Publications (28)64.54 Total impact
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Article: Rituximab Improves Subclinical Temporal Dispersion of Distal Compound Muscle Action Potential in Anti-MAG/SGPG Neuropathy Associated with Waldenström Macroglobulinemia: A Case Report.
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ABSTRACT: Patients with anti-myelin-associated glycoprotein (MAG)/sulfated glucuronyl paragloboside (SGPG) neuropathy associated with Waldenström macroglobulinemia show demyelinating neuropathy, but the temporal dispersion of distal compound muscle action potential (CMAP) in motor nerve conduction studies (NCS), which represents heterogeneous demyelination at the motor nerve terminal, is rare. We report on a 70-year-old man with anti-MAG/SGPG neuropathy associated with Waldenström macroglobulinemia; he had a 2-year history of mild dysesthesia of the foot sole without any motor symptoms. He showed marked temporal dispersion of distal CMAP in the tibial nerve with other demyelinating findings in the NCS. The temporal dispersion of distal CMAP in the tibial nerve improved significantly, and motor function was again normal 1 year after rituximab monotherapy. The temporal dispersion of distal CMAP in anti-MAG/SGPG neuropathy is rare, but it could occur from an early stage when the patients show mild or no motor symptoms. Rituximab therapy before secondary axonal degeneration has great potential to reverse the effects of the demyelination including the temporal dispersion of distal CMAP, and to prevent the deterioration of neuropathy in anti-MAG/SGPG neuropathy.Case Reports in Neurology 01/2013; 5(1):34-9. -
Article: First attack of Kleine-Levin syndrome triggered by influenza B mimicking influenza-associated encephalopathy.
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ABSTRACT: Six days after the onset of influenza B symptoms, a 14-year-old Japanese boy presented with encephalopathy-like symptoms, somnolence, irritability, and childishness, which we first considered was an atypical type of influenza-associated encephalopathy because the infection symptoms disappeared by day 4. His encephalopathy-like symptoms gradually improved, although he had repetitive hypersomnia attacks. Owing to the patient's clinical presentation and normal interleukin-6 levels in the cerebrospinal fluid during the first period of hypersomnia, we diagnosed him with Kleine-Levin syndrome (KLS) triggered by influenza B. The preceding influenza infection was not only a diagnostic clue of KLS but also a diagnostic confounding factor.Internal Medicine 01/2012; 51(12):1605-8. · 0.94 Impact Factor -
Article: Reversible extensive leukoencephalopathy in Sweet disease: a case report.
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ABSTRACT: Sweet disease, also known as acute febrile neutrophilic dermatosis, is an idiopathic multisystem inflammatory disorder characterised by erythematous skin lesions and fever. "Neuro-Sweet disease" is a rare central nervous system involvement that coexists with the characteristic cutaneous lesions of Sweet disease. Here, we report a case of Sweet disease complicated with acute encephalopathy. This case showed extensive lesions in the cerebral white matter on magnetic resonance imaging (MRI) and high protein concentration in the cerebrospinal fluid (CSF) without pleocytosis. After steroid therapy, the patient's clinical syndrome recovered completely with no significant neurological deficits or abnormal findings on brain MRI, and normalization of the high CSF protein concentration. Both clinical and MRI findings suggested that the marked leukoencephalopathy in this case was mainly due to reversible oedema rather than destructive structural alterations in the cerebral parenchyma.Journal of the Neurological Sciences 10/2008; 275(1-2):178-80. · 2.35 Impact Factor -
Article: Pictures in clinical medicine. Spinal epidural abscess with osteomyelitis as a cause of bacterial meningitis.
Internal Medicine 02/2008; 47(9):883. · 0.94 Impact Factor -
Article: Rituximab therapy in chronic inflammatory demyelinating polyradiculoneuropathy with anti-SGPG IgM antibody.
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ABSTRACT: We report a patient with chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) who showed high titers of anti-sulfated glucuronyl paragloboside (SGPG) IgM antibody without M-protein in serum. The patient was resistant to corticosteroids and immunosuppressants, but after administration of rituximab, clinical symptoms improved and the patient remained in a stable state for approximately 10 months. Rituximab may be a potent therapeutic option for refractory cases of CIDP irrespective of detectable M-protein in either serum or urine.Journal of Clinical Neuroscience 08/2006; 13(6):683-7. · 1.25 Impact Factor -
Article: High-dose melphalan followed by autologous stem cell support in primary systemic AL amyloidosis with multiple organ involvement.
