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DOI 10.48095/cccg202344

Медицинская библиотека · Ceska gynekologie · 2023

Cryopreservation of ovarian tissue as a method for fertility preservation in women.

Chen Yin-Huai, Grigelioniene Giedre, Newton Phillip T, Gullander Jacob, Elfving Maria, Hammarsjö Anna, Batkovskyte Dominyka, Alsaif Hessa S, Kurdi Wesam I Y, Abdulwahab Firdous, Shanmugasundaram Veerabahu, Devey Luke, Bacrot Séverine, Brodszki Jana, Huber Celine, Hamel Ben, Gisselsson David, Papadogiannakis Nikos, Jedrycha Katarina, Gürtl-Lackner Barbara, Chagin Andrei S, Nishimura Gen, Aschenbrenner Dominik, Alkuraya Fowzan S, Laurence Arian, Cormier-Daire Valérie, Uhlig Holm H

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Аннотация

The gene IL6ST encodes GP130, the common signal transducer of the IL-6 cytokine family consisting of 10 cytokines. Previous studies have identified cytokine-selective IL6ST defects that preserve LIF signaling. We describe three unrelated families with at least five affected individuals who presented with lethal Stüve-Wiedemann-like syndrome characterized by skeletal dysplasia and neonatal lung dysfunction with additional features such as congenital thrombocytopenia, eczematoid dermatitis, renal abnormalities, and defective acute-phase response. We identified essential loss-of-function variants in IL6ST (a homozygous nonsense variant and a homozygous intronic splice variant with exon skipping). Functional tests showed absent cellular responses to GP130-dependent cytokines including IL-6, IL-11, IL-27, oncostatin M (OSM), and leukemia inhibitory factor (LIF). Genetic reconstitution of GP130 by lentiviral transduction in patient-derived cells reversed the signaling defect. This study identifies a new genetic syndrome caused by the complete lack of signaling of a whole family of GP130-dependent cytokines in humans and highlights the importance of the LIF signaling pathway in pre- and perinatal development.

Для цитирования: Chen Yin-Huai, Grigelioniene Giedre, Newton Phillip T, Gullander Jacob, Elfving Maria, Hammarsjö Anna, Batkovskyte Dominyka, Alsaif Hessa S, Kurdi Wesam I Y, Abdulwahab Firdous, Shanmugasundaram Veerabahu, Devey Luke, Bacrot Séverine, Brodszki Jana, Huber Celine, Hamel Ben, Gisselsson David, Papadogiannakis Nikos, Jedrycha Katarina, Gürtl-Lackner Barbara, Chagin Andrei S, Nishimura Gen, Aschenbrenner Dominik, Alkuraya Fowzan S, Laurence Arian, Cormier-Daire Valérie, Uhlig Holm H. Cryopreservation of ovarian tissue as a method for fertility preservation in women.. Ceska gynekologie. 2023. DOI: 10.48095/cccg202344.

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