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Three cases with torticollis caused by an infratentorial tumour are presented. A father and his son both had a Lindau tumour, and another patient had an ependymoma. It is stressed that torticollis may be the predominant or only sign of an infratentorial tumour, and that torticollis should always be suspected to have an organic origin.
Congenital muscular torticollis may present in late childhood or adulthood with cervical contractures, limited range of motion, pain, and muscular hypertrophy. Seven of eight pediatric and adult patients with congenital muscular torticollis were injected with botulinum toxin type A. Only two patients had a modest benefit with improved range of motion. Early recognition and treatment of congenital muscular torticollis is crucial to prevent subsequent contractures and other complications.
Presented are four cases of familial spasmodic torticollis, comprising members of three families. The age of onset tended to be family-specific, and no afflicted family member had evidence of more widespread disease (dystonia musculorum deformans). Familial incidence supports the conclusion that spasmodic torticollis is organic in origin. Familial spasmodic torticollis occurs more often than has been generally recognized.
There are numerous well-known causes of acquired torticollis. However, an extensive review of the literature reveals only a rare, brief remark that associates torticollis with syringomyelia. We present three children found to have syringomyelia and spinal cord tumor, with torticollis as an early finding in each.
We examined suppression of EMG activity in the contracting sternocleidomastoid muscles, produced by electrical stimulation of the supraorbital nerve in 10 normal subjects and 9 patients with spasmodic torticollis. This exteroceptive reflex in the sternocleidomastoid muscle consisted of 2 or 3 phases: (1) an early, small, and unstable phase of facilitation, followed by (2) a period of suppression beginning 35 msec after the stimulus, lasting for 35 msec with a reduction in EMG activity to approximately 40% of the prestimulus level, and (3) a further phase of facilitation at a latency of 70 msec, with duration 35 msec and an increase in EMG activity to approximately 35% above prestimulus levels. The latency and duration of the suppressive phase of this reflex were similar to the exteroceptive suppression of EMG activity in the masseter muscle after supraorbital nerve stimulation (masseter silent period). In patients with spasmodic torticollis, the depth of this exteroceptive suppression in the sternocleidomastoid muscles was less than that observed in an age-matched cohort of normal subjects, although the latency and duration were normal. In contrast, exteroceptive suppression in the masseter muscle was normal. These findings suggest abnormal function of inhibitory interneuronal networks between the 5th cranial nerve and the motor neurons of the spinal accessory and upper cervical nerves which mediate exteroceptive suppression in the sternocleidomastoid muscle in patients with spasmodic torticollis.
OBJECTIVE: The purpose of this work was to assess the frequency of clinically relevant findings from plain films of infants evaluated for torticollis. PATIENTS AND METHODS: After institutional review board approval, radiology records were searched for infants 0 to 12 months of age who underwent plain film study for torticollis or "head tilt." Infants evaluated for trauma or Down syndrome were excluded. All of the studies were reviewed, demographic data was recorded, and any additional imaging studies were examined. RESULTS: A total of 502 patients (189 girls and 313 boys) were identified with an average age of 0.37 +/- 0.2 years. Head tilt was to the left in two thirds of patients. Ten patients had abnormal findings reported. Six of these proved normal on subsequent studies (3 suspected occipital-C1 fusions, 2 suspected cervical fusions, and 1 suspected hemivertebra). Four patients had true bony vertebral abnormalities including absent left C7 pedicle, multiple fusion anomalies from C4 to T2, C3 hemivertebra and thoracic spine anomalies, and C4 hypoplasia. This last patient had abnormal kyphosis on physical examination and demonstrated instability with dynamic testing. Twenty-five additional patients with normal plain films underwent spine computed tomography or magnetic resonance imaging; all were normal. CONCLUSIONS: The true-positive yield of plain films in nontraumatic infant torticollis was low (4 of 502). There were more false-positive than true-positive results. A common rationale for imaging is to exclude craniocervical or other unstable abnormalities that might contraindicate physical therapy, seen in only 1 of the 502 cases. Close physical examination could safely eliminate most patients sent for radiography.
