Yuzuru Yasuda

Paramedian thalamic and midbrain infarcts associated with palilalia.

A patient with paramedian thalamic and midbrain infarcts developed palilalia.

Amnesia following infarction in the right retrosplenial region.

We present a case of retrosplenial amnesia following an infarction in the right retrosplenial region. A 62-year-old male showed mild weakness of the left hand, dizziness and gait disturbance. He also noticed that he could not perceive objects that he saw as real, but could perceive an object as real only by touching it. Magnetic resonance imaging (MRI) showed an infarction in the splenium of the corpus callosum and retrosplenial region on the right side. There was no aphasia or apraxia, but mild topographic disturbance was present. Intelligence was normal, but amnesia was noted. Both verbal and visual memory were disturbed equally. This case suggests that memory plays a role in the right retrosplenial region.

Sulindac-induced aseptic meningitis in mixed connective tissue disease

A 21-year-old female with mixed connective tissue disease (MCTD) experienced nausea, headache, consciousness disturbance, nuchal rigidity, and a temperature of 38.5 less than or equal to C three days after the intake of sulindac (300 mg/day). Cerebrospinal fluid analysis revealed an opening pressure of 310 mm of water, a predominantly lymphocytic pleocytosis, and elevated protein content of 89 mg/dl. After discontinuing sulindac, the aseptic meningitis improved in five days. In the acute stage, CT scan disclosed contrast enhancement in the cerebral hemispheres, which suggests that hypersensitivity may be involved in the pathogenesis of nonsteroidal antiinflammatory drug (NSAID) induced aseptic meningitis.

Cheiro-Oral-Pedal Syndrome

The cheiro-oral syndrome is a well-known peculiar sensory disturbance seen around the corner of the mouth and in the palm of the hand on the same side. However, sensory disturbance around the corner of the mouth, in the palm of the hand and in the foot on the same side (cheiro-oral-pedal syndrome) has not been reported until now. We examined 2 cases of cheiro-oral-pedal syndrome; the lesion responsible was confirmed by magnetic resonance imaging to be in the medial lemniscus and in the ventral ascending tract of the trigeminal nerve.

Unusual sensory disturbance in the thoracic region after stroke : relationship to cheiro-oral and cheiro-oral-pedal syndrome

Three patients with unusual unilateral sensory disturbances in the thorax, in addition to hand and mouth, hand and foot, and hand, mouth and foot regions, respectively, after stroke, are described. Magnetic resonance imaging (MRI) showed a causative lesion in the ventral posteromedial and ventral posterolateral nucleus of the contralateral thalamus in two patients and the contralateral thalamocortical projections in one patient. Symptoms in three patients were due to infarction. Objective sensory loss was not found, but subjective paresthesia was observed in the thorax, in addition to the hand and mouth, hand and foot, and hand, mouth and foot regions, respectively. Paresthesia recovery time was evaluated in these three patients in addition to seven patients with cheiro-oral syndrome and eight patients with cheiro-oral-pedal syndrome. All thirteen patients who demonstrated recovery showed regional improvement of paresthesia in the following sequences: thorax, foot, mouth and then hand, which suggests that the detection threshold, from highest to lowest, occurs in the following sequences: thorax>foot>mouth>hand. These three cases with thoracic sensory disturbance form the explanatory links between the cheiro-oral syndrome and the cheiro-oral-pedal syndrome; cases with sensory disturbance in the hand, mouth, foot or thorax could be regarded as cases of sensory hemisyndrome in which the subjective experience of symptoms was restricted to the hand, mouth, foot or thorax. These symptoms may possibly be attributable to differential detection thresholds.

Polymyositis Associated with Asymptomatic Primary Biliary Cirrhosis

We studied a 67-year-old female who suffered from polymyositis associated with primary biliary cirrhosis (PBC). Liver dysfunction was revealed by a screening test. Antimitochondrial antibodies (AMA) and antinuclear antibodies were positive. Liver biopsy was compatible with PBC (Scheuer stage I). Four years later she showed severe weakness and atrophy on her four extremities. Laboratory examination showed a creatinine kinase level of 312 IU/l, IgM 416 mg/dl, and AMA titer 1:320. Muscle biopsy findings were compatible with polymyositis. Electron microscopic examination disclosed diffuse increase of mitochondria in subsarcolemma and intermyofibrils, which has not been reported until now in polymyositis associated with PBC.

