U.S. flag

An official website of the United States government

Format

Send to:

Choose Destination

Links from Gene

Mendelian susceptibility to mycobacterial diseases due to complete IL12RB1 deficiency(IMD30)

MedGen UID:
862386
Concept ID:
C4013949
Disease or Syndrome
Synonyms: IL12RB1 DEFICIENCY; Immunodeficiency 30
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).
 
Gene (location): IL12RB1 (19p13.11)
 
Monarch Initiative: MONDO:0013955
OMIM®: 614891
Orphanet: ORPHA319552

Definition

IMD30 results from autosomal recessive IL12RB1 deficiency and is the most common form of susceptibility to mycobacterial disease. Activated T and natural killer lymphocytes from IMD30 patients do not express IL12RB1 on their surface or, more rarely, express nonfunctional IL12RB1 on their surface. IMD30 patients therefore lack responses to IL12 (see 161560) and IL23 (see 605580). The clinical presentation of IL12RB1-deficient patients is similar to that of IL12B-deficient patients (see IMD29, 614890). Bacillus Calmette-Guerin (BCG) disease and salmonellosis are the most frequent infections. Salmonellosis is present in about half of IL12RB1-deficient patients, and significant numbers of patients present with isolated salmonellosis. Severe tuberculosis has been reported in several unrelated patients, and other infections have been reported in single patients. IMD30 has low penetrance, and patients have relatively mild disease and good prognosis (review by Al-Muhsen and Casanova, 2008). [from OMIM]

Clinical features

From HPO
Immunodeficiency
MedGen UID:
7034
Concept ID:
C0021051
Disease or Syndrome
Failure of the immune system to protect the body adequately from infection, due to the absence or insufficiency of some component process or substance.
Recurrent infections
MedGen UID:
65998
Concept ID:
C0239998
Finding
Increased susceptibility to infections.
Recurrent mycobacterial infections
MedGen UID:
869021
Concept ID:
C4023438
Finding
Increased susceptibility to mycobacterial infections, as manifested by recurrent episodes of mycobacterial infection.

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVMendelian susceptibility to mycobacterial diseases due to complete IL12RB1 deficiency

Recent clinical studies

Diagnosis

Patel S, Uppuluri R, Vellaichamy Swaminathan V, Ravichandran N, Melarcode Ramanan K, Raj R
Pediatr Blood Cancer 2020 May;67(5):e28187. Epub 2020 Jan 21 doi: 10.1002/pbc.28187. PMID: 31965686
Rosain J, Oleaga-Quintas C, Deswarte C, Verdin H, Marot S, Syridou G, Mansouri M, Mahdaviani SA, Venegas-Montoya E, Tsolia M, Mesdaghi M, Chernyshova L, Stepanovskiy Y, Parvaneh N, Mansouri D, Pedraza-Sánchez S, Bondarenko A, Espinosa-Padilla SE, Yamazaki-Nakashimada MA, Nieto-Patlán A, Kerner G, Lambert N, Jacques C, Corvilain E, Migaud M, Grandin V, Herrera MT, Jabot-Hanin F, Boisson-Dupuis S, Picard C, Nitschke P, Puel A, Tores F, Abel L, Blancas-Galicia L, De Baere E, Bole-Feysot C, Casanova JL, Bustamante J
J Clin Immunol 2018 Jul;38(5):617-627. Epub 2018 Jul 11 doi: 10.1007/s10875-018-0527-6. PMID: 29995221Free PMC Article
Alinejad Dizaj M, Mortaz E, Mahdaviani SA, Mansouri D, Mehrian P, Verhard EM, Varahram M, Babaie D, Adcock IM, Garssen J, van de Vosse E, Velayati A
Immunogenetics 2018 Jun;70(6):373-379. Epub 2017 Dec 18 doi: 10.1007/s00251-017-1041-3. PMID: 29256176Free PMC Article
Senanayake MP, Kumararatne DS, Doffinger R, Barcenas-Morales G
Paediatr Int Child Health 2015 Feb;35(1):69-71. Epub 2014 May 27 doi: 10.1179/2046905514Y.0000000129. PMID: 24863105
Remus N, Reichenbach J, Picard C, Rietschel C, Wood P, Lammas D, Kumararatne DS, Casanova JL
Pediatr Res 2001 Jul;50(1):8-13. doi: 10.1203/00006450-200107000-00005. PMID: 11420412

