Prolonged Disturbance Of Consciousness Research Articles

Hoarding medication and suicide attempts with 1180 tablets of mirtazapine overdose: A case report

AbstractBackgroundConfirming a patient's consistent adherence to prescribed medication remains a persistent challenge for healthcare professionals. One reason for nonadherence is that patients accumulate their prescribed medicines with the intent of taking large quantities as a suicidal attempt. We present a case involving hoarding and overdosing on the highest quantity of mirtazapine ever reported.Case PresentationOur patient, a 44‐year‐old man suffering from depression, was admitted to our emergency department due to an overdose of 1180 mirtazapine tablets, equivalent to 17,700 mg. He exhibited a prolonged disturbance of consciousness, liver enzyme abnormalities, and serotonin syndrome. After 7 weeks in the intensive care unit, he was transferred to a psychiatric ward. Over time, his depressive symptoms improved, as did his suicidal ideation. On the 73rd day of his illness, he was discharged without any lasting effects, although he experienced retrograde amnesia.ConclusionsIn this case, ensuring the patient's medication adherence is paramount. Implementing electronic prescription technologies may offer the potential to enhance healthcare by avoiding medication duplication, preventing stockpiling of duplicated medicine, and preventing overdosing.

Recovery after Prolonged Disturbance of Consciousness and Repeated Cerebral Perfusion Changes in Neuronal Intranuclear Inclusion Disease.

Encephalitic episodes are a clinical manifestation of neuronal intranuclear inclusion disease (NIID) and often show transient disturbance of consciousness. We herein report a genetically confirmed patient with NIID who initially presented progressive dementia and showed prolonged disturbance of consciousness preceded by an acute-onset headache. During that time, we performed N-isopropyl-p-[123I] iodoamphetamine single-photon-emission computed tomography twice and found that the blood flow increased in different regions. Prolonged disturbance of consciousness following an encephalitic episode may be associated with repeated hyperperfusion in various regions resulting from mitochondrial dysfunction. NIID patients presenting with encephalitic episodes can recover gradually and spontaneously even after prolonged disturbances of consciousness.

Recovery of consciousness under therapy with benzodiazepines. A case report and literature review

Treatment of patients with prolonged and permanent disturbance of consciousness is still an extremely difficult problem. Nowadays, management is based on pathophysiological and molecular mechanisms of impaired consciousness. Several electrophysiological and pharmacological methods were proposed to restore consciousness in appropriate patients. We present recovery of clear consciousness under therapy with phenazepam and literature review devoted to therapy of these disorders. This case confirms available data on drug neuromodulation in complex treatment of patients with prolonged impairment of consciousness and substantiates the need for individual multimodal assessment of structural and functional disorders in prolonged and chronic impairment of consciousness for adequate therapy.

Malignancy-related Hypercalcemia Caused by Metameric Cutaneous Metastasis of Parathyroid Hormone-related Protein-producing Bladder Carcinoma with Squamous Cell Differentiation: An Autopsy Case of Cobb Syndrome.

A 74-year-old woman was admitted with hypercalcemia and prolonged disturbance of consciousness. The left buttock to the anterior aspect of the left thigh was swollen and erythematous, with a collection of 1.0-cm large, firm, elastic nodules distributed in a zosteriform pattern in the L1-L4 region. Based on autopsy findings, a very rare case of Cobb syndrome was diagnosed due to a spinal vascular malformation at the Th12-L4 level and L5 vertebral hemangioma. Cobb syndrome-associated cutaneous metastasis extending along the same metamere was complicated by immunohistochemically proven parathyroid hormone-related protein-producing advanced bladder carcinoma in this case.

ODP285 A Case of Encephalitis and Adrenal Insufficiency in a Patient With Panhypopituitarism After COVID-19 Vaccination

