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Immunodeficiency 105(IMD105)

MedGen UID:
1809425
Concept ID:
C5677005
Disease or Syndrome
Synonyms: IMD105; Immunodeficiency 105, severe combined
 
Gene (location): PTPRC (1q31.3-32.1)
 
Monarch Initiative: MONDO:0800104
OMIM®: 619924

Definition

Immunodeficiency-105 (IMD105) is an autosomal recessive disorder characterized by onset of recurrent infections in early infancy. Manifestations may include pneumonia, dermatitis, and lymphadenopathy. B-cell lymphoma was reported in 1 patient. Laboratory studies show decreased or absent numbers of nonfunctional T cells, normal or increased levels of B cells, hypogammaglobulinemia, and normal or low NK cells. The disorder is caused by a deficiency of transmembrane protein CD45 (PTPRC) on leukocytes, which plays an important role in T- and B-cell development (Cale et al., 1997; Kung et al., 2000). For a general phenotypic description and a discussion of genetic heterogeneity of autosomal recessive SCID, see 601457. [from OMIM]

Clinical features

From HPO
B-cell lymphoma
MedGen UID:
86953
Concept ID:
C0079731
Neoplastic Process
A type of lymphoma that originates in B-cells.
Hepatosplenomegaly
MedGen UID:
9225
Concept ID:
C0019214
Sign or Symptom
Simultaneous enlargement of the liver and spleen.
Pancytopenia
MedGen UID:
18281
Concept ID:
C0030312
Disease or Syndrome
An abnormal reduction in numbers of all blood cell types (red blood cells, white blood cells, and platelets).
Lymphopenia
MedGen UID:
7418
Concept ID:
C0024312
Disease or Syndrome
A reduced number of lymphocytes in the blood.
Decreased circulating IgA concentration
MedGen UID:
57934
Concept ID:
C0162538
Disease or Syndrome
Decreased levels of immunoglobulin A (IgA).
Decreased circulating total IgM
MedGen UID:
116095
Concept ID:
C0239989
Finding
An abnormally decreased level of immunoglobulin M (IgM) in blood.
Absence of lymph node germinal center
MedGen UID:
376112
Concept ID:
C1847383
Finding
Absence of germinal centers in lymph nodes. Germinal centers are the parts of lymph nodes in which B lymphocytes proliferate, differentiate, mutate through somatic hypermutation and class switch during antibody responses.
B lymphocytopenia
MedGen UID:
340780
Concept ID:
C1855067
Finding
An abnormal decrease from the normal count of B cells.
Reduced natural killer cell count
MedGen UID:
383765
Concept ID:
C1855767
Finding
Less than normal number of natural killer cells, a type of lymphocyte in the innate immune system with an ability to mediate cytotoxicity and produce cytokines after the ligation of a germline-encoded activation receptor.
Increased B cell count
MedGen UID:
349067
Concept ID:
C1858972
Finding
An abnormal increase from the normal count of B cells.
T lymphocytopenia
MedGen UID:
419385
Concept ID:
C2931322
Finding
An abnormally low count of T cells.
Impaired lymphocyte transformation with phytohemagglutinin
MedGen UID:
871152
Concept ID:
C4025625
Finding
Normal peripheral blood lymphocytes, when stimulated by phytohemagglutinin (PHA) are cytotoxic for homologous and heterologous cells but not for autologous cells in monolayer culture. The cytotoxic effect is thought to be indicative of the immunological competence of the lymphocytes.
Decreased circulating antibody concentration
MedGen UID:
892481
Concept ID:
C4048270
Finding
An abnormally decreased level of immunoglobulin in blood.
Decreased circulating IgG concentration
MedGen UID:
1720114
Concept ID:
C5234937
Finding
An abnormally decreased level of immunoglobulin G (IgG) in blood.
Skin rash
MedGen UID:
1830322
Concept ID:
C5779628
Sign or Symptom
A red eruption of the skin.
Fever
MedGen UID:
5169
Concept ID:
C0015967
Sign or Symptom
Body temperature elevated above the normal range.