Muscular torticollis is a medically well-known condition that is usually diagnosed in early childhood and in which early surgical intervention is recommended to prevent the development of facial asymmetries. The purpose of this study is to examine head posture and possible dentofacial asymmetries in patients who have undergone surgical treatment for muscular torticollis in early childhood. Natural head position roentgenograms were taken in frontal projection, a clinical examination of oral status was performed, and dental casts were made. Marked craniofacial and dental asymmetries were observed, combined with a deviant head posture, in spite of surgical treatment for muscular torticollis earlier in childhood.
A multi-adjustable torticollis orthosis is described for the post-operative bracing of patients after surgical correction of congenital muscular torticollis. The orthosis can be put on in the early post-operative period and the head and neck position can be maintained in the corrected, and later over-corrected position by the built-in multi-adjustable joint-mechanism. The details of the manufacturing are described. Twenty-five patients (13 girls and 12 boys) from age 1 to 22 with congenital muscular torticollis were fitted with the orthosis post-operatively for an average duration of 10 weeks. Satisfactory compliance with the orthosis was found in 23 cases. Complications were minimal (3 cases) and were related to scalp irritation which improved after minor adjustments of the halo.
The exact pathophysiologic mechanisms of spasmodic torticollis and other idiopathic torsion dystonias remain largely unknown. Thus, a variety of drugs have been used alone or in combination on an empirical basis to treat these disorders, but to date none have efficacy that is proven and consistent. The drugs in use include anticholinergics, benzodiazepines, dopaminergics and dopamine antagonists with variable degrees of clinical improvement. Botulinum toxin A injection treatment for spasmodic torticollis is safe and efficacious with minimal adverse effect. However, it is expensive and beneficial effects are short-lasting. Only when a spasmodic torticollis patient's symptoms are refractory to combined treatment, using various drugs and Botulinum toxin injections, should the patient be considered a candidate for neurosurgical procedures.
Eighty-four patients with congenital muscular torticollis were treated surgically in a 10-year period with a mean followup of 5 years (range, 2-13 years). All patients were classified into subgroups according to the type of congenital torticollis, the limitation of passive rotation of the neck, and other parameters, including head tilt and craniofacial asymmetry. Twenty-two (26.2%) patients underwent surgery before they were 1 year of age, 22.6% were between 1 and 3 years of age, 38.1% were between 3 and 10 years, and 13.1% were older than 10 years. Postoperative management included physiotherapy for 3 to 4 months and the application of a multiply adjustable torticollis brace for 10 weeks in children older than 2 years of age. Loss of the sternomastoid column was found in 82.6%, poor scar in 2.4%, lateral band in 47.2% and 1.2% required a second operation. The final overall score showed excellent results in 88.1%, good results in 8.3%, and fair to poor results in 3.6%. The most important factor affecting the overall result and outcome was found to be the age of the patient at the time of operation. However, this series also showed that for patients who were 10 years or older at the time of surgery, 63.6% had excellent results and 81.8% had good to excellent results, indicating the benefit of surgery even in the late cases.
In summary, then, without consideration of specific circuits or transmitter agents, one can conceive of a hypothetical model that involves both learning and the functional nature of the defect in torticollis and focal dystonia to describe the results obtained. The model must be further elaborated upon and tested, preferably in a quantitative manner. Naturally, the specific finding of a defective transmitter agent (e.g., GABA) such as described in parkinsonian syndrome (dopamine) or the interruption of a specific pathway that causes and improves a dyskinesia is desirable. In this chapter we have described the use of integrated EMG feedback for the treatment of focal dystonia or spasmodic torticollis. Although we have achieved significant results, it remains clear that further research in the treatment of these disorders is required. However, since this treatment does not require medication or surgery and the possibility for significant improvement is greater than 40%, it should be attempted in patients with focal dystonia or torticollis prior to other forms of therapy. SFT should be considered as a standard mode in the medical armamentarium used for the treatment of these disorders, either primarily or in conjunction with other forms of medical, surgical, and physical therapy.