Abstract Background Since COVID-19 vaccination was introduced, various adverse effects have been linked to the vaccines. In patients with hypopituitarism, adrenal insufficiency due to the side reactions including fever of COVID-19 vaccination is concerned. Clinical Case A 33-year woman was on medical therapy including hydrocortisone (HC) for panhypopituitarism arising from surgical treatment of a pituitary adenoma in 2006. She received a COVID-19 vaccination on day X-3. On day X-2, she developed fever in the morning and became unconscious in the evening. She was brought to our hospital by her family at night on day X. She had fever of 40.5°C, low blood pressure, and Glasgow Coma Scale (GCS) of 11. Her neck was supple and she had no quadriplegia. A COVID-19 PCR test was negative. Blood tests showed elevated white blood cell count (8900/μL; reference range: 3300–8600/µL) and C-reactive protein (138.3 mg/l; reference range: 0-1.44 mg/l). Blood glucose (81 mg/dL), ACTH (<3. 00 pg/mL; reference range: 7.2–63.3 pg/mL), and cortisol (1.9 μg/dL; reference range: 2.9–19.4 µg/dL) were low. Serum electrolytes were normal. A computed tomography scan showed no abnormality. Adrenal insufficiency was suspected, and she received HC intravenously. Her blood glucose and blood pressure increased, but her disorientation persisted. Lumbar puncture with cerebrospinal fluid (CSF) examination revealed slightly elevated cell counts (8 μ/L; reference range ≤4 μ/L) with average protein and glucose levels. Magnetic resonance imaging (MRI) of the brain revealed abnormal hyperintensity in the splenium of the corpus callosum on diffusion-weighted images and decreased apparent diffusion coefficient in the lesion, suggesting clinically mild encephalitis/encephalopathy with a reversible splenial lesion (MERS). During her hospital stay, she received a 7-day course of meropenem and acyclovir for suspected meningoencephalitis. Her consciousness disturbance improved to GCS of 15 on day X+1 and her fever decreased on day X+2. HSV and VZV PCR tests were negative on CSF examination, and antibiotics and antivirals were discontinued on day X+7. On day X+8, brain MRI showed complete resolution of the corpus callosum lesion. She discharged on day X+18 without any neurological sequelae. Conclusions For most vaccines, the incidence rates of encephalitis are low at 0.1–0.2 per 100,000 vaccinated individuals (1). The present patient developed fever and adrenal insufficiency after COVID-19 vaccination, and her prolonged disturbance of consciousness after HC administration led to the diagnosis of MERS. MERS should be considered in patients with adrenocortical insufficiency who show delayed recovery from unconsciousness with HC administration after COVID-19 vaccination. Reference: (1) Huynh W, Cordato DJ, Kehdi E, Masters LT, Dedousis C. Post-vaccination encephalomyelitis: literature review and illustrative case. J Clin Neurosci. 2008 Dec;15(12): 1315-22. Presentation: No date and time listed

Tacrolimus-associated posterior reversible encephalopathy syndrome after kidney transplantation

Calcineurin inhibitors are essential immunosuppressive agents for kidney transplant patients. Posterior reversible leukoencephalopathy syndrome is a rare complication of treatment with calcineurin inhibitors. Herein, we report three cases of tacrolimus-associated posterior reversible leukoencephalopathy syndrome after kidney transplantation and their unique clinical courses. Patients in Cases 1 and 2 presented with headache and visual field disturbance on the fourth day after transplantation. Both patients were successfully treated with antihypertensive therapy and a change in medications from tacrolimus to cyclosporine and everolimus. Case 3 was a kidney transplant patient at 12 years post-transplant who developed fever and conjugate eye deviation to the right. The patient was initially treated with antihypertensive medication and a reduction in tacrolimus dose, but she experienced prolonged disturbance of consciousness, which eventually improved. These cases highlight the importance of considering the possibility of posterior reversible leukoencephalopathy syndrome in kidney transplant recipients taking calcineurin inhibitors who present with acute neurological dysfunction. They also demonstrate the need to perform appropriate imaging to confirm the diagnosis and initiate treatment, even if a significant amount of time has passed since transplantation. The introduction of an mTOR inhibitor may be practical when changing or discontinuing calcineurin inhibitors.

