Article

Multidisciplinary Management of Pediatric Nonepileptic Seizures

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Abstract

In May 2005 the National Institute of Neurological Disorders and Stroke, the National Institute of Mental Health, and the American Epilepsy Society sponsored a multidisciplinary workshop for a group of experts to develop a research agenda for nonepileptic seizure (NES) treatment trials. In this article, we use the term NESs as recommended by this workgroup (LaFrance et al., 2006) and focus solely on NESs associated with the DSM-IV diagnosis of conversion disorder. We are not using the term psychogenic because some find it prematurely dismissive of possible biological processes. Although NESs have historically been widely recognized, they continue to present dilemmas for clinicians. Differing views regarding etiology, associated vague terminology, and the lack of standardized diagnostic criteria and treatment guidelines leave clinicians perplexed and discouraged. Arriving at an accurate NES diagnosis takes about 3.5 years (median delay), by which time 50% to 95% of the children are taking antiepileptic drugs (AEDs; Paolicchi, 2002; Wyllie et al., 1999). Over time, episodes can become more stereotyped and increasingly resemble epileptic seizures, making diagnosis more difficult. Consequently, early diagnosis and treatment of NESs may prevent unnecessary diagnostic procedures and treatments as well as iatrogenic complications. The aim of this article is to share our clinical experience with the management of children with NESs in tertiary urban pediatric hospitals.

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... 31 It is critical to ensure that patients and families understand the interdisciplinary nature of evaluation and the multifactorial nature of SSRDs to normalize the involvement of mental health professionals. 6,11,32 Delayed mental health involvement results in patients and families perceiving that their care is being "handed off" to mental health and further stigmatizes the condition. 10 The presence of inconsistent physical symptoms and/or psychosocial stressors, although raising the concern for possible somatization, is insufficient to make a diagnosis of SSRD. ...
... 7 Ensuring effective, interdisciplinary communication with the family has been found to be associated with improved treatment adherence, participation in outpatient follow-up, and improved patient outcomes. 32 Team meetings have been shown to be associated with high patient and caregiver satisfaction in the pediatric inpatient medical setting, particularly for patients with comorbid medical and mental health diagnoses. 34 A coordinated approach by 7,32 Using terminology that is consistent, understandable, and based on the DSM-5 and providing explanation regarding the biopsychosocial model is critical to effective symptom evaluation and management. ...
... 32 Team meetings have been shown to be associated with high patient and caregiver satisfaction in the pediatric inpatient medical setting, particularly for patients with comorbid medical and mental health diagnoses. 34 A coordinated approach by 7,32 Using terminology that is consistent, understandable, and based on the DSM-5 and providing explanation regarding the biopsychosocial model is critical to effective symptom evaluation and management. 7 Therefore, practical scripts are included in the CP to guide clinicians at all levels of training to effectively discuss the conceptual framework of symptom development, diagnosis, and treatment regarding pediatric SSRDs. ...
Article
Somatic symptom and related disorders (SSRDs) are commonly encountered in pediatric hospital settings. There is, however, a lack of standardization of care across institutions for youth with these disorders. These patients are diagnostically and psychosocially complex, posing significant challenges for medical and behavioral health care providers. SSRDs are associated with significant health care use, cost to families and hospitals, and risk for iatrogenic interventions and missed diagnoses. With sponsorship from the American Academy of Child and Adolescent Psychiatry and input from multidisciplinary stakeholders, we describe the first attempt to develop a clinical pathway and standardize the care of patients with SSRDs in pediatric hospital settings by a working group of pediatric consultation-liaison psychiatrists from multiple institutions across North America. The authors of the SSRD clinical pathway outline 5 key steps from admission to discharge and include practical, evidence-informed approaches to the assessment and management of children and adolescents who are medically hospitalized with SSRDs.
... Benbadis et al. (2000) describe the use of an effective procedure involving the use of suggestion, hyperventilation, and photic stimulation in 19 adults with suspected PNES, and because these techniques are also used to induce epileptic seizures, there is an absence of deception and thus could be seen as more ethical than placebo induction. Plioplys et al. (2007) emphasize that PNES are not a default diagnosis based on negative video-telemetry, as the mental health clinician also needs to show evidence of an underlying conversion disorder. The importance of taking a detailed history (i.e., history of paroxysmal events, child medical/developmental history, and family history) from multiple sources (e.g., child, parents and school staff) has been emphasized (Plioplys et al., 2007), and may allow health care professionals to differentiate between PNES and epilepsy (Reuber & Mayor, 2012). ...
... Plioplys et al. (2007) emphasize that PNES are not a default diagnosis based on negative video-telemetry, as the mental health clinician also needs to show evidence of an underlying conversion disorder. The importance of taking a detailed history (i.e., history of paroxysmal events, child medical/developmental history, and family history) from multiple sources (e.g., child, parents and school staff) has been emphasized (Plioplys et al., 2007), and may allow health care professionals to differentiate between PNES and epilepsy (Reuber & Mayor, 2012). Plioplys et al. (2007) argue that interviewing the child and parents separately may be helpful and also suggest that it is important to address the degree to which the parents are invested in the child having medical problems (or not having a psychological problem). ...
