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MHC class II deficiency 5

MedGen UID:
349183
Concept ID:
C1859538
Disease or Syndrome
Synonyms: Bare lymphocyte syndrome type 2, complementation group E; BARE LYMPHOCYTE SYNDROME, TYPE II, COMPLEMENTATION GROUP E
 
Monarch Initiative: MONDO:0971016
OMIM®: 620818

Definition

MHC class II deficiency-5 (MHC2D5) is an autosomal recessive disorder characterized by defective cell surface expression of class II HLA molecules on the surface of peripheral blood B cells, monocytes, and activated T cells. Affected individuals may present in infancy with recurrent infections and hypogammaglobulinemia, but patients do not develop severe infections, as observed in other forms of MHC class II deficiency. Some individuals may be asymptomatic (Wolf et al., 1995). 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
Howard TE, Yanover C, Mahlangu J, Krause A, Viel KR, Kasper CK, Pratt KP
Haemophilia 2011 Sep;17(5):721-8. Epub 2011 Jun 8 doi: 10.1111/j.1365-2516.2011.02517.x. PMID: 21649795Free PMC Article

Recent clinical studies

Etiology

Bhakat B, Pal J, Das S, Charaborty SK, SircarMedical NR, Kolkata, RGKar, NorthBengal, Siliguri
J Assoc Physicians India 2023 Jan;71(1):1. PMID: 37116030
Guo L, Shen S, Rowley JW, Tolley ND, Jia W, Manne BK, McComas KN, Bolingbroke B, Kosaka Y, Krauel K, Denorme F, Jacob SP, Eustes AS, Campbell RA, Middleton EA, He X, Brown SM, Morrell CN, Weyrich AS, Rondina MT
Blood 2021 Aug 5;138(5):401-416. doi: 10.1182/blood.2020008958. PMID: 33895821Free PMC Article
Howard TE, Yanover C, Mahlangu J, Krause A, Viel KR, Kasper CK, Pratt KP
Haemophilia 2011 Sep;17(5):721-8. Epub 2011 Jun 8 doi: 10.1111/j.1365-2516.2011.02517.x. PMID: 21649795Free PMC Article
Abiru N, Kawasaki E, Eguch K
Diabetes Metab Res Rev 2002 Sep-Oct;18(5):357-66. doi: 10.1002/dmrr.323. PMID: 12397578
Wilson WA, Gharavi AE
Lupus 1996 Oct;5(5):398-403. doi: 10.1177/096120339600500513. PMID: 8902769

Diagnosis

Radhika R, Tullu MS, Karande S, Bargir UA
Indian J Pediatr 2024 Sep;91(9):967-969. Epub 2023 Jul 20 doi: 10.1007/s12098-023-04750-2. PMID: 37470959
Mousavi Khorshidi MS, Seeleuthner Y, Chavoshzadeh Z, Behfar M, Hamidieh AA, Alimadadi H, Sherkat R, Momen T, Behniafard N, Eskandarzadeh S, Mansouri M, Behnam M, Mahdavi M, Heydarazad Zadeh M, Shokri M, Alizadeh F, Movahedi M, Momenilandi M, Keramatipour M, Casanova JL, Cobat A, Abel L, Shahrooei M, Parvaneh N
J Clin Immunol 2023 Nov;43(8):1941-1952. Epub 2023 Aug 16 doi: 10.1007/s10875-023-01562-z. PMID: 37584719
Bhakat B, Pal J, Das S, Charaborty SK, SircarMedical NR, Kolkata, RGKar, NorthBengal, Siliguri
J Assoc Physicians India 2023 Jan;71(1):1. PMID: 37116030
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
Abiru N, Kawasaki E, Eguch K
Diabetes Metab Res Rev 2002 Sep-Oct;18(5):357-66. doi: 10.1002/dmrr.323. PMID: 12397578

