Autoimmune polyglandular syndrome type 1 and pregnancy: Clinical types

Logutova, L. S.; Petrukhin, V. A.; Burumkulova, F. F.; Shidlovskaya, N. V.; Barinova, I V; Башакин, Н. Ф.

doi:10.17116/rosakush201515359-64

Cited by 2 publications

(3 citation statements)

References 3 publications

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“…A Russian case study described two pregnant APS-1 patients on appropriate hormone replacement therapy who suffered threatened miscarriages in the first trimester, fetoplacental insufficiency, and fetal growth restriction. 74 Several APS-1 patients had placentas with diffuse lymphocytic infiltration of the decidual plate, suggestive of autoimmunity. 75 Unlike other endocrine organs, whose function can be replaced by hormone therapy, the placenta has both a hormonal and a critical structural function: providing nutrients to the embryo.…”

Section: Aire Mutati On S and Infertilit Ymentioning

confidence: 99%

“…Perhaps APS‐1 patients with severe symptomatology, including ovarian insufficiency necessitating oocyte donation, are more likely to suffer pregnancy complications. A Russian case study described two pregnant APS‐1 patients on appropriate hormone replacement therapy who suffered threatened miscarriages in the first trimester, fetoplacental insufficiency, and fetal growth restriction 74 . Several APS‐1 patients had placentas with diffuse lymphocytic infiltration of the decidual plate, suggestive of autoimmunity 75 .…”

Section: Aire Mutations and Infertilitymentioning

confidence: 99%

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Thymic and extrathymic Aire‐expressing cells in maternal‐fetal tolerance*

Gillis-Buck

MacKenzie

Gardner

2022

Immunological Reviews

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Healthy pregnancy requires maternal immune tolerance to both fetal and placental tissues which contain a range of self‐ and non‐self‐antigens. While many of the components and mechanisms of maternal‐fetal tolerance have been investigated in detail and previously and thoroughly reviewed (Erlebacher A. Annu Rev Immunol. 2013;31:387–411), the role of autoimmune regulator (Aire), a critical regulator of central tolerance expressed by medullary thymic epithelial cells (mTECs), has been less explored. Aire is known to facilitate the expression of a range of otherwise tissue‐specific antigens (TSAs) in mTECs, and here we highlight recent work showing a role for mTEC‐mediated thymic selection in maintaining maternal‐fetal tolerance. Recently, however, our group and others have identified additional populations of extrathymic Aire‐expressing cells (eTACs) in the secondary lymphoid organs. These hematopoietic antigen‐presenting cells possess the ability to induce functional inactivation and/or deletion of cognate T cells, and deletion of maternal eTACs during pregnancy increases T‐cell activation in the lymph nodes and lymphocytic infiltration of the uterus, leading to pregnancy complications including intrauterine growth restriction (IUGR) and fetal resorption. In this review, we briefly summarize findings related to essential Aire biology, discuss the known roles of Aire‐deficiency related to pregnancy complications and infertility, review the newly discovered role for eTACs in the maintenance of maternal‐fetal tolerance—as well as recent work defining eTACs at the single‐cell level—and postulate potential mechanisms by which eTACs may regulate this process.

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Section: Aire Mutati On S and Infertilit Ymentioning

confidence: 99%

Section: Aire Mutations and Infertilitymentioning

confidence: 99%

Thymic and extrathymic Aire‐expressing cells in maternal‐fetal tolerance*

Gillis-Buck

MacKenzie

Gardner

2022

Immunological Reviews

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“…Roles for Aire in decidualization and embryo implantation have also been proposed (10)(11)(12). Although patients with AIRE mutations on appropriate hormone replacement therapy can achieve pregnancy, these are nonetheless complicated by fetoplacental insufficiency, IUGR, and placentas with diffuse lymphocytic infiltration of the decidual plate, suggestive of placental autoimmunity (13,14). However, whether Aire-expressing cells are functionally involved in maternal-fetal tolerance has not been investigated.…”

Section: Introductionmentioning

confidence: 99%

Extrathymic Aire -expressing cells support maternal-fetal tolerance

et al. 2021

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Healthy pregnancy requires tolerance to fetal alloantigens as well as syngeneic embryonic and placental antigens. Given the importance of the autoimmune regulator (Aire) gene in self-tolerance, we investigated the role of Aire-expressing cells in maternal-fetal tolerance. We report that maternal ablation of Aire-expressing (Aire+) cells during early mouse pregnancy caused intrauterine growth restriction (IUGR) in both allogeneic and syngeneic pregnancies. This phenotype is immune mediated, as IUGR was rescued in Rag1-deficient mice, and involved a memory response, demonstrated by recurrence of severe IUGR in second pregnancies. Single-cell RNA sequencing demonstrated that Aire+ cell depletion in pregnancy results in expansion of activated T cells, particularly T follicular helper cells. Unexpectedly, selective ablation of either Aire-expressing medullary thymic epithelial cells or extrathymic Aire-expressing cells (eTACs) mapped the IUGR phenotype exclusively to eTACs. Thus, we report a previously undescribed mechanism for the maintenance of maternal-fetal immune homeostasis and demonstrate that eTACs protect the conceptus from immune-mediated IUGR.

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Autoimmune polyglandular syndrome type 1 and pregnancy: Clinical types

Cited by 2 publications

References 3 publications

Thymic and extrathymic Aire‐expressing cells in maternal‐fetal tolerance*

Thymic and extrathymic Aire‐expressing cells in maternal‐fetal tolerance*

Extrathymic Aire -expressing cells support maternal-fetal tolerance

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