Received: 15 April 2018 | Accepted: 5 July 2018

DOI: 10.1002/ppul.24138

ORIGINAL ARTICLE: SLEEP & BREATHING

Outpatient initiation of long-term continuous positive airway

pressure in children

Alessandro Amaddeo MD, PhD1,2,3 | Annick Frapin MSN1 | Samira Touil BSc1 |
Sonia Khirani PhD1,4 | Lucie Griffon MD1 | Brigitte Fauroux MD, PhD1,2,3

1 PediatricNoninvasive Ventilation and Sleep

Unit, AP-HP, Hôpital Necker-Enfants Malades, Abstract
Paris, France
Introduction: Current guidelines recommend initiating continuous positive airway
2 Paris Descartes University, Paris, France
pressure (CPAP) in children during an overnight in-hospital titration study. Due to a
3 INSERM U 955, Team 13, Créteil, France
4 ASV Santé, Gennevilliers, France
shortage of hospital beds and economic constraints, we started a program for
outpatient initiation of CPAP in selected children with obstructive sleep apnea (OSA).
Correspondence
Methods: Objective CPAP compliance and efficacy were evaluated in consecutive
Alessandro Amaddeo, Pediatric noninvasive
ventilation and sleep unit, AP-HP, Hôpital children enrolled in an outpatient CPAP program when they fulfilled the following
Necker Enfants Malades, 149 rue de Sèvres,
criteria: persistent OSA, age >6 months, stable condition, family living in the Parisian
Paris, 75015 France.
Email: alessandro.amaddeo@aphp.fr area and agreeing with a regular follow-up.
Results: Thirty-one children, median (range) age 8.9 years (0.8-17.5), were included in the
program. The most common diagnosis were Down syndrome (n = 7), achondroplasia
(n = 3), and obesity (n = 3). Median baseline obstructive apnea-hypopnea index (OAHI) was
12.5 events/h (5-100). Median duration of follow-up was 12.3 months (2.2-25.2). Four
subjects (three adolescents with Down syndrome) were not compliant at 2-month follow-
up with a compliance <4 h/night. In the other 27 subjects, median compliance was
08:21 h:min/night (05:45-12:20), with a median number of night use per month of 25 (18-
30). CPAP efficacy at the end of follow-up was excellent in the compliant subjects with a
median AHI of two events/h (0-4) and normal gas exchange with CPAP therapy. Three
subjects were successfully weaned from CPAP treatment during the study period.
Conclusion: Initiating CPAP in an outpatient setting in children is feasible and effective in
selected subjects. A high rate of compliance can be achieved as well as a correction of OSA.

KEYWORDS
children, continuous positive airway pressure (CPAP), obstructive sleep apnea OSA,
outpatient

Abbreviations: APAP, autotitrating positive airway pressure; CPAP, continuous positive
airway pressure; OAHI, obstructive apnea-hypopnea index; OSA, obstructive sleep apnea;
PG, polygraphy; PSG, polysomnography; PtcCO2, transcutaneous carbon dioxide; SpO2,
pulse oximetry.
All authors have seen and approved the manuscript
1 | I NTRODU C TI ON
Continuous positive airway pressure (CPAP) is increasingly used in
children with persistent obstructive sleep apnea (OSA) despite optimal
medical and/or surgical management.1–3 CPAP has proven its efficacy
for treating OSA in children with various underlying disorders such as

Pediatric Pulmonology. 2018;1–7. wileyonlinelibrary.com/journal/ppul © 2018 Wiley Periodicals, Inc. | 1

