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[Surgical treatment of nystagmus with horizontal torticollis].

PURPOSE: To study the effect of modified Anderson's procedure in nystagmus surgery with pure horizontal torticollis. METHOD: Retrospective study of 8 patients with nystagmus and pure horizontal torticollis that were surgically treated in our hospital in a 4 years period. Mean age of the sample was 9 years, SD 8.6 (range 4-30). Large bilateral recessions were made in the yoke rectus muscles that are responsible for the versions to obtain the null position. The amount of surgery was variable according to the torticollis intensity and the association of strabismus. Follow-up length ranged from 15 to 45 months (average 31.14, SD 11.55). RESULTS: Torticollis improved in all patients at the end of the study. A good result was achieved in 62.5% of the cases, with a residual torticollis inferior to 10 degrees. In two cases, severe torticollis improved to a moderate one (10-20 degrees ). Torticollis reversal occurred in one patient, who needed a second operation. Neither clinically significant ductions limitations, nor secondary strabismus were found. CONCLUSIONS: The modified Anderson's procedure was effective for treating moderate horizontal torticollis with nystagmus. Severe torticollis were not totally corrected. Large muscle recessions were not associated with clinically significant ductions limitations. There was not secondary strabismus (Arch Soc Esp Oftalmol 2003; 78: 481-486).

Adult↗

Outcome of selective ramisectomy for botulinum toxin resistant torticollis.

OBJECTIVE: To investigate the long term outcome of selective ramisectomy denervation in patients with botulinum toxin resistant spasmodic torticollis. BACKGROUND: The published surgical series of ramisectomy treatment for torticollis do not provide systematic information on patients who develop resistance to the current standard of treatment-botulium toxin injections. Moreover, there is little information on surgical outcome using rating scale measurements of torticollis, or assessments of functional and occupational capacity. METHODS: Using a structured interview format and videotape assessments of severity of dystonia in a retrospective fashion, detailed follow up information was obtained on 16 patients who underwent open label selective denervation for severe, disabling torticollis, refractory to injections of botulinum toxin. RESULTS: Of 16 patients with disabling torticollis followed up postoperatively for a mean of 5 years, six (37.5%) had a moderate or complete return of normal neck function, as determined using functional capacity scales, whereas 10 had only minimal relief of dystonia or gain in function. Six of the 16 patients (37.5%) underwent a second peripheral denervation operation, and one required a third. Of 11 patients working outside the home before surgery, nine were disabled by dystonia, and only one continued to work after surgery. Dystonia rating scale scores of videotaped examinations using a modification of the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) improved in 12 of 14 patients (85.7%) who underwent selective ramisectomy. When patients with primary botulinum toxin resistance were excluded, the magnitude of benefit for this subgroup was 31.9% of the baseline dystonia score (p<0.0002), comparable with the degree of improvement in a group of control patients receiving botulinum toxin treatment for torticollis. CONCLUSION: About one third of patients with torticollis resistant to injections of botulinum toxin may derive modest long term functional improvement from selective denervation, with a reduction in dystonia by about 30%, but remain unable to work.

Adult↗

Pattern of premature degenerative changes of the cervical spine in patients with spasmodic torticollis and the impact on the outcome of selective peripheral denervation.

