Literature DB >> 15932358

Phenotypic variability in Kennedy's disease: implication of the early diagnostic features.

Jae-Hyeok Lee1, Jin-Hong Shin, Kyung-Pil Park, In-Joo Kim, Cheol-Min Kim, Jeong-Geun Lim, Young-Chul Choi, Dae-Seong Kim.   

Abstract

OBJECTIVES: The clinical diagnosis of Kennedy's disease (KD) is not easy when the typical manifestations are lacking, especially in early stage of the disease. In our study, we tried to identify the relative frequency of common clinical features and early symptoms in KD.
METHOD: Eighteen Korean patients with KD were included. Clinical findings were subdivided into two parts: the age at onset of each clinical symptoms and characteristic signs on investigations. With detailed clinical examinations, the serum creatine kinase (CK) level, electrophysiologic study and DNA analysis were performed and analyzed in detail.
RESULTS: In KD, the most consistent clinical findings at evaluations included perioral fasciculation with variable bulbar paresis, limb weakness with wasting, hyporeflexia, hand tremor, and elevated CK level. Some distinguishing features, such as X-linked family history, gynecomastia, and sensory abnormalities were absent in a half of cases. Frequent initial clinical findings include tremor (50%) and symptoms other than weakness, such as cramps and fatigability (33.3%).
CONCLUSION: We conclude that KD shows variable clinical and electrophysiological features. Our description on the onset and subsequent progression of each clinical finding might help to identify KD in early stage and avoid misdiagnosis. Copyright Blackwell Munksgaard 2005.

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Year:  2005        PMID: 15932358     DOI: 10.1111/j.1600-0404.2005.00428.x

Source DB:  PubMed          Journal:  Acta Neurol Scand        ISSN: 0001-6314            Impact factor:   3.209


  10 in total

1.  Brain MRI shows white matter sparing in Kennedy's disease and slow-progressing lower motor neuron disease.

Authors:  Edoardo G Spinelli; Federica Agosta; Pilar M Ferraro; Giorgia Querin; Nilo Riva; Cinzia Bertolin; Ilaria Martinelli; Christian Lunetta; Andrea Fontana; Gianni Sorarù; Massimo Filippi
Journal:  Hum Brain Mapp       Date:  2019-03-28       Impact factor: 5.038

2.  The metabolic and endocrine characteristics in spinal and bulbar muscular atrophy.

Authors:  Angela Rosenbohm; Susanne Hirsch; Alexander E Volk; Torsten Grehl; Julian Grosskreutz; Frank Hanisch; Andreas Herrmann; Katja Kollewe; Wolfram Kress; Thomas Meyer; Susanne Petri; Johannes Prudlo; Carsten Wessig; Hans-Peter Müller; Jens Dreyhaupt; Jochen Weishaupt; Christian Kubisch; Jan Kassubek; Patrick Weydt; Albert C Ludolph
Journal:  J Neurol       Date:  2018-02-20       Impact factor: 4.849

3.  Gene therapy with AR isoform 2 rescues spinal and bulbar muscular atrophy phenotype by modulating AR transcriptional activity.

Authors:  Wooi F Lim; Mitra Forouhan; Thomas C Roberts; Jesse Dabney; Ruth Ellerington; Alfina A Speciale; Raquel Manzano; Maria Lieto; Gavinda Sangha; Subhashis Banerjee; Mariana Conceição; Lara Cravo; Annabelle Biscans; Loïc Roux; Naemeh Pourshafie; Christopher Grunseich; Stephanie Duguez; Anastasia Khvorova; Maria Pennuto; Constanza J Cortes; Albert R La Spada; Kenneth H Fischbeck; Matthew J A Wood; Carlo Rinaldi
Journal:  Sci Adv       Date:  2021-08-20       Impact factor: 14.136

4.  Tremor in X-linked recessive spinal and bulbar muscular atrophy (Kennedy's disease).

Authors:  Francisco A Dias; Renato P Munhoz; Salmo Raskin; Lineu César Werneck; Hélio A G Teive
Journal:  Clinics (Sao Paulo)       Date:  2011       Impact factor: 2.365

Review 5.  Targeted Molecular Therapies for SBMA.

Authors:  Carlo Rinaldi; Bilal Malik; Linda Greensmith
Journal:  J Mol Neurosci       Date:  2015-11-17       Impact factor: 3.444

6.  Abnormal eye movements in Kennedy disease.

Authors:  Matthew J Thurtell; Erik P Pioro; R John Leigh
Journal:  Neurology       Date:  2009-04-28       Impact factor: 9.910

7.  Clinical Characteristics and Genotype-Phenotype Correlation of Korean Patients with Spinal and Bulbar Muscular Atrophy.

Authors:  Ju Sun Song; Kyung-Ah Kim; Ju-Hong Min; Chang-Seok Ki; Jong-Won Kim; Duk Hyun Sung; Byoung Joon Kim
Journal:  Yonsei Med J       Date:  2015-07       Impact factor: 2.759

Review 8.  More than a bystander: the contributions of intrinsic skeletal muscle defects in motor neuron diseases.

Authors:  Justin G Boyer; Andrew Ferrier; Rashmi Kothary
Journal:  Front Physiol       Date:  2013-12-18       Impact factor: 4.566

Review 9.  Beyond motor neurons: expanding the clinical spectrum in Kennedy's disease.

Authors:  Raquel Manzano; Gianni Sorarú; Christopher Grunseich; Pietro Fratta; Emanuela Zuccaro; Maria Pennuto; Carlo Rinaldi
Journal:  J Neurol Neurosurg Psychiatry       Date:  2018-01-20       Impact factor: 10.154

10.  The French national protocol for Kennedy's disease (SBMA): consensus diagnostic and management recommendations.

Authors:  Pierre-François Pradat; Emilien Bernard; Philippe Corcia; Philippe Couratier; Christel Jublanc; Giorgia Querin; Capucine Morélot Panzini; François Salachas; Christophe Vial; Karim Wahbi; Peter Bede; Claude Desnuelle
Journal:  Orphanet J Rare Dis       Date:  2020-04-10       Impact factor: 4.123

  10 in total

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