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Article: Does one size fit all? An update on chronic ventilatory support in different respiratory illnesses.

Carlucci, Annalisa / Patout, Maxime / Winck, João Carlos

2023 Volume 19, Issue 2, Page(s) 230046

Abstract: Home noninvasive ventilation (HNIV) improves outcomes in different disease categories. In this article, we discuss indications for when and how to initiate HNIV in COPD, obesity hypoventilation syndrome (OHS) and neuromuscular disorders (NMD). While in ... ...

Abstract	Home noninvasive ventilation (HNIV) improves outcomes in different disease categories. In this article, we discuss indications for when and how to initiate HNIV in COPD, obesity hypoventilation syndrome (OHS) and neuromuscular disorders (NMD). While in COPD, significant diurnal hypercapnia and high-intensity HNIV are essential ingredients for success, in NMD and OHS, early respiratory changes are best detected during sleep through oxy-capnography associated (or not) with respiratory polygraphy. In COPD and OHS, it is crucial to consider the coexistence of obstructive sleep apnoea because treatment with continuous positive airway pressure may be the simplest and most effective treatment that should be proposed even in hypercapnic patients as first-line therapy. In NMD, the need for continuous HNIV and eventual switching to tracheostomy ventilation makes this group's management more challenging. Achieving successful HNIV by improving quality of sleep, quality of life and keeping a good adherence to the therapy is a challenge, above all in COPD patients. In OHS patients, on top of HNIV, initiation of other interventions such as weight loss management is crucial. More resources should be invested in improving all these aspects. Telemonitoring represents a promising method to improve titration and follow-up of HNIV.
Language	English
Publishing date	2023-07-11
Publishing country	England
Document type	Journal Article ; Review
ZDB-ID	2562899-9
ISSN	2073-4735 ; 1810-6838
ISSN (online)	2073-4735
ISSN	1810-6838
DOI	10.1183/20734735.0046-2023
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Article ; Online: Higher baseline heart rate variability in CCHS patients with progestin-associated recovery of hypercapnic ventilatory response.

Sevoz-Couche, Caroline / Patout, Maxime / Charbit, Beny / Similowski, Thomas / Straus, Christian

Respiratory research

2024 Volume 25, Issue 1, Page(s) 87

Abstract: After a fortuitous observation of two cases of chemosensitivity recovery in women with congenital central hypoventilation syndrome (CCHS) who took desogestrel, we aimed to evaluate the ventilatory response to hypercapnia of five CCHS patients with or ... ...

Abstract	After a fortuitous observation of two cases of chemosensitivity recovery in women with congenital central hypoventilation syndrome (CCHS) who took desogestrel, we aimed to evaluate the ventilatory response to hypercapnia of five CCHS patients with or without treatment consisting of desogestrel (DESO) or levonorgestrel (LEVO). Only two patients became responsive to hypercapnia under treatment, according to their basal vagal heart rate variability. These results suggest that heart rate variability may be promising tool to discriminate patients susceptible to become responsive to hypercapnia under DESO-LEVO treatment.Clinical Trials Identifier NCT01243697.
MeSH term(s)	Humans ; Female ; Progestins/therapeutic use ; Hypercapnia/diagnosis ; Hypercapnia/drug therapy ; Desogestrel/therapeutic use ; Heart Rate ; Sleep Apnea, Central ; Hypoventilation/congenital ; Homeodomain Proteins/therapeutic use
Chemical Substances	Progestins ; Desogestrel (81K9V7M3A3) ; Homeodomain Proteins
Language	English
Publishing date	2024-02-09
Publishing country	England
Document type	Letter
ZDB-ID	2041675-1
ISSN	1465-993X ; 1465-993X
ISSN (online)	1465-993X
ISSN	1465-993X
DOI	10.1186/s12931-023-02625-w
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Article ; Online: Paradoxical breathing during sleep is associated with increased sleep apnea and reduced ventilatory capacities in high-level spinal cord injury.

Vivodtzev, Isabelle / Rong, Sophie / Ely, Matthew R / Patout, Maxime / Taylor, J Andrew

Journal of sleep research

2024 , Page(s) e14171

Abstract: Sleep-disordered breathing is highly prevalent in individuals with high-level spinal cord injury. In addition, chest mechanics are known to be altered, leading to paradoxical breathing. Here we investigated the interaction between paradoxical breathing ... ...