OBJECTIVES: To characterise the pattern of and risk factors for degenerative changes of the cervical spine in patients with spasmodic torticollis and to assess whether these changes affect outcome after selective peripheral denervation. METHODS: Preoperative CT of the upper cervical spine of 34 patients with spasmodic torticollis referred for surgery were reviewed by two radiologists blinded to the clinical findings. Degenerative changes were assessed for each joint separately and rated as absent, minimal, moderate, or severe. Patients were clinically assessed before surgery and 3 months postoperatively by an independent examiner using standardised clinical rating scales. For comparison of means a t test was carried out. To determine whether an association exists between the side of degenerative changes and type of spasmodic torticollis a chi(2) test was used. Changes in severity, disability, and pain before and after surgery were calculated using a Wilcoxon matched pairs signed ranks test. RESULTS: Fourteen out of 34 patients had moderate or severe degenerative changes. They were predominantly found at the C2/C3 and C3/C4 level and were significantly more likely to occur on the side of the main direction of the spasmodic torticollis (p = 0.015). There was no significant difference in age, sex, duration of torticollis, overall severity, degree of disability, or pain between the group with either no or minimal changes and the group with moderate or severe changes. However, in the second group the duration of inadequate treatment was longer (10.1 v 4.8 years; p=0.009), head mobility was more restricted (p = 0.015), and head tremor was more severe (p = 0.01). At 3 months postoperatively, patients with n or minimal degenerative changes showed a significant improvement in pain and severity whereas no difference was found in those with moderate or severe changes. CONCLUSIONS: Patients with spasmodic torticollis have an increased risk of developing premature degenerative changes of the upper cervical spine that tend to be on the side towards which the head is turned or tilted and compromise outcome after surgery. Effective early treatment of spasmodic torticollis with botulinum toxin seems to have a protective effect. Patients with spasmodic torticollis and restricted head mobility who do not adequately respond to treatment should undergo imaging of the upper cervical spine. Patients with imaging evidence of moderate or severe degenerative changes seem to respond poorly to selective peripheral denervation.

Adult↗

Cerebrospinal fluid protein patterns in spasmodic torticollis.

In order to investigate the value of CSF-protein analyses in spasmodic torticollis CSF from six patients with probable organic and two patients with probable psychogenic torticollis was examined by isoelectric focusing and electrophoresis. In all the patients with organic torticollis two pathological CSF-protein fractions were found in the alkaline region on electrofocusing and in four cases aberrant fractions occurred also in the acidic pH range. An increasing number of abnormal fractions were noted during at least the first year after onset of symptoms. Lithium treatment of three patients resulted in a striking decrease of torticollis as well as of the number of abnormal CSF-protein fractions. During placebo treatment of two cases, torticollis and the pathological CSF-proteins recurred. Some observations, including a few previous autopsy findings, might indicate that an encephalitogenic agent is involved in the pathogenesis of organic torticollis. In the patients with psychogenic torticollis the CSF-protein pattern was normal. This investigation supports a recent suggestion that organic and psychogenic torticollis might be distinguished by electrofocusing of the CSF-protein.

Adult↗

[Torticollis in a group of strabismic patients in Cameroon].

PURPOSE: Our aim was to study the characteristics of torticollis in a group of strabismic patients in Cameroon. PATIENTS AND METHODS: This was a prospective study carried out in the ophthalmology service of the General Hospital, Douala from January 1991 to December 2004. All strabismic patients followed up in our service were included in this study and they all had a complete strabologic and ophthalmological examination. Torticollis was analysed using photographs and classified according to its configuration, degree and variability. RESULTS: We examined 379 strabismic patients of which 227 were divergent (59.9%) and 152 convergent (40.1%). We found 166 cases of torticollis (43.8%). This pathology was found in 52.6% of patients with esotropia versus 37.9% in exotropia. Torticollis was classified as severe in 14.5% of patients, moderate in 45.8% and mild in 39.7%. Two out of three early esotropias had alternating torticollis. In congenital monophthalmus syndrome, torticollis was found in 68.4% of cases and in 100% of cases with the alphabetic syndrome. Moreover 63.8% of strabismics with torticollis have astigmatism. CONCLUSION: Torticollis in strabismic patients is a cause for concern as it is frequent and its functional consequences are severe.

Astigmatism↗

Dissociation between motor potential and contingent negative variation after lesions of some basal ganglia in man: electrophysiological study of torticollis.

Studied slow cortical potentials associated with voluntary movements ("readiness potential", RP and "motor potential", MP) and discrimination tasks (contingent negative variation", CNV, "expectancy wave", EW) in 11 normal subjects and 11 patients with torticollis (every person going through 2 experimental sessions). The following was found: 1. In torticollis-patients, the RP appeared as a complex, irregular wave with low--voltage, ataxic course. 2. The mean amplitude of the RP was significantly lower in the torticollis-group than in the control group. 3. The mean amplitude of the late positive component P2 of the MP was also significantly lower in torticollis-patients than in controls. 4. The CNV showed a high-voltage course in patients with torticollis. 5. The mean amplitude of the CNV was significantly higher in torticollis--patients than in controls. 6. The mean amplitude of the late positive component P2 after the resolution of the discrimination task was also higher in the torticollis--group than in the control group, but the difference did not reach the level of significance.--This marked dissociation between the adynamic RP-MP field generators and the very responsive CNV field generators is being interpreted partly as an electrophysiological symptom of impairment in programming and performance of sequential motor acts (in the case of the RP-MP), partly as a disruption of the control of emotional and affective processes, as well as non-specific (extralemniscal) afferences (in the case of the CNV) resulting from a basal ganglia lesion in torticollis.