Rapidly progressive brain atrophy in septic ICU patients: a retrospective descriptive study using semiautomatic CT volumetry

BackgroundSepsis is often associated with multiple organ failure; however, changes in brain volume with sepsis are not well understood. We assessed brain atrophy in the acute phase of sepsis using brain computed tomography (CT) scans, and their findings’ relationship to risk factors and outcomes.MethodsPatients with sepsis admitted to an intensive care unit (ICU) and who underwent at least two head CT scans during hospitalization were included (n = 48). The first brain CT scan was routinely performed on admission, and the second and further brain CT scans were obtained whenever prolonged disturbance of consciousness or abnormal neurological findings were observed. Brain volume was estimated using an automatic segmentation method and any changes in brain volume between the two scans were recorded. Patients with a brain volume change < 0% from the first CT scan to the second CT scan were defined as the “brain atrophy group (n = 42)”, and those with ≥ 0% were defined as the “no brain atrophy group (n = 6).” Use and duration of mechanical ventilation, length of ICU stay, length of hospital stay, and mortality were compared between the groups.ResultsAnalysis of all 42 cases in the brain atrophy group showed a significant decrease in brain volume (first CT scan: 1.041 ± 0.123 L vs. second CT scan: 1.002 ± 0.121 L, t (41) = 9.436, p < 0.001). The mean percentage change in brain volume between CT scans in the brain atrophy group was –3.7% over a median of 31 days, which is equivalent to a brain volume of 38.5 cm3. The proportion of cases on mechanical ventilation (95.2% vs. 66.7%; p = 0.02) and median time on mechanical ventilation (28 [IQR 15–57] days vs. 15 [IQR 0–25] days, p = 0.04) were significantly higher in the brain atrophy group than in the no brain atrophy group.ConclusionsMany ICU patients with severe sepsis who developed prolonged mental status changes and neurological sequelae showed signs of brain atrophy. Patients with rapidly progressive brain atrophy were more likely to have required mechanical ventilation.

Informed DEcision for cerebrospinal fluid analysis after epiLeptic seizures- the IDEAL-score: A development and validation study

BackgroundThis observational study was done to develop a score based on clinical predictors that enables a guided decision for the necessity of cerebrospinal fluid (CSF) analysis after first unprovoked epileptic seizures and to validate this score in a retrospective patient cohort. MethodsClinical predictors were identified by two panels of epilepsy experts and selected according to content validity ratios. Based on these predictors a score was created and applied to a cohort of patients with first epileptic seizures. ResultsThe “IDEAL score” consists of 9 items (fever, prolonged disturbance of consciousness, headache, imaging results, cognitive dysfunction, status epilepticus, malignancy, autoimmune encephalitis symptoms) that are collected at two different time points (< 3 h [A-score]; > 3 h [B-score] after hospital admittance). A CSF analysis is recommended, if at least one clinical finding is present, either one of the items evaluated during the acute phase (A-score) or later in the diagnostic process (B-score). In 41 patients (13%) CSF analysis provided essential clues to the cause of the seizure. The combined IDEAL score reached a sensitivity of 98%, a specificity of 53%, a positive predictive value of 24% and a negative predictive value of 99% in this patient cohort. ConclusionsA CSF analysis after first epileptic seizures provided decisive etiological findings in only 13% of all investigated patients. The IDEAL score offers clinicians a simple and easy-to-implement algorithm to assess the necessity of a CSF analysis, and to prevent unnecessary diagnostic procedures.

アモキサピン中毒による痙攣重積発作により重篤なアシドーシス（pH 6.499）を呈するも，救命できた1症例(A survival case of amoxapine overdose presented severe acidosis (pH 6.499) due to status epilepticus)

要旨 第二世代三環系抗うつ薬の一つであるアモキサピンは，中毒により重篤な中枢神経毒性を呈することが知られている。とくに痙攣は難治性であり，ベンゾジアゼピンに抵抗性を示すことがある。症例は51歳の女性。アモキサピン1,500mgを服用し，3時間後に痙攣重積状態で搬送された。来院時pH 6.499，PaCO2 208mmHg，HCO3− 15.2mmol/L，Lac 24mmol/Lと著しい混合性アシドーシスを認めた。ミダゾラム計20mgを投与したが不応であり，プロポフォール100mgを投与したところ痙攣は停止した。入院後は，プロポフォール，ミダゾラムを持続投与し，持続脳波モニタリングでは痙攣の再発はなかった。鎮静薬を漸減し，第6病日に従命反応を確認したが，意識障害遷延のため気管切開を実施した。合併症として横紋筋融解症を来したが，急性腎傷害には至らず軽快した。第13病日にICUを退室し緩徐に意識レベルは回復したが，第35病日に精神科病院へ転院した際には，高次脳機能障害と運動失調が残存した。頭部CTでは，来院時に脳浮腫を認めたものの第4病日に改善していた。画像上明らかではないが，長時間の痙攣重積に伴う神経学的後遺症と考えられた。アモキサピン中毒による痙攣重積発作は，長時間により持続重篤な転帰をたどることがある。痙攣発作が難治性であることに留意し，可及的速やかに痙攣を停止させることが重要である。

Prolonged mild disturbance of consciousness and acute encephalopathy.

