Home LiteratureArticle Details
PMID: 331324 Published · ppublish English Case Reports Journal Article Research Support, U.S. Gov't, P.H.S.

Rationale for combined use of fetal liver and thymus for immunological reconstitution in patients with variants of severe combined immunodeficiency.

Pahwa R, Pahwa S, Good RA, Incefy GS, O'Reilly RJ

Abstract

Bone marrow cells from a patient with severe combined immunodeficiency were studied in vitro for thymus-dependent lymphocyte (T cell) differentiation by using, at varying times, thymic epithelial monolayers and culture supernatants, thymopoietin, ubiquitin, and thymic extract as inducing agents. On initial evaluation, with thymopoietin or human thymic extract, only a partial differentiation of marrow cells was achieved into cells bearing the human T cell antigenicity without the capacity to form rosettes with sheep erythrocytes, suggesting that the stem cells were defective. Two fetal liver transplantations aimed at reconstitution were unsuccessful, despite evidence of chimerism. Induction studies at that time demonstrated rosetting capacity (with sheep erythrocytes) of the patient's bone marrow cells after coculture with thymic epithelial monolayers but not with their supernatants. An 18-week fetal thymus (irradiated) was then transplanted, but the transplantation was unsuccessful and no clear evidence of chimerism was demonstrated. Subsequently, transplantation of another fetal liver resulted in chimerism and immunologic reconstitution. Serum thymic factor activity rose from 1:2 before transplantation to 1:16 after reconstitution. The combined use of fetal thymus and liver may provide effective immunological reconstitution in some variants of severe combined immunodeficiency.

