Harvard Medical School, Boston, MA, USA Behavioral Neurology Unit, Beth Israel Deaconess Medical Center, 330 Brookline Avenue, KS-2, Boston, MA 02215, USA
Patients with psychogenic nonepileptic seizures (NES) have episodes of abnormal movements, alterations of awareness and unresponsiveness that appear similar to epileptic seizures. Unlike epileptic seizures, however, these spells occur in the absence of concurrent epileptiform activity in the brain. Such episodes are common, occurring in approximately 20% of those presenting to academic epilepsy centers for evaluation [1]. Although there is no gold standard process for diagnosis, the most common approach involves capturing the spell during electroencephalography (EEG), to demonstrate the lack of corresponding epileptiform discharges. Often patients with NES will not have spells while undergoing EEG monitoring, however, which creates difficulty in establishing a diagnosis and often leads to the use of induction procedures. Historically, the neurologists’ approach to the diagnosis of NES or other forms of conversion disorder has been controversial. In the 19th century, Charcot developed criteria for the differentiation of ‘‘hysterical attacks” from epilepsy, which included the precipitation or termination of spells by pressing on the ovaries or other ‘‘hysterogenic” areas [2]. A review of his works revealed that he was ‘‘not above slight trickery” or suggestion, as when he spoke offensively to a patient with psychogenic hearing loss to elicit a response [3]. Others in this time period were reported to use more radical methods. Gowers’ approach included applying electric shocks, covering the face with a towel to the point of asphyxiation, pouring water in the mouth, inducing vomiting with apomorphine, and pulling on pubic hair to abort spells and establish a diagnosis [4]. Perhaps the most colorful story from this era is that of Mitchell’s consultation on a puzzling case of paralysis. After examining the patient, he requested that the other doctors step into the hallway. When he joined them a moment later, they asked if the patient would ever walk again. He responded ‘‘yes, in a moment,” at which time the patient ran out of the room. When asked what happened, Dr. Mitchell replied, ‘‘I set the bedclothes on fire” [5]. In the mid- to late-20th century, diagnostic methods for conversion disorder may have been more ‘‘humane,” but nevertheless deceptive. Many physicians advocated the use of placebos for the diagnosis of conversion disorder in its various forms. Levy and Jankovic [6] reported a particularly striking case in which they documented a ‘‘dose dependent” reaction to a placebo injection, resulting in psychogenic nonfluent aphasia, NES, and profound * Fax: +1 617 667 7981. E-mail address: [email protected]. 1525-5050/$ - see front matter Ó 2009 Elsevier Inc. All rights reserved. doi:10.1016/j.yebeh.2009.01.013
unresponsiveness, in evaluation of a psychogenic reaction to phenytoin. Monday and Jankovic [7] reported a series of patients with psychogenic myoclonus whose symptoms initially worsened with saline injections given to ‘‘enhance the abnormal movements” and later improved with additional saline injections given to ‘‘ameliorate” the movements. They encouraged the use of ‘‘positive criteria,” such as response to placebo, when considering the diagnosis of a psychogenic movement disorder. Similarly, Fahn and Williams [8] proposed a classification system to reflect certainty of diagnosis with respect to psychogenic dystonia. Criteria for ‘‘documented psychogenic dystonia” included the response to placebo. Multiple successful trials of placebo treatment were taken as the ‘‘best evidence for documenting [the] diagnosis.” The repeated response to placebo trials has been advanced as a diagnostic method by other authors as well, with Platt [9] providing the example of its use in psychogenic headache. Although much of this literature dates pre-1995, if social psychology research may serve as a guide, publications from 1969 to 1992 did not demonstrate any large, sustained shifts in the use of deceptive practices to indicate that ideas would have changed in recent years [10]. Currently, an estimated 39–73% of epileptologists have adopted the practice of inducing spells during EEG recording, typically by the administration of placebo agents such as saline injections, alcohol pads applied transdermally, tuning forks placed on the forehead, and head tilting maneuvers [11,12]. These methods commonly involve misleading the patient to believe that an active, proconvulsant agent is being administered and that an ‘‘antidote” (also a placebo) would be provided if a spell is prolonged or severe. These methods may involve omissions of the truth or direct lies. Burack et al. [13], for example, describe an induction procedure in patient RB: RB had EEG electrodes and a blood pressure cuff attached and an intravenous catheter placed. A resident physician. . .asked RB’s consent to administer intravenously ‘‘an electrolyte solution that may bring on spells”. . .. The solution, unbeknownst to RB, was normal saline. RB was asked to pump his arm vigorously while an infusion was begun, to help the ‘‘infusion serum” circulate. A sense of drama and expectancy pervaded the procedure: the physician’s voice and manner were tense, and the patient was perspiring and appeared anxious. The physician narrated the patient’s movements and responses, and noted a point at which the infusion reached ‘‘full therapeutic levels.” RB then had a typical seizure. . .. The physician stated loudly
B.A. Leeman / Epilepsy & Behavior 15 (2009) 110–114
that serum levels should be back to zero; seconds later, the muscular jerking tapered off. The patients may or may not be debriefed after the procedure, although patients who are not told of the deception could easily discover the truth later via other means (e.g., medical records or other health professionals involved in their care). Proponents of this approach argue that use of deception in induction procedures is justified, based primarily on claims that such procedures will clarify the diagnosis in a safe and efficient manner, thereby sparing the patient from the harm of an erroneous diagnosis of epilepsy. Most recently, Ribaï et al. [14] took an extreme stance, concluding that the investigation of NES should ‘‘always include” a provocative saline test. Although little has been published on this issue, the majority of the authors have conceded that use of deception may be justifiable in certain limited situations, but that the ethical issues must be carefully considered [13,15–18]. The reader should question, however, whether establishing a diagnosis should ever outweigh the violations of autonomy and nonmaleficence caused by deception. 1. Are there benefits? Some believe that documenting a response to placebo will more definitively establish the diagnosis of NES. Confirming a diagnosis is important, as patients may then be directed toward appropriate treatments, such as cognitive-behavioral therapies and antidepressants for comorbid depression and anxiety. In addition, it is hoped that patients with NES would avoid the social and financial hardships that result from a diagnosis of epilepsy. They would also be spared potentially harmful treatments for epileptic seizures, as anticonvulsant medications would be ineffective for treatment of NES. Antiepileptic medications are often costly and have potential side effects. These side effects can be life-threatening, such as aplastic anemia and Stevens–Johnson syndrome. Patients presenting with nonepileptic status could also avoid the excessive sedative medications, intubations, and ICU admissions that occur with true status epilepticus. To hasten the diagnosis by induction may also prevent long, costly hospital stays and the concomitant risks of nosocomial infections. Those adopting a paternalistic approach would state that beneficence in these regards would supersede the need to respect patient autonomy. 2. Are there harms? Supporters of induction may claim that the diagnosis is made with little risk, assuming that those employing placebos with malintent (i.e., to anger an objectionable patient) are removed from the equation. In their view, the duty of nonmaleficence, to treat the patient in the least harmful manner, is upheld. Saline is an inert substance, injected through an intravenous line that is already placed for clinical purposes. Alcohol pads are not irritating to the skin. Although people may sustain injuries during induced spells, this risk is no different than that incurred during spontaneous spells captured by routine video/EEG monitoring. Hence, proponents of deceptive induction procedures would argue that these methods act in the patients’ best interests and with little potential for harm. The counterargument, however, is that the principle of nonmaleficence has indeed been violated, in that deceptive methods actually cause more psychological harm than good. To deceive a patient risks irreparable damage to the physician– patient relationship. The patient may have distrust of that physician, which may extend to other physicians or the medical system as a whole. This distrust could potentially compromise adherence to the treatment plan for NES. This places the patient at risk for all of the consequences of untreated NES and depression, including