Acute encephalopathy (AE) is defined by the Japanese guidelines as the acute disturbance of consciousness (Glasgow coma scale [GCS] score ≤11) that persists for >24h. We have often encountered, however, cases of prolonged mild disturbance of consciousness (PMDC) with GCS score >11, meaning that they do not fit the guideline definition of AE. The reports of these cases have been relatively sparse, and the nosological position, prognosis, and other characteristics remain unknown. To clarify the characteristics of PMDC we compared cases of PMDC with cases of AE. This study was a retrospective observational study at a tertiary children's hospital in Japan. We studied children with a diagnosis of AE or PMDC between January 2011 and August 2016. Thirteen cases of PMDC and 19 cases of AE were identified during the study period. PMDC patients more frequently had hyponatremia (P < 0.01), paradoxical arousal response on electroencephalogram (P = 0.010), normal computed tomography (CT; P = 0.025), and normal magnetic resonance imaging (MRI; P < 0.01) than the AE patients. Sequelae were more frequently observed in AE than in PMDC patients (P = 0.011). PMDC has different characteristics to AE with regard to hyponatremia, paradoxical arousal response, CT or MRI findings, and prognosis. Despite the differences, PMDC might also be regarded as being a milder member of the wide variety of AE and related diseases.

Efficacy of Thyrotropin-Releasing Hormone Analog for Protracted Disturbance of Consciousness due to Aneurysmal Subarachnoid Hemorrhage

Objective: The efficacy of thyrotropin-releasing hormone tartrate (TRH-T) for treating prolonged disturbance of consciousness due to aneurysmal subarachnoid hemorrhage (SAH) remains unclear. The purpose of the present study was to determine whether TRH-T was really effective, and what was the recovery factor when it was valid. This was a retrospective study of a single facility. Methods: We treated 208 patients with aneurysmal SAH at our hospital between 2011 and 2017. Among them, we investigated 97 cases in which TRH-T was administered to prolonged disturbance of consciousness. Thirty one patients with Hasegawa dementia rating scale-revised (HDS-R) score less than 20 were included. Patients’ HDS-R scores were evaluated 7 days after clipping the aneurysm and 2 days after completing a course of TRH-T treatment. HDS-R score increases of greater than or over equal to 8 and less than 8 were defined as good and poor outcomes, respectively. Outcomes were compared to 11 patients who did not receive TRH-T treatment. Results: Average initial and post-treatment HDS-R scores were 9 ± 6.6 and 19 ± 9.5, respectively. The good outcome group included 19 patients. Statistically significant differences in HDS-R score changes were observed between the group with initial HDS-R scores of 0-4 and the other groups. Poor outcomes were significantly correlated with age of greater than 60 years and initial HDS-R scores less than oroverequal to 4 points. The improvement in HDS-R score was significantly greater in the TRH-T administration group than the control group. Conclusions: TRH-T was effective for treating prolonged disturbance of consciousness due to aneurysmal SAH, especially in young patients with HDS-R scores between 5 and 20.

Effectiveness of combined use of levodopa and amantadine on prolonged consciousness disturbance due to traumatic brain injury in children—A case report

Prolonged consciousness disturbance associated with traumatic brain injury (TBI) in children is one of the most serious complications for rehabilitation in order to perform cognitive/behavioral therapy. We report two cases of young TBI who got the remarkable recover by combined use of Levodopa and Amantadine. Case: TBI in two children. Case 1 was a 17-year-old man with diffuse brain injury following traffic accident showing prolonged consciousness disturbance and easy irritability due to damage of bilateral orbital part of inferior frontal gyrus. Case 2 exhibited similar symptoms at 14 years of age. After admission to our recovery rehabilitation ward, they had daily 6 hours of rehabilitation, and at the same time, and took Levodopa and Amantadine for 10 weeks. In case 1, the disorder of consciousness was changed from E1V2M4 to E4V5M6 Glasgow Coma Scale, WAIS - III also to VIQ 72 → 96, PIQ 48 → 80, FIQ 58 → 87, FIM improved from 40 to 93, he discharged to his home in 3 months after hospitalization, and then returned to high school. In addition, he succeeded in getting a driver's license. Case 2 also followed a similar course and succeeded in successful recovery. It is said that deficiency of dopamine, a mediator in the brain, is involved in prolonged consciousness disturbance and easy irritability after TBI of children. It is thought that Levodopa was converted into dopamine in the brain, and Amantadine was activated dopamine in the brain by enhancing the neurotransmission of dopamine in the brain. It suggests that this therapy will be recommended as one of treatment methods for TBI in children.

