This is the first study to assess the parent-reported experience of in-laboratory PSG specifically in children with NDD and highlights the challenges for this population. Several key findings have important clinical implications.

Firstly, in keeping with reports from others who examined the experiences of children (although not specifically those with NDD) undergoing PSG [13], our study found more than a third of parents/carers felt that their child’s sleep on the night of the PSG was not typical of sleep in their home environment. This may be due to an increased propensity for sleep fragmentation and night waking in younger children [14] which parents may become aware of on the night of the PSG when they are required to sleep in the same room as the child, as well as due to the ‘first-night effect’, a well-documented phenomenon of poorer sleep experienced on the first night in a new location, observed in both children and adults [15]. Atypical sleep during a diagnostic test suggests that PSG may not accurately capture usual sleep architecture and sleep stages, which could lead to underestimating the severity of SDB and suboptimal treatment decisions. A retrospective study of 200 participants undertaken over a decade ago found that more than a third of respondents felt their child’s breathing on the night of the PSG did not reflect that usually seen at home [16]. In-home testing is likely to result in more typical patterns of sleep, given the usual sleep environment, particularly for children with NDD in whom small changes in routine may be difficult.

Secondly, we found that prior experience of PSG resulted in reduced worry amongst children and their parents. The higher reported parent and child worry regarding first-time PSG demonstrated in our study is similar to patterns of greater reported anxiety during other first-time procedures such as radiological imaging [17] and surgeries [18]. A common thread across these studies is reference to desensitisation protocols as an avenue for reducing anxiety in participants. Research with a particular focus on PSG desensitisation has demonstrated that after an average of approximately four sessions involving home practice and a tour of the sleep laboratory, 19 out of 23 child participants successfully completed PSG without sedation [19]. PSG desensitisation strategies may therefore be of benefit to some children. Evaluating differing PSG desensitisation approaches for children with NDD is an important area for future research.

A previously unreported finding from our study is the higher reported parent and child worry scores and increased likelihood of atypical sleep observed in the sub-populations of children with ASD and/or neuromuscular disease. We speculate that reasons for this could include children with ASD having propensity for sensory sensitivities being triggered by PSG monitoring leads, comorbid anxiety disorders, difficulty tolerating changes to routines, and children with neuromuscular weakness having decreased ability to co-ordinate motor withdrawal from noxious stimuli, perhaps increasing fear of PSG-related procedures [20, 21]. It should also be noted that there is a well-recognised overlap between neuromuscular disease and ASD – particularly in children with Duchenne Muscular Dystrophy [22]. In some cases, worry expressed by parents and children may point to a feedback loop where the discomfort of the child increases the anxiety of the parent and vice versa, thereby making experiences for both worse overall [23]. Limited desensitisation sessions alone may not be sufficient to address this problem, although extensive parental education and emotional support could assist in breaking this cycle.

A final novel finding from our study is the clear preference reported by parents for an in-home mat-based diagnostic option compared to only 7% of families favouring the in-laboratory evaluation. We speculate that this is likely motivated by the distress and anxiety brought about by additional hospital visits, the burden of care associated with transporting children with NDD, and the impact of a new test/medical environment. It should be noted the questionnaire specifically enquired about home sleep mat-based testing, rather than other more traditional forms of home sleep apnoea tests (HSATs), such as Level 2 HSATs. Important future work should explore in greater detail the factors driving parental preference for home-based testing, with qualitative studies providing the option to examine families’ experiences and attitudes towards in-laboratory and in-home testing of various types.

This study has a number of limitations. Firstly, while we assessed the experiences of parents and other caregivers, we did not evaluate the experiences of children directly. Whilst some children with NDD may be too young or unable to effectively communicate their feelings regarding PSG experience, many children with neurodevelopmental conditions may be able to reliably provide feedback, and evaluating this in future research is strongly recommended. Second, the questionnaires’ PSG equipment set-up and overnight tolerance report was intrinsically subjective because it relied on parent report rather than objective assessment. By accompanying their child for the study and sleeping in the same room, parents may become more acutely aware of any sleep disturbances, potentially heightening the degree of perceived distress/worsened sleep quality compared to what might be perceived in the home environment. Previous research has assessed PSG set-up and monitoring lead tolerance in children with NDD more objectively [8]. These reports may or may not align with parental perception; as an example, caregiver reports of sleep in children with ASD frequently differed from actigraphy data [24]. Caregiver reports are subject to recall bias. A further limitation is the potential variability in the PSG setup across sites. Minor differences in the way technicians demonstrated or used the equipment, the amount of support provided, or environmental factors such as the arrangement, and room ambience could impact the child’s experience and tolerance. Additionally, although we aimed to examine PSG experiences from three sites, the number of patients from QCH (72% of participants), was disproportionately large, making it difficult to draw conclusions about differences between locations. A final limitation is the lack of a direct comparison group of neurotypical children.