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Absent specific antibody response

MedGen UID:
354937
Concept ID:
C1863246
Finding
HPO: HP:0005424

Definition

Absence of specific immunoglobulins directed against a specific antigen or microorganism. [from HPO]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVAbsent specific antibody response

Conditions with this feature

Severe combined immunodeficiency, autosomal recessive, T cell-negative, B cell-negative, NK cell-negative, due to adenosine deaminase deficiency
MedGen UID:
354935
Concept ID:
C1863236
Disease or Syndrome
Adenosine deaminase (ADA) deficiency is a systemic purine metabolic disorder that primarily affects lymphocyte development, viability, and function. The clinical phenotypic spectrum includes: Severe combined immunodeficiency disease (SCID), often diagnosed by age six months and usually by age 12 months; Less severe "delayed" onset combined immune deficiency (CID), usually diagnosed between age one and ten years; "Late/adult onset" CID, diagnosed in the second to fourth decades; Benign "partial ADA deficiency" (very low or absent ADA activity in erythrocytes but greater ADA activity in nucleated cells), which is compatible with normal immune function. Infants with typical early-onset ADA-deficient SCID have failure to thrive and opportunistic infections associated with marked depletion of T, B, and NK lymphocytes, and an absence of both humoral and cellular immune function. If immune function is not restored, children with ADA-deficient SCID rarely survive beyond age one to two years. Infections in delayed- and late-onset types (commonly, recurrent otitis, sinusitis, and upper respiratory) may initially be less severe than those in individuals with ADA-deficient SCID; however, by the time of diagnosis these individuals often have chronic pulmonary insufficiency and may have autoimmune phenomena (cytopenias, anti-thyroid antibodies), allergies, and elevated serum concentration of IgE. The longer the disorder goes unrecognized, the more immune function deteriorates and the more likely are chronic sequelae of recurrent infection.
Immunodeficiency 99 with hypogammaglobulinemia and autoimmune cytopenias
MedGen UID:
1801342
Concept ID:
C5676971
Disease or Syndrome
Immunodeficiency-99 with hypogammaglobulinemia and autoimmune cytopenias (IMD99) is an autosomal recessive immunologic disorder characterized by the onset of recurrent sinopulmonary infections in early childhood. Laboratory studies reveal hypogammaglobulinemia with decreased memory B cells that show impaired class-switch recombination (CSR) and decreased somatic hypermutation (SHM). Due to abnormal antibody production and impaired self-tolerance, patients may develop autoimmune cytopenias, such as thrombocytopenia, or autoimmune features, such as vitiligo. There are also defects in the T-cell compartment (Kuhny et al., 2020).

Professional guidelines

PubMed

Subramaniam S, Fares-Gusmao R, Sato S, Cullen JM, Takeda K, Farci P, McGivern DR
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Recent clinical studies

Etiology

Simula ER, Zarbo IR, Arru G, Sechi E, Meloni R, Deiana GA, Solla P, Sechi LA
Int J Mol Sci 2023 Dec 28;25(1) doi: 10.3390/ijms25010446. PMID: 38203616Free PMC Article
Wagner A, Jasinska J, Tomosel E, Zielinski CC, Wiedermann U
Vaccine 2021 Dec 17;39(51):7375-7378. Epub 2021 Nov 1 doi: 10.1016/j.vaccine.2021.10.068. PMID: 34785100Free PMC Article
Alaniz ME, Lardone RD, Yudowski SL, Farace MI, Nores GA
Infect Immun 2004 Apr;72(4):2148-51. doi: 10.1128/IAI.72.4.2148-2151.2004. PMID: 15039337Free PMC Article
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Peterson RG, Handschumacher RE, Mitchell MS
J Clin Invest 1971 May;50(5):1080-90. doi: 10.1172/JCI106579. PMID: 4101590Free PMC Article

Diagnosis

Saikia B, Gupta S
Indian J Pediatr 2016 Apr;83(4):338-44. Epub 2016 Feb 12 doi: 10.1007/s12098-016-2038-x. PMID: 26868026
Iversen R, Di Niro R, Stamnaes J, Lundin KE, Wilson PC, Sollid LM
J Immunol 2013 Jun 15;190(12):5981-91. Epub 2013 May 20 doi: 10.4049/jimmunol.1300183. PMID: 23690478Free PMC Article
Alaniz ME, Lardone RD, Yudowski SL, Farace MI, Nores GA
Infect Immun 2004 Apr;72(4):2148-51. doi: 10.1128/IAI.72.4.2148-2151.2004. PMID: 15039337Free PMC Article
Bliss RD, Kirby JA, Browell DA, Lennard TW
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Therapy

