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PubMed1991

[Diffuse axonal injury (DAI) in an autopsy case of head trauma with long survival].

The authors reported a clinico-pathological case survived 11 months after a traffic accident. A 41-year-old man had been hit by a motor car and was found in a state of semicoma. On admission, his consciousness level was III-100 to 200 (Japan Coma Scale). Pupils were isocoric; light reflex was present. Linear fracture of occipital bone was disclosed by Skull X-ray and subarachnoid hemorrhage was revealed on CT scan. This comatose state, lasting 24 hours, slowly improved and eventually he presented the so-called Korsakoff's syndrome until his death. He could not recognized his relatives, only uttered some meaningless words. He was unable to obey simple verbal orders. The patient was incontinent and right pyramidal sign was positive. On repeated CT scans, cerebral ventricles gradually increased in size; especially the enlargement of the fourth ventricle was remarkable. He expired of septic shock caused by bed sores. At autopsy brain weighed 1190 g. Old gloss contusional scars were observed on the bilateral frontal lobes including the orbital area and on the left temporal pole. Gliding contusions were revealed in the subcortical white matter beneath the left superior frontal convolution. Fibrillary gliosis was noted in this region, the deep white matter underlying the left temporal pole and the tissue surrounding the anterior horn of the left lateral ventricle. Nerve fibers were fragmented and lacerated at corpus callosum, anterior commissure and posterior limb of the left internal capsule. Bilateral pyramidal tracts showed mild myelin pallor at the brainstem. Loss of Purkinje cells were observed. This case would correspond to mile type of diffuse axonal injury proposed by Adams and Gennarelli. (ABSTRACT TRUNCATED AT 250 WORDS)

Accidents, Traffic↗
PubMed1991

Long term remission of intrinsic brainstem gliomas: the case reports of two children.

Intrinsic brainstem gliomas carry the worst prognosis of all pediatric CNS tumors; only 10-25% of patients are expected to survive for more than two years. Over a period of four years seven intrinsic brainstem gliomas were diagnosed in children in one institution. Four of them underwent a rapidly fatal course, whilst one was diagnosed only two years ago, which is too recent for long-term evaluation. We report the case histories of the remaining two boys, who showed a favorable course of their disease. Presenting symptoms were headaches and signs of brainstem dysfunction with multiple bilateral cranial nerve palsies, ataxia and pyramidal tract signs. Diagnosis rested on neuroimaging (CAT scans and/or MRI scans). Both tumors were intrinsic brainstem gliomas, one diffuse and the other focal. They responded to treatment (radiotherapy and chemotherapy in the former patient and radiotherapy alone in the latter patient). The two boys became long-term survivors and have remained well, without evidence of disease, for more than 71 and 61 months, respectively, after completion of treatment. They are probably cured. Prompt therapy with curative intention is recommended, with consistent adherence to the chosen antitumor regimen even in poor-risk brainstem gliomas.

Adolescent↗
PubMed1991

[Hypothyroidism with increased serum levels of cholestanol and bile alcohol--analogous symptoms to cerebrotendinous xanthomatosis].

We reported a 53-year-old woman with hypothyroidism due to ectopic thyroid gland. She showed intellectual impairment, bilateral pyramidal tract sings, slight cerebellar signs, and degenerative changes of brain white matter on CT and MRI, which were similar to symptoms and signs in cerebrotendinous xanthomatosis (CTX). We found increases of serum cholestanol in the patient and additional 3 patients with hypothyroidism. Total bile alcohol was also increased in the serum of the patients. We speculate that hypothyroidism and CTX might have a similar pathophysiological background on the development of neurological complications and atherosclerosis.

Alcohols↗
PubMed1991

[A case of adrenomyeloneuropathy with marked spinal cord atrophy on magnetic resonance imaging].