... The importance of taking a detailed history (i.e., history of paroxysmal events, child medical/developmental history, and family history) from multiple sources (e.g., child, parents and school staff) has been emphasized (Plioplys et al., 2007), and may allow health care professionals to differentiate between PNES and epilepsy (Reuber & Mayor, 2012). Plioplys et al. (2007) argue that interviewing the child and parents separately may be helpful and also suggest that it is important to address the degree to which the parents are invested in the child having medical problems (or not having a psychological problem). ...
Article
One of the considerations when a child presents with paroxysmal events is psychogenic nonepileptic seizures (PNES). PNES are discernible changes in behavior or consciousness that resemble epileptic seizures but are not accompanied by electrophysiologic changes. They are usually understood as the manifestation of a conversion disorder that reflects underlying psychological distress. There is a lack of population-based data on the prevalence or incidence of PNES in pediatric populations. The prevalence of PNES in children would appear to be lower than that in the adult population, but the prevalence of PNES seems to increase with age, and nonepileptic paroxysmal events are more likely to be PNES in adolescence than earlier in childhood. In terms of manifestation, PNES in childhood have been described using various categorizations and terminology, making comparisons across studies difficult. There is some evidence that events are more likely to involve unresponsiveness in younger children and prominent motor symptoms in older children. The most common precipitating factors would appear to be school-related difficulties and interpersonal conflict within the child's family. In terms of psychopathology, children with PNES are at high risk for symptoms of depression and anxiety. Accurate diagnosis of PNES in children is likely to involve taking a comprehensive description of the episodes, garnering the child's medical/developmental history, video-electroencephalography (video-EEG) to rule out epileptic seizures, and an evaluation of family functioning. The importance of effective and sensitive communication of the diagnosis of PNES has been emphasized and management approaches will typically involve multidisciplinary efforts to safely manage the events at home and at school. Interventions to reduce the effect of precipitating psychosocial stressors and the involvement of a mental health professional to treat comorbid psychopathology will also form part of an effective management plan. Outcome at follow-up is reported to be largely positive, although studies have not been able to follow all children, and few studies have focused on predictors of a good outcome. Future controlled intervention studies using a range of outcome measures are needed to identify efficacious approaches and predictors of best outcome.
... It is certainly important to gather a detailed clinical history (i.e., a history of paroxysmal events, medical and developmental history, family history) from various sources (the child and parents) [12], to help doctors formulate an accurate diagnosis [13]. A history of abuse (32%) [14] or stress in the family (40%) [15] and a concomitant psychopathology (social anxiety in particular) [16,17] are common in children and adolescents with PNES. ...
... Seven were female, and three were male. The average age was 12 (8)(9)(10)(11)(12)(13)(14)(15)(16)(17) years. All patients had a normal intelligence quotient. ...
Article
The differential diagnosis of epileptic seizures (ES) and psychogenic nonepileptic seizures (PNES) is often difficult, especially in pediatric and adolescent settings. Conversation analysis (CA) can be a worthwhile diagnostic tool in adults. The aim of this study was to assess the diagnostic value of CA in Italian children and adolescents. Ten patients (seven females and three males), diagnosed using video-EEG as having either ES or PNES, underwent a video-recorded interview by a physician from outside the center specifically trained for this purpose. An external linguistic rater then examined the video recordings and transcripts using CA. Diagnoses formulated on the basis of interactional and linguistic features of the patients' speech were compared with diagnoses made by seizure experts on the basis of all available clinical information including the video-EEG findings. Conversation analysis diagnoses corresponded to the video-EEG diagnoses in 8 out of 10 cases. In conclusion, while some conversational adaptation is necessary to enable children and adolescents to share their seizure experiences with an adult health professional, this study indicates the differential diagnostic potential of a CA approach in these young people with PNES or epilepsy. Larger samples are obviously needed to confirm these findings.
... In our study, the treatment of five patients (26.3%), started with a preliminary diagnosis of epilepsy, was terminated after confirming the diagnosis of shuddering attack. The ideal routine EEG recording in the differential diagnosis of epileptic and PNEs should be obtained in sleep and wakeful states, while falling asleep [15]. In our study, epileptic activities were observed in only one case in short-term EEG, while simultaneous epileptiform activity was not observed in the same patient's Video-EEG. ...
... In the differential diagnosis of epileptic seizures from PNEs, routine biochemistry, basal metabolic tests, antiepileptic drug level in patients with epilepsy, toxicology screening, EEG, and ineligible patients, brain MRI tests may be performed [15]. Serological tests, biochemistry, and metabolic tests were performed on our cases, but could not help in making a diagnosis. ...
Article
Background Shuddering attacks (SA) are one of the most common childhood paroxysmal nonepileptic events (PNEs). These attacks usually start between the first 4th and 6th months of life with rapid tremors of the head and adduction of the arms and knees. A number of factors including eating, breastfeeding, and playing stimulating games have been shown to trigger the attacks; however, the exact pathogenesis remains unknown. It has been stated that there is no need for further research in patients diagnosed, and spontaneous regression is expected. Purpose This study aimed to identify the causes, accompanying clinical conditions, possible differential diagnosis of SA, and the role of video-electroencephalogram (V-EEG) recording for accurate diagnosis. Methods Nineteen cases with SA have been collected from the database of Erciyes University Pediatric Neurology Clinic, where 52.6% are boys (n = 10) and 47.6% are girls (n = 9). The relationship between the onset and disappearance of SA symptoms and variables including family history, birth history, age, sleep, teething during SA, video-EEG recordings, brain imaging, and accompanying conditions such as epilepsy have been investigated by retrospective analysis. Results Four cases were found to have gastroesophageal reflux, one had epilepsy, and one had Marcus Gunn Jaw Winking Syndrome. No accompanying conditions could be identified for rest of the cases. It was observed that onset of symptoms in 15 (78.9%) of 19 cases coincided remarkably with the period of teething. Conclusion We speculate that there might be an indirect link between SA and teething and teething may be a triggering or an aggravating factor for SA.
