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PubMed1991

Neuroleptic malignant syndrome: a report of a case and review of the literature.

A case of NMS was reported. NMS is an uncommon but potentially lethal complication of treatment with neuroleptics. The diagnosis of NMS should seriously be considered in any individual receiving neuroleptic medications who develops unexplained fever associated with muscle rigidity. The discontinuation of neuroleptics and the use of general supportive measures are crucial. On the basis of more rapid clinical response, either bromocriptine or dantrolene could be added to traditional supportive care.

Adult↗
PubMedDate not supplied

A case report of neuroleptic malignant syndrome without fever in a patient given aripiprazole.

Neuroleptic malignant syndrome (NMS) is a rare disorder seen most often in patients exposed to antipsychotic medications. This syndrome is generally manifested by hyperthermia, muscle rigidity, autonomic instability, altered mental status, tremors, elevated serum creatinine phosphokinase and leucocytosis. It was first described by Delay during the 1960s. It is considered a medical emergency and is fatal if not promptly addressed. It is clinically relevant not only to psychiatrists but all clinicians since patients taking neuroleptics are seen by physicians from virtually every specialty. Relevant studies report a mortality rate of 10-20%. Conditions that share some features of NMS but have different treatment regimens include serotonergic syndrome, lethal catatonia, malignant hyperthermia, infections and various heat disorders. The importance of recognition and prompt intervention can not be overemphasized. Fever is a predominant symptom in NMS. The authors present an unusual case of NMS in a schizophrenic patient without fever who had been on aripiprazole. To date, there are only three possible reported cases of NMS related to aripiprazole. This case report serves to remind clinicians of the essential features in the diagnosis and management of NMS.

Adult↗
PubMed2006

[7-year experience in usage of mirapex in patients with different forms of primary parkinsonism].

The results of mirapex (pramipexol) treatment of 402 patients with Parkinson's disease and juvenile parkinsonism during the period from 6 months to 7 years are summarized. Mirapex was used in monotherapy as well as in combination with levadopa and other antiparkinsonic drugs. The drug was well tolerated and effective in rest tremor, hypokinesia, muscle rigidity and depression, the more pronounced effect being seen at the early stage of the disease. The use of mirapex allows an effective control of motor fluctuations developing during long-term continuous levodopa therapy. The results obtained characterize mirapex as a drug of choice in the treatment of juvenile parkinsonism. In case of a break in mirapex treatment, the recommencement of treatment usually is not accompanied by reduced sensitivity to drug effect.

Adult↗
PubMed1991

[Hodgkin's disease associated with LDH-M subunit deficiency].

The patient was a 60-year-old man who had complained of left cervical swelling from the end of June, 1984. He was diagnosed as having Hodgkin's disease (mixed-cell type) by lymph node biopsy, and was admitted to our hospital for thorough examination and treatment on July 23rd. Diagnosis of stage IA Hodgkin's disease involving only the left cervical lymph node was made. On admission, laboratory data revealed that the LDH activity in his serum and erythrocytes consisted of only type 1 (LDH-H4), hence LDH-M subunit deficiency (homogeneous type) was diagnosed. However, the muscle rigidity, myoglobinuria, and skin lesions that are usually seen in this abnormality were not observed. After two course of C-MOPP therapy, beginning on August 1st. complete remission was obtained. He was subsequently given a total of 6 courses of C-MOPP therapy and 40 Gy of irradiation on an outpatient basis. He eventually died of heart failure on November 3rd, 1986. Only five families with LDH-M subunit deficiency have been reported to date in Japan. This is the first report in Japan of the condition being complicated by Hodgkin's disease.

Antineoplastic Combined Chemotherapy Protocols↗
PubMed1991

[A family with autosomal dominant hereditary myoedema, muscular irritability, stiffness and hypertrophy].

A familial case with autosomal dominantly transmitted myoedema, muscular irritability, stiffness and hypertrophy was reported. The patient is 54 years old and his father, two sisters and niece had suffered from the similar symptoms. He had noticed a feeling of stiffness and pain in the femoral muscles after several minutes of erect position from the age of 5-6 years. He had observed that light tapping of muscles caused a bulge which persisted for several seconds in the whole body. These symptoms were not progressive. The patients had an athletic appearance, hypertrophy of gastrocnemius muscles, pes cavus and hammer toes. Mild muscular weakness and wasting were noted in the intrinsic hand muscles and the anterior tibial muscles. Myoedema was seen in the muscles of the whole body. His thyroid functions were normal. EMG studies showed no myotonic discharges. Light microscopy of biopsy specimens from the biceps brachii muscle showed unspecific myopathic changes. Electron microscopy showed many vacuoles between myofibrils. The symptoms and signs of this case are very like to those of the cases which Torbergsen described in 1975 and only four families were reported after that. The family presented here is the first report in Japan.