2 |
Down syndrome, congenital bone diseases, metabolic diseases, Prader
1,4–8
Willi syndrome, and craniofacial malformations.
Current guidelines recommend CPAP initiation at an accredited
facility with an overnight supervised polysomnography (PSG) titration
study.9 The decision on the setting where to start CPAP varies largely
according to local practice. Outpatient programs that involve home
acclimation to the interface and the device, followed by an in-hospital
titration PSG have been developed in the USA10–12 and in Australia.13
Some experience has been reported on the initiation of mechanical
ventilation in an outpatient or home setting in adult patients, but
experience in children is scarce.10,12–15 In Europe, in-hospital CPAP
initiation represents the standard practice for children.6,16 However,
this approach is not always feasible because of shortage of hospital
beds, limited access to PSG, and economic constraints. Moreover,
repeated hospitalizations may be stressful for children with chronic
conditions who spend already a large amount of time in hospital.
Hospitalizations are associated with school and parent's work loss, and
a risk of nosocomial infection, particularly during the winter season. In
addition, with regard to comfort and quality of life, patients and
families inevitably prefer outpatient versus inpatient care.17
Our standard inpatient CPAP initiation program consists of a 2-3
days hospitalization. During the first day, the indication and rationale
of CPAP treatment are explained to the patient and the family by the
CPAP team which comprises of a pediatrician and a nurse having an
expertise in CPAP. This is followed by two to three CPAP short trials
with progressively increasing CPAP pressures while the patient is
awake, after careful choice of an appropriate interface. The patient and
the parents are educated to put on and take off the mask and CPAP
device. The patient is then encouraged to sleep with CPAP during the
first night. If the patient is able to sleep with CPAP >3 h, a respiratory
polygraphy (PG) is performed during the second night in order to check
the CPAP pressure. Mask leaks and other potential problems are
discussed with the patient and the parents. The patient may stay
during a third night in case of CPAP duration <3 h. This program has
been shown to be very efficient in terms of compliance18 and
19
correction of respiratory events. However, dramatic improvements
in the technology of home CPAP devices have been made over the last
decade. The built-in software of CPAP devices provides now a large
20
amount of data regarding treatment compliance and efficacy and
manual analysis of the built-in data coupled with a pulse oximetry
(SpO2) recording may be used as a PG.21 This led us to change our
practice with the development of an outpatient CPAP initiation
program.
The aim of the present study was to evaluate the objective
compliance and efficacy of CPAP initiated through an outpatient CPAP
program in selected children with OSA.
2 | M E TH O D S
2.1 | Selection and description of participants
The study was conducted between March 2015 and January 2017 at
the noninvasive ventilation (NIV) and sleep unit of Necker Children's
AMADDEO ET AL.
Hospital. All consecutive children who underwent a PSG or PG in our
sleep laboratory and who presented persistent OSA defined as an
obstructive apnea-hypopnea index (OAHI) >5 events/h despite
optimal surgical and/or medical treatment, were included in an
outpatient CPAP initiation program if they fulfilled the following
inclusion criteria: 1) stable medical condition; 2) age>6 month of age; 3)
French-speaking family living in the Parisian area and agreeing with
regular follow-up visits. Written informed consent was obtained
before enrolment from patient's parents or from the patient's legal
guardian. The protocol was approved by the local ethical committee
(CPP Ile de France II, n° 2014-03-09 SC).
2.2 | Outpatient initiation program
The program consists of an individual approximately 2-h outpatient
visit. During the first 30 min, the results of the sleep study and the
principles of CPAP treatment are explained to the patient (if age >2-3
years) and the parents by a pediatric pulmonologist and a nurse
specialized in NIV and therapeutic education, by means of specific age-
adapted educational tools. These tools consisted of educational boards
and cartoons, a booklet explaining CPAP, and a teddy bear breathing
with a CPAP device (Figures 2 and 3).
The choice of the device was made according to manufacturers’
recommendations, that is, based on a minimal weight.22–25 This choice
was made in order to avoid an underestimation of objective
compliance due to a too low flow rate in young children, as previously
reported by our group.18 Heated humidification was systematically
prescribed. The most appropriate and comfortable interface according
to patient's age, facial morphology and preference was chosen.
The interface was then tried first without the CPAP device. The
parents (and the patient if possible) were trained to put on and take off
the interface. CPAP was then started at a minimal pressure of
4 cmH2O, which was progressively increased to the highest tolerated
pressure level, while the patient was lying down calmly, according to
our previous published experience.26 The patient was then asked to
keep the CPAP with the chosen pressure for at least 30 min. According
to manufacturers’ indications, auto-titrating positive airway pressure
