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  • The following term was not found in MedGen: com4.

MHC class II deficiency 4

MedGen UID:
347176
Concept ID:
C1859537
Disease or Syndrome
Synonym: Bare Lymphocyte Syndrome, Type II, Complementation Group D
 
Monarch Initiative: MONDO:0971015
OMIM®: 620817

Definition

MHC class II deficiency-4 (MHC2D4) is an autosomal recessive immunodeficiency characterized by the onset of recurrent, persistent infections from birth, resulting in failure to thrive. Infectious agents include bacteria, viruses, fungi, and protozoa, usually affecting the respiratory and gastrointestinal tract. Laboratory studies show decreased CD4+ T cells, hypogammaglobulinemia, an inverted CD4:CD8 ratio, and absence of MHC type II antigens (HLA-DR, -DQ, and DP) on the surface of antigen-presenting cells. Expression of MHC type I antigens may also be decreased (referred to as 'BLS type III'). Patients may die in infancy or early childhood, unless they undergo bone marrow transplantation, which can be curative, although complications are common. MHC2D4 may not be detected by newborn T-cell receptor excision circle (TREC) screening (summary by Mousavi Khorshidi et al., 2023). For a discussion of genetic heterogeneity of MHC class II deficiency, see MHC2D1 (209920). [from OMIM]

Professional guidelines

PubMed

Korkmaz SB, Karaselek MA, Aytekin SE, Tokgoz H, Reisli I, Guner S, Keles S
Int J Immunogenet 2023 Aug;50(4):177-184. Epub 2023 Jun 12 doi: 10.1111/iji.12624. PMID: 37308802
Wang X, Schoenhals JE, Li A, Valdecanas DR, Ye H, Zang F, Tang C, Tang M, Liu CG, Liu X, Krishnan S, Allison JP, Sharma P, Hwu P, Komaki R, Overwijk WW, Gomez DR, Chang JY, Hahn SM, Cortez MA, Welsh JW
Cancer Res 2017 Feb 15;77(4):839-850. Epub 2016 Nov 7 doi: 10.1158/0008-5472.CAN-15-3142. PMID: 27821490Free PMC Article

Recent clinical studies

Etiology

Gulec Koksal Z, Bilgic Eltan S, Topyildiz E, Sezer A, Keles S, Celebi Celik F, Ozhan Kont A, Gemici Karaaslan B, Sefer AP, Karali Z, Arik E, Ozek Yucel E, Akcal O, Karakurt LT, Yorgun Altunbas M, Yalcin K, Uygun V, Ozek G, Babayeva R, Aydogmus C, Ozcan D, Cavkaytar O, Keskin O, Kilic SS, Kiykim A, Arikoglu T, Genel F, Gulez N, Guner SN, Karaca NE, Reisli I, Kutukculer N, Altintas DU, Ozen A, Karakoc Aydiner E, Baris S
J Allergy Clin Immunol Pract 2024 Sep;12(9):2490-2502.e6. Epub 2024 Jul 10 doi: 10.1016/j.jaip.2024.06.046. PMID: 38996837
Hanna S, Etzioni A
J Allergy Clin Immunol 2014 Aug;134(2):269-75. Epub 2014 Jul 4 doi: 10.1016/j.jaci.2014.06.001. PMID: 25001848
Nekrep N, Fontes JD, Geyer M, Peterlin BM
Immunity 2003 Apr;18(4):453-7. doi: 10.1016/s1074-7613(03)00086-4. PMID: 12705848
Abiru N, Kawasaki E, Eguch K
Diabetes Metab Res Rev 2002 Sep-Oct;18(5):357-66. doi: 10.1002/dmrr.323. PMID: 12397578
Fischer A
Immunodefic Rev 1992;3(2):83-100. PMID: 1554500