Abstract	Sleep-disordered breathing is highly prevalent in individuals with high-level spinal cord injury. In addition, chest mechanics are known to be altered, leading to paradoxical breathing. Here we investigated the interaction between paradoxical breathing and sleep quality in these patients, and its association with measurements of respiratory function, hypercapnic ventilatory response and peak exercise ventilation. Home-based polysomnography was performed in 13 patients with spinal cord injury (C4 to T4) untreated for sleep-disordered breathing. We defined paradoxical breathing as counterphase between thoracic and abdominal movements during slow-wave and rapid eye movement sleep. Sleep quality, pulmonary function, hypercapnic ventilatory responses and peak exercise ventilation were compared between those with and without paradoxical breathing. Half of individuals presented with nocturnal paradoxical breathing. Despite similar age, body mass index, injury level, time since injury, and respiratory function, those with paradoxical breathing had higher apnea-hypopnea index (13 ± 8 versus 5 ± 3 events per hr) and average sleep heart rate (67 ± 12 versus 54 ± 4 bpm; p < 0.05). Moreover, paradoxical breathing was associated with lower hypercapnic ventilatory response (slope: 0.35 ± 0.17 versus 0.96 ± 0.38) and lower peak exercise ventilation (33 ± 4 versus 48 ± 12 L min
Language	English
Publishing date	2024-02-14
Publishing country	England
Document type	Journal Article
ZDB-ID	1122722-9
ISSN	1365-2869 ; 0962-1105
ISSN (online)	1365-2869
ISSN	0962-1105
DOI	10.1111/jsr.14171
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Article: Diagnosing sleep disordered breathing in patients with chronic pulmonary disease: which test for which patient?

Grassion, Léo / Gonzalez-Bermejo, Jésus / Arnulf, Isabelle / Patout, Maxime

Breathe (Sheffield, England)

2023 Volume 19, Issue 1, Page(s) 220199

Abstract: Overnight polysomnography should be the first-line diagnostic test in patients with severe respiratory disease. However, if access to polysomnography is limited, overnight polygraphy can be used as an alternative first-line diagnostic. ...

Abstract	Overnight polysomnography should be the first-line diagnostic test in patients with severe respiratory disease. However, if access to polysomnography is limited, overnight polygraphy can be used as an alternative first-line diagnostic.
Language	English
Publishing date	2023-04-11
Publishing country	England
Document type	Journal Article
ZDB-ID	2562899-9
ISSN	2073-4735 ; 1810-6838
ISSN (online)	2073-4735
ISSN	1810-6838
DOI	10.1183/20734735.0199-2022
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Article ; Online: Effect of Facemask in Congenital Central Hypoventilation Syndrome.

Dudoignon, Benjamin / Patout, Maxime / Couque, Nathalie / Delclaux, Christophe / Bokov, Plamen

Respiration; international review of thoracic diseases

2023 Volume 102, Issue 12, Page(s) 991–994

Abstract: Introduction: Congenital central hypoventilation syndrome (CCHS) is a rare genetic disorder with a mutation in the PHOX2B gene. Patients need ventilatory support by noninvasive ventilation or tracheostomy to treat alveolar hypoventilation. Patients with ...

Abstract	Introduction: Congenital central hypoventilation syndrome (CCHS) is a rare genetic disorder with a mutation in the PHOX2B gene. Patients need ventilatory support by noninvasive ventilation or tracheostomy to treat alveolar hypoventilation. Patients with CCHS have a defect in chemosensitivity signal integration. Recently, due to the COVID-19 pandemic, the entire world has had to get used to wearing medical masks (MM). Objectives: The aim of the study was to evaluate the effect of an MM on gas exchange and to determine the role of central and peripheral chemoresponsiveness on the partial pressure of transcutaneous carbon dioxide (PtcCO2) in patients with CCHS wearing an MM. Methods: This study was based on the analysis of recordings obtained without and with an MM during hospitalization and was conducted to assess the impact of MM on PtcCO2 and SpO2 recordings with the SenTec Digital Monitor and their relationships with peripheral CO2 chemosensitivity obtained during tidal breathing measurement and with the hypercapnic hyperoxic ventilatory response. Results: Sixteen patients were included (13 boys) and were 10.2 (7.5; 18.5) years old. The use of an MM had a negative impact on gas exchange in patients with CCHS. The median PtcCO2 increased significantly. Peripheral chemosensitivity correlated with MM-induced PtcCO2 changes (R = -0.72, p = 0.005), but central chemosensitivity (the hypercapnic ventilator response slope) did not (R = -0.22, p = 0.510). Conclusion: The use of an MM had a negative impact on gas exchange in patients with CCHS.
MeSH term(s)	Male ; Humans ; Adolescent ; Hypoventilation/therapy ; Hypoventilation/congenital ; Masks ; Pandemics ; Sleep Apnea, Central/therapy ; Hypercapnia/therapy ; Homeodomain Proteins/genetics
Chemical Substances	Homeodomain Proteins
Language	English
Publishing date	2023-12-01
Publishing country	Switzerland
Document type	Journal Article
ZDB-ID	206674-9
ISSN	1423-0356 ; 0025-7931
ISSN (online)	1423-0356
ISSN	0025-7931
DOI	10.1159/000535127
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Article: Statement from the Early Career Member Committee (ECMC) Chair and Co-chair and introduction of the new ECMC members.