Therapy

Luo Y, Xia Y, Liu D, Li X, Li H, Liu J, Zhou D, Dong Y, Li X, Qian Y, Xu C, Tao K, Li G, Pan W, Zhong Q, Liu X, Xu S, Wang Z, Liu R, Zhang W, Shan W, Fang T, Wang S, Peng Z, Jin P, Jin N, Shi S, Chen Y, Wang M, Jiao X, Luo M, Gong W, Wang Y, Yao Y, Zhao Y, Huang X, Ji X, He Z, Zhao G, Liu R, Wu M, Chen G, Hong L; COCPO Consortium, Ma D, Fang Y, Liang H, Gao Q
Cell 2024 Sep 5;187(18):4905-4925.e24. Epub 2024 Jul 5 doi: 10.1016/j.cell.2024.06.013. PMID: 38971151
Bhakat B, Pal J, Das S, Charaborty SK, SircarMedical NR, Kolkata, RGKar, NorthBengal, Siliguri
J Assoc Physicians India 2023 Jan;71(1):1. PMID: 37116030
Abolhassani H, Lim CK, Aghamohammadi A, Hammarström L
Front Immunol 2020;11:14. Epub 2020 Jan 24 doi: 10.3389/fimmu.2020.00014. PMID: 32038658Free 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
Howard TE, Yanover C, Mahlangu J, Krause A, Viel KR, Kasper CK, Pratt KP
Haemophilia 2011 Sep;17(5):721-8. Epub 2011 Jun 8 doi: 10.1111/j.1365-2516.2011.02517.x. PMID: 21649795Free PMC Article

Prognosis

Dennison TW, Edgar RD, Payne F, Nayak KM, Ross ADB, Cenier A, Glemas C, Giachero F, Foster AR, Harris R, Kraiczy J, Salvestrini C, Stavrou G, Torrente F, Brook K, Trayers C, Elmentaite R, Youssef G, Tél B, Winton DJ, Skoufou-Papoutsaki N, Adler S, Bufler P, Azabdaftari A, Jenke A, G N, Thomas N, Miele E, Al-Mohammad A, Guarda G, Kugathasan S, Venkateswaran S, Clatworthy MR, Castro-Dopico T, Suchanek O, Strisciuglio C, Gasparetto M, Lee S, Xu X, Bello E, Han N, Zerbino DR, Teichmann SA, Nys J, Heuschkel R, Perrone F, Zilbauer M
Gut 2024 Aug 8;73(9):1464-1477. doi: 10.1136/gutjnl-2024-332043. PMID: 38857990Free PMC Article
Radhika R, Tullu MS, Karande S, Bargir UA
Indian J Pediatr 2024 Sep;91(9):967-969. Epub 2023 Jul 20 doi: 10.1007/s12098-023-04750-2. PMID: 37470959
Mousavi Khorshidi MS, Seeleuthner Y, Chavoshzadeh Z, Behfar M, Hamidieh AA, Alimadadi H, Sherkat R, Momen T, Behniafard N, Eskandarzadeh S, Mansouri M, Behnam M, Mahdavi M, Heydarazad Zadeh M, Shokri M, Alizadeh F, Movahedi M, Momenilandi M, Keramatipour M, Casanova JL, Cobat A, Abel L, Shahrooei M, Parvaneh N
J Clin Immunol 2023 Nov;43(8):1941-1952. Epub 2023 Aug 16 doi: 10.1007/s10875-023-01562-z. PMID: 37584719
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
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

Luo Y, Xia Y, Liu D, Li X, Li H, Liu J, Zhou D, Dong Y, Li X, Qian Y, Xu C, Tao K, Li G, Pan W, Zhong Q, Liu X, Xu S, Wang Z, Liu R, Zhang W, Shan W, Fang T, Wang S, Peng Z, Jin P, Jin N, Shi S, Chen Y, Wang M, Jiao X, Luo M, Gong W, Wang Y, Yao Y, Zhao Y, Huang X, Ji X, He Z, Zhao G, Liu R, Wu M, Chen G, Hong L; COCPO Consortium, Ma D, Fang Y, Liang H, Gao Q
Cell 2024 Sep 5;187(18):4905-4925.e24. Epub 2024 Jul 5 doi: 10.1016/j.cell.2024.06.013. PMID: 38971151
Baschal EE, Eisenbarth GS
J Autoimmun 2008 Aug;31(1):1-6. Epub 2008 May 1 doi: 10.1016/j.jaut.2008.03.003. PMID: 18450419Free PMC Article
Dong F, Pirbhai M, Zhong Y, Zhong G
Mol Microbiol 2004 Jun;52(5):1487-94. doi: 10.1111/j.1365-2958.2004.04072.x. PMID: 15165249
Lovitch SB, Petzold SJ, Unanue ER
J Immunol 2003 Sep 1;171(5):2183-6. doi: 10.4049/jimmunol.171.5.2183. PMID: 12928360
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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