Therapy

Patel S, Uppuluri R, Vellaichamy Swaminathan V, Ravichandran N, Melarcode Ramanan K, Raj R
Pediatr Blood Cancer 2020 May;67(5):e28187. Epub 2020 Jan 21 doi: 10.1002/pbc.28187. PMID: 31965686
Hatipoglu N, Güvenç BH, Deswarte C, Koksalan K, Boisson-Dupuis S, Casanova JL, Bustamante J
Pediatrics 2017 Nov;140(5) Epub 2017 Oct 12 doi: 10.1542/peds.2016-1668. PMID: 29025965Free PMC Article
Senanayake MP, Kumararatne DS, Doffinger R, Barcenas-Morales G
Paediatr Int Child Health 2015 Feb;35(1):69-71. Epub 2014 May 27 doi: 10.1179/2046905514Y.0000000129. PMID: 24863105
Aytekin C, Dogu F, Tuygun N, Tanir G, Guloglu D, Boisson-Dupuis S, Bustamante J, Feinberg J, Casanova JL, Ikinciogullari A
J Investig Allergol Clin Immunol 2011;21(5):401-4. PMID: 21905505Free PMC Article

Prognosis

Nekooie-Marnany N, Deswarte C, Ostadi V, Bagherpour B, Taleby E, Ganjalikhani-Hakemi M, Le Voyer T, Rahimi H, Rosain J, Pourmoghadas Z, Sheikhbahaei S, Khoshnevisan R, Petersheim D, Kotlarz D, Klein C, Boisson-Dupuis S, Casanova JL, Bustamante J, Sherkat R
J Clin Immunol 2018 Oct;38(7):787-793. Epub 2018 Sep 25 doi: 10.1007/s10875-018-0548-1. PMID: 30255293Free PMC Article
Rosain J, Oleaga-Quintas C, Deswarte C, Verdin H, Marot S, Syridou G, Mansouri M, Mahdaviani SA, Venegas-Montoya E, Tsolia M, Mesdaghi M, Chernyshova L, Stepanovskiy Y, Parvaneh N, Mansouri D, Pedraza-Sánchez S, Bondarenko A, Espinosa-Padilla SE, Yamazaki-Nakashimada MA, Nieto-Patlán A, Kerner G, Lambert N, Jacques C, Corvilain E, Migaud M, Grandin V, Herrera MT, Jabot-Hanin F, Boisson-Dupuis S, Picard C, Nitschke P, Puel A, Tores F, Abel L, Blancas-Galicia L, De Baere E, Bole-Feysot C, Casanova JL, Bustamante J
J Clin Immunol 2018 Jul;38(5):617-627. Epub 2018 Jul 11 doi: 10.1007/s10875-018-0527-6. PMID: 29995221Free PMC Article
Senanayake MP, Kumararatne DS, Doffinger R, Barcenas-Morales G
Paediatr Int Child Health 2015 Feb;35(1):69-71. Epub 2014 May 27 doi: 10.1179/2046905514Y.0000000129. PMID: 24863105
Döffinger R, Altare F, Casanova JL
Microbes Infect 2000 Nov;2(13):1553-7. doi: 10.1016/s1286-4579(00)01311-3. PMID: 11113374

Clinical prediction guides

Rosain J, Oleaga-Quintas C, Deswarte C, Verdin H, Marot S, Syridou G, Mansouri M, Mahdaviani SA, Venegas-Montoya E, Tsolia M, Mesdaghi M, Chernyshova L, Stepanovskiy Y, Parvaneh N, Mansouri D, Pedraza-Sánchez S, Bondarenko A, Espinosa-Padilla SE, Yamazaki-Nakashimada MA, Nieto-Patlán A, Kerner G, Lambert N, Jacques C, Corvilain E, Migaud M, Grandin V, Herrera MT, Jabot-Hanin F, Boisson-Dupuis S, Picard C, Nitschke P, Puel A, Tores F, Abel L, Blancas-Galicia L, De Baere E, Bole-Feysot C, Casanova JL, Bustamante J
J Clin Immunol 2018 Jul;38(5):617-627. Epub 2018 Jul 11 doi: 10.1007/s10875-018-0527-6. PMID: 29995221Free PMC Article
Döffinger R, Altare F, Casanova JL
Microbes Infect 2000 Nov;2(13):1553-7. doi: 10.1016/s1286-4579(00)01311-3. PMID: 11113374

Supplemental Content

Recent activity

Your browsing activity is empty.

Activity recording is turned off.

Turn recording back on

See more...