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Charcot-Marie-Tooth disease type 1B(HMSN IB; CMT1B)

MedGen UID:
124377
Concept ID:
C0270912
Disease or Syndrome
Synonyms: CHARCOT-MARIE-TOOTH DISEASE, AUTOSOMAL DOMINANT, WITH FOCALLY FOLDED MYELIN SHEATHS, TYPE 1B; Charcot-Marie-Tooth disease, demyelinating, type 1b; CHARCOT-MARIE-TOOTH DISEASE, SLOW NERVE CONDUCTION TYPE, LINKED TO DUFFY; Charcot-Marie-Tooth disease, type IB; CHARCOT-MARIE-TOOTH NEUROPATHY, TYPE 1B; Hereditary motor and sensory neuropathy 1B; HEREDITARY MOTOR AND SENSORY NEUROPATHY I; HEREDITARY MOTOR AND SENSORY NEUROPATHY IB; Peroneal muscular atrophy
SNOMED CT: Charcot-Marie-Tooth disease, type IB (42986003)
Modes of inheritance:
Autosomal dominant inheritance
MedGen UID:
141047
Concept ID:
C0443147
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in heterozygotes. In the context of medical genetics, an autosomal dominant disorder is caused when a single copy of the mutant allele is present. Males and females are affected equally, and can both transmit the disorder with a risk of 50% for each child of inheriting the mutant allele.
 
Gene (location): MPZ (1q23.3)
 
Monarch Initiative: MONDO:0007307
OMIM®: 118200
Orphanet: ORPHA101082

Definition

Charcot-Marie-Tooth disease is a sensorineural peripheral polyneuropathy. Affecting approximately 1 in 2,500 individuals, Charcot-Marie-Tooth disease is the most common inherited disorder of the peripheral nervous system (Skre, 1974). Autosomal dominant, autosomal recessive, and X-linked forms have been recognized. Classification On the basis of electrophysiologic properties and histopathology, CMT has been divided into primary peripheral demyelinating (type 1, or HMSNI) and primary peripheral axonal (type 2, or HMSNII) neuropathies. The demyelinating neuropathies classified as CMT type 1 are characterized by severely reduced motor NCVs (less than 38 m/s) and segmental demyelination and remyelination with onion bulb formations on nerve biopsy. The axonal neuropathies classified as CMT type 2 are characterized by normal or mildly reduced NCVs and chronic axonal degeneration and regeneration on nerve biopsy (see CMT2A1; 118210). Distal hereditary motor neuropathy (dHMN) (see 158590), or spinal CMT, is characterized by exclusive motor involvement and sparing of sensory nerves (Pareyson, 1999). McAlpine (1989) proposed that the forms of CMT with very slow nerve conduction be given the gene symbol CMT1A (118220) and CMT1B, CMT1A being the gene on chromosome 17 and CMT1B being the gene on chromosome 1. CMT2 was the proposed symbol for the autosomal locus responsible for the moderately slow nerve conduction form of the disease (axonal). For a phenotypic description and discussion of genetic heterogeneity of the various subtypes of CMT, see CMTX1 (302800), CMT2A1 (118210), CMT3 (DSS; 145900), CMT4A (214400), and CMTDIB (606482). Genetic Heterogeneity of Autosomal Dominant Demyelinating CMT1 Autosomal dominant demyelinating CMT1 is a genetically heterogeneous disorder and can be caused by mutations in different genes; see CMT1A (118220), CMT1C (601098), CMT1D (607678), CMT1E (607734), CMT1F (607734), CMT1G (618279), CMT1H (619764), CMT1I (619742), and CMT1J (620111). See also 608236 for a related phenotype characterized by isolated slowed nerve conduction velocities (NCVs). [from OMIM]