Cheiro-oral-pedal syndrome in the lesion of thalamocortical projections

Unilateral sensory disturbance in the hand and mouth (cheiro-oral syndrome) is well known, but one in the hand, mouth and foot region (cheiro-oral-pedal syndrome) has been reported only in 2 patients with brainstem lesion and in 2 patients with thalamic infarction. We report the first case of cheiro-oral-pedal syndrome due to involvement of thalamocortical projections, which favors somatotopy in thalamocortical projections.

Pontine Infarction with Pure Millard-Gubler Syndrome: Precise Localization with Magnetic Resonance Imaging

Pontine Infarction with Pure Millard-Gubler Syndrome: Precise Localization with Magnetic Resonance Imaging Dr. Yuzuru Yasuda, Department of Neurology, Otsu Red Cross Hospital, 1-1-35 Nagara, Otsu 520 (Japan) Millard-Gubler syndrome [1] is characterized by ipsilateral facial palsy, probably owing to involvement of the root fibers, and contralateral hemiplegia resulting from involvement of the corticospinal fibers. Most patients with Millard-Gubler syndrome show some other associated neurological abnormalities because many nuclei or fibers exist near the root fibers of the facial nerve [2]. This is the first case report of pure Millard-Gubler syndrome whose responsible lesion was confirmed by magnetic resonance imaging (MRI). A 60-year-old man developed a throbbing headache on 16 December 1991. Then the headache increased gradually, but nausea or vomiting was not seen. He showed paresthesia on the extremities of the left side 2 days later and vertigo 4 days later. He noticed left facial weakness on 27 December and was admitted to our hospital. On admission, his blood pressure was 186/92 mm Hg. He was alert, and his intelligence was normal. He showed a peripheral facial nerve palsy on the right. Eye movement was normal (fig. 1). He showed left hemiparesis, and his tongue was deviated to the left. The deep tendon reflex was exaggerated, and Babinski’s reflex was equivocal on the left. Objective sensory loss or cerebellar ataxia was not seen. Routine blood and urine examinations were normal. An electroencephalogram, auditory brainstem response and somatosensory evoked response were normal. Computed tomography of the head was normal. T2-weighted MRI showed a high-signal area in the right ventral pons (fig. 2). A vertebral antiogram showed occlusion of both vertebral arteries. The lesion responsible for Millard-Gubler syndrome [ 1 ] is probably located in the root fibers of the facial nerve and the corticospinal fibers. Therefore the lateral inferior and medial inferior pons must be involved together for the appearance of the syndrome. However, as near the root fibers of the facial nerve there are the medial longitudinal fasciculus, paramedian pontine reticular formation, abducens nucleus, superior cerebellar peduncle, dorsal spinothalamic tract, medial lemniscus and secondary ascending tract of the trigeminal nerve [2], Millard-Gubler syndrome often associates with one-and-a-half syndrome, abducens palsy, contralateral cerebellar ataxia, contralateral sensory deficits and so on. Millard-Gubler syndrome is usually seen in brainstem tumor or bleeding but rarely in infarction. Moreover, pontine infarction with pure Millard-Gubler syndrome like in our case is

Cheiro-oral-pedal syndrome in thalamic infarction

Sensory disturbance around the corner of the mouth, in the palm of the hand and in the foot on the same side (cheiro-oral-pedal syndrome) has been reported only in 2 patients with brainstem lesion. We report 2 cases of cheiro-oral-pedal syndrome; the causative lesion was revealed by magnetic resonance imaging to be in the ventral postero-lateral and ventral posteromedial nuclei of the thalamus.

Sphenoid sinus mucocele with recurrent visual disturbance

We present a case of sphenoid sinus mucocele with recurrent visual disturbance on the same side. A 22-year-old female showed two episodes of visual disturbance in the left eye for 3 months, and acute retrobulbar optic neuritis was diagnosed. With corticosteroid, visual disturbance improved in 1 week. MRI and CT scans showed mucocele in the left sphenoid sinus, and left optic nerve swelling with high intensity was observed in T2-weighted MRI. No destruction of the optic canal was found. The contiguous inflammation in the optic nerve rather than compression was considered as pathogenesis.