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ABSTRACT: A patient with primary systemic AL amyloidosis achieved partial hematological response after 2 courses of VAD (vincristine, doxorubicin and dexamethasone) and subsequent high-dose melphalan followed by autologous peripheral blood stem cell transplantation despite involvement of multiple organs, including the heart. In this patient natriuretic peptides and free light chains in serum were useful as markers of cardiac involvement and therapeutic effects, respectively. When amyloidosis-related dysfunction is seen in multiple organs, this intensive chemotherapy might be a possible therapeutic option, although several modifications in the regimen and careful management are necessary.Internal Medicine 06/2005; 44(5):484-9. · 0.94 Impact Factor -
Article: Nonparaneoplastic, nonherpetic limbic encephalitis with severe episodic hypothermia: a case report.
European Neurology 02/2005; 54(3):170-4. · 1.81 Impact Factor -
Article: Chronic inflammatory demyelinating polyradiculoneuropathy with autonomic involvement.
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ABSTRACT: We report a case of chronic acquired neuropathy predominantly affecting sensory and autonomic nerves. Investigations showed a demyelinating polyradiculoneuropathy with axonal degeneration and depletion of postganglionic noradrenergic fibers in the rectal mucosa. Intravenous immunoglobulin and corticosteroid administration were effective in alleviating symptoms and improving electrophysiological abnormalities. This neuropathy may be a novel variant of chronic inflammatory demyelinating polyradiculoneuropathy (CIDP), in which autoimmunoreactivity is directed not only against myelin but also against axon- or ganglion-composing protein. Autonomic nerve involvement does not exclude a diagnosis of CIDP.Muscle & Nerve 02/2005; 31(1):108-12. · 2.37 Impact Factor -
Article: MRI analysis on a patient with the V30M mutation is characteristic of leptomeningeal amyloid.
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ABSTRACT: We report a characteristic finding in gadolinium-enhanced magnetic resonance images (MRIs) of the central nervous system (CNS) in a 61-year-old man with a homozygous transthyretin (TTR) Val30Met mutation. Although he presented with polyneuropathy accompanied by autonomic dysfunction and vitreous opacities in both eyes, he has shown no overt signs or symptoms of CNS involvement. Total protein level in the cerebrospinal fluid was moderately elevated. In the gadolinium-enhanced T1-weighted MRIs of the brain and spinal cord, leptomeningeal enhancement was seen along the surfaces of the brain stem and more clearly in the spinal cord, suggesting leptomeningeal TTR-related amyloid deposition. Our result indicates that gadolinium-enhanced MRI of the CNS may be a very sensitive and useful method for detecting leptomeningeal amyloid deposition, since abnormal findings can be detected even at a presymptomatic stage of CNS involvement.Amyloid 01/2005; 11(4):265-7. · 2.66 Impact Factor -
Article: Bilateral localized amyloidosis of the ureters: clinicopathology and therapeutic approaches in two cases.
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ABSTRACT: Localized amyloidosis in the ureter is a rare condition, in which immunoglobulin light chain is locally synthesized, causing thickening of ureteric walls by deposits of immunoglobulin-related amyloid. Since the clinical features of ureteral amyloidosis with ureteric stricture and/or hydroureteronephrosis closely resemble those of malignancy involving the ureters, nephroureterctomy is usually performed for this disease. We describe two aged patients with localized amyloidosis on the bilateral ureters. In both cases, left hydronephrosis with left ureteral stricture was found. They were treated with total nephroureterctomy and Alambda amyloid deposition was confirmed in the resected ureters. Several months later right ureteral stenosis was found. One patient was treated with percutaneous nephrostomy to preserve his renal function and the other with corticosteroids. This appeared to result in significant regression of the stenotic lesion. In both cases, all examinations for systemic involvement of organs were negative. Corticosteroids may be of use in treating immunoglobulin-derived localized amyloidosis in the ureters.Amyloid 01/2005; 11(4):260-4. · 2.66 Impact Factor -
Article: Mixed connective tissue disease with interstitial pneumonia in HTLV-1 carrier: case report and review of the literature.