... The term PNES refers to seizure-like events that are due to underlying psychological stressors or conflicts rather than epilepsy [32]. The flurry of research over the last decade has advanced clinical understanding of PNES, but this continues to be a field in development, particularly in pediatric and adolescent patients [3,[27][28][32][33][34][35][36][37][38][39][40][41][42][43][44][45][46][47][48]. Current management recommendations highlight that acceptance of the diagnosis, which depends on the exclusion of epilepsy and other disorders, is a critical first step to successful treatment [3,28,48]. ...
... The flurry of research over the last decade has advanced clinical understanding of PNES, but this continues to be a field in development, particularly in pediatric and adolescent patients [3,[27][28][32][33][34][35][36][37][38][39][40][41][42][43][44][45][46][47][48]. Current management recommendations highlight that acceptance of the diagnosis, which depends on the exclusion of epilepsy and other disorders, is a critical first step to successful treatment [3,28,48]. In order for the patient and family to accept a PNES diagnosis however, several diagnostic challenges must be overcome [17,[23][24][45][46][47]. ...
Article
To date, only a very narrow window of ethical dilemmas in psychogenic nonepileptic seizures (PNES) has been explored. Numerous distinct ethical dilemmas arise in diagnosing and treating pediatric and adolescent patients with PNESs. Important ethical values at stake include trust, transparency, confidentiality, professionalism, autonomy of all stakeholders, and justice. In order to further elucidate the ethical challenges in caring for this population, an ethical analysis of the special challenges faced in four specific domains is undertaken: (1) conducting and communicating a diagnosis of PNESs, (2) advising patients about full transparency and disclosure to community including patients' peers, (3) responding to requests to continue antiepileptic drugs, and (4) managing challenges arising from school policy and procedure. An analysis of these ethical issues is essential for the advancement of best care practices that promote the overall well-being of patients and their families.
... Treatment should be multidisciplinary and include the mental health professional, the child, parents, school, and a pediatrician/ pediatric neurologist [19,[43][44][45]. The first step of treatment is providing psychoeducation regar-ding PNES to the family and school [19,44]. ...
Article
Context: Though psychogenic non-epileptic seizures (PNES) are seen commonly during evaluation of children and adolescents with epilepsy, the literature regarding developmental changes in PNES is limited. Evidence acquisition: Literature search was conducted in PubMed. Key search terms included: Pseudoseizure* OR PNES OR [(non-epileptic or nonepileptic or psychogenic or non-epileptic attack disorder) AND (seizure*)], resulting in 3,236 articles. Filters included human, ages 1-18 years, English language and last 15 years (2004-2019), resulting in 533 articles. We reviewed 33 articles, which included 19 articles that involved children (1-18 years), with 10 or more children with PNES in their study group. 21 articles obtained in cross references that were outside the filter setting (including time frame and age range) were also reviewed, for a total of 54 articles. Results: Majority of the studies were retrospective. We detail clinical features, predisposing factors and appropriate workup for children and adolescents with possible PNES. There is no consensus regarding frequency of psychiatric comorbidities in children with PNES. No controlled trials of treatment of PNES in children are available, but cognitive behavioral therapy is the consensus for adult PNES. Outcome appears to be better in children with PNES. Conclusion: There is a need for be long-term prospective studies to document various clinical features and outcome of pediatric and adolescent PNES, and also the comorbid conditions.
... Video EEG, the gold standard in the differential diagnosis of paroxysmal events [11,12], plays a central role in clinical work [10,[13][14][15]. With VEEG, the diagnostic accuracy rate can reach up to 88.0% [16], whereas that of ambulatory EEG is only 67.5% [17]. ...
Article
Full-text available
Background The diagnosis of paroxysmal events in infants is often challenging. Reasons include the child’s inability to express discomfort and the inability to record video electroencephalography at home. The prevalence of mobile phones, which can record videos, may be beneficial to these patients. In China, this advantage may be even more significant given the vast population and the uneven distribution of medical resources. Objective The aim of this study is to investigate the value of mobile phone videos in increasing the diagnostic accuracy and cost savings of paroxysmal events in infants. Methods Clinical data, including descriptions and home videos of episodes, from 12 patients with paroxysmal events were collected. The investigation was conducted in six centers during pediatric academic conferences. All 452 practitioners present were asked to make their diagnoses by just the descriptions of the events, and then remake their diagnoses after watching the corresponding home videos of the episodes. The doctor’s information, including educational background, profession, working years, and working hospital level, was also recorded. The cost savings from accurate diagnoses were measured on the basis of using online consultation, which can also be done easily by mobile phone. All data were recorded in the form of questionnaires designed for this study. ResultsWe collected 452 questionnaires, 301 of which met the criteria (66.6%) and were analyzed. The mean correct diagnoses with and without videos was 8.4 (SD 1.7) of 12 and 7.5 (SD 1.7) of 12, respectively. For epileptic seizures, mobile phone videos increased the mean accurate diagnoses by 3.9%; for nonepileptic events, it was 11.5% and both were statistically different (P=.006 for epileptic events; P
... Nonepileptic seizures (NES) are observed in youth with [29] and without intellectual disability [30]. In children and youth with normal intelligence, NES are associated primarily with conversion disorder, as well as with comorbid depression, anxiety, and ADHD diagnoses (see reviews in [31,32]). ...
