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KEGG   DISEASE: Alternative complement pathway component defects
Entry
H00104                      Disease                                
Name
Alternative complement pathway component defects
  Subgroup
Factor B deficiency
Factor D deficiency
Factor H deficiency
Properdin deficiency
  Supergrp
Disorders of innate immunity [DS:H02525]
Primary immunodeficiency disease [DS:H01725]
Description
The alternative pathway (AP) is antibody independent and relies on native C3 undergoing minimal spontaneous hydrolysis. Hydrolyzed C3 binds factor B. Factor B, when bound to hydrolyzed C3, is cleaved by factor D into Ba and Bb. Hydrolyzed C3Bb is responsible for a constant low level of C3 cleavage into C3b. If C3b binds to an appropriate surface, factor B will bind with C3b to form C3bBb, a highly efficient C3-cleaving enzyme. This overall series of successive proteolytic steps is enhanced by the serum protein properdin, which stabilizes protein:protein interactions during the process. Factor H is essential in controlling the function of the alternative pathway by inhibiting the formation of and degrading C3bBb. Deficiencies of alternative pathway-specific components are rare, and usually lead to an increased frequency of Neisseria infections.
Category
Primary immunodeficiency
Brite
Human diseases in ICD-11 classification [BR:br08403]
 04 Diseases of the immune system
  Primary immunodeficiencies
   4A00  Primary immunodeficiencies due to disorders of innate immunity
    H00104  Alternative complement pathway component defects
Pathway-based classification of diseases [BR:br08402]
 Immune system
  nt06513  Complement cascade
   H00104  Alternative complement pathway component defects
Pathway
hsa04610  Complement and coagulation cascades
Network
nt06513 Complement cascade
Gene
(CFBD) CFB [HSA:629] [KO:K01335]
(CFDD) CFD [HSA:1675] [KO:K01334]
(CFHD) CFH [HSA:3075] [KO:K04004]
(CFPD) CFP [HSA:5199] [KO:K15412]
Other DBs
ICD-11: 4A00.1
OMIM: 615561 613912 609814 312060
Reference
  Authors
Pettigrew HD, Teuber SS, Gershwin ME
  Title
Clinical significance of complement deficiencies.
  Journal
Ann N Y Acad Sci 1173:108-23 (2009)
DOI:10.1111/j.1749-6632.2009.04633.x
Reference
  Authors
Kumar A, Teuber SS, Gershwin ME.
  Title
Current perspectives on primary immunodeficiency diseases.
  Journal
Clin Dev Immunol 13:223-59 (2006)
DOI:10.1080/17402520600800705
Reference
  Authors
Geha RS, Notarangelo LD, Casanova JL, Chapel H, Conley ME, Fischer A, Hammarstrom L, Nonoyama S, Ochs HD, Puck JM, Roifman C, Seger R, Wedgwood J.
  Title
Primary immunodeficiency diseases: an update from the International Union of Immunological Societies Primary Immunodeficiency Diseases Classification Committee.
  Journal
J Allergy Clin Immunol 120:776-94 (2007)
DOI:10.1016/j.jaci.2007.08.053
Reference
PMID:24152280 (CFBD)
  Authors
Slade C, Bosco J, Unglik G, Bleasel K, Nagel M, Winship I
  Title
Deficiency in complement factor B.
  Journal
N Engl J Med 369:1667-9 (2013)
DOI:10.1056/NEJMc1306326
Reference
PMID:11457876 (CFDD)
  Authors
Biesma DH, Hannema AJ, van Velzen-Blad H, Mulder L, van Zwieten R, Kluijt I, Roos D
  Title
A family with complement factor D deficiency.
  Journal
J Clin Invest 108:233-40 (2001)
DOI:10.1172/JCI12023
Reference
PMID:9312129 (CFHD)
  Authors
Ault BH, Schmidt BZ, Fowler NL, Kashtan CE, Ahmed AE, Vogt BA, Colten HR
  Title
Human factor H deficiency. Mutations in framework cysteine residues and block in H protein secretion and intracellular catabolism.
  Journal
J Biol Chem 272:25168-75 (1997)
DOI:10.1074/jbc.272.40.25168
Reference
PMID:8530058 (CFPD)
  Authors
Westberg J, Fredrikson GN, Truedsson L, Sjoholm AG, Uhlen M
  Title
Sequence-based analysis of properdin deficiency: identification of point mutations in two phenotypic forms of an X-linked immunodeficiency.
