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RANDOMIZED CONTROLLED TRIAL
A Gordon Smith, J Robinson Singleton, Adrienne Aperghis, Christopher S Coffey, Peter Creigh, Merit Cudkowicz, Robin Conwit, Dixie Ecklund, Janel K Fedler, Anna Gudjonsdottir, Peter Hauer, David N Herrmann, Marianne Kearney, John Kissel, Elizabeth Klingner, Adam Quick, Cathy Revere, Amro Stino
IMPORTANCE: Cryptogenic sensory peripheral neuropathy (CSPN) is highly prevalent and often disabling due to neuropathic pain. Metabolic syndrome and its components increase neuropathy risk. Diet and exercise have shown promise but are limited by poor adherence. OBJECTIVE: To determine whether topiramate can slow decline in intraepidermal nerve fiber density (IENFD) and/or neuropathy-specific quality of life measured using the Norfolk Quality of Life-Diabetic Neuropathy (NQOL-DN) scale...
December 1, 2023: JAMA Neurology
#2
JOURNAL ARTICLE
Guangyi Gao, Jo A Wick, Alexandra R Brown, Richard J Barohn, Byron J Gajewski
The PAIN-CONTRoLS trial compared four medications in treating Cryptogenic sensory polyneuropathy. The primary outcome was a utility function that combined two outcomes, patients' pain score reduction and patients' quit rate. However, additional analysis of the individual outcomes could also be leveraged to inform selecting an optimal medication for future patients. We demonstrate how joint modeling of longitudinal and time-to-event data from PAIN-CONTRoLS can be used to predict the effects of medication in a patient-specific manner and helps to make patient-focused decisions...
2023: Communications in Statistics. Case Studies, Data Analysis and Applications
#3
JOURNAL ARTICLE
Ling Yi Lee, Cheng Yin Tan, Kum Thong Wong, Khean Jin Goh, Nortina Shahrizaila
BACKGROUND: With progress made in neurogenetics and neuroinflammation, the indications and value of nerve biopsies in the diagnostic evaluation of peripheral neuropathies are less clear. In this study, we aimed to evaluate the diagnostic yield of nerve biopsies in patients with peripheral neuropathies. METHODS: We performed a retrospective review of nerve biopsy reports from April 1998 to June 2021 of patients with peripheral neuropathies presenting to the Department of Pathology, University of Malaya Medical Centre, Kuala Lumpur, Malaysia...
January 2023: Journal of Clinical Neuroscience: Official Journal of the Neurosurgical Society of Australasia
#4
JOURNAL ARTICLE
Lawrence A Zeidman, Pravesh Saini, Peter Mai
OBJECTIVES: Up to 50% of small fiber neuropathy (SFN) cases are idiopathic, but novel antibodies to Trisulfated Heparin Disaccharide (TS-HDS) and fibroblast growth factor receptor 3 (FGFR-3) have been implicated in half of these cases; the role of anti-Plexin D1 is less clear. We aimed to clarify presentation and management of these patients. METHODS: An 18-month retrospective analysis revealed 54 cases of cryptogenic SFN who had testing for the 3 autoantibodies...
September 1, 2022: Journal of Clinical Neuromuscular Disease
#5
RANDOMIZED CONTROLLED TRIAL
Salman F Bhai, Alexandra Brown, Byron Gajewski, Kim S Kimminau, Lemuel R Waitman, Mamatha Pasnoor, Richard J Barohn
INTRODUCTION/AIMS: Peripheral neuropathies commonly affect quality of life of patients due to pain, sleep disturbances, and fatigue, although trials have not adequately explored these domains of care. The aim of this study was to assess the impact of nortriptyline, duloxetine, pregabalin, and mexiletine on pain, sleep, and fatigue in patients diagnosed with cryptogenic sensory polyneuropathy (CSPN). METHODS: We implemented a Bayesian adaptive design to perform a 12-wk multisite, randomized, prospective, open-label comparative effectiveness study in 402 CSPN patients...
October 2022: Muscle & Nerve
#6
JOURNAL ARTICLE
Lawrence A Zeidman, Konrad Kubicki
OBJECTIVE: Novel antibodies to trisulfated heparin disaccharide (TS-HDS) and fibroblast growth factor receptor 3 (FGFR-3) have been recently described in otherwise cryptogenic small fiber neuropathy (SFN) cases. Our goal was to further describe clinical features in such cases and to analyze treatment responses. METHODS: In a retrospective analysis, 40 cases of cryptogenic SFN in a university neuropathy clinic were identified. Of these, TS-HDS and FGFR-3 cases were identified, and clinical features and treatment responses were analyzed...
