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PubMed1987

Congenital muscular torticollis: results of conservative management with long-term follow-up in 85 cases.

A retrospective review of 277 patients with congenital muscular torticollis seen between 1970 and 1982 was conducted. In 85 cases this was supplemented by questionnaires and recent photographs, permitting a two- to 13-year follow-up. The first visit for 81.6% of patients was before six months of age. All were enrolled in a specific physical therapy program at the time of the first visit, unless they presented with severe torticollis after 12 months of age. Torticollis was mild to moderately severe in 90.6% of cases. Sternomastoid fibrotic nodules were present in 38.6%, more frequently in the more severe cases. Hip dysplasia increased in direct relation to severity and occurred in 10.5% of cases. At 12 months the torticollis had been conservatively resolved in nearly 70% of patients regardless of severity and presence or absence of focal fibrosis. Tenotomies were indicated in only ten children, eight of whom had first been seen after 12 months of age. Long-term sequelae were mild and consisted of craniofacial asymmetry, intermittent head tilt, and mild scoliosis. Developmental asymmetry or high tone due to limited mobility in the cervical spine were noted in 25.3% of infants initially and tended to subside with appropriate therapy. However, 11.8% of patients with long-term follow-up showed persistent functional asymmetry of the involved body side despite mild or moderate severity, early diagnosis, and complete resolution of the torticollis. Long-term observations indicate that congenital torticollis rarely requires surgical treatment.

Female↗

PubMed1988

Brain-stem auditory-evoked potentials in spasmodic torticollis.

The pathophysiology of idiopathic spasmodic torticollis is uncertain. Cerebral, basal ganglia, brain-stem, and cervicomedullary lesions have been implicated. Some investigators have found evoked-potential abnormalities, while others have not. We recorded brain-stem auditory-evoked potentials in six patients with otherwise normal result of examinations and laboratory studies. Brain-stem auditory-evoked potentials were recorded from Cz-A1 and Cz-A2 with rarefaction clicks delivered at 11.1/s and 70 dB above sensory threshold with 40-dB contralateral masking. Analysis time was 10 ms, filter bandpass was 150 to 3000 Hz, and 4000 averages were replicated. Patients and age-matched controls were compared by Student's t test. One patient had I-III and I-V interpeak latencies (IPLs) beyond clinical norms (99% tolerance limit). Mean IPLs ipsilateral to torticollis were 2.32 +/- 0.2 (I-III), 1.96 +/- 0.4 (III-V), and 4.16 +/- 0.3 (I-V). Contralateral IPLs were 2.0 +/- 0.2 (I-III), 2.0 +/- 0.2 (III-V), and 3.9 +/- 0.5 (I-V). Control values were 2.04 +/- 0.2 (I-III), 1.86 +/- 0.2 (III-V), and 3.86 +/- 0.4 (I-V). Absolute latencies, V/I amplitude ratios, and III-V IPLs did not differ significantly between patients and controls, nor did IPLs contralateral to torticollis. Ipsilateral I-III and I-V IPLs were greater in patients than in controls. These findings are consistent with those of some clinical reports and with experimental evidence that brain-stem lesions produce torticollis. They imply brain-stem dysfunction ipsilateral to head deviation in at least some patients with torticollis.

Brain Stem↗

PubMed1990

Congenital muscular torticollis. A spectrum of disease.

Sternocleidomastoid muscle fibrosis has been recognized for centuries, but its pathogenesis and treatment remains controversial. Pseudotumor of infancy is a firm fibrous mass in the sternocleidomastoid muscle appearing at 2 to 3 weeks of age. Congenital muscular torticollis is less common and appears later in life. Pseudotumor and congenital muscular torticollis probably represent different manifestations of sternocleidomastoid muscle fibrosis. Pseudotumor will usually resolve with conservative therapy; however, some patients will subsequently develop torticollis. Congenital muscular torticollis usually requires surgical release of the sternocleidomastoid muscle to achieve a good cosmetic result and to prevent plagiocephaly, facial asymmetry, and scoliosis. This report provides guidelines for the management of congenital muscular torticollis and pseudotumor of infancy based on the authors' experience and review of the medical literature. Representative case histories from the neonate through the adult are presented, and the pathogenesis, diagnosis, treatment, and prognosis are discussed.