111
injury or even suicide. The patient may now be less willing to seek care for other medical issues, as well. Establishing the diagnosis of NES in this manner could also affect the public’s view of the medical system more broadly. Patients may already be suspicious of the diagnosis of NES, and providing further reason for mistrust would be ill-advised. Furthermore, deceptive practices in the diagnosis of NES prey upon a particularly vulnerable patient population. These patients are often depressed and anxious, and may have personality or other psychiatric disorders. Histories of physical or sexual abuse, unstable home environments, and other traumatic life events are common. Hence, patients may already have difficulty in trusting others. Dishonesty from a physician, who has pledged to deliver honest, compassionate care, may be even more destructive to the relationships of these patients, with physicians or others, than anticipated. Stagno and Smith [18] take this argument a step further and assert that for patients with a history of sexual abuse, ‘‘the insertion of a needle and infusion of a fluid (in the case of IV inductions) may be a psychologic symbol of the abuse.” Some authors advocate psychiatric screening for personality traits that may make a patient susceptible to psychological harm, to be performed prior to the decision to use deceptive induction. Although an estimated 43% of centers using induction obtain a psychiatric screen prior to the procedure [12], it is not clear that such screens are adequately predictive. While debriefing may explain the rationale for deception, and provide potentially persuasive arguments for its use, it cannot be known how a given patient, particularly one who is psychiatrically ill, will react. Burack et al. [13] noted that harms may affect the physician as well, as (s) he may be ‘‘burdened by the unforeseeable and cumulative strains and risk of the further deceptions required to support the initial one.” This may be especially true for those who use additional deception after induction, telling patients that the diagnosis of NES was established in some other way. The physician’s integrity, as viewed by the patients, the community, and him- or herself, may be threatened. Conversely, those who routinely perform placebo induction may become dulled to the ethical dilemmas involved. Another concern involves the message sent, not just to patients, but to those in training. How would one explain this behavior to a student? Furthermore, if we teach dishonesty in one domain of medicine, would this behavior indicate that it is acceptable to be less than honest in other situations? How would one prevent the slippery slope that might ensue? As Kirk stated [19], ‘‘if we want our junior colleagues to treat patients with respect, we must lead by example.” The example may be set by clearly outlining why deceptive induction techniques are unacceptable and presenting appropriate alternative diagnostic tools.
3. Are there alternatives? Although the benefits of establishing a diagnosis are clear, it is not clear that these procedures actually provide, or are necessary to provide, the needed information. While some studies report the sensitivity of placebo induction to be as high as 72–84% [20– 23], these numbers might overestimate the utility of this approach. In many cases no spells are elicited. In the study by Ribaï et al. [14], only 9 of 16 subjects had NES on provocative testing when no such spells were captured on routine monitoring. They also found that 9 of 28 patients (32%) did not have any events in response to saline injection. Bazil et al. [24], despite concluding that ‘‘the saline provocation test is a. . .sensitive method of evaluating seizure patients for NES,” found that of 52 patients, 40% had no response to the procedure. Patients with NES may have increased medical knowledge due to frequent hospitalizations, making these procedures of reduced value [25].
112
B.A. Leeman / Epilepsy & Behavior 15 (2009) 110–114
In other patients, atypical spells may be generated, which could complicate the diagnosis. Walczak et al. [23] found that 15% of subjects with epilepsy and 8% of subjects with NES had atypical nonepileptic spells elicited by saline injection. Bazil et al. [24] found that 23% of patients had responses unlike their usual events, and Gumnit [1] noted that ‘‘the number of false-positive results in our group [was] far too high.” Drake [16] also noted the high false-positive rates and suggested that psychiatric evaluation may more readily identify features typical of NES. In surveys regarding the use of placebo induction, the most common reason for not employing such procedures was not an ethical concern, but rather poor test reliability [12]. Some authors have stated that the response to suggestion means nothing more than that the patient is suggestible. It seems unreasonable to subject a patient to deception and its possible harms for a test of poor predictive value. Moreover, most epileptologists would agree that only in limited situations would provoking one event alter management. The clinician must obtain confirmation that a single event is typical of all of the patient’s spells. Often the patient experiences multiple spell types, and 10–60% of patients with NES have both epileptic and nonepileptic seizures [26–32]. Each spell type must be evaluated (e.g., by history and/or EEG correlate). It is a misconception among some clinicians that a single provocative procedure is sufficient to establish the diagnosis when patients may suffer from both nonepileptic and epileptic seizures. Patients with frontal lobe seizures present a particular challenge, in that the semiology of the seizures may be unusual and recordings during spells may be unremarkable, calling NES into question. Therefore, capturing many examples of spells may be necessary to ensure that a diagnosis of epilepsy is not missed. This discussion is predicated on the assumption that deception is integral to the induction procedure, in that the procedure would be ineffective without it. It is not clear, however, that this is the case. Devinsky and Fisher [33] advocated use of induction procedures, but only with full disclosure. Their methods included hyperventilation and photic stimulation, as often used to elicit abnormalities in routine EEG. Induction was performed in carefully selected patients, after obtaining consent and explaining the procedure with honesty. They found this to be an effective means of eliciting nonepileptic spells, although the frequency of response was not noted. Benbadis et al. [34,35] documented that 66–84% of attempted inductions resulted in a typical spell using hyperventilation and photic stimulation. Their technique did not involve overt deception, and yielded response rates similar to that of saline injections. Similarly, an older body of literature investigated the response to placebo agents and found that patients may still derive benefit even when they know a substance is inert [36,37]. Eliciting the placebo response may be less related to deception and more related to patient awareness of the disorder, sense of control, and belief that the physician is caring [38]. Many epilepsy centers do not perform placebo inductions, but rather rely on clinical history, background EEG, video/EEG recordings of spontaneous events, psychiatric interviews, and MRI scans to arrive at a diagnosis. In the Parra et al. study [39] of time to spontaneous events, all patients did ultimately have a spontaneous spell after the first 58 h of EEG monitoring. This underscores the notion that effective alternatives to deception are available; therefore, a need for deceit cannot be justified.