Article
This paper first summarizes the main findings of clinical studies conducted over the past two and a half decades on psychopathology (i.e., psychiatric diagnoses, behavior and emotional problems) in children with new onset and chronic epilepsy both with and without intellectual disability who are treated medically and surgically. Although impaired social relationships are core features of the psychiatric disorders found in pediatric epilepsy, few studies have examined social competence (i.e., social behavior, social adjustment, and social cognition) in these children. There also is a dearth of treatment studies on the frequent psychiatric comorbidities of pediatric epilepsy, attention deficit hyperactivity disorder, anxiety disorders, and depression. Drs. Hamiwka and Jones then describe their current and planned studies on social competence and cognitive behavioral treatment of anxiety disorders, respectively, in these children and how they might mitigate the poor long-term psychiatric and social outcome of pediatric epilepsy.
... Video-EEG monitoring to obtain an ictal recording plays a central role in the management of paroxysmal events in children (10)(11)(12)(13). This method is considered as the golden standard in the differential diagnosis of paroxysmal events (14,15) . The diagnostic accuracy rate of video-EEG monitoring can reach up to 88.0% (16), whereas that of ambulatory EEG is only 67.5% (17). ...
Preprint
Full-text available
BACKGROUND The diagnosis of paroxysmal events in infants is often challenging. Reasons include the child’s inability to express discomfort and the inability to record video electroencephalography at home. The prevalence of mobile phones, which can record videos, may be beneficial to these patients. In China, this advantage may be even more significant given the vast population and the uneven distribution of medical resources. OBJECTIVE The aim of this study is to investigate the value of mobile phone videos in increasing the diagnostic accuracy and cost savings of paroxysmal events in infants. METHODS Clinical data, including descriptions and home videos of episodes, from 12 patients with paroxysmal events were collected. The investigation was conducted in six centers during pediatric academic conferences. All 452 practitioners present were asked to make their diagnoses by just the descriptions of the events, and then remake their diagnoses after watching the corresponding home videos of the episodes. The doctor’s information, including educational background, profession, working years, and working hospital level, was also recorded. The cost savings from accurate diagnoses were measured on the basis of using online consultation, which can also be done easily by mobile phone. All data were recorded in the form of questionnaires designed for this study. RESULTS We collected 452 questionnaires, 301 of which met the criteria (66.6%) and were analyzed. The mean correct diagnoses with and without videos was 8.4 (SD 1.7) of 12 and 7.5 (SD 1.7) of 12, respectively. For epileptic seizures, mobile phone videos increased the mean accurate diagnoses by 3.9%; for nonepileptic events, it was 11.5% and both were statistically different (P=.006 for epileptic events; P<.001 for nonepileptic events). Pediatric neurologists with longer working years had higher diagnostic accuracy; whereas, their working hospital level and educational background made no difference. For patients with paroxysmal events, at least US $673.90 per capita and US $128 million nationwide could be saved annually, which is 12.02% of the total cost for correct diagnosis. CONCLUSIONS Home videos made on mobile phones are a cost-effective tool for the diagnosis of paroxysmal events in infants. They can facilitate the diagnosis of paroxysmal events in infants and thereby save costs. The best choice for infants with paroxysmal events on their initial visit is to record their events first and then show the video to a neurologist with longer working years through online consultation.
... The PNES becomes an expression of what is wrong within the family rather than the individual. There may be modeling of parental somatizing behaviors (Plioplys et al., 2007). ...
Article
Psychogenic nonepileptic seizures (PNES) are a common presentation to the emergency room and neurology department. They are often misdiagnosed and treated as epileptic seizures. Inappropriate treatment leads to poor patient outcomes and iatrogenic complications, as the underlying mechanisms and treatments are distinctly different. Most causes involve a conversion or dissociative reaction to a prior traumatic experience or abuse. Recognition of the characteristic clinical features of PNES and utilization of video-electroencephalography to confirm the diagnosis is critical. Discontinuation of treatment for epilepsy (unless comorbid PNES and epilepsy is present), acceptance of the diagnosis, and a multidisciplinary treatment plan with clinical psychologists, neurologist, and psychiatrist improve patient and healthcare outcomes.
... Neurologist and epileptologist should educate the patient and their relatives that these values are not easily reconcilable and deciding whether to disclose the diagnosis involves an inherent tradeoff, and the prognosis of these patients is still relatively poor, and a good outcome seems dependent on a young age at diagnosis, early diagnosis, less severe psychological comorbidities, and continued follow-up and management by the diagnosing neurologist, epileptologist, or clinician [8,49,105,[108][109][110][111][112][113][114][115][116]. ...
... Parents of affected children may prefer the terms functional seizures, nonepileptic events, and NEAD 4 whilst proposed management guidelines use the term NES. 5 Although these names are often used interchangeably, it is not clear that they are always used to indicate the same phenomena. Some terms may refer to non-epileptic events of specifically psychogenic aetiology whereas others may encompass nonepileptic events which are not psychogenic in origin, such as syncope, tremors, myoclonus, dystonia, and parasomnias. ...
