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Familial cold autoinflammatory syndrome(FCAS)

MedGen UID:
137986
Concept ID:
C0343068
Disease or Syndrome
Synonyms: COLD-INDUCED AUTOINFLAMMATORY SYNDROME, FAMILIAL; Familial cold urticaria; Familial polymorphous cold eruption; FCAS
SNOMED CT: Familial cold urticaria (238687000); Familial cold autoinflammatory syndrome (238687000); FCU - familial cold urticaria (238687000); FCAS - familial cold autoinflammatory syndrome (238687000)
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.
 
Related genes: NLRP3, NLRP12, NLRC4, PLCG2
 
Monarch Initiative: MONDO:0018768
OMIM®: 120100
OMIM® Phenotypic series: PS120100
Orphanet: ORPHA47045

Definition

Familial cold autoinflammatory syndrome is characterized clinically by recurrent attacks of a maculopapular rash associated with arthralgias, myalgias, fever and chills, and swelling of the extremities after exposure to cold. Despite the first description of 'cold urticaria' (Kile and Rusk, 1940) the rash in most patients is nonpruritic and nonurticarial. Rarely, some patients may also develop late-onset renal amyloidosis (Hoffman et al., 2000). Overlapping syndromes also caused by mutation in the NLRP3 gene include Muckle-Wells syndrome (CAPS2; 191900), which has a high frequency of amyloidosis and late-onset sensorineural deafness, and chronic neurologic cutaneous and articular syndrome (CINCA, CAPS3; 607115), which shows earlier onset and a more severe phenotype. Genetic Heterogeneity of Familial Cold Autoinflammatory Syndrome See also FCAS2 (611762), caused by mutation in the NLRP12 gene (609648) on chromosome 19q13; FCAS3 (614468), caused by mutation in the PLCG2 gene (600220) on chromosome 16q23; and FCAS4 (616115), caused by mutation in the NLRC4 gene (606831) on chromosome 2p22. [from OMIM]

Professional guidelines

PubMed

Diaz VL, Gribbons KB, Yazdi-Nejad K, Kuemmerle-Deschner J, Wanderer AA, Broderick L, Hoffman HM
J Allergy Clin Immunol Pract 2023 Aug;11(8):2275-2285. Epub 2023 Jun 7 doi: 10.1016/j.jaip.2023.05.040. PMID: 37290539
Koné-Paut I, Galeotti C
Expert Rev Clin Immunol 2015;11(10):1083-92. Epub 2015 Aug 27 doi: 10.1586/1744666X.2015.1077702. PMID: 26312542
Miyamae T
Paediatr Drugs 2012 Apr 1;14(2):109-17. doi: 10.2165/11595040-000000000-00000. PMID: 22335455

Recent clinical studies

Etiology

Wang HF
Pediatr Rheumatol Online J 2022 Feb 5;20(1):9. doi: 10.1186/s12969-022-00669-8. PMID: 35123508Free PMC Article
Williams R, Hawkins P, Lane T
Br J Nurs 2019 Oct 10;28(18):1180-1186. doi: 10.12968/bjon.2019.28.18.1180. PMID: 31597069
Marzano AV, Tavecchio S, Venturini M, Sala R, Calzavara-Pinton P, Gattorno M
G Ital Dermatol Venereol 2015 Feb;150(1):41-50. Epub 2015 Jan 14 PMID: 25586657
Miyamae T
Paediatr Drugs 2012 Apr 1;14(2):109-17. doi: 10.2165/11595040-000000000-00000. PMID: 22335455
Neven B, Prieur AM, Quartier dit Maire P
Nat Clin Pract Rheumatol 2008 Sep;4(9):481-9. Epub 2008 Jul 29 doi: 10.1038/ncprheum0874. PMID: 18665151

Diagnosis

Diaz VL, Gribbons KB, Yazdi-Nejad K, Kuemmerle-Deschner J, Wanderer AA, Broderick L, Hoffman HM
J Allergy Clin Immunol Pract 2023 Aug;11(8):2275-2285. Epub 2023 Jun 7 doi: 10.1016/j.jaip.2023.05.040. PMID: 37290539
Wang HF
Pediatr Rheumatol Online J 2022 Feb 5;20(1):9. doi: 10.1186/s12969-022-00669-8. PMID: 35123508Free PMC Article
Maltseva N, Borzova E, Fomina D, Bizjak M, Terhorst-Molawi D, Košnik M, Kulthanan K, Meshkova R, Thomsen SF, Maurer M; COLD-CE Steering Committee
Allergy 2021 Apr;76(4):1077-1094. Epub 2020 Dec 24 doi: 10.1111/all.14674. PMID: 33249577
Davis MDP, van der Hilst JCH
J Allergy Clin Immunol Pract 2018 Jul-Aug;6(4):1162-1170. Epub 2018 Jun 2 doi: 10.1016/j.jaip.2018.05.006. PMID: 29871797
Manners PJ, Guttinger R
Indian J Pediatr 2010 Oct;77(10):1173-81. Epub 2010 Oct 16 doi: 10.1007/s12098-010-0206-y. PMID: 20953850