  Journal
Genomics 29:1-8 (1995)
DOI:10.1006/geno.1995.1208
LinkDB

» Japanese version

KEGG   DISEASE: Age-related macular degeneration
Entry
H00821                      Disease                                
Name
Age-related macular degeneration
Description
Macular degeneration is the physical breakdown of the central portion of the retina called the macula. Age-related macular degeneration (AMD/ARMD) is the leading cause of blindness. AMD is a complex disease caused by the combination of genetic predisposition and environmental factors. Using genome linkage scan and association studies, multiple potentially causative genes have been identified. In AMD, there are two phenotypes, atrophic/ dry and neovascular/ wet. The former is characterized by the geographic atrophy due to death of retinal pigment epithelium, and the latter is usually characterized by the abnormal growth of new blood vessels under the macula, which causes severe loss of vision. While wet AMD can be treated by the inhibition of vascular endothelial growth factor or photodynamic therapy, so far there are no available treatments for dry AMD.
Category
Nervous system disease
Brite
Human diseases in ICD-11 classification [BR:br08403]
 09 Diseases of the visual system
  Disorders of the eyeball posterior segment
   Disorders of the retina
    9B75  Macular disorders
     H00821  Age-related macular degeneration
Pathway-based classification of diseases [BR:br08402]
 Cellular process
  nt06535  Efferocytosis
   H00821  Age-related macular degeneration
  nt06527  Necroptosis
   H00821  Age-related macular degeneration
 Immune system
  nt06513  Complement cascade
   H00821  Age-related macular degeneration
  nt06517  TLR signaling
   H00821  Age-related macular degeneration
Pathway
hsa04610  Complement and coagulation cascades
hsa04620  Toll-like receptor signaling pathway
hsa04217  Necroptosis
Network
nt06513 Complement cascade
nt06517 TLR signaling
nt06527 Necroptosis
nt06535 Efferocytosis
Gene
(ARMD1) HMCN1 [HSA:83872] [KO:K17341]
(ARMD1) CFHR1 [HSA:3078] [KO:K23815]
(ARMD1) CFHR3 [HSA:10878] [KO:K23815]
(ARMD2) ABCA4 [HSA:24] [KO:K05644]
(ARMD3) FBLN5 [HSA:10516] [KO:K17340]
(ARMD4) CFH [HSA:3075] [KO:K04004]
(ARMD5) ERCC6 [HSA:2074] [KO:K10841]
(ARMD6) RAX2 [HSA:84839] [KO:K09333]
(ARMD7) HTRA1 [HSA:5654] [KO:K08784]
(ARMD8) ARMS2 [HSA:387715] [KO:K25179]
(ARMD9) C3 [HSA:718] [KO:K03990]
(ARMD10) TLR4 [HSA:7099] [KO:K10160]
(ARMD11) CST3 [HSA:1471] [KO:K13899]
(ARMD12) CX3CR1 [HSA:1524] [KO:K04192]
(ARMD13) CFI [HSA:3426] [KO:K01333]
(ARMD14) C2 [HSA:717] [KO:K01332]
(ARMD14) CFB [HSA:629] [KO:K01335]
(ARMD15) C9 [HSA:735] [KO:K04000]
Drug
Aflibercept [DR:D09574]
Verteporfin [DR:D01162]
Ranibizumab [DR:D05697]
Brolucizumab [DR:D11083]
Faricimab [DR:D11516]
Avacincaptad pegol sodium [DR:D11748]
Comment
A haplotype carrying deletion of the complement factor H-related genes CFHR1 and CFHR3 is also associated with reduced risk of ARMD.
Other DBs
ICD-11: 9B75.0
ICD-10: H35.3
MeSH: D008268
OMIM: 603075 153800 608895 610698 613761 613757 610149 613778 611378 611488 611953 613784 615439 615489 615591
Reference
  Authors
Montezuma SR, Sobrin L, Seddon JM
  Title
Review of genetics in age related macular degeneration.
  Journal
Semin Ophthalmol 22:229-40 (2007)
DOI:10.1080/08820530701745140
Reference
  Authors
Virgili G, Bini A
  Title
Laser photocoagulation for neovascular age-related macular degeneration.
  Journal
Cochrane Database Syst Rev CD004763 (2007)
DOI:10.1002/14651858.CD004763.pub2
Reference
PMID:14570714 (HMCN1)
  Authors
Schultz DW, Klein ML, Humpert AJ, Luzier CW, Persun V, Schain M, Mahan A, Runckel C, Cassera M, Vittal V, Doyle TM, Martin TM, Weleber RG, Francis PJ, Acott TS
  Title
Analysis of the ARMD1 locus: evidence that a mutation in HEMICENTIN-1 is associated with age-related macular degeneration in a large family.