Adolescent↗

[Acquired torticollis in hospitalized children].

Torticollis results from various pathological mechanisms, and its elucidation depends on identifying diseases of musculoskeletal, neural and ocular tissues. This study characterized the underlying diseases of children hospitalized with torticollis, excluding congenital torticollis. Records of 36 children with torticollis seen during 4 years were reviewed and categorized according to presumed etiology. Most could be classified into 2 categories: in 39% it was due to trauma and in 36% to upper respiratory tract infection. Most girls were in the first group and most boys in the second group. There were 3 cases of ocular torticollis due to superior-oblique muscle palsy, 1 with a post-burn eschar, 2 with neurological disorders (intramedullary cervical astrocytoma and leukodystrophy with macrencephaly), and in 3 no associated cause was found. There was a clear seasonal trend with 58% of cases presenting from November through February, 33% from April through July, and the rest, of neurological or ocular origin, during the rest of the year. In cases of post-traumatic torticollis 21% had neurological symptoms such as weakness of the limbs, headaches or incontinence. Only a few had prior upper respiratory tract infection. All children whose torticollis was assigned to infection had had fever. Only 8% had had neurological complaints or vomiting, half of whom presented with fever exceeding 37.5 degrees C. 46% had restriction of movement and 38% had tenderness. In over 60% of those in this group there were signs of an upper respiratory tract infection, such as lymphadenopathy or a white blood cell count exceeding 15,000/microliter, 3 patients with recurrent torticollis were diagnosed as having severe neurological diseases. Mean hospitalization time was 4 days (range 1-28). Hospitalization periods were similar for all kinds of patients and treatment by traction or fixation did not affect this period.

Child↗

The aetiological relationship between congenital torticollis and obstetrical paralysis.

A statistical study was done to determine the incidence of congenital torticollis. The aetiological relationship between torticollis and obstetrical paralysis was analysed in an attempt to clarify the possible cause of torticollis. There were forty-three cases of torticollis in the 2,160 babies delivered at the Otsu Red Cross Hospital from 1978 to 1981. The overall incidence of torticollis was 1.9%; 1.2% in cephalic presentations, 6.5% in double breech presentations, 4.2% in footling presentations and 34.0% in single (frank) breech presentations. In addition, forty-three cases of obstetrical paralysis treated in Kyoto University Hospital from 1961 to 1981 were reviewed. Torticollis was found in 26% of the cephalic presentations (6 out of 23) and in 80% of the breech presentations (16 out of 20) respectively. These findings suggest that a foetal posture with both the knees and the cervical spine extended seems to be closely related to the development of torticollis, which indicates that a direct cause may be stretching of the sternocleidomastoid muscle during delivery.

Breech Presentation↗

Congenital torticollis in association with craniosynostosis.

The incidence of congenital torticollis in association with plagiocephaly is 1 in 300 newborns, with the torticollis resulting from pathologically sustained contraction of the sternocleidomastoid. Such conditions as facial asymmetries, craniovertebral anomalies, cervical hemivertebra, and mono- or polydysostoses may also be associated with torticollis diagnosed during the neonatal period. With particular reference to synostotic (coronal and/or lambdoidal) plagiocephaly, a clear distinction is made in this paper between posterior neurocranial flattening secondary to the sustained rotation of the skull resulting from torticollis and that seen in synostotic plagiocephaly. The rarity of torticollis with sustained contraction of the sternocleidomastoid muscle relative to the frequency of occipital-parietal flattening in newborn kept in the supine position has not been discussed in the literature and is therefore of clinical importance. In light of the fact that the prognosis and, consequently, the treatment plan vary directly with the presence or absence of synostoses, clinical evaluation also includes cephalometrics, plain skull X-rays, and CT imaging. If the torticollis is associated with neurocranial deformity but synostosis is absent, cervical traction and physiotherapy resolve the symptoms. When, however, the clinical picture is complicated by synostotic plagiocephaly, corrective surgery is necessary, though cervical traction and physiotherapy are essential to provide early and complete cure of the torticollis.