Clinical features

From HPO
Limb muscle weakness
MedGen UID:
107956
Concept ID:
C0587246
Finding
Reduced strength and weakness of the muscles of the arms and legs.
Pes cavus
MedGen UID:
675590
Concept ID:
C0728829
Congenital Abnormality
An increase in height of the medial longitudinal arch of the foot that does not flatten on weight bearing (i.e., a distinctly hollow form of the sole of the foot when it is bearing weight).
Hammertoe
MedGen UID:
209712
Concept ID:
C1136179
Anatomical Abnormality
Hyperextension of the metatarsal-phalangeal joint with hyperflexion of the proximal interphalangeal (PIP) joint.
Foot dorsiflexor weakness
MedGen UID:
356163
Concept ID:
C1866141
Finding
Weakness of the muscles responsible for dorsiflexion of the foot, that is, of the movement of the toes towards the shin. The foot dorsiflexors include the tibialis anterior, the extensor hallucis longus, the extensor digitorum longus, and the peroneus tertius muscles.
Split hand
MedGen UID:
397570
Concept ID:
C2699510
Congenital Abnormality
A condition in which middle parts of the hand (fingers and metacarpals) are missing giving a cleft appearance. The severity is very variable ranging from slightly hypoplastic middle fingers over absent middle fingers as far as oligo- or monodactyl hands.
Ulnar claw
MedGen UID:
871311
Concept ID:
C4025799
Congenital Abnormality
An abnormal hand position characterized by hyperextension of the fourth and fifth fingers at the metacarpophalangeal joints and flexion of the interphalangeal joints of the same fingers such that they are curled towards the palm.
Peripheral neuropathy
MedGen UID:
18386
Concept ID:
C0031117
Disease or Syndrome
Peripheral neuropathy is a general term for any disorder of the peripheral nervous system. The main clinical features used to classify peripheral neuropathy are distribution, type (mainly demyelinating versus mainly axonal), duration, and course.
Areflexia
MedGen UID:
115943
Concept ID:
C0234146
Finding
Absence of neurologic reflexes such as the knee-jerk reaction.
Steppage gait
MedGen UID:
98105
Concept ID:
C0427149
Finding
An abnormal gait pattern that arises from weakness of the pretibial and peroneal muscles due to a lower motor neuron lesion. Affected patients have footdrop and are unable to dorsiflex and evert the foot. The leg is lifted high on walking so that the toes clear the ground, and there may be a slapping noise when the foot strikes the ground again.
Hyporeflexia
MedGen UID:
195967
Concept ID:
C0700078
Finding
Reduction of neurologic reflexes such as the knee-jerk reaction.
Peripheral demyelination
MedGen UID:
451074
Concept ID:
C0878575
Pathologic Function
A loss of myelin from the internode regions along myelinated nerve fibers of the peripheral nervous system.
Hypertrophic nerve changes
MedGen UID:
322038
Concept ID:
C1832776
Finding
Myelin outfoldings
MedGen UID:
334341
Concept ID:
C1843168
Finding
The presence of excessive redundant myelin in the peripheral nerve sheath.
Distal sensory impairment
MedGen UID:
335722
Concept ID:
C1847584
Finding
An abnormal reduction in sensation in the distal portions of the extremities.
Onion bulb formation
MedGen UID:
376237
Concept ID:
C1847906
Finding
Repeated episodes of segmental demyelination and remyelination lead to the accumulation of supernumerary Schwann cells around axons, which is referred to as onion bulb formation. This finding affects peripheral nerves.
Decreased number of peripheral myelinated nerve fibers
MedGen UID:
346872
Concept ID:
C1858285
Finding
A loss of myelinated nerve fibers in the peripheral nervous system (in general, this finding can be observed on nerve biopsy).
Decreased motor nerve conduction velocity
MedGen UID:
388130
Concept ID:
C1858729
Finding
A type of decreased nerve conduction velocity that affects the motor neuron.
Distal muscle weakness
MedGen UID:
140883
Concept ID:
C0427065
Finding
Reduced strength of the musculature of the distal extremities.
Kyphoscoliosis
MedGen UID:
154361
Concept ID:
C0575158
Anatomical Abnormality
An abnormal curvature of the spine in both a coronal (lateral) and sagittal (back-to-front) plane.
Distal amyotrophy
MedGen UID:
338530
Concept ID:
C1848736
Disease or Syndrome
Muscular atrophy affecting muscles in the distal portions of the extremities.
Cold-induced muscle cramps
MedGen UID:
396193
Concept ID:
C1861675
Finding
Sudden and involuntary contractions of one or more muscles brought on by exposure to cold temperatures.
Tonic pupil
MedGen UID:
52779
Concept ID:
C0040416
Sign or Symptom
An abnormality of the pupillary light reaction characterized by a marked slowing of the light reaction of usually just one pupil. The pupil tends to be relatively dilated, and there is reduced accommodation.

