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Case Report

One-Sided Chronic Intervillositis of Unknown Etiology in Dizygotic Twins: A Description of 3 Cases

1
Department of Pathology, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands
2
Department of Pathology, University Medical Center Utrecht, 3584 CX Utrecht, The Netherlands
3
Department of Immunology, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands
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Department of Obstetrics, Birth Center Wilhelmina’s Children Hospital, University Medical Center Utrecht, 3584 CX Utrecht, The Netherlands
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Elizabeth Garrett Anderson Institute for Women’s Health, University College London, London WC1E 6DB, UK
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Department of Gynecology and Obstetrics, Leiden University Medical Center, 2333 ZA Leiden, The Netherlands
*
Author to whom correspondence should be addressed.
Academic Editor: Martin Gauster
Int. J. Mol. Sci. 2021, 22(9), 4786; https://doi.org/10.3390/ijms22094786
Received: 4 April 2021 / Revised: 23 April 2021 / Accepted: 27 April 2021 / Published: 30 April 2021
(This article belongs to the Special Issue Molecular Aspects of Inflammation in Pregnancy 2.0)
Chronic intervillositis of unknown etiology (CIUE) is a rare, poorly understood, histopathological diagnosis of the placenta that is frequently accompanied by adverse pregnancy outcomes including miscarriage, fetal growth restriction, and intrauterine fetal death. CIUE is thought to have an immunologically driven pathophysiology and may be related to human leukocyte antigen mismatches between the mother and the fetus. Dizygotic twins with one-sided CIUE provide an interesting context to study the influence of immunogenetic differences in such cases. The main immune-cell subsets were investigated using immunohistochemistry. We identified three dizygotic twin pregnancies in which CIUE was present in only one of the two placentas. Two of the pregnancies ended in term delivery and one ended in preterm delivery. Presence of CIUE was correlated with lower placental weight and lower birthweight. Relative number of CD68, CD56, CD20, and CD3 positive cells were comparable between co-twins. The presence of one-sided CIUE in dizygotic twin pregnancy was associated with selective growth restriction in the affected twin. This suggests a unique fetal immunogenetic contribution to the pathogenesis of CIUE. Further study of dizygotic and monozygotic placentas affected by CIUE could identify new insights into its pathophysiology and into the field of reproductive immunology. View Full-Text
Keywords: placenta; chronic intervillositis of unknown etiology; twin placenta; chronic intervillositis of unknown etiology; twin
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MDPI and ACS Style

van der Meeren, L.E.; Krop, J.; Dijkstra, K.L.; Bloemenkamp, K.W.M.; Cornish, E.F.; Nikkels, P.G.J.; van der Hoorn, M.-L.P.; Bos, M. One-Sided Chronic Intervillositis of Unknown Etiology in Dizygotic Twins: A Description of 3 Cases. Int. J. Mol. Sci. 2021, 22, 4786. https://doi.org/10.3390/ijms22094786

AMA Style

van der Meeren LE, Krop J, Dijkstra KL, Bloemenkamp KWM, Cornish EF, Nikkels PGJ, van der Hoorn M-LP, Bos M. One-Sided Chronic Intervillositis of Unknown Etiology in Dizygotic Twins: A Description of 3 Cases. International Journal of Molecular Sciences. 2021; 22(9):4786. https://doi.org/10.3390/ijms22094786

Chicago/Turabian Style

van der Meeren, Lotte E., Juliette Krop, Kyra L. Dijkstra, Kitty W.M. Bloemenkamp, Emily F. Cornish, Peter G.J. Nikkels, Marie-Louise P. van der Hoorn, and Manon Bos. 2021. "One-Sided Chronic Intervillositis of Unknown Etiology in Dizygotic Twins: A Description of 3 Cases" International Journal of Molecular Sciences 22, no. 9: 4786. https://doi.org/10.3390/ijms22094786

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