Professional guidelines

PubMed

Lorenzini T, Fliegauf M, Klammer N, Frede N, Proietti M, Bulashevska A, Camacho-Ordonez N, Varjosalo M, Kinnunen M, de Vries E, van der Meer JWM, Ameratunga R, Roifman CM, Schejter YD, Kobbe R, Hautala T, Atschekzei F, Schmidt RE, Schröder C, Stepensky P, Shadur B, Pedroza LA, van der Flier M, Martínez-Gallo M, Gonzalez-Granado LI, Allende LM, Shcherbina A, Kuzmenko N, Zakharova V, Neves JF, Svec P, Fischer U, Ip W, Bartsch O, Barış S, Klein C, Geha R, Chou J, Alosaimi M, Weintraub L, Boztug K, Hirschmugl T, Dos Santos Vilela MM, Holzinger D, Seidl M, Lougaris V, Plebani A, Alsina L, Piquer-Gibert M, Deyà-Martínez A, Slade CA, Aghamohammadi A, Abolhassani H, Hammarström L, Kuismin O, Helminen M, Allen HL, Thaventhiran JE, Freeman AF, Cook M, Bakhtiar S, Christiansen M, Cunningham-Rundles C, Patel NC, Rae W, Niehues T, Brauer N, Syrjänen J, Seppänen MRJ, Burns SO, Tuijnenburg P, Kuijpers TW; NIHR BioResource, Warnatz K, Grimbacher B; NIHR BioResource
J Allergy Clin Immunol 2020 Oct;146(4):901-911. Epub 2020 Apr 9 doi: 10.1016/j.jaci.2019.11.051. PMID: 32278790Free PMC Article
Devanathan AS, Anderson DJC, Cottrell ML, Burgunder EM, Saunders AC, Kashuba ADM
Clin Pharmacol Ther 2019 Jun;105(6):1362-1377. Epub 2019 Mar 25 doi: 10.1002/cpt.1393. PMID: 30739315
Hurst JR, Verma N, Lowe D, Baxendale HE, Jolles S, Kelleher P, Longhurst HJ, Patel SY, Renzoni EA, Sander CR, Avery GR, Babar JL, Buckland MS, Burns S, Egner W, Gompels MM, Gordins P, Haddock JA, Hart SP, Hayman GR, Herriot R, Hoyles RK, Huissoon AP, Jacob J, Nicholson AG, Rassl DM, Sargur RB, Savic S, Seneviratne SL, Sheaff M, Vaitla PM, Walters GI, Whitehouse JL, Wright PA, Condliffe AM
J Allergy Clin Immunol Pract 2017 Jul-Aug;5(4):938-945. Epub 2017 Mar 25 doi: 10.1016/j.jaip.2017.01.021. PMID: 28351785

Recent clinical studies

Etiology

DiGiacomo DV, Roelstraete B, Lebwohl B, Green PHR, Hammarström L, Farmer JR, Khalili H, Ludvigsson JF
Ann Allergy Asthma Immunol 2024 Jun;132(6):752-758.e2. Epub 2024 Feb 6 doi: 10.1016/j.anai.2024.01.019. PMID: 38331244
Reimer-Mcatee M, Ramirez D, Mcatee C, Granillo A, Hasbun R
J Neurol 2023 Aug;270(8):3914-3933. Epub 2023 Apr 28 doi: 10.1007/s00415-023-11735-w. PMID: 37115358Free PMC Article
Ku CL, Chi CY, von Bernuth H, Doffinger R
Hum Genet 2020 Jun;139(6-7):783-794. Epub 2020 May 17 doi: 10.1007/s00439-020-02180-0. PMID: 32419033Free PMC Article
Mäntylä J, Mazur W, Törölä T, Bergman P, Saarinen T, Kauppi P
Respir Med 2019 Jun;152:105-111. Epub 2019 Apr 30 doi: 10.1016/j.rmed.2019.04.022. PMID: 31128603
Vaz A, Pineda-Roman M, Thomas AR, Carlson RW
Hosp Pract (1995) 1998 Sep 15;33(9):105-8, 113-5, 119-20. doi: 10.3810/hp.1998.09.107. PMID: 9750556

