Correction of both immunodeficiency and hypoparathyroidism by thymus transplantation in complete DiGeorge syndrome.
clinical research/practice
endocrinology/diabetology
immune deficiency
immunobiology
major histocompatibility complex
thymus/thymic biology
tolerance: mechanisms
translational research/science
Journal
American journal of transplantation : official journal of the American Society of Transplantation and the American Society of Transplant Surgeons
ISSN: 1600-6143
Titre abrégé: Am J Transplant
Pays: United States
ID NLM: 100968638
Informations de publication
Date de publication:
05 2020
05 2020
Historique:
received:
23
08
2019
revised:
06
10
2019
accepted:
16
10
2019
pubmed:
31
10
2019
medline:
22
6
2021
entrez:
31
10
2019
Statut:
ppublish
Résumé
Combined immune deficiency due to athymia in patients with complete DiGeorge syndrome can be corrected by allogeneic thymus transplantation. Hypoparathyroidism is a frequent concomitant clinical problem in these patients, which persists after thymus transplantation. Cotransplantation of allogeneic thymus and parental parathyroid tissue has been attempted but does not achieve durable correction of the patients' hypoparathyroidism due to parathyroid graft rejection. Surprisingly, we observed correction of hypoparathyroidism in one patient after thymus transplantation. Immunohistochemical analysis and fluorescence in situ hybridization confirmed the presence of allogeneic parathyroid tissue in the patient's thymus transplant biopsy. Despite a lack of HLA-matching between thymus donor and recipient, the reconstituted immune system displays tolerance toward the thymus donor. Therefore we expect this patient's hypoparathyroidism to be permanently cured. It is recognised that ectopic parathyroid tissue is not infrequently found in the thymus. If such thymuses could be identified, we propose that their use would offer a compelling approach to achieving lasting correction of both immunodeficiency and hypoparathyroidism.
Identifiants
pubmed: 31663273
doi: 10.1111/ajt.15668
pii: S1600-6135(22)22331-4
doi:
Types de publication
Case Reports
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
1447-1450Informations de copyright
© 2019 The American Society of Transplantation and the American Society of Transplant Surgeons.
Références
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Chinn IK, Markert ML. Induction of tolerance to parental parathyroid grafts using allogeneic thymus tissue in patients with DiGeorge anomaly. J Allergy Clin Immunol. 2011;127(6):1351-1355.
Brandi ML, Bilezikian JP, Shoback D, et al. Management of hypoparathyroidism: summary statement and guidelines. J Clin Endocrinol Metab. 2016;101(6):2273-2283.
US FDA. Natpara (parathyroid hormone) for injection: US prescribing information. 2015. https://www.fda.gov/media/91054/download November 12, 2019.
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