4. A violation of autonomy and inherent morality Deception violates the principle of autonomy, which provides that patients have the right to participate in decisions that have consequences that may affect them. Even if consent for deception is obtained, by definition the consent is not fully informed [40],
and it is unknown what idiosyncratic bits of information may alter a given patient’s desire for participation. Some supporters of deceptive induction procedures claim that if patients could give full informed consent, they would choose to do so. It is not clear on what this information is based. A patient who is not upset by the procedure, or is happy to have received a diagnosis, is not equivalent to the counterfactual experiment of being able to go back in time and provide consent. Patients who have been manipulated in this way may in fact be pleased with the outcome, but may be quite displeased with the manner in which it was obtained. To predict a patient’s reaction to deceptive induction, one may survey a surrogate population [10]. In a survey of epileptologists regarding their use of induction procedures, Schachter et al. [11] found that one of the most common side effects of induction was ‘‘anger” and that 13% felt that their patients had ‘‘difficulty in accepting” the procedure. In a study of attitudes toward the use of deception in antidepressant drug trials [41], a balanced placebo design with post hoc debriefing was considered to be ‘‘unacceptable by the overwhelming majority” of patients with depression, psychiatrists, and general practitioners polled. Three of the five patients interviewed would not agree to participation in such a trial. In a study of outpatient stroke care [42], patients were not told of a specific question that would be asked at a later date. Although most subjects were not upset to later learn by letter that information was withheld, there were a small number of participants who had negative feelings or decreased willingness to participate in future studies after learning of the omission. In a study of subjects’ attitudes toward use of deception in disguised alcohol questionnaires, participants were misled regarding the purpose of the questions. The subjects, including both alcoholics and nonalcoholics, were told that the survey would evaluate general health practices in an effort to design better questionnaires, when in reality the focus was to assess drinking behavior. Twenty-five percent of subjects were not comfortable with being misinformed regarding the true intent of the survey, 22% felt an invasion of privacy, 17% believed that the methods were unethical and they would not participate in future questionnaire studies, and a subset of subjects indicated that use of such procedures would diminish trust in medical professionals [43]. It may be that a patient’s reaction to deceptive practices depends on the social acceptability of the revealed or induced behaviors, with more negative behaviors making deception more objectionable [10]. Perhaps deceptive induction of a nonepileptic event, an often socially stigmatized behavior, would be viewed more negatively by the patient than if socially positive or neutral behaviors were elicited. Hence, it is not clear that if given the option, a patient with NES would consent to deceptive induction, and in some cases patients have in fact declined to undergo the procedure [23]. Furthermore, to consider placebo induction as an exception to informed consent is not justified. It is generally accepted that in life-threatening emergencies or intraoperative complications, requirements for informed consent may be waived. Placebo induction, however, does not fall into these categories. Perhaps a more compelling argument for waiving informed consent is that of ‘‘therapeutic privilege,” which indicates that consent may be unnecessary when the information withheld would be damaging to the patient. In the case of placebo inductions, however, withholding the truth, rather than disclosure, causes the damage. Stagno and Smith [18] asserted that ‘‘when the procedure is considered justifiable, it should be done only if. . .a debriefing of the patients. . .is to take place following the procedure.” It is unclear that debriefing makes the lack of informed consent any more acceptable, however, akin to it being easier to ask for forgiveness than permission. Debriefing a patient regarding deceptive techniques does not eliminate the psychological risks. As Sieber et al. [10] describe,
B.A. Leeman / Epilepsy & Behavior 15 (2009) 110–114
debriefing involves two components: dehoaxing and desensitizing. Dehoaxing entails explanation of the deceptive nature of the procedure, whereas desensitization refers to removal of any emotional harm caused by the deception. As the authors state, ‘‘Unfortunately, dehoaxing is not always. . .harmless, and it is not always obvious what emotional harm remains or how to remove it.” Those who espouse consequentialism, who judge actions by their consequences, would state that if no apparent harm was done, then deception was a reasonable approach. Harm may not be apparent, however, unless subjects are directly questioned after debriefing. The reader may draw a parallel to the Lustig et al. [44] study, in which healthy subjects underwent a sham procedure. They were told that an electric current would pass through their heads, possibly causing pain. When the deception was revealed they felt, at least transiently, ‘‘shame, embarrassment, anger and disappointment.” One may extrapolate to patients with NES and imagine that they might feel the same way after deceptive induction. The final, and perhaps strongest, argument against the use of deception in diagnosis is that it is inherently wrong to deceive. Moreover, as Burack et al. [13] state, ‘‘inherent inequities in power and knowledge . . . generate special obligations not to deceive.” Patients trust physicians with information of great sensitivity, and the physician must behave in a manner to maintain that trust. The Hippocratic Oath states that ‘‘I will keep [patients] from harm and injustice. . ..In purity and holiness I will guard my life and my art. . ..Whatever houses I may visit, I will come for the benefit of the sick, remaining free of all intentional injustice. . ..” Little is more harmful, more unjust, less pure, and less holy than deceiving a patient. Those who allow for the procedure in certain situations, by preceding mental health examinations or postinduction debriefing, cannot escape that deception is always inherently wrong. On that basis alone, its use in the clinical setting is never justified. 5. What can be learned from other disciplines? The common underlying pathologic process in NES and other forms of conversion is psychiatric in nature, and the views of psychiatrists regarding deception may help to guide us. Historically, although opinions were divided, much of the psychiatric literature argued against the use of placebos. Specifically, Shapiro’s review of the literature [45] found that most nonpsychiatric articles supported use of placebos in treatment and diagnosis, whereas the psychiatric articles did not. Multiple issues were raised against the use of placebos, including concerns about the ethics of deception and its effects on the doctor–patient relationship. Hofling [46] cautioned against use of placebos as a diagnostic device because of the risk that truly organic symptoms may be elicited by the placebo, causing the underlying disease to go unrecognized. He also warned against their use in patients with paranoia, as such patients may be particularly suspect of the physicians’ motives and the doctor–patient relationship may be at higher risk. Salfield [47] argued that placebos should be avoided in patients with psychiatric issues, as it would make future therapy difficult. If the patient was led to believe that the administered ‘‘medication” was efficacious in eliciting or aborting the illness, (s)he may be less likely to believe that there is a psychological etiology. More recently, Kirk [19] noted that alternative approaches to deception should be sought, as treatment of patients with conversion disorder should not risk compromise of the doctor–patient relationship.