Diagnosis

Gulec Koksal Z, Bilgic Eltan S, Topyildiz E, Sezer A, Keles S, Celebi Celik F, Ozhan Kont A, Gemici Karaaslan B, Sefer AP, Karali Z, Arik E, Ozek Yucel E, Akcal O, Karakurt LT, Yorgun Altunbas M, Yalcin K, Uygun V, Ozek G, Babayeva R, Aydogmus C, Ozcan D, Cavkaytar O, Keskin O, Kilic SS, Kiykim A, Arikoglu T, Genel F, Gulez N, Guner SN, Karaca NE, Reisli I, Kutukculer N, Altintas DU, Ozen A, Karakoc Aydiner E, Baris S
J Allergy Clin Immunol Pract 2024 Sep;12(9):2490-2502.e6. Epub 2024 Jul 10 doi: 10.1016/j.jaip.2024.06.046. PMID: 38996837
Lum SH, Anderson C, McNaughton P, Engelhardt KR, MacKenzie B, Watson H, Al-Mousa H, Al-Herz W, Al-Saud B, Mohammed R, Al-Zahrani DM, Alghamdi HA, Goronfolah L, Nademi Z, Habibollah S, Flinn AM, Shillitoe B, Owens S, Williams E, Emonts M, Hambleton S, Abinun M, Flood T, Cant A, Gennery AR, Slatter M
Blood 2020 Mar 19;135(12):954-973. doi: 10.1182/blood.2019002690. PMID: 31932845
Hanna S, Etzioni A
J Allergy Clin Immunol 2014 Aug;134(2):269-75. Epub 2014 Jul 4 doi: 10.1016/j.jaci.2014.06.001. PMID: 25001848
Abiru N, Kawasaki E, Eguch K
Diabetes Metab Res Rev 2002 Sep-Oct;18(5):357-66. doi: 10.1002/dmrr.323. PMID: 12397578
Fischer A
Immunodefic Rev 1992;3(2):83-100. PMID: 1554500

Therapy

Tsilifis C, Schim van der Loeff I, Williams E, Owens S, Powell S, Gennery A, Slatter M
Arch Dis Child Educ Pract Ed 2022 Aug;107(4):274-275. Epub 2020 Dec 18 doi: 10.1136/archdischild-2020-320883. PMID: 33355234
Walline CC, Deffit SN, Wang N, Guindon LM, Crotzer VL, Liu J, Hollister K, Eisenlohr LC, Brutkiewicz RR, Kaplan MH, Blum JS
Immunology 2014 Apr;141(4):531-9. doi: 10.1111/imm.12210. PMID: 24205828Free PMC Article
Engelhorn ME, Guevara-Patiño JA, Merghoub T, Liu C, Ferrone CR, Rizzuto GA, Cymerman DH, Posnett DN, Houghton AN, Wolchok JD
Mol Ther 2008 Apr;16(4):773-81. Epub 2008 Feb 26 doi: 10.1038/mt.2008.8. PMID: 18301399Free PMC Article
MacDonald KP, Rowe V, Clouston AD, Welply JK, Kuns RD, Ferrara JL, Thomas R, Hill GR
J Immunol 2005 Feb 15;174(4):1841-50. doi: 10.4049/jimmunol.174.4.1841. PMID: 15699110
Fischer A
Immunodefic Rev 1992;3(2):83-100. PMID: 1554500

