Current status of the congenital myasthenic syndromes

Neuromuscular Disorders - Tập 22 Số 2 - Trang 99-111 - 2012
Andrew G. Engel1
1Department of Neurology, Mayo Clinic, Rochester, MN 55905, United States. [email protected]

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Engel, 1977, A new myasthenic syndrome with end-plate acetylcholinesterase deficiency, small nerve terminals, and reduced acetylcholine release, Ann Neurol, 1, 315, 10.1002/ana.410010403

Beeson, 2006, Dok-7 mutations underlie a neuromuscular junction synaptopathy, Science, 313, 1975, 10.1126/science.1130837

Selcen, 2008, Dok-7 myasthenia: phenotypic and molecular genetic studies in 16 patients, Ann Neurol, 64, 71, 10.1002/ana.21408

Maselli, 2009, Mutations in LAMB2 causing a severe form of synaptic congenital myasthenic syndrome, J Med Genet, 46, 203, 10.1136/jmg.2008.063693

Liewluck, 2010, Sporadic centronuclear myopathy with muscle pseudohypertrophy, neutropenia, and necklace fibers due to a DNM2 mutation, Neuromuscul Disord, 20, 801, 10.1016/j.nmd.2010.07.273

Engel, 1996, New mutations in acetylcholine receptor subunit genes reveal heterogeneity in the slow-channel congenital myasthenic syndrome, Hum Mol Genet, 5, 1217, 10.1093/hmg/5.9.1217

Ohno, 1998, Myasthenic syndromes in Turkish kinships due to mutations in the acetylcholine receptor, Ann Neurol, 44, 234, 10.1002/ana.410440214

Ohno, 2003, A frameshifting mutation in CHRNE unmasks skipping of the preceding exon, Hum Mol Genet, 12, 3055, 10.1093/hmg/ddg334

Senderek, 2011, Hexosamine biosynthetic pathway mutations cause neuromuscular transmission defect, Am J Hum Genet, 88, 162, 10.1016/j.ajhg.2011.01.008

Denning, 2007, High throughput genetic analysis of congenital myasthenic syndromes using resequencing microarrays, PLoS One, 2, e918, 10.1371/journal.pone.0000918

Kaiser, 2010, Affordable ‘exomes’ fill gaps in a catalog of rare diseases, Science, 330, 903, 10.1126/science.330.6006.903

Shinawi M, 2008, The array CGH and its clinical applications, Drug Discov Today, 13, 760, 10.1016/j.drudis.2008.06.007

Ohno, 2001, Choline acetyltransferase mutations cause myasthenic syndrome associated with episodic apnea in humans, Proc Natl Acad Sci USA, 98, 2017, 10.1073/pnas.98.4.2017

Shen, 2011, Functional consequences and structural interpretation of mutations in human choline acetyltransferase, Hum Mutat, 32, 1259, 10.1002/humu.21560

Byring, 2002, Congenital myasthenic syndrome associated with episodic apnea and sudden infant death, Neuromuscul Disord, 12, 548, 10.1016/S0960-8966(01)00336-4

Maselli, 2003, Choline acetyltransferase mutations in myasthenic syndrome due to deficient acetylcholine resynthesis, Muscle Nerve, 27, 180, 10.1002/mus.10300

Mallory, 2009, Congenital myasthenic syndrome with episodic apnea, Pediatr Neurol, 41, 42, 10.1016/j.pediatrneurol.2009.02.017

Schara, 2010, Long-term follow-up in patients with congenital myasthenic syndrome due to CHAT mutations, Eur J Paediatr Neurol, 14, 326, 10.1016/j.ejpn.2009.09.009

Kraner, 2003, Congenital myasthenic syndrome with episodic apnea in patients homozygous for a CHAT missense mutation, Arch Neurol, 60, 761, 10.1001/archneur.60.5.761

Engel, 1999, Congenital myasthenic syndromes, 251

Walls, 1993, Congenital myasthenic syndrome associated with paucity of synaptic vesicles and reduced quantal release, Ann NY Acad Sci, 681, 461, 10.1111/j.1749-6632.1993.tb22930.x

Bady, 1987, Congenital Lambert–Eaton myasthenic syndrome, J Neurol Neurosurg Psychiatry, 50, 476, 10.1136/jnnp.50.4.476

Ohno, 1998, Human endplate acetylcholinesterase deficiency caused by mutations in the collagen-like tail subunit (ColQ) of the asymmetric enzyme, Proc Natl Acad Sci USA, 95, 9654, 10.1073/pnas.95.16.9654