Edema↗
PubMed1991

[A case of progressive systemic sclerosis and Sjögren's syndrome complicated by parkinsonism with special reference to the beneficial effect of corticosteroid].

A 79-year-old woman was consulted by us because of 2 years' history of difficulty in neck flexion and frozen gait. On admission, the neurological examination disclosed findings compatible with parkinsonism such as bradykinesia and muscle rigidity. In addition, she felt thirsty and hard skin, cyanosis and Raynaud phenomenon in the limbs. These were consistent with diagnosis of progressive systemic sclerosis and Sjögren's syndrome, which was then confirmed by high titers of anti-nuclear antibody and anti-ENA antibody and abnormal sialogram; marked decrease of saliva and tear secretion was demonstrated. Brain CT and angiography showed no evidence of ischemic stroke. Parkinson symptoms were not relieved by a month's treatment with L-dopa, but was improved by 3 weeks' administration of corticosteroids; the latter also provided a benefit to the poor circulation in her finger tips. Parkinsonism in the present case is likely related to Sjögren's syndrome. Our report proposes a possibility of autoimmune disease as one of underlying causes for drug-resistant parkinsonism.

Aged↗
PubMed1976

Probenecid-induced accumulation of cyclic nucleotides, 5-hydroxyindoleacetic acid, and homovanillic acid in cisternal spinal fluid of genetically nervous dogs.

These studies have been conducted on 40 dogs, twenty each of a genetically nervous strain and of a normal strain of short-haired pointers. The nervous strain after about age 3 months displays extreme hypervigilance, timidity, human avoidance, and often shows catatonic-like muscle rigidity when in the presence of humans or novel stimuli. Measurements of probenecid-induced accumulation of acid metabolites in cisternal cerebrospinal fluid (CSF) have been carried out. Among the compounds measured at from 1.5 hr to 6.0 hr after probenecid treatment, homovanillic acid (HVA) was similar for the two strains, 5-hydroxy-indoleacetic acid (5-HIAA) was lower, but cyclic adenosine-3',5'-monophosphate (cAMP) and cyclic guanosine-3',5'-monophosphate (cGMP) were higher for the nervous strain when compared with age- and sex-matched behaviorally normal dogs. Probenecid levels in CSF were similar at all points in time from 1.5 to 6.0 hr after its intravenous administration in a dose of 50 mg/kg body weight. These findings coupled with previously observed differences in the two strains suggest that hyperresponsiveness of the central nervous system (CNS) noradrenergic and cholinergic systems and a hyporesponsiveness of the serotoninergic system are related to the genetically expressed aberrant behavior.

Animals↗
PubMed1991

[A case of metoclopramide-induced neuroleptic malignant syndrome with cerebrospinal fluid lactic acidosis].

A case of metoclopramide-induced neuroleptic malignant syndrome with cerebrospinal fluid (CSF) lactic acidosis was reported. A 44-year-old Japanese woman noted tarry stool on July 2, 1988 and was treated with metoclopramide and cimetidine for nausea and vomiting. Hydroxyzine pamoate was also administered for insomnia at 3:10 am and she became comatose with muscle rigidity at 3:40-4:30 am on July 3. Tachycardia and high fever (39.5 degrees C) were evident at 8:00 am on July 4. She was transferred to the Kyushu University Hospital. On admission, serum creatine kinase was elevated to 1640 IU/1; MM fraction was 100%. She was diagnosed as malignant syndrome. Cerebrospinal fluid was normocellular with protein 38 mg/dl and glucose 122 mg/dl. Cerebrospinal fluid lactate increased markedly to 3.43 mmol/l, CSF pH was 7.264, HCO3- 14.4 mEq/l, indicating CSF metabolic acidosis. She became afebrile after the 10th hospital day, and gradually but completely recovered within a month. She was discharged on August 16, 1988. The anti-dopaminergic activity of metoclopramide was considered to be primarily responsible for the development of malignant syndrome in this case. Cerebrospinal fluid lactic acidosis seemed to reflect hyperpyrexia or malignant syndrome induced derangement of the brain metabolism.

Acidosis, Lactic↗
PubMed1991

Neuroleptic malignant syndrome and Escherichia coli urosepsis.

