Abstract
Kumari S, Garg D, Sharma S, Pemde HK. Epilepsy Res. 2022;186:106992. doi:10.1016/j.eplepsyres.2022.106992. PMID: 35970090. To characterize the effects of sudden unexpected death in epilepsy (SUDEP) counselling on epilepsy-related childcare behaviour and practices, and emotional distress, measured by the Depression, Anxiety and Stress scale-21 (DASS-21), among parents of children with epilepsy (CWE), and to obtain feedback about SUDEP counselling. We conducted a single-arm, pre- and post-intervention study design. We enrolled 120 parents of CWE. At enrolment, we collected clinical and demographic data, assessed 18 epilepsy-related childcare and behaviour and practices and the DASS-21 score. Parents underwent a three-phase counselling, comprising SUDEP information via written material, followed by an informative video, and then a detailed face-to-face counselling session with treating clinicians. Changes in epilepsy-related childcare behaviour and practices, and DASS-21 scores were reassessed at one month. Qualitative feedback was obtained about SUDEP counselling. At one month, 84.2% of parents reported a change in >2 epilepsy-related childcare behaviour and practices. Significant improvement compared to baseline was observed in reported administration of anti-seizure medications at fixed time (100% versus 57.5%; p < 0.0001), regular exercise (87.5% versus 60%; p < 0.0001), supervision of activity as advised (56.7%-39.2%; p = 0.0096), having a seizure action plan (97.5% versus 20%; p < 0.0001), rescue medications (95% versus 25.8%; p < 0.0001), providing information of child’s epilepsy to schools (100% versus 62%; p < 0.0001), providing an emergency contact number in schools (100% versus 58%; p < 0.0001), and a seizure action plan in their child’s school (54% versus 8%; p < 0.0001). There was no significant change in DASS-21 scores at one month. Parents expressed a preference for face-to-face interaction, in combination with other audiovisual modes, for SUDEP counselling, and being counselled when they were comfortable with their child’s diagnosis of epilepsy. SUDEP disclosure among Indian parents of CWE was associated with significant improvement in self-reported epilepsy-related childcare behaviour and practices, without increase in depression, stress or anxiety.Objectives:
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Commentary
Sudden unexpected death in epilepsy (SUDEP) is the term used for epilepsy-related death without identifiable cause in postmortem examination, which may or may not be related to a witnessed seizure. 1 In adult persons with epilepsy (PWE), it occurs in approximately 1 in 1000 patients per year, while in children with epilepsy (CWE), it occurs in approximately 1 in 4500 patients per year. 1 Cardiorespiratory failure and brainstem arousal system impairment are the presumed mechanisms, but no direct prevention strategy has been established. 2 Yet, young males with learning disability (LD) and drug-resistant epilepsy (DRE) of early onset characterized by frequent, nocturnal, convulsive seizures who have poor adherence to anti-seizure medications (ASMs) and suboptimal supervision are at particular risk. 2 Therefore, optimizing seizure control, improving ASMs compliance, avoiding triggers and addressing psychiatric, sleep-related and cardiopulmonary comorbidities constitute potentially modifiable factors. 2
Given the lack of definitive preventative therapies, the nonuniform risk and the concern of provoking distress, there has been much debate on the need to discuss SUDEP with all PWE and their caregivers, 3 resulting in reduced rates of disclosure by Neurologists globally. 4 The current study 5 adds further to the vexed topic of SUDEP counseling, particularly with regard to parents of CWE. One hundred twenty parents of CWE were recruited from an epilepsy referral center in India. Many children had DRE and LD. Milder forms of seizures such as absences were excluded. The recruited caregivers were predominantly of low socioeconomic status and mostly oblivious of SUDEP at baseline. Those with personal history of epilepsy or psychiatric disorders were excluded. Their baseline anxiety and depression levels, and epilepsy management habits were assessed through a validated survey and a developed 18-item-long questionnaire, respectively. Subsequently, parental counseling regarding SUDEP was performed using sequentially a written brochure, a brief educational video and face-to-face interaction. At 1 month, a quantitative and qualitative evaluation of the counseling content and process was undertaken. It was found that 84% of parents changed in >2 epilepsy-related childcare behavior and practice items, primarily related to set administration of ASMs, regular exercise, and seizure action plan. No psychological repercussions of the disclosure were observed. Most of the parents opined that SUDEP should be discussed, via a combination of methods that includes two-way communication, regardless of seizure control, and when the family feels comfortable with the diagnosis of epilepsy.