Kimbell, 2004, C-terminal and heparin-binding domains of collagenic tail subunit are both essential for anchoring acetylcholinesterase at the synapse, J Biol Chem, 279, 10997, 10.1074/jbc.M305462200

Mihaylova, 2008, Clinical and molecular genetic findings in COLQ-mutant congenital myasthenic syndromes, Brain, 131, 747, 10.1093/brain/awm325

Hutchinson, 1993, Congenital endplate acetylcholinesterase deficiency, Brain, 116, 633, 10.1093/brain/116.3.633

Ohno, 2000, The spectrum of mutations causing endplate acetylcholinesterase deficiency, Ann Neurol, 47, 162, 10.1002/1531-8249(200002)47:2<162::AID-ANA5>3.0.CO;2-Q

Donger, 1998, Mutation in the human acetylcholinesterase-associated gene, COLQ, is responsible for congenital myasthenic syndrome with end-plate acetylcholinesterase deficiency, Am J Hum Genet, 63, 967, 10.1086/302059

Bestue-Cardiel, 2005, Congenital endplate acetylcholinesterase deficiency responsive to ephedrine, Neurology, 65, 144, 10.1212/01.wnl.0000167132.35865.31

Brengman, 2006, Ephedrine treatment of seven patients with congenital endplate acetylcholinesterase deficiency, Neuromuscul Disord, 16, S129

Liewluck T, Selcen D, Engel AG. Beneficial effects of albuterol in congenital endplate acetylcholinesterase deficiency and DOK-7 myasthenia. Muscle Nerve; in press.

Engel, 1996, End-plate acetylcholine receptor deficiency due to nonsense mutations in the ε subunit, Ann Neurol, 40, 810, 10.1002/ana.410400521

Ohno, 1997, Congenital myasthenic syndromes due to heteroallelic nonsense/missense mutations in the acetylcholine receptor epsilon subunit gene: identification and functional characterization of six new mutations, Hum Mol Genet, 6, 753, 10.1093/hmg/6.5.753

Milone, 1998, Mode switching kinetics produced by a naturally occurring mutation in the cytoplasmic loop of the human acetylcholine receptor ε subunit, Neuron, 20, 575, 10.1016/S0896-6273(00)80996-4

Abicht, 1999, A common mutation (ε1267delG) in congenital myasthenic patients of Gypsy ethnic origin, Neurology, 53, 1564, 10.1212/WNL.53.7.1564

Middleton, 1999, Congenital myasthenic syndromes linked to chromosome 17p are caused by defects in acetylcholine receptor ε subunit gene, Neurology, 53, 1076, 10.1212/WNL.53.5.1076

Croxen, 1999, Novel functional ε-subunit polypeptide generated by a single nucleotide deletion in acetylcholine receptor deficiency congenital myasthenic syndrome, Ann Neurol, 46, 639, 10.1002/1531-8249(199910)46:4<639::AID-ANA13>3.0.CO;2-1

Abicht, 2002, A newly identified chromosomal microdeletion and an N-box mutation of the AChR ε gene cause a congenital myasthenic syndrome, Brain, 125, 1005, 10.1093/brain/awf095

Ealing, 2002, Mutations in congenital myasthenic syndromes reveal an ε subunit C-terminal cysteine, C470, crucial for maturation and surface expressions of adult AChR, Hum Mol Genet, 11, 3087, 10.1093/hmg/11.24.3087

Ohno, 1999, Congenital myasthenic syndrome caused by a mutation in the Ets-binding site of the promoter region of the acetylcholine receptor ε subunit gene, Neuromuscul Disord, 9, 131, 10.1016/S0960-8966(99)00007-3

Nichols, 1999, Mutation of the acetylcholine receptor ε-subunit promoter in congenital myasthenic syndrome, Ann Neurol, 45, 439, 10.1002/1531-8249(199904)45:4<439::AID-ANA4>3.0.CO;2-W

Ohno, 1996, Congenital myasthenic syndrome caused by decreased agonist binding affinity due to a mutation in the acetylcholine receptor ε subunit, Neuron, 17, 157, 10.1016/S0896-6273(00)80289-5

Quiram, 1999, Mutation causing congenital myasthenia reveals acetylcholine receptor βδ subunit interaction essential for assembly, J Clin Invest, 104, 1403, 10.1172/JCI8179