(APAP) was only used in children weighing more than 30 kg.24,25
FIGURE 1 AHI before and after treatment. Apnea Hypopnea
Index = AHI (events/h)
AMADDEO ET AL.
FIGURE 2 Teddy bear breathing with a CPAP device
Concerning APAP, the initiation session was started with a fixed CPAP
and then, after 10 min, switched to the final pressure range with a delta
of at least 4 cmH2O between minimal and maximal pressure. During that
period, relaxation or distraction was offered with the patient's favorite
nursery rhymes, music, or calm videos, according to the age and
preference of the patient. Afterwards, the nurse reviewed the use and
maintenance of the interface and CPAP device with the parents. An
information and follow-up booklet on CPAP was explained and given to
the parents. The parents were asked to contact the NIV unit by phone or
email after 1 week and earlier if they encountered any problem at home.
A home care provider trained in pediatric NIV performed a first
scheduled visit at home within the 3 days following the CPAP initiation
and then on a weekly basis during the first month. During these visits,
objective compliance, efficacy and comfort were assessed as well as the
correct use and maintenance of the equipment. The data recorded on the
built-in software of the CPAP device was systematically downloaded and
transmitted to the hospital team. An overnight recording of SpO2 and
transcutaneous carbon dioxide (PtcCO2) was performed (SenTec Digital
Monitor, Therwil, Switzerland) at home when the CPAP use reached ≥6 h/
night or at least once within the first 2 months. CPAP and/or interface
adjustments were performed at home according to the results.
A follow-up outpatient visit with the pediatrician and the NIV
nurse was scheduled 1 month after CPAP initiation. Objective
compliance and recorded data were downloaded from the device
and analysed via the respective built-in software (Encore Basic Version
2.2 and DirectView 2.2, Philips Herrsching, Germany, for Philips
devices and ResScan Data Management software 5.0 and further
versions, ResMed, NSW, Australia for ResMed devices). Potential
problems or difficulties with CPAP were analyzed and the interface
and/or CPAP settings were changed if necessary. Follow-up
outpatient visits were then performed every month during the first
| 3
two months and then every three months. Patients and families were
contacted by phone or email (when preferred) every two weeks during
the first 2 months. CPAP compliance and efficacy were checked via the
built-in software data at each visit. In-hospital PG or PSG with CPAP
was only performed in case of persistent symptoms of OSA despite an
objective compliance ≥6 h per night or when the nocturnal gas
exchange and built-in software data were not conclusive or not
interpretable.
Objective compliance was calculated as the median number of
hours of CPAP use per night and the percentage of nights with a CPAP
use ≥4 h per night after 2 months. Non-compliance to CPAP was
arbitrarily defined as an objective CPAP use <4 h per night after the
first 2 months. CPAP efficacy was evaluated using the OAHI on a PG/
PSG or the AHI recorded by the built-in software data of the CPAP
device that was checked for accuracy as previously demonstrated by
our group,21 and with overnight gas exchange during CPAP. Data are
presented as median and range or mean and standard deviation.
3 | RESULTS
Thirty-one children were included in the outpatient program, which
represented 35% of the children started on long term CPAP during the
study period (Table 1). Fifty-seven subjects were not included because
of an age ≤6 month (n = 16), an unstable clinical state with an initiation
in the intensive care unit (n = 8), non-French-speaking family or a
family living outside the Parisian area (n = 33). The median age of the
population at CPAP initiation was 8.9 years (range 0.8-17.5) with a
predominance of male gender (n = 19). The median BMI centile was
87.3 (range 5.7-99.6). The most common diagnosis was Down
syndrome (n = 7), followed by achondroplasia (n = 3) and obesity
(n = 3). Median baseline OAHI was 12.5 events/h (range 4-100).
Baseline gas exchange data are shown in Table 1.
Twenty seven (87%) subjects achieved an excellent compliance
after the first 2 months, with a median use of 08:21 h:min (range 05:45-
12:20) per night (Table 2) at the follow-up visit. CPAP was used a median
of 25 (range 18-30) nights per month and ≥4 h/night during a median of
83 ± 17% of nights. The majority of patients (81%) used a nasal mask.
Fifteen patients (55%) used a constant CPAP with a mean pressure of
8.5 ± 1.0 cmH2O. APAP was used in the 12 (45%) oldest patients. APAP
pressure ranges were 8-12 cmH2O in 8 (67%) of the 12 patients.
Four patients never achieved a CPAP use ≥4 h per night. Three of
these patients were adolescents with Down syndrome (a boy of 16.2
years and two girls of 16.6 and 10.2 years, respectively) and the last
patient was an 8-year-old girl with an arteriovenous cervico-facial
malformation. These four patients presented variable degrees of
developmental delay, behavior problems and social stressors.
CPAP efficacy was assessed after 2 months by nocturnal gas
exchange in all the subjects, and by the AHI given by the built-in
software of the CPAP device in 17 (63%) subjects and on a PSG/PG in 4
(15%) subjects (Table 3). The built-in software automatic AHI was not
taken into account for six patients (22%) because their weight was below
the minimal recommended weight by the manufacturer. None of these
4 | AMADDEO ET AL.