Adult↗

PubMed1997

Cerebral glucose metabolism in patients with spasmodic torticollis.

The pathophysiology of spasmodic torticollis is not clear. Basal ganglia dysfunction has been suggested to underlie this clinical syndrome. We studied resting cerebral glucose metabolism in 10 spasmodic torticollis patients and 10 healthy controls by using positron-emission tomography and [18F]2-fluoro-2-deoxy-D-glucose. Statistical parametric mapping (SPM95) was used to compare both groups on a pixel-by-pixel basis. Torticollis patients showed a significantly higher glucose metabolism bilaterally in the lentiform nucleus (p < 0.005). Analyses performed using normalization of regional to global glucose metabolism confirmed this finding (controls, 1.26 +/- 0.06, and patients, 1.35 +/- 0.06; p < 0.01). The torticollis score did not correlate with glucose metabolism, nor did disease duration or side of chin direction. Our results indicate that the lentiform nucleus plays a predominant role in the pathophysiology of idiopathic spasmodic torticollis.

Adult↗

PubMed1987

EMG investigations in patients with torticollis.

EMGs have been performed on patients suffering from organic torticollis, hysterical torticollis and on normal control subjects. The EMG activity of the sternomastoid muscles during head rotation in control subjects and those with hysterical torticollis showed similar characteristics and neither group showed a response to body tilt. Subjects suffering from organic torticollis, however, did show a response to tilt. The results suggest that the response to backward tilt might aid in distinguishing the organic and hysterical forms of torticollis.

Electromyography↗

PubMed1998

Cortico-cortical inhibition of the motor cortical area projecting to sternocleidomastoid muscle in normals and patients with spasmodic torticollis or essential tremor.

OBJECTIVES: To investigate whether the cortico-cortical inhibition originally reported for the human hand motor area is present in the motor cortex for sternocleidomastoid muscle (SCM) and to evaluate the amount of inhibition in spasmodic torticollis and essential tremor. METHODS: Subjects were 14 normal healthy volunteers, 10 patients with spasmodic torticollis and 5 with essential tremor involving neck muscles. A paired-pulse magnetic stimulation was performed for the SCMs and first dorsal interosseous muscles (FDIs). RESULTS: In normal subjects, a subthreshold magnetic conditioning stimulus suppressed responses to a suprathreshold magnetic test stimulus when their interval was 1-5 ms in SCM. This indicates that the similar cortico-cortical inhibitory mechanism is present in the motor cortex for SCM as in the hand motor area. In the patients with spasmodic torticollis, the cortico-cortical inhibitory effect was reduced or absent in SCM, but normal in the FDI. In contrast, in patients with essential tremor, normal cortico-cortical inhibition was seen in both the SCM and FDI. CONCLUSIONS: The cortico-cortical inhibitory mechanisms of the motor cortex for SCM can be studied by a paired-pulse magnetic stimulation method. Our result of reduced cortico-cortical inhibition in torticollis patients suggests abnormal excitability (hyperexcitable or disinhibited) of the motor cortex for SCM in spasmodic torticollis.

Adult↗

PubMed1988

Depression in torticollis: a controlled study.

Eighty-five patients with idiopathic, persistent spasmodic torticollis were compared to a control group of 49 patients with cervical spondylosis to assess (1) the prevalence of psychiatric disorder prior to and following the onset of their physical complaint, and (2) to establish the effect of torticollis on self-reports of depression on the Beck Depression Inventory, and self-rated hopelessness on the Hopelessness Scale. The prevalence of psychiatric disorder in the two groups did not differ. The torticollis patients had significantly higher mean scores on the Beck Depression Inventory, but did not differ from the control group in terms of hopelessness. Self-referent negative cognitions such as self-blame, self-accusation, self-punitive thoughts, and negative body-image emerged as the prominent component of depression in torticollis, which has implications for the management of depression in torticollis.