Müller, 2006, CHRND mutation causes a congenital myasthenic syndrome by impairing co-clustering of the acetylcholine receptor with rapsyn, Brain, 129, 2784, 10.1093/brain/awl188

Ohno, 1998, Frameshifting and splice-site mutations in acetylcholine receptor ε subunit gene in 3 Turkish kinships with congenital myasthenic syndromes, Ann NY Acad Sci, 841, 189, 10.1111/j.1749-6632.1998.tb10926.x

Sadeh, 2011, Beneficial effect of albuterol in congenital myasthenic syndrome with ε subunit mutations, Muscle Nerve, 44, 289, 10.1002/mus.22153

Engel, 2003, Sleuthing molecular targets for neurological diseases at the neuromuscular junction, Nature Rev Neurosci, 4, 339, 10.1038/nrn1101

Ohno, 1995, Congenital myasthenic syndrome caused by prolonged acetylcholine receptor channel openings due to a mutation in the M2 domain of the ε subunit, Proc Natl Acad Sci USA, 92, 758, 10.1073/pnas.92.3.758

Harper, 1998, Quinidine sulfate therapy for the slow-channel congenital myasthenic syndrome, Ann Neurol, 43, 480, 10.1002/ana.410430411

Harper, 2003, Treatment of slow channel congenital myasthenic syndrome with fluoxetine, Neurology, 60, 170, 10.1212/01.WNL.0000061483.11417.1B

Colomer, 2006, Long-term improvement of slow-channel myasthenic syndrome with fluoxetine, Neuromuscul Disord, 16, 329, 10.1016/j.nmd.2006.02.009

Chaouch A, Muller JS, Guergueltcheva V, et al. A retrospective clinical study of the treatment of slow-channel congenital myasthenic syndrome. J Neurol 2011, August 7, 2011 [Epub ahead of print].

Shen, 2008, Congenital myasthenia-related AChR δ subunit mutation interferes with intersubunit communication essential for channel gating, J Clin Invest, 118, 1867, 10.1172/JCI34527

Sine, 2006, Recent advances in Cys-loop receptor structure and function, NAT, 440, 448, 10.1038/nature04708

Engel, 2011, Highly fatal low-affinity fast-channel congenital myasthenic syndrome caused by a novel AChR ε subunit mutation at the agonist binding site, Neurology, 76, A644

Hesselmans, 1993, Development of innervation of skeletal muscle fibers in man: relation to acetylcholine receptors, Anat Rec, 236, 553, 10.1002/ar.1092360315

Morgan, 2006, Mutations in the embryonal subunit of the acetylcholine receptor (CHNRG) cause lethal and Escobar variants of the multiple pterygium syndrome, Am J Hum Genet, 79, 390, 10.1086/506256

Vogt, 2008, Mutation Analysis of CHRNA1, CHRNB1, CHRND, and RAPSN genes in multiple pterygium syndrome/fetal akinesia patients, Am J Hum Genet, 82, 222, 10.1016/j.ajhg.2007.09.016

Michalk, 2008, Acetylcholine receptor pathway mutations explain various fetal akinesia deformation sequence disorders, Am J Hum Genet, 82, 464, 10.1016/j.ajhg.2007.11.006

Vogt, 2009, Germline mutation in DOK7 associated with fetal akinesia deformation sequence, J Med Genet, 46, 338, 10.1136/jmg.2008.065425

Froehner, 1990, The postsynaptic 43K protein clusters muscle nicotinic acetylcholine receptors in Xenopus oocytes, Neuron, 5, 403, 10.1016/0896-6273(90)90079-U

Cartaud, 1998, Evidence for in situ and in vitro association between β-dystroglycan and the subsynaptic 43K rapsyn protein. Consequence for acetylcholine receptor clustering at the synapse, J Biol Chem, 273, 11321, 10.1074/jbc.273.18.11321

Mittaud, 2004, A single pulse of agrin triggers a pathway that acts to cluster acetylcholine receptors, Mol Cell Biol, 24, 7841, 10.1128/MCB.24.18.7841-7854.2004

Okada, 2006, The muscle protein Dok-7 is essential for neuromuscular synaptogenesis, Science, 312, 1802, 10.1126/science.1127142

Zhang, 2008, LRP4 serves as a coreceptor of agrin, Neuron, 60, 285, 10.1016/j.neuron.2008.10.006

Kim, 2008, Lrp4 is a receptor for Agrin and forms a complex with MuSK, Cell, 135, 334, 10.1016/j.cell.2008.10.002