INTRODUCTION: There are many causes of acquired non-dystonic torticollis: cervical bony anomalies, nasopharyngeal infections, tumours of the spinal cord, and posterior fossa, ocular, vestibular and gastrointestinal disorders. In children, non-dystonic is commoner than dystonic torticollis, except for in adverse reactions to drugs. Palatopharyngeal lesions due to a rigid object being impacted in the mouth (pencil-injury) are common in childhood. Many are not dangerous and require no specific treatment, although there is a risk of perforation of the pharyngeal wall and of a retropharyngeal abscess. CLINICAL CASE: We report the case of a 9 year old boy who was seen in the Emergency Department complaining of fever present for four days and progressive cervical rigidity for the past two days which did not improve with myorelaxant drugs. A lateral X-ray of the spine showed prevertebral air and on the oesophagogram a fistula tract was seen. On further questioning the boy admitted that a stick he had had in his mouth had caused damage the day before his fever started. CT showed the extent of the abscess. After a week of treatment with antibiotics and corticosteroids the clinical features disappeared, and on CT after two weeks it was seen that the abscess had resolved. CONCLUSIONS: We suggest that a lateral radiograph of the neck should be done in cases of acquired torticollis, even though there is no suspicious clinical history. Early diagnosis and treatment of retropharyngeal abscesses is essential to prevent extension to adjacent structures, and atlanto-axoid subluxation secondary to the oedema and tension of the ligaments caused by the abscess (Grisel's syndrome).
BACKGROUND: The inflammatory process in torticollis involves the cervical muscles, nerves, and/or the vertebral synovia causing painful and abnormal head position. Most cases of acute acquired non-traumatic torticollis are of benign etiology, but some patients may suffer from a serious disease requiring thorough investigation and hospitalization. OBJECTIVES: To describe the epidemiology, clinical features and the etiology of acute acquired non-traumatic torticollis in our center. METHODS: A retrospective chart review of 45 hospitalized children in the Sapir Medical Center over a 10 year period. RESULTS: We studied 45 children; 23 girls and 22 boys. Their mean age was 6 years (range 6 months to 16 years). All patients reported marked neck pain as their main complaint. Local infection was the most frequent etiology, and 33 (73%) patients were hospitalized during the fall-winter season. Twenty six patients underwent x-ray imaging, and of these, ten (38%) had pathology. The main findings were some degree of atlanto-axial subluxation, and widening of the prevertebral space. Treatment included analgesic and antibiotics for suspected bacterial infections. Two patients required surgical drainage. Cervical neck traction was performed on 15 patients. Mean admission time was 4.1 days and was shorter in the pediatric department compared to the ENT and orthopedic departments. CONCLUSIONS: Careful clinical and radiological evaluation for the wide spectrum of clinical entities should be done. Local infections of the respiratory tract are the most common etiology. Conservative treatment usually leads to complete resolution in a short time.
For children with torticollis, dynamic computed tomography scanning (DCTS) is the imaging modality of choice in diagnosing atlantoaxial rotatory subluxation (AARS). At present, there is no grouping system based on DCTS to determine severity and direct treatment. Fifty children with torticollis underwent DCTS in the workup for AARS. The relative rotation of C1 versus C2 was compared for left and right rotation views. Each DCTS was classified: stage 0, torticollis but normal DCTS; stage 1, limitation of motion (<15 degrees difference between C1 and C2, but C1 crosses midline of C2); and stage 2, fixed (C1 does not cross midline of C2). Duration of symptoms and treatment were compared. There were 27 girls and 23 boys with a mean age of 8.2 years. There were 8 stage 0, 30 stage 1, and 12 stage 2 DCTS. Average onset of symptoms to diagnosis was 6.7 days for stage 0, 8.6 days for stage 1, and 20 days for stage 2. A significant trend was found between increasing intensity of treatment and stage. Using this grouping system, the authors found that patients with a higher stage had an increase in the mean duration of symptoms and intensity of treatment.
