Home sleep studies in children with neurodisabilities: success rates and parental perception

Extract

The coronavirus disease 2019 (COVID-19) pandemic resulted in a shift in how sleep medicine was delivered. Many centres, including ours, pivoted to home studies during lockdown. Despite the subsequent reopening of sleep laboratories, many families requested to continue with home sleep studies. However, data on feasibility of home studies in children with complex comorbidities are lacking. Many centres only perform home studies on children with suspected sleep disordered breathing who are otherwise well, but emerging data indicate that they could represent a viable alternative to inpatient studies [1], including in children with comorbidities [2, 3].

Shareable abstract

Home sleep studies in children with neurodisabilities have a high success rate (85.4% in our cohort), particularly in patients with limited mobility, have the advantage of reducing the burden of hospital admissions and are the family preferred option https://bit.ly/46t8aWN

To the Editor:

The coronavirus disease 2019 (COVID-19) pandemic resulted in a shift in how sleep medicine was delivered. Many centres, including ours, pivoted to home studies during lockdown. Despite the subsequent reopening of sleep laboratories, many families requested to continue with home sleep studies. However, data on feasibility of home studies in children with complex comorbidities are lacking. Many centres only perform home studies on children with suspected sleep disordered breathing who are otherwise well, but emerging data indicate that they could represent a viable alternative to inpatient studies [1], including in children with comorbidities [2, 3].

We report the success rates of home sleep studies in children with suspected sleep disordered breathing and neurological comorbidities and parental views on their experience of such studies.

We retrospectively collected information on 48 home studies done on 48 patients between July 2021 and November 2022: eight oxicapnographies (oxygen (O₂)/carbon dioxide (CO₂)) and 40 cardiorespiratory sleep studies (CRSS); all but four CRSS included measurement of transcutaneous CO₂. If patients had more than one home study in this period, only the first one was considered. The information collected included age of the child, diagnosis, indication, technical details and whether changes were made based on the home study. In a subset of patients (n=22), parental telephonic feedback was obtained.

The equipment for CRSS (Somnotouch; Somnomedics, Randersacker, Germany) consisted of chest and abdominal respiratory inductance plethysmography bands, nasal cannula, oxygen saturation probe and transcutaneous CO₂ sensor; for oxicapnography, oxygen saturation probe (Masimo, Irvine, CA, USA) and transcutaneous CO₂ sensor (TCM5; Radiometer, Broønshøj, Denmark). Parents either collected the equipment from the hospital and were shown how to attach it, or the equipment was couriered to their home and parents were contacted by the respiratory physiologist on the same day to go through the setup and identify any issues. All parents received written instructions, including a video link for further information.

Sleep studies were scored using American Academy of Sleep Medicine criteria (version 2.6) [4].

Statistical analysis was done using the Fisher exact test calculator (MedCalc).

The indications for home study were routine monitoring (n=28, 58.3%), change in clinical condition (n=7, 14.6%), following change in ventilator settings (n=2, 4.2%) and other (n=11, 22.9%). The children had no respiratory support (n=25, 52%), or used bilevel positive airway pressure (BIPAP) (n=21, 44%), continuous positive airway pressure (CPAP) (n=1) or split BIPAP/no support (n=1). Seven (14.5%) patients were tracheostomy ventilated.

Studies were defined as technically successful if ≥4 h of artefact-free sleep data were obtained. 85.4% (41 out of 48) studies were successful, compared with 91% (312 out of 343) home studies in children without comorbidities (CRSS without CO₂) done in our unit between July 2021 and November 2022.

Diagnostic groups were divided into neuromuscular conditions (e.g. spinal muscular atrophy, muscular dystrophy, myopathy), cerebral palsy and other neurological conditions (e.g. maple syrup urine disease with profound central hypotonia and dystonia, Haddad syndrome and epilepsy, Tay–Sachs disease with developmental regression and epilepsy, muscle eye brain disease, epilepsy and ventriculoperitoneal shunt, EEF1A2 gene defect with microcephaly, hypotonia and epilepsy, chromosomal abnormality with developmental delay and epilepsy). In children with neuromuscular conditions and cerebral palsy, success rates were 94.1% and 92.3%, respectively. Overall, in patients with limited mobility (neuromuscular conditions and cerebral palsy), the success rate of home studies tended to be higher than in the group with other neurological conditions (p=0.08).

21 patients had successful home studies on CPAP/BIPAP which allowed settings to be changed in five cases (inspiratory positive airway pressure (IPAP), back-up rate or more than one change) and the mode of ventilation to be changed in two cases (figure 1).

FIGURE 1.

Home sleep studies in patients with neurological comorbidity. CRSS: cardiorespiratory sleep studies; CO₂: carbon dioxide; O₂: oxygen.

23 patients subsequently underwent further inpatient sleep studies (only the first inpatient study was considered) and the success rate was 78.2% (n=18: one O₂/CO₂, 17 CRSS). Five studies were <4 h because of wakefulness. In our cohort, the success rate of inpatient studies (18 out of 23) versus home studies (41 out of 48) was not statistically significant (p=0.50), but it was lower than our overall 96% (872 out of 909) success rate of inpatient sleep studies (p=0.01).

We contacted 22 parents who verbally consented to answer a nine-item questionnaire regarding the equipment for home studies (who attached it and time taken, instructions, issues), preference for sleep studies (at home/hospital and why; if influenced by the COVID-19 pandemic), experience of previous home studies and any recommendations. The median time from home study to telephone interview was 17 weeks.

20 parents set up the equipment themselves (five O₂/CO₂, 15 CRSS) on average in 20 min (range 2–40 min) and it was “very easy” (n=4, 20%), “easy” (n=13, 65%), “neither easy nor difficult” (n=2, 10%), “difficult” (n=1, 5%); none found setting up the equipment to be “very difficult”. The parent who found setting up the equipment “difficult”, had no prior experience of a home study, but would prefer home studies in the future.

17 (77%) responders would prefer future studies to be done at home, most commonly because the child sleeps better at home (n=12), but other reasons were: carer available to monitor the child overnight (n=5), no need to arrange transport for inpatient study (n=3), take time off work to accompany the child to hospital (n=3) or to arrange child care for sibling (n=3), or other reason (n=6). Three parents did not have any preferences and only two preferred inpatient studies “to make sure the sleep study is done properly” or “because in hospital there is staff who knows what to do if there are any issues and you also get the result of the study quicker, on the following day”. Only two parents would not have considered a home study before the COVID-19 pandemic.

Home sleep studies in patients with neurodisabilities have a high success rate (85.4% in our cohort), in particular in children with limited mobility, and can be used for routine monitoring and to guide changes in ventilation. A limitation of our study is the lack of validation compared to the gold standard represented by polysomnography [5]. 85% of parents found setting up the equipment to be “very easy” or “easy” and 77% would prefer home studies, despite the extra care commitment in a group of children with already a big care burden.

Home studies represent a valid alternative to inpatient studies, have the advantage of reducing the burden of hospital admissions, represent the family preferred option and may be more representative of a typical night's sleep for the child. If the study fails, the option of a hospital study still exists.