MeSH Terms
Bone Marrow/immunology Bone Marrow Cells Chimera HLA Antigens/analysis Humans Immunologic Deficiency Syndromes/immunology,therapy Infant Liver/embryology Liver Transplantation Male T-Lymphocytes/immunology Thymus Gland/embryology,transplantation Transplantation, Homologous
Chemicals
HLA Antigens
Authors & Affiliations
5 authors, click to expand affiliations / ORCID
Pahwa R
Pahwa S
Good R A
Incefy G S
O'Reilly R J
References (36)
36 references, click to expand
  1. ONTOGENY AND PHYLOGENY OF ADAPTIVE IMMUNITY.
    Adv Immunol. 1964;27:1-115 PMID: 14333018
  2. Immunological function of the thymus.
    Lancet. 1961 Sep 30;2(7205):748-9 PMID: 14474038
  3. THE LYMPHOD TISSUES AND IMMUNE RESPONSES OF NEONATALLY THYMECTOMIZED MICE BEARING THYMUS TISSUE IN MILLIPORE DIFFUSION CHAMBERS.
    J Exp Med. 1964 Jan 1;119:177-94 PMID: 14113112
  4. The role of the thymus in development of immunologic capacity in rabbits and mice.
    J Exp Med. 1962 Nov 1;116:773-96 PMID: 13949053
  5. Strain variation in mortality and runt disease in mice thymectomized at birth.
    Transplant Bull. 1962 Apr;29:102-4 PMID: 14484061
  6. Role of the thymus in immune ractions in rats. I. The immunologic response to bovine serum albumin (antibody formation, Arthus reactivity, and delayed hypersensitivity) in rats thymectomized or splenectomized at various times after birth.
    J Exp Med. 1962 Aug 1;116:159-76 PMID: 14451146
  7. Estimation of radiation effects at small exposures.
    Fed Proc. 1961 Jul;20(Suppl 8):26-8 PMID: 13790530
  8. Perclusion of secondary phase of irradiation syndrome by inoculation of fetal hematopoietic tissue following lethal total-body x-irradiation.
    J Natl Cancer Inst. 1958 Mar;20(3):625-32 PMID: 13539613
  9. Protein measurement with the Folin phenol reagent.
    J Biol Chem. 1951 Nov;193(1):265-75 PMID: 14907713
  10. Thymic activity in severe combined immunodeficiency diseases.
    Proc Natl Acad Sci U S A. 1977 Mar;74(3):1250-3 PMID: 322151
  11. Reconstitution of B and T lymphocyte function in severe combined immunodeficiency disease after transplantation with thymic epithelium.
    Lancet. 1976 Dec 11;2(7998):1270-2 PMID: 63746
  12. Long survival and immunologic reconstitution following transplantation with syngeneic or allogeneic fetal liver and neonatal spleen cells.
    Transplant Proc. 1976 Dec;8(4):521-5 PMID: 11582
  13. Leukocyte migration inhibitory factor (LMIF) profile in primary and secondary immunodeficiency disease.
    Clin Exp Immunol. 1976 Dec;26(3):505-10 PMID: 795576
  14. Heterogeneity of stem cells in severe combined immunodeficiency.
    Clin Exp Immunol. 1976 Sep;25(3):462-71 PMID: 786520
  15. Morphological and functional maturation of human thymic epithelium in culture.
    Nature. 1974 Oct 4;251(5474):421-3 PMID: 4138493
  16. Bone marrow transplantation. Therapeutic usefulness and complications.
    Am J Med. 1971 Oct;51(4):513-32 PMID: 4265085
  17. Isolation of bovine thymin: a polypeptide hormone of the thymus.
    Nature. 1974 Jan 4;247(5435):11-4 PMID: 4128781
  18. Primary immunodeficiencies. Report of a World Health Organization Committee.
    Pediatrics. 1971 May;47(5):927-46 PMID: 4102352
  19. Immunological restitution in lymphopenic immunological deficiency syndrome.
    Lancet. 1968 Mar 9;1(7541):503-6 PMID: 4171200
  20. Thymus: an essential factor in lymphoid repopulation.
    Transplant Proc. 1969 Mar;1(1):614-5 PMID: 4400068
  21. Grafting of fetal thymus and hematopoietic tissue in infants with immune deficiency syndromes.
    Transplantation. 1973 May;15(5):427-34 PMID: 4145548
  22. Thymus transplantation. Permanent reconstitution of cellular immunity in a patient with sex-linked combined immunodeficiency.
    N Engl J Med. 1973 Jul 5;289(1):5-9 PMID: 4145369
  23. Leukocyte migration inhibitory factor (LMIF) induced by concanavalin A: standardized microassay for production in vitro.
    Proc Natl Acad Sci U S A. 1975 Aug;72(8):3197-200 PMID: 1103141
  24. Isolation of a polypeptide that has lymphocyte-differentiating properties and is probably represented universally in living cells.
    Proc Natl Acad Sci U S A. 1975 Jan;72(1):11-5 PMID: 1078892
  25. Successful fetal liver transplantation in a child with severe combined immunodeficiency.
    Lancet. 1975 Nov 1;2(7940):850-3 PMID: 53333
  26. Immunodeficiency in developmental perspective.
    Harvey Lect. 1973;67:1-107 PMID: 4581934
  27. Enhancement of T-lymphocyte differentiation in vitro by thymic extracts after bone marrow transplantation in severe combined immunodeficiencies.
    Clin Immunol Immunopathol. 1975 Jul;4(2):258-68 PMID: 49234
  28. Correction of severe combined immunodeficiency by fetal liver cells.
    N Engl J Med. 1976 May 13;294(20):1076-81 PMID: 3737
  29. Protection of lethally irradiated mice with allogeneic fetal liver cells: influence of irradiation dose on immunologic reconstitution.
    Proc Natl Acad Sci U S A. 1975 Oct;72(10):4100-4 PMID: 673
  30. Human lymphocyte-sheep erythrocyte rosette formation: some characteristics of the interaction.
    Clin Immunol Immunopathol. 1973 Jul;1(4):511-22 PMID: 4549205
  31. Purification and properties of bovine thymosin.
    Ann N Y Acad Sci. 1975 Feb 28;249:125-44 PMID: 236717
  32. The thymus and the cellular basis of immunity.
    Transplant Rev. 1969;1:43-91 PMID: 4945476
  33. Experimental studies on the development of the thymus.
    J Exp Med. 1967 Oct 1;126(4):715-26 PMID: 4861748
  34. Demonstration of an intrathymic defect in a case of severe combined immunodeficiency disease.
    N Engl J Med. 1975 Aug 28;293(9):424-8 PMID: 1097926
  35. In vitro differentiation of human marrow cells into T lymphocytes by thymic extracts using the rosette technique.
    Clin Exp Immunol. 1975 Mar;19(3):475-83 PMID: 1081929
  36. Hereditary sever combined immunodeficiency and adenosine deaminase deficiency.
    Pediatr Res. 1976 Jan;10(1):67-70 PMID: 1246464
Article Info
Journal
Proceedings of the National Academy of Sciences of the United States of America
Abbr.
Proc Natl Acad Sci U S A
ISSN
0027-8424
Published
1977-07-00
Pages
3002-5
Language
English
Region
United States
NLM ID
7505876
PMCID
PMC431383
Subset
IM
Analysis Services
Analysis Services

Contact

No. 2 Wenbo Road, Zhangqiu District, Jinan, Shandong

Qilu Normal University · Genelibs Bioinformatics Lab

750 Shunhua Rd, Jinan

2F, Bldg F, University Science Park

Tel: 0531-88819269

WeChat Official Account

Follow our WeChat subscription account for real-time updates and the latest in medical and biological research.


Business Email

E-mail: [email protected]