  Journal
Hum Mol Genet 12:3315-23 (2003)
DOI:10.1093/hmg/ddg348
Reference
PMID:16998489 (CFHR1 CFHR3, reduced risk)
  Authors
Hughes AE, Orr N, Esfandiary H, Diaz-Torres M, Goodship T, Chakravarthy U
  Title
A common CFH haplotype, with deletion of CFHR1 and CFHR3, is associated with lower risk of age-related macular degeneration.
  Journal
Nat Genet 38:1173-7 (2006)
DOI:10.1038/ng1890
Reference
PMID:9295268 (ABCA4)
  Authors
Allikmets R, Shroyer NF, Singh N, Seddon JM, Lewis RA, Bernstein PS, Peiffer A, Zabriskie NA, Li Y, Hutchinson A, Dean M, Lupski JR, Leppert M
  Title
Mutation of the Stargardt disease gene (ABCR) in age-related macular degeneration.
  Journal
Science 277:1805-7 (1997)
DOI:10.1126/science.277.5333.1805
Reference
PMID:15269314 (FBLN5)
  Authors
Stone EM, Braun TA, Russell SR, Kuehn MH, Lotery AJ, Moore PA, Eastman CG, Casavant TL, Sheffield VC
  Title
Missense variations in the fibulin 5 gene and age-related macular degeneration.
  Journal
N Engl J Med 351:346-53 (2004)
DOI:10.1056/NEJMoa040833
Reference
PMID:15761122 (CFH)
  Authors
Klein RJ, Zeiss C, Chew EY, Tsai JY, Sackler RS, Haynes C, Henning AK, SanGiovanni JP, Mane SM, Mayne ST, Bracken MB, Ferris FL, Ott J, Barnstable C, Hoh J
  Title
Complement factor H polymorphism in age-related macular degeneration.
  Journal
Science 308:385-9 (2005)
DOI:10.1126/science.1109557
Reference
PMID:16754848 (ERCC6)
  Authors
Tuo J, Ning B, Bojanowski CM, Lin ZN, Ross RJ, Reed GF, Shen D, Jiao X, Zhou M, Chew EY, Kadlubar FF, Chan CC
  Title
Synergic effect of polymorphisms in ERCC6 5' flanking region and complement factor H on age-related macular degeneration predisposition.
  Journal
Proc Natl Acad Sci U S A 103:9256-61 (2006)
DOI:10.1073/pnas.0603485103
Reference
PMID:15028672 (RAX2)
  Authors
Wang QL, Chen S, Esumi N, Swain PK, Haines HS, Peng G, Melia BM, McIntosh I, Heckenlively JR, Jacobson SG, Stone EM, Swaroop A, Zack DJ
  Title
QRX, a novel homeobox gene, modulates photoreceptor gene expression.
  Journal
Hum Mol Genet 13:1025-40 (2004)
DOI:10.1093/hmg/ddh117
Reference
PMID:17053108 (HTRA1)
  Authors
Dewan A, Liu M, Hartman S, Zhang SS, Liu DT, Zhao C, Tam PO, Chan WM, Lam DS, Snyder M, Barnstable C, Pang CP, Hoh J
  Title
HTRA1 promoter polymorphism in wet age-related macular degeneration.
  Journal
Science 314:989-92 (2006)
DOI:10.1126/science.1133807
Reference
PMID:16174643 (ARMS2)
  Authors
Rivera A, Fisher SA, Fritsche LG, Keilhauer CN, Lichtner P, Meitinger T, Weber BH
  Title
Hypothetical LOC387715 is a second major susceptibility gene for age-related macular degeneration, contributing independently of complement factor H to disease risk.
  Journal
Hum Mol Genet 14:3227-36 (2005)
DOI:10.1093/hmg/ddi353
Reference
PMID:17634448 (C3)
  Authors
Yates JR, Sepp T, Matharu BK, Khan JC, Thurlby DA, Shahid H, Clayton DG, Hayward C, Morgan J, Wright AF, Armbrecht AM, Dhillon B, Deary IJ, Redmond E, Bird AC, Moore AT
  Title
Complement C3 variant and the risk of age-related macular degeneration.