Diagnosis

Gupta SK, Berhe M, Crofoot G, Benson P, Ramgopal M, Sims J, McDonald C, Ruane P, Sanchez WE, Scribner A, Liu SY, VanderVeen LA, Dvory-Sobol H, Rhee MS, Baeten JM, Koenig E
Lancet HIV 2023 Jan;10(1):e15-e23. doi: 10.1016/S2352-3018(22)00291-0. PMID: 36566079
Bellanti JA, Settipane RA
Allergy Asthma Proc 2021 Mar 1;42(2):105-107. doi: 10.2500/aap.2021.42.210009. PMID: 33685554Free PMC Article
Heusinkveld LE, Majumdar S, Gao JL, McDermott DH, Murphy PM
J Clin Immunol 2019 Aug;39(6):532-556. Epub 2019 Jul 16 doi: 10.1007/s10875-019-00665-w. PMID: 31313072Free PMC Article
Hurst JR, Verma N, Lowe D, Baxendale HE, Jolles S, Kelleher P, Longhurst HJ, Patel SY, Renzoni EA, Sander CR, Avery GR, Babar JL, Buckland MS, Burns S, Egner W, Gompels MM, Gordins P, Haddock JA, Hart SP, Hayman GR, Herriot R, Hoyles RK, Huissoon AP, Jacob J, Nicholson AG, Rassl DM, Sargur RB, Savic S, Seneviratne SL, Sheaff M, Vaitla PM, Walters GI, Whitehouse JL, Wright PA, Condliffe AM
J Allergy Clin Immunol Pract 2017 Jul-Aug;5(4):938-945. Epub 2017 Mar 25 doi: 10.1016/j.jaip.2017.01.021. PMID: 28351785
Vaz A, Pineda-Roman M, Thomas AR, Carlson RW
Hosp Pract (1995) 1998 Sep 15;33(9):105-8, 113-5, 119-20. doi: 10.3810/hp.1998.09.107. PMID: 9750556

Therapy

Gupta SK, Berhe M, Crofoot G, Benson P, Ramgopal M, Sims J, McDonald C, Ruane P, Sanchez WE, Scribner A, Liu SY, VanderVeen LA, Dvory-Sobol H, Rhee MS, Baeten JM, Koenig E
Lancet HIV 2023 Jan;10(1):e15-e23. doi: 10.1016/S2352-3018(22)00291-0. PMID: 36566079
Bellanti JA, Settipane RA
Allergy Asthma Proc 2021 Mar 1;42(2):105-107. doi: 10.2500/aap.2021.42.210009. PMID: 33685554Free PMC Article
Jarrar D, Song GY
Thorac Surg Clin 2018 Feb;28(1):105-108. doi: 10.1016/j.thorsurg.2017.09.005. PMID: 29150032
Ling Y, Puel A
Actas Dermosifiliogr 2014 Oct;105 Suppl 1:34-40. doi: 10.1016/S0001-7310(14)70016-X. PMID: 25398490
Cristalli G, Costanzi S, Lambertucci C, Lupidi G, Vittori S, Volpini R, Camaioni E
Med Res Rev 2001 Mar;21(2):105-28. doi: 10.1002/1098-1128(200103)21:2<105::aid-med1002>3.0.co;2-u. PMID: 11223861

Prognosis

Oyewusi L, Zeng C, Seung KJ, Mpinda S, Kunda M, Mitnick CD, Kanu M, Tamirat M, Makaka J, Mofolo M, Maime R, Maama L, Senyo N, Oguntoyinbo B, Mayombo L, Franke MF
Glob Health Action 2024 Dec 31;17(1):2305930. Epub 2024 Feb 2 doi: 10.1080/16549716.2024.2305930. PMID: 38305025Free PMC Article
Gupta SK, Berhe M, Crofoot G, Benson P, Ramgopal M, Sims J, McDonald C, Ruane P, Sanchez WE, Scribner A, Liu SY, VanderVeen LA, Dvory-Sobol H, Rhee MS, Baeten JM, Koenig E
Lancet HIV 2023 Jan;10(1):e15-e23. doi: 10.1016/S2352-3018(22)00291-0. PMID: 36566079
Andrews B, Semler MW, Muchemwa L, Kelly P, Lakhi S, Heimburger DC, Mabula C, Bwalya M, Bernard GR
JAMA 2017 Oct 3;318(13):1233-1240. doi: 10.1001/jama.2017.10913. PMID: 28973227Free PMC Article
Baxter C, Moodley D
S Afr Med J 2015 Nov;105(11):948-52. doi: 10.7196/samj.2015.v105i11.10126. PMID: 26937508
Malone M
Histopathology 1991 Aug;19(2):105-19. doi: 10.1111/j.1365-2559.1991.tb00001.x. PMID: 1757066