Ohno, 2002, Rapsyn mutations in humans cause endplate acetylcholine receptor deficiency and myasthenic syndrome, Am J Hum Genet, 70, 875, 10.1086/339465

Ohno, 2003, E-box mutations in RAPSN promoter region in eight cases with congenital myasthenic syndrome, Hum Mol Genet, 12, 739, 10.1093/hmg/ddg089

Maselli, 2003, Rapsyn mutations in myasthenic syndrome due to impaired receptor clustering, Muscle Nerve, 28, 293, 10.1002/mus.10433

Burke, 2003, Rapsyn mutations in hereditary myasthenia. Distinct early- and late-onset phenotypes, Neurology, 61, 826, 10.1212/01.WNL.0000085865.55513.AE

Müller, 2004, A newly identified chromosomal microdeletion of the rapsyn gene causes a congenital myasthenic syndrome, Neuromuscul Disord, 14, 744, 10.1016/j.nmd.2004.06.010

Müller, 2003, Rapsyn N88K is a frequent cause of CMS in European patients, Neurology, 60, 1805, 10.1212/01.WNL.0000072262.14931.80

Cossins, 2006, Diverse molecular mechanisms involved in AChR deficiency due to rapsyn mutations, Brain, 129, 2773, 10.1093/brain/awl219

Muller, 2006, Impaired receptor clustering in congenital myasthenic syndrome with novel RAPSN mutations, Brain, 129, 2784, 10.1093/brain/awl188

Maselli, 2007, Congenital myasthenic syndrome caused by two non-N88K rapsyn mutations, Clin Genet, 72, 63, 10.1111/j.1399-0004.2007.00824.x

Milone, 2009, Myasthenic syndrome due to defects in rapsyn: clinical and molecular findings in 39 patients, Neurology, 73, 228, 10.1212/WNL.0b013e3181ae7cbc

Banwell, 2004, Novel truncating RAPSN mutation causing congenital myasthenic syndrome responsive to 3,4-diaminopyridine, Neuromuscul Disord, 14, 202, 10.1016/j.nmd.2003.11.004

Skeie, 2006, Unusual features in a boy with rapsyn N88K mutation, Neurology, 67, 2262, 10.1212/01.wnl.0000249184.09369.c2

Elliott, 1997, Plectin transcript diversity: identification and tissue distribution of variants with distinct first coding exons and rodless isoforms, Genomics, 42, 115, 10.1006/geno.1997.4724

Fuchs, 1999, Unusual 5′ transcript complexity of plectin isoforms: novel tissue-specific exons modulate actin binding activity, Hum Mol Genet, 8, 2461, 10.1093/hmg/8.13.2461

Konieczny, 2008, Myofiber integrity depends on desmin network targeting to Z-disks and costameres via distinct plectin isoforms, J Cell Biol, 181, 667, 10.1083/jcb.200711058

Banwell, 1999, Myopathy, myasthenic syndrome, and epidermolysis bullosa simplex due to plectin deficiency, J Neuropathol Exp Neurol, 58, 832, 10.1097/00005072-199908000-00006

Selcen, 2011, Myasthenic syndrome caused by plectinopathy, Neurology, 76, 327, 10.1212/WNL.0b013e31820882bd

Forrest, 2010, Congenital muscular dystrophy, myasthenic symptoms and epidermolysis bullosa simplex (EBS) associated with mutations in the PLEC1 gene encoding plectin, Neuromuscul Disord, 20, 709, 10.1016/j.nmd.2010.06.003

Maselli R, Arredondo J, Cagney O, et al. Congenital myasthenic syndrome associated with epidermolysis bullosa caused by homozygous mutaions in PLEC1 and CHRNE. Clin Genet; in press.

Gundesli, 2010, Mutation in exon 1f of PLEC, leading to disruption of plectin isoform 1f, causes autosomal-recessive limb-girdle muscular dystrophy, Am J Hum Genet, 87, 834, 10.1016/j.ajhg.2010.10.017

Tsujino, 2003, Myasthenic syndrome caused by mutation of the SCN4A sodium channel, Proc Natl Acad Sci USA, 100, 7377, 10.1073/pnas.1230273100

Huze, 2009, Identification of an agrin mutation that causes congenital myasthenia and affects synapse function, Am J Hum Genet, 85, 155, 10.1016/j.ajhg.2009.06.015

DeChiara, 1996, The receptor tyrosine kinase MuSK is required for neuromuscular junction formation in vivo, Cell, 85, 501, 10.1016/S0092-8674(00)81251-9

