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World Journal of Clinical Cases 2024年第3期12卷 601-606页

作者： Jia-Rong Li Lei-Yu Feng Jian-Wei Li Yu Liao Fei-Qi Liu Department of General Medicine Xiangtan Central HospitalXiangtan 411100Hunan ProvinceChina

BACKGROUND polyneuropathy,organomegaly,endocrinopathy,M-protein,skin changes(POEMS)syndrome is a rare paraneoplastic syndrome that encompass multiple systems.The most common clinical symptoms of POEMS syndrome are pro-gressive sensorimotor polyneuropathy,organ enlargement,endocrine disorders,darkening skin,a monoclonal plasma cell proliferative disorder,and lymph node hyperplasia.The organomegaly consists of hepatosplenomegaly and/or lym-phadenopathy;cases of cardiomyopathy are rare.Diagnoses are often delayed because of the atypical nature of the syndrome,exposing patients to possibly severe disability.Therefore,identifying atypical symptoms can improve the prognosis and quality of life among POEMS syndrome patients.lenalidomide and dexamethasone.CONCLUSION When patients with cardiomyopathy have systemic manifestations such as numb limbs and darkening skin,the POEMS syndrome is the most possible diagnosis.

关键词： polyneuropathy organomegaly endocrinopathy M-protein skin changes syndrome Dilated cardiomyopathy Lenalidomide Dexamethasone Case report

Intensive care unit-acquired weakness and mechanical ventilation:A reciprocal relationship

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World Journal of Clinical Cases 2024年第18期12卷 3644-3647页

作者： Ranjeet Kumar Sinha Sony Sinha Prateek Nishant Arvind Kumar Morya Department of Community Medicine Patna Medical CollegeBiharPatna 800004India Department of Ophthalmology-Vitreo-Retina Neuro-Ophthalmology and OculoplastyAll India Institute of Medical SciencesBiharPatna 801507India Department of Ophthalmology ESIC Medical CollegeBiharPatna 801113India Department of Ophthalmology All India Institute of Medical SciencesTelanganaHyderabad 508126India

Intensive care unit-acquired weakness(ICU-AW;ICD-10 Code:G72.81)is a syndrome of generalized weakness described as clinically detectable weakness in critically ill patients with no other credible cause.The risk factors for ICU-AW include hyperglycemia,parenteral nutrition,vasoactive drugs,neuromuscular blocking agents,corticosteroids,sedatives,some antibiotics,immobilization,the disease severity,septicemia and systemic inflammatory response syndrome,multiorgan failure,prolonged mechanical ventilation(MV),high lactate levels,older age,female sex,and pre-existing systemic morbidities.There is a definite association between the duration of ICU stay and MV with ICU-AW.However,the interpretation that these are modifiable risk factors influencing ICU-AW,appears to be flawed,because the relationship between longer ICU stays and MV with ICU-AW is reciprocal and cannot yield clinically meaningful strategies for the prevention of ICU-AW.Prevention strategies must be based on other risk factors.Large multicentric randomized controlled trials as well as meta-analysis of such studies can be a more useful approach towards determining the influence of these risk factors on the occurrence of ICU-AW in different populations.

关键词： Critical illness Meta-analysis Morbidity Myopathy polyneuropathy Risk factors

Clinical and electrophysiological profiles in early recognition of polyneuropathy, organomegaly, endocrinopathy, M-protein, and skin changes syndrome

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Chinese Medical Journal 2019年第14期132卷 1666-1672页

作者： Qin Wang Peng Liu Li-Li Ji Shuai Wu Guo-Dong Feng Xin Wang Ji-Hong Dong Department of Neurology Zhongshan Hospital Fudan UniversityShanghai 200032China Department of Haematology Zhongshan Hospital Fudan UniversityShanghai 200032China

