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Systematic review of the nonsurgical management of atlantoaxial rotatory fixation in childhood

Sae-Huang, M; Borg, A; Hill, CS; (2021) Systematic review of the nonsurgical management of atlantoaxial rotatory fixation in childhood. Journal of Neurosurgery: Pediatrics , 27 (1) pp. 108-119. 10.3171/2020.6.peds20396. Green open access

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Abstract

OBJECTIVE: Atlantoaxial rotatory fixation (AARF) is an acquired fixed abnormality of C1–2 joint rotation associated with torticollis in childhood. If the condition is left uncorrected, patients are at risk for developing C1–2 fusion with permanent limitation in the cervical range of movement, cosmetic deformity, and impact on quality of life. The management of AARF and the modality of nonsurgical treatment are poorly defined in both primary care and specialized care settings, and the optimal strategy is not clear. This systematic review aims to examine the available evidence to answer key questions relating to the nonsurgical management of AARF. METHODS: A systematic review was performed using the following databases: PubMed, MEDLINE, Healthcare Management Information Consortium (HMIC), EMCare, Embase, Cumulative Index to Nursing and Allied Health Literature (CINAHL), British Nursing Index (BNI), and Allied and Complementary Medicine Database (AMED). Search criteria were created and checked independently among the authors. All articles with a radiological diagnosis of AARF and primary outcome data that met the study inclusion criteria were included and analyzed by the authors. RESULTS: Search results did not yield any level I evidence such as a meta-analysis or randomized controlled trial. The initial search yielded 724 articles, 228 of which were screened following application of the core exclusion criteria. A total of 37 studies met the full criteria for inclusion in this review, consisting of 4 prospective studies and 33 retrospective case reviews. No articles directly compared outcomes between modalities of nonsurgical management. Six studies compared the outcome of AARF based on duration of symptoms before initiation of treatment. Comparative analysis of studies was hindered by the wide variety of treatment modalities described and the heterogeneity of outcome data. CONCLUSIONS: The authors did not identify any level I evidence comparing different nonsurgical management approaches for AARF. There were few prospective studies, and most studies were uncontrolled, nonrandomized case series. Favorable outcomes were often reported regardless of treatment methods, with early treatment of AARF tending to yield better outcomes independent of the treatment modality. There is a lack of high-quality data, and further research is required to determine the optimal nonsurgical treatment strategy.

Type: Article
Title: Systematic review of the nonsurgical management of atlantoaxial rotatory fixation in childhood
Open access status: An open access version is available from UCL Discovery
DOI: 10.3171/2020.6.peds20396
Publisher version: https://doi.org/10.3171/2020.6.peds20396
Language: English
Additional information: This version is the version of record. For information on re-use, please refer to the publisher’s terms and conditions.
Keywords: systematic review; atlantoaxial rotatory fixation; torticollis; nonsurgical; conservative; spine
UCL classification: UCL
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Brain Sciences
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Brain Sciences > UCL Queen Square Institute of Neurology
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Brain Sciences > UCL Queen Square Institute of Neurology > Department of Neuromuscular Diseases
URI: https://discovery-pp.ucl.ac.uk/id/eprint/10112284
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