Adolescent↗

PubMed2005

Persistent fixed torticollis due to Atlanto-axial rotatory fixation: report of 4 pediatric cases.

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.

Adolescent↗

PubMed1981

[Numerical reflections concerning alternating torticollis with nystagmus and binocular functions (author's transl)].

The authors describe a patient suffering from torticollis with an eccentric neutral point of relative rest caused by a congenital nystagmus (rotation to the left for long-range vision and to the right for close-range vision - nystagmus alternans); the torticollis is not aggravated in close-range vision by an asymmetrical Anderson-Kestenbaum operation at a suitable range (asymmetrical correction of the torticollis for long-range vision by exploiting the range of fusion into divergence). Surgical alternatives are discussed and trigonometric relations are correlated with clinical experience (for torticollis with binocular functions). Torticollis without binocular functions is discussed in brief.

Child↗

PubMed1986

Progressive supranuclear palsy: report of a case with torticollis, blepharospasm, and dysfluency.

A 76-year-old man is reported with advanced progressive supranuclear palsy (PSP) who developed a persistent, gradually progressive torticollis over a period of several months. Blepharospasm and dysfluency of the extrapyramidal type antedated the torticollis. This first report of torticollis in PSP reinforces previous notions that torticollis is related to pathologic changes in the striatum and brainstem. In addition, the combination of torticollis and blepharospasm in our patient supports the previous concept that these two "focal dystonias" have a common pathophysiologic mechanism. This also suggests that dysfluency in PSP may be an expression of a focal dystonia involving the muscles of articulation.

Aged↗

PubMedDate not supplied

A pharmacoeconomic evaluation of botulinum toxin in the treatment of spasmodic torticollis.

We performed a prospective study in 21 patients to evaluate the cost of treatment of spasmodic torticollis (cervical dystonia) before and after botulinum toxin type A (BTA) treatment and to assess the impact of BTA treatment on quality of life. Data were recorded for the analysis over a period starting 8 months before and ending 7.2 +/- 0.2 months (mean +/- SEM) after the first injection of BTA. All patients received at least two BTA injections (2.9 +/- 0.2 injections per patient). We studied direct medical costs (drugs, outpatient and inpatient visits, diagnostic procedures, physiotherapy), clinical effects of BTA (clinical rating scale and patient's global assessment), quality of life (French version of the Nottingham Health Profile [NHP]), and adverse reactions. Costs associated with the treatment of spasmodic torticollis before the first BTA injection were 479 +/- 143 French Francs (FF)/patient/month (97 +/- 29 US $/pt/mo). During BTA treatment, costs were 1,126 +/- 147 FF/pt/mo (228 +/- 30 US $/pt/mo), including a mean cost of BTA of 771 +/- 131 FF/pt/mo (157 +/- 27 US $/pt/mo). Treatment with BTA significantly decreased clinical symptoms of spasmodic torticollis and improved the emotional, social, and pain-related domains of the quality of life assessment. Botulinum toxin type A treatment increases the cost of treating spasmodic torticollis but improves quality of life in terms of pain, social, and psychologic functioning in patients with spasmodic torticollis.

Adult↗

PubMedDate not supplied

The prevalence of nonmuscular causes of torticollis in children.

Torticollis in children may result from a wide variety of pathologic processes. We retrospectively analyzed 288 patients seen in a tertiary care pediatric orthopedic facility for the evaluation of torticollis over a 10-year period to ascertain the frequency of nonmuscular causes of this condition. Fifty-three of these children (18.4% of the study population) had a nonmuscular etiology for their torticollis. Of these 53 patients, Klippel-Feil anomalies were present in 16 (30%), and an underlying neurologic disorder was present in 27 (51%). These neurologic conditions included ocular disorders in 12 (23%) patients, brachial plexus palsies in nine (17%) patients, and lesions involving the central nervous system in six (11%) patients. We conclude that nonmuscular causes of torticollis are collectively not rare. In a child without an identifiable muscular etiology for torticollis, Klippel-Feil anomalies or an underlying neurologic disorder is likely to be the cause of the deformity in the majority of patients.

