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Hereditary angioedema with C1Inh deficiency

MedGen UID:
1812520
Concept ID:
C4552294
Disease or Syndrome
Synonyms: angioedema, hereditary, 1 and 2; angioedema, hereditary, type 1; angioedema, hereditary, type 1/2; angioedema, hereditary, type 2; angioedema, hereditary, type I; angioneurotic edema, hereditary; C1 esterase inhibitor, deficiency of; HAE (hereditary angioedema) with C1Inh (C1 esterase inhibitor) deficiency; HAE with C1 inhibitor deficiency; HAE with C1Inh deficiency; HAE1; Hereditary angioedema with C1 esterase inhibitor deficiency; Hereditary angioedema with C1Inh (C1 esterase inhibitor) deficiency; hereditary angioedema with C1Inh deficiency; Hereditary angioneurotic edema with C1 inhibitor deficiency; Hereditary angioneurotic edema with C1Inh deficiency
SNOMED CT: Hereditary angioedema with C1Inh (C1 esterase inhibitor) deficiency (1230015008); Hereditary angioedema with C1 esterase inhibitor deficiency (1230015008); HAE (hereditary angioedema) with C1Inh (C1 esterase inhibitor) deficiency (1230015008); Hereditary angioneurotic edema with C1 inhibitor deficiency (1230015008)
Modes of inheritance:
Not genetically inherited
MedGen UID:
988794
Concept ID:
CN307044
Finding
Source: Orphanet
clinical entity without genetic inheritance.
 
Monarch Initiative: MONDO:0033946
Orphanet: ORPHA528623

Definition

A rare hereditary angio-oedema characterised by potentially life-threatening episodes of subcutaneous and/or submucosal oedema without urticaria, associated with C1 esterase inhibitor (C1-INH) deficiency. Hereditary angio-oedema (HAE) type 1 is caused by quantitative, HAE type 2 by qualitative defects of C1-INH. The two subtypes are clinically indistinguishable. Patients may present at any age (but most commonly in childhood) with recurrent attacks of nonpitting oedema of the skin, severe abdominal symptoms such as pain and swelling, and/or respiratory distress due to upper respiratory airways involvement. Genital, bladder, muscle or joint swelling may occur in some cases. [from SNOMEDCT_US]

Term Hierarchy

Professional guidelines

PubMed

Longhurst HJ, Bork K
Br J Hosp Med (Lond) 2019 Jul 2;80(7):391-398. doi: 10.12968/hmed.2019.80.7.391. PMID: 31283393
Perego F, Wu MA, Valerieva A, Caccia S, Suffritti C, Zanichelli A, Bergamaschini L, Cicardi M
Expert Opin Biol Ther 2019 Jun;19(6):517-526. Epub 2019 Mar 26 doi: 10.1080/14712598.2019.1595581. PMID: 30912460
Cicardi M, Suffritti C, Perego F, Caccia S
J Investig Allergol Clin Immunol 2016;26(4):212-21; quiz two pages after page 221. doi: 10.18176/jiaci.0087. PMID: 27470642

Recent clinical studies

Etiology

Christiansen SC, Wilmot J, Castaldo AJ, Zuraw BL
Ann Allergy Asthma Immunol 2023 Dec;131(6):766-774.e8. Epub 2023 Aug 23 doi: 10.1016/j.anai.2023.08.012. PMID: 37619776
Veronez CL, Christiansen SC, Smith TD, Riedl MA, Zuraw BL
Allergy Asthma Proc 2021 Nov 1;42(6):506-514. doi: 10.2500/aap.2021.42.210083. PMID: 34871158Free PMC Article
Billebeau A, Fain O, Launay D, Boccon-Gibod I, Bouillet L, Gobert D, Plu-Bureau G, Gompel A; French National Reference Center for Hereditary Angioedema (CREAK)
J Clin Immunol 2021 Jan;41(1):163-170. Epub 2020 Oct 31 doi: 10.1007/s10875-020-00902-7. PMID: 33130967
Proper SP, Lavery WJ, Bernstein JA
Allergy Asthma Proc 2020 Nov 1;41(Suppl 1):S03-S07. doi: 10.2500/aap.2020.41.200040. PMID: 33109317
Bygum A
Dermatology 2019;235(4):263-275. Epub 2019 Jun 5 doi: 10.1159/000500196. PMID: 31167185