TABLE 1 Demographic data and characteristics of the subjects patients had undergone a follow-up PG/PSG at 2 months. The OAHI on
(n = 31) the PSG/PG, as well as the AHI given by the built-in software data on the
Female to male ratio 12/19 CPAP device, improved dramatically (Figure 1 and Table 3). None of the
Age, years (median, range) 8.9 (0.8-17.5) patients spent ≥2% of night time with a SpO2 <90% or a PtcCO2
BMI, centile (median, range) 87.3 (5.7-99.6) >50 mmHg. One patient developed central sleep apneas with a central
Predisposing conditions apnea index of 7 events/h which resolved with a decrease of CPAP

Down syndrome 7
pressure. With the exception of this patient, no other patients required
several overnight assessments with CPAP in order to achieve OSA
Achondroplasia 3
correction.
Obesity 3
Three patients could be weaned from CPAP treatment after 21.5,
22q11 deletion 2
16.9, and 13.6 months of follow-up. One patient with mucopolysac-
Prader Willi syndrome 2
charidosis improved after mandibular distraction osteogenesis while
Pycnodysostosis 2
the other two subjects, one with 22q11 deletion and the other with a
Mucolipidosis type 1 2 polymalformative syndrome, improved spontaneously with age.
Mucopolysaccaridosis type 2 1 Concerning the four non-compliant children, the boy with Down
Crouzon syndrome 1 syndrome had a second adenoidectomy that resolved his OSA. The
Chiari malformation type 2 1 two girls with Down syndrome were switched to high flow air by nasal
Pierre Robin sequence 1 cannula but did not comply either with this type of respiratory support.
Polymalformative syndrome 1 The oldest girl had persistent OSA with an OAHI of 9 events/h and the
Cervicofacial venous malformation 1 mother decided to stop the follow-up at the last visit. The youngest girl,

Idiopathic OSA
Immunodeficiency with lymphoid tissue hyperplasia
OAHI, events/h (median, range)
Nocturnal gas exchange, n = 31
1
whose OAHI remains high at 45 events/h at the last PG control, will
have an orthodontic treatment with surgical palatal enlargement. The
1
girl with the arteriovenous cervico-facial malformation had a laser
12.5 (5-100)
reduction of the mass with a follow up PSG planned in the next months.

Mean SpO2 (%) 96 (92-98)

Minimal SpO2 (%) 85 (55-93) 4 | DISCUSSION

Time spent with SpO2 <90% (%)
Oxygen Desaturation Index (events/h)
Mean PtcCO2 (mmHg)
Maximal PtcCO2 (mmHg)
Time spent with PtcCO2 >50 mmHg (%)
BMI, body mass index; OAHI, obstructive apnea-hypopnea index; OSA,
obstructive sleep apnea.
0 (0-31)
Our study shows that an outpatient CPAP initiation program, with an
12 (1-95)
integrated age-adapted education program and a strict follow up, is
44 (32-50)
feasible in selected children with OSA with an excellent compliance in
48 (43-65)
the majority of patients and a correction of OSA. Indeed, 87% of the
0 (0-26)
patients achieved an excellent compliance with a median objective use
of CPAP of about 8 h per night and the correction of OSA.
Outpatient CPAP initiation programs have been reported in other
countries, namely in the USA10,11 and in Autralia.13 The main difference

TABLE 2 Continuous positive airway pressure (CPAP) compliance TABLE 3 Continuous positive airway pressure efficacy data of the
and equipment (n = 27) 27 compliant subjects

