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Immune dysregulation-inflammatory bowel disease-arthritis-recurrent infections-lymphopenia syndrome

MedGen UID:
1799956
Concept ID:
C5568533
Disease or Syndrome
Synonyms: Immune dysregulation, inflammatory bowel disease, arthritis, recurrent infection, lymphopenia syndrome; immune dysregulation-inflammatory bowel disease-arthritis-recurrent infections-lymphopenia syndrome
SNOMED CT: Immune dysregulation, inflammatory bowel disease, arthritis, recurrent infection, lymphopenia syndrome (1186654001)
Modes of inheritance:
Autosomal recessive inheritance
MedGen UID:
141025
Concept ID:
C0441748
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 individuals with two pathogenic alleles, either homozygotes (two copies of the same mutant allele) or compound heterozygotes (whereby each copy of a gene has a distinct mutant allele).
 
Monarch Initiative: MONDO:0033968
Orphanet: ORPHA529977

Definition

A rare genetic immune disease characterised by early onset of recurrent bacterial, viral and fungal infections, chronic inflammatory bowel disease, gastritis and inflammatory polyarthritis. Patients present with diarrhoea, vomiting, hepatosplenomegaly, mouth ulcers, perianal abscesses, chronic lung disease with bronchiectasis and failure to thrive. Occurrence of a skin rash associated with lymphocytic vasculitis has also been reported. Immunologic abnormalities include variable T-cell lymphopenia, decreased natural killer cells, and decreased B-cells with variable hypogammaglobulinaemia. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVImmune dysregulation-inflammatory bowel disease-arthritis-recurrent infections-lymphopenia syndrome

Professional guidelines

PubMed

Camilleri M, Boeckxstaens G
Gut 2023 Mar;72(3):590-599. Epub 2022 Oct 28 doi: 10.1136/gutjnl-2022-328515. PMID: 36307180Free PMC Article
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Expert Rev Gastroenterol Hepatol 2020 Jan;14(1):39-46. Epub 2020 Jan 2 doi: 10.1080/17474124.2020.1708718. PMID: 31893959
Craven MD, Washabau RJ
J Vet Intern Med 2019 Mar;33(2):383-402. Epub 2019 Feb 14 doi: 10.1111/jvim.15406. PMID: 30762910Free PMC Article

Recent clinical studies

Etiology

Camilleri M, Zheng T
Clin Gastroenterol Hepatol 2023 Dec;21(13):3217-3229. Epub 2023 Sep 9 doi: 10.1016/j.cgh.2023.07.031. PMID: 37678488Free PMC Article
Mamieva Z, Poluektova E, Svistushkin V, Sobolev V, Shifrin O, Guarner F, Ivashkin V
World J Gastroenterol 2022 Mar 28;28(12):1204-1219. doi: 10.3748/wjg.v28.i12.1204. PMID: 35431513Free PMC Article
Holtmann GJ, Ford AC, Talley NJ
Lancet Gastroenterol Hepatol 2016 Oct;1(2):133-146. Epub 2016 Sep 8 doi: 10.1016/S2468-1253(16)30023-1. PMID: 28404070
Qin HY, Cheng CW, Tang XD, Bian ZX
World J Gastroenterol 2014 Oct 21;20(39):14126-31. doi: 10.3748/wjg.v20.i39.14126. PMID: 25339801Free PMC Article
Saper RB, Rash R
Am Fam Physician 2009 May 1;79(9):768-72. PMID: 20141096Free PMC Article

Diagnosis

Di Tommaso N, Gasbarrini A, Ponziani FR
Int J Environ Res Public Health 2021 Dec 6;18(23) doi: 10.3390/ijerph182312836. PMID: 34886561Free PMC Article
Eraksoy H
Gastroenterol Clin North Am 2021 Jun;50(2):341-360. doi: 10.1016/j.gtc.2021.02.004. PMID: 34024445
Clark R, Johnson R
Nurs Clin North Am 2018 Sep;53(3):361-374. Epub 2018 Jul 11 doi: 10.1016/j.cnur.2018.05.001. PMID: 30100002
Chey WD, Kurlander J, Eswaran S
JAMA 2015 Mar 3;313(9):949-58. doi: 10.1001/jama.2015.0954. PMID: 25734736
Qin HY, Cheng CW, Tang XD, Bian ZX
World J Gastroenterol 2014 Oct 21;20(39):14126-31. doi: 10.3748/wjg.v20.i39.14126. PMID: 25339801Free PMC Article