A 29-year-old man complained of increasing paraplegia and sphincter disturbances. On admission, he was 23 years old. He had moderate pigmentation of the skin, and his neurological examinations revealed spastic paraplegia, hyperreflexia of both legs with Babinski's signs, a pinprick sensation deficit below the L-1 level, loss of vibration sense in the lower extremities and horizontal nystagmus on lateral gaze. Endocrinological examinations revealed adrenocortical insufficiency. CSF, EEG, EMG, brain-CT and myelography did not show any abnormalities, but metrizamide CT myelography at the low thoracic spinal cord revealed decreased cord diameter. Nerve conduction velocities showed impairment in the tibial and peroneal motor fibers. Auditory brain-stem response revealed elongated III-V interval. A cystometrogram disclosed a reflex neurogenic bladder. In the analysis of the fatty acid component of plasma sphingomyelin, the C26/C22, C25/C22, C24/C22 ratios were found to be increased, and the diagnosis of adrenomyeloneuropathy (AMN) was confirmed. The patient's mother was also found to be asymptomatic carrier of AMN on the basis of long chain fatty acid plasma levels. The MRI performed in his age of 29 years, showed marked spinal atrophy from low cervical to low thoracic regions and mild cerebellar atrophy. This findings seems to correspond with chronic progressive demyelination of spinal white matter such as pyramidal tract and fasciculus gracilis.

Adrenoleukodystrophy↗
PubMedDate not supplied

[Case of spinal cord injury at the site of its junction with the medulla].

The authors describe a case of spinal cord injury close to the site of its junction with the medulla. The injury was caused by the sharp end of joiner file. Following the injury a neurological syndrome developed indicating selective direct injury to the pyramidal tract in the left lateral funiculi of the cord and to the pathways of deep sensation on the left side, as well a transient injury to the pathways of superficial sensation on the side.

Accidents, Home↗
PubMed1991

[A mechanism of cheiro-oral syndrome due to brainstem lesions, a case of a dissecting aneurysm of the basilar artery].

A 42-year-old woman developed an abrupt onset of severe headache, nausea, vomiting, unstable gait and numbness around the right side of her mouth and in her right hand. Neurological examination revealed bilateral pyramidal tract signs and hypesthesia of her right palmar tip and the right side of her mouth. However, pain and temperature sensibility was preserved. Cerebrospinal fluid was clear and colorless. CT scan showed an enhancing mass in the prepontine cistern compressing the pontine base. Vertebral angiography revealed irregular narrowing of bilateral vertebral arteries (string sign) proximal to a fusiform aneurysm on the entire length of the basilar artery. MRI showed double lumina in the wall of the aneurysm. The medial lemniscus conducts the discriminatory tactile and the deep sensory impulses from the extremities. The ventral ascending tract of the trigeminal nerve conducts the discriminatory tactile sensory impulses from the face. These two tracts lie close together in the pontine tegmentum, which is also a watershed area of the paramedian branches and circumferential branches of the basilar artery. We suggest that in this case the dissecting aneurysm caused ischemia of these two tracts in the left pontine tegmentum, presenting right cheiro-oral syndrome.

Adult↗
PubMed1977

[Clinical and histopathological studies of cases of lafora-like inclusion bodies].

In case 1, 41-year-old male, developed progressive demetia, paretic gait disturbance and pyramidal signs with the duration of three years. The neuropathological study revealed systemic atrophy as type Pick-disease i.e., lobal atrophy in the frontal and the parieto-occipital regions, degenerative changes in the basal ganglia and in the thalamus, nerve cell loss in the substantia nigra and myelin pallor in the pyramidal tract. Lafora-like inclusions were found in the cerebral cortex and in the cochleal nucleus. In case 2, 45-year-old male, showed character change, cerebellar symptomes and mental deteriotation, and ulcers on the oral mucosa during about 15 years long period. Neuropathological examination showed chronic encephalitis in the brain stem, vacuolar change in the neuron in the olivary nucleus and Lafora-like inclusions in the cochlear nucleus. Though neither generalized conversion nor myoclonus were clinicaly observed in these cases, the inclusions showed histochemically strong similarity with that of the Lafora-disease. These Lafora-like inclusions were compared with those in the literatur, which were reported on various disease of CNS. Finally in respect of predilection of the inclusions, it is likely that the inclusions result from same metabolic disturbance in the cochlear neurons in the Lafora-disease as well as in the present cases.

Adult↗
PubMed1991

[Evoked potentials in diagnosis of ischemic brain stem lesions].

