RT Journal Article SR Electronic T1 Short-term cognitive loading deteriorates breathing pattern and gas exchange in adult patients with congenital central hypoventilation syndrome JF ERJ Open Research JO erjor FD European Respiratory Society SP 00408-2022 DO 10.1183/23120541.00408-2022 VO 9 IS 2 A1 Jessica Taytard A1 Marie-Cécile Niérat A1 Camille Gand A1 Sophie Lavault A1 Capucine Morélot-Panzini A1 Maxime Patout A1 Laure Serresse A1 Nicolas Wattiez A1 Laurence Bodineau A1 Christian Straus A1 Thomas Similowski YR 2023 UL http://openres.ersjournals.com/content/9/2/00408-2022.abstract AB Question Human PHOX2B mutations result in life-threatening sleep-related hypoventilation (congenital central hypoventilation syndrome, CCHS). Most patients retain ventilatory activity when awake through a respiratory-related cortical network. We hypothesised that this need to mobilise cortical resources to breathe would lead to breathing-cognition interferences during cognitive loading.Patients and methods Seven adult CCHS patients (five women; median age 21) performed standard neuropsychological tests (paced auditory serial addition test – calculation capacity, working memory, sustained and divided attention; trail making test – visuospatial exploration capacity, cognitive processing speed, attentional flexibility; Corsi block-tapping test – visuospatial memory, short-term memory, working memory) during unassisted breathing and under ventilatory support. Ventilatory variables and transcutaneous haemoglobin oxygen saturation were recorded. Cortical connectivity changes between unassisted breathing and ventilatory support were assessed using electroencephalographic recordings (EEG).Results Baseline performances were lower than expected in individuals of this age. During unassisted breathing, cognitive loading coincided with increased breathing variability, and decreases in oxygen saturation inversely correlated with an increasing number of apnoeic cycles per minute (rho −0.46, 95% CI −0.76 to −0.06, p=0.01). During ventilatory support, cognitive tasks did not disrupt breathing pattern and were not associated with decreased oxygen saturation. Ventilatory support was associated with changes in EEG cortical connectivity but not with improved test performances.Conclusions Acute cognitive loads induce oxygen desaturation in adult CCHS patients during unassisted breathing, but not under ventilatory support. This justifies considering the use of ventilatory support during mental tasks in CCHS patients to avoid repeated episodes of hypoxia.In adult CCHS patients, acute cognitive loads disorganise breathing and induce oxygen desaturation during unassisted breathing but not ventilatory support. Ventilatory support should be considered in this setting, to avoid repeated hypoxia. https://bit.ly/3Mlp0yH