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Hyper-IgM syndrome with susceptibility to opportunistic infections

MedGen UID:
1673500
Concept ID:
C5190885
Disease or Syndrome
Synonyms: HIGM with susceptibility to opportunistic infections; Hyperimmunoglobulin M syndrome with susceptibility to opportunistic infection
SNOMED CT: Hyper-IgM syndrome with susceptibility to opportunistic infections (783248004); Hyperimmunoglobulin M syndrome with susceptibility to opportunistic infection (783248004)
 
Orphanet: ORPHA183663

Definition

A rare genetic non-severe combined immunodeficiency disorder with characteristics of normal or elevated IgM serum levels with low or absent IgG, IgA and IgE serum concentrations, which manifests with recurrent or severe bacterial infections and increased susceptibility to opportunistic infections (in particular, pneumonia due to P. jiroveci, but also chronic cryptosporidial, cryptococcal, cytomegalovirus and toxoplasma infections). Haematologic disorders (neutropenia, anaemia, thrombocytopenia) are frequently associated. Immunologic findings reveal decreased numbers of CD27+ memory B cells and lack of germinal centre formation. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVHyper-IgM syndrome with susceptibility to opportunistic infections

Professional guidelines

PubMed

França TT, Barreiros LA, Al-Ramadi BK, Ochs HD, Cabral-Marques O, Condino-Neto A
Expert Rev Clin Immunol 2019 May;15(5):529-540. Epub 2019 Feb 18 doi: 10.1080/1744666X.2019.1573674. PMID: 30681380

Recent clinical studies

Etiology

Drgona L, Gudiol C, Lanini S, Salzberger B, Ippolito G, Mikulska M
Clin Microbiol Infect 2018 Jun;24 Suppl 2:S83-S94. Epub 2018 Mar 20 doi: 10.1016/j.cmi.2018.03.022. PMID: 29572070
Lee PP
Ann Acad Med Singap 2015 Aug;44(8):297-301. PMID: 26477962
Montella S, Maglione M, Giardino G, Di Giorgio A, Palamaro L, Mirra V, Ursini MV, Salerno M, Pignata C, Caffarelli C, Santamaria F
Ital J Pediatr 2012 Sep 19;38:45. doi: 10.1186/1824-7288-38-45. PMID: 22992442Free PMC Article
Davies EG, Thrasher AJ
Br J Haematol 2010 Apr;149(2):167-80. Epub 2010 Feb 23 doi: 10.1111/j.1365-2141.2010.08077.x. PMID: 20180797Free PMC Article
Tomizawa D, Imai K, Ito S, Kajiwara M, Minegishi Y, Nagasawa M, Morio T, Nonoyama S, Mizutani S
Am J Hematol 2004 May;76(1):33-9. doi: 10.1002/ajh.20044. PMID: 15114594

Diagnosis

Yan H, Mo Y, Liu S, Luo X, Liu L, Zhou L, Zhang X, Chen Y, Cao K
Front Immunol 2022;13:1038354. Epub 2022 Dec 2 doi: 10.3389/fimmu.2022.1038354. PMID: 36532052Free PMC Article
Palterer B, Salvati L, Capone M, Mecheri V, Maggi L, Mazzoni A, Cosmi L, Volpi N, Tiberi L, Provenzano A, Giglio S, Parronchi P, Maggiore G, Gallo O, Bartoloni A, Annunziato F, Zammarchi L, Liotta F
Front Immunol 2022;13:840767. Epub 2022 Apr 28 doi: 10.3389/fimmu.2022.840767. PMID: 35572607Free PMC Article
Lee PP
Ann Acad Med Singap 2015 Aug;44(8):297-301. PMID: 26477962
Montella S, Maglione M, Giardino G, Di Giorgio A, Palamaro L, Mirra V, Ursini MV, Salerno M, Pignata C, Caffarelli C, Santamaria F
Ital J Pediatr 2012 Sep 19;38:45. doi: 10.1186/1824-7288-38-45. PMID: 22992442Free PMC Article
Winkelstein JA, Marino MC, Ochs H, Fuleihan R, Scholl PR, Geha R, Stiehm ER, Conley ME
Medicine (Baltimore) 2003 Nov;82(6):373-84. doi: 10.1097/01.md.0000100046.06009.b0. PMID: 14663287

Therapy

Pacharn P, Phongsamart W, Boonyawat B, Jirapongsananuruk O, Visitsunthorn N, Chokephaibulkit K
Asian Pac J Allergy Immunol 2021 Mar;39(1):31-34. doi: 10.12932/AP-140218-0265. PMID: 30447657
França TT, Barreiros LA, Al-Ramadi BK, Ochs HD, Cabral-Marques O, Condino-Neto A
Expert Rev Clin Immunol 2019 May;15(5):529-540. Epub 2019 Feb 18 doi: 10.1080/1744666X.2019.1573674. PMID: 30681380
Drgona L, Gudiol C, Lanini S, Salzberger B, Ippolito G, Mikulska M
Clin Microbiol Infect 2018 Jun;24 Suppl 2:S83-S94. Epub 2018 Mar 20 doi: 10.1016/j.cmi.2018.03.022. PMID: 29572070
Lee PP
Ann Acad Med Singap 2015 Aug;44(8):297-301. PMID: 26477962
Winkelstein JA, Marino MC, Ochs H, Fuleihan R, Scholl PR, Geha R, Stiehm ER, Conley ME
Medicine (Baltimore) 2003 Nov;82(6):373-84. doi: 10.1097/01.md.0000100046.06009.b0. PMID: 14663287

Prognosis

França TT, Barreiros LA, Al-Ramadi BK, Ochs HD, Cabral-Marques O, Condino-Neto A
Expert Rev Clin Immunol 2019 May;15(5):529-540. Epub 2019 Feb 18 doi: 10.1080/1744666X.2019.1573674. PMID: 30681380
Tomizawa D, Imai K, Ito S, Kajiwara M, Minegishi Y, Nagasawa M, Morio T, Nonoyama S, Mizutani S
Am J Hematol 2004 May;76(1):33-9. doi: 10.1002/ajh.20044. PMID: 15114594

Clinical prediction guides

Drgona L, Gudiol C, Lanini S, Salzberger B, Ippolito G, Mikulska M
Clin Microbiol Infect 2018 Jun;24 Suppl 2:S83-S94. Epub 2018 Mar 20 doi: 10.1016/j.cmi.2018.03.022. PMID: 29572070
Subauste CS, Wessendarp M, Sorensen RU, Leiva LE
J Immunol 1999 Jun 1;162(11):6690-700. PMID: 10352287
Revy P, Geissmann F, Debré M, Fischer A, Durandy A
Eur J Immunol 1998 Nov;28(11):3648-54. doi: 10.1002/(SICI)1521-4141(199811)28:11<3648::AID-IMMU3648>3.0.CO;2-U. PMID: 9842907

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