2014
DOI: 10.2350/14-04-1476-cr.1
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Trisomy 21–Associated Transient Abnormal Myelopoiesis Involving the Maternal Space of the Placenta: A Case Report and Literature Review

Abstract: We report a case of a male newborn with trisomy 21 and transient abnormal myelopoiesis at birth whose placenta showed extravasated fetal blasts in the perivillous (maternal) space. Concern for possible maternal spread of fetal malignancy prompted a Kleihauer-Betke test and flow cytometric analysis of the maternal peripheral blood on postpartum day 2. Notably, no evidence of the persistence of fetal cells in the maternal blood was identified, a finding that likely reflected successful maternal immunologic clear… Show more

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Cited by 10 publications
(9 citation statements)
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“…As stated in the literature and encountered in our first case, poor prognostic factors in fetal TAM are hepatosplenomegaly, hypoechoic hepatomegaly and non‐immune fetal hydrops. In addition, the presence of fetal abnormal myeloblasts in the maternal space of the placenta in the first case may also represent a poor prognostic factor for the neonate. This finding is likely to reflect the higher burden of circulating immature cells, as confirmed by maternal flow cytometry, and therefore a more aggressive fetal disease.…”
supporting
confidence: 69%
“…As stated in the literature and encountered in our first case, poor prognostic factors in fetal TAM are hepatosplenomegaly, hypoechoic hepatomegaly and non‐immune fetal hydrops. In addition, the presence of fetal abnormal myeloblasts in the maternal space of the placenta in the first case may also represent a poor prognostic factor for the neonate. This finding is likely to reflect the higher burden of circulating immature cells, as confirmed by maternal flow cytometry, and therefore a more aggressive fetal disease.…”
supporting
confidence: 69%
“…Although permission to perform a fetal autopsy may not be granted, TAM can be diagnosed by the presence of abnormal cells within umbilical and chorionic vessels [2,21,22]. In some cases, proliferating blast cells of fetal origin invade the vascular wall of the placenta and exist in the maternal space [2,21,23]. The presence of fetal blast cells in the maternal space is associated with a 90% rate of stillbirth or death in early life [21].…”
Section: Discussionmentioning
confidence: 99%
“…In some cases, proliferating blast cells of fetal origin invade the vascular wall of the placenta and exist in the maternal space [2,21,23]. The presence of fetal blast cells in the maternal space is associated with a 90% rate of stillbirth or death in early life [21]. In stillborn cases of TAM in Down syndrome, hydrops fetalis, periventricular leukomalacia, or maternal involvement of fetal blast cells, as well as liver fibrosis, as seen in the present case, may be the cause of intrauterine