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
Follow this link to review classifications for Charcot-Marie-Tooth disease type 1B in Orphanet.

Professional guidelines

PubMed

Lei L, Xiaobo L, Zhiqiang L, Yongzhi X, Shunxiang H, Huadong Z, Beisha T, Ruxu Z
Eur J Neurol 2023 Apr;30(4):1069-1079. Epub 2023 Feb 5 doi: 10.1111/ene.15700. PMID: 36692866
Sanmaneechai O, Feely S, Scherer SS, Herrmann DN, Burns J, Muntoni F, Li J, Siskind CE, Day JW, Laura M, Sumner CJ, Lloyd TE, Ramchandren S, Shy RR, Grider T, Bacon C, Finkel RS, Yum SW, Moroni I, Piscosquito G, Pareyson D, Reilly MM, Shy ME; Inherited Neuropathies Consortium - Rare Disease Clinical Research Consortium (INC-RDCRC)
Brain 2015 Nov;138(Pt 11):3180-92. Epub 2015 Aug 25 doi: 10.1093/brain/awv241. PMID: 26310628Free PMC Article

Curated

Aretz S, Rautenstrauss B, Timmerman V
Eur J Hum Genet 2010 Sep;18(9) Epub 2010 May 26 doi: 10.1038/ejhg.2010.75. PMID: 20512157Free PMC Article

Recent clinical studies

Etiology

Lei L, Xiaobo L, Zhiqiang L, Yongzhi X, Shunxiang H, Huadong Z, Beisha T, Ruxu Z
Eur J Neurol 2023 Apr;30(4):1069-1079. Epub 2023 Feb 5 doi: 10.1111/ene.15700. PMID: 36692866
Sanmaneechai O, Feely S, Scherer SS, Herrmann DN, Burns J, Muntoni F, Li J, Siskind CE, Day JW, Laura M, Sumner CJ, Lloyd TE, Ramchandren S, Shy RR, Grider T, Bacon C, Finkel RS, Yum SW, Moroni I, Piscosquito G, Pareyson D, Reilly MM, Shy ME; Inherited Neuropathies Consortium - Rare Disease Clinical Research Consortium (INC-RDCRC)
Brain 2015 Nov;138(Pt 11):3180-92. Epub 2015 Aug 25 doi: 10.1093/brain/awv241. PMID: 26310628Free PMC Article
Speevak MD, Farrell SA
Eur J Med Genet 2013 Oct;56(10):566-9. Epub 2013 Jun 25 doi: 10.1016/j.ejmg.2013.06.004. PMID: 23811036
Huang LW, Lin KP, Chang MH, Liao YC, Liao KK, Soong BW, Lee YC
J Chin Med Assoc 2012 May;75(5):197-202. Epub 2012 Apr 29 doi: 10.1016/j.jcma.2012.03.005. PMID: 22632984
Roa BB, Warner LE, Garcia CA, Russo D, Lovelace R, Chance PF, Lupski JR
Hum Mutat 1996;7(1):36-45. doi: 10.1002/(SICI)1098-1004(1996)7:1<36::AID-HUMU5>3.0.CO;2-N. PMID: 8664899

Diagnosis

Cortese R, Zoccolella S, Muglia M, Patitucci A, Scarafino A, Paolicelli D, Simone IL
Brain Behav 2016 Dec;6(12):e00580. Epub 2016 Sep 25 doi: 10.1002/brb3.580. PMID: 28032003Free PMC Article
Huang LW, Lin KP, Chang MH, Liao YC, Liao KK, Soong BW, Lee YC
J Chin Med Assoc 2012 May;75(5):197-202. Epub 2012 Apr 29 doi: 10.1016/j.jcma.2012.03.005. PMID: 22632984
Lancaster E, Elman LB, Scherer SS
Muscle Nerve 2010 Apr;41(4):555-8. doi: 10.1002/mus.21546. PMID: 19918771Free PMC Article
Cartwright MS, Brown ME, Eulitt P, Walker FO, Lawson VH, Caress JB
Muscle Nerve 2009 Jul;40(1):98-102. doi: 10.1002/mus.21292. PMID: 19533637
Suter U, Patel PI
Hum Mutat 1994;3(2):95-102. doi: 10.1002/humu.1380030203. PMID: 7515304