Diagnosis

Sinnathamby ES, Issa PP, Roberts L, Norwood H, Malone K, Vemulapalli H, Ahmadzadeh S, Cornett EM, Shekoohi S, Kaye AD
Adv Ther 2023 Mar;40(3):814-827. Epub 2023 Jan 7 doi: 10.1007/s12325-022-02401-0. PMID: 36609679Free PMC Article
Billebeau A, Fain O, Launay D, Boccon-Gibod I, Bouillet L, Gobert D, Plu-Bureau G, Gompel A; French National Reference Center for Hereditary Angioedema (CREAK)
J Clin Immunol 2021 Jan;41(1):163-170. Epub 2020 Oct 31 doi: 10.1007/s10875-020-00902-7. PMID: 33130967
Proper SP, Lavery WJ, Bernstein JA
Allergy Asthma Proc 2020 Nov 1;41(Suppl 1):S03-S07. doi: 10.2500/aap.2020.41.200040. PMID: 33109317
Patel G, Pongracic JA
Allergy Asthma Proc 2019 Nov 1;40(6):441-445. doi: 10.2500/aap.2019.40.4267. PMID: 31690390
Bygum A
Dermatology 2019;235(4):263-275. Epub 2019 Jun 5 doi: 10.1159/000500196. PMID: 31167185

Therapy

Longhurst HJ, Bork K
Br J Hosp Med (Lond) 2019 Jul 2;80(7):391-398. doi: 10.12968/hmed.2019.80.7.391. PMID: 31283393
Perego F, Wu MA, Valerieva A, Caccia S, Suffritti C, Zanichelli A, Bergamaschini L, Cicardi M
Expert Opin Biol Ther 2019 Jun;19(6):517-526. Epub 2019 Mar 26 doi: 10.1080/14712598.2019.1595581. PMID: 30912460
Magerl M, Germenis AE, Maas C, Maurer M
Immunol Allergy Clin North Am 2017 Aug;37(3):571-584. doi: 10.1016/j.iac.2017.04.004. PMID: 28687110
Obtułowicz K
Pol Arch Med Wewn 2016;126(1-2):76-85. doi: 10.20452/pamw.3273. PMID: 26842379
Farnam J, Grant JA
Dermatol Clin 1985 Jan;3(1):85-95. PMID: 2868818

Prognosis

Riedl MA
Immunol Allergy Clin North Am 2023 Feb;43(1):145-157. Epub 2022 Oct 28 doi: 10.1016/j.iac.2022.05.011. PMID: 36411000
Veronez CL, Christiansen SC, Smith TD, Riedl MA, Zuraw BL
Allergy Asthma Proc 2021 Nov 1;42(6):506-514. doi: 10.2500/aap.2021.42.210083. PMID: 34871158Free PMC Article
Zanichelli A, Farkas H, Bouillet L, Bara N, Germenis AE, Psarros F, Varga L, Andrási N, Boccon-Gibod I, Castiglioni Roffia M, Rutkowski M, Cancian M
Clin Rev Allergy Immunol 2021 Aug;61(1):77-83. Epub 2021 Mar 31 doi: 10.1007/s12016-021-08855-4. PMID: 33791951Free PMC Article
Billebeau A, Fain O, Launay D, Boccon-Gibod I, Bouillet L, Gobert D, Plu-Bureau G, Gompel A; French National Reference Center for Hereditary Angioedema (CREAK)
J Clin Immunol 2021 Jan;41(1):163-170. Epub 2020 Oct 31 doi: 10.1007/s10875-020-00902-7. PMID: 33130967
Csuka D, Veszeli N, Varga L, Prohászka Z, Farkas H
Mol Immunol 2017 Sep;89:59-68. Epub 2017 Jun 7 doi: 10.1016/j.molimm.2017.05.020. PMID: 28595743

Clinical prediction guides

Gramstad OR, Schjalm C, Mollnes TE, Nielsen EW
Clin Exp Immunol 2023 Dec 12;214(2):170-181. doi: 10.1093/cei/uxad091. PMID: 37561062Free PMC Article
Miyata T, Horiuchi T
Allergol Int 2023 Jul;72(3):375-384. Epub 2023 May 9 doi: 10.1016/j.alit.2023.04.004. PMID: 37169642
Billebeau A, Fain O, Launay D, Boccon-Gibod I, Bouillet L, Gobert D, Plu-Bureau G, Gompel A; French National Reference Center for Hereditary Angioedema (CREAK)
J Clin Immunol 2021 Jan;41(1):163-170. Epub 2020 Oct 31 doi: 10.1007/s10875-020-00902-7. PMID: 33130967
Csuka D, Veszeli N, Varga L, Prohászka Z, Farkas H
Mol Immunol 2017 Sep;89:59-68. Epub 2017 Jun 7 doi: 10.1016/j.molimm.2017.05.020. PMID: 28595743
Hofman ZL, Relan A, Zeerleder S, Drouet C, Zuraw B, Hack CE
J Allergy Clin Immunol 2016 Aug;138(2):359-66. Epub 2016 May 28 doi: 10.1016/j.jaci.2016.02.041. PMID: 27246526

Recent systematic reviews

Bork K, Steffensen I, Machnig T
Allergy Asthma Proc 2013 Jul-Aug;34(4):312-327. Epub 2013 May 22 doi: 10.2500/aap.2013.34.3677. PMID: 23710659

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