Child, Preschool↗

PubMedDate not supplied

Congenital muscular torticollis in infants: ultrasound-assisted diagnosis and evaluation.

Ultrasonography is considered the modality of choice for differentiating congenital muscular torticollis from other pathologies in the neck. The authors present their experience with ultrasound examination for the evaluation and management of congenital muscular torticollis. Twenty-six infants, 14 boys and 12 girls, age ranging from 1 to 16 weeks, with torticollis and a palpable mass were examined. Ultrasound showed a well-defined mass in the sternocleidomastoid muscle. The lesions ranged in size from 8 to 15.8 mm on maximal transverse diameter, with length ranging from 13.7 to 45.8 mm. Clinically the torticollis disappeared between 1 to 6 weeks, with complete clinical reduction of the palpated mass between 2 and 8.5 weeks. The ultrasonographic disappearance of the mass was delayed by an average of 2 weeks in comparison to the clinical disappearance of the mass. Ultrasound is advocated for the diagnosis and follow-up of congenital muscular torticollis because it noninvasively provides reliable and dynamic information without sedation.

Diagnosis, Differential↗

PubMed2000

Factors that ameliorate or aggravate spasmodic torticollis.

A sample of 72 patients with adult onset torticollis were asked to complete a checklist to indicate how a list of situations and activities affected the severity of their torticollis. Stress and self consciousness were reported as aggravating factors by more than 80% of the sample, whereas walking, fatigue, and carrying objects were noted as exacerbators by over 70% of the patients. For more than 40% of the sample, torticollis improved in the supine position, by relaxation, sleep, and lying on the side. However, the last four factors also worsened the head deviation in 16% to 25% of the patients. Use of a "geste antagoniste" to maintain the head in the body midline, was reported by 64 (88.9%) of the patients, which was still effective in correcting head position in 47%. The sensitivity of torticollis to social and emotional factors can be best explained in terms of a possible link between extrapyramidal and affective disorders through overlapping changes in catecholamine metabolism. The worsening of torticollis with peripheral motor activity (walking, running, writing) or its improvement with changes in body posture or with the geste antagoniste is best viewed in terms of alterations of peripheral proprioceptive feedback or central corollary discharge provoked by the motor output or command.

Adult↗

PubMed1981

Plasma dopamine-beta-hydroxylase activity in spasmodic torticollis. A sibship study.

Spasmodic torticollis remains an ill-understood disease. A central deficit in neurotransmitter activity in the basal ganglia may be present in this disorder. In the present study, dopamine-beta-hydroxylase (DBH) activity has been evaluated in the plasma of patients suffering from spasmodic torticollis and in their sibship in 4 informative families. In 1 of these families, 2 brothers are affected with torticollis and they have high plasma DBH levels. In the 3 other families the plasma DBH values are within normal ranges in the probands and their first-degree relatives. The genetic study of plasma DBH activity allowed to discriminate between two forms of spasmodic torticollis, one, which is most likely linked to familial torsion dystonia, appears to be associated with altered dopaminergic activity, while the other, nonfamilial with normal sympathetic activity, is probably an idiopathic or sporadic form of spasmodic torticollis.

Aged↗

PubMed2002

Fever and acquired torticollis in hospitalized children.

