eAppendix. Trial Protocol
eTable 1. Modified Ramsay Sedation Scale
eTable 2. Modified Aldrete Recovery Score
Data Sharing Statement
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Sahyoun C, Krauss B, Bevacqua M, Antonsen A, Jardinier L, Barbi E. Safety and Efficacy Associated With a Family-Centered Procedural Sedation Protocol for Children With Autism Spectrum Disorder or Developmental Delay. JAMA Netw Open. 2023;6(5):e2315974. doi:10.1001/jamanetworkopen.2023.15974
Routine health care, whether physical examinations, blood sampling, vaccination, or dental care, is challenging and often traumatic for children with autism spectrum disorder (ASD) and children with developmental delay (DD), often requiring physical restraint or being indefinitely postponed. Behavioral interventions are critical for effective treatment of these children; however, in many children who have had traumatic medical experiences, these interventions may not be successful.1 Pharmacologic treatment with oral sedatives may be insufficient to perform common minor procedures, while the intramuscular route, requiring physical restraint for administration, can deepen the fear and lack of trust these children have toward the health care system.2 We describe a consecutive case series using a family-centered integrated behavioral and sedation protocol for common medical procedures in these children.
This case series was approved by the Swiss Association of Ethics Committees. Informed consent was obtained from the children's parents or legal guardians. This study is reporting following the reporting guideline for case series. We developed a procedural sedation protocol for children ages 2 to 16 years with ASD or DD described by parents as previously unapproachable for routine immunization and venipuncture due to extreme agitation. After discussion with the patient’s referring pediatrician or pediatric subspecialist and with the parents, children were enrolled for participation in the protocol. Our hypothesis was that an integrated behavioral-pharmacologic protocol is safe and effective in such patients. The main outcome measure was the need for physical restraint.
The protocol uses home desensitization to intranasal drug delivery, environmental modification, and intranasal dexmedetomidine combined with nitrous oxide (n2o), as dexmedetomidine alone would not allow painful procedures to be performed comfortably. Details of the protocol are provided in the eAppendix in Supplement 1. Patient, procedure, and sedation data were collected with each sedation. Study data were collected from June 2021 to December 2022 and managed using Qualtrics software (Qualtrics). Descriptive statistics were calculated using SPSS version 29.0 (IBM). Data were analyzed from January to February 2023.
During the study period, 44 children and their parents were approached for enrollment, and 43 consented. Median (IQR) patient age was 7.2 (4.5-11.4) years, and 31 patients (72%) were male. Of 42 patients who underwent the planned procedure, 39 patients (93%) had their procedures without physical restraint. Three patients required brief restraint (1 patient by 1 person and 2 patients by 2 persons), lasting less than 30 seconds. In 1 patient, the procedure was aborted because the sedation was suboptimal. There were no intermediate or sentinel adverse events during sedation or at the 24-hour follow-up call. Patient, procedure, and sedation characteristics are detailed in the Table.
In this case series assessing a family-centered integrated protocol for managing children with ASD and children with DD requiring common medical procedures, 93% of the patients required no physical restraint, and procedures were successfully completed in 98% of patients. The drug regimen was perceived as well tolerated, safe, and effective.
Children with ASD are often frightened by medical situations, particularly if they have a previous traumatic experience. Behavioral approaches have been used with variable success, are often time-consuming, and require multiple visits prior to the actual procedure.3
Intranasal dexmedetomidine has been used effectively and safely in children with ASD for nonpainful procedures.4 Although intranasal drug administration could be perceived as invasive and frightening, previous home desensitization, as in our protocol, could lead to greater acceptance if performed by a parent in a safe, familiar environment. Few studies have explored the combination of n2o and dexmedetomidine, and to our knowledge, none have explored it in children with ASD.5,6
The main limitation of this study is the lack of a control group with a different behavioral and pharmacologic approach. However, this study represents proof of concept of the safety and efficacy of an integrated behavioral and pharmacologic approach of home desensitization, environmental modification, and an intranasal and inhalational pharmacologic regimen. Future studies comparing this approach with other techniques are needed.
Accepted for Publication: April 17, 2023.
Published: May 30, 2023. doi:10.1001/jamanetworkopen.2023.15974
Open Access: This is an open access article distributed under the terms of the CC-BY License. © 2023 Sahyoun C et al. JAMA Network Open.
Corresponding Author: Cyril Sahyoun, MD, Division of Pediatric Emergency Medicine, Children’s Hospital of Geneva, Geneva University Hospitals, Rue Gabrielle-Perret-Gentil, 4, 1205 Geneva, Switzerland (firstname.lastname@example.org).
Author Contributions: Dr Sahyoun had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.
Concept and design: Sahyoun, Krauss, Jardinier, Barbi.
Acquisition, analysis, or interpretation of data: Sahyoun, Krauss, Bevacqua, Antonsen, Jardinier.
Drafting of the manuscript: All authors.
Critical revision of the manuscript for important intellectual content: Sahyoun, Krauss, Jardinier, Barbi.
Statistical analysis: Sahyoun.
Administrative, technical, or material support: Sahyoun, Antonsen, Jardinier.
Supervision: Sahyoun, Krauss, Bevacqua, Barbi.
Conflict of Interest Disclosures: None reported.
Data Sharing Statement: See Supplement 2.