6. Summary In conclusion, the use of deceptive provocative procedures in the diagnosis of NES should be discouraged. The potential benefit is small, in that eliciting a single spell is often of limited clinical
113
utility. The possible harms, however, are great. Most importantly, such behavior endangers the physician–patient relationship, and may result in patients’ failing to comply with NES treatment or seeking medical attention in the future. Furthermore, deceptive induction is inherently wrong, and should be avoided by physicians in all cases on that basis alone. Available alternative approaches that do not require deception should be employed. References [1] Gumnit RJ. The differential diagnosis of epilepsy in nonepileptic paroxysmal disorders. In: Wyllie E, editor. The treatment of epilepsy: principles and practices. Philadelphia: Lea & Febiger; 1993. p. 692–6. [2] Massey EW, McHenry LC. Hysteroepilepsy in the nineteenth century: Charcot and Gowers. Neurology 1986;36:65–7. [3] Goetz CG. J.-M. Charcot and simulated neurologic disease: attitudes and diagnostic strategies. Neurology 2007;69:103–9. [4] LaFrance WC, Devinsky O. The treatment of nonepileptic seizures: historical perspectives and future directions. Epilepsia 2004;45(Suppl. 2):15–21. [5] Tucker BR. Speaking of Weir Mitchell. Am J Psychiatry 1936;93:341–6. [6] Levy RS, Jankovic J. Placebo-induced conversion reaction: a neurobehavioral and EEG study of hysterical aphasia, seizure, and coma. J Abnorm Psychol 1983;92:243–9. [7] Monday K, Jankovic J. Psychogenic myoclonus. Neurology 1993;43:349–52. [8] Fahn S, Williams DT. Psychogenic dystonia. Adv Neurol 1988;50:431–55. [9] Platt R. Two essays on the practice of medicine. Lancet 1947;250:305–7. [10] Sieber JE, Iannuzzo R, Rodriguez B. Deception methods in psychology: have they changed in 23 years? Ethics Behav 1995;5:67–85. [11] Schachter SC, Brown F, Rowan AJ. Provocative testing for nonepileptic seizures: attitudes and practices in the United States among American Epilepsy Society members. J Epilepsy 1996;9:249–52. [12] Stagno SJ, Smith ML. Use of induction procedures in diagnosing psychogenic seizures. J Epilepsy 1996;9:153–8. [13] Burack JH, Back AL, Pearlman RA. Provoking nonepileptic seizures: the ethics of deceptive diagnostic testing. Hastings Cent Rep 1997;27:24–33. [14] Ribaï P, Tugendhaft P, Legros B. Usefulness of prolonged video-EEG monitoring and provocative procedure with saline injection for the diagnosis of non epileptic seizures of psychogenic origin. J Neurol 2006;253:328–32. [15] Howe EG. Deceiving patients for their own good. J Clin Ethics 1997;8:211–6. [16] Drake Jr ME. Saline activation of pseudoepileptic seizures: clinical EEG and neuropsychiatric observations. Clin Electroencephalogr 1985;16:171–6. [17] Smith ML, Stagno SJ, Dolske M, et al. Induction procedures for psychogenic seizures: ethical and clinical considerations. J Clin Ethics 1997;8:217–29. [18] Stagno SJ, Smith ML. The use of placebo in diagnosing psychogenic seizures: who is being deceived? Semin Neurol 1997;17:213–8. [19] Kirk EP. Treatment by deception is bad medicine. Lancet 2003;362:668. [20] Cohen RJ, Suter C. Hysterical seizures: suggestion as a provocative EEG test. Ann Neurol 1982;11:391–5. [21] Dericiog˘lu N, Saygi S, Cig˘er A. The value of provocation methods in patients suspected of having non-epileptic seizures. Seizure 1999;8:152–6. [22] Lancman ME, Asconape JJ, Craven WT, Howard G, Penry JK. Predictive value of induction of psychogenic seizures by suggestion. Ann Neurol 1994;35:359–61. [23] Walczak TS, Williams DT, Berten W. Utility and reliability of placebo infusion in the evaluation of patients with seizures. Neurology 1994;44:394–9. [24] Bazil CW, Kothari M, Luciano D, et al. Provocation of nonepileptic seizures by suggestion in a general seizure population. Epilepsia 1994;35:768–70. [25] Zaidi A, Crampton S, Clough P, Fitzpatrick A, Scheepers B. Head-up tilting is a useful provocative test for psychogenic non-epileptic seizures. Seizure 1999;8:353–5. [26] O’Sullivan SS, Spillane JE, McMahon EM, et al. Clinical characteristics and outcome of patients diagnosed with psychogenic nonepileptic seizures: a 5year review. Epilepsy Behav 2007;11:77–84. [27] Kotagal P, Costa M, Wyllie E, Wolgamuth B. Paroxysmal nonepileptic events in children and adolescents. Pediatrics 2002;110:e46. [28] Benbadis SR, Agrawal V, Tatum WO. How many patients with psychogenic nonepileptic seizures also have epilepsy? Neurology 2001;57:915–7. [29] Ozkara C, Dreifuss FE. Differential diagnosis in pseudoepileptic seizures. Epilepsia 1993;34:294–8. [30] Meierkord H, Will B, Fish D, Shorvon S. The clinical features and prognosis of pseudoseizures diagnosed using video-EEG telemetry. Neurology 1991;41:1643–6. [31] Reuber M, Fernandez G, Bauer J, Helmstaedter C, Elger CE. Diagnostic delay in psychogenic nonepileptic seizures. Neurology 2002;58:493–5. [32] Schachter SC. Psychiatric disorders that resemble seizures. In: Fisch B, editor. Epilepsy and intensive care monitoring: principles and practice. New York: Demos Medical Publishing; in press. [33] Devinsky O, Fisher R. Ethical use of placebos and provocative testing in diagnosing nonepileptic seizures. Neurology 1996;47:866–70. [34] Benbadis SR, Johnson K, Anthony K, et al. Induction of psychogenic nonepileptic seizures without placebo. Neurology 2000;55:1904–5. [35] Benbadis SR, Siegrist K, Tatum WO, Heriaud L, Anthony K. Short-term outpatient EEG video with induction in the diagnosis of psychogenic seizures. Neurology 2004;63:1728–30.
114
B.A. Leeman / Epilepsy & Behavior 15 (2009) 110–114
[36] Park LC, Covi L. Nonblind placebo trial: an exploration of neurotic outpatients’ response to placebo when its inert content is disclosed. Arch Gen Psychiatry 1965;12:336–45. [37] Vogel AV, Goodwin JS, Goodwin JM. The therapeutics of placebo. Am Fam Physician 1980;22:105–9. [38] Brody H. The lie that heals: the ethics of giving placebos. Ann Intern Med 1982;97:112–8. [39] Parra J, Kanner AM, Iriarte J, Gil-Nagel A. When should induction protocols be used in the diagnostic evaluation of patients with paroxysmal events? Epilepsia 1998;39:863–7. [40] Evans M. Justified deception? The single blind placebo in drug research. J Med Ethics 2000;26:188–93. [41] Dowrick CF, Hughes JG, Hiscock JJ, Wigglesworth M, Walley TJ. Considering the case for an antidepressant drug trial involving temporary deception: a
[42]
[43]
[44] [45] [46] [47]
qualitative enquiry of potential participants. BMC Health Serv Res 2007;7:64–76. Boter H, van Delden JJM, de Haan RJ, Rinkel GJE. Patients’ evaluation of informed consent to postponed information: cohort study. Br Med J 2004;329:86–7. Fleming MF, Bruno M, Barry K, Fost N. Informed consent, deception, and the use of disguised alcohol questionnaires. Am J Drug Alcohol Abuse 1989;15:309–19. Lustig BA, Coverdale J, Bayer T, Chiang E. Attitudes toward the use of deception in psychologically induced pain. IRB 1993;15:6. mv8. Shapiro AK. Attitudes toward the use of placebos in treatment. J Nerv Ment Dis 1960;130:200–11. Hofling CK. The place of placebos in medical practice. GP 1955;11:103–7. Salfield DJ. The placebo. Lancet 1953;262:940.