... 50 The communication of the diagnosis to the patient and the parents is also of vital importance with emphasis being placed that the child is not faking these events. 51 Typically, in children and adolescents, PNES are thought to be a manifestation of conversion disorder 52 and the diagnosis should be conveyed positively without blame placed on the patient. 53 Adolescence represents an awkward period of time when a child morphs into an adult both physically and psychologically. ...
Article
Paroxysmal nonepileptic spells refer to events that clinically can resemble epileptic seizures but are not caused by abnormal epileptiform activity in the brain. These spells can often be difficult to diagnose owing to clinical similarities, but a correct diagnosis is needed to prevent unnecessary treatment with antiepileptic drugs. We review the most common diagnoses in adolescents that can be mistaken for epilepsy, including migraine, syncope, movement disorders, sleep disorders, and psychologic disorders.
Article
Objective: No formal guidelines for diagnosing psychogenic nonepileptic seizures (PNES) in children exist, and little is known about the clinical practice of diagnosing PNES in the pediatric setting. We therefore performed a national survey as a first step to document pediatricians' current diagnostic practice for PNES. Methods: A questionnaire was distributed to all pediatricians (n=64) working in the field of neuropediatrics and/or social pediatrics in the Danish hospital setting to uncover their use of terminology and of the International Classification of Diseases, 10th Revision (ICD-10) codes as well as their clinical diagnostic approach to pediatric PNES. The questionnaire included questions on 18 history and 24 paroxysmal event characteristics. Results: The response rate was 95% (61/64). There was no consensus on which terminology and diagnostic codes to use. Five history characteristics (psychosocial stressors/trauma, sexual abuse, paroxysmal events typically occur in stressful situations, no effect of antiepileptic drugs, and physical abuse) and six paroxysmal event characteristics (resisted eyelid opening, avoidance/guarding behavior, paroxysmal events occur in the presence of others, closed eyes, rarely injury related to paroxysmal event, and absence of postictal change) were agreed to be very predictive of PNES by at least 50% of the pediatricians. Supplementary diagnostic tests such as blood chemistry measurements (e.g., blood glucose or acute phase reactants; i.e., white blood cell count and C-reactive protein) and electrocardiography were inconsistently used. Only 49% of the respondents reported to use video-electroencephalography (VEEG) frequently as part of their diagnostic procedure. Significance: To our knowledge, this is the first national survey that offers a systematic insight into the diagnostic practices for children with PNES in the hospital setting. The results demonstrate a need for clinical guidelines to improve and systematize the diagnostic approach for PNES in children.
Article
Full-text available
Based primarily on consensus due to lack of relevant clinical studies, it is difficult to distinguish PNES from epileptic seizures. Failure to make this distinction may result in lack of appropriate management and a decline in the quality of life for the child and family. Based on some research evidence as well as consensus, common associated stressors in children include school difficulties, family discord, and interpersonal conflicts with peers and friends, with sexual abuse being less common than in adults. Anxiety and depression commonly are associated with PNES. (2)(3)(4) PNES should be differentiated from other paroxysmal nonepileptic events. Based on strong research evidence, prolonged video-EEG monitoring is the gold standard in making a definitive diagnosis. (5)(6) Based on some research evidence as well as consensus, early diagnosis and referral to a psychiatrist or psychologist for treatment of the underlying psychopathology with individual and family therapy are the mainstay of successful management of pediatric NES due to psychological causes. Children who have PNES have a better prognosis than adults.
Conference Paper
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Purpose: Paroxysmal non-epileptic events (PNEs) are observable, abrupt changes in consciousness or behavior that present similar to seizures, although are not accompanied by electro physiologic changes. They are thought to rise from psychological etiology and significantly impact both the pediatric and adult populations. Frequency of PNEs in the pediatric literature ranges between 10-23% of individuals referred to an epilepsy clinic. Cases are likely underreported due to the difficulty in accurately defining PNEs symptoms. This leads to inappropriate use of anti-epileptic drugs and unnecessary diagnostic procedures. Moreover, inaccurate diagnosis prevents correct assessment and treatment from being implemented as well as imposing a heavy and avoidable financial burden on the medical system. Unlike the adult literature, very little information is available regarding the clinical characteristics, treatment and outcome of PNEs in children and adolescents. Aims: We strove to better understand the etiology, features and medical care pathway of PNEs in a pediatric population of patients seen at Alberta Children’s Hospital through following aims: 1) describe demographics, clinical characteristics and presenting symptoms, 2) describe psychological characteristics 3) describe management and outcomes. Methods: Our group embarked on a retrospective chart review of children referred to clinical psychology with a final diagnosis of PNEs. Charts were examined for demographics, non-epileptic presenting symptoms, psychological testing results, neurological testing, co-morbidities, management and outcomes. All patient underwent similar assessment involving parental ratings and self-report measures including the; Behavior Assessment System for Children-Version II and Beck’s Youth Inventory. Patients 13 years of age or older were administered the Millon Adolescent Personality Inventory, a self-report personality assessment. In cases where an EEG study was completed, video recordings of the events were used along-side EEG data to aid in diagnosis. The most apparent diagnostic signs described in the charts were reported. Treatment was consistent with current practices described in adult and pediatric literature and occurred over periods of treatment ranging from brief periods to several months in a non-linear fashion. In addition to medical diagnosis and education, all patients received psychological assessment, diagnosis and PNEs education. Results: The majority of patients identified were adolescents, Caucasian (72%) and female (76%). Initial entry into the medical system was through the emergency room department (66%) and outpatient neurology clinic (31%). Participants’ levels of suggested distress on a personality inventory were highly discrepant from those indicated on parent and self report measures. The most prevalent personality findings were elevated inhibited and submissive personality types with clinical evaluations depicting a tendency for anxiety and depression. It was also displayed that a high number of medical contacts as compared to psychology contacts correlated with refractory PNEs, where predominantly psychological care led to remission status. Lastly, appropriate psychological treatment facilitated a high rate of remission (59%) and partial remission (25%) leading to a significant (84%) decrease in emergency department visits. Conclusions: Accurate identification, diagnosis and treatment is essential in order to provide appropriate care for children and adolescents with PNEs. This study has furthered knowledge around common traits and treatment avenues effective for this population. This study supports that appropriate care expedites rates of remission, decreases health care costs and mediates inappropriate use of AEDs. Currently, a care pathway is not well-established despite significant costs to the healthcare system. Future implementation of clinical decision tools will greatly improve efficiency, cost and treatment for children and adolescents with PNEs.