Duration of follow-up, months (median, range) 12.3 Poly(somno)graphy apnea-hypopnea index 2 (0-2)
(2.2-25.2) (events/h), n = 4

Objective CPAP compliance over the last month (n = 27) In-built software apnea-hypopnea index (events/h), 2 (0-4)
n = 17
Average use per night, h:min (median, range) 08:21
(05:45- Nocturnal gas exchange, n = 27 97 (93-100)
12:20) Mean SpO2 (%) 91 (84-96)
Percentage of nights with CPAP use >4 h, % (mean 83 ± 17 Minimal SpO2 (%) 0 (0-1)
± standard deviation)
Time spent with SpO2 <90% (%) 1 (0-9)
Average nights use per month, nights (median, range) 25 (18-30)
Oxygen Desaturation Index (events/h) 41 (35-48)
Constant CPAP pressure, cmH2O (mean ± SD) (n = 15) 8.5 ± 1.0
Mean PtcCO2 (mmHg) 46 (42-52)
Interface (n = 27)
Maximal PtcCO2 (mmHg) 0 (0-0)
Nasal mask 22
Time spent with PtcCO2 >50 mmHg (%)
Facial mask 3
PtcCO2, transcutaneous carbon dioxide; SpO2, pulse oximetry.
Nasal prongs 2
Data are presented as median (range).
AMADDEO ET AL.
| 5