Contents lists available at ScienceDirect
Epilepsy & Behavior journal homepage: www.elsevier.com/locate/yebeh
Controversies in Epilepsy and Behavior
Provocative techniques should not be used for the diagnosis of psychogenic nonepileptic seizures Beth A. Leeman * Departments of Neurology, Beth Israel Deaconess Medical Center, Massachusetts General Hospital, Harvard Medical School, Boston, MA, USA Behavioral Neurology Unit, Beth Israel Deaconess Medical Center, 330 Brookline Avenue, KS-2, Boston, MA 02215, USA
Patients with psychogenic nonepileptic seizures (NES) have episodes of abnormal movements, alterations of awareness and unresponsiveness that appear similar to epileptic seizures. Unlike epileptic seizures, however, these spells occur in the absence of concurrent epileptiform activity in the brain. Such episodes are common, occurring in approximately 20% of those presenting to academic epilepsy centers for evaluation [1]. Although there is no gold standard process for diagnosis, the most common approach involves capturing the spell during electroencephalography (EEG), to demonstrate the lack of corresponding epileptiform discharges. Often patients with NES will not have spells while undergoing EEG monitoring, however, which creates difficulty in establishing a diagnosis and often leads to the use of induction procedures. Historically, the neurologists’ approach to the diagnosis of NES or other forms of conversion disorder has been controversial. In the 19th century, Charcot developed criteria for the differentiation of ‘‘hysterical attacks” from epilepsy, which included the precipitation or termination of spells by pressing on the ovaries or other ‘‘hysterogenic” areas [2]. A review of his works revealed that he was ‘‘not above slight trickery” or suggestion, as when he spoke offensively to a patient with psychogenic hearing loss to elicit a response [3]. Others in this time period were reported to use more radical methods. Gowers’ approach included applying electric shocks, covering the face with a towel to the point of asphyxiation, pouring water in the mouth, inducing vomiting with apomorphine, and pulling on pubic hair to abort spells and establish a diagnosis [4]. Perhaps the most colorful story from this era is that of Mitchell’s consultation on a puzzling case of paralysis. After examining the patient, he requested that the other doctors step into the hallway. When he joined them a moment later, they asked if the patient would ever walk again. He responded ‘‘yes, in a moment,” at which time the patient ran out of the room. When asked what happened, Dr. Mitchell replied, ‘‘I set the bedclothes on fire” [5]. In the mid- to late-20th century, diagnostic methods for conversion disorder may have been more ‘‘humane,” but nevertheless deceptive. Many physicians advocated the use of placebos for the diagnosis of conversion disorder in its various forms. Levy and Jankovic [6] reported a particularly striking case in which they documented a ‘‘dose dependent” reaction to a placebo injection, resulting in psychogenic nonfluent aphasia, NES, and profound * Fax: +1 617 667 7981. E-mail address: [email protected]. 1525-5050/$ - see front matter Ó 2009 Elsevier Inc. All rights reserved. doi:10.1016/j.yebeh.2009.01.013
unresponsiveness, in evaluation of a psychogenic reaction to phenytoin. Monday and Jankovic [7] reported a series of patients with psychogenic myoclonus whose symptoms initially worsened with saline injections given to ‘‘enhance the abnormal movements” and later improved with additional saline injections given to ‘‘ameliorate” the movements. They encouraged the use of ‘‘positive criteria,” such as response to placebo, when considering the diagnosis of a psychogenic movement disorder. Similarly, Fahn and Williams [8] proposed a classification system to reflect certainty of diagnosis with respect to psychogenic dystonia. Criteria for ‘‘documented psychogenic dystonia” included the response to placebo. Multiple successful trials of placebo treatment were taken as the ‘‘best evidence for documenting [the] diagnosis.” The repeated response to placebo trials has been advanced as a diagnostic method by other authors as well, with Platt [9] providing the example of its use in psychogenic headache. Although much of this literature dates pre-1995, if social psychology research may serve as a guide, publications from 1969 to 1992 did not demonstrate any large, sustained shifts in the use of deceptive practices to indicate that ideas would have changed in recent years [10]. Currently, an estimated 39–73% of epileptologists have adopted the practice of inducing spells during EEG recording, typically by the administration of placebo agents such as saline injections, alcohol pads applied transdermally, tuning forks placed on the forehead, and head tilting maneuvers [11,12]. These methods commonly involve misleading the patient to believe that an active, proconvulsant agent is being administered and that an ‘‘antidote” (also a placebo) would be provided if a spell is prolonged or severe. These methods may involve omissions of the truth or direct lies. Burack et al. [13], for example, describe an induction procedure in patient RB: RB had EEG electrodes and a blood pressure cuff attached and an intravenous catheter placed. A resident physician. . .asked RB’s consent to administer intravenously ‘‘an electrolyte solution that may bring on spells”. . .. The solution, unbeknownst to RB, was normal saline. RB was asked to pump his arm vigorously while an infusion was begun, to help the ‘‘infusion serum” circulate. A sense of drama and expectancy pervaded the procedure: the physician’s voice and manner were tense, and the patient was perspiring and appeared anxious. The physician narrated the patient’s movements and responses, and noted a point at which the infusion reached ‘‘full therapeutic levels.” RB then had a typical seizure. . .. The physician stated loudly
B.A. Leeman / Epilepsy & Behavior 15 (2009) 110–114
that serum levels should be back to zero; seconds later, the muscular jerking tapered off. The patients may or may not be debriefed after the procedure, although patients who are not told of the deception could easily discover the truth later via other means (e.g., medical records or other health professionals involved in their care). Proponents of this approach argue that use of deception in induction procedures is justified, based primarily on claims that such procedures will clarify the diagnosis in a safe and efficient manner, thereby sparing the patient from the harm of an erroneous diagnosis of epilepsy. Most recently, Ribaï et al. [14] took an extreme stance, concluding that the investigation of NES should ‘‘always include” a provocative saline test. Although little has been published on this issue, the majority of the authors have conceded that use of deception may be justifiable in certain limited situations, but that the ethical issues must be carefully considered [13,15–18]. The reader should question, however, whether establishing a diagnosis should ever outweigh the violations of autonomy and nonmaleficence caused by deception. 1. Are there benefits? Some believe that documenting a response to placebo will more definitively establish the diagnosis of NES. Confirming a diagnosis is important, as patients may then be directed toward appropriate treatments, such as cognitive-behavioral therapies and antidepressants for comorbid depression and anxiety. In addition, it is hoped that patients with NES would avoid the social and financial hardships that result from a diagnosis of epilepsy. They would also be spared potentially harmful treatments for epileptic seizures, as anticonvulsant medications would be ineffective for treatment of NES. Antiepileptic medications are often costly and have potential side effects. These side effects can be life-threatening, such as aplastic anemia and Stevens–Johnson syndrome. Patients presenting with nonepileptic status could also avoid the excessive sedative medications, intubations, and ICU admissions that occur with true status epilepticus. To hasten the diagnosis by induction may also prevent long, costly hospital stays and the concomitant risks of nosocomial infections. Those adopting a paternalistic approach would state that beneficence in these regards would supersede the need to respect patient autonomy. 2. Are there harms? Supporters of induction may claim that the diagnosis is made with little risk, assuming that those employing placebos with malintent (i.e., to anger an objectionable patient) are removed from the equation. In their view, the duty of nonmaleficence, to treat the patient in the least harmful manner, is upheld. Saline is an inert substance, injected through an intravenous line that is already placed for clinical purposes. Alcohol pads are not irritating to the skin. Although people may sustain injuries during induced spells, this risk is no different than that incurred during spontaneous spells captured by routine video/EEG monitoring. Hence, proponents of deceptive induction procedures would argue that these methods act in the patients’ best interests and with little potential for harm. The counterargument, however, is that the principle of nonmaleficence has indeed been violated, in that deceptive methods actually cause more psychological harm than good. To deceive a patient risks irreparable damage to the physician– patient relationship. The patient may have distrust of that physician, which may extend to other physicians or the medical system as a whole. This distrust could potentially compromise adherence to the treatment plan for NES. This places the patient at risk for all of the consequences of untreated NES and depression, including