Therapy

Stoeva MK, Garcia-So J, Justice N, Myers J, Tyagi S, Nemchek M, McMurdie PJ, Kolterman O, Eid J
Gut Microbes 2021 Jan-Dec;13(1):1-28. doi: 10.1080/19490976.2021.1907272. PMID: 33874858Free PMC Article
Cristofori F, Dargenio VN, Dargenio C, Miniello VL, Barone M, Francavilla R
Front Immunol 2021;12:578386. Epub 2021 Feb 26 doi: 10.3389/fimmu.2021.578386. PMID: 33717063Free PMC Article
Hidalgo-Cantabrana C, Delgado S, Ruiz L, Ruas-Madiedo P, Sánchez B, Margolles A
Microbiol Spectr 2017 Jun;5(3) doi: 10.1128/microbiolspec.BAD-0010-2016. PMID: 28643627
Chey WD, Kurlander J, Eswaran S
JAMA 2015 Mar 3;313(9):949-58. doi: 10.1001/jama.2015.0954. PMID: 25734736
Bischoff SC, Barbara G, Buurman W, Ockhuizen T, Schulzke JD, Serino M, Tilg H, Watson A, Wells JM
BMC Gastroenterol 2014 Nov 18;14:189. doi: 10.1186/s12876-014-0189-7. PMID: 25407511Free PMC Article

Prognosis

Shulpekova YO, Nechaev VM, Popova IR, Deeva TA, Kopylov AT, Malsagova KA, Kaysheva AL, Ivashkin VT
Nutrients 2021 Sep 15;13(9) doi: 10.3390/nu13093207. PMID: 34579083Free PMC Article
Ford AC, Sperber AD, Corsetti M, Camilleri M
Lancet 2020 Nov 21;396(10263):1675-1688. Epub 2020 Oct 10 doi: 10.1016/S0140-6736(20)31548-8. PMID: 33049223
Clark R, Johnson R
Nurs Clin North Am 2018 Sep;53(3):361-374. Epub 2018 Jul 11 doi: 10.1016/j.cnur.2018.05.001. PMID: 30100002
Joyce E, Glasner P, Ranganathan S, Swiatecka-Urban A
Pediatr Nephrol 2017 Apr;32(4):577-587. Epub 2016 May 7 doi: 10.1007/s00467-016-3394-5. PMID: 27155873Free PMC Article
Qin HY, Cheng CW, Tang XD, Bian ZX
World J Gastroenterol 2014 Oct 21;20(39):14126-31. doi: 10.3748/wjg.v20.i39.14126. PMID: 25339801Free PMC Article

Clinical prediction guides

Man AM, Orăsan MS, Hoteiuc OA, Olănescu-Vaida-Voevod MC, Mocan T
Int J Mol Sci 2023 Nov 8;24(22) doi: 10.3390/ijms242216095. PMID: 38003284Free PMC Article
Vanuytsel T, Bercik P, Boeckxstaens G
Gut 2023 Apr;72(4):787-798. Epub 2023 Jan 19 doi: 10.1136/gutjnl-2020-320633. PMID: 36657961Free PMC Article
Kinashi Y, Hase K
Front Immunol 2021;12:673708. Epub 2021 Apr 22 doi: 10.3389/fimmu.2021.673708. PMID: 33968085Free PMC Article
Spiller R
Nutrients 2021 Feb 9;13(2) doi: 10.3390/nu13020575. PMID: 33572262Free PMC Article
Glissen Brown JR, Singh P
Paediatr Int Child Health 2019 Feb;39(1):23-31. Epub 2018 Aug 13 doi: 10.1080/20469047.2018.1504431. PMID: 30099930

Recent systematic reviews

Li M, Wan J, Xu Z, Tang B
Front Immunol 2023;14:1103053. Epub 2023 Jan 25 doi: 10.3389/fimmu.2023.1103053. PMID: 36761731Free PMC Article
Cheng CY, Baritussio A, Giordani AS, Iliceto S, Marcolongo R, Caforio ALP
Autoimmun Rev 2022 Apr;21(4):103037. Epub 2022 Jan 5 doi: 10.1016/j.autrev.2022.103037. PMID: 34995763
Shigesi N, Kvaskoff M, Kirtley S, Feng Q, Fang H, Knight JC, Missmer SA, Rahmioglu N, Zondervan KT, Becker CM
Hum Reprod Update 2019 Jul 1;25(4):486-503. doi: 10.1093/humupd/dmz014. PMID: 31260048Free PMC Article
Bashashati M, Moossavi S, Cremon C, Barbaro MR, Moraveji S, Talmon G, Rezaei N, Hughes PA, Bian ZX, Choi CH, Lee OY, Coëffier M, Chang L, Ohman L, Schmulson MJ, McCallum RW, Simren M, Sharkey KA, Barbara G
Neurogastroenterol Motil 2018 Jan;30(1) Epub 2017 Aug 29 doi: 10.1111/nmo.13192. PMID: 28851005
Kechichian E, Haber R, Mourad N, El Khoury R, Jabbour S, Tomb R
Int J Dermatol 2017 May;56(5):486-495. Epub 2017 Feb 23 doi: 10.1111/ijd.13584. PMID: 28233293

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