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PubMedDate not supplied

[Intraoperative circulatory collapse due to a coronary spasm following successful coronary revascularization].

Coronary artery spasm is recognized cause of angina and circulatory collapse during the operative and early postoperative period following cardiopulmonary bypass for coronary artery surgery. We present our experience with hemodynamical collapse during cardiopulmonary bypass weaning, which were refractory to treatment with inotropics such as noradrenaline and adrenalin and vasodilators such as nitroglycerin. The electrocardiographic changes, poor answer to medical treatment following successful cardiopulmonary bypass for coronary artery surgery, good myocardial protection and complete revascularization suggested severe coronary artery spasm. We used sublingual nifedipine, achieving hemodynamical stability and stopping inotropic support and cardiopulmonary bypass. We review the literature about pathophysiologic mechanism and treatment of coronary artery spasm.

Coronary Disease↗
PubMed1993

[Successful documentation by coronary angiography of spontaneous simultaneous multivessel coronary spasm in a variant angina patient: a case report].

A 59-year old female who complained of chest discomfort was admitted to our hospital. Electrocardiogram (ECG) on admission and treadmill exercise test showed negative for ischemia. She underwent coronary arteriography. Initial angiography showed there was no significant coronary arterial stenosis. However, when we were preparing the spasm provocation test, she complained of the same kind of chest discomfort as she had felt before. We found that ST segment was elevated in both the anterior and inferior leads on the ECG. Coronary arteriography showed that severe spasm occurred in both the left anterior descending artery (Seg. 6) and the right coronary artery (Seg. 1). Heart rate decreased and electromechanical dissociation occurred. She temporarily lost consciousness. After cardiopulmonary resuscitation, she recovered and the elevation of ST segment returned on the ECG. This is the first case report which has documented spontaneous simultaneous multivessel coronary spasm by coronary arteriography.

Angina Pectoris, Variant↗
PubMed1995

[Coronary artery spasm as a cause of perioperative myocardial infarction and stunned myocardium].

We report a male patient who had recurrent coronary artery spasm on withdrawal from cardiopulmonary bypass, which led to myocardial infarction and stunned myocardium. The spasm responded to conventional medication. Transesophageal echocardiogram showed no remarkable asynergy during the operation. However, MB fraction of creatine kinase was 132 IU.l-1 and electrocardiogram showed new Q waves. After stopping intravenous catecholamines, hypokinesis of the anteroseptal segment appeared in the echocardiogram. Spotty myocardial necrosis and stunned myocardium might have resulted from recurrent coronary artery spasm.

Aged↗
PubMed1995

[Spasm of normal or irregular coronary arteries. Long-term outcome of 277 patients].

The outcome of patients presenting with spasm of normal or subnormal coronary arteries is only known in small series of patients at medium-term. The authors reviewed the outcome of 277 successive patients over an average period of 86 +/- 43 months (12 to 174 months). There were 206 men and 71 women with a mean age of 54 +/- 9 years. Coronary angiography was performed in all cases and did not show any stenosis greater than 50%. Spasm was documented during coronary angiography in 157 cases (57%) by a positive Methergin test after coronary angiography in 113 cases (41%) and by an ECG recording of Prinzmetal angina in 7 cases (2.5%). Nearly all patients (264: 95%) were treated by calcium antagonists. At the end of follow-up, there were: 35 lost to follow-up (12.6%), 20 deaths (7.2%) of which 10 were cardiac (3.6%), 18 myocardial infarctions (6.5%): 11 had repeat coronary angiography which showed one or more new significant (> 70%) coronary lesions in all cases; 109 patients had persistence of chest pain (39%). The severity of symptoms in 52 cases (over one attack of chest pain per month) led to repeat coronary angiography which showed significant coronary disease in 19 cases; 95 patients (34%) were asymptomatic. A multivariate statistical analysis showed hypertension or subnormal appearances on the initial coronary angiography to be significant predictive factors for new coronary events (death, myocardial infarction or angina requiring repeat coronary angiography). The authors conclude that coronary spasm of angiographically normal or subnormal arteries is only well controlled at long-term in 39% of patients not lost to follow-up and is responsible for death or myocardial infarction in 11.6% of cases (nearly 1.5% per year).