Therapy

Demir F, Sözeri B
Clin Exp Rheumatol 2023 Oct;41(10):2115-2121. Epub 2023 Oct 9 doi: 10.55563/clinexprheumatol/kzggp3. PMID: 37877365
Maltseva N, Borzova E, Fomina D, Bizjak M, Terhorst-Molawi D, Košnik M, Kulthanan K, Meshkova R, Thomsen SF, Maurer M; COLD-CE Steering Committee
Allergy 2021 Apr;76(4):1077-1094. Epub 2020 Dec 24 doi: 10.1111/all.14674. PMID: 33249577
Koné-Paut I, Galeotti C
Expert Rev Clin Immunol 2015;11(10):1083-92. Epub 2015 Aug 27 doi: 10.1586/1744666X.2015.1077702. PMID: 26312542
Miyamae T
Paediatr Drugs 2012 Apr 1;14(2):109-17. doi: 10.2165/11595040-000000000-00000. PMID: 22335455
Shinkai K, McCalmont TH, Leslie KS
Clin Exp Dermatol 2008 Jan;33(1):1-9. Epub 2007 Oct 9 doi: 10.1111/j.1365-2230.2007.02540.x. PMID: 17927785

Prognosis

Kuemmerle-Deschner JB
Semin Immunopathol 2015 Jul;37(4):377-85. Epub 2015 May 12 doi: 10.1007/s00281-015-0491-7. PMID: 25963520
Miyamae T
Paediatr Drugs 2012 Apr 1;14(2):109-17. doi: 10.2165/11595040-000000000-00000. PMID: 22335455
Hedrich CM, Bruck N, Paul D, Hahn G, Gahr M, Rösen-Wolff A
Rheumatol Int 2012 Sep;32(9):2629-36. Epub 2011 Jul 22 doi: 10.1007/s00296-011-2019-3. PMID: 21833523
Jesus AA, Oliveira JB, Hilário MO, Terreri MT, Fujihira E, Watase M, Carneiro-Sampaio M, Silva CA
J Pediatr (Rio J) 2010 Sep-Oct;86(5):353-66. doi: 10.2223/JPED.2015. PMID: 20938587
Maksimovic L, Stirnemann J, Caux F, Ravet N, Rouaghe S, Cuisset L, Letellier E, Grateau G, Morin AS, Fain O
Rheumatology (Oxford) 2008 Mar;47(3):309-10. Epub 2008 Jan 3 doi: 10.1093/rheumatology/kem318. PMID: 18174231

Clinical prediction guides

Samson JM, Ravindran Menon D, Vaddi PK, Kalani Williams N, Domenico J, Zhai Z, Backos DS, Fujita M
Front Immunol 2020;11:584364. Epub 2020 Nov 19 doi: 10.3389/fimmu.2020.584364. PMID: 33329557Free PMC Article
Landmann EC, Walker UA
Expert Rev Clin Pharmacol 2017 Aug;10(8):855-864. Epub 2017 Jun 20 doi: 10.1080/17512433.2017.1338946. PMID: 28586272
Yadlapati S, Efthimiou P
Mod Rheumatol 2016;26(1):3-8. Epub 2015 Aug 3 doi: 10.3109/14397595.2015.1069459. PMID: 26140469
Rosengren S, Mueller JL, Anderson JP, Niehaus BL, Misaghi A, Anderson S, Boyle DL, Hoffman HM
J Allergy Clin Immunol 2007 Apr;119(4):991-6. Epub 2007 Feb 22 doi: 10.1016/j.jaci.2006.12.649. PMID: 17320940Free PMC Article
Tunca M, Ozdogan H
Curr Drug Targets Inflamm Allergy 2005 Feb;4(1):77-80. doi: 10.2174/1568010053622957. PMID: 15720239

Recent systematic reviews

Rodrigues F, Cuisset L, Cador-Rousseau B, Giurgea I, Neven B, Buob D, Quartier P, Hachulla E, Lequerré T, Cam G, Boursier G, Hervieu V, Grateau G, Georgin-Lavialle S
Rheumatology (Oxford) 2022 Nov 28;61(12):4827-4834. doi: 10.1093/rheumatology/keac145. PMID: 35262642

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