Acute torticollis due to non-traumatic atlanto-axial subluxation (AAS) is often seen in children presenting with inflammatory conditions of the upper respiratory tract and the neck. Grisel's syndrome is the eponym given to this condition. These patients may present earlier in the disease process without evident subluxation. Thus, early recognition of the condition with prompt commencement of appropriate conservative treatment could halt the progression into Grisel's syndrome. The purpose of this study is to address the importance of early recognition of inflammatory torticollis that can be treated successfully by conservative methods. A retrospective review was made of the case files and radiological investigations of 13 children with fever and torticollis who were treated in the neurosurgery unit of Hamad General Hospital in Qatar, over a two-year period from July 1996 to July 1998. The children were aged between three and 12 years with a male to female ratio of 3:1. All patients arrived at the hospital within 48 hours of onset of torticollis and almost all had manifestations of upper respiratory tract or head and neck infections. Radiological examination by cervical spine X-rays, computerized tomography (CT) or magnetic resonance images (MRI) revealed that only three cases out of 13 had AAS. All patients underwent conservative treatment that included rest, neck collar, simple analgesics and antibiotics, where appropriate. A muscle relaxant was used in nine cases and Halter traction was applied to the three with AAS. All patients responded well to treatment and none required surgical intervention for AAS. We conclude that the majority of children presenting acutely with inflammatory torticollis have rotational deformity only without AAS. Progression to the latter, i.e. Grisel's syndrome, may be aborted should the diagnosis be made early and conservative treatment initiated in time. On the other hand, delay in diagnosis would deprive these children an opportunity of receiving effective conservative treatment.

Acute Disease↗

PubMed1999

Epidemiology, presentation and management of congenital muscular torticollis.

AIM OF STUDY: Congenital muscular torticollis is a condition of debatable aetiology and management. Untreated, cervical function and facial cosmesis may be severely compromised. The aim of this study was to establish the epidemiology, presentation and management of congenital muscular torticollis in Singapore. PATIENTS: Ninety-one patients with torticollis were seen at the National University Hospital (NUH) from January 1994 to December 1997. Torticollis was first noted at a median age of 2 months with the median age of presentation being 6 months. At presentation, a sternomastoid tumour was noted in 33 patients and 62 patients had facial asymmetry. Thirteen of 22 patients with neonatal records available had mandibular hypoplasia at birth on the side where the sternomastoid was affected. Half of the patients (45) had a right sided lesion, with 46 being left sided. The rates of assisted breech delivery, instrumental deliveries (forceps and vacuum) and Caesarean section were higher in the study group. Nine (59.1%) of 13 patients with vertex presentation, had a lesion on the side of the presenting shoulder. Forty-eight of 72 patients responded well to therapy with improvement; 20 underwent surgery and the median age of presentation of 19.5 months in this group was significantly later than that of 4 months in the group which responded to physiotherapy alone. CONCLUSION: Birth trauma appears to be the main aetiological factor in congenital muscular torticollis. Patients generally respond well to physiotherapy. This study revealed 2 findings hitherto unreported: (1) mandibular hypoplasia may be an useful early sign of this condition, and (2) the side affected may depend on the side of shoulder delivered first. More studies, however, are required to confirm these findings.

Age of Onset↗

PubMedDate not supplied

Ultrasonographic study of the coexistence of muscular torticollis and dysplasia of the hip.

The consistent relationship between congenital muscular torticollis and dysplasia of the hip is now widely accepted. However, the coexistence rate of these two disorders has been reported with variations from 0 to 20%. To obtain a more accurate coexistence rate, ultrasound scanner was used as the diagnostic tool for both of these disorders.Sixty-three children (30 boys and 33 girls) younger than 6 months who had undergone ultrasound scanning of both bilateral sternocleidomastoid muscle and bilateral hips were included in this study. Forty-seven children were confirmed to have muscular torticollis, and the remaining 16 cases were diagnosed as postural torticollis. Only these 47 cases were included for analysis of the coexistence rate. Eight children were found with dysplasia of the hips associated with muscular torticollis, which included Graf's type IIa for four hips, type IIb for two hips, type IIIa for one hip, and type IIIb for one hip. From the results of the present ultrasonographic study, the coexistence rate of congenital muscular torticollis and dysplasia of the hip was concluded to be 17%. If only those dysplastic hips (type IIb, IIIa, IIIb) that required treatment were included, the coexistence rate would be lowered to 8.5%.

Female↗