Effect of co-occurring conditions on the pediatric manifestations of catatonia: systematic analysis of individual patient data

Importance: Catatonia is a rare psychomotor syndrome, of which three main subtypes are described, "excited", "retarded" and "malignant". Its diagnosis is challenging in pediatric patients, due to its multitude of presentations, and the overlap of its symptoms with those of the co-occurring conditions. Improved knowledge of the impact of co-occurring conditions on the symptoms of catatonia would allow a more precise diagnosis. Objective To investigate the impact of developmental and psychiatric co-occurring conditions, or of their absence (unspecified catatonia) on the symptoms of catatonia, age of occurrence and sex-ratio in pediatric patients. For comparison purposes, patients with catatonia following NMDAR-antibody encephalitis (NMDARE), were included in the analyses, as a model of a medical condition with a known neurotransmitter-related mechanism. Data Sources: We searched PubMed, EMBASE and PsychINFO for studies published between January 1, 1995, and September 10, 2021. Study Selection: case-reports of patients under 18 years old with catatonia and the selected co-occurring conditions. Data Extraction and Synthesis: Co-occurring conditions, catatonia DSM-5 diagnostic criteria and “malignant” symptoms, age at first catatonic signs and sex were individually extracted. From these symptoms, we inferred the "excited", "retarded" and "malignant" catatonia subtypes. Main Outcome and Measure: We estimated the effect of co-occurring conditions on the catatonia symptoms and subtypes. Results On 1,425 study records,184 (237 patients) were eligible for analysis. The median age was 15 years old. There was a higher ratio of males to females in all conditions except for NMDARE. The "excited" form was more common in neurodevelopmental conditions (beta = 0.608, 95%CI: 0.416, 0.799), psychiatric conditions (beta = 0.261, 95%CI: 0.093, 0.429), and NMDARE (beta = 0.266, 95%CI: 0.047, 0.485), than in “unspecified catatonia’’ (beta=-0,062, 95%CI: -0.236, -0.112); the "retarded" form was more prevalent in psychiatric disease (beta = 0.291, 95%CI: 0.099, 0.483) and less in the medical condition (beta=-0.592, 95%CI: -0.846, -0.339). The "malignant" form was found more in psychiatric (beta = 0.367, 95%CI: 0.230, 0.505) and medical conditions (beta = 0.861, 95%CI: 0.632, 1.090), and less in unspecified condition (beta=-0.551, 95%CI: -0.687, -0.415). Within the different types of catatonia, some symptoms were more specific to one condition. Overall, stupor, mutism and negativism were the symptoms most independent of co-occurring conditions. Conclusions and Relevance: Searching for catatonic symptoms most frequently associated with a specific condition may unravel a catatonia otherwise unnoticed. Conversely, analyzing catatonic symptoms may help the clinician in the search for co-occurring conditions.