  Journal
N Engl J Med 357:553-61 (2007)
DOI:10.1056/NEJMoa072618
Reference
PMID:15829498 (TLR4)
  Authors
Zareparsi S, Buraczynska M, Branham KE, Shah S, Eng D, Li M, Pawar H, Yashar BM, Moroi SE, Lichter PR, Petty HR, Richards JE, Abecasis GR, Elner VM, Swaroop A
  Title
Toll-like receptor 4 variant D299G is associated with susceptibility to age-related macular degeneration.
  Journal
Hum Mol Genet 14:1449-55 (2005)
DOI:10.1093/hmg/ddi154
Reference
PMID:11815350 (CST3)
  Authors
Zurdel J, Finckh U, Menzer G, Nitsch RM, Richard G
  Title
CST3 genotype associated with exudative age related macular degeneration.
  Journal
Br J Ophthalmol 86:214-9 (2002)
DOI:10.1136/bjo.86.2.214
Reference
PMID:15208270 (CX3CR1)
  Authors
Tuo J, Smith BC, Bojanowski CM, Meleth AD, Gery I, Csaky KG, Chew EY, Chan CC
  Title
The involvement of sequence variation and expression of CX3CR1 in the pathogenesis of age-related macular degeneration.
  Journal
FASEB J 18:1297-9 (2004)
DOI:10.1096/fj.04-1862fje
Reference
PMID:23685748 (CFI)
  Authors
van de Ven JP, Nilsson SC, Tan PL, Buitendijk GH, Ristau T, Mohlin FC, Nabuurs SB, Schoenmaker-Koller FE, Smailhodzic D, Campochiaro PA, Zack DJ, Duvvari MR, Bakker B, Paun CC, Boon CJ, Uitterlinden AG, Liakopoulos S, Klevering BJ, Fauser S, Daha MR, Katsanis N, Klaver CC, Blom AM, Hoyng CB, den Hollander AI
  Title
A functional variant in the CFI gene confers a high risk of age-related macular degeneration.
  Journal
Nat Genet 45:813-7 (2013)
DOI:10.1038/ng.2640
Reference
PMID:16518403 (C2 CFB)
  Authors
Gold B, Merriam JE, Zernant J, Hancox LS, Taiber AJ, Gehrs K, Cramer K, Neel J, Bergeron J, Barile GR, Smith RT, Hageman GS, Dean M, Allikmets R
  Title
Variation in factor B (BF) and complement component 2 (C2) genes is associated with age-related macular degeneration.
  Journal
Nat Genet 38:458-62 (2006)
DOI:10.1038/ng1750
Reference
PMID:24036952 (C9)
  Authors
Seddon JM, Yu Y, Miller EC, Reynolds R, Tan PL, Gowrisankar S, Goldstein JI, Triebwasser M, Anderson HE, Zerbib J, Kavanagh D, Souied E, Katsanis N, Daly MJ, Atkinson JP, Raychaudhuri S
  Title
Rare variants in CFI, C3 and C9 are associated with high risk of advanced age-related macular degeneration.
  Journal
Nat Genet 45:1366-70 (2013)
DOI:10.1038/ng.2741
LinkDB

» Japanese version

KEGG   DISEASE: Atypical hemolytic uremic syndrome
Entry
H01434                      Disease                                
Name
Atypical hemolytic uremic syndrome
Description
The haemolytic uraemic syndrome (HUS) is characterized by the triad of thrombocytopenia, microangiopathic haemolytic anaemia and acute renal failure. HUS may be classified as either diarrhoeal-associated (D+HUS) or non-diarrhoeal/atypical (aHUS). Approximately half of the patients with aHUS have mutations in genes that regulate the complement system. Several other conditions and factors, such as infection, drugs, pregnancy, and malignancy, have been reported to cause aHUS.