Therapy

Patzkó A, Bai Y, Saporta MA, Katona I, Wu X, Vizzuso D, Feltri ML, Wang S, Dillon LM, Kamholz J, Kirschner D, Sarkar FH, Wrabetz L, Shy ME
Brain 2012 Dec;135(Pt 12):3551-66. doi: 10.1093/brain/aws299. PMID: 23250879Free PMC Article

Prognosis

Corrado L, Magri S, Bagarotti A, Carecchio M, Piscosquito G, Pareyson D, Varrasi C, Vecchio D, Zonta A, Cantello R, Taroni F, D'Alfonso S
Neuromuscul Disord 2016 Aug;26(8):516-20. Epub 2016 May 24 doi: 10.1016/j.nmd.2016.05.011. PMID: 27344971
Crehalet H, Latour P, Bonnet V, Attarian S, Labauge P, Bonello N, Bernard R, Millat G, Rousson R, Bozon D
Neurogenetics 2010 Feb;11(1):13-9. Epub 2009 May 28 doi: 10.1007/s10048-009-0199-8. PMID: 19475438
Magot A, Latour P, Mussini JM, Mourtada R, Guiheneuc P, Pereon Y
Muscle Nerve 2008 Aug;38(2):1055-9. doi: 10.1002/mus.21050. PMID: 18663734
Eggers SD, Keswani SC, Melli G, Cornblath DR
Muscle Nerve 2004 Jun;29(6):867-9. doi: 10.1002/mus.20034. PMID: 15170620
Roa BB, Warner LE, Garcia CA, Russo D, Lovelace R, Chance PF, Lupski JR
Hum Mutat 1996;7(1):36-45. doi: 10.1002/(SICI)1098-1004(1996)7:1<36::AID-HUMU5>3.0.CO;2-N. PMID: 8664899

Clinical prediction guides

Lei L, Xiaobo L, Zhiqiang L, Yongzhi X, Shunxiang H, Huadong Z, Beisha T, Ruxu Z
Eur J Neurol 2023 Apr;30(4):1069-1079. Epub 2023 Feb 5 doi: 10.1111/ene.15700. PMID: 36692866
Choi BO, Kim SB, Kanwal S, Hyun YS, Park SW, Koo H, Yoo JH, Hyun JW, Park KD, Choi KG, Chung KW
Int J Mol Med 2011 Sep;28(3):389-96. Epub 2011 Apr 18 doi: 10.3892/ijmm.2011.678. PMID: 21503568
Mazzeo A, Muglia M, Rodolico C, Toscano A, Patitucci A, Quattrone A, Messina C, Vita G
Acta Neurol Scand 2008 Nov;118(5):328-32. Epub 2008 Apr 12 doi: 10.1111/j.1600-0404.2008.01021.x. PMID: 18422810
Eggers SD, Keswani SC, Melli G, Cornblath DR
Muscle Nerve 2004 Jun;29(6):867-9. doi: 10.1002/mus.20034. PMID: 15170620
Roa BB, Warner LE, Garcia CA, Russo D, Lovelace R, Chance PF, Lupski JR
Hum Mutat 1996;7(1):36-45. doi: 10.1002/(SICI)1098-1004(1996)7:1<36::AID-HUMU5>3.0.CO;2-N. PMID: 8664899

Supplemental Content

Table of contents

    Clinical resources

    Practice guidelines

    • PubMed
      See practice and clinical guidelines in PubMed. The search results may include broader topics and may not capture all published guidelines. See the FAQ for details.
    • Bookshelf
      See practice and clinical guidelines in NCBI Bookshelf. The search results may include broader topics and may not capture all published guidelines. See the FAQ for details.

    Curated

    • EuroGenetest, 2010
      Clinical utility gene card for: HMSN/HNPP HMSN types 1, 2, 3, 6 (CMT1,2,4, DSN, CHN, GAN, CCFDN, HNA); HNPP

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