Background:The detection of polyneuropathy,organomegaly,endocrinopathy,M-protein,and skin changes (POEMS) syndrome at early stage is challenging for neurologists.Since polyneuropathy could be the first manifestation,it could be misdiagnosed as chronic inflammatory demyelinating polyneuropathy (CIDP).The present study aimed to determine the clinical and electrophysiological features of POEMS syndrome to distinguish from CIDP.Methods:The data of a group of patients with POEMS (n =17) and patients with CIDP (n =17) in Zhongshan Hospital Fudan University from January 2015 to September 2017 were analyzed in this retrospective study.The clinical features,neurological symptoms,and electrophysiological findings were compared between the two groups.Results:Clinically,patients with POEMS demonstrated significantly more neuropathic pain in the lower extremities than patients with CIDP (58.8 % vs.11.8 %,P =0.01).Multisystem features like edema,skin change,organomegaly,and thrombocytosis were also pointed towards the diagnosis of POEMS syndrome.Electrophysiologically,terminal latency index (TLI) was significantly higher in patients with POEMS than that in patients with CIDP (median nerve:0.39 [0.17-0.52] vs.0.30 (0.07-0.69),Z =-2.413,P =0.016;ulnar nerve:0.55 [0.23-0.78] vs.0.42 [0.12-0.70],Z =-2.034,P =0.042).Patients with POEMS demonstrated a higher frequency of absent compound muscle action potential of the tibial nerve (52.9% vs.17.6%,P =0.031),less conduction block (ulnar nerve:0 vs.35.3%,P =0.018),and less temporal dispersion (median nerve:17.6% vs.58.8%,P =0.032) than CIDP group.The combination of positive serum monoclonal protein and high TLI (if either one or both were present) discriminated POEMS from CIDP with a sensitivity of 94.1% and 47.1% and specificity of 76.5% and 100.0%,respectively.Conclusions:POEMS syndrome could be distinguished from CIDP through typical clinical and electrophysiological characteristics in practice.The combination of serum monoclonal protein and high TLI might raise the sensitivity of de

关键词： polyneuropathy Organomegaly Endocrinopathy M protein and skin changes syndrome Chronic inflammatory demyelinating polyradiculoneuropathy Terminal latency index

Demyelinating polyneuropathy and lymphoplasmacytic lymphoma coexisting in 36-year-old man:A case report

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World Journal of Clinical Cases 2020年第12期8卷 2566-2573页

作者： Lesia Rozlucka Elzbieta Semik-Grabarczyk Marta Pietrukaniec Agnieszka Zak-Golab Malgorzata Grabarczyk Sebastian Grosicki Michal Holecki Department of Internal Medicine Allergology and Clinical ImmunologyFaculty of Medical Sciences in ZabrzeMedical University of SilesiaKatowice 40-752Poland Department of Internal Medicine Autoimmune and Metabolic DiseasesFaculty of Medical Sciences in KatowiceMedical University of SilesiaKatowice 40-752Poland Student Scientific Society at the Department of Internal Medicine Autoimmune and Metabolic DiseasesFaculty of Medical Sciences in KatowiceMedical University of SilesiaKatowice 40-752Poland Department of Hematology and Cancer Prevention Faculty of Health Sciences in BytomMedical University of SilesiaBytom41-902Poland

BACKGROUND Lymphoplasmacytic lymphoma is a rare non-Hodgkin’s lymphoma,occurring mostly in the elderly.It develops slowly and leads to malignant proliferation of lymphoid line cells in the bone marrow,lymph nodes and spleen.It may also affect nerve roots and meninges;some patients develop sensorimotor polyneuropathy which may precede general symptoms of lymphoma.CASE SUMMARY We present a case of a 36-year-old man diagnosed in 2012 with chronic inflammatory demyelinating polyneuropathy(CIDP),then he was hospitalized in 2019 due to progressive symptoms of heart failure and significant weight loss over the previous four months.Based on clinical and laboratory findings a diagnosis of lymphoplasmacytic lymphoma was suspected and confirmed by bone marrow flow cytometry.There was no improvement in the results of laboratory tests and the patient's condition after immediate implementation of chemotherapy.Patient died on the fifth day of treatment.CONCLUSION While CIDP and malignant disease co-occurrence is rare,it should be suspected and investigated in patients with atypical neuropathy symptoms.

关键词： Lymphoplasmacytic lymphoma Chronic inflammatory demyelinating polyneuropathy polyneuropathy Paraneoplastic neuropathy Autoimmunity Case report

Conductive hearing loss in chronic inflammatory demyelinating polyneuropathy(CIDP):A case report

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Journal of Otology 2018年第4期13卷 141-144页

作者： Mark A.Fadel Kevin Y.Zhan Edward E.Dodson University of Toledo College of Medicine&Life Sciences 3000 Arlington AveToledoOH43614USA The Ohio State University Wexner Medical Center Department of Otolaryngology-Head&Neck Surgery915 Olentangy River RdColumbusOH43212USA

Chronic inflammatory demyelinating polyneuropathy(CIDP) is a progressive autoimmune disorder that targets peripheral nerves. It commonly presents with motor-predominant dysfunction and enlargement of cranial nerves. With regards to hearing loss, a few cases of sensorineural loss have been described. We present a novel case of conductive hearing loss caused by a mass on the tympanic segment of the facial nerve in the setting of CIDP.