111
injury or even suicide. The patient may now be less willing to seek care for other medical issues, as well. Establishing the diagnosis of NES in this manner could also affect the public’s view of the medical system more broadly. Patients may already be suspicious of the diagnosis of NES, and providing further reason for mistrust would be ill-advised. Furthermore, deceptive practices in the diagnosis of NES prey upon a particularly vulnerable patient population. These patients are often depressed and anxious, and may have personality or other psychiatric disorders. Histories of physical or sexual abuse, unstable home environments, and other traumatic life events are common. Hence, patients may already have difficulty in trusting others. Dishonesty from a physician, who has pledged to deliver honest, compassionate care, may be even more destructive to the relationships of these patients, with physicians or others, than anticipated. Stagno and Smith [18] take this argument a step further and assert that for patients with a history of sexual abuse, ‘‘the insertion of a needle and infusion of a fluid (in the case of IV inductions) may be a psychologic symbol of the abuse.” Some authors advocate psychiatric screening for personality traits that may make a patient susceptible to psychological harm, to be performed prior to the decision to use deceptive induction. Although an estimated 43% of centers using induction obtain a psychiatric screen prior to the procedure [12], it is not clear that such screens are adequately predictive. While debriefing may explain the rationale for deception, and provide potentially persuasive arguments for its use, it cannot be known how a given patient, particularly one who is psychiatrically ill, will react. Burack et al. [13] noted that harms may affect the physician as well, as (s) he may be ‘‘burdened by the unforeseeable and cumulative strains and risk of the further deceptions required to support the initial one.” This may be especially true for those who use additional deception after induction, telling patients that the diagnosis of NES was established in some other way. The physician’s integrity, as viewed by the patients, the community, and him- or herself, may be threatened. Conversely, those who routinely perform placebo induction may become dulled to the ethical dilemmas involved. Another concern involves the message sent, not just to patients, but to those in training. How would one explain this behavior to a student? Furthermore, if we teach dishonesty in one domain of medicine, would this behavior indicate that it is acceptable to be less than honest in other situations? How would one prevent the slippery slope that might ensue? As Kirk stated [19], ‘‘if we want our junior colleagues to treat patients with respect, we must lead by example.” The example may be set by clearly outlining why deceptive induction techniques are unacceptable and presenting appropriate alternative diagnostic tools.
3. Are there alternatives? Although the benefits of establishing a diagnosis are clear, it is not clear that these procedures actually provide, or are necessary to provide, the needed information. While some studies report the sensitivity of placebo induction to be as high as 72–84% [20– 23], these numbers might overestimate the utility of this approach. In many cases no spells are elicited. In the study by Ribaï et al. [14], only 9 of 16 subjects had NES on provocative testing when no such spells were captured on routine monitoring. They also found that 9 of 28 patients (32%) did not have any events in response to saline injection. Bazil et al. [24], despite concluding that ‘‘the saline provocation test is a. . .sensitive method of evaluating seizure patients for NES,” found that of 52 patients, 40% had no response to the procedure. Patients with NES may have increased medical knowledge due to frequent hospitalizations, making these procedures of reduced value [25].
112
B.A. Leeman / Epilepsy & Behavior 15 (2009) 110–114
In other patients, atypical spells may be generated, which could complicate the diagnosis. Walczak et al. [23] found that 15% of subjects with epilepsy and 8% of subjects with NES had atypical nonepileptic spells elicited by saline injection. Bazil et al. [24] found that 23% of patients had responses unlike their usual events, and Gumnit [1] noted that ‘‘the number of false-positive results in our group [was] far too high.” Drake [16] also noted the high false-positive rates and suggested that psychiatric evaluation may more readily identify features typical of NES. In surveys regarding the use of placebo induction, the most common reason for not employing such procedures was not an ethical concern, but rather poor test reliability [12]. Some authors have stated that the response to suggestion means nothing more than that the patient is suggestible. It seems unreasonable to subject a patient to deception and its possible harms for a test of poor predictive value. Moreover, most epileptologists would agree that only in limited situations would provoking one event alter management. The clinician must obtain confirmation that a single event is typical of all of the patient’s spells. Often the patient experiences multiple spell types, and 10–60% of patients with NES have both epileptic and nonepileptic seizures [26–32]. Each spell type must be evaluated (e.g., by history and/or EEG correlate). It is a misconception among some clinicians that a single provocative procedure is sufficient to establish the diagnosis when patients may suffer from both nonepileptic and epileptic seizures. Patients with frontal lobe seizures present a particular challenge, in that the semiology of the seizures may be unusual and recordings during spells may be unremarkable, calling NES into question. Therefore, capturing many examples of spells may be necessary to ensure that a diagnosis of epilepsy is not missed. This discussion is predicated on the assumption that deception is integral to the induction procedure, in that the procedure would be ineffective without it. It is not clear, however, that this is the case. Devinsky and Fisher [33] advocated use of induction procedures, but only with full disclosure. Their methods included hyperventilation and photic stimulation, as often used to elicit abnormalities in routine EEG. Induction was performed in carefully selected patients, after obtaining consent and explaining the procedure with honesty. They found this to be an effective means of eliciting nonepileptic spells, although the frequency of response was not noted. Benbadis et al. [34,35] documented that 66–84% of attempted inductions resulted in a typical spell using hyperventilation and photic stimulation. Their technique did not involve overt deception, and yielded response rates similar to that of saline injections. Similarly, an older body of literature investigated the response to placebo agents and found that patients may still derive benefit even when they know a substance is inert [36,37]. Eliciting the placebo response may be less related to deception and more related to patient awareness of the disorder, sense of control, and belief that the physician is caring [38]. Many epilepsy centers do not perform placebo inductions, but rather rely on clinical history, background EEG, video/EEG recordings of spontaneous events, psychiatric interviews, and MRI scans to arrive at a diagnosis. In the Parra et al. study [39] of time to spontaneous events, all patients did ultimately have a spontaneous spell after the first 58 h of EEG monitoring. This underscores the notion that effective alternatives to deception are available; therefore, a need for deceit cannot be justified.