Actuarial Analysis↗
PubMed1994

Infantile spasms: a pathophysiological hypothesis

Clinical and electroencephalogram (EEG) observations permit a hypothesis for the pathophysiology of infantile spasms. The triggering area(s) for spasms is probably cortical. Diffuse hyperexcitability of the immature cortex and/or diffusion of the epileptic process from a focal abnormality would allow development of hypsarrhythmia. Cortical maturation from posterior to anterior might account for the location of causative focal abnormalities. In cryptogenic West syndrome, the predominant pathophysiologic process may be diffuse cortical hyperexcitability, explaining the severe developmental deterioration at the onset of spasms. In symptomatic West syndrome with focal pathology, the initial deterioration is often less striking, suggesting that the predominant effect is diffusion of the epileptic process. The pattern of eventual cognitive deficits follows a similar pattern.

Journal Article↗
PubMed1998

[Use of the radial artery graft in coronary artery bypass grafting: harvesting technique and spasm prevention].

The use of radial artery (RA) in coronary artery bypass grafting (CABG) has been increasing recently as a revival. In this report, we describe several practical suggestions for improving patency rate of the graft. Between April of 1997 and February of 1998, 41 CABGs were performed using RA graft, totalling 56 anastomoses. The early patency rate of the graft has been 100% (graft: 38/38, anastomosis: 53/53). Harvesting technique: with the use of Harmonic Scalpel, it is possible to atraumatically harvest the vessel in a short time. Although longitudinal fasciotomy of the adventitia has been recently reported to be effective in releasing spasm, the nature of the vessel raise concern that the fasciotomy may even induce spasm. We hypothesize that leaving the adventitia intact, preserving vasa vasorum, rather than performing fasciotomy leads to improvement of long-term patency. Spasm prevention: we consider the body temperature to be the most important factor. Therefore, we utilize normothermic cardiopulmonary bypass (CPB). Another important factor is that the arterial CO2 is kept at a high level during CPB. For dilation of RA graft, milrinone is used instead of papaverine. For the intra- and postoperative management, intravenous continuous administration of diltiazem was changed to nicorandil. Technically, essential resolution for improvement of patency rate is either to allow for large proximal anastomosis, or to make sequential anastomosis with another coronary artery which has a good run off. For these purposes, the proximal anastomosis on the ascending aorta seems to have the advantage over placing it on ITA.

Adult↗
PubMed2026

RFX3 Pathogenic Variants as a Rare Cause of Infantile Epileptic Spasms Syndrome.

Regulatory Factor X3 (RFX3-OMIM#601337) encodes a transcription factor that is highly expressed in the human brain, particularly during neurodevelopment. It has been previously associated with neurodevelopmental disorders, including autism spectrum disorder (ASD), intellectual developmental disorder, and attention-deficit/hyperactivity disorder. However, the neurological and epileptic features remain poorly characterized, and no phenotype has yet been formally annotated in OMIM. Here, we report the second known case of Infantile Epileptic Spasms Syndrome (IESS) associated with RFX3 variants. The patient developed clusters of extensor spasms associated with eye deviation and achieved complete remission within two weeks following vigabatrin and ACTH therapy, remaining seizure-free thereafter. During follow-up, he presented with global developmental delay, ASD, and facial dysmorphisms. Genetic analysis by array comparative genomic hybridization identified a de novo heterozygous microdeletion of approximately 147 kb at 9p24.2, involving the initial exons of RFX3 (NM_134428). This case expands the clinical spectrum associated with RFX3 variants, supporting a potential role in IESS and early neurodevelopmental disruption. It highlights the relevance of including RFX3 in the genetic evaluation of patients with IESS and co-occurring neurodevelopmental disorders.

Humans↗
PubMed1986

Infantile spasms. Comparative trial of nitrazepam and corticotropin.