Craniosynostoses↗

Frequency analysis of EMG activity in patients with idiopathic torticollis.

The pathophysiology of idiopathic dystonic torticollis is unclear and there is no simple test that confirms the diagnosis and excludes a psychogenic or voluntary torticollis in individual patients. We recorded EMG activity in the sternocleidomastoid (SCM) and splenius capitis (SPL) muscles of eight patients with rotational torticollis and eight age-matched controls, and analysed the signals in the frequency and time domains. All control subjects but one showed a significant peak in the autospectrum of the SPL EMG at 10-12 Hz, which was absent in all patients with torticollis. Conversely, patients with torticollis had evidence of a 4-7 Hz drive to the SPL and SCM that was absent in coherence spectra from controls. The pooled cumulant density estimates revealed a peak in both groups, and within the patient group there was a second narrow subpeak with a width of 13 ms. The activity in the SCM and SPL was in phase in the patients but not in the controls. The lack of any phase difference and the suggestion of short-term synchronization between SCM and SPL are consistent with an abnormal corticoreticular and corticospinal drive in dystonic torticollis. Clinically, the pattern of SPL EMG autospectra and of SCM-SPL coherence may provide a sensitive and specific feature distinguishing dystonic from psychogenic torticollis.

Electromyography↗

Diagnosis and management of torticollis.

Clearly, torticollis may stem from a variety of factors involving a number of primary structures -- bones, ligaments, muscles, and the nervous system. The crucial point is to differentiate torticollis that may lead to structural changes from that of a compensatory or functional nature. The key criterion is whether torticollis persists or resolves. This can be readily determined by treating torticollis initially with head halter traction of three to four pounds' weight and observing whether it resolves in five of seven days. If the torticollis and related symptoms persist beyond this time, the physician should consider possible lesions other than common rotary subluxation. Diagnosis of unresolved torticollis requires extensive and often repetitive investigation procedures. Perseverance in the diagnosis and management of persistent torticollis is a commendable virtue.

Adolescent↗

[Torticollis as a cause of consultation in neuropediatrics].

INTRODUCTION AND OBJECTIVE: Torticollis is a very non-specific symptom occurring in different conditions and may therefore be the reason for consultation in many specialties including neuropaediatrics. Analysis of torticollis as a cause for consultation in neuropaediatrics may contribute to the establishment of a suitable strategy for diagnosis. PATIENTS AND METHODS: We review, from the diagnostic point of view, the clinical histories of cases in which the reasons for consultation included torticollis. These cases were included in the database of all the patients assessed by the neuropaediatric department of the Hospital Miguel Servet in Zaragoza between May 1990 and February 1999. RESULTS: Of the 4,138 new patients evaluated during the period studied, in 60 patients torticollis was either the sole symptom or was one of the symptoms leading to consultation. The diagnoses established were: 30 congenital torticollis (50%), 6 secondary to space-occupying intracranial lesions (10%), 5 benign paroxystic torticollis (8.3%), 4 post-traumatic, 3 secondary to ocular disorders, 3 Sandifer syndrome, 1 focal dystonia of the neck, 1 secondary to a submandibular adeno-phlegmon, 1 secondary to an epidural hematoma of the cervical spine, 1 to encephalomyelitis and 1 to spondylodiscitis, with 4 cases unclassified. CONCLUSIONS: The clinical history, physical examination and follow-up of the course of the disorder orientate or permit the diagnosis to be established in many cases of torticollis. The indications for complementary investigations, particularly neuroimaging, should be considered individually in each case.

Adolescent↗

Natural history of adult-onset idiopathic torticollis.

