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Currently, there are few and qualitatively unsatisfactory epidemiological data on the prevalence of sleep-disordered breathing (SDB) in children with cerebral palsy (CP).
The investigators aim to conduct a multicentre retrospective study to assess the presence of SDB in children with CP undergoing sleep studies (polysomnography or polygraphy).
A recent review of the scientific literature shows that in the 61 studies on sleep-disordered breathing (SDB) in children with cerebral palsy (CP), epidemiological data on the diagnosis of SDB are scarce and of unsatisfactory quality, as the data are mainly based on the use of targeted questionnaires such as the Pediatric Sleep Questionnaire (PSQ). A study published by Garcia in Developmental Medicine and Child Neurology in 2016 shows that children with CP, especially if they have epilepsy as a comorbidity, have a higher risk of SDB than healthy children.
In a study published in the Annals of Indian Academy of Neurology in 2013, Elsayed found that children with CP are at higher risk for SDB with increasing age, with a higher incidence in the school-age group than in the preschool age group. In the same study, the investigator observed a direct correlation between the severity of motor impairment (levels IV and V versus levels I - III of the Gross Motor Function Classification System scale, GMFCS) and the risk of SDB.
Dum found in 2023 in the Journal of Child Neurology a 2.91-fold (p < 0.001) increased risk of SDB in obese individuals with CP compared to normal weight individuals with CP.
In a retrospective study published in Sleep and Breathing in 2024, Kalyoncu showed good sensitivity (88.8%) of the PSQ in patients with CP, but unacceptable specificity of this instrument (30.7%) compared to polysomnography.
The primary objective of the proposed multicentre retrospective study is to assess the prevalence of SDB in children with diagnosed CP and suspected SDB who underwent polygraphic or polysomnographic examination in the period from 01/01/2020 to 12/31/2023.
A secondary objective is to assess the correlation between SDB and clinical outcome in cases where a follow-up of at least 12 months is available.
Data collected anonymously include clinical aspects (age, gender, GMFCS score, epilepsy diagnosis, adenotonsillar hypertrophy, previous adenotonsillar hypertrophy surgery, spinal changes, previous spinal surgery, oxygen therapy at home, use of Non-Invasive Ventilation/Continuous Positive Airway Pressure (NIV/CPAP) at home, presence of tracheostomy, reported snoring, reported findings of sleep apnea, Assessment of Complex Clinical-Assistance Needs in Pediatrics (ACCAPED) score, referral to a regional palliative care centre) and instrumental (evidence of obstructive sleep apnea in the final diagnosis, mean and minimum oxygen saturation (SPO2), percentage of time with SPO2 <90%, Apnea-Hypopnea Index (AHI), Mixed and Obstructive Apnea-Hypopnea Index (MOAHI), Central Apnea-Hypopnea Index (CAHI), Oxygen Desaturation Index (ODI), heart rate changes).
The following data is collected for the 12-month follow-up after the sleep study: deaths, number of emergency room visits or hospitalisations due to breathing problems, initiation of NIV/CPAP, surgery for adenotonsillar hypertrophy, tracheostomy packing.
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| Label | Type | Description | Intervention Names |
|---|---|---|---|
| Children (0-17 years) with cerebral palsy who had a sleep study at an Italian reference Center. | Children (0-17 years) of either sex, diagnosed with cerebral palsy who had a sleep study (polysomnography or polygraphy) at an Italian Center during the period 01/01/2020 - 12/31/2023. |
|
| Name | Type | Description | Arm Group Labels | Other Names |
|---|---|---|---|---|
| Polysomnography | Diagnostic Test | Sleep studies (polysomnography and polygraphy) will be performed and reported according to current American Academy of Sleep Medicine guidelines |
|
| Measure | Description | Time Frame |
|---|---|---|
