Multiple thromboembolic events in fetofetal transfusion syndrome in triplets contributing to the understanding of pathogenesis of hydranencephaly in combination with polymicrogyria |
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Authors: | Mittelbronn Michel Beschorner Rudi Schittenhelm Jens Capper David Goeppert Benjamin Meyermann Richard Meyer-Wittkopf Matthias Mackensen-Haen Susanne |
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Affiliation: | Institute of Brain Research, University of Tuebingen, Tuebingen, Germany. michel-guy-andr.mittelbronn@med.uni-tuebingen.de |
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Abstract: | Over the last 180 years, several theories concerning the origin of hydranencephaly have been proposed with an emphasis on infectious, aplastic, and vascular etiologies. In this report, we present a case of triplets with fetofetal transfusion syndrome of which 2 fetuses (1 and 2) developed almost similar hydranencephaly, whereas the third exhibited the features of a fetus papyraceus (3). In the monochorial triamniotic placenta, multiple arteriovenous anastomoses were detected, representing a probable route for the transmission of thrombi originating from fetus 3 causing visceral lesions in fetus 2. Hydranencephaly was histologically characterized by necrosis, macrophage invasion, and endothelial proliferation. In addition, polymicrogyria was seen in fetuses 1 and 2. The combination of multiple visceral thromboembolic events and the death of fetus 3 approximately in the 11th week of gestation suggested a vascular thrombotic pathogenesis of hydranencephaly. Polymicrogyria can be considered as postmigratory laminar necrosis. Our findings contribute to the pathogenetic understanding of combined hydranencephaly and polymicrogyria. |
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Keywords: | Fetofetal transfusion syndrome Hydranencephaly Thromboembolism Polymicrogyria Triplets |
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