Secondary Logo

Share this article on:

Bilateral congenital torticollis: a case report with 25 years of follow-up

Matuszewski, Lukasz; Pietrzyk, Damian; Kandzierski, Grzegorz; Wilczynski, Michal

Journal of Pediatric Orthopaedics B: November 2017 - Volume 26 - Issue 6 - p 585–588
doi: 10.1097/BPB.0000000000000407
MISCELLANEOUS

Unilateral congenital sternocleidomastoid muscle (SCM) contracture causing torticollis is well known. Although the unilateral muscular torticollis is quite often recognized, a bilateral contracture of SCM muscle is very rare. A review of the literature showed only three cases of bilateral congenital torticollis reported over the last two decades. We present a case report of a boy with congenital bilateral torticollis with 25 years of follow-up. Bilateral tenotomies of the right SCM were performed and the child was immobilized in Schanz’s cervical orthosis. Three months after operative procedure, the physical examination indicated an increasing contracture of the left SCM with time. A similar operative procedure was applied to the left SCM. The follow-up examinations showed good wound healing and a positive outcome. Bilateral congenital muscular torticollis is a very rare form of muscle skeletal disorder. We describe a surgical treatment of such deformation that ended with a satisfactory result confirmed through a 25-year follow-up.

Department of Pediatric Orthopedics, Pediatric Clinical Hospital in Lublin, Medical University of Lublin, Lublin, Poland

Correspondence to Lukasz Matuszewski, PhD, Gebali 6, 20-093, Lublin, Poland Tel: +48 81 7185570; fax: +48 81 7415653; e-mail: lukasz.matuszewski@am.lublin.pl

This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0/

Back to Top | Article Outline

Introduction

Unilateral congenital muscular torticollis, also known as wry neck, is a common condition caused by shortening and fibrosis of the sternocleidomastoid muscle (SCM). This condition is usually discovered during the first few weeks of life. It is characterized by the tilt of the infant’s head to one side, with difficulties turning to the opposite side. A nontender mass attached of the SCM at the side toward the head is tilting can also be noted.

The bilateral contracture of the SCM is a very rare condition. We present a case of bilateral muscular torticollis first diagnosed and operated in a child aged 12 and a 25-year follow-up.

Back to Top | Article Outline

Case report

A 12-year-old boy was referred to the orthopedic department from a general-doctor practice. He was a second child of a nonconsanguineous marriage from full-term vaginal delivery in breech position. There was no family history of congenital deformities. He showed no other abnormalities. That particular patient had not been treated by any doctor before. All data considering his health status were provided by his parents, who reported that first symptoms of torticollis appeared in a preschool age.

The physical examination of the child indicated asymmetry of the head and face and a smaller right cheek. The lines of the mouth and eyes were crossing on the right part of the face. The head presented an altered, compulsory position of bending to the right side with the chin rotated to the left by ∼20° (Figs 1–3). The observed aggravated tension of both SCM widened the shape of the neck and restricted its movements. The difference in shortening on both SCM was 2 cm. Range of motion (ROM) in the neck was limited for rotation to 30° and for lateral flexion to 20°. ROM on the left side was better and was 40° for rotation and 30° for lateral flexion. Infants of 2 months of age had a median muscle function score of 1, which makes it difficult for the baby to turn the head side to side. Examination of the spine showed compensatory deformation in the cervical and upper thoracic part, an increased thoracic kyphosis, and elevation of the right shoulder. Radiographs did not show any structural deformation of the vertebrae (Figs 4 and 5). As a treatment, the upper and lower tenotomies of the right SCM were performed and the child was immobilized in Schanz’s cervical orthosis. Three months after the operative procedure, the physical examination indicated an increasing contracture of the left SCM with time. The head again presented an altered, compulsory position of bending now to the left side and the chin rotation to the right. A similar operative procedure was applied to the left SCM. The child was placed in cervical orthosis in maximal extension, without any rotation or head lateral bending. The follow-up examinations during a period of 6 months showed good wound healing and a positive outcome. The child and the parents were satisfied with the result of the treatment.

Fig. 1

Fig. 1

Fig. 2

Fig. 2

Fig. 3

Fig. 3

Fig. 4

Fig. 4

Fig. 5

Fig. 5

The patient was invited for re-examination 25 years after the initial treatment. The physical examination at the age of 37 years showed symmetrical rotation of the head and no restriction to flexion or to extension of the cervical spine (Figs 6 and 7). The ROM for rotation was to 90° on both sides. ROM for lateral flexion showed its highest range to about 60° both sides. The initial asymmetry of the face was hardly noticed. The patient is an agricultural physical worker. The previous treatment has no present effect on his well-being.