June 1, 2021: Journal of Clinical Neuromuscular Disease
#7
JOURNAL ARTICLE
Marie-Christine Reinert, David Pacheu-Grau, Claudia B Catarino, Thomas Klopstock, Andreas Ohlenbusch, Michael Schittkowski, Ekkehard Wilichowski, Peter Rehling, Knut Brockmann
BACKGROUND: Leber hereditary optic neuropathy (LHON) is the most common mitochondrial disorder and characterized by acute or subacute painless visual loss. Environmental factors reported to trigger visual loss in LHON mutation carriers include smoking, heavy intake of alcohol, raised intraocular pressure, and some drugs, including several carbonic anhydrase inhibitors. The antiepileptic drug sulthiame (STM) is effective especially in focal seizures, particularly in benign epilepsy of childhood with centrotemporal spikes, and widely used in pediatric epileptology...
February 4, 2021: Orphanet Journal of Rare Diseases
#8
REVIEW
Mohamed Kazamel, Amro Maher Stino, Albert Gordon Smith
Diabetic peripheral neuropathy and metabolic syndrome (MetS) are both global health challenges with well-established diagnostic criteria and significant impacts on quality of life. Clinical observations, epidemiologic evidence, and animal models of disease have strongly suggested MetS is associated with an elevated risk for cryptogenic sensory peripheral neuropathy (CSPN). MetS neuropathy preferentially affects small unmyelinated axons early in its course, and it may also affect autonomic and large fibers. CSPN risk is linked to MetS and several of its components including obesity, dyslipidemia, and prediabetes...
March 2021: Muscle & Nerve
#9
RANDOMIZED CONTROLLED TRIAL
Richard J Barohn, Byron Gajewski, Mamatha Pasnoor, Alexandra Brown, Laura L Herbelin, Kim S Kimminau, Dinesh Pal Mudaranthakam, Omar Jawdat, Mazen M Dimachkie, Stanley Iyadurai, Amro Stino, John Kissel, Robert Pascuzzi, Thomas Brannagan, Matthew Wicklund, Aiesha Ahmed, David Walk, Gordon Smith, Dianna Quan, Darryl Heitzman, Alejandro Tobon, Shafeeq Ladha, Gil Wolfe, Michael Pulley, Ghazala Hayat, Yuebing Li, Pariwat Thaisetthawatkul, Richard Lewis, Suur Biliciler, Khema Sharma, Kian Salajegheh, Jaya Trivedi, William Mallonee, Ted Burns, Mark Jacoby, Vera Bril, Tuan Vu, Sindhu Ramchandren, Mark Bazant, Sara Austin, Chafic Karam, Yessar Hussain, Christen Kutz, Paul Twydell, Stephen Scelsa, Hani Kushlaf, James Wymer, Michael Hehir, Noah Kolb, Jeffrey Ralph, Alexandru Barboi, Navin Verma, Moiz Ahmed, Anza Memon, David Saperstein, Jau-Shin Lou, Andrea Swenson, Tiyonnoh Cash
IMPORTANCE: Cryptogenic sensory polyneuropathy (CSPN) is a common generalized slowly progressive neuropathy, second in prevalence only to diabetic neuropathy. Most patients with CSPN have significant pain. Many medications have been tried for pain reduction in CSPN, including antiepileptics, antidepressants, and sodium channel blockers. There are no comparative studies that identify the most effective medication for pain reduction in CSPN. OBJECTIVE: To determine which medication (pregabalin, duloxetine, nortriptyline, or mexiletine) is most effective for reducing neuropathic pain and best tolerated in patients with CSPN...
January 1, 2021: JAMA Neurology
#10
REVIEW
W N Löscher, B Iglseder
The peripheral nervous system is subject to changes during the aging process, e.g., deep tendon reflexes decrease, as proprioception does. In contrast, polyneuropathies have to be distinguished from age-associated changes as independent diseases with etiologies similar to those in younger ages. Incidence of polyneuropathies is reported about 118/100,000, the overall prevalence in the general population is estimated to be about 1% and rises to up to 7% in the elderly. Etiology includes metabolic disorders, primary inflammatory polyneuropathies, systemic disorders and vasculitic neuropathies...
March 2020: Der Internist
#11
JOURNAL ARTICLE
Todd D Levine, Jafar Kafaie, Lawrence A Zeidman, David S Saperstein, Reyanna Massaquoi, Ruth J Bland, Alan Pestronk
INTRODUCTION: Causes of small-fiber peripheral neuropathies (SFN) are often undefined. In this study we investigated associations of serum autoantibodies, immunoglobulin G (IgG) vs fibroblast growth factor receptor-3 (FGFR-3), and immunoglobulin M (IgM) vs trisulfated heparan disaccharide (TS-HDS) in cryptogenic SFN. METHODS: One hundred fifty-five patients with biopsy-proven SFN and no identified cause for their neuropathy were blindly tested for serum IgM vs TS-HDS and IgG vs FGFR-3...
April 2020: Muscle & Nerve
#12
REVIEW
Ch S Pavlov, I V Damulin, Yu O Shulpekova, E A Andreev
The review discusses thesteps of vitamin B12 metabolism and its role in maintaining of neurological functions. The term "vitamin B12 (cobalamin)" refers to several substances (cobalamins) of a very similar structure. Cobalamin enters the body with animal products. On the peripherу cobalamin circulates only in binding with proteins transcobalamin I and II (complex cobalamin-transcobalamin II is designated as "holotranscobalamin"). Holotranscobalamin is absorbed by different cells, whereas transcobalamin I-binded vitamin B12 - only by liver and kidneys...
