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PubMed1981

The universal, muscular chain reaction, muscle spasm, torsions, ruptures and extravasations. Chameleons of pathology and some manifestations of simple muscular disorders.

Most bodily functions require the coordinated actions of complementary and supplementary paired muscle groups. Where this essential muscular cooperation is lacking, hollow organs may burst and others become literally screwed up, giving rise to many similar spastic diseases such as Torticollis, Twisted ovarian cyst, Torsion of the Testis, Volvulus of the intestines, Varicose Veins, Megacolon, Aortamegaly, Scoliosis, Erb's Palsy, Peyronie's Disease, Main-en-Griffe, Undescended Foot (Pes Cavus), Talipes, Strabismus. Spasm is "panenepidemic" and unclassified examples of Torsion Dystonia and Dyskinesia really are as common as debt and taxes.

Female↗
PubMed1991

Altered behavioral responses mediated by serotonin receptors in the genetically dystonic (dt) rat.

The genetically dystonic (dt) rat is an animal model of dystonia that displays sustained abnormal movements that include: torticollis, clasping of the hindlimbs, rigidity of the limbs, and contortions of the trunk. Since serotonin (5-HT) has been shown to be involved in some animal models of movement disorders, the functional responsiveness of the 5-HT system in dt rats and phenotypical normal littermates was examined by administering 5-HT agonists selective for different receptor subtypes and observing behavioral responses associated with the activation of specific 5-HT receptor subtypes. The dt rats were 6-fold more sensitive to the ability of the 5-HT1A agonist 8-OH-2-(di-n-propylamino)tetralin (8-OH-DPAT) to produce the 5-HT behavioral syndrome. The dt rats demonstrated a diminished head-shaking response following administration of the 5-HT2 agonist 1-(2,5-dimethoxy-4-bromophenyl)-2-aminopropane (DOB). However, the dt rats also displayed significantly fewer head shakes following mechanical stimulation of the aural pinnae. The inability of the dt rats to demonstrate head-shaking behavior following stimulation of 5-HT2 receptors is probably due to the dt rat's difficulty in producing the motor responses involved in this behavioral response and do not reflect alterations in 5-HT2 receptor sensitivity. These results suggest that the 5-HT system, particularly 5-HT1A receptors, may have an integral role in the abnormal movements displayed by the genetically dystonic rat and movement disorders in general.

Animals↗
PubMed1996

Infantile spasms associated with proximal duplication of chromosome 15q.

We describe a case of infantile spasms associated with a chromosome abnormality (supernumerary inverted duplication of chromosome 15 [47,XX,+inv dup(15)]). The patient was nondysmorphic and presented with mild hypotonia and delay in acquisition of gross motor milestones before the diagnosis of seizures at age 7 months. Additional features included unilateral sensorineural deafness and torticollis. Molecular cytogenetic studies confirmed that the patient has a large inv dup(15). Inv dup(15) chromosomes are variable with respect to the size and genetic composition of the chromosome and in their phenotypic effects. Patients with small inv dup(15s) may have no phenotypic abnormalities, whereas patients with large inv dup(15s) may have multiple abnormalities. ACTH therapy resulted in prompt remission of seizures and resolution of EEG abnormalities. This is the second report of a patient with IS and a supernumerary inv dup(15). Several genes code for neurotransmitter receptor subunits located in the duplicated region of chromosome 15, and abnormal dosage of these genes may be involved in the genesis of seizure activity in carriers of the inv dup(15). Chromosome analysis may lead to a specific diagnosis in infants with unexplained infantile spasms.

Adrenocorticotropic Hormone↗
PubMed2006

Abdominal pain with rigidity secondary to the anti-emetic drug metoclopramide.

We report a case of abdominal pain with rigidity, mimicking an acute abdomen, caused by metoclopramide, a common anti-emetic drug. Extrapyramidal symptoms are commonly reported side-effects of this medication. They generally include involuntary movements of limbs, torticollis, oculogyric crisis, rhythmic protrusion of tongue, trismus, or dystonic reactions resembling tetanus, etc. Abdominal rigidity due to this medication, resembling an acute abdomen, has not been reported previously. This case report illustrates the importance of considering medication side-effects when evaluating a patient with abdominal pain and rigidity.

Abdomen, Acute↗
PubMed2005

A case report of manipulation under anesthesia of posttraumatic type II occipital-atlantoaxial rotatory subluxation in a 4-year-old girl.