Chapter
Children and adolescents commonly experience physical symptoms that are not readily explained by identifiable medical illness or tissue pathology. Such symptoms can be transient and benign, or they can be persistent, severe, and disabling. Whether or not the symptoms are explained by a medical problem, some children have great difficulty coping with them. Among children that appear more distressed or disabled by physical symptoms than expected, a diagnosis of Somatoform Disorder can be made. There are presently seven identified Somatoform Disorders that can be applied to children, despite the lack of empirical data regarding the appropriateness of such diagnoses in this age group. A large and growing literature on pediatric chronic pain has informed the conceptualization, assessment, and treatment of childhood somatoform disorders. Most clear regarding the current understanding of chronic somatic symptoms is their multifaceted etiological nature and the necessity of interventions to address biological, psychological, and social precipitating and maintaining factors. Important areas of assessment include the nature of the physical symptoms themselves, including their location, duration, quality, variability, and intensity; the social, emotional, physical, cognitive, and academic functioning of the symptomatic child; and, family factors such as parenting style, response to child symptoms, and psychopathology. Frequently employed treatment strategies include medications, individual and/or family cognitive behavioral interventions, and body-based therapies. Ideally, these assessment and treatment methodologies are collaboratively implemented within a biopsychosocial framework by medical and psychological professionals who are familiar with the related research evidence base.
Article
Background This article systematically reviews the literature on the effectiveness of psychological interventions for NES in children and adolescents. Methods Embase, Medline and PsycINFO were searched to December 2020, for articles published in English, which used an objective measure to evaluate the outcome of a psychological intervention for NES. Further studies were identified from reference lists and experts in the field were contacted for unpublished studies. Results Twelve studies met inclusion criteria. Of these, four were set up with the primary aim of evaluating an intervention (Flewelling, Koehler, & Shaffer, 2020; Fobian, Long, & Szaflarski, 2020; Kozlowska et al., 2018; Terry et al., 2020) of which two used a control group, two were prospective outcome studies and eight looked at retrospective clinical data. Two studies evaluated a single treatment modality, the others multiple treatment components. Overall, psychoeducation, and top-down psychotherapy, such as cognitive therapies, were the most frequent interventions, with recent studies describing body-oriented (bottom-up) approaches. Analysis across all studies identified a range of additional intervention components which included assessment and / or treatment for co-morbidities, liaison with school and support for parents, highlighting the importance of individualised treatment packages. Conclusions There is a paucity of studies specifically evaluating interventions for NES. Though a range of approaches have been described in managing this patient group, with generally positive outcomes, it is not possible to conclude from the available literature that one treatment approach is superior to another, though the information may be helpful in developing management guidelines.
Article
Full-text available
The differential diagnosis of epileptic seizures (ES) and psychogenic nonepileptic seizures (PNES) is often difficult, especially in pediatric and adolescent settings. Conversation analysis (CA) can be a worthwhile diagnostic tool in adults. The aim of this study was to assess the diagnostic value of CA in Italian children and adolescents. Ten patients (seven females and three males), diagnosed using video-EEG as having either ES or PNES, underwent a video-recorded interview by a physician from outside the center specifically trained for this purpose. An external linguistic rater then examined the video recordings and transcripts using CA. Diagnoses formulated on the basis of interactional and linguistic features of the patients' speech were compared with diagnoses made by seizure experts on the basis of all available clinical information including the video-EEG findings. Conversation analysis diagnoses corresponded to the video-EEG diagnoses in 8 out of 10 cases. In conclusion, while some conversational adaptation is necessary to enable children and adolescents to share their seizure experiences with an adult health professional, this study indicates the differential diagnostic potential of a CA approach in these young people with PNES or epilepsy. Larger samples are obviously needed to confirm these findings.