Heitmann JS, Bilich T, Tandler C, Nelde A, Maringer Y, Marconato M, Reusch J, Jäger S, Denk M, Richter M, Anton L, Weber LM, Roerden M, Bauer J, Rieth J, Wacker M, Hörber S, Peter A, Meisner C, Fischer I, Löffler MW, Karbach J, Jäger E, Klein R, Rammensee HG, Salih HR, Walz JS
Nature 2022 Jan;601(7894):617-622. Epub 2021 Nov 23 doi: 10.1038/s41586-021-04232-5. PMID: 34814158Free PMC Article
Klyushnenkova EN, Riabov VB, Kouiavskaia DV, Wietsma A, Zhan M, Alexander RB
Prostate 2014 Oct;74(14):1423-32. Epub 2014 Aug 11 doi: 10.1002/pros.22858. PMID: 25111463
Höhn Y, Sudowe S, Reske-Kunz AB
Methods Mol Biol 2013;940:199-213. doi: 10.1007/978-1-62703-110-3_17. PMID: 23104345
Wiertsema SP, Veenhoven RH, Walraven V, Uiterwaal CS, Schilder AG, Rijkers GT, Sanders EA
Vaccine 2006 Feb 6;24(6):792-7. Epub 2005 Aug 22 doi: 10.1016/j.vaccine.2005.08.029. PMID: 16198451
Peterson RG, Handschumacher RE, Mitchell MS
J Clin Invest 1971 May;50(5):1080-90. doi: 10.1172/JCI106579. PMID: 4101590Free PMC Article

Prognosis

Guo Y, Endmayr V, Zekeridou A, McKeon A, Leypoldt F, Hess K, Kalinowska-Lyszczarz A, Klang A, Pakozdy A, Höftberger E, Hametner S, Haider C, De Simoni D, Peters S, Gelpi E, Röcken C, Oberndorfer S, Lassmann H, Lucchinetti CF, Höftberger R
Acta Neuropathol 2024 Feb 3;147(1):31. doi: 10.1007/s00401-023-02678-7. PMID: 38310187Free PMC Article
Charakidas A, Kalogeraki A, Tsilimbaris M, Koukoulomatis P, Brouzas D, Delides G
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Holevinsky KO, Nelson DJ
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Simon M, Green H
Cell 1984 Apr;36(4):827-34. doi: 10.1016/0092-8674(84)90032-1. PMID: 6200236
Peterson RG, Handschumacher RE, Mitchell MS
J Clin Invest 1971 May;50(5):1080-90. doi: 10.1172/JCI106579. PMID: 4101590Free PMC Article

Clinical prediction guides

Guo Y, Endmayr V, Zekeridou A, McKeon A, Leypoldt F, Hess K, Kalinowska-Lyszczarz A, Klang A, Pakozdy A, Höftberger E, Hametner S, Haider C, De Simoni D, Peters S, Gelpi E, Röcken C, Oberndorfer S, Lassmann H, Lucchinetti CF, Höftberger R
Acta Neuropathol 2024 Feb 3;147(1):31. doi: 10.1007/s00401-023-02678-7. PMID: 38310187Free PMC Article
Federica G, Giuseppina F, Veronica L, Gianpaolo Z, Massimo T, Veronica M, Giuseppe S, Maria TA
Sci Rep 2023 Jan 12;13(1):629. doi: 10.1038/s41598-022-26156-4. PMID: 36635345Free PMC Article
Heitmann JS, Bilich T, Tandler C, Nelde A, Maringer Y, Marconato M, Reusch J, Jäger S, Denk M, Richter M, Anton L, Weber LM, Roerden M, Bauer J, Rieth J, Wacker M, Hörber S, Peter A, Meisner C, Fischer I, Löffler MW, Karbach J, Jäger E, Klein R, Rammensee HG, Salih HR, Walz JS
Nature 2022 Jan;601(7894):617-622. Epub 2021 Nov 23 doi: 10.1038/s41586-021-04232-5. PMID: 34814158Free PMC Article
Tankersley DL
Immunol Rev 1994 Jun;139:159-72. doi: 10.1111/j.1600-065x.1994.tb00861.x. PMID: 7927410
Peterson RG, Handschumacher RE, Mitchell MS
J Clin Invest 1971 May;50(5):1080-90. doi: 10.1172/JCI106579. PMID: 4101590Free PMC Article

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