OBJECTIVE: To discuss a case of occipital-atlantoaxial rotatory subluxation successfully treated with manipulation under general anesthesia. CLINICAL FEATURES: A 4-year-old girl presented to the Taichung Veterans General Hospital with acute torticollis and neck stiffness for 1 week after she had fallen. Although some nonsteroidal anti-inflammatory drugs had been prescribed for her, her neck still tilted to the right side and her chin inclined to the left side. There were no neurological signs, no significant pain if she did not move her neck and head, and no muscular hypertonocity. There was painful guarding in the right sternocleidomatoid muscle when manipulation was attempted. Three-dimensional computer tomography revealed uneven joint space between the C1 anterior arch and odontoid process and confirmed a type II atlantoaxial rotatory subluxation. INTERVENTION AND OUTCOME: Manipulation under anesthesia was performed by a medical doctor trained in manual therapy. The low-velocity, right rotational manipulation applied to the occiput included axial traction. The neck symptoms were relieved immediately after treatment. CONCLUSION: Under general anesthesia, manipulation may be a good method for treating noncomplicated type II atlantoaxial rotatory subluxation. Additional investigations may be necessary to evaluate the treatment effect.

Accidental Falls↗
PubMed2006

Fatigue in the sternocleidomastoid muscle and hip dysplasia: a surface electromyographic assessment in adult women.

OBJECTIVE: The aim of this study is to assess a possible relationship between unilateral hip dysplasia and sternocleidomastoid muscle function during a fatiguing task. METHODS: In this patient-control, cross-sectional study, 11 female patients (aged 37-63 years) with unilateral coxoarthrosis subsequent to hip dysplasia and 9 female control subjects matched for age were assessed. Surface electromyography of the right and left sternocleidomastoid muscles was measured in the women performing a standardized endurance test. The median power frequency was calculated at the beginning (T0) and at the end of the task (T1, endurance time). For each subject and muscle, percentage decrements in the median power frequency were computed at T1. RESULTS: Endurance time ranged between 15 and 125 s (control group), and 10 and 200 s (patient group), without a significant difference between the 2 groups (Mann-Whitney test, P > .05). In the patient group, no significant differences in the median power frequency between the ispilateral to coxoarthrosis and contralateral sides were found (Wilcoxon test, P > .05). The percentage decrements of the median power frequency in the control (right and left side pooled) and patient group did not significantly differ either in the ispilateral to coxoarthrosis and contralateral sides (Mann-Whitney test, P > .05). CONCLUSIONS: The lack of significant differences between the sternocleidomastoid muscles of healthy women and those of women with hip dysplasia does not support the presence of a common noxa causing both congenital muscular torticollis and hip joint alterations. Alternatively, the coexistence of the 2 alterations in infancy may disappear during adult life.

Adult↗
PubMed2006

Intrafamilial phenotypic and genetic heterogeneity of dystonia.

Most cases of early-onset primary torsion dystonia are caused by the same 3-bp (GAG) deletion in the DYT1 gene. We describe a large Serbian family with significant intrafamilial variability of the DYT1 phenotype, from asymptomatic carrier status to late-onset focal, and generalized jerky dystonia. Seven mutation carriers (six proven by direct analysis and one by inferred haplotype) were identified, but only two of them were affected by dystonia (penetrance reduced to 29%). In addition, three GAG-deletion-negative family members also developed dystonia (two multifocal dystonia and one torticollis), suggesting that their involuntary movements are due to some other etiological factor(s) (i.e., another dystonia gene), or may be psychogenic.

Adult↗
PubMed2005

Passive muscle stiffness may be influenced by active contractility of intramuscular connective tissue.

The article introduces the hypothesis that intramuscular connective tissue, in particular the fascial layer known as the perimysium, may be capable of active contraction and consequently influence passive muscle stiffness, especially in tonic muscles. Passive muscle stiffness is also referred to as passive elasticity, passive muscular compliance, passive extensibility, resting tension, or passive muscle tone. Evidence for the hypothesis is based on five indications: (1) tonic muscles contain more perimysium and are therefore stiffer than phasic muscles; (2) the specific collagen arrangement of the perimysium is designed to fit a load-bearing function; (3) morphological considerations as well as histological observations in our laboratory suggest that the perimysium is characterized by a high density of myofibroblasts, a class of fibroblasts with smooth muscle-like contractile kinetics; (4) in vitro contraction tests with fascia have demonstrated that fascia, due to the presence of myofibroblasts, is able to actively contract, and that the resulting contraction forces may be strong enough to influence musculoskeletal dynamics; (5) the pronounced increase of the perimysium in muscle immobilization and in the surgical treatment of distraction osteogenesis indicates that perimysial stiffness adapts to mechanical stimulation and hence influences passive muscle stiffness. In conclusion, the perimysium seems capable of response to mechanostimulation with a myofibroblast facilitated active tissue contraction, thereby adapting passive muscle stiffness to increased tensional demands, especially in tonic musculature. If verified, this new concept may lead to novel pharmaceutical or mechanical approaches to complement existing treatments of pathologies which are accompanied by an increase or decrease of passive muscle stiffness (e.g., muscle fibroses such as torticollis, peri-partum pelvic pain due to pelvic instability, and many others). Methods for testing this new concept are suggested, including histological examinations and specific in vitro contraction tests.