The strength of this study is that it addresses an important topic focusing on the pediatric population, where it has been relatively underexplored, particularly in the world region where it was conducted. By quantitatively and qualitatively assessing the views and perceptions on SUDEP counseling in the primary caregivers of this age-group, it provides useful insight regarding their educational needs. Contrary to the vast majority of research in this field that is cross-sectional at a single time point, this study evaluates the impact of an educational intervention on SUDEP to families living with epilepsy.
This study is not free of limitations. Generalizability of the results is threatened by the selectivity of the study population. The risk of response bias from “information seekers” is lurking. Classification and presentation of certain demographic and disease-related characteristics was somewhat ambiguous. While the scale used to assess the psychological impact of the intervention on the caregivers was previously validated, that was not the case with the questionnaire developed for the intervention itself. It is unclear whether the arbitrary cutoff of >2 correct answers pre- and post-education constitute a meaningful change. Moreover, the impact on other important psychosocial aspects of caregivers, such as quality of life and functionality, was not assessed. Despite good intents to incorporate a qualitative component, no formal qualitative methodology was deployed. With 1 month follow-up time, the sustainability of the identified positive impact of the educational intervention remains under question, and it is uncertain if it would lead to seizure frequency and SUDEP rates reduction in the long run. Foremost, the lack of a control group of counseling about epilepsy management in general versus SUDEP specifically prevents us from differentiating whether the observed changes were a mere outcome of improved caregiver knowledge and engagement, rather than directed SUDEP-related counseling.
These drawbacks notwithstanding, the findings of this study are noteworthy and corroborate published literature pertaining to SUDEP revelation. Distilling the bibliography, one converges to the following answers to key questions involved in this thorny issue: (1) Why? Both caregivers and patients consistently prefer to be informed. That promotes increased knowledge and vigilance, which, in turn, results in empowerment, effective collaboration, and improved self-management. 2,6 Even if SUDEP occurs, better understanding alleviates the feelings of shock, frustration, and guilt. 7 The presumed negative consequences of perpetually heightened anxiety for a rare entity, particularly in well-controlled cases, and of unnecessary, subsequent restriction of activities have not been substantiated. 5 -9 Even in such low-risk cases, a discussion provides reassurance to the family, medicolegal coverage to the provider, and forges a transparent therapeutic relationship between them 2 ; (2) When? Though the optimal counseling time is better determined on a case-by-case basis factoring patient and caregiver comfort level and the potential of mental and emotional saturation, earlier than delayed disclosure is recommended, as close to the time of diagnosis as possible 2,10 ; (3) Where? SUDEP discussions require time and concentrated attention. Hence, they should not occur in the emergency department or critical care setting, but should be ideally saved for an unhurried clinic appointment 10 ; (4) What? A disclosure should ideally include the knowns (epidemiology, predisposing, and protective factors, and how these can be modified accordingly) and the unknowns (mechanism, definitive prevention measures), balancing the message of risk with one of hope 7,10 ; (5) Who? All studies unanimously identify Neurologists as the most suitable providers to share this information, rather than general practitioners. 5 Logistical and psychological support can be further provided by nurses and social workers, where available. 7 Within the confines of cognitive capacity, emotional maturity, cultural idiosyncrasies, and parental consent, participation of older children in such discussions may be considered 8,10 ; (6) How? Candid, thoughtful and compassionate, face-to-face interactions between the family and the Neurologists consistently emerge as the preferred dissemination mode of SUDEP information. 10 That can be coupled with printed 6 or video 5 material and credible online resources by formal organizations or foundations, 2 with the option for follow-up in-person clarifications. 8
On the whole, the amalgamate of SUDEP counseling studies, including the one commented herein, in addition to professional societies and clinical practice guidelines, 1 highlight that patient and family education is critical and converge on its content and process. It behooves future investigations to assess its long-lasting impact on epilepsy management and on the psychosocial well-being of adult and pediatric PWE and their caregivers of various cultural and ethnic backgrounds, as well as on SUDEP rates themselves. In that regard, the effect of novel technologies (e.g., mobile devices) and newer health care models (e.g., telemedicine) should also be evaluated. While the scientific community is arduously working on elucidating the pathophysiology and prevention strategy of this dreaded eventuality, such longitudinal studies may inform practice and policy alike, and act as a catalyst to bridge the currently existing gap between the family-desired and the clinician-provided counseling.