Article
Objectives: Psychogenic nonepileptic seizures (PNES) are a manifestation of conversion disorder among children but can be difficult to distinguish from epilepsy. We sought to identify characteristics that differentiate children with PNES from those with epilepsy. Methods: We conducted a retrospective cohort study of children admitted with epilepsy or PNES to 45 children's hospitals from 2004 to 2014. Children with PNES (n = 399) versus those with epilepsy (n = 13 241) were compared on demographic and clinical characteristics, testing, treatment, and health care use. Hierarchical multivariable logistic regression was used to identify characteristics associated with PNES diagnosis. Results: Children with PNES were more likely to be female (adjusted odds ratio [aOR] 2.3; 95% confidence interval [CI] 1.8-3.0), older (aOR 3.8; 95% CI 2.7-5.3 for 14-16 years old), African American (aOR 2.0; 95% CI 1.5-2.7), and have diagnosis codes for psychiatric disorders (aOR 7.1; 95% CI 5.6-9.1) and pain (aOR 2.6; 95% CI 1.9-3.4). They were also more likely to be admitted in the fall (aOR 2.0; 95% CI 1.4-2.8) or spring (aOR 1.9; 95% CI 1.4-2.6) versus summer. Total adjusted hospitalization costs were greater for children with epilepsy ($4724, 95% CI $4413-$5057 vs $5326, 95% CI $5259-$5393; P = .001); length of stay was similar. Conclusions: Demographic and clinical characteristics differed among children with PNES versus those with epilepsy, including significantly increased odds of psychiatric and pain diagnoses among children with PNES. To better inform treatment and prognostication for children with PNES, research is needed to understand reasons for these differences, seasonal admission patterns, and the relationship between PNES and other psychiatric disorders.
Article
Objective: To explore paediatricians' attitudes to and treatment practice for children with functional seizures (FS). Methods: In a nationwide survey, all 64 Danish neuro-paediatricians and social paediatricians were invited to complete a structured questionnaire encompassing FS-related issues that included beliefs and attitudes about aetiology and diagnostic assessment, current strategies for management, experienced need for clinical guidelines and better treatment options. Results: A total of 61 paediatricians (95%) participated in the study. Nearly half (46%) had seen more than 30 children with FS during their career. Most (65%) believed in a primarily psychogenic aetiology. More than half (57%) stated that they could make the diagnosis by solely observing a seizure, and 18% indicated the children faked their symptoms. The paediatricians' responses to these issues did not significantly vary according to their level of clinical experience. Furthermore, the majority (78%) expressed a need for clinical guidelines, and only 13% rated existing treatment options as sufficient. Collaborative care between different specialties or management in a child and adolescent mental health services (CAMHS) setting was seen as the best model for treatment. However, only 23% reported often referring these children to CAMHS after making the diagnosis. Conclusion: The findings suggest that introduction of clinical guidelines in this area is highly needed. Such guidelines could promote more formal training of paediatricians in understanding and assessing FS and increased collaboration between paediatrics and CAMHS regarding care for children with this challenging and potentially costly and disabling disorder.
Chapter
This chapter covers some of the more common consultation-liaison psychiatry scenarios, including the patient with “somatic” symptoms of uncertain etiology requiring a thorough evaluation for “other medical” etiologies and the patient with behavioral symptoms more clearly attributable to “other medical” or “biological” (in the biopsychosocial formulation) etiologies. The consultant can play a key role in adequately investigating differential possibilities, in addressing all relevant aspects of the biopsychosocial formulation, in establishing an expected course of recovery, and in insuring that the child and family feel supported. The case vignettes will review the traditional somatic symptom and related disorders as well as other situations where the psychiatrist can help guide the team in considering other medical etiologies for behavioral symptoms.
Article
The goal of this study was to identify assessment tools and associated behavioral domains that differentiate children with psychogenic nonepileptic seizures (PNES) from those with epilepsy. A sample of 24 children with PNES (mean age 14.0 years, 14 female), 24 children with epilepsy (mean age 13.6 years, 13 female), and their parents were recruited from five epilepsy centers in the United States. Participants completed a battery of behavioral questionnaires including somatization, anxiety, and functional disability symptoms. Children with PNES had significantly higher scores on the Childhood Somatization and Functional Disability Inventories, and their parents reported more somatic problems on the Child Behavior Checklist (CBCL). Depression, anxiety, and alexithymia instruments did not differentiate the groups. Measures of somatization and functional disability may be promising tools for differentiating the behavioral profile of PNES from that of epilepsy. Increased somatic awareness and perceived disability emphasize the similarity of PNES to other pediatric somatoform disorders.
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In this preliminary clinical investigation, hypnosis was used in the differential diagnosis of epileptic versus psychogenic seizures (PS). Eight patients with a clinical profile suggesting the presence of PS were given a hypnotic suggestion in which they had to go back in time to the exact moment of their last seizure. They were then asked to concentrate their attention on any unusual feeling or bodily sensation. All 8 patients presented a PS during the age regression protocol. In 6 cases, independent testimony from family members corroborated the morphological similarity of the induced attack and the ones presented in their natural environment. Also, the seizures ended abruptly after a command was given to stop them. A control group of 5 epileptic subjects did not present any signs of discomfort or seizure behavior during the hypnotic protocol. It is argued that a simple procedure as the one described in this investigation can be useful as a diagnostic tool in the differentiation of epileptic from PS attacks.