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ABSTRACT: We report on a carrier of human T-lymphotropic virus type 1 (HTLV-1) who developed mixed connective tissue disease (MCTD). This patient suddenly manifested clinical symptoms and interstitial pneumonia ascribable to MCTD following long-term infection with HTLV-1. After initiation of oral prednisolone all manifestations quickly improved in parallel with a decrease in inflammatory reactions. In this patient HTLV-1 infection might have played an important role in the pathogenesis of MCTD. Since HTLV-1 can cause adult T-cell leukemia and HTLV-1-associated myelopathy, and also collagen diseases including MCTD, careful observation is necessary even in a carrier, particularly when autoantibodies are detectable in serum.Clinical Rheumatology 01/2005; 23(6):548-51. · 2.00 Impact Factor -
Article: Chronic pancreatitis associated with adult-onset type II citrullinemia: clinical and pathologic findings.
Annals of internal medicine 11/2004; 141(7):W109-10. · 16.73 Impact Factor -
Article: [Cervical myelopathy in a patient with congenital cervico-cerebral vascular malformation].
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ABSTRACT: We report a 50 year-old woman with cervical myelopathy. The patient, who had cutaneous angiomas in the right orbital area, became aware of left upper limb weakness when she woke up, followed by painful abnormal sensation in both axilla and arms. MRI revealed an intramedullar lesion mainly located in cervical cord at the level of C3-C4. Angiography showed that serpentine left vertebral artery entered the canalis vertebralis at C3 and fed the blood flow of bilateral middle cerebral arteries. In this case, the upper cervical spinal cord ischemia might be induced by hemodynamic insufficiency of the anterior spinal artery ascribed to congenital cervico-cerebral vascular malformation.Rinsho shinkeigaku = Clinical neurology 10/2004; 44(9):623-5. -
Article: [Serial brain MRI findings of a patient with thrombotic thrombocytopenic purpura complicated by malignant hypertension and reversible posterior leukoencephalopathy syndrome].
Nihon Naika Gakkai Zasshi 08/2004; 93(7):1445-7. -
Article: Sarcoidosis with high serum levels of vascular endothelial growth factor (VEGF), showing RS3PE-like symptoms in extremities.
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ABSTRACT: We report a patient with sarcoidosis who showed edema in the distal portion of all extremities, particularly the legs, as seen in remitting seronegative symmetrical synovitis with pitting edema (RS3PE). Magnetic resonance imaging demonstrated diffuse abnormal intensity in subcutaneous tissues of both legs, and skin biopsy led to a diagnosis of sarcoidosis. Vascular endothelial growth factor (VEGF) showed a high serum level, which decreased soon after starting oral prednisolone, in parallel with an improvement in the limb edema. In this patient VEGF as well as infiltration by sarcoid granuloma in the skin might have played an important role in the pathogenesis of RS3PE-like symptoms in the extremities. When painful pitting edema is seen predominantly in the distal portion of all extremities, sarcoidosis as well as RS3PE should be considered as a possible diagnosis.Clinical Rheumatology 07/2004; 23(3):246-8. · 2.00 Impact Factor -
Article: IgM AL amyloidosis due to B cell lymphoproliferative disorder: efficacy of high-dose melphalan followed by autologous stem cell transplantation.
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ABSTRACT: This report concerns a patient with IgM AL amyloidosis due to a B cell lymphoproliferative disorder who was successfully treated with VAD and subsequent high-dose melphalan followed by autologous stem cell support. After this chemotherapeutic regimen, the patient showed complete hematological remission and improvement in nephrotic syndrome. These findings suggest that high-dose melphalan may also be effective for lymphoplasmacytoid cells producing monoclonal IgM which are phenotypically distinct from plasma cells. Myeloablative therapies, such as high-dose melphalan, should definitely be considered as a treatment option for AL amyloidosis, irrespective of the type of precursor immunoglobulin.Amyloid 07/2004; 11(2):130-5. · 2.66 Impact Factor -
Article: Efficacy of tacrolimus in treatment of polymyositis associated with myasthenia gravis.
