KEGG   DISEASE: Meckel syndrome
Entry
H00261                      Disease                                
Name
Meckel syndrome;
Meckel-Gruber syndrome
Description
Meckel syndrome (MKS) is a lethal, autosomal recessive disorder characterized by anomalies of the central nervous system, cystic dysplasia of the kidneys, and malformations of the hands and feet.
Category
Congenital malformation
Brite
Human diseases in ICD-11 classification [BR:br08403]
 20 Developmental anomalies
  Multiple developmental anomalies or syndromes
   LD2F  Syndromes with multiple structural anomalies, without predominant body system involvement
    H00261  Meckel syndrome
Gene
(MKS1) MKS1 [HSA:54903] [KO:K19332]
(MKS2) TMEM216 [HSA:51259] [KO:K19385]
(MKS3) TMEM67 [HSA:91147] [KO:K19348]
(MKS4) CEP290 [HSA:80184] [KO:K16533]
(MKS5) RPGRIP1L [HSA:23322] [KO:K16550]
(MKS6) CC2D2A [HSA:57545] [KO:K19352]
(MKS7) NPHP3 [HSA:27031] [KO:K19360]
(MKS8) TCTN2 [HSA:79867] [KO:K19361]
(MKS9) B9D1 [HSA:27077] [KO:K16744]
(MKS10) B9D2 [HSA:80776] [KO:K16745]
(MKS11) TMEM231 [HSA:79583] [KO:K19362]
(MKS12) KIF14 [HSA:9928] [KO:K17915]
(MKS13) TMEM107 [HSA:84314] [KO:K22764]
(MKS14) TXNDC15 [HSA:79770] [KO:K25389]
Other DBs
ICD-11: LD2F.13
MeSH: C536133 C536131 C536132
OMIM: 249000 603194 607361 611134 611561 612284 267010 613885 614209 614175 615397 616258 617562 619879
Reference
  Authors
Chen CP
  Title
Meckel syndrome: genetics, perinatal findings, and differential diagnosis.
  Journal
Taiwan J Obstet Gynecol 46:9-14 (2007)
DOI:10.1016/S1028-4559(08)60100-X
Reference
  Authors
Alexiev BA, Lin X, Sun CC, Brenner DS
  Title
Meckel-Gruber syndrome: pathologic manifestations, minimal diagnostic criteria, and differential diagnosis.
  Journal
Arch Pathol Lab Med 130:1236-8 (2006)
DOI:10.5858/2006-130-1236-MS
Reference
PMID:16415886 (MKS1)
  Authors
Kyttala M, Tallila J, Salonen R, Kopra O, Kohlschmidt N, Paavola-Sakki P, Peltonen L, Kestila M
  Title
MKS1, encoding a component of the flagellar apparatus basal body proteome, is mutated in Meckel syndrome.
  Journal
Nat Genet 38:155-7 (2006)
DOI:10.1038/ng1714
Reference
PMID:20512146 (TMEM216)
  Authors
Valente EM, Logan CV, Mougou-Zerelli S, Lee JH, Silhavy JL, Brancati F, Iannicelli M, Travaglini L, Romani S, Illi B, Adams M, Szymanska K, Mazzotta A, Lee JE, Tolentino JC, Swistun D, Salpietro CD, Fede C, Gabriel S, Russ C, Cibulskis K, Sougnez C, Hildebrandt F, Otto EA, Held S, Diplas BH, Davis EE, Mikula M, Strom CM, Ben-Zeev B, Lev D, Sagie TL, Michelson M, Yaron Y, Krause A, Boltshauser E, Elkhartoufi N, Roume J, Shalev S, Munnich A, Saunier S, Inglehearn C, Saad A, Alkindy A, Thomas S, Vekemans M, Dallapiccola B, Katsanis N, Johnson CA, Attie-Bitach T, Gleeson JG
  Title
Mutations in TMEM216 perturb ciliogenesis and cause Joubert, Meckel and related syndromes.
  Journal
Nat Genet 42:619-25 (2010)
DOI:10.1038/ng.594
Reference
PMID:26191240 (TMEM67)
  Authors
Zhang M, Cheng J, Liu A, Wang L, Xiong L, Chen M, Sun Y, Li J, Lu Y, Yuan H, Li Y, Lu Y
  Title
A missense mutation in TMEM67 causes Meckel-Gruber syndrome type 3 (MKS3): a family from China.
  Journal
Int J Clin Exp Pathol 8:5379-86 (2015)
Reference
PMID:17705300 (CEP290)
  Authors
Frank V, den Hollander AI, Bruchle NO, Zonneveld MN, Nurnberg G, Becker C, Du Bois G, Kendziorra H, Roosing S, Senderek J, Nurnberg P, Cremers FP, Zerres K, Bergmann C
  Title
Mutations of the CEP290 gene encoding a centrosomal protein cause Meckel-Gruber syndrome.