Neuroleptic malignant syndrome is a rare but potentially life-threatening reaction to neuroleptic drugs. The syndrome develops rapidly, and may occur at the initiation of neuroleptic therapy or after long-term use; its pathogenesis is unclear. The signs and symptoms associated with the syndrome are hyperpyrexia, extreme muscle rigidity, an altered level of consciousness, and autonomic dysfunction. We describe a case of neuroleptic malignant syndrome in a patient who had Escherichia coli urosepsis caused by thioridazine.

Aged↗
PubMed1991

[An emergency cesarean section using general anesthesia for a patient with schizophrenia].

A 27 year-old female in 39th week gestation with schizophrenia underwent an emergency Cesarean section using general anesthesia. A diagnosis of schizophrenia was made two years previously, since then oral anti-psychotic drugs such as chlorpromazine had been given to her. In June 1989 she suddenly became excited and generalized muscle rigidity was observed without any triggering episodes. Her excitement was so marked that we had to administer intramuscular levomepromazine 75 mg and diazepam 10 mg to her, but they failed to sedate her adequately. Emergency Cesarean section was scheduled to overcome this situation. Spinal or epidural anesthesia was not indicated because of her vigorous excitement, and anesthesia was induced with thiopental 350 mg and succinylcholine 40 mg. Induction-delivery time was 12 minutes. Pentazocine 30 mg in combination with nitrous oxide was given for the maintenance of anesthesia. Plasma levomepromazine levels were 46.9 ng.ml-1 in the mother and 11.3 ng.ml-1 in the umbilical vein, respectively. The baby's Apgar score was 9 and 1 min and 9 at 5 min after the delivery. The baby developed slight generalized tremor until next day, probably due to effect of levomepromazine given before the Cesarean section. The patient was discharged without any cardiorespiratory trouble and her baby has been doing well so far.

Adult↗
PubMed1991

[Effects of enflurane on the elastic properties of frog skinned skeletal muscle fiber under relaxed and rigor conditions].

We studied the effects of enflurane on the elastic property of the mechanical skinned fiber prepared from M. Semitendinosus of bullfrog. Longitudinal and lateral elastic properties of the skinned fibers were measured under relaxed and rigor conditions to clarify the effects of enflurane on actomyosin complex. Under the rigor condition, enflurane did not change the lateral elasticity of the fibers, but enflurane decreased the longitudinal elasticity. On the other hand, under the relaxed condition, enflurane did not change the elasticity. The results suggest that enflurane probably decreases the force of binding of cross-bridges to actin, which is one of the factors to reduce the maximal force.

Animals↗
PubMed1990

[Motor neuron disease with Parkinson's disease--case report].

The patient was a 64-year-old woman who showed muscle weakness and tremor of upper extremities and gait disturbance at the age of 62 years. The symptoms progressed and she was admitted to our hospital. Neurological examination revealed muscle weakness, muscle atrophy and fasciculation bilaterally in the upper extremities. The deep tendon reflexes were reduced in the upper extremities and increased in the lower extremities, but Babinski's sign was not present. There was mild hand tremor at rest (right greater than left). Muscle rigidity was also evident. Her gait was small-stepped and her trunk was bent forward. She showed hypomimia, but no dementia was detected. She died of respiratory failure 7 months after admission. The duration of the illness was about 2 years. At autopsy, macroscopic examination showed depigmentation of the substantia nigra and locus ceruleus, and atrophy of the anterior roots of the spinal cord. Microscopic examination revealed a few senile plaques in the temporal cortex. In the substantia nigra, the number of melanin-containing cells was decreased in its central parts. A few Lewy bodies were found in some of the remaining neurons, and melanin pigment migrated into the parenchyma. In the locus ceruleus and dorsal motor nucleus of vagus, abundant Lewy bodies and mild astrocytosis were seen. A few Lewy bodies were also seen in the nucleus raphe, nucleus basalis of Meynert and hypothalamic nuclei. Severe neuronal loss of the anterior horn cells was observed in the cervical segment, and to a lesser degree, in the lumbo-sacral segments. segments.(ABSTRACT TRUNCATED AT 250 WORDS)

Brain↗
PubMedDate not supplied

Stiff-man syndrome in a patient with Hodgkin's disease. An unusual paraneoplastic syndrome.