4. A violation of autonomy and inherent morality Deception violates the principle of autonomy, which provides that patients have the right to participate in decisions that have consequences that may affect them. Even if consent for deception is obtained, by definition the consent is not fully informed [40],
and it is unknown what idiosyncratic bits of information may alter a given patient’s desire for participation. Some supporters of deceptive induction procedures claim that if patients could give full informed consent, they would choose to do so. It is not clear on what this information is based. A patient who is not upset by the procedure, or is happy to have received a diagnosis, is not equivalent to the counterfactual experiment of being able to go back in time and provide consent. Patients who have been manipulated in this way may in fact be pleased with the outcome, but may be quite displeased with the manner in which it was obtained. To predict a patient’s reaction to deceptive induction, one may survey a surrogate population [10]. In a survey of epileptologists regarding their use of induction procedures, Schachter et al. [11] found that one of the most common side effects of induction was ‘‘anger” and that 13% felt that their patients had ‘‘difficulty in accepting” the procedure. In a study of attitudes toward the use of deception in antidepressant drug trials [41], a balanced placebo design with post hoc debriefing was considered to be ‘‘unacceptable by the overwhelming majority” of patients with depression, psychiatrists, and general practitioners polled. Three of the five patients interviewed would not agree to participation in such a trial. In a study of outpatient stroke care [42], patients were not told of a specific question that would be asked at a later date. Although most subjects were not upset to later learn by letter that information was withheld, there were a small number of participants who had negative feelings or decreased willingness to participate in future studies after learning of the omission. In a study of subjects’ attitudes toward use of deception in disguised alcohol questionnaires, participants were misled regarding the purpose of the questions. The subjects, including both alcoholics and nonalcoholics, were told that the survey would evaluate general health practices in an effort to design better questionnaires, when in reality the focus was to assess drinking behavior. Twenty-five percent of subjects were not comfortable with being misinformed regarding the true intent of the survey, 22% felt an invasion of privacy, 17% believed that the methods were unethical and they would not participate in future questionnaire studies, and a subset of subjects indicated that use of such procedures would diminish trust in medical professionals [43]. It may be that a patient’s reaction to deceptive practices depends on the social acceptability of the revealed or induced behaviors, with more negative behaviors making deception more objectionable [10]. Perhaps deceptive induction of a nonepileptic event, an often socially stigmatized behavior, would be viewed more negatively by the patient than if socially positive or neutral behaviors were elicited. Hence, it is not clear that if given the option, a patient with NES would consent to deceptive induction, and in some cases patients have in fact declined to undergo the procedure [23]. Furthermore, to consider placebo induction as an exception to informed consent is not justified. It is generally accepted that in life-threatening emergencies or intraoperative complications, requirements for informed consent may be waived. Placebo induction, however, does not fall into these categories. Perhaps a more compelling argument for waiving informed consent is that of ‘‘therapeutic privilege,” which indicates that consent may be unnecessary when the information withheld would be damaging to the patient. In the case of placebo inductions, however, withholding the truth, rather than disclosure, causes the damage. Stagno and Smith [18] asserted that ‘‘when the procedure is considered justifiable, it should be done only if. . .a debriefing of the patients. . .is to take place following the procedure.” It is unclear that debriefing makes the lack of informed consent any more acceptable, however, akin to it being easier to ask for forgiveness than permission. Debriefing a patient regarding deceptive techniques does not eliminate the psychological risks. As Sieber et al. [10] describe,
B.A. Leeman / Epilepsy & Behavior 15 (2009) 110–114
debriefing involves two components: dehoaxing and desensitizing. Dehoaxing entails explanation of the deceptive nature of the procedure, whereas desensitization refers to removal of any emotional harm caused by the deception. As the authors state, ‘‘Unfortunately, dehoaxing is not always. . .harmless, and it is not always obvious what emotional harm remains or how to remove it.” Those who espouse consequentialism, who judge actions by their consequences, would state that if no apparent harm was done, then deception was a reasonable approach. Harm may not be apparent, however, unless subjects are directly questioned after debriefing. The reader may draw a parallel to the Lustig et al. [44] study, in which healthy subjects underwent a sham procedure. They were told that an electric current would pass through their heads, possibly causing pain. When the deception was revealed they felt, at least transiently, ‘‘shame, embarrassment, anger and disappointment.” One may extrapolate to patients with NES and imagine that they might feel the same way after deceptive induction. The final, and perhaps strongest, argument against the use of deception in diagnosis is that it is inherently wrong to deceive. Moreover, as Burack et al. [13] state, ‘‘inherent inequities in power and knowledge . . . generate special obligations not to deceive.” Patients trust physicians with information of great sensitivity, and the physician must behave in a manner to maintain that trust. The Hippocratic Oath states that ‘‘I will keep [patients] from harm and injustice. . ..In purity and holiness I will guard my life and my art. . ..Whatever houses I may visit, I will come for the benefit of the sick, remaining free of all intentional injustice. . ..” Little is more harmful, more unjust, less pure, and less holy than deceiving a patient. Those who allow for the procedure in certain situations, by preceding mental health examinations or postinduction debriefing, cannot escape that deception is always inherently wrong. On that basis alone, its use in the clinical setting is never justified. 5. What can be learned from other disciplines? The common underlying pathologic process in NES and other forms of conversion is psychiatric in nature, and the views of psychiatrists regarding deception may help to guide us. Historically, although opinions were divided, much of the psychiatric literature argued against the use of placebos. Specifically, Shapiro’s review of the literature [45] found that most nonpsychiatric articles supported use of placebos in treatment and diagnosis, whereas the psychiatric articles did not. Multiple issues were raised against the use of placebos, including concerns about the ethics of deception and its effects on the doctor–patient relationship. Hofling [46] cautioned against use of placebos as a diagnostic device because of the risk that truly organic symptoms may be elicited by the placebo, causing the underlying disease to go unrecognized. He also warned against their use in patients with paranoia, as such patients may be particularly suspect of the physicians’ motives and the doctor–patient relationship may be at higher risk. Salfield [47] argued that placebos should be avoided in patients with psychiatric issues, as it would make future therapy difficult. If the patient was led to believe that the administered ‘‘medication” was efficacious in eliciting or aborting the illness, (s)he may be less likely to believe that there is a psychological etiology. More recently, Kirk [19] noted that alternative approaches to deception should be sought, as treatment of patients with conversion disorder should not risk compromise of the doctor–patient relationship.