Fifty-two patients were enrolled in a four-week randomized multicenter study comparing nitrazepam and corticotropin in the treatment of infantile spasms. The drugs' efficacy was evaluated in 48 patients, all less than 2 years of age. Both treatments resulted in a statistically significant reduction in spasm frequency from that at baseline, but the difference between treatments was not significant. The number of patients who experienced side effects was similar in the two treatment groups, but the adverse effects encountered among the patients treated with corticotropin were qualitatively more severe and required the discontinuation of treatment in six patients.

Adrenocorticotropic Hormone↗
PubMed2002

Infantile spasms: West syndrome.

West syndrome is an epileptic syndrome with a devastating clinical course. In recent years, anatomic and functional neuroimaging studies have helped to diagnose the cause of the spasms in most children, but with little progress in improving the poor developmental outcome associated with this syndrome. This article cites 4 seminal observations regarding the clinical presentation, diagnosis, and treatment of infantile spasms.

History, 19th Century↗
PubMed2006

A comparative study of primary and secondary hemifacial spasm.

BACKGROUND: Hemifacial spasm (HFS) is a common movement disorder. OBJECTIVE: To evaluate possible differences in the demographic and clinical features between primary and secondary HFS. DESIGN: In-person interview using a standardized questionnaire to collect demographic and clinical data. SETTING: A multicenter study that included patients with HFS attending 3 Italian academic centers. Patients Two hundred fourteen patients with HFS. MAIN OUTCOME MEASURE: A complete neurological examination assessed the current muscle distribution of spasm and the presence of synkinetic movements between upper and lower facial muscles. RESULTS: The study sample comprised 214 patients with HFS, 81 men and 133 women, having a mean +/- SD age of 65.9 +/- 12.3 years; 164 patients were classified as having primary HFS and 50 patients (48 postparalytic and 2 symptomatic cases) were classified as having secondary HFS. Patients with primary and those with secondary HFS had similar mean +/- SD ages at onset (54.9 +/- 13.5 vs 57.0 +/- 12.8 years), male-female ratios (63:101 vs 18:32), right-sided-left-sided HFS (77:86 [1 bilateral] vs 21:28 [1 bilateral]), and frequencies of familial cases (2.9% vs 2.0%), respectively. Most patients (65.0%) with primary HFS had initial symptoms of periocular muscle contractions alone and had subsequent involvement of the lower facial muscles. Most patients (72.0%) with secondary HFS reported initial involvement of the upper and lower facial muscles simultaneously. Signs of synkinesis were present in primary (43.3%) and secondary (58.0%) HFS. CONCLUSIONS: Patients with primary and those with secondary HFS share common demographic and clinical features, including sex distribution, age at onset, affected side of HFS, synkinesis, and rarity of familial cases. Signs of synkinesis were present in significant proportions of patients with primary or secondary HFS. The 2 forms differed in clinical presentation.

Adolescent↗
PubMed1985

Hemifacial spasm treated with botulinum A toxin injection.

Fifteen patients with hemifacial spasm were treated with botulinum A toxin injections. All patients experienced relief from spasm, with the effect lasting an average of 12.2 weeks. Complications were tearing in four patients, inability to close the involved eye in three patients, corneal exposure in one patient, and ectropion in one patient. All complications were transient and deemed minor by the patients.

Adult↗
PubMed1999

Severe spasm of a radial artery coronary bypass graft during coronary intervention.

Coronary spasm is a well-recognized observation during coronary angiography. This report describes a patient who developed spasm of a radial artery bypass graft during coronary intervention. There are many reports on the angiographic follow-up to confirm the surgical results for patency in radial arterial conduits. We share our experience with this first case in the literature noted to have severe vasospasm during PTCA. Cathet. Cardiovasc. Intervent. 47:331-335, 1999.

Angioplasty, Balloon, Coronary↗
PubMed1999

Bilateral hemifacial spasm: a report of five cases and a literature review.

