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DOI: 10.1055/s-2004-830533
Georg Thieme Verlag KG Stuttgart · New York
Persistent Fixed Torticollis due to Atlanto-Axial Rotatory Fixation: Report of 4 Pediatric Cases
Publication History
Received: May 24, 2004
Accepted after Revision: November 10, 2004
Publication Date:
09 February 2005 (online)

Abstract
Atlanto-axial rotatory fixation (AARF) is a rare cause of childhood torticollis that may occur spontaneously or in association with trauma and upper respiratory infections. We describe the clinical findings, as well as the effectiveness of imaging in the diagnosis and the treatment of 4 children with AARF, in whom acute fixed non-dystonic torticollis was the presenting symptom. Onset of torticollis was spontaneous in Case 1, after general anesthesia for cholesteatoma surgery in Case 2, after a trauma in Case 3, and during hypersomnia in Case 4. Duration of torticollis prior to diagnosis was 3 months in the first two patients and 20 days in the other two. All the patients underwent cervical X-rays examinations, which were not contributory to the diagnosis, followed by CT, which demonstrated C1-C2 rotatory fixation. One patient had a spontaneous resolution; treatment with Gardner's tongs and soft collar permitted restoration of the normal alignment in the other 3 patients. AARF must be considered in all the patients with persistent painful torticollis.
Key words
Atlanto-axial rotatory fixation - torticollis - computed tomography - conservative treatment
References
- 1 Deliganis A, Baxter A, Hanson J A. et al . Radiologic spectrum of craniocervical distraction injuries. Radiographics. 2000; 20 S237-S250
- 2 Fidler M W, de Lange J. Atlanto-axial rotatory fixation. A cause of torticollis. Clin Neurol Neurosurg. 1979; 81 114-118
- 3 Fielding J W, Hawkins R J. Atlanto-axial rotatory fixation (fixed rotatory subluxation of the atlanto-axial joint). J Bone Joint Surg. 1977; 59 37-44
- 4 Jayakrishnan V K, Teasdale E. Torticollis due to atlanto-axial rotatory fixation following general anaesthesia. Br J Neurosurg. 2000; 14 583-585
- 5 Kowalski H M, Cohen W A, Cooper P, Wisoff J H. Pitfalls in the CT diagnosis of atlantoaxial rotatory subluxation. Am J Roentgenol AJR. 1987; 149 595-600
- 6 Lee S C, Lui T N, Lee S T. Atlantoaxial rotatory subluxation in skeletally immature patients. Br J Neurosurg. 2002; 16 154-157
- 7 Mihara H, Onari K, Hachiya M, Toguchi A, Yamada K. Follow-up study of conservative treatment for atlantoaxial rotatory displacement. J Spinal Disord. 2001; 14 494-499
- 8 Murray J B, Ziervogel M. The value of computed tomography in the diagnosis of atlanto-axial rotatory fixation. Br J Radiol. 1990; 63 894-897
- 9 Olivero W C. A unique presentation of nontraumatic atlanto-axial instability: a case report. Child's Nerv Syst. 1993; 9 117-118
- 10 Subach B R, McLaughlin M R, Albright A L, Pollack I F. Current management of pediatric atlantoaxial rotatory subluxation. Spine. 1998; 23 2174-2179
-
11 Suchowersky O, Calne D B.
Non dystonic causes of torticollis. Fahn S, Marsden CD, Calne DB Advances in Neurology, Dystonia 2. Vol 50. New York; Raven Press 1988: 501-508 - 12 Wortzman G, Dewar F P. Rotatory fixation of the atlanto-axial joint: rotational atlantoaxial subluxation. Radiology. 1968; 90 479-487
- 13 Wurm G, Aichholzer M, Nussbaumer K. Acquired torticollis due to Grisel's syndrome: case report and follow-up of non-traumatic atlantoaxial rotatory subluxation. Neuropediatrics. 2004; 35 134-138
MD Luisa Chiapparini
Department of Neuroradiology
Istituto Nazionale Neurologico „C. Besta“
Via Celoria 11
20133 Milano
Italy
Email: lchiapparini@istituto-besta.it