May 16, 2019: Terapevticheskiĭ Arkhiv
#13
EDITORIAL
Mamatha Pasnoor, A Gordon Smith
No abstract text is available yet for this article.
March 2019: Muscle & Nerve
#14
JOURNAL ARTICLE
Kumi Fujita, Kazuhiro Hatta
We had encountered the case of membranous glomerulonephritis (MGN) with dilated cardiomyopathy due to LMNA gene mutation. LMNA mutation was known as a cause of 'laminopathy' such as dilated cardiomyopathy, muscular dystrophy, neuropathy and so on. LMNA gene might be a candidate of genetic basis in cryptogenic MGN.
May 2018: CEN Case Reports
#15
JOURNAL ARTICLE
B Oyanguren, R Segoviano, E Alegria, E Besada, M Gonzalez-Salaices, M Eimil-Ortiz, C Lopez de Silanes
INTRODUCTION: Fabry's disease is an infrequent metabolic pathology linked to the X chromosome which causes a wide variety of signs and symptoms. CASE REPORT: A 39-year-old male who was admitted to our stroke unit with right-side hemiparesis (1 + 0) and dysarthria (1). The score on the National Institute of Health Stroke Scale was 2. The patient presented angiokeratomas in both thighs. A computerised axial tomography scan of the head showed left thalamic acute infarction...
May 16, 2017: Revista de Neurologia
#16
REVIEW
Amro M Stino, Albert G Smith
Peripheral neuropathy is a major cause of disability worldwide. Diabetes is the most common cause of neuropathy, accounting for 50% of cases. Over half of people with diabetes develop neuropathy, and diabetic peripheral neuropathy (DPN) is a major cause of reduced quality of life due to pain, sensory loss, gait instability, fall-related injury, and foot ulceration and amputation. Most patients with non-diabetic neuropathy have cryptogenic sensory peripheral neuropathy (CSPN). A growing body of literature links prediabetes, obesity and metabolic syndrome to the risk of both DPN and CSPN...
September 2017: Journal of Diabetes Investigation
#17
JOURNAL ARTICLE
Anna L Emanuel, Mariska D Nieuwenhoff, Erica S Klaassen, Ajay Verma, Mark H H Kramer, Rob Strijers, Alexander F J E Vrancken, Etto Eringa, Geert Jan Groeneveld, Erik H Serné
OBJECTIVE: This study investigated whether the relationship between neuropathy and microvascular dysfunction in patients with type 2 diabetes is independent of diabetes-related factors. For this purpose, we compared skin microvascular function in patients with type 2 diabetes with that of patients with cryptogenic axonal polyneuropathy (CAP), a polyneuropathy of unknown etiology. RESEARCH DESIGN AND METHODS: Cross-sectional information was collected from 16 healthy controls (HCs), 16 patients with CAP, 15 patients with type 2 diabetes with polyneuropathy (DPN), and 11 patients with type 2 diabetes without polyneuropathy...
April 2017: Diabetes Care
#18
REVIEW
Panagiotis Zis, Ptolemaios Georgios Sarrigiannis, Dasappaiah Ganesh Rao, Channa Hewamadduma, Marios Hadjivassiliou
Chronic idiopathic axonal polyneuropathy (CIAP) is a term describing neuropathies with both sensory and motor involvement in a length dependant distribution where neurophysiology reveals axonal damage, neuropathy onset is insidious and shows slow or no progression of the disease over at least 6 months with no aetiology being identified despite appropriate investigations. This entity merits further consideration given how common it is, the absence of clarity regarding aetiopathogenesis, natural history and therapies...
October 2016: Journal of Neurology
#19
Sanjay Pandey
Painful leg and moving toes (PLMT) syndrome is a distinct clinical entity and in majority of the patients there are some underlying causes related to spinal cord, cauda equina, or peripheral neuropathy. However, some cases are cryptogenic with no identifiable underlying cause. Response to treatment is disappointing in most of the cases. We report a 60-year-old lady who presented with very severe type of painful legs and moving toes (PLMT) with no underlying cause.
October 2015: Annals of Indian Academy of Neurology
#20
Partha Pal, Sayantan Ray, Sisir Kumar Patra, Anjan Kumar Das, Subhasis Dey, Rintu George
Authors describe the case of a 60-year-old diabetic man who presented with jaundice, ascites and significant weight loss over a period of 2 months. Physical examination revealed firm hepatomegaly with ascites. On evaluation, nephropathy, axonal neuropathy, carpal tunnel syndrome and decompensated cryptogenic liver disease with portal hypertension were found fitting with the diagnosis of diabetic nephropathy and neuropathy and nonalcoholic steato-hepatitis-associated cirrhosis, respectively. It was only after tissue diagnosis and serum protein electrophoresis that a definitive diagnosis of myeloma-related amyloidosis was made...
April 2015: Oxford Medical Case Reports
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