Fig. 6

Fig. 6

Fig. 7

Fig. 7

Back to Top | Article Outline

Discussion

Congenital muscular torticollis, or wry neck, is described as the most common form of painless torticollis. First descriptions were reported by Cheselden in 1749 1 and later by Anderson 1893 2. Unilateral SCM contracture causing torticollis is well known. However the etiology of CMT remains unknown. Possible causes include a lack of space in utero, resulting in local compartment syndrome or ischemia producing fibrotic SCM 3. As the child’s head is tilted toward the involved neck muscle and the chin rotated toward the contralateral shoulder, the diagnosis is made on physical examination at birth or shortly after 1,4. The finding of a hardened mass of SCM muscle on the involved side facilitates diagnostics. The unilateral contracture is almost universally present after infancy and may be replaced later with a fibrous contracted band 5.

Many authors agree that the nonsurgical treatment with stretching and massage yields an excellent result in more than 90% of patients 1,6–10. Others emphasize the fact that for such treatment to be very successful, it should be started within a few months after birth. If this or other treatments do not yield positive results, surgery can sometimes correct the problem. Conservative management is rarely successful in patients presented in childhood 7,11. Less than 10% of cases require a surgical approach if a significant restriction of motion (above 30°) or facial asymmetry is present at school age 4,10,12. Ferkel bipolar lengthening of SCM is a procedure of choice for those patients 13. We are of the opinion that in the patients seen in childhood, surgical intervention should be considered the treatment of choice to avoid further irreversible changes.

Bilateral congenital muscular torticollis is a very rare form of congenital muscular torticollis. The medical literature is scarce on cases of bilateral torticollis, with just three cases described in Hungary in 1993 14, in Singapore in 2009 15, and in China in 2016 16. A birth injury or a defective embryogenesis could likely be a cause of unilateral condition. Other theories of the origin of bilateral torticollis include fibrosis of the SCM muscle, resulting from venous occlusion because of intrauterine persistent position. Finally, we should take into consideration a genetic, growth retardation, infectious myositis, or mix of a variety of factors caused. Torticollis is a well-known deformity; however, bilateral malformation might present a differential diagnostic problem to orthopedic surgeons as well as ophthalmologists and neurologists.

Back to Top | Article Outline

Acknowledgements

The authors have contributed substantially to the submitted work, have made a significant contribution to the findings and methods in the paper and have reviewed and agree with the submission of the manuscript for review. All authors have read and approved the final draft.

Back to Top | Article Outline

Conflicts of interest

There are no conflicts of interest.

Back to Top | Article Outline

References

1. Morrison DL, MacEwen GD. Congenital muscular torticollis: observations regarding clinical findings, associated conditions, and results of results of treatment. J Pediatr Orthop 1982; 2:500.
2. Anderson W. Clinical lecture on sternocleidomastoid torticollis. Lancet 1893; 1:9.
3. Hummer CD, MacEwen GD. The coexistence of torticollis and congenital dysplasia of the hip. J Bone Joint Surg Am 1985; 67:30.
4. Ling CM. The influence of age on the result of open sternomastoid tenotomy in muscular torticollis. Clin Orthop Relat Res 1976; 116:142.
5. Coventry MB, Harris LE. Congenital muscular torticollis in infancy: some observations regarding treatment. J Bone Joint Surg Am 1959; 41:815.
6. Binder H, Eng GD, Gaiser JF, Koch B. Congenital muscular torticollis: results of conservative management with long-term follow-up in 85 cases. Arch Phys Med Rehabil 1987; 68:222.
7. Canale ST, Griffin DW, Hubbard CN. Congenital muscular torticollis: a long-term follow-up. J Bone Joint Surg Am 1982; 64:810.
8. Celayir AC. Congenital muscular torticollis: early and intensive treatment is critical. A prospective study. Pediatr Int 2000; 42:504.
9. Cheng JC, Tang SP. Outcome of surgical treatment congenital muscular torticollis. Clin Orthop Relat Res 1999; 362:190.
10. Ippolito E, Tudisco C, Massobrio M. Long-term results of open sternocleidomastoid tenotomy for idiopathic muscular torticollis. J Bone Joint Surg Am 1985; 67:30.
11. Minamitani K, Inoue A, Okuno T. Results of surgical treatment of muscular torticollis for patients greater than 6 years of age. J Pediatr Orthop 1990; 10:754.
12. Masłon A, Lebiedzinski R, Domzalski M, Synder M, Grzegorzewski A. Facial asymmetry in children with congenital muscular torticollis after surgical treatment. Chir Narzadow Ruchu Ortop Pol 2009; 74:31–34.
13. Do TT. Congenital muscular torticollis: current concepts and review of treatment. Curr Opin Pediatr 2006; 18:26–29.
14. Kustos T, Magdics M. Bilateral torticollis. Orv Hetil 1993; 134:2817–2820.
15. Babu MK, Lee P, Mahadev A, Lee EH. Congenital bilateral sternocleidomastoid contracture: a case report. J Pediatr Orthop B 2009; 18:145–147.
16. Shi XL, Li CS, Zhou KP, Yang C, Qi X, Li SQ. Congenital bilateral sternocleidomastoid contracture: a case report. Zhongguo Gu Shang 2016; 29:86–88.
Keywords:

children; congenital; torticollis

Copyright © 2017 Wolters Kluwer Health, Inc. All rights reserved.