6. Summary In conclusion, the use of deceptive provocative procedures in the diagnosis of NES should be discouraged. The potential benefit is small, in that eliciting a single spell is often of limited clinical
113
utility. The possible harms, however, are great. Most importantly, such behavior endangers the physician–patient relationship, and may result in patients’ failing to comply with NES treatment or seeking medical attention in the future. Furthermore, deceptive induction is inherently wrong, and should be avoided by physicians in all cases on that basis alone. Available alternative approaches that do not require deception should be employed. References [1] Gumnit RJ. The differential diagnosis of epilepsy in nonepileptic paroxysmal disorders. In: Wyllie E, editor. The treatment of epilepsy: principles and practices. Philadelphia: Lea & Febiger; 1993. p. 692–6. [2] Massey EW, McHenry LC. Hysteroepilepsy in the nineteenth century: Charcot and Gowers. Neurology 1986;36:65–7. [3] Goetz CG. J.-M. Charcot and simulated neurologic disease: attitudes and diagnostic strategies. Neurology 2007;69:103–9. [4] LaFrance WC, Devinsky O. The treatment of nonepileptic seizures: historical perspectives and future directions. Epilepsia 2004;45(Suppl. 2):15–21. [5] Tucker BR. Speaking of Weir Mitchell. Am J Psychiatry 1936;93:341–6. [6] Levy RS, Jankovic J. Placebo-induced conversion reaction: a neurobehavioral and EEG study of hysterical aphasia, seizure, and coma. J Abnorm Psychol 1983;92:243–9. [7] Monday K, Jankovic J. Psychogenic myoclonus. Neurology 1993;43:349–52. [8] Fahn S, Williams DT. Psychogenic dystonia. Adv Neurol 1988;50:431–55. [9] Platt R. Two essays on the practice of medicine. Lancet 1947;250:305–7. [10] Sieber JE, Iannuzzo R, Rodriguez B. Deception methods in psychology: have they changed in 23 years? Ethics Behav 1995;5:67–85. [11] Schachter SC, Brown F, Rowan AJ. Provocative testing for nonepileptic seizures: attitudes and practices in the United States among American Epilepsy Society members. J Epilepsy 1996;9:249–52. [12] Stagno SJ, Smith ML. Use of induction procedures in diagnosing psychogenic seizures. J Epilepsy 1996;9:153–8. [13] Burack JH, Back AL, Pearlman RA. Provoking nonepileptic seizures: the ethics of deceptive diagnostic testing. Hastings Cent Rep 1997;27:24–33. [14] Ribaï P, Tugendhaft P, Legros B. Usefulness of prolonged video-EEG monitoring and provocative procedure with saline injection for the diagnosis of non epileptic seizures of psychogenic origin. J Neurol 2006;253:328–32. [15] Howe EG. Deceiving patients for their own good. J Clin Ethics 1997;8:211–6. [16] Drake Jr ME. Saline activation of pseudoepileptic seizures: clinical EEG and neuropsychiatric observations. Clin Electroencephalogr 1985;16:171–6. [17] Smith ML, Stagno SJ, Dolske M, et al. Induction procedures for psychogenic seizures: ethical and clinical considerations. J Clin Ethics 1997;8:217–29. [18] Stagno SJ, Smith ML. The use of placebo in diagnosing psychogenic seizures: who is being deceived? Semin Neurol 1997;17:213–8. [19] Kirk EP. Treatment by deception is bad medicine. Lancet 2003;362:668. [20] Cohen RJ, Suter C. Hysterical seizures: suggestion as a provocative EEG test. Ann Neurol 1982;11:391–5. [21] Dericiog˘lu N, Saygi S, Cig˘er A. The value of provocation methods in patients suspected of having non-epileptic seizures. Seizure 1999;8:152–6. [22] Lancman ME, Asconape JJ, Craven WT, Howard G, Penry JK. Predictive value of induction of psychogenic seizures by suggestion. Ann Neurol 1994;35:359–61. [23] Walczak TS, Williams DT, Berten W. Utility and reliability of placebo infusion in the evaluation of patients with seizures. Neurology 1994;44:394–9. [24] Bazil CW, Kothari M, Luciano D, et al. Provocation of nonepileptic seizures by suggestion in a general seizure population. Epilepsia 1994;35:768–70. [25] Zaidi A, Crampton S, Clough P, Fitzpatrick A, Scheepers B. Head-up tilting is a useful provocative test for psychogenic non-epileptic seizures. Seizure 1999;8:353–5. [26] O’Sullivan SS, Spillane JE, McMahon EM, et al. Clinical characteristics and outcome of patients diagnosed with psychogenic nonepileptic seizures: a 5year review. Epilepsy Behav 2007;11:77–84. [27] Kotagal P, Costa M, Wyllie E, Wolgamuth B. Paroxysmal nonepileptic events in children and adolescents. Pediatrics 2002;110:e46. [28] Benbadis SR, Agrawal V, Tatum WO. How many patients with psychogenic nonepileptic seizures also have epilepsy? Neurology 2001;57:915–7. [29] Ozkara C, Dreifuss FE. Differential diagnosis in pseudoepileptic seizures. Epilepsia 1993;34:294–8. [30] Meierkord H, Will B, Fish D, Shorvon S. The clinical features and prognosis of pseudoseizures diagnosed using video-EEG telemetry. Neurology 1991;41:1643–6. [31] Reuber M, Fernandez G, Bauer J, Helmstaedter C, Elger CE. Diagnostic delay in psychogenic nonepileptic seizures. Neurology 2002;58:493–5. [32] Schachter SC. Psychiatric disorders that resemble seizures. In: Fisch B, editor. Epilepsy and intensive care monitoring: principles and practice. New York: Demos Medical Publishing; in press. [33] Devinsky O, Fisher R. Ethical use of placebos and provocative testing in diagnosing nonepileptic seizures. Neurology 1996;47:866–70. [34] Benbadis SR, Johnson K, Anthony K, et al. Induction of psychogenic nonepileptic seizures without placebo. Neurology 2000;55:1904–5. [35] Benbadis SR, Siegrist K, Tatum WO, Heriaud L, Anthony K. Short-term outpatient EEG video with induction in the diagnosis of psychogenic seizures. Neurology 2004;63:1728–30.
114
B.A. Leeman / Epilepsy & Behavior 15 (2009) 110–114
[36] Park LC, Covi L. Nonblind placebo trial: an exploration of neurotic outpatients’ response to placebo when its inert content is disclosed. Arch Gen Psychiatry 1965;12:336–45. [37] Vogel AV, Goodwin JS, Goodwin JM. The therapeutics of placebo. Am Fam Physician 1980;22:105–9. [38] Brody H. The lie that heals: the ethics of giving placebos. Ann Intern Med 1982;97:112–8. [39] Parra J, Kanner AM, Iriarte J, Gil-Nagel A. When should induction protocols be used in the diagnostic evaluation of patients with paroxysmal events? Epilepsia 1998;39:863–7. [40] Evans M. Justified deception? The single blind placebo in drug research. J Med Ethics 2000;26:188–93. [41] Dowrick CF, Hughes JG, Hiscock JJ, Wigglesworth M, Walley TJ. Considering the case for an antidepressant drug trial involving temporary deception: a
[42]
[43]
[44] [45] [46] [47]
qualitative enquiry of potential participants. BMC Health Serv Res 2007;7:64–76. Boter H, van Delden JJM, de Haan RJ, Rinkel GJE. Patients’ evaluation of informed consent to postponed information: cohort study. Br Med J 2004;329:86–7. Fleming MF, Bruno M, Barry K, Fost N. Informed consent, deception, and the use of disguised alcohol questionnaires. Am J Drug Alcohol Abuse 1989;15:309–19. Lustig BA, Coverdale J, Bayer T, Chiang E. Attitudes toward the use of deception in psychologically induced pain. IRB 1993;15:6. mv8. Shapiro AK. Attitudes toward the use of placebos in treatment. J Nerv Ment Dis 1960;130:200–11. Hofling CK. The place of placebos in medical practice. GP 1955;11:103–7. Salfield DJ. The placebo. Lancet 1953;262:940.