We present an overview on the impact of evoked potentials in diagnosis of ischemic brainstem lesions. Brainstem auditory evoked potentials and somatosensory evoked potentials depict abnormalities, whereas visual evoked potentials are normal in most cases. In patients with basilar artery thrombosis and primary pontine hemorrhage these evoked potentials are mostly abnormal and can indicate the location of the lesion. Furthermore, they are of prognostic value. Whether evoked potentials are abnormal in strokes with branch occlusion of the basilar artery, depends on the location of the infarction. In infarctions of the basis pontis BAEP and SEP may be normal. Hemiparesis in brainstem strokes are associated with abnormalities in transcranial magnetic stimulation. However, no further evaluation of the level of the pyramidal tract lesion is possible by this method. During fibrinolytic therapy of basilar artery thrombosis a continuous monitoring is possible by means of BAEP. Thus, information can be obtained that is not available from neurological examination of the patient under sedative drugs.

Brain Ischemia↗
PubMed1991

High aluminum deposition in the central nervous system of patients with amyotrophic lateral sclerosis from the Kii Peninsula, Japan: two case reports.

We report two cases of amyotrophic lateral sclerosis (ALS), in which metal analysis revealed markedly higher aluminum concentration in the central nervous system (CNS) as well as higher calcium and lower magnesium concentration and higher Ca/Mg ratios compared with controls. Case 1 was a 55-year-old housewife and total duration of illness was 2 years and 2 mon from onset of clinical symptom. Case 2 was a 80-year-old woman and total duration of illness was 10 mon. Results showed that neither were exposed to toxic environments nor any neurologic disease in the past history. Postmortem examination disclosed motor neuron death and degeneration of pyramidal tracts. The significance of metal metabolism in pathogenesis of amyotrophic lateral sclerosis is discussed.

Aged↗
PubMed1991

[SEP monitoring during balloon occlusion test or operation for vertebro-basilar aneurysms].

SEP (somatosensory evoked potential) monitoring was carried out on seven patients with vertebro-basilar aneurysms during balloon occlusion test, during operation, or after operation. In the patient (case 5) with basilar tip aneurysm, the amplitude of N20 remarkably decreased and this finding closely correlated with disturbed consciousness during transient balloon occlusion of the basilar artery. In another patient (case 6) with vertebral dissecting aneurysm, cerebellar retraction caused transient prolongation of N20 latency during operation. In another case, postoperative SEP monitoring revealed marked reduction of N20 amplitude in the patient (case 7) who showed disturbed consciousness and bilateral oculomotor palsy after operation for basilar aneurysms, but who showed no abnormality in postoperative ABR (auditory evoked brainstem response). The other four patients showed no neurological deterioration and no SEP change during transient balloon occlusion of the parent arteries. Because of the high rate of "false-negative" findings, it remains unclear whether SEP monitoring during surgery for vertebrobasilar aneurysms is of value to predict postoperative deficit due to brainstem ischemia. In our study, however, the changes of SEP were well correlated with neurological deterioration and/or the location of postoperative infarction. In conclusion, SEP monitoring during balloon occlusion tests or operations for vertebro-basilar aneurysms is considered to be useful in predicting ischemic complication of the brainstem caused by the occlusion of the parent artery. However other methodologies have to be developed in order to monitor the pyramidal tract and reticular activating system of the brainstem more accurately.

Adolescent↗
PubMed1990

[A clinical study of a family affected with HLA-linked hereditary spinocerebellar ataxia].