关键词： Chronic inflammatory demyelinating polyneuropathy Hearing loss Ossicular erosion Cranial neuropathy

Dissecting pathophysiology of a human dominantly inherited disease, familial amyloidotic polyneuropathy, by using genetically engineered mice

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Frigid Zone Medicine 2022年第2期2卷 65-75页

作者： Zhenghua Li Kenichi Yamamura Department of Histology and Embryology Harbin Medical UniversityHarbin 150081China Institute of Resource Development and Analysis Kumamoto UniversityKumamoto 860-0811Japan TransGenic Inc.Fukuoka 810-0001Japan

Familial amyloidotic polyneuropathy(FAP)is a type of systemic amyloidosis characterized by peripheral and autonomic neuropathy.Although FAP is a typical autosomal dominant disorder caused by a point mutation in the TTR gene,the average age at onset varies significantly among different countries.This discrepancy clearly suggests that a combination of intrinsic factors as well as extrinsic(environmental)factors shapes the development of FAP.However,these factors are difficult to analyze in humans,because detailed pathologic tissue analysis is only possible at autopsy.Thus,mouse models have been produced and used to disentangle these factors.This review covers the mouse models produced thus far and how these models are applied to analyze intrinsic and extrinsic factors involved in disease development and to test drug efficacy.

关键词： amyloid polyneuropathy neuropathy

De novo intraocular amyloid deposition after hepatic transplantation in familial amyloidotic polyneuropathy

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World Journal of Transplantation 2017年第4期7卷 243-249页

作者： Ivo Filipe Gama Leonor Duarte Almeida Ophthalmology Department Santa Maria University Hospital (North Lisbon Hospital Center)

The familiar amyloid polyneuropathy(FAP) is a rare autosomal-dominant systemic amyloidosis. Amyloid deposition occurs more frequently and extensively in the vitq. The increase in intraocular pressure(IOP) is a result of deposition of transthyretin(TTR) in trabecular meshwork. Rarely, the amyloid deposition in anterior segment can be more exuberant than in posterior segment. A 42 years old man, with FAP(Val30Met mutation), liver transplantation in 1997. He was asymptomatic, without any significant ocular abnormality until 2011. In 2011 he had an episode of pain in right eye(RE). Scalloped pupils, pupillary amyloid deposits and subtle vitreous opacities were detected. The IOP was 40 mmHg in RE and 28 mmHg in left eye(LE) with open angle. Optical coherence tomography detected a temporal superior retinal nerve fiber layer defect in LE and perimetry was normal. Topical timolol was initiated, and brimonidine was subsequently added to improve IOP control, which was achieved with topical medication until last evaluation. No progression occurred since 2011. Actually, with longer life expectancies, there is an increased risk of ocular involvement in FAP, even after liver transplantation. Although rare, a more exuberant amyloid deposition in anterior segment vs posterior segment can occur, and supports an important role of amyloid production in ciliary pigment epithelium in these patients. Medical control of IOP and a stable course are unusual in this secondary glaucoma. Ophthalmologists have an important task in the follow-up of patients and early diagnosis of risk factors for secondary glaucoma, such as scalloped pupils with amyloid deposits.

关键词： Familial amyloid polyneuropathy Glaucoma Scalloped pupils Pupillary amyloid deposits Liver transplantation

Pericarditis and chronic inflammatory demyelinating polyneuropathy during therapy with pegylated interferon alfa-2a for chronic hepatitis C

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World Journal of Hepatology 2010年第9期2卷 358-361页

作者： Kazuaki Nishio Takeshi Konndo Shunichi Okada Machiko Enchi The Third Department of Internal Medicine School of Medicine Showa University Department of Neurology Toyoko HospitalSt.Marianna University Kawasaki Social Insurance Hospital

We report a case of pericarditis and chronic inflam- matory demyelinating polyneuropathy with biological signs of a lupus-like syndrome due to pegylated interferon alfa-2a therapy during treatment for chronic hepatitis C.The patient developed moderate weakness in the lower limbs and dyspnea.He was hospitalized for congestive heart failure.An electrocardiogram showed gradual ST-segment elevation in leads V1 through V6 without coronary artery disease.A transthoracic cardiac ultrasonographic study revealed moderate pericardial effusion with normal left ventricular function.Anti-DNA antibody and anti-ds DNA IgM were positive.Neu ro logical examination revealed a symmetrical predomina ntly sensory polyneuropathy with impairment of light touch and pin prick in globe and stoking-like distribution.Treatment with prednisolone improved the pericarditis and motor nerve disturbance and the treatment with intravenous immunoglobulin improved the sensory nerve disturbance.