Ubags, Niki / Patout, Maxime / Dumas, Orianne / Cruz, Joana

Breathe (Sheffield, England)

2021 Volume 17, Issue 1, Page(s) 200281

Abstract: Statement from the Chair and Co-chair of the ERS Early Career Member Committee (ECMC) and introduction to the new members of this ... ...

Abstract	Statement from the Chair and Co-chair of the ERS Early Career Member Committee (ECMC) and introduction to the new members of this committee
Language	English
Publishing date	2021-05-08
Publishing country	England
Document type	Journal Article
ZDB-ID	2562899-9
ISSN	2073-4735 ; 1810-6838
ISSN (online)	2073-4735
ISSN	1810-6838
DOI	10.1183/20734735.0281-2020
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Article: Lessons from COVID-19 in the management of acute respiratory failure.

Crimi, Claudia / Murphy, Patrick / Patout, Maxime / Sayas, Javier / Winck, Joao Carlos

Breathe (Sheffield, England)

2023 Volume 19, Issue 1, Page(s) 230035

Abstract: Accumulated evidence supports the efficacy of noninvasive respiratory support therapies in coronavirus disease 2019 (COVID-19)-related acute hypoxaemic respiratory failure, alleviating admissions to intensive care units. Noninvasive respiratory support ... ...

Abstract	Accumulated evidence supports the efficacy of noninvasive respiratory support therapies in coronavirus disease 2019 (COVID-19)-related acute hypoxaemic respiratory failure, alleviating admissions to intensive care units. Noninvasive respiratory support strategies, including high-flow oxygen therapy, continuous positive airway pressure
Language	English
Publishing date	2023-05-23
Publishing country	England
Document type	Journal Article ; Review
ZDB-ID	2562899-9
ISSN	2073-4735 ; 1810-6838
ISSN (online)	2073-4735
ISSN	1810-6838
DOI	10.1183/20734735.0035-2023
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Article ; Online: Central CO

Bokov, Plamen / Dudoignon, Benjamin / Patout, Maxime / Matrot, Boris / Gallego, Jorge / Delclaux, Christophe

Journal of applied physiology (Bethesda, Md. : 1985)

2023 Volume 135, Issue 2, Page(s) 343–351

Abstract: Whether peripheral chemoreceptor response is altered in congenital central hypoventilation syndrome (CCHS) remains debated. Our aim was to prospectively evaluate both peripheral and central ... ...

Abstract	Whether peripheral chemoreceptor response is altered in congenital central hypoventilation syndrome (CCHS) remains debated. Our aim was to prospectively evaluate both peripheral and central CO
MeSH term(s)	Female ; Humans ; Carbon Dioxide ; Sleep Apnea, Central ; Hypoventilation/congenital ; Hypoventilation/genetics ; Respiration
Chemical Substances	Carbon Dioxide (142M471B3J)
Language	English
Publishing date	2023-06-30
Publishing country	United States
Document type	Journal Article
ZDB-ID	219139-8
ISSN	1522-1601 ; 0021-8987 ; 0161-7567 ; 8750-7587
ISSN (online)	1522-1601
ISSN	0021-8987 ; 0161-7567 ; 8750-7587
DOI	10.1152/japplphysiol.00182.2023
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Article ; Online: Reorganizing the respiratory high dependency unit for pandemics.

Masa, Juan Fernando / Patout, Maxime / Scala, Raffaele / Winck, Joao Carlos

Expert review of respiratory medicine

2021 Volume 15, Issue 12, Page(s) 1505–1515

Abstract: Introduction: Respiratory high dependency units (RHDUs) set up in European countries in the last decade are based on being a transitional step between the intensive care units (ICUs) and the conventional hospital ward in terms of staffing, level of ... ...