The gene locus of hereditary olivopontocerebellar atrophy (OPCA) has been mapped to the short arm of chromosome 6. This locus has been termed as SCA 1 and it is linked to HLA locus. Linkage for OPCA locus to HLA was first been reported by Yakura et al. We had a chance to re-investigate the original family reported by them. Among the members affected, 6 cases were autopsied and their clinical and pathological features were reported by Fujimoto et al. Kudoh et al, and Ikeda. We clinically studied 2 additional patients in this pedigree. The clinical features of these patients, including 6 previously reported cases, were characterized by 1) cerebellar ataxia predominating throughout clinical course; 2) pyramidal tract involvement, characterized by pathological reflexes, with hyper-reflexia or terminal hyporeflexia; 3) generalized muscle atrophy; 4) slow eye movement; 5) facial and tongue atrophy; 6) optic disc pallor; 7) terminal external ophthalmoparesis; 8) mydriasis and sluggish light reflex; 9) mild peripheral neuropathy; 10) mild reduction of deep sense; 11) bulbar symptom; 12) emotional lability, irritation, or euphoria dominating terminally. Clinically, there are certain similarities between this pedigree and other reported pedigrees of which linkage for OPCA locus to HLA have been proved. Furthermore, clinico-pathological reports of hereditary OPCA showing slow eye movement share numbers of clinical characteristics observed in the patients of this pedigree. Accumulating linkage data suggest that hereditary OPCA might be heterogenous disorder genetically. Whether there are any correlation between linkage data and clinico-pathological findings is essential to establish disease entities, and to re-classify hereditary OPCA and its related disorders.

Adolescent↗
PubMedDate not supplied

The long loop reflex in spinocerebellar degeneration and motor neuron disease--its changes with TRH therapy.

This study aimed to define the characteristics of the long loop reflex (LLR) in patients with spinocerebellar degeneration (SCD) and motor neuron disease (MND), and observe changes in LLR caused by thyrotropin releasing hormone (TRH), a facilitator of cerebellar and motor neurons. The markers used for LLR were: V1-2 peak Latency (the latency between the V1 and V2 peaks); V2 peak-P24 Latency (the latency between the V2 peak and P24 of a somatosensory evoked potential); V2 Amplitude, and V2 Square (the area of the V2 wave). V1-2 peak Latency was significantly longer, and V2 Amplitude was significantly lower than the control in SCD. We attributed these alterations of the LLR to cerebellar ataxia, since all SCD cases had cerebellar ataxia, and extrapyramidal symptoms were only present in one SCD case; the MND cases with motor neuron disturbance showed no significant difference from the control. TRH injection resulted in an increase in V2 Square and a decrease in V2 peak-P24 Latency in SCD and other neurological disease patients. We regarded these changes as activation of the LLR by TRH. With TRH therapy, activation of the LLR coincided with improvement of cerebellar ataxia in SCD. Symptomatic improvement, however, was not observed and the LLR changes were not stable in MND. These results suggest that TRH-induced activation of LLR is caused by the activation of cerebellar function, and indirectly concerns with upper motor neurons because V2 Square increased in MND without pyramidal tract signs.

Aged↗
PubMedDate not supplied

MR imaging of Wallerian degeneration in the brainstem: temporal relationships.

Degeneration of the myelin sheath and axon distal to the most proximal site of axonal interruption secondary to axonal disease has been called wallerian degeneration. On MR imaging, wallerian degeneration of the pyramidal tract can be observed as an abnormal signal intensity, showing prolonged T1 and T2 relaxation times that correspond to the corticospinal tract, with or without shrinkage of the ipsilateral cerebral peduncle and pons. Review of MR studies in 150 cases of supratentorial cerebrovascular accidents showed abnormal signal alterations in the ipsilateral brainstem in 33 of the cases. Abnormal intensity in the ipsilateral brainstem was seen as early as 5 weeks after the supratentorial ictus and was fully evident after 10 weeks in all 33 cases. Signal alterations were strongest at about 3-6 months when compared with alterations seen at 10 weeks or even 10 months after the ictus. Shrinkage of the ipsilateral brainstem appeared as early as 8 months and was demonstrated in all cases 13 months after the ictus. MR seems to be the most effective technique for early detection of wallerian degeneration and may provide insight into its pathophysiological and chemical changes.

Adult↗
PubMedDate not supplied

Cerebral hemiatrophy: a clinicopathological report of two cases with a contribution to pathogenesis and differential diagnosis.

"Cerebral hemiatrophy" describes a condition of different etiologies that is characterized by a marked asymmetry of cerebral hemispheres. Case reports of two different forms of cerebral hemiatrophy are presented. In the first case perinatal asphyxia led to severe white matter lesions with predominance on the left side and marked asymmetry of the pyramidal tracts. Symptoms were present immediately after birth ("primary cerebral hemiatrophy"). The second case displayed postictal cerebral hemiatrophy with a widespread loss of cortical neurons of the entire left hemisphere. The disease process started at the age of two years after a widely normal early development ("secondary cerebral hemiatrophy"). A modified classification of cerebral hemiatrophy is presented, and concepts of pathogenesis and differential diagnosis are discussed.