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ABSTRACT: We report a patient with polymyositis (PM) associated with myasthenia gravis (MG). Both disorders had been controlled for around 15 years by oral prednisolone and a cholinesterase inhibitor following surgical removal of invasive thymoma and radiotherapy, but muscular weakness due to myalgia and an increase in serum levels of myogenic enzymes, mainly ascribable to the recurrence of PM, reappeared immediately after cessation of these drugs, which was done because the patient had multiple bone fractures and severe osteoporosis due to the long-term corticosteroid therapy. Oral tacrolimus was therefore tried, and produced an improvement in muscular symptoms in association with normalization of myogenic enzymes. PM associated with MG as in this patient might be the best indication for tacrolimus, considering its efficacy in MG, but this drug should also be actively considered as a therapeutic option in refractory cases of PM alone, particularly when either corticosteroids or other immunosuppressive agents are not usable.Clinical Rheumatology 07/2004; 23(3):262-5. · 2.00 Impact Factor -
Article: A patient with severe renal amyloidosis associated with an immunoglobulin gamma-heavy chain fragment.
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ABSTRACT: The authors report a patient with progressive renal dysfunction caused by severe renal amyloidosis associated with a gamma-heavy chain variable region (V(H)) fragment. The patient was a 71-year-old woman who had renal insufficiency without nephrotic syndrome. Laboratory data showed a monoclonal IgG lambda component in her serum and urine. Renal biopsy results showed massive amyloid deposition in the mesangial region, but the glomerular basement membranes and epithelial cells were preserved. Because immunohistochemical studies using antibodies against a number of known amyloid fibril proteins failed to detect the amyloid protein, the amyloid protein extracted from a small piece of the biopsied renal tissue was subjected to biochemical analysis. Sodium dodecyl sulfate-polyacrylamide gel electrophoresis analysis of the extracted amyloid protein showed a prominent band at 11-kDa, and this protein was identified by amino acid sequence analysis as a gamma-heavy chain variable region fragment (V(H)3 subgroup) without the first N-terminal residue. Our results indicate that the patient's renal amyloidosis was associated with a gamma-heavy chain variable region fragment. Microextraction and biochemical characterization of amyloid fibrils was of great use for reaching a definitive diagnosis.American Journal of Kidney Diseases 06/2004; 43(5):e23-8. · 5.43 Impact Factor -
Article: Feasibility of auxiliary partial orthotopic liver transplantation from living donors for patients with adult-onset type II citrullinemia.
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ABSTRACT: More than 20 patients with adult-onset type II citrullinemia have undergone liver transplantation, showing dramatic therapeutic effects. In Japan, living donor liver transplantation is the standard technique of liver transplantation because of the rare availability of cadaveric donors. The feasibility of auxiliary partial orthotopic liver transplantation (APOLT) for adult-onset type II citrullinemia to overcome the problem of a small-for-size graft in living donor liver transplantation has not been defined. We recently performed APOLT for patients with type II citrullinemia. Here, we present 2 patients: patient 1 was a 32-year-old man and patient 2 was a 43-year-old woman. Both patients suffered from hepatic encephalopathy, and laboratory data showed highly elevated plasma levels of ammonia and citrulline. In patient 1, the liver graft was obtained from a patient with familial amyloid polyneuropathy as a domino liver transplant. In patient 2, APOLT was performed after graft donation from her husband. The postoperative clinical courses of both patients were uneventful, and the neurological symptoms were completely resolved. The plasma concentrations of ammonia and citrulline normalized rapidly in both patients. APOLT can provide an adequate hepatocyte mass to correct the underlying enzyme deficiency in adult patients with type II citrullinemia. In addition, APOLT can be carried out safely to overcome the limitation of graft volume in living donor liver transplantation.Liver Transplantation 05/2004; 10(4):550-4. · 3.39 Impact Factor -
Article: Malignant lymphoma associated with rheumatoid arthritis, developing shortly after initiation of oral methotrexate.
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ABSTRACT: We report a patient with rheumatoid arthritis (RA) who developed malignant lymphoma of the diffuse large B-cell type in the right submandibular region shortly after initiation of oral methotrexate (MTX). Despite cessation of MTX, the lymphadenopathy did not regress, and only reached complete remission after 3 courses of CHOP therapy followed by irradiation. In this patient highly active RA itself was considered to be the main cause of malignant lymphoma, and MTX might have contributed to the development by modifying the immune system. When RA is highly active, MTX should be used carefully because of the possible development of malignant lymphoma as well as other serious complications.Internal Medicine 03/2004; 43(2):135-8. · 0.94 Impact Factor
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- Internal Medicine (5)
- Clinical Rheumatology (3)
- Amyloid (3)
- European Neurology (2)
- Internal Medicine (2)
Institutions
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2003–2008
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Shinshu University
- Division of Neurology and Rheumatology
Matsumoto, Nagano-ken, Japan
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