Torticollis as a presenting symptom of pediatric CNS tumors: A systematic review

Abstract

To assess the frequency of torticollis as a presenting symptom of pediatric CNS tumors and its impact on pre-diagnostic symptom intervals (PSIs) and patient outcomes. We performed a systematic review of studies reporting torticollis in children with various CNS tumors. We searched PubMed for studies published from January 1972 to March 2021 in English. Case reports were included in the analysis if the following criteria were met (1) torticollis was the presenting symptom secondary to a CNS tumor (2) children <18 years of age, (3) underwent imaging intervention and (4) outcome data provided. Of 1,365 relevant articles, 45 were eligible for analysis according to our inclusion criteria representing 95 patients. Two independent investigators extracted the data, and a third investigator arbitrated discrepancies. We found that 38.9% of CNS tumors exhibited torticollis as the only presenting symptom. PSI lengths ranged from 0 to 4 years (median, 5 months) for low-grade tumors, and patient age and PSI length were inversely associated. Of patients with low-grade tumors, 23.8% received physiotherapy, in contrast with 0% of patients with high-grade tumors. PSI length also increased for patients who received rehabilitation, and 39.3% and 7.1% of patients with high-grade and low-grade tumors, respectively, died of their disease. Patients with low-grade tumors and torticollis as a primary presenting symptom were more likely to have a prolonged PSI. Therefore, CNS tumors should be considered in the differential diagnosis of acquired torticollis in children.

Keywords: CNS tumors; Low grade tumors; Pediatric cervical dystonia; Posterior fossa tumors; Torticollis; stiff neck.