“Emplotted Narratives” and Structured “Behavioral Observations” Supporting the Diagnosis of Willis‐Ekbom Disease/Restless Legs Syndrome in Children with Neurodevelopmental Conditions

Abstract

SummaryBackgroundWillis‐Ekbom disease/restless legs syndrome (WED/RLS) seems to be a frequent cause of intractable chronic insomnia (ICI) but is under‐recognized in children/adolescents with neurodevelopmental conditions (NDCs), as many patients do not have the ability to express the underlying “urge‐to‐move”. In light of this, we aim to develop a protocol for behavioral observations supporting the diagnosis of WED/RLS.MethodsWe investigated 26 pediatric patients (age 1–16 years, median 8) with NDCs, ICI and evidence of familial WED/RLS employing (1) “emplotted narratives” for description of the various “urge‐to‐move” presentations and (2) self‐description and “behavioral observations” during a “suggested clinical immobilization test” (SCIT).ResultsParental narratives reflected typical WED/RLS‐related “urge‐to‐move” symptoms during day‐, bed‐, and nighttime in all patients. Fifteen out of 26 patients could describe the “urge‐to‐move” during the SCIT. Ten out of 26 patients, unable to describe their symptoms due to cognitive disabilities, showed patterns of “relieving‐movements” upon observation. Sensory processing abnormalities were reported in all patients, with tactile sensitivities (26/26) (including shifted pain threshold) as the most common sensory domain.Conclusion“Emplotted narratives” and structured “behavioral observations” support recognition of familial WED/RLS associated movement patterns and provide a useful tool for the diagnosis of WED/RLS in children with NDCs in a clinical office setting.

Prenatal alcohol exposure and sleep-wake behaviors: exploratory and naturalistic observations in the clinical setting and in an animal model

“…We analyzed retrospectively data from assessments performed between 2011 and 2014 at the Sleep/Wake-Behavior Clinic (British Columbia Children's Hospital, the only quaternary care center in British Columbia), on 40 pediatric patients with major sleep problems who were referred by community-based practitioners after unsuccessful treatment attempts. The novelty of the structured assessment methodology as a clinical standard of care, utilizing the naturalistic listening, observing, exploring and describing methodology, with an assessor and trainee observer, has been explained in detail previously [13,14,29,31,68]. The clinical data were extracted from our database, which has been developed under UBC REB#: H15-00323 study (a retrospective chart study for patients seen at the Sleep-Wake Behavior Clinic between 2008 and 2015, based on the documentation of assessments).…”

“…To capture the coexistence of disruptive sleep and wake behaviors in children and adolescents with neurodevelopmental conditions (NDCs) [13], we apply methods that allow for observation, exploration and description of naturalistic real-life situations [29]: emplotted narratives [13], structured daytime observations [30,31], and structured, videobased nighttime observations in the home setting [29,[32][33][34]. In this context, emplotted is defined as working collaboratively with parents and caregivers (via exploration and negotiation of symptoms) to describe challenging/ disruptive behaviors and then sharing the final summary for quality control.…”

“…In this context, emplotted is defined as working collaboratively with parents and caregivers (via exploration and negotiation of symptoms) to describe challenging/ disruptive behaviors and then sharing the final summary for quality control. This approach facilitates self-description and observation of nighttime symptoms, which are otherwise not unveiled in time-constricted clinic visits, thus increasing our understanding of the interrelation between day and nighttime symptoms [31]. Notably, this methodology allowed us to capture the high prevalence of probable restless legs syndrome (RLS) in children with NDCs, including FASD [13,30,31].…”

“…This approach facilitates self-description and observation of nighttime symptoms, which are otherwise not unveiled in time-constricted clinic visits, thus increasing our understanding of the interrelation between day and nighttime symptoms [31]. Notably, this methodology allowed us to capture the high prevalence of probable restless legs syndrome (RLS) in children with NDCs, including FASD [13,30,31]. RLS is a diagnosis based on the patient's verbal description of subjective symptoms, such as discomfort and urge to move in a resting state [33,35].…”

“…To achieve our aim of capturing presentations of disruptive sleep-/wake-behaviors in reallife, natural settings, we applied a structured behavioral observation protocol (ie, naturalistic observations) in the clinical setting [31,34], and a similar behavioral observation protocol in our well-established PAE animal model in the home-cage environment [67]. This collaborative work investigates dysregulated sleep-wake behaviors and compares outcomes from the experimental and clinical studies utilizing the naturalistic observational methodology.…”