The rates of spontaneous remission and progression of dystonia to other sites were studied in 72 patients who first presented with adult-onset torticollis, and who were followed up for a mean of 7.7 years. Dystonia had progressed to sites other than the neck (mainly the face and upper limbs) in 23 patients (32%). The latter cases were not differentiated from those with isolated torticollis in terms of any of the demographic or clinical features studied, although they tended to have suffered from torticollis longer. Fifteen patients (20.8%) had experienced a spontaneous remission of their torticollis, which was sustained for a median period of 3 years in 9 cases (12.5%). Eighty-seven percent of the 15 remissions had occurred during the first 5 years of the illness. In the 9 cases with sustained remission, the duration of torticollis before spontaneous remission was significantly longer and remission had mostly occurred after 2 years of illness compared with the 6 who had relapsed. The 15 cases with spontaneous remission tended to have an earlier age of onset compared with those with no remission. Sixty-five percent of cases were correctly classified on the basis of age at onset, which emerged as the only salient variable in the discrimination of the 15 patients with spontaneous remission from the 57 without spontaneous remission. Age at onset, form of torticollis, gender, and direction of head deviation resulted in a correct classification rate of 70%, in the discrimination of the 9 cases with sustained remission from those with no remission.(ABSTRACT TRUNCATED AT 250 WORDS)

Adult↗

Torticollis secondary to posterior fossa and cervical spinal cord tumors: report of five cases and literature review.

Torticollis is either congenital or acquired in childhood. Acquired torticollis is not a diagnosis but rather a sign of an underlying disorder. The causes of acquired torticollis include ligamentous, muscular, osseous, ocular, psychiatric, and neurologic disorders and may be a symptom of significant abnormalities of the spinal cord and brain, such as spinal syrinx or central nervous system neoplasia. Torticollis is rarely considered to be an initial clinical presentation of posterior fossa and cervical spinal cord tumors. We report five cases of pediatric tumors with torticollis at the onset: an astrocytoma originating from the medulla oblongata, another presumptive astrocytoma of the spinal cord located between C1 and C6 cervical vertebrae (not operated), an ependymoma located throughout the whole cervical spinal cord extending into the bulbomedullary junction, an astrocytoma originating from the bulbus and extending into the posterior fossa, and another case of a eosinophilic granuloma located extradurally through the anterior and posterior portions of the vertebral bodies from C3 to C7 producing the collapse of the sixth cervical vertebra. All five cases were seen in children, aged between 3 and 12 years. All these cases reflect the misinterpretation of this neurological sign and the lack of association with the possibility of spinal or posterior fossa tumor. This delay in the diagnosis of these diseases led to progressive neurological deterioration and to the increase in the tumor size, which made surgical intervention difficult and the prognosis unfavorable. Although torticollis secondary to tumors is rarely seen, it is necessary to be kept in mind in the differential diagnosis.

Astrocytoma↗

Psychosocial factors and depression in torticollis.

Sixty-seven patients suffering from spasmodic torticollis, a neurological disorder in which involuntary contractions of the head produce an abnormal head posture, completed a booklet of questionnaires aiming to assess psychosocial adjustment. Twenty-four per cent of the patients were moderately to severely depressed. Overall, as a sample the patients were not acceptant of or adjusted to their illness. Cognitive and emotion-focused strategies were slightly more widely used than instrumental ways of coping. Both adaptive and maladaptive coping strategies were more frequently used by female than male sufferers. The number of individuals who were potential sources of social support was not high (mean = 2.7, SD = 1.2). The patients were, however, generally satisfied with the available support. Self-depreciation, lack of control over head position/movement and the resulting disability in activities of daily living, lack of satisfaction with available social support, and the use of maladaptive coping strategies accounted for 75% of the variance of depression in torticollis. Self-depreciation, which was the most salient predictor of depression in torticollis, accounting for 59% of its variance, was itself shown to arise from the patient's negative body concept relating to the postural abnormality of the head. As the severity of illness and resulting disability contributed to depression, successful symptomatic treatment of torticollis would be expected to ameliorate mood. However, as the currently available medical treatments are not effective for all treated cases, and as the self-depreciation resulting from a negative body concept formed the core of depression in torticollis, cognitive-behavioural therapy could improve the quality of life of the depressed torticollis patient.

Adaptation, Psychological↗

Atlantoaxial rotatory fixation-subluxation revisited: a computed tomographic analysis of acute torticollis in pediatric patients.

