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PubMed1993

Clinical application of a dysregulation model of illness and disease: a case of spasmodic torticollis.

A case of spasmodic torticollis is described to demonstrate the clinical usefulness of a new theoretical model of somatic illness and disease. Based on a conception of the living organism as a self-regulating system, the model attributes somatic disorders to a variety of regulatory disturbances including deficits in affect regulation and in the ability to maintain a stable and cohesive self. Throughout the analytical treatment, attention was given to repairing deficits in the patient's self-organisation, and to elevating chaotic impulses and inchoate emotions from a primitive sensorimotor level of experience to a mature representational level where they could be valued for their signal function and modulated through imaginal activity and communication with others. Analysis of the patient's pathologic internal object relations modified his self-organisation, and enabled him to establish regulatory interpersonal relationships that complemented his enhanced self-regulatory capacities. These changes were accompanied by a gradual resolution of the patient's torticollis.

Adult↗
PubMed1995

[The clinico-electromyographic characteristics of spastic torticollis].

The aim of the study was the investigation of central and peripheral pathogenetic mechanisms of torticollis spasmodic. It was examined 68 patients-37 with left pathology and 31-with right one. Electromyograms (EMG) were registered in m. sternocleidomastoideus at rest and under postural loadings (at sitting and lying position of patients). Statistically significant differences were found in the pattern of EMG in connection of the side of torticollis. In the case of the left side of pathology EMG changes were presumably in "participating" muscle, in the right disorders-in "participating" muscle as well as in "nonparticipating" one (this change can be compensatory reaction which direct to the correction of a torsion).

Adult↗
PubMed1995

Cervical dystonia (spasmodic torticollis). Some aspects of the natural history.

A literature survey was done to outline the natural history of cervical dystonia (spasmodic torticollis). The disorder starts between 25 and 60 years with head deviation or neck pain. Sometimes, there are personal or familial extrapyramidal antecedents (tremor, dystonia). The role of a preceding neck or head trauma is unsettled. Often, there is a delay in diagnosis of more than one year. The majority of patients show steady progression of their focal dystonia and reach maximal disability after five years; neck pain occurring in 70-80% contributes significantly to disability. In a third of the cases, there is a progression to segmental dystonia. In about 20% of the patients a spontaneous, sustained or unsustained remission of the torticollis can be observed; this occurs particularly in cases with earlier age of onset. Cervical dystonia has important psychosocial consequences: many patients have to withdraw from their job or from social activities.

Adult↗
PubMed1996

Surgical treatment of spasmodic torticollis: is there a role for microvascular decompression? With an illustrative case report.

Spasmodic torticollis (ST) is a puzzling movement disorder, characterized by involuntary tonic or clonic contractions of various neck muscles. From time to time, psychogenic, extrapyramidal and neuroperipheral origins have been postulated and reflecting the variety of theories proposed, as many different treatments have been attempted, none of which has shown absolute effectiveness. Surgery of ST classically includes destructive procedures such as myotomies, stereotactically placed lesions, rhizotomies and neurectomies. The recent application of the concept of "neurovascular conflict" to ST induced several authors to perform microvascular decompression (MVD) in these patients, with encouraging and in some cases long-lasting results. Our case report joins this group. From the analysis of pertinent literature we conclude that: a. Spasmodic torticollis is probably a collection of separate clinical entities; b. Even in the so-called "ST of neuroperipheral origin" some coexising central factor must be admitted; c. Neurovascular compression underlies at least some cases of ST. Historical, anatomical, physiopathological and clinical aspects of ST are extensively discussed.

Decompression, Surgical↗
PubMedDate not supplied

[Benign paroxysmal torticollis in childhood].

The Author describes one case of a male infant aged 12 months with recurrent short lasting episodes of torticollis, accompanied by vomiting, pallor, vacuous stare, sudden cry, without any electroencephalographical or neuroradiological alterations. At first torticollis attacks appeared with monthly frequency, afterwards they began to become less frequent and at last they disappeared at the age of about 30 months.

Age Factors↗
PubMed1976

Dystonia and tremor in spasmodic torticollis.

The occurence of extranuchal dystonia, facial spasm, parkinsonian symptoms (facial masking, bradykinesia, rigidity), tremor and family history of tremor was tabulated in a group of 30 patients with IST. The incidence of extranuchal dystonia increased as severity of IST increased. There was a strong trend for severity of extranuchal dystonia to increase as severity of torticollis increased, which was significant (p less than 0.001). There was a similar trend for severity of facial spasm to increase with increasing severity of torticollis (p less than 0.025). Parkinsonian features were seen in 10 of 30 patients, and in three the diagnosis of Parkinson's disease could be entertained. Tremor was seen in 26 of 30 patients being mild in 12, moderate in 11, and severe in three. A family history of tremor was present in 16 of 28 cases for whom history was available (12 primary, four secondary relations). The results are most consistent with the hypothesis that IST is a variant of DMD with tremor as an integral part of the disease and tremor represents a forme of the disease in family members.