Category
Hematologic disease
Brite
Human diseases in ICD-11 classification [BR:br08403]
 03 Diseases of the blood or blood-forming organs
  Anaemias or other erythrocyte disorders
   Haemolytic anaemias
    Congenital haemolytic anaemia
     3A10  Hereditary haemolytic anaemia
      H01434  Atypical hemolytic uremic syndrome
Pathway-based classification of diseases [BR:br08402]
 Immune system
  nt06513  Complement cascade
   H01434  Atypical hemolytic uremic syndrome
Pathway
hsa04610  Complement and coagulation cascades
Network
nt06513 Complement cascade
Gene
(AHUS1) CFH [HSA:3075] [KO:K04004]
(AHUS1) CFHR3 [HSA:10878] [KO:K23815]
(AHUS1) CFHR1 [HSA:3078] [KO:K23815]
(AHUS2) CD46, MCP [HSA:4179] [KO:K04007]
(AHUS3) CFI, IF [HSA:3426] [KO:K01333]
(AHUS4) CFB [HSA:629] [KO:K01335]
(AHUS5) C3 [HSA:718] [KO:K03990]
(AHUS6) THBD [HSA:7056] [KO:K03907]
Drug
Eculizumab [DR:D03940]
Ravulizumab [DR:D11054]
Other DBs
ICD-11: 3A10.Y
ICD-10: D58.8
MeSH: D065766
OMIM: 235400 612922 612923 612924 612925 612926
Reference
  Authors
Kavanagh D, Goodship TH, Richards A
  Title
Atypical haemolytic uraemic syndrome.
  Journal
Br Med Bull 77-78:5-22 (2006)
DOI:10.1093/bmb/ldl004
Reference
PMID:9551389 (CFH)
  Authors
Warwicker P, Goodship TH, Donne RL, Pirson Y, Nicholls A, Ward RM, Turnpenny P, Goodship JA
  Title
Genetic studies into inherited and sporadic hemolytic uremic syndrome.
  Journal
Kidney Int 53:836-44 (1998)
DOI:10.1111/j.1523-1755.1998.00824.x
Reference
PMID:17367211 (CFHR3 CFHR1)
  Authors
Zipfel PF, Edey M, Heinen S, Jozsi M, Richter H, Misselwitz J, Hoppe B, Routledge D, Strain L, Hughes AE, Goodship JA, Licht C, Goodship TH, Skerka C
  Title
Deletion of complement factor H-related genes CFHR1 and CFHR3 is associated with atypical hemolytic uremic syndrome.
  Journal
PLoS Genet 3:e41 (2007)
DOI:10.1371/journal.pgen.0030041
Reference
PMID:14615110 (CD46)
  Authors
Noris M, Brioschi S, Caprioli J, Todeschini M, Bresin E, Porrati F, Gamba S, Remuzzi G
  Title
Familial haemolytic uraemic syndrome and an MCP mutation.
  Journal
Lancet 362:1542-7 (2003)
DOI:10.1016/S0140-6736(03)14742-3
Reference
PMID:15173250 (CFI)
  Authors
Fremeaux-Bacchi V, Dragon-Durey MA, Blouin J, Vigneau C, Kuypers D, Boudailliez B, Loirat C, Rondeau E, Fridman WH
  Title
Complement factor I: a susceptibility gene for atypical haemolytic uraemic syndrome.
  Journal
J Med Genet 41:e84 (2004)
DOI:10.1136/jmg.2004.019083
Reference
PMID:17182750 (CFB)
  Authors
Goicoechea de Jorge E, Harris CL, Esparza-Gordillo J, Carreras L, Arranz EA, Garrido CA, Lopez-Trascasa M, Sanchez-Corral P, Morgan BP, Rodriguez de Cordoba S
  Title
Gain-of-function mutations in complement factor B are associated with atypical hemolytic uremic syndrome.
  Journal
Proc Natl Acad Sci U S A 104:240-5 (2007)
DOI:10.1073/pnas.0603420103
Reference
PMID:18796626 (C3)
  Authors
Fremeaux-Bacchi V, Miller EC, Liszewski MK, Strain L, Blouin J, Brown AL, Moghal N, Kaplan BS, Weiss RA, Lhotta K, Kapur G, Mattoo T, Nivet H, Wong W, Gie S, Hurault de Ligny B, Fischbach M, Gupta R, Hauhart R, Meunier V, Loirat C, Dragon-Durey MA, Fridman WH, Janssen BJ, Goodship TH, Atkinson JP
  Title
Mutations in complement C3 predispose to development of atypical hemolytic uremic syndrome.
  Journal
Blood 112:4948-52 (2008)
DOI:10.1182/blood-2008-01-133702
Reference
PMID:19625716 (THBD)
  Authors
Delvaeye M, Noris M, De Vriese A, Esmon CT, Esmon NL, Ferrell G, Del-Favero J, Plaisance S, Claes B, Lambrechts D, Zoja C, Remuzzi G, Conway EM
  Title
Thrombomodulin mutations in atypical hemolytic-uremic syndrome.
  Journal
N Engl J Med 361:345-57 (2009)
DOI:10.1056/NEJMoa0810739
LinkDB

» Japanese version

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