STUDY DESIGN: Cross-sectional clinical and radiologic study with a normal control group. OBJECTIVES: To compare the range of motion of the atlantoaxial joint in patients with acute torticollis with those of normals as measured from computed tomography scans, to look for the existence of atlantoaxial rotatory fixation in any position (subluxation or normal range of motion) in this group of patients, and to clarify the definition of atlantoaxial rotatory subluxation by measuring the atlantodental interval and analyzing the location of the center of rotation in patients as well as normal controls. SUMMARY OF BACKGROUND DATA: Although acute acquired torticollis is usually termed atlantoaxial rotatory subluxation or atlantoaxial rotatory fixation, the radiologic definition of these conditions is not clear. PATIENTS AND METHODS: Thirty-three consecutive pediatric patients (average age 8.5 years, range 2-18 years) with acute acquired torticollis were analyzed. All were neurologically intact. Anteroposterior and lateral radiographs were obtained in all atlantoaxial computed tomography scans in 31 patients (dynamic in 23 and static in 8). Twelve age-matched patients with normal cervical spines were also analyzed with dynamic computed tomography as normal controls. Atlantoaxial rotatory subluxation, atlantoaxial angle, center of rotation, and presence of atlantoaxial rotatory fixation were analyzed in each computed tomography. All patients were treated conservatively. Eight had control dynamic computed tomography scans at the end of the treatment. RESULTS: All patients had atlantoaxial rotatory subluxation <or=3 mm. On dynamic computed tomography, the range of atlantoaxial rotation was 30.4 degrees (range 11-54 degrees) toward deformity and 28.3 degrees (range 18-54 degrees) away from deformity (P = 0.333). Atlantoaxial rotatory fixation was not noted in any of the patients. The same measurement for the normal control group was 28 degrees (range 5-41 degrees) (P = 0.770). Of the eight patients with repeat control computed tomography, the atlantoaxial rotatory subluxation was 26 degrees before and 29 degrees after treatment (P = 0.691 to right and P= 0.199 to left). The center of rotation was within dens in 15 of 19 patients, outside dens in 2 of 19, and undetectable in 2 of 19. In the control group, it was within dens in 8 of 11, outside dens in 2 of 11, and undetectable in 1 of 11. All patients were symptom free at the end of the conservative treatment. CONCLUSION: We could not demonstrate the presence of atlantoaxial rotatory subluxation or atlantoaxial rotatory fixation in our series of 33 consecutive pediatric patients with acute torticollis. Our findings suggest that the existence of these phenomena are doubtful, although not associated with acute acquired torticollis. Acute acquired torticollis is not necessarily the sign of a pathologic condition of the atlantoaxial joint. Finally, it is probably not necessary to obtain computed tomography scans (static or dynamic) in this group of patients at the time of presentation.

Acute Disease↗

The Ponseti method for treatment of congenital club foot.

PURPOSE OF REVIEW: This review of the Ponseti technique for the treatment of congenital club foot covers a topic of recently renewed interest. Pediatric orthopedists and parents have become increasingly enthusiastic about the success of this technique, which has been practiced continuously at the University of Iowa since 1948 but only recently become widely utilized. RECENT FINDINGS: Current literature has emphasized the reproducibility of these excellent results among multiple centers throughout the world. In addition, the role of the Internet, and accessibility of medical information have been central elements in the development of this phenomenon. SUMMARY: The current research should convince physicians that all children should be managed by the Ponseti technique at the outset. This research should also reassure physicians and parents that the overwhelming majority of children with club feet can be successfully managed, without the need for major reconstructive surgery.

Braces↗

Selective resection and denervation of cervical muscles in the treatment of spasmodic torticollis: results in 60 cases.

The author presents a series of 60 patients with torticollis who were treated successfully by operation. He describes the clinical types of torticollis: rotational torticollis, retrocollis, antecollis, lateroflexional torticollis, and combined types. The muscular mechanism of torticollis is detailed, and the operative technique of resecting or denervating only the principal or motive muscles is explained and illustrated for each type of torticollis. The results in this series were very good: 83.3% recovered completely or were improved markedly, and there were no complications.

Adolescent↗