  Journal
Hum Mutat 29:45-52 (2008)
DOI:10.1002/humu.20614
Reference
PMID:17558409 (RPGRIP1L)
  Authors
Delous M, Baala L, Salomon R, Laclef C, Vierkotten J, Tory K, Golzio C, Lacoste T, Besse L, Ozilou C, Moutkine I, Hellman NE, Anselme I, Silbermann F, Vesque C, Gerhardt C, Rattenberry E, Wolf MT, Gubler MC, Martinovic J, Encha-Razavi F, Boddaert N, Gonzales M, Macher MA, Nivet H, Champion G, Bertheleme JP, Niaudet P, McDonald F, Hildebrandt F, Johnson CA, Vekemans M, Antignac C, Ruther U, Schneider-Maunoury S, Attie-Bitach T, Saunier S
  Title
The ciliary gene RPGRIP1L is mutated in cerebello-oculo-renal syndrome (Joubert syndrome type B) and Meckel syndrome.
  Journal
Nat Genet 39:875-81 (2007)
DOI:10.1038/ng2039
Reference
PMID:18513680 (CC2D2A)
  Authors
Tallila J, Jakkula E, Peltonen L, Salonen R, Kestila M
  Title
Identification of CC2D2A as a Meckel syndrome gene adds an important piece to the ciliopathy puzzle.
  Journal
Am J Hum Genet 82:1361-7 (2008)
DOI:10.1016/j.ajhg.2008.05.004
Reference
PMID:18371931 (NPHP3)
  Authors
Bergmann C, Fliegauf M, Bruchle NO, Frank V, Olbrich H, Kirschner J, Schermer B, Schmedding I, Kispert A, Kranzlin B, Nurnberg G, Becker C, Grimm T, Girschick G, Lynch SA, Kelehan P, Senderek J, Neuhaus TJ, Stallmach T, Zentgraf H, Nurnberg P, Gretz N, Lo C, Lienkamp S, Schafer T, Walz G, Benzing T, Zerres K, Omran H
  Title
Loss of nephrocystin-3 function can cause embryonic lethality, Meckel-Gruber-like syndrome, situs inversus, and renal-hepatic-pancreatic dysplasia.
  Journal
Am J Hum Genet 82:959-70 (2008)
DOI:10.1016/j.ajhg.2008.02.017
Reference
PMID:21462283 (TCTN2)
  Authors
Shaheen R, Faqeih E, Seidahmed MZ, Sunker A, Alali FE, AlQahtani K, Alkuraya FS
  Title
A TCTN2 mutation defines a novel Meckel Gruber syndrome locus.
  Journal
Hum Mutat 32:573-8 (2011)
DOI:10.1002/humu.21507
Reference
PMID:21763481 (B9D1 B9D2)
  Authors
Dowdle WE, Robinson JF, Kneist A, Sirerol-Piquer MS, Frints SG, Corbit KC, Zaghloul NA, van Lijnschoten G, Mulders L, Verver DE, Zerres K, Reed RR, Attie-Bitach T, Johnson CA, Garcia-Verdugo JM, Katsanis N, Bergmann C, Reiter JF
  Title
Disruption of a ciliary B9 protein complex causes Meckel syndrome.
  Journal
Am J Hum Genet 89:94-110 (2011)
DOI:10.1016/j.ajhg.2011.06.003
Reference
PMID:23349226 (TMEM231)
  Authors
Shaheen R, Ansari S, Mardawi EA, Alshammari MJ, Alkuraya FS
  Title
Mutations in TMEM231 cause Meckel-Gruber syndrome.
  Journal
J Med Genet 50:160-2 (2013)
DOI:10.1136/jmedgenet-2012-101431
Reference
PMID:24128419 (KIF14)
  Authors
Filges I, Nosova E, Bruder E, Tercanli S, Townsend K, Gibson WT, Rothlisberger B, Heinimann K, Hall JG, Gregory-Evans CY, Wasserman WW, Miny P, Friedman JM
  Title
Exome sequencing identifies mutations in KIF14 as a novel cause of an autosomal recessive lethal fetal ciliopathy phenotype.
  Journal
Clin Genet 86:220-8 (2014)
DOI:10.1111/cge.12301
Reference
PMID:26595381 (TMEM107)
  Authors
Lambacher NJ, Bruel AL, van Dam TJ, Szymanska K, Slaats GG, Kuhns S, McManus GJ, Kennedy JE, Gaff K, Wu KM, van der Lee R, Burglen L, Doummar D, Riviere JB, Faivre L, Attie-Bitach T, Saunier S, Curd A, Peckham M, Giles RH, Johnson CA, Huynen MA, Thauvin-Robinet C, Blacque OE
  Title
TMEM107 recruits ciliopathy proteins to subdomains of the ciliary transition zone and causes Joubert syndrome.
  Journal
Nat Cell Biol 18:122-31 (2016)
DOI:10.1038/ncb3273
Reference
PMID:30851085 (TXNDC15)
  Authors
Ridnoi K, Sois M, Vaidla E, Pajusalu S, Kelder L, Reimand T, Ounap K
  Title
A prenatally diagnosed case of Meckel-Gruber syndrome with novel compound heterozygous pathogenic variants in the TXNDC15 gene.
  Journal
Mol Genet Genomic Med 7:e614 (2019)
DOI:10.1002/mgg3.614
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