We describe five patients with bilateral hemifacial spasm evaluated in a Movement Disorders Clinic to illustrate the clinical characteristics and to draw attention to the differential diagnosis of this condition. All patients had unilateral onset followed by bilateral, asymmetric, and asynchronous facial contractions. The mean age of the patients (4 women and 1 man) was 70.6 years (range, 54-81 yrs), and the mean duration of symptoms was 17 years (range, 2-30 yrs). The facial twitching started in the left eyelid in all cases and the opposite side of the face began to twitch on the average 8.4 years (range, 0.2-15 yrs) later. Imaging studies revealed tortuous vertebrobasilar arteries in three patients. Four patients were successfully treated with botulinum toxin injections. Bilateral hemifacial spasm is a rare, peripherally induced disorder that must be differentiated from tics, dystonia including blepharospasm and other cranial dystonia, and other facial dyskinesias. Botulinum toxin injection appears to be the treatment of choice.

Aged↗
PubMed1999

Painful tonic/dystonic spasms in Sjogren's syndrome.

Three patients with Sjogren's syndrome are presented in whom frequent tonic/dystonic spasms of the limbs developed during the course of the illness. These patients' clinical findings suggested spinal cord involvement, a localization that was confirmed by magnetic resonance imaging in two patients. In one patient the painful movements responded to treatment with phenytoin and in one other to baclofen. Sjogren's syndrome should be considered in the differential diagnosis of conditions that produce tonic/dystonic limb spasms.

Adult↗
PubMed2005

Cri du chat syndrome and complex karyotype in a patient with infantile spasms, hypsarrhythmia, nonketotic hyperglycinemia, and heterotopia.

Seizures are rarely reported in association with deletion or duplication syndromes of the short arm of chromosome 5, or with chromosome 5 rings. We report on the clinical and cytogenetic findings in a girl with Cri du chat syndrome associated with complex abnormalities in chromosome 5, dysmorphic features, flexor infantile spasms, hypsarrhythmia, nonketotic hyperglycinemia, and heterotopia in her brain. Peripheral blood cytogenetic analysis indicates a mosaic karyotype with de novo deletion of varying amounts of 5p and pericentric inversion of the same chromosome 5. The deleted segment on 5p includes the region implicated in the catlike cry as well as sequences implicated in development of facial dysmorphism and mental retardation. This is the first case with Cri du chat syndrome associated with nonketotic hyperglycinemia, infantile spasms, hypsarrhythmia, and heterotopia.

Abnormalities, Multiple↗
PubMed1978

Infantile spasms in Down syndrome: a report of 5 cases and review of the literature.

Five patients with Down syndrome (DS) and infantile spasms were examined in a four-year interval. Previous reports of this association have been infrequent except during experimental treatment with 5-hydroxytryptophan, a precursor of serotonin. Malformations of the brain in DS are associated with an increased incidence of epilepsy, and both gross and cytoarchitectural anomalies have been described. The severity and distribution of these central nervous system anomalies presumably predispose to the infantile spasms. The contribution of altered serotonin metabolism is uncertain.

5-Hydroxytryptophan↗
PubMed1979

Choroid plexus papilloma and infantile spasms.

A 7-month-old infant had the infantile spasm syndrome, consisting of flexor and extensor spasms, developmental delay, and hypsarrhythmia. Corticotropin produced delay, and hypsarrhythmia. Corticotropin produced improvement in the clinical symptoms and reverted the generalized electroencephalographic abnormalities to more focal ones. Removal of a choroid plexus papilloma of the left lateral ventricle was followed by clinical recovery. One year later the child was normal developmentally and neurologically and was seizure free on anticonvulsant therapy.

Cerebral Ventricle Neoplasms↗
PubMed1980

Masticatory spasm in facial hemiatrophy.

Spontaneous spasms began in the left masseter muscle six years following the onset of facial hemiatrophy in a young woman. A dental procedure had preceded the original facial wasting by four weeks. The electromyographic findings were similar to those described in hemifacial spasm and consisted of brief bursts of one to four morphologically different potentials, each firing at rates of up to 200 Hz. The findings demonstrate that spontaneous activity due to nerve injury can arise in muscles innervated by the trigeminal nerve.

Adult↗