Adult↗
PubMed1998

Isolated atlantoaxial rotatory fixation in a child with seronegative spondyloarthropathy presenting with torticollis.

Children presenting with torticollis demand thorough investigation. Atlantoaxial subluxation and rotatory fixation should be included in the differential diagnosis even in the absence of neurologic findings. We describe a 9-year-old boy with this condition associated with HLA-B27 positive seronegative spondyloarthropathy that went undiagnosed for 11 weeks. No other radiographic changes were seen, including no erosions. No neurologic findings were present. The child was treated with Halter head traction, which successfully reduced the atlantoaxial joint. This was followed by posterior C1-C2 fusion secondary to instability. Rheumatologists should be aware of this condition when examining a child presenting with torticollis.

Arthritis↗
PubMed1998

[Psychosomatic aspects of idiopathic spasmodic torticollis. Results of a multicenter study].

Idiopathic spasmodic torticollis (IST) is one of the most frequent dystonic movement disorders. Its classification as a focal dystonia, as well as its treatment with botulinum toxin resulted in groups of patients being regularly seen by neurologic specialists. In a multicentre study, we investigated psychosocial changes, coping and psychopathology, and their interrelations with signs, symptoms and course. 256 patients were included in the study (59.3% women, 40.7% men). The mean age was 49.1 years. Rotating torticollis occurred more often than latero-retrocollis and antero-retrocollis. A family history of IST was seen in 3.1% of the total sample. 34% of the patients had additional dystonic symptoms. Most frequently, these affected the upper extremities (13%), less often the legs. 19.1% of the patients had experienced a period of complete remission. The General Symptom Index of the SCL 90-R in 27% of the patients ranged above the double standard deviation of the normal controls, indicating a clinically significant psychopathology.

Adaptation, Psychological↗
PubMed1998

[Two children with unusual causes of torticollis: primitive neuroectodermal tumor and Grisel's syndrome].

Two children were presented with a torticollis. One, a 2-year-old girl, had a primitive neuroectodermal tumour of the cervical spinal cord which could not be radically removed surgically (the patient died three days later), the other, a 9-year-old boy, had the syndrome of Grisel. He was treated with antibiotics, head traction and atlantoaxial posterior spondylodesis. Acquired torticollis is a frequent innocent symptom in children with cervical lymphadenopathy following a respiratory tract infection. These cases demonstrate that additional investigation should not be delayed if relief of symptoms is not achieved very soon.

Atlanto-Axial Joint↗
PubMedDate not supplied

Congenital dislocation of the hip associated with congenital muscular torticollis.

Four recently identified cases of congenital dislocation of the hip associated with congenital muscular torticollis out of 49 consecutive cases of frank dislocation of the hip suggest that: the coexistence of congenital muscular torticollis and congenital dislocation of the hip occurs more commonly than is generally appreciated; the presence of either abnormality should initiate a careful appraisal for the presence of the other; although both conditions are seen more commonly in children delivered by breech extraction, a direct causal relationship has not yet been established although the abnormal uterine position is definitely related to each condition.

Breech Presentation↗
PubMed1998

Synostotic plagiocephaly causing pseudoparalysis of the superior oblique and ocular torticollis: report of a case with unique sensory findings.

PURPOSE: To report a case in which strabismus surgery did not eliminate the abnormal head posture although it did improve the vertical strabismus; and to consider the causes of this situation. CASE REPORT: The patient had a right eso-hypertropia and ocular torticollis to the left shoulder as a result of a right superior oblique mechanical deficiency, secondary to her craniosynostosis. A myectomy of the right inferior oblique muscle and a tucking procedure of the right superior oblique muscle were performed. Postoperatively, the abnormal head posture persisted, although strabismus surgery improved the vertical deviation. However, improvement of the abnormal head posture upon patching the deviated eye only indicated that the origin of the persistent abnormal head posture must be ocular. Further tests revealed that an anomalous retinal correspondence (ARC) with eccentric fixation was responsible. CONCLUSIONS: In patients with ocular torticollis, ARC with eccentric fixation must be ruled out prior to surgery as these may negatively affect the outcome.

Child↗
PubMed1978

Midification of tardive dyskinesia and spasmodic torticollis by apomorphine. Possible role of dopamine autoreceptors.

