Case report: C1/2 rotational instability progressing to extreme subaxial hyperkyphosis in an adolescent with severe catatonia

Samuel F Schaible; Christoph Hamann; Sebastian Grunt; Fabian C Aregger; Moritz C Deml

doi:10.1016/j.bas.2024.102747

Case report: C1/2 rotational instability progressing to extreme subaxial hyperkyphosis in an adolescent with severe catatonia

Brain Spine. 2024 Jan 6:4:102747. doi: 10.1016/j.bas.2024.102747. eCollection 2024.

Authors

Samuel F Schaible¹, Christoph Hamann², Sebastian Grunt³, Fabian C Aregger¹, Moritz C Deml¹

Affiliations

¹ Department of Orthopaedic Surgery and Traumatology, University Hospital Bern, Inselspital, University of Bern, CH-3010 Bern, Switzerland.
² Division of Child and Adolescent Psychiatry and Psychosomatic Medicine, Department of Paediatrics, Inselspital, Bern University Hospital, University of Bern, CH-3010 Bern, Switzerland.
³ Division of Neuropaediatrics, Development and Rehabilitation, Department of Paediatrics, Inselspital, Bern University Hospital, University of Bern, CH-3010 Bern, Switzerland.

Abstract

Introduction: Autism spectrum disorder (ASD) is characterized by deficits in social communication, repetitive behaviors, and can be accompanied by a spectrum of psychiatric symptoms, such as schizophrenia and catatonia. Rarely, these symptoms, if left untreated, can result in spinal deformities.

Research question and case description: This case report details the treatment of a 16-year-old male ASD patient with catatonic schizophrenia and mutism, presenting with neck pain, left-rotated torticollis, and fever. MRI revealed atlantoaxial rotational instability and spinal cord compression from a dislocated dens axis. After inconclusive biopsies, empirical antibiotics, hard collar and halo fixation treatment, persistent instability necessitated C1/2 fusion. The ongoing catatonia was addressed with electroconvulsive therapy. Concurrently, he developed severe subaxial hyperkyphosis. The report examines the decision-making between conservative and surgical management for an adolescent with significant psychiatric comorbidity and progressive spinal symptoms against a backdrop of uncertain etiology.

Materials and methods: A case report and review of the literature.

Results: Posterior C1-C7 stabilization was successfully executed, effectively restoring cervical sagittal alignment, which was maintained throughout a two-year follow-up. Concurrently, the catatonia resolved.

Discussion and conclusion: To our knowledge, this is the third reported case of severe cervical deformity associated with fixed posture in a psychiatric patient. This case report emphasizes the critical importance of multidisciplinary collaboration in managing the interplay between neuropsychiatric disorders and severe spinal deformities. It showcases the practicality and efficacy of surgical intervention for persistent cervical deformity in pediatric schizophrenia patients, highlighting the necessity for a comprehensive risk-benefit analysis.

Keywords: Atlantoaxial instability; Autism spectrum disorder; Catatonia; Cervical hyperkyphosis; Pediatric deformity; Schizophrenia.

Publication types

Case Reports