Atrophy↗
PubMedDate not supplied

[Neurological examinations of workers with chronic exposure to manganese dioxide during the production of piles and batteries].

The authors examined 118 workers making piles and batteries under conditions of exposure to toxic action of manganese dioxide dust. Neurological and EEG investigations showed presence of pyramidal tract lesion signs in 16.9% of workers, extrapyramidal syndromes in 6.8%, peripheral nervous system involvement in 13.5% and neurotic complaints in 42.4%. The frequency of neurological manifestations was directly proportional to the duration of work in exposure. The difference in the frequency of neurotic complaints and signs of peripheral and central nervous system damage was statistically significant between the group exposed to manganese dioxide and the control group. EEG investigations were performed in 51 workers and abnormalities were found in 21 cases.

Adult↗
PubMed1990

Electrophysiological correlates of movement initiation.

By averaging the electroencephalogram (EEG) or the magnetoencephalogram (MEG) of waking man in a manner time-locked to the onset of self-initiated volitional acts, movement-related potentials (or magnetic fields) can be recorded. In the preparation period, a slowly increasing cortical negatively called the Bereitschaftspotential (BP) or readiness potential, can be recorded. The BP precedes all our self-initiated (i.e. endogenous or willed) movements and actions by 1 sec or more prior to the onset of muscular contraction. The BP has 2 principal components, an early one which is bihemispherically symmetric, even prior to unilateral movements, and a late one which is lateralized towards the contralateral hemisphere. We have provided evidence (which was confirmed by regional cerebral blood flow studies) that the principal cortical generator producing the early component is the so called supplementary motor area ("SMA"), whereas the late asymmetric (contralateral) component is generated by the primary (rolandic) motor area, MI. The early "SMA"-related BP component may have motivational, intentional or timing properties, while the late component is related to synaptic facilitation in the contralateral MI area in preparation for the final pyramidal tract volley. In motor learning, the frontal areas are activated in both DC-potential recordings and emission CT experiments. The complete role of the "SMA" is not yet clear. However, it obviously has to determine the right moment to start a movement including the timing of single movements in motor sequences. It is particularly strongly activated in bimanual tapping of difficult rhythms.

Electroencephalography↗
PubMed1990

[The role of magnetic resonance imaging in the diagnosis of Hallervorden-Spatz disease].

A mentally-retarded 21-year-old female with narrowing of the visual field was admitted to our hospital because of dystonia in the body and extremities which appeared 6 months before and was gradually exacerbating. On admission, torsion dystonia, pyramidal tract sign, and retinitis pigmentosa with optic atrophy, urinary incontinence, hyperhidrosis and insomnia were noted. On the Wechsler Adult Intelligence Scale, she achieved less than 60 for verbal-IQ. Laboratory tests including serum copper and ceruloplasmin, were all normal. A computed tomography of the brain showed small hyperdense spot in the globus pallidus on the both sides. There was neither cortical atropy nor ventricular dilatation. A Magnetic Resonance Imaging was obtained using both moderate-field (0.5 Tesla) and high-field (1.5 Tesla) superconducting MR systems. T2-weighted spin echo images demonstrated markedly decreased signal intensity area restricted in the globus pallidus and substantia nigra on both system. Moreover, in the center of the globus pallidus, a small high signal spot was seen ("eye-of the-tiger" sign coined by Sethi et al). There was no abnormal signal in the putamen, caudate nucleus, red nucleus, and dentate nucleus. On the other hand, T1-weighted inversion recovery images were not remarkable. These striking low attenuation on T2-weighted findings suggests an increased iron deposition limited to the globus pallidus and substantia nigra which is characteristic of Hallervorden-Spatz disease/syndrome (H-S). Moreover, "eye-of-the-tiger" sign on T2-weighted MR images in the globus pallidus is previously described in H-S.(ABSTRACT TRUNCATED AT 250 WORDS)

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↗