| Number of Participants with obstructive sleep apnea in the study group | According to the American Academy of Sleep Medicine (AASM), pediatric obstructive sleep apnea (OSA) is diagnosed and classified based on the Apnea-Hypopnea Index (AHI) in the sleep study (polysomnography or polygraphy), which quantifies the number of apnea and hypopnea events per hour of sleep. The sleep study is diagnosed: Normal if AHI < 1 event per hour; Mild OSA if AHI is between 1 and 5 events per hour; Moderate OSA if AHI is between 5 and 10 events per hour and Severe OSA if AHI is greater than 10 events per hour. | The primary outcome measure refers to the first or only sleep study performed by participants in the time interval between January 2020 and December 2023. |
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Inclusion Criteria:
Exclusion Criteria:
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The study population consists of children (0-17 years) with cerebral palsy who underwent a sleep study (polysomnography or polygraphy) at one of the Italian centers participating in the study during the period from January 2020 to December 2023
| Name | Role | Phone | Extension | |
|---|---|---|---|---|
| GUIDO CAMANNI, MD | Contact | +393406730100 | guidocamanni@serafico.it | |
| ORNELLA CICCONE, MD | Contact | +39075812411 | ornellaciccone@serafico.it |
| Name | Affiliation | Role |
|---|---|---|
| LUIGI MASINI, MD | OSPEDALE UMBERTO I, SALERNO | Study Chair |
| Facility | Status | City | State | ZIP | Country | Contacts |
|---|---|---|---|---|---|---|
| Uoc Hospice Pediatrico - Azienda Ospedale-Universita' Padova | Padova | PD | 35121 | Italy |
| PubMed Identifier | Type | Citation | Retractions |
|---|---|---|---|
| 37733254 | Background | Kalyoncu M, Namli N, Yegit CY, Yanaz M, Gulieva A, Ergenekon AP, Selcuk M, Atag E, Ikizoglu NB, Sabanci M, Lale K, Gokdemir Y, Ersu R, Karakoc F, Karadag B, Eralp EE. Is the Pediatric Sleep Questionnaire sensitive for sleep-disordered breathing in children with complex chronic disease? Sleep Breath. 2024 Mar;28(1):331-337. doi: 10.1007/s11325-023-02915-z. Epub 2023 Sep 21. | |
| 26991829 |
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All collected IPD.
IPD will be made available for a period of 5 years after completion of the study.
Substantiated applications for IPD from interested researchers should be sent by e-mail to the sponsor-investigator.
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| ID | Term |
|---|---|
| D012891 | Sleep Apnea Syndromes |
| D002547 | Cerebral Palsy |
| ID | Term |
|---|---|
| D001049 | Apnea |
| D012120 | Respiration Disorders |
| D012140 | Respiratory Tract Diseases |
| D020919 | Sleep Disorders, Intrinsic |
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| ID | Term |
|---|---|
| D017286 | Polysomnography |
| ID | Term |
|---|---|
| D008991 | Monitoring, Physiologic |
| D019937 | Diagnostic Techniques and Procedures |
| D003933 | Diagnosis |
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| Uoc Pronto Soccorso Pediatrico Azienda Ospedale-Universita' Padova | Padova | PD | 35128 | Italy |
|
| Istituto Serafico Di Assisi | Assisi | PG | 06081 | Italy |
|
| Ospedale Pediatrico Bambino Gesu' | Roma | RM | 00165 | Italy |
|
| Azienda Sanitario-Universitaria Sant'Andrea | Roma | RM | 00189 | Italy |
|
| Ospedale Umberto I | Salerno | SA | 84014 | Italy |
|
| Irccs Materno Infantile Burlo Garofolo | Trieste | TS | 34137 | Italy |
|
| Universita' Degli Studi Dell'Insubria | Varese | VR | 21100 | Italy |
|
| Azienda Sanitario-Universitaria Federico Ii | Naples | 80138 | Italy |
|
| Background |
| Garcia J, Wical B, Wical W, Schaffer L, Wical T, Wendorf H, Roiko S. Obstructive sleep apnea in children with cerebral palsy and epilepsy. Dev Med Child Neurol. 2016 Oct;58(10):1057-62. doi: 10.1111/dmcn.13091. Epub 2016 Mar 16. |
| 37016991 | Background | Dum R, Walter V, Thomas NJ, Krawiec C. Odds of Cardiometabolic Diseases and Medications in Children With Cerebral Palsy. J Child Neurol. 2023 Mar;38(3-4):239-246. doi: 10.1177/08830738231167534. Epub 2023 Apr 5. |
| 23661965 | Background | Elsayed RM, Hasanein BM, Sayyah HE, El-Auoty MM, Tharwat N, Belal TM. Sleep assessment of children with cerebral palsy: Using validated sleep questionnaire. Ann Indian Acad Neurol. 2013 Jan;16(1):62-5. doi: 10.4103/0972-2327.107708. |
| D020920 |
| Dyssomnias |
| D012893 | Sleep Wake Disorders |
| D009422 | Nervous System Diseases |
| D001925 | Brain Damage, Chronic |
| D001927 | Brain Diseases |
| D002493 | Central Nervous System Diseases |