A Case of Transient Hypercortisolism Simultaneously Occurring With the Syndrome of Inappropriate Antidiuretic Hormone Secretion Induced by Olanzapine

We herein present a case of a 71-year-old woman who was referred to our hospital with the prolonged disturbance of consciousness after two convulsive seizures the day before admission. She was in long-term hospitalization because of schizophrenia. Blood tests showed severe low-osmolar hyponatremia (Osm: 226 mOsm/kg and Na: 110.3 mEq/L) and hypokalemia (K: 2.67 mEq/L). An endocrine examination revealed that the syndrome of inappropriate antidiuretic hormone secretion (SIADH), which was induced secondary to olanzapine, and transient hypercortisolism had simultaneously occurred. Low- and high-dose overnight dexamethasone suppression tests and the corticotropin-releasing hormone (CRH) test showed similar endocrine responses to those in patients with Cushing’s syndrome. However, no abnormality in the pituitary gland was detected using contrast magnetic resonance imaging (MRI). After admission, elevated adrenocorticotropic hormone (ACTH) and cortisol levels gradually decreased and normalized by the time of discharge. Transient hypercortisolism sometimes occurs in patients with schizophrenia. Hypercortisolism in the present case was considered to be induced by stress such as severe hypo-osmotic hyponatremia and convulsions in addition to the underlying disorder of schizophrenia. Clinicians need to routinely check electrolytes in patients taking olanzapine in order to prevent insidious progression to severe hyponatremia. To the best of our knowledge, this is the first case report in which an endocrine examination and its interpretation were performed for SIADH accompanied by transient hypercortisolism due to the stimulation of ACTH. J Med Cases. 2017;8(8):259-263 doi: https://doi.org/10.14740/jmc2877w

A Case of Transient Hypercortisolism Simultaneously Occurring With the Syndrome of Inappropriate Antidiuretic Hormone Secretion Induced by Olanzapine

We herein present a case of a 71-year-old woman who was referred to our hospital with the prolonged disturbance of consciousness after two convulsive seizures the day before admission. She was in long-term hospitalization because of schizophrenia. Blood tests showed severe low-osmolar hyponatremia (Osm: 226 mOsm/kg and Na: 110.3 mEq/L) and hypokalemia (K: 2.67 mEq/L). An endocrine examination revealed that the syndrome of inappropriate antidiuretic hormone secretion (SIADH), which was induced secondary to olanzapine, and transient hypercortisolism had simultaneously occurred. Low- and high-dose overnight dexamethasone suppression tests and the corticotropin-releasing hormone (CRH) test showed similar endocrine responses to those in patients with Cushing’s syndrome. However, no abnormality in the pituitary gland was detected using contrast magnetic resonance imaging (MRI). After admission, elevated adrenocorticotropic hormone (ACTH) and cortisol levels gradually decreased and normalized by the time of discharge. Transient hypercortisolism sometimes occurs in patients with schizophrenia. Hypercortisolism in the present case was considered to be induced by stress such as severe hypo-osmotic hyponatremia and convulsions in addition to the underlying disorder of schizophrenia. Clinicians need to routinely check electrolytes in patients taking olanzapine in order to prevent insidious progression to severe hyponatremia. To the best of our knowledge, this is the first case report in which an endocrine examination and its interpretation were performed for SIADH accompanied by transient hypercortisolism due to the stimulation of ACTH. J Med Cases. 2017;8(8):259-263 doi: https://doi.org/10.14740/jmc2877w

Severe hyponatremia in a patient with schizophrenia associated with prolonged consciousness disturbance