Prognosis

Gulec Koksal Z, Bilgic Eltan S, Topyildiz E, Sezer A, Keles S, Celebi Celik F, Ozhan Kont A, Gemici Karaaslan B, Sefer AP, Karali Z, Arik E, Ozek Yucel E, Akcal O, Karakurt LT, Yorgun Altunbas M, Yalcin K, Uygun V, Ozek G, Babayeva R, Aydogmus C, Ozcan D, Cavkaytar O, Keskin O, Kilic SS, Kiykim A, Arikoglu T, Genel F, Gulez N, Guner SN, Karaca NE, Reisli I, Kutukculer N, Altintas DU, Ozen A, Karakoc Aydiner E, Baris S
J Allergy Clin Immunol Pract 2024 Sep;12(9):2490-2502.e6. Epub 2024 Jul 10 doi: 10.1016/j.jaip.2024.06.046. PMID: 38996837
Lin M, Zhang XL, You R, Yang Q, Zou X, Yu K, Liu YP, Zou RH, Hua YJ, Huang PY, Wang J, Zhao Q, Jiang XB, Tang J, Gu YK, Yu T, He GP, Xie YL, Wang ZQ, Liu T, Chen SY, Zuo ZX, Chen MY
Theranostics 2021;11(13):6427-6444. Epub 2021 Apr 19 doi: 10.7150/thno.53229. PMID: 33995666Free PMC Article
Lum SH, Anderson C, McNaughton P, Engelhardt KR, MacKenzie B, Watson H, Al-Mousa H, Al-Herz W, Al-Saud B, Mohammed R, Al-Zahrani DM, Alghamdi HA, Goronfolah L, Nademi Z, Habibollah S, Flinn AM, Shillitoe B, Owens S, Williams E, Emonts M, Hambleton S, Abinun M, Flood T, Cant A, Gennery AR, Slatter M
Blood 2020 Mar 19;135(12):954-973. doi: 10.1182/blood.2019002690. PMID: 31932845
Iizuka A, Nonomura C, Ashizawa T, Kondou R, Ohshima K, Sugino T, Mitsuya K, Hayashi N, Nakasu Y, Maruyama K, Yamaguchi K, Akiyama Y
Clin Cancer Res 2019 May 1;25(9):2925-2934. Epub 2019 Feb 8 doi: 10.1158/1078-0432.CCR-17-3123. PMID: 30737243
Abiru N, Kawasaki E, Eguch K
Diabetes Metab Res Rev 2002 Sep-Oct;18(5):357-66. doi: 10.1002/dmrr.323. PMID: 12397578

Clinical prediction guides

Gulec Koksal Z, Bilgic Eltan S, Topyildiz E, Sezer A, Keles S, Celebi Celik F, Ozhan Kont A, Gemici Karaaslan B, Sefer AP, Karali Z, Arik E, Ozek Yucel E, Akcal O, Karakurt LT, Yorgun Altunbas M, Yalcin K, Uygun V, Ozek G, Babayeva R, Aydogmus C, Ozcan D, Cavkaytar O, Keskin O, Kilic SS, Kiykim A, Arikoglu T, Genel F, Gulez N, Guner SN, Karaca NE, Reisli I, Kutukculer N, Altintas DU, Ozen A, Karakoc Aydiner E, Baris S
J Allergy Clin Immunol Pract 2024 Sep;12(9):2490-2502.e6. Epub 2024 Jul 10 doi: 10.1016/j.jaip.2024.06.046. PMID: 38996837
Iizuka A, Nonomura C, Ashizawa T, Kondou R, Ohshima K, Sugino T, Mitsuya K, Hayashi N, Nakasu Y, Maruyama K, Yamaguchi K, Akiyama Y
Clin Cancer Res 2019 May 1;25(9):2925-2934. Epub 2019 Feb 8 doi: 10.1158/1078-0432.CCR-17-3123. PMID: 30737243
Walline CC, Deffit SN, Wang N, Guindon LM, Crotzer VL, Liu J, Hollister K, Eisenlohr LC, Brutkiewicz RR, Kaplan MH, Blum JS
Immunology 2014 Apr;141(4):531-9. doi: 10.1111/imm.12210. PMID: 24205828Free PMC Article
Brown GR, Lee EL, Thiele DL
J Immunol 2003 May 15;170(10):5082-8. doi: 10.4049/jimmunol.170.10.5082. PMID: 12734353
Abiru N, Kawasaki E, Eguch K
Diabetes Metab Res Rev 2002 Sep-Oct;18(5):357-66. doi: 10.1002/dmrr.323. PMID: 12397578

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