A case of stiff-man syndrome (SMS), a rare and dramatic CNS disease characterized by continuous muscle activity and painful spasms resembling a chronic form of tetanus, occurring in a patient with Hodgkin's disease (HD) is reported. The patient developed the clinical features of SMS at the same time as the HD relapse. A satisfactory improvement was obtained with diazepam, but the complete recovery from stiffness was achieved only after chemotherapy was started. Cerebellar autoantibodies were found in the serum of the patient. With chemotherapy the patient achieved a second complete remission (CR). Eighteen months later the patient developed a second HD relapse, and at that time no signs of SMS were detected.

Antineoplastic Combined Chemotherapy Protocols↗
PubMed1990

[Left auricular hypertrophy in aortic stenosis in adults].

Left atrial hypertrophy (LAH) was noted from the electrocardiograms of 72 of 98 adult patients (81%) who underwent hemodynamic evaluation of calcified aortostenosis (CAS). The relations between LAH and clinical, echographic and hemodynamic findings are specified. The frequency of LAH was not higher in cases of a history of hypertension, angina pectoris, lipothymia or exercise-induced syncope. In contrast, dyspnea was more frequently associated with LAH (84%) than not (17%). An approximately linear relation was seen between LAH and the mean pulmonary capillary pressure, the mean rate of circumferential decrease (RCF), the coefficient of muscle rigidity (ks of Mirsky), the left ventricular mass (LVM) and the left ventricle-aorta gradient. LAH is, therefore, a frequent sign in patients presenting CAS. Its origin is multifactorial, with a predominance of increased mean capillary pressure in cases of clinical signs of poor safety.

Aortic Valve Stenosis↗
PubMed1990

[A contribution to "stiff man" syndrome].

A patient with the clinical symptoms of the "Stiff-man"-syndrome, but an atypical course was introduced. Symptoms and course were compared with similar cases mentioned in literature. The "Stiff-man"-syndrome is probably a disease of central origin affecting the relationship between inhibitory and excitatory regulation of the muscle tonus, especially the exteroceptive reflex mechanisms. Several pathological processes of the CNS can be held responsible for the disturbance of the balance in this regulatory system.

Deglutition Disorders↗
PubMed1990

Neuroleptic malignant syndrome.

NMS is an uncommon disorder characterized by hyperthermia, muscle rigidity, autonomic imbalance, altered levels of consciousness and significant mortality. Successful treatment of NMS requires a high degree of suspicion, rapid recognition of clinical signs and symptoms and institution of therapy with dantrolene and possibly dopaminergic agonists. This condition is precipitated by neuroleptic drugs, which are commonly used in many medical specialties. All medical practitioners responsible for primary care, psychiatrists and anesthesiologists should be familiar with the manifestations of the hypermetabolic syndromes of Neuroleptic Malignant Syndrome, Malignant Hyperthermia, and Neuroleptic Malignant-like Syndrome and should be prepared to initiate appropriate therapy.

Adult↗
PubMed1990

[Parkinsonism associated with cerebrotendinous xanthomatosis].

Two sibling cases of cerebrotendinous xanthomatosis with parkinsonism were reported. One was a woman of 39 years old, and another was her sister of 36 years old. In both cases, febrile convulsion appeared on 1.5 year old, and mental deterioration, ataxic -spastic gait, cataract and swelling of Achilles tendons developed in order since entrance into elementary school. Five years ago, while they were in hospital at the first time, they were diagnosed as cerebrotendinous xanthomatosis by mental disturbance, cerebellar ataxia, pyramidal tract sign, histologically xanthomatous granuloma of Achilles tendons and hypercholestanolemia and family history of autosomal recessive trait. After the second admission, parkinsonism was noticed in addition to those findings above. Parkinsonism consisted of the following: Resting tremor of parkinsonian type, mild muscle rigidity of forearm and intrinsic-plus hand were observed in the elder sister, and generalized severe rigidity and bradykinesia in the younger sister. In both cases, brain CT showed the pontocerebellar atrophy, and the bilateral low density area in corona radiata, posterior portion of internal capsule, cerebral peduncle, tegmentum of midbrain and deep matter of cerebellum. Brain MRI also showed abnormal intensity in the same regions as on the brain CT. Administration of anti-parkinsonian drugs was challenged for the parkinsonism. Oral L-dopa test (500 mg) moderately improved parkinsonism in both cases. Therapy of diphenylpyraline hydrochloride (10 mg/day) entirely inhibited parkinsonian tremor and mild rigidity in the elder sister but was less effective for severe rigidity in the younger sister than administration of L-dopa.(ABSTRACT TRUNCATED AT 250 WORDS)

Achilles Tendon↗