Hyponatremia presents with various central nervous system symptoms during its course and treatment. We treated a patient who presented with a prolonged consciousness disorder and was suspected of having complications of neuroleptic malignant syndrome and osmotic demyelination syndrome (ODS) during the treatment for his hyponatremia, which was caused by syndrome of inappropriate secretion of antidiuretic hormone (SIADH). The patient was a 30-year-old Japanese man who had been under treatment for schizophrenia. He presented with profound hyponatremia (96 mEq/L) and a consciousness disorder. Because he was taking a number of antipsychotic drugs and since psychogenic polydipsia was present along with laboratory findings, the patient was diagnosed with SIADH. However, the consciousness disorder reappeared after his serum sodium concentrations were normalized, and it persisted over a long period. Although ODS was suspected from the clinical course and imaging findings, there were several inconsistencies, such as the lack of quadriplegia. The patient also showed muscular rigidity and fever, and we, therefore, diagnosed complications of malignant hyperthermia syndrome caused by the discontinuation of all antipsychotic drugs at the time of onset. There have been no reports of complications of these two conditions, and we report this case for its clinically valuable information.

Therapeutic Hypothermia With the Use of Intracranial Pressure Monitoring for Acute Disseminated Encephalomyelitis With Brainstem Lesion

Acute disseminated encephalomyelitis confined to the brainstem is associated with poor prognosis. We describe a case of a 10-year-old boy with acute disseminated encephalomyelitis in the brainstem that developed after influenza A infection. A 10-year-old boy presented with fever and prolonged disturbance of consciousness and was admitted to our hospital. Magnetic resonance imaging (MRI) of the midbrain, with T2-weighted and fluid-attenuated inversion recovery images, suggested acute disseminated encephalomyelitis accompanied by a brainstem lesion. Lumbar puncture showed pleocytosis and increased protein content, including myelin basic protein, interleukin-6, and immunoglobulin G, all suggestive of acute disseminated encephalomyelitis. Treatments such as methylprednisolone pulse therapy, intravenous immunoglobulin, and therapeutic hypothermia were performed. Although the patient presented with anisocoria with increased intracranial pressure monitoring during hypothermia, prompt therapy with d-mannitol and dopamine was effective. Our case results suggest that hypothermia could be included in the choice of therapy for acute disseminated encephalomyelitis with brainstem lesions.

Regional Cerebral Blood Flow Abnormalities in Patients With Kawasaki Disease

Kawasaki disease (KD) is an acute febrile disorder of unknown etiology. Brain single-photon emission computed tomography (SPECT) and magnetic resonance imaging (MRI) help in detecting regional cerebral blood flow abnormalities and brain damage. The usefulness of SPECT and MRI in patients with KD was evaluated. All 22 patients with KD underwent brain SPECT using Tc-99m-hexamethyl propylene amine oxime from 6 days to 3 years after onset, and 8 patients underwent brain MRI. Of the 22 patients, 4 had neurologic symptoms. Case 1 showed prolonged apnea; case 2, prolonged disturbance of consciousness; and cases 3 and 4 generalized tonic-clonic seizures. Initial brain SPECT showed localized hypoperfusion in 4 and 13 patients with and without neurologic symptoms, respectively. All patients with neurologic symptoms underwent follow-up SPECT; localized hypoperfusion was detected between 1- and 6-month follow-up in 3 of these patients. Six patients without neurologic symptoms underwent follow-up SPECT. Localized hypoperfusion was detected at approximately 1- to 11-month follow-up in 4 of these patients. Diffusion-weighted imaging revealed abnormal high-intensity areas in the corpus callosum in case 1. Case 2 showed a bilateral chronic subdural hematoma with decreased size and ischemic changes, and case 3 showed bilateral hippocampal atrophy and left hippocampal sclerosis. Because the occurrence of localized hypoperfusion is possibly not restricted to only the acute phase in KD, brain SPECT and MRI should also be performed in KD patients with neurologic symptoms.