关键词： Chronic hepatitis C Chronic inflammatory demyelinating polyneuropathy Peginterferon alfa-2a Pericarditis

HIV-Associated polyneuropathy in Resource-Limited Settings: Genetic Predisposition and Vitamin Variations

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World Journal of AIDS 2017年第2期7卷 106-121页

作者： Frank N. Ndakala Julius O. Oyugi Margaret O. Oluka Directorate of Research Management and Development State Department of Science and Technology Nairobi Kenya Institute of Tropical Infectious Diseases (UNITID) University of Nairobi Nairobi Kenya Department of Pharmacology and Pharmacognosy University of Nairobi Nairobi Kenya

Human immunodeficiency virus-related polyneuropathy remains a painful condition resulting from damaged nerve endings. HIV infection strongly associates with a predominantly polyneuropathy that is attributed to HIV infection itself, or a toxic neuropathy associated with combination antiretroviral therapy (CART). In non-HIV-infected individuals, both deficiency and high intake of vitamins have been associated with polyneuropathy. For that reason, clinicians recommend vitamin supplements before and during CART. Although some, but not all, HIV-related vitamin deficiencies may replete during treatment with CART, it is predictable that high vitamin supplement intakes may contribute to nerve disorders. In resource-limited settings where the diagnosis of polyneuropathy heavily relies on symptoms, data on risk factors for polyneuropathy including vitamin status, alcohol consumption, and co-infections are limited. In addition, studies on genetic influence on the concentration of micronutrients in the blood of long-term users of CART are scarce. Possible sources of high intakes of vitamins could arise from the fact that a number of HIV-infected persons self-medicate. In addition, since HIV-infected individuals have an increased lifespan, relying on symptoms alone to specifically diagnose HIV-associated neuropathies could be a barrier to effective treatment in recourse-poor settings. This paper reviews evidence on single nucleotide polymorphisms (SNPs) with the potential to influence bioavailability of vitamins in HIV-infected patients. Genome-wide association studies have reported SNPs in alkaline phosphatase, fucosyltransferase 2, cubilin, transcobalamin 1, and tumor necrosis factor as potential determinants of various blood levels of vitamin B-6, B-12 and E. As long term CART increasingly become, personalized, future research should focus on SNPs, which influence vitamin blood levels, and with potential to augment long-term treatment with CART.

关键词： Human Immunodeficiency Virus Combination Antiretroviral Therapy Single Nucleotide Polymorphisms and polyneuropathy

Frequency of Distal Sensory polyneuropathy among Diabetics in Parakou in 2012

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Neuroscience & Medicine 2015年第3期6卷 90-97页

作者： Adoukonou Thierry Kouna-Ndouongo Philomène Sourokou Boni Justine Imorou Abdoulaye Houinato Dismand Unité d’Enseignement et de Recherche de Neurologie Faculté de Médecine Université de Parakou Parakou Benin Unité de Neurologie du service de Médecine du Centre Hospitalier Universitaire de Parakou Parakou Benin Service de Neurologie CHU Libreville Libreville Gabon Unité Diabète du Centre Hospitalier Universitaire de Parakou Parakou Benin Unité d’Enseignement et de Recherche de Neurologie Faculté des Sciences de la Santé Université d’Abomey-Calavi Cotonou Benin

Background: The distal sensory polyneuropathy (DSP) was more frequent among diabetics, although its determinant was not well known among diabetics in Benin. Objective: We aimed to assess the frequency of DSP and its determinants among diabetics at Parakou. Methods: It was a cross-sectional study carried out from 1 March to 31 August 2012 and included 336 diabetics followed at the diabetes unit of Parakou hospital. The diagnosis of DSP was based on DNS (Diabetic Neuropathy Score) criteria and other criteria. All data concerning the diabetes mellitus were recorded. We used Epi-Info and SPSS 16.0 software to perform analysis. Results: They were 187 females (56.0%) with the mean age of 54.9 ± 10.9 years. 298 patients fulfilled criteria for DSP;the overall prevalence of DSP was 88.7%. The main associated factors in multivariate analysis were treatment duration of diabetes more than 4 years: OR = 36.7 [4.0 - 336.9];the elevated glycaemia: OR = 3.1 [1.4 - 7.1];the activity with high income: OR = 0.2 [0.0 - 0.8];the ethnicity: nagots/fon: OR: 3.7 [1.4 - 12.5]. Conclusion: Those results suggested the high frequency of DSP among diabetics at Parakou.

关键词： Distal Sensory polyneuropathy Diabetes Neuropathy Frequency