Nodular fasciitis is a benign pseudosarcomatous proliferative lesion which is frequently misdiagnosed as malignant tumor clinically and microscopically. It usually occurs as a rapidly enlarging subcutaneous mass on the upper extremities, especially on the forearm. Here we report a patient showing muscular torticollis caused by nodular fasciitis in the sternocleidomastoid muscle (SCM). A 17-year-old woman was hospitalized because of rapidly progressive torticollis. The right SCM was markedly enlarged and firm on palpation. Muscle biopsy taken from the right SCM revealed massive proliferation of spindle shaped fibroblasts infiltrating into the endomysium. These findings coincided with the intramuscular nodular fasciitis. However, different from typical nodular fasciitis, no apparent nodule formation was found in this patient. Instead, diffuse proliferative lesion extended widely into the neck soft tissue. To our knowledge, this is the first report of muscular torticollis caused by nodular fasciitis involving the SCM.
We reviewed the efficacy and adverse effects of repeated botulinum toxin injections into hyperactive neck muscles of 107 successive patients with spasmodic torticollis. They received 510 injection treatments over a median period of 15 months (range 3-42 months). One patient failed to benefit at all, but 101 (95%) patients reported considerable (moderate or excellent) benefit from at least one treatment. On a global subjective response rating, 93% of 429 treatments resulted in some improvement and 76% in moderate or excellent improvement. Pain reduction followed 89% of 190 treatments with moderate or excellent reduction after 66%. Median duration of benefit was 9 weeks. All torticollis types responded equally well and injections into two (or more) involved neck muscles were more effective than injection into a single muscle. The most frequent adverse effect was dysphagia, occurring after 44% of all treatments, but this was severe after only 2%. Antibodies to botulinum toxin were detected in the serum of three out of the five patients in whom loss of treatment efficacy occurred. We conclude that botulinum toxin treatment is the most effective available therapy for spasmodic torticollis and practical advice is provided for anyone wishing to set up the technique.
Patients with spasmodic torticollis secondary to athetoid cerebral palsy may develop symptomatic degenerative cervical disc disease or spondylosis. In these cases, peri-operative stabilization of the cervical spine is considered to be difficult and recurrence occurs frequently. We reported a case with cervical spondylosis induced by athetoid cerebral palsy. A 44-year-old female with athetoid cerebral palsy had suffered from secondary spasmodic torticollis. 7 years previously, she had been admitted to our hospital for the first time with complaints of gait disturbance, dysesthesia and hypesthesia in all extremities. CT-myelography and MRI showed disc degeneration and spondylosis at the level of C4/5 and C5/6, compressing the spinal cord. She underwent C5 vertebrectomy and C4-6 anterior fusion. However, due to cervical dystonia, it was difficult for her to wear a Philadelphia collar, so she had to under go complete bed rest for postoperative neck stabilization. Her symptoms improved after the operation. 5 years after the first operation, she complained of gait disturbance and sensory disturbance, again. MRI showed compression of the spinal cord at C2-C4, rostral to the level of the previous surgical area. So we planned the second operation. Prior to the operation, we used botulinum A toxin to reduce the athetoid movement of the neck. And we performed C3-6 expansive laminoplasty and partial laminectomy of C2 and C7 laminae. After the second operation, she was able to wear the Philadelphia collar without trouble and her neurological condition improved again. Botulinum A toxin was very effective to control the spasmodic torticollis peri-operatively in this case with cervical dystonia.
Torticollis is a common condition presenting to the general or pediatric orthopedist. We describe the case of a child with severe torticollis caused by a hereditary unilateral absence of the sternocleidomastoid and trapezius muscles. Both his father and paternal grandfather had a forme fruste, with similar but milder findings. Electromyography and cross-sectional imaging were valuable in making the diagnosis. We believe this is the first reported case of hereditary unilateral muscle aplasia presenting as torticollis in an infant.