Early Findings on the Effectiveness of Novel Awakening Stimuli for Patients with Congenital Central Hypoventilation Syndrome

Congenital Central Hypoventilation Syndrome (CCHS) is a rare disorder that impairs autonomic breathing control, leading to alveolar hypoventilation and sometimes to central apnoea, predominantly during sleep. Patients typically require nocturnal ventilatory support and alarms to prevent life-threate...

Full description

Saved in:
Bibliographic Details
Main Authors: Silvia Rapella, Caterina Piazza, Francesco Morandi, Alessandro Carcano, Cinzia Arzilli, Niccolò Nassi, Igor Catalano, Francesca Formica, Emilia Biffi
Format: Article
Language:English
Published: MDPI AG 2025-03-01
Series:Sensors
Subjects:
Online Access:https://www.mdpi.com/1424-8220/25/6/1759
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1850280038895714304
author Silvia Rapella
Caterina Piazza
Francesco Morandi
Alessandro Carcano
Cinzia Arzilli
Niccolò Nassi
Igor Catalano
Francesca Formica
Emilia Biffi
author_facet Silvia Rapella
Caterina Piazza
Francesco Morandi
Alessandro Carcano
Cinzia Arzilli
Niccolò Nassi
Igor Catalano
Francesca Formica
Emilia Biffi
author_sort Silvia Rapella
collection DOAJ
description Congenital Central Hypoventilation Syndrome (CCHS) is a rare disorder that impairs autonomic breathing control, leading to alveolar hypoventilation and sometimes to central apnoea, predominantly during sleep. Patients typically require nocturnal ventilatory support and alarms to prevent life-threatening desaturation events. However, current alarm systems integrated into pulse oximeters do not provide adequate assistance at home. To address these limitations, we developed an assistive device with customizable multisensory stimulation that activates based on the severity and duration of desaturation episodes. In a multicenter clinical trial involving 4 children and 11 young adults with CCHS, we assessed the device’s effectiveness and the role of arousals over three nights: one baseline and two test nights. The results showed that the device significantly improved awakening rates and enabled faster recovery from desaturations in young adults. However, no such improvements were observed in children compared to the baseline. Arousal events and sleep efficiency were unaffected by the device in both groups. These findings suggest that the device can enhance the safety and autonomy of young adults with CCHS but may be more effective in alerting caregivers in pediatric cases than directly waking children. Further studies are needed to refine its application across different age groups, given the limited sample size.
format Article
id doaj-art-c04a738fb9814cc48d47c629b4b70a0e
institution OA Journals
issn 1424-8220
language English
publishDate 2025-03-01
publisher MDPI AG
record_format Article
series Sensors
spelling doaj-art-c04a738fb9814cc48d47c629b4b70a0e2025-08-20T01:48:54ZengMDPI AGSensors1424-82202025-03-01256175910.3390/s25061759Early Findings on the Effectiveness of Novel Awakening Stimuli for Patients with Congenital Central Hypoventilation SyndromeSilvia Rapella0Caterina Piazza1Francesco Morandi2Alessandro Carcano3Cinzia Arzilli4Niccolò Nassi5Igor Catalano6Francesca Formica7Emilia Biffi8Scientific Institute, IRCCS Eugenio Medea, 23842 Bosisio Parini, LC, ItalyScientific Institute, IRCCS Eugenio Medea, 23842 Bosisio Parini, LC, ItalyItalian Association for Congenital Central Hypoventilation Syndrome (AISICC), 50126 Florence, FI, ItalyItalian Association for Congenital Central Hypoventilation Syndrome (AISICC), 50126 Florence, FI, ItalyMeyer Children’s Hospital IRCCS, 50139 Florence, FI, ItalyMeyer Children’s Hospital IRCCS, 50139 Florence, FI, ItalyPediatric Palliative Care, Pain Service, Department of Women’s and Children’s Health, University of Padua, 35128 Padua, PD, ItalyScientific Institute, IRCCS Eugenio Medea, 23842 Bosisio Parini, LC, ItalyScientific Institute, IRCCS Eugenio Medea, 23842 Bosisio Parini, LC, ItalyCongenital Central Hypoventilation Syndrome (CCHS) is a rare disorder that impairs autonomic breathing control, leading to alveolar hypoventilation and sometimes to central apnoea, predominantly during sleep. Patients typically require nocturnal ventilatory support and alarms to prevent life-threatening desaturation events. However, current alarm systems integrated into pulse oximeters do not provide adequate assistance at home. To address these limitations, we developed an assistive device with customizable multisensory stimulation that activates based on the severity and duration of desaturation episodes. In a multicenter clinical trial involving 4 children and 11 young adults with CCHS, we assessed the device’s effectiveness and the role of arousals over three nights: one baseline and two test nights. The results showed that the device significantly improved awakening rates and enabled faster recovery from desaturations in young adults. However, no such improvements were observed in children compared to the baseline. Arousal events and sleep efficiency were unaffected by the device in both groups. These findings suggest that the device can enhance the safety and autonomy of young adults with CCHS but may be more effective in alerting caregivers in pediatric cases than directly waking children. Further studies are needed to refine its application across different age groups, given the limited sample size.https://www.mdpi.com/1424-8220/25/6/1759congenital central hypoventilation syndromeassistive devicedesaturation episodesarousal
spellingShingle Silvia Rapella
Caterina Piazza
Francesco Morandi
Alessandro Carcano
Cinzia Arzilli
Niccolò Nassi
Igor Catalano
Francesca Formica
Emilia Biffi
Early Findings on the Effectiveness of Novel Awakening Stimuli for Patients with Congenital Central Hypoventilation Syndrome
Sensors
congenital central hypoventilation syndrome
assistive device
desaturation episodes
arousal
title Early Findings on the Effectiveness of Novel Awakening Stimuli for Patients with Congenital Central Hypoventilation Syndrome
title_full Early Findings on the Effectiveness of Novel Awakening Stimuli for Patients with Congenital Central Hypoventilation Syndrome
title_fullStr Early Findings on the Effectiveness of Novel Awakening Stimuli for Patients with Congenital Central Hypoventilation Syndrome
title_full_unstemmed Early Findings on the Effectiveness of Novel Awakening Stimuli for Patients with Congenital Central Hypoventilation Syndrome
title_short Early Findings on the Effectiveness of Novel Awakening Stimuli for Patients with Congenital Central Hypoventilation Syndrome
title_sort early findings on the effectiveness of novel awakening stimuli for patients with congenital central hypoventilation syndrome
topic congenital central hypoventilation syndrome
assistive device
desaturation episodes
arousal
url https://www.mdpi.com/1424-8220/25/6/1759
work_keys_str_mv AT silviarapella earlyfindingsontheeffectivenessofnovelawakeningstimuliforpatientswithcongenitalcentralhypoventilationsyndrome
AT caterinapiazza earlyfindingsontheeffectivenessofnovelawakeningstimuliforpatientswithcongenitalcentralhypoventilationsyndrome
AT francescomorandi earlyfindingsontheeffectivenessofnovelawakeningstimuliforpatientswithcongenitalcentralhypoventilationsyndrome
AT alessandrocarcano earlyfindingsontheeffectivenessofnovelawakeningstimuliforpatientswithcongenitalcentralhypoventilationsyndrome
AT cinziaarzilli earlyfindingsontheeffectivenessofnovelawakeningstimuliforpatientswithcongenitalcentralhypoventilationsyndrome
AT niccolonassi earlyfindingsontheeffectivenessofnovelawakeningstimuliforpatientswithcongenitalcentralhypoventilationsyndrome
AT igorcatalano earlyfindingsontheeffectivenessofnovelawakeningstimuliforpatientswithcongenitalcentralhypoventilationsyndrome
AT francescaformica earlyfindingsontheeffectivenessofnovelawakeningstimuliforpatientswithcongenitalcentralhypoventilationsyndrome
AT emiliabiffi earlyfindingsontheeffectivenessofnovelawakeningstimuliforpatientswithcongenitalcentralhypoventilationsyndrome