between these outpatient programs and our program is the complete

absence of the need for an in-hospital titration PSG. Indeed, outpatient
programs in the USA and Australia involve a 2 h education session and
then a home acclimation to the mask and an in-hospital setting of the
device during one or more nights in the hospital. A recent study by Riley
et al12 describes an intensive CPAP initiation program, in which patient is
started directly with a low pressure in order to acclimatize to treatment.
After this initial visit, the patient and the parents are contacted via phone
calls for troubleshooting and are then seen for a second visit before an
in-hospital titration PSG. On the contrary, our program consists in a 2 h
education session during which the CPAP pressure is set at the optimal
pressure according to the patient's comfort and tolerance. CPAP
efficacy is then checked on the built-in software data and home
overnight SpO2 and PtcCO2 recordings without the need for a titration
PSG. In-hospital titration PSG or PG are restricted to patients with
persistent OSA symptoms or with not interpretable built-in software
data. The cost of our program is not comparable to those previously
described given the different organization of health care systems in the
USA and Europe. The staff of our program includes two full-time
paediatric pulmonologists, a full-time nurse specialised in NIV and
therapeutic education and a home NIV program coordinator. Moreover,
according to the recent French reimbursement law for the treatment of
OSA in paediatric patients, home care providers receive a weekly refund
which includes planned visits at 1 week, 1 month, 3 months and then at
least every 6 months after CPAP initiation.27
One of the main concerns about CPAP is compliance with
treatment. Despite a behavioral program and a close follow-up most
studies28,29 reported a suboptimal compliance with CPAP, with a mean
utilization ranging from 4.7 to 5.3 h/night. The routine use of CPAP
educational program may have contributed to the excellent compli-
ance observed in our study. Different types of intervention such as
supportive interventions during follow-up, educational interventions
and behavioral therapy have proven their efficacy to improve CPAP
30
compliance in adult patients. To our knowledge, only one study has
evaluated the effect of educational interventions on CPAP compliance
in children, showing that the benefit of education intervention is
maintained over time in three out of four preschool children.31 We
have developed this type of intervention over several years, which may
explain the excellent compliance (over 8 h/night) reported in a
previous study from our group.25 Importantly, especially for children,
it is crucial to choose the age- and developmental-adjusted
interventions that best match individual patient needs in order to
reach the most successful and cost-effective therapy
30
(Figures 2 and 3). Second, few children in our study were initiated
to CPAP due to obesity (n = 3), which has been recognized as a risk
29,32
factor for poor adherence. Finally, the close collaboration with
home care providers trained in pediatric CPAP may have contributed
to the excellent compliance. These home care providers perform
regular home visits and transmit to the hospital team the objective
CPAP compliance data which allows prompt adjustments during the
first weeks of treatment. Indeed, the first weeks of CPAP treatment
have been shown to be crucial for CPAP success.10,33 The close follow-
up visits during the first weeks with overnight recording of nocturnal
FIGURE 3 Booklet for educational intervention
gas exchange during CPAP allows immediate and efficacious adjust-
ment of the interface and the CPAP settings.34
Four (13%) patients never accepted CPAP despite repeated visits
and treatment adjustments. Three of these subjects had Down
syndrome and all had some degree of developmental delay and
behavioral problems. CPAP failure was particularly high in a recent
study where five out of 10 children with Down syndrome and OSA did
not accept CPAP.35 All these children were deficient on tests of
adaptive behavior, visual-motor integration, and academic achieve-
ment. Importantly, investment and motivation of the family is crucial
for the success of long term CPAP. In our experience, a supportive
family is probably one of the greatest predictor of CPAP success.36
Unfortunately, we were not able to retrospectively provide objective
data about family structure for these patients.
One could argue that, since the population of our study is
represented mainly by children with complex-OSA a possible
explanation for the success of our program is that parents of these
children are more motivated for CPAP than parents of children with
idiopathic OSA. Although we are not able to exclude this hypothesis,
our impression is that the success of our program is more related to the
CPAP intensive program rather than to the underlying diseases.
Another reason for the high CPAP compliance to treatment may
be cultural,37 with French parents being more prone to persuade their
children to adhere to treatment. However, we are not aware of data
comparing French parents to those from other countries. Moreover, as
a tertiary university hospital, families come from all around the world
and are therefore not culturally homogeneous.
We recognize that because of the lack of a titration PSG some
patients may have persistent OSA due to low pressure, or in the contrary
sleep fragmentation because of a CPAP pressure higher than needed.
However, we have already demonstrated that with our approach,
persistent respiratory events in children under CPAP are rare and usually
with little or no consequences.19 Moreover, unintentional leaks and
6 |
respiratory events could be diagnosed using built-in software data.21 For
this reason, we believe that PSG or PG may be restricted to patients with
discordance between gas exchange and built-in software or for children
with persistent OSA symptoms despite a good CPAP compliance.
The efficacy of the outpatient CPAP program with regard to the
correction of OSA, even in the absence of a titration study, was
excellent and comparable to previous work from our group. CPAP was
associated with a normalization of nocturnal gas exchange18,19 and a
dramatic decrease of the baseline OAHI and AHI.19 Moreover, at the
present time, we observed no long-term failure among the compliant
subjects and none of the patients required a tracheotomy.
Our study has some limitations. First, the patients included in the
outpatient initiation program were not compared to a control group. This
was explained by the selection criteria of the outpatient population. Indeed,
in clinical practice, because of medical and practical constraints, only around
35% of the total population started on long term CPAP was eligible for this
program. Finally, in a previous study from our group, during which CPAP was
initiated in the hospital for all the subjects, the mean objective CPAP
compliance was found to be as high as 8:17 ± 2:30 h/min per night.25
In conclusion, the present study confirms that the initiation of
CPAP in an outpatient setting is feasible and effective in selected
pediatric patients. A high rate of compliance can be achieved as well as
a correction of OSA. We strongly believe that our results highlight the
importance of a dedicated pediatric CPAP/NIV unit working in close
collaboration with trained pediatric homecare providers for the
initiation and the follow-up of these patients.
ACKNOWLEDGMENTS
The research of Brigitte Fauroux is supported by the Association
Française contre les Myopathies (AFM), Assistance Publique-Hôpitaux
de Paris, INSERM, ADEP Assistance, ASV Santé, Elivie, S2A Santé and
Université Paris Descartes—Paris V.
CONFLICT OF INTEREST
None.
ORCID
Alessandro Amaddeo http://orcid.org/0000-0003-2117-0781
Sonia Khirani http://orcid.org/0000-0003-4163-5021
Brigitte Fauroux http://orcid.org/0000-0001-6092-2662
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How to cite this article: Amaddeo A, Frapin A, Touil S,
Khirani S, Griffon L, Fauroux B. Outpatient initiation of long-
term continuous positive airway pressure in children.
Pediatric Pulmonology. 2018;1–7.
https://doi.org/10.1002/ppul.24138