Clinical prediction guides

Oyewusi L, Zeng C, Seung KJ, Mpinda S, Kunda M, Mitnick CD, Kanu M, Tamirat M, Makaka J, Mofolo M, Maime R, Maama L, Senyo N, Oguntoyinbo B, Mayombo L, Franke MF
Glob Health Action 2024 Dec 31;17(1):2305930. Epub 2024 Feb 2 doi: 10.1080/16549716.2024.2305930. PMID: 38305025Free PMC Article
Petersen RS, Bordone L, Riedl MA, Tachdjian R, Craig TJ, Lumry WR, Manning ME, Bernstein JA, Raasch J, Zuraw BL, Deng Y, Newman KB, Alexander VJ, Lui C, Schneider E, Cohn DM
Allergy 2024 Mar;79(3):724-734. Epub 2023 Nov 27 doi: 10.1111/all.15948. PMID: 38009241
Yimer NB, Mohammed MA, Solomon K, Tadese M, Grutzmacher S, Meikena HK, Alemnew B, Sharew NT, Habtewold TD
Public Health 2021 Jun;195:105-111. Epub 2021 May 31 doi: 10.1016/j.puhe.2021.04.014. PMID: 34082174
Hurst JR, Verma N, Lowe D, Baxendale HE, Jolles S, Kelleher P, Longhurst HJ, Patel SY, Renzoni EA, Sander CR, Avery GR, Babar JL, Buckland MS, Burns S, Egner W, Gompels MM, Gordins P, Haddock JA, Hart SP, Hayman GR, Herriot R, Hoyles RK, Huissoon AP, Jacob J, Nicholson AG, Rassl DM, Sargur RB, Savic S, Seneviratne SL, Sheaff M, Vaitla PM, Walters GI, Whitehouse JL, Wright PA, Condliffe AM
J Allergy Clin Immunol Pract 2017 Jul-Aug;5(4):938-945. Epub 2017 Mar 25 doi: 10.1016/j.jaip.2017.01.021. PMID: 28351785
Ling Y, Puel A
Actas Dermosifiliogr 2014 Oct;105 Suppl 1:34-40. doi: 10.1016/S0001-7310(14)70016-X. PMID: 25398490

Recent systematic reviews

Yimer NB, Mohammed MA, Solomon K, Tadese M, Grutzmacher S, Meikena HK, Alemnew B, Sharew NT, Habtewold TD
Public Health 2021 Jun;195:105-111. Epub 2021 May 31 doi: 10.1016/j.puhe.2021.04.014. PMID: 34082174
Hassan AS, Pybus OG, Sanders EJ, Albert J, Esbjörnsson J
AIDS 2017 Jun 1;31(9):1211-1222. doi: 10.1097/QAD.0000000000001470. PMID: 28353537Free PMC Article
Iroh PA, Mayo H, Nijhawan AE
Am J Public Health 2015 Jul;105(7):e5-16. Epub 2015 May 14 doi: 10.2105/AJPH.2015.302635. PMID: 25973818Free PMC Article
Ha S, Paquette D, Tarasuk J, Dodds J, Gale-Rowe M, Brooks JI, Kim J, Wong T
Can J Public Health 2014 Jan 15;105(1):e53-62. doi: 10.17269/cjph.105.4128. PMID: 24735698Free PMC Article
Plessis SD, Vink M, Joska JA, Koutsilieri E, Stein DJ, Emsley R
AIDS 2014 Mar 27;28(6):803-11. doi: 10.1097/QAD.0000000000000151. PMID: 24300546

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