Pseudoseizures may occur as a somatoform disorder in children and adolescents as well as adults. However, few data are available about psychiatric features or outcome in pediatric patients. We studied 34 patients (25 girls [74%]) who were evaluated by a child psychiatrist at our institution immediately after diagnosis of pseudoseizures by ictal video electroencephalogram (EEG) at ages 9 to 18 years (mean age, 14 years). Each patient had at least 1 pseudoseizure recorded by video EEG that was judged by the patient and family as typical, characterized by unresponsiveness plus limb twitching or limpness and other features, with EEG showing persistence of normal cortical background rhythms. In addition to conversion disorder, 11 patients (32%) had mood disorders including major depression, bipolar disorder, or dysthymic disorder, usually with severe psychosocial stressors. Eight children (24%) had separation anxiety and school refusal with moderate psychosocial stressors. Two patients (6%) had brief reactive psychosis or schizophreniform disorder. A few (1-3) patients each had panic disorder, overanxious disorder, adjustment disorder, oppositional/defiant disorder, or impulse control disorder. Four patients (12%) also had personality disorders. Eleven patients (32%) had a history of sexual abuse. This was especially frequent in the subgroup with mood disorders (7 [64%] of 11 patients). Fifteen patients (44%) had severe family stressors including recent parental divorce, parental discord, or death of a close family member. Two patients (6%) had a history of physical abuse. Freedom from pseudoseizures for the preceding 9 to 55 months (mean, 30 months) was achieved for 15 (72%) of the 21 patients who could be reached for telephone follow-up. For 8 (53%) of these 15 patients, the last pseudoseizure was within 1 month of diagnosis by video EEG. Major mood disorders and severe environmental stress, especially sexual abuse, are common among children and adolescents with pseudoseizures and should be considered in every case. A subgroup of children with separation anxiety and school refusal had less severe psychiatric problems and moderate psychosocial stressors. Clear diagnosis by video EEG, together with prompt psychiatric evaluation and treatment, may result in freedom from pseudoseizures for most children and adolescents.
Article
Autosomal dominant partial epilepsy with auditory features (ADPEAF) is a rare form of nonprogressive lateral temporal lobe epilepsy characterized by partial seizures with auditory disturbances. The gene predisposing to this syndrome was localized to a 10-cM region on chromosome 10q24. We assessed clinical features and linkage evidence in four newly ascertained families with ADPEAF, to refine the clinical phenotype and confirm the genetic localization. We genotyped 41 individuals at seven microsatellite markers spanning the previously defined 10-cM minimal genetic region. We conducted two-point linkage analysis with the ANALYZE computer package, and multipoint parametric and nonparametric linkage analyses as implemented in GENEHUNTER2. In the four families, the number of individuals with idiopathic epilepsy ranged from three to nine. Epilepsy was focal in all of those with idiopathic epilepsy who could be classified. The proportion with auditory symptoms ranged from 67 to 100%. Other ictal symptoms also were reported; of these, sensory symptoms were most common. Linkage analysis showed a maximum 2-point LOD score of 1.86 at (theta=0.0 for marker D10S603, and a maximum multipoint LOD score of 2.93. These findings provide strong confirmation of linkage of a gene causing ADPEAF to chromosome 10q24. The results suggest that the susceptibility gene has a differential effect on the lateral temporal lobe, thereby producing the characteristic clinical features described here. Molecular studies aimed at the identification of the causative gene are underway.
Article
Nonepileptic events (NEE) are common in children, and can be difficult to distinguish from epileptic events. Several strategies can assist in differentiation. The first is an age-based approach to the differential of commonly presenting EEs in neonates, infants, and adolescents. The next strategy is to identify key elements of the patient's history to narrow the possibilities, and third is a rational approach to ancillary testing. There are additional challenges to the diagnosis and evaluation of NEEs in patients with cognitive impairments or mental retardation (MR). Twenty to 25% of neurologically normal patients (34), and up to 60% of children with MR (35) referred for an evaluation of seizures, have NEE. In most instances, the clinical history leads to the diagnosis, and ancillary testing serves as confirmation. But in certain populations, neonates, children with concurrent epilepsy, children in whom pseudoseizures are suspected, and children with MR, early use of video-EEG telemetry is indicated to establish the diagnosis and avoid overtreatment with antiepileptic drugs (AEDs).
Article
Only a few studies have been reported in which suggestion was used to provoke pseudoseizures (PS). In these studies PS were video EEG monitored, and saline injections were administered as placebo. This method may be somewhat unethical and carries a low success rate. The authors, two child psychiatrists (GZ and DS) and a neurologist (NG), applied hypnosis to provoke PS which were monitored by video-EEG. Pre-, intra- and post-ictal serum prolactin levels were determined. The first hypnotic session was diagnostic and for this reason featured controlled attempts to determine whether there might be childhood trauma material. The following sessions easily provoked PS during which the EEG was normal and pre-, intra- and post-ictal serum prolactin levels were identical and within normal values. We conclude that hypnosis (with informed consent) for PS monitored by video-EEG telemetry, seems to be an ethical, cheap and quite easy way to demonstrate PS.
Article
In May 2005, an international, interdisciplinary group of researchers gathered in Bethesda, MD, USA, for a workshop to discuss the development of treatments for patients with nonepileptic seizures (NES). Specific subgroup topics that were covered included: pediatric NES; presenting the diagnosis of NES, outcome measures for NES trials; classification of NES subtypes; and pharmacological treatment approaches and psychotherapies. The intent was to develop specific research strategies that can be expanded to involve a large segment of the epilepsy and psychiatric treatment communities. Various projects have resulted from the workshop, including the initial development of a prospective randomized clinical trial for NES.
Nonepileptic seizures treatment workshop summary Epilepsy Behav
  • WC LaFrance
  • K Alper
  • D Babcock
  • for the NES Treatment Workshop participants