Elasticity↗
PubMed2006

Obsessive-compulsive disorder and executive deficits in two patients with primary dystonia.

In spite of the high prevalence of behavioral and cognitive disturbances found in most basal ganglia disorders and attributed to fronto-striatal dysfunction, the existence of psychiatric and cognitive symptoms in patients with primary dystonia remains controversial. We present a 42-year-old female with primary writer's cramp and obsessive-compulsive disorder (OCD) and a 59-year-old male with Meigs syndrome, idiopathic torticollis and OCD. Both patients had mild executive dysfunction. The coexistence of psychiatric, cognitive and motor symptoms of different intensity may be explained by variable dysfunction on different frontal-striatal loops, as proposed by the open interconnected model of fronto-striatal circuits.

Adult↗
PubMed2006

Acute dystonia after initial doses of ziprasidone: a case report.

We present the case of an 18-year-old man, who was admitted with an obsessive compulsive disorder (OCD) since 17 years old. He had symmetry and contamination obsessions and cleaning compulsions, and had been treated with fluoxetine 40 mg/day for 2 months. The patient was not taking additional psychotropic medication. We made ziprasidone augmentation to the treatment. First day, ziprasidone 40 mg bid was applied. Four hours after the evening dose, the patient complained about distress. He demonstrated a notable torticollis and dystonic posture to his left side. Intramuscular biperiden lactate (5 mg) was given, and dystonic symptoms resolved within 30 min. Ziprasidone is a new atypical antipsychotic with reported low potential for extrapyramidal side effects. To our knowledge, acute dystonic reaction with initial doses of ziprasidone in adult has not been reported yet. This case report describes a male patient with OCD developed acute dystonia shortly after initial doses (80 mg/day) of ziprasidone and discusses precautions in antipsychotic use.

Adolescent↗
PubMed2007

Acute dystonia resulting from abrupt bupropion discontinuation.

Acute dystonia is commonly associated with high-potency antipsychotics. Some cases of acute dystonia had been reported to be associated with antidepressant. However, only few reported cases are related to bupropion. As reported herein, the patient with major depression suffered from acute dystonia twice, which resulted from abrupt bupropion discontinuation. The first episode occurred when medicament was shifted from bupropion to duloxetine abruptly. The patient was requested with nothing per mouth (NPO) due to panendoscopic examination. Therefore, the second one emerged after the patient was suspended from two doses of bupropion. The symptoms of dysphagia, trismus and torticollis in these two episodes were resolved after bupropion reinstitution or biperiden injection. So far as we know by our documents, this is the first report concerning an acute dystonia resulting from bupropion discontinuation.

Antidepressive Agents, Second-Generation↗
PubMed2006

Unusual presentations of craniovertebral junction tuberculosis: a report of 2 cases and literature review.

BACKGROUND: CVJ tuberculosis is a described entity requiring challenging ways of management. Severe neck pain, causing restricted neck movements and torticollis, is a characteristic presentation of neurologically asymptomatic suboccipital Pott's disease. CASE DESCRIPTION: Two patients with unusual CVJ tuberculosis form the basis for the present communication. The first patient presented with tubercular otitis media, causing progressive erosion of the petrous part of temporal bone, and destruction of the occipital condyle, along with the lateral mass of atlas, leading to CVJ instability. This is a first report of such a presentation, according to our knowledge. Detailed bony architectural destruction demonstrable on CT scan has been described. The second patient, with CVJ tuberculosis, presented with skull base syndrome and with multiple cranial nerve palsies. Both patients were managed without surgical intervention and showed clinical and radiological recovery. CONCLUSION: In such patients with unusual clinical presentations, histopathologic examination is necessary to arrive at a correct diagnosis. The management of patients with tubercular involvement of CVJ remains controversial. In the present communication, both the patients were managed successfully with full dose of antitubercular drugs and immobilization.

Adult↗
PubMedDate not supplied

Skew deviation revisited.

Skew deviation is a vertical misalignment of the eyes caused by damage to prenuclear vestibular input to ocular motor nuclei. The resultant vertical ocular deviation is relatively comitant in nature, and is usually seen in the context of brainstem or cerebellar injury from stroke, multiple sclerosis, or trauma. Skew deviation is usually accompanied by binocular torsion, torticollis, and a tilt in the subjective visual vertical. This constellation of findings has been termed the ocular tilt reaction. In the past two decades, a clinical localizing value for skew deviation has been assigned, and a cogent vestibular mechanism for comitant and incomitant variants of skew deviation has been proposed. Our understanding of skew deviation as a manifestation of central otolithic dysfunction in different planes of three-dimensional space is evolving. The similar spectrum of vertical ocular deviations arising in patients with congenital strabismus may further expand the nosology of skew deviation to include vergence abnormalities caused by the effects of early binocular visual imbalance on the developing visual system.