A group of 16 patients afflicted with involuntary movement disorders received subcutaneous injections of the direct dopamine agonist, apomorphine hydrochloride. Paradoxically, these injections were generally followed by a reduction of dyskinesia; this was most noticeable in patients with tardive dyskinesia and was only mild in some patients with spasmodic torticollis. Preferential stimulation by apomorphine of inhibitory dopamine presynaptic receptors (so-called dopamine autoreceptors) is proposed as the most likely explanation for the observed antidyskinetic effect of this drug. The results of this study also suggest that direct dopamine agonists may be used clinically to attenuate CNS dopaminergic transmission, especially when use of antidopminergic drugs such as the nuroleptics is contraindicated, as in the case of tardive dyskinesia.

Adult↗
PubMed1982

Plagiocephaly causing superior oblique deficiency and ocular torticollis. A new clinical entity.

Unilateral coronal suture stenosis provokes a shortening of the orbital roof of one side. It is argued that the effectiveness of the superior oblique muscle is reduced because of reduced length of its unreflected part and because of the increased angle between the reflected part of the tendon and ocular axis. This condition causes a clinical picture of superior oblique palsy, with ocular torticollis. We report four cases of this newly described clinical entity with various degrees of involvement of ocular motility.

Adolescent↗
PubMed1997

Evidence for DYT7 being a common cause of cervical dystonia (torticollis) in Central Europe.

Adult-onset focal idiopathic torsion dystonias (AFITD), such as torticollis, have a prevalence similar to that of multiple sclerosis and usually seem sporadic. Only recently has one large AFITD pedigree "K" with autosomal dominant inheritance and reduced penetrance from Northwest Germany provided the opportunity to identify a gene locus on chromosome 18p. We have now tested the relevance of this DYT7 gene locus in a collective of 18 nuclear AFITD families from Central Europe who were genotyped with chromosome 18p microsatellites. In three families, the affected relatives did not share a chromosome 18p haplotype, suggesting locus heterogeneity in AFITD. In the remaining 15 families, significant allelic association was observed for marker D18S1098. This result suggests that DYT7 is a common cause for AFITD at least in Central Europe, that many patients are descended from a common ancestor, and that the DYT7 gene is mapped in a 4.4 centimorgan subregion of chromosome 18p.

Adolescent↗
PubMed1996

Blink reflex recovery curves in blepharospasm, torticollis spasmodica, and hemifacial spasm.

R1 and R2 blink reflex responses to single and paired stimuli were investigated in 23 control subjects, 21 patients with blepharospasm (BSP), 20 patients with torticollis spasmodica (TS), and 23 with hemifacial spasm (HFS). For paired stimuli, we compared measurements of area and peak responses at two and three times R2 threshold. R1 and R2 indices were calculated as the average of the recovery values at 0.5-, 0.3-, and 0.21-s interstimulus intervals to test individual patients. Peak amplitude measurements at three times R2 threshold were optimal. The R2 index was abnormal in 67% of BSP patients, 37% of TS patients, and 50% of HFS patients on the affected side and 20% on the unaffected side. A normal R2 index in one third of patients with BSP may indicate that different pathophysiological mechanisms are involved in this type of focal dystonia.

Adult↗
PubMed1981

Iris pigmentation (melanin) in idiopathic dystonic syndromes including torticollis.

Iris pigmentation was evaluated in 153 Caucasian patients with torticollis and other focal, segmental, or generalized dystonias of unknown cause. Since these disorders are rare in non-Caucasians, it was hypothesized that a relationship might exist between decreased melanin metabolism, reflected by iris pigmentation, and a genetic predisposition to these disorders of voluntary movement. Patients were separated into two groups on the basis of iris pigmentation. Compared to control groups, there was a statistically significant reversal in the ratio of patients with light eyes versus dark eyes. Further comparisons were made in two groups of patients with drug-induced nonparkinsonian dyskinesias; no significant correlation was found between their dyskinetic syndrome and iris pigmentation. Data concerning race, ethnic origin, and other disorders of the basal ganglia support the hypothesis that there may be genetic predilection in individuals whose metabolic pathways produce less systemic melanin.

Adolescent↗
PubMed1981

Effect of L-glutamine and isoniazid on torticollis and segmental dystonia.

Fourteen patients with spasmodic torticollis and other segmental dystonic syndromes, who were refractory to previous forms of therapy, were selected for treatment with drugs intended to elevate brain gamma-aminobutyric acid (GABA) levels. These patients were simultaneously given diazepam, isoniazid, pyridoxine, and large doses of L-glutamine. Involuntary spasmodic activity improved to varying degrees in 7 patients; in 2 the dyskinesia became worse. Transient alteration of renal or hepatic function occurred in 6 patients and mild euphoria unrelated to neurological improvement in 8. Two patients are still being treated. Deficiency of GABA may be a factor in some patients with these disorders.

Adult↗