Sitting Without Back Support Position for Prolonged Consciousness Disturbance Patients

The purpose of this research was to document and compare the before and after levels of consciousness of patients with prolonged consciousness disturbance (PCD) who sat for a specified length of time in a specially designed backless chair (Suwarou-Kun). Three patients were selected as participants using the PCD criteria described by Jennet and Plum in 1972. The Kohnan Vegetative Score (henceforth referred to as the Kohnan PCD Scale) and electroencephalography (EEG) measurements, together with direct observation of subtle changes, were used to record each participant's overt behavior and autonomic responses. A single-case observation/intervention time series design was used in this study. Length of exposure to the intervention and mean exposure time per session were as follows: case 1, 65 days and 30 minutes; case 2, 36 days and 11 minutes; and case 3, 43 days and 36 minutes. The Wilcoxon's rank sum test was used to analyze the pair of Kohnan PCD Scale and EEG scores collected before and at the midway point of each intervention session. Because more than two variables were being measured, the data were reanalyzed using repeated-measure analysis of variance. In cases 2 and 3, there were significant differences in the Kohnan PCD Scale and EEG scores during the "sitting without back support position" (SB) intervention period as well as at the midway point of each session (p < .05) compared with the measurements taken before the intervention. In all three cases, there also were subtle changes during the intervention, for example, eye movement, finger or thumb movement, strength of voice, and salivation. Alpha and beta waves were greater after the introduction of the SB intervention and preceded the behavioral response changes. The SB intervention resulted in at least some improvement in the level of consciousness for each participant. It remains an open question, however, whether longer exposure would have brought about further change. The SB intervention is costly in terms of human time and effort, and its beneficial effects beyond those measured in this study will require additional research.

抗Ｈｕ抗体，抗ＧｌｕＲε２抗体ともに陽性で辺縁系脳炎を合併した末梢神経障害の１例

We report a case of paraneoplastic neurological syndrome with anti-neuronal antibodies, namely anti-Hu and anti-GluR epsilon 2 antibodies in sera. A 72-year-old male had a transient history of eye movement disorder and sensory neuropathy, which improved spontaneously. Two years later, he was admitted to another hospital because of gait disturbance, numbness of the hands and an attack of unconsciousness with generalized convulsion. He was admitted to our hospital with prolonged consciousness disturbance and muscular weakness of all extremities. On admission his consciousness deteriorated slightly without neck stiffness. His cranial nervous system was normal except for incomplete abduction and elevation of both eyes. The patient had severe distal dominant weakness and atrophy in the muscles of all four limbs. Muscle tonus was decreased and hyporeflexia was noted in the four extremities. Plantar response was extensor. Neither sensory disturbance nor ataxia was observed. Cranial MRI showed T2-weighted high intensity lesions in the bilateral mesial temporal lobes, including the hippocampi. A nerve conduction study revealed motor-dominant peripheral neuropathy with prolonged latency; the amplitudes of compound muscle action potentials were severely reduced in all four limbs and those of sensory nerve action potentials were moderately reduced in the right upper and lower extremities. We also found a left hilar lymphadenopathy showing accumulation of FDG on PET, suggesting a possibility of malignancy. Anti-Hu and anti-GluR epsilon 2 antibodies were detected in sera but not in CSF. We diagnosed him with limbic encephalitis and peripheral neuropathy due to paraneoplastic neurological syndrome and treated him with two courses of intravenous immunoglobulin (IVIg) (400 mg/kg, 5 days). The consciousness disturbance, and prolonged distal latency revealed by motor nerve conduction studies improved slightly. Although the roles of anti-neuronal antibodies in paraneoplastic conditions remain unknown, we consider that IVIg may be worth using to treat cases with anti-Hu and anti-GluR epsilon 2 antibodies.

EFA6A-like antibodies in paraneoplastic encephalitis associated with immature ovarian teratoma: a case report

We describe a 19-year-old patient with paraneoplastic encephalitis associated with immature ovarian teratoma (OT), who presented with psychiatric symptoms, prolonged disturbance of consciousness, refractory status epilepticus, central hypoventilation, and various abnormal involuntary movements. Immunological characterization of the patient's serum and cerebrospinal fluid (CSF) demonstrated the presence of an autoantibody that colocalized with EFA6A, a brain-specific protein involved in the regulation of dendritic development of hippocampal neurons. Despite the severity of the symptoms, the patient showed significant neurological improvement following removal of the tumor and chemotherapy. This case suggests that physicians should rule out an OT in young women with encephalitis who present with the subacute-onset of psychiatric symptoms. Antibodies that colocalize with EFA6A are a valuable marker for early diagnosis of a potentially reversible paraneoplastic encephalitis associated with OT.