Diplopia↗
PubMed1974

Lymphoepithelioma in childhood.

Nine black children with lymphoepithelioma, a rare malignancy of childhood, are the subject of this report. Unique clinical features included tender cervical lymphadenopathy with torticollis, trismus, epistaxis, and change in voice quality. A nasopharyngeal mass was demonstrable in seven children on careful examination, but none was resectable. Treatment with radiation alone or radiation plus cyclophosphamide resulted in complete tumor regression in eight of the nine children. Local recurrence or distant metastases occurred in four within 10 months, two of whom responded to additional radiation plus cyclophosphamide or adriamycin. At present, four children are alive without disease for periods of seven to 78 months, two are alive for seven to 53 months and are in remission from recurrent disease, and three have died with distant metastases. Freedom from disease for one year was associated with a favorable prognosis. Adjuvant chemotherapy appears warranted in view of the high incidence of local recurrence and distant metastases.

Adolescent↗
PubMed1993

Posttraumatic cervical dystonia.

Posttraumatic cervical dystonia has been described as a distinct syndrome with some similarities to idiopathic nontraumatic cervical dystonia (torticollis). We describe five patients in whom cervical dystonia developed immediately after relatively mild trauma to the neck. Four of the five patients had persistent contractions of all cervical muscles including the trapezius muscles, which almost completely prevented motion of the neck and resulted in muscle hypertrophy. The condition persisted unabated in all patients for the entire period of follow-up (duration, 1 1/2 to 3 years). Pharmaceutical interventions, which had been used previously for idiopathic nontraumatic cervical dystonia, failed to benefit these patients. Two patients who received injections of botulinum toxin had no more than mild benefit. Selective denervation was inapplicable because of the widespread involvement of all cervical muscles in all but one patient. Physical therapy was essentially ineffective. Because of the unusual features and possible medicolegal setting, clinicians may tend to diagnose this condition as a psychogenic disorder or litigation-oriented behavior. The clinical picture, however, is consistent with an organic dystonia that may render the patient functionally and occupationally disabled.

Accidents, Traffic↗
PubMed1998

Cervical deformity: rationale for selecting the appropriate fusion technique (anterior, posterior, and 360 degree).

This article examines cervical deformities and their treatments, such as iatrogenic deformities, posttraumatic deformities, ankylosing spondylitis, rheumatoid arthritis, degenerative subaxial spondylolisthesis, myopathy, infectious spondylitis, and tumors. Congenital scoliosis and kyphosis and torticollis and rotatory atlanto-axial subluxation also are discussed.

Diskectomy↗
PubMed1997

Pharmacologic characterization of botulinum toxin for basic science and medicine.

The use of Botulinum neurotoxin (BoNT) is increasing in both clinical and basic science. Clinically, intramuscular injection of nanogram quantities of BoNT is fast becoming the treatment of choice for a spectrum of disorders including movement disorders such as torticollis, blepharospasm, Meige Disease, and hemifacial spasm (Borodic et al., 1991, 1994a; Jankovic and Brin, 1991; Clarke, 1992). Neuroscientists are using BoNTs as tools to develop a better understanding of the mechanisms underlying the neurotransmitter release process. Consequently, our ability to accurately and reliably quantify the biologic activity of botulinum toxin has become more important than ever. The accurate measurement of the pharmacologic activity of BoNTs has become somewhat problematic with the most significant problems occurring with the clinical use of the toxins. The biologic activity of BoNTs has been measured using a variety of techniques including assessment of whole animal responses to in vitro effects on neurotransmitter release. The purpose of this review is to examine the approaches employed to characterize, quantify and investigate the actions of the BoNTs and to provide a guide to aid investigators in determining which of these methods is most appropriate for their particular application or use.

Animals↗
PubMed2001

Symptomatic cavernous hemangioma of the occipital condyle treated with methacrylate embolization.

BACKGROUND: Cavernous hemangiomas of the cranial base are rare tumors. No case of symptomatic intraosseous angioma affecting the occipital condyle has been reported. This particular case was treated with surgical embolization using acrylic resin. CASE DESCRIPTION: A 20-year-old man with a 1-year history of neck pain and torticollis was referred to our hospital. Neuroradiological examination revealed the typical picture of an intraosseous cavernous hemangioma located in the right occipital condyle. The patient was operated through a suboccipital approach. Biopsy and direct embolization with methacrylate was performed. The definitive pathological diagnosis confirms the neuro-radiological suspicion of intraosseous cavernous hemangioma. The follow-up of the patient (4 years) revealed no recurrence of pain or abnormal posture. CONCLUSIONS: A rare case of cranial base cavernous hemangioma is reported. Methacrylate embolization can be a good option for the treatment of this uncommon lesion.

Adult↗