Scoping Review on the Diagnosis, Prognosis, and Treatment of Pediatric Disorders of Consciousness

Abstract

Background and objectives: Comprehensive guidelines for the diagnosis, prognosis, and treatment of disorders of consciousness (DoC) in pediatric patients have not yet been released. We aimed to summarize available evidence for DoC with >14 days duration to support the future development of guidelines for children, adolescents and young adults aged 6 months-18 years.

Methods: This scoping review was reported based on Preferred Reporting Items for Systematic reviews and Meta-Analyses-extension for Scoping Reviews guidelines. A systematic search identified records from 4 databases: PubMed, Embase, Cochrane Library, and Web of Science. Abstracts received 3 blind reviews. Corresponding full-text articles rated as "in-scope" and reporting data not published in any other retained article (i.e., no double reporting) were identified and assigned to 5 thematic evaluating teams. Full-text articles were reviewed using a double-blind standardized form. Level of evidence was graded, and summative statements were generated.

Results: On November 9, 2022, 2,167 documents had been identified; 132 articles were retained, of which 33 (25%) were published over the past 5 years. Overall, 2,161 individuals met the inclusion criteria; female patients were 527 of 1,554 (33.9%) cases included, whose sex was identifiable. Of 132 articles, 57 (43.2%) were single case reports and only 5 (3.8%) clinical trials; the level of evidence was prevalently low (80/132; 60.6%). Most studies included neurobehavioral measures (84/127; 66.1%) and neuroimaging (81/127; 63.8%); 59 (46.5%) were mainly related to diagnosis, 56 (44.1%) to prognosis, and 44 (34.6%) to treatment. Most frequently used neurobehavioral tools included the Coma Recovery Scale-Revised, Coma/Near-Coma Scale, Level of Cognitive Functioning Assessment Scale, and Post-Acute Level of Consciousness scale. EEG, event-related potentials, structural CT, and MRI were the most frequently used instrumental techniques. In 29/53 (54.7%) cases, DoC improvement was observed, which was associated with treatment with amantadine.

Discussion: The literature on pediatric DoCs is mainly observational, and clinical details are either inconsistently presented or absent. Conclusions drawn from many studies convey insubstantial evidence and have limited validity and low potential for translation in clinical practice. Despite these limitations, our work summarizes the extant literature and constitutes a base for future guidelines related to the diagnosis, prognosis, and treatment of pediatric DoC.

Figure 1. Workflow Illustrating the Generation of Themes, Subthemes, and Statements Through the Study

The IBIA DoC-SIG Diagnosis and Prognosis Subgroup conceptualized the study and established 5 themes a priori, based on the previous work and recommendations for the general population with DoC and based on the terms included into the database search. Three systematic searches were repeated during the study, with 4 different databases interrogated at any search. Subthemes were iteratively generated, which informed the statements formulation. DoC = disorder of consciousness; IBIA-DoC SIG = Special interest Group on Disorders of Consciousness of the International Brain Injury.

Figure 2. Flowchart Illustrating the Extraction of Evidence on the Diagnosis, Prognosis, Treatment, and Techniques Used in the Care of Pediatric Patients With DoC

The selected full texts (n = 132, of which n = 127 informative) were in most cases reviewed by more than 1 team of experts, as relevant to more than 1 topic. Full texts were reviewed for information about clinical aim (diagnosis, prognosis, and/or treatment) and for the techniques used (behavioral tests, neuroimaging, and neurophysiologic examination). DoC = disorder of consciousness.