Herbst, 2000, The juxtamembrane region of MuSK has a critical role in agrin-mediated signaling, EMBO J, 19, 67, 10.1093/emboj/19.1.67

Nguyen, 2000, Nerve terminals form but fail to mature when postsyanptic differentiation is blocked: in vivo analysis using mammalian nerve–muscle chimeras, J Neurosci, 20, 6077, 10.1523/JNEUROSCI.20-16-06077.2000

Chevessier, 2004, MUSK, a new target for mutations causing congenital myasthenic syndrome, Hum Mol Genet, 13, 3229, 10.1093/hmg/ddh333

Chevessier, 2008, A mouse model for congenital myasthenic syndrome due to MuSK mutations reveals defects in structure and function of neuromuscular junctions, Hum Mol Genet, 17, 3577, 10.1093/hmg/ddn251

Maselli, 2010, Mutations in MUSK causing congenital myasthenic syndrome impair MuSK-Dok-7 interaction, Hum Mol Genet, 19, 2370, 10.1093/hmg/ddq110

Mihaylova, 2009, Refinement of the clinical phenotype in musk-related congenital myasthenic syndromes, Neurology, 73, 1926, 10.1212/WNL.0b013e3181c3fce9

Muller, 2007, Phenotypical spectrum of DOK7 mutations in congenital myasthenic syndromes, Brain, 130, 1497, 10.1093/brain/awm068

Anderson, 2008, Variable phenotypes associated with mutations in DOK7, Muscle Nerve, 37, 448, 10.1002/mus.20944

Ammar, 2010, Phenotype–genotype analysis in 15 patients presenting a congenital myasthenic syndrome due to mutaions in DOK7, J Neurol, 257, 754, 10.1007/s00415-009-5405-y

Hallock, 2010, Dok-7 regulates neuromuscular synapse formation by recruiting Crk and Crk-L, Genes Dev, 24, 2451, 10.1101/gad.1977710

Slater, 2006, Pre- and postsynaptic abnormalities associated with impaired neuromuscular transmission in a group of patients with ‘limb-girdle myasthenia’, Brain, 127, 2061, 10.1093/brain/awl200

Palace, 2007, Clinical features of the DOK7 neuromuscular junction synaptopathy, Brain, 130, 1507, 10.1093/brain/awm072

Srour, 2010, DOK7 mutations presenting as a proximal myopathy in French Canadians, Neuromuscul Disord, 20, 453, 10.1016/j.nmd.2010.05.007

Jephson, 2010, Congenital stridor with feeding difficulty as a presenting symptom of Dok7 congenital myasthenic syndrome, Int J Pediatr Otolaryng, 74, 991, 10.1016/j.ijporl.2010.05.022

Wu, 2010, To build a synapse: signaling pathways in neuromuscular junction assembly, Development, 137, 1017, 10.1242/dev.038711

Bergamin, 2010, The cytoplasmic adaptor protein Dok-7 activates the receptor tyrosine kinase MuSK via dimerization, Mol Cell, 39, 100, 10.1016/j.molcel.2010.06.007

Romero, 2010, Centronucelar myopathies: a widening concept, Neuromuscul Disord, 20, 223, 10.1016/j.nmd.2010.01.014

Claeys, 2010, Phenotype of a patient with recessive centronuclear myopathy and a novel BIN1 mutation, Neurology, 74, 519, 10.1212/WNL.0b013e3181cef7f9

Baradello, 1989, Adult-onset centronuclear myopathy: evidence against a neurogenic pathology, Acta Neurol Scand, 80, 162, 10.1111/j.1600-0404.1989.tb03860.x

Liewluck, 2011, Endplate structure and parameters of neuromuscular transmission in sporadic centronuclear myopathy associated with myasthenia, Neuromuscul Disord, 21, 387, 10.1016/j.nmd.2011.03.002

Robb, 2011, Impaired neuromuscular transmission and response to aceylcholinesterase inhibitors in centronuclear myopathy, Neuromuscul Disord, 21, 379, 10.1016/j.nmd.2011.02.012

Croxen, 2002, Myasthenia gravis in a woman with congenital AChR deficiency due to ε-subunit mutations, Neurology, 58, 1563, 10.1212/WNL.58.10.1563

Wintzen, 1998, Acquired slow-channel syndrome: a form of myasthenia gravis with prolonged open time of the acetylcholine receptor channel, Ann Neurol, 44, 657, 10.1002/ana.410440412