Abstract	Introduction: Respiratory high dependency units (RHDUs) set up in European countries in the last decade are based on being a transitional step between the intensive care units (ICUs) and the conventional hospital ward in terms of staffing, level of monitoring, and patients' severity. In the pre-COVID-19 era, its main use has been the treatment of hypercapnic acute-on-chronic respiratory failure with noninvasive respiratory support, and more recently, for hypoxemic acute respiratory failure. Areas covered: We searched the following databases: MEDLINE, EMBASE, Cochrane Central Register of Controlled Trials, limited to the terms: COVID-19 and RHDU, Respiratory Intermediate care Unit, acute respiratory distress syndrome (ARDS), noninvasive ventilation (NIV), high flow nasal cannula (HFNC), prone position, and monitoring. In this review, we summarize RHDU´s dual purpose: on the one hand, to decrease the number of admissions into ICU, and on the other hand, early discharges of patients from ICU with prolonged admissions due to the need of care or laborious weaning from invasive mechanical ventilation. Although this dual purpose of RHDUs has contributed to decrease the overload of the ICUs during the pandemic, the hundreds of patients admitted in hospitals, with approximately 20%-30% needing critical care, has exceeded the forecasts of many hospitals. Expert opinion: It seems clear that a reorganization and optimization of the care of patients with severe COVID-19 is necessary, minimizing admissions to the ICU and facilitating an early discharge. During the pandemic, several hospitals have spontaneously created new RHDUs or extended preexisting RHDUs or up-graded respiratory wards in order to receive less sick patients requiring lower levels of monitoring and nurse-to-patient ratios. This article reviews under a European expert perspective this topic and proposes an adaptation and optimization of the RHDUs to meet the emergent needs caused by the pandemic emphasizing the role of the expert application of noninvasive respiratory therapies in preventing intubation and ICU access.
MeSH term(s)	COVID-19 ; Humans ; Intensive Care Units ; Noninvasive Ventilation ; Oxygen Inhalation Therapy ; Pandemics ; Respiratory Insufficiency/diagnosis ; Respiratory Insufficiency/epidemiology ; Respiratory Insufficiency/therapy ; SARS-CoV-2
Language	English
Publishing date	2021-11-29
Publishing country	England
Document type	Journal Article ; Review
ZDB-ID	2479146-5
ISSN	1747-6356 ; 1747-6348
ISSN (online)	1747-6356
ISSN	1747-6348
DOI	10.1080/17476348.2021.1997596
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Article ; Online: Heart rate variability in congenital central hypoventilation syndrome: relationships with hypertension and sinus pauses.

Dudoignon, Benjamin / Denjoy, Isabelle / Patout, Maxime / Matrot, Boris / Gallego, Jorge / Bokov, Plamen / Delclaux, Christophe

Pediatric research

2022 Volume 93, Issue 4, Page(s) 1003–1009

Abstract: Background: Autonomic nervous system (ANS) dysregulation has been described in congenital central hypoventilation syndrome (CCHS). The objectives were to describe heart rate variability (HRV) analyses in children suffering from CCHS both while awake and ...

Abstract	Background: Autonomic nervous system (ANS) dysregulation has been described in congenital central hypoventilation syndrome (CCHS). The objectives were to describe heart rate variability (HRV) analyses in children suffering from CCHS both while awake and asleep and their relationships with both ambulatory blood pressure (BP) and ECG monitoring results. Methods: This retrospective study enrolled children with CCHS (n = 33, median age 8.4 years, 18 girls) who had BP and ECG monitored during the same 24 h. From the latter, HRV analyses were obtained during daytime and nighttime. Results: The prevalences of hypertension and sinus pauses were 33% (95% confidence interval [CI]: 18-52) and 18% (95% CI: 7-35), respectively. The decrease in systolic BP at night negatively correlated with an increase in very low frequency (VLF) and LF powers at night, and the longest RR interval positively correlated with daytime VLF and LF powers. Among the three groups of children (polyalanine repeat expansion mutation [PARM], moderate [20/25 and 20/26], severe [20/27 and 20/33], and non-PARMs), the prevalence of elevated BP or hypertension was different: in PARM subjects: 6/18 moderate, 7/9 severe versus 0/6 in non-PARM (p = 0.002). Conclusion: Modifications of cardiac ANS are associated with systemic hypertension and the occurrence of sinus pauses in CCHS. Impact: Children with congenital central hypoventilation syndrome (CCHS) exhibit an increased prevalence of hypertension and sinus pauses that are linked to cardiac autonomic nervous system dysfunction. Sinus pauses are the main manifestation of sinus nodal dysfunction in children with CCHS. The increased prevalence of hypertension, especially at nighttime, is a new finding in CCHS. Sinus nodal dysfunction can be due to the sole impairment of the cardiac autonomic nervous system. Ambulatory blood pressure and ECG monitoring are mandatory in patients with CCHS.
MeSH term(s)	Child ; Female ; Humans ; Homeodomain Proteins/genetics ; Transcription Factors/genetics ; Heart Rate ; Retrospective Studies ; Blood Pressure Monitoring, Ambulatory ; Sleep Apnea, Central/diagnosis ; Sleep Apnea, Central/epidemiology ; Hypertension/epidemiology
Chemical Substances	Homeodomain Proteins ; Transcription Factors
Language	English
Publishing date	2022-07-26
Publishing country	United States
Document type	Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	4411-8
ISSN	1530-0447 ; 0031-3998
ISSN (online)	1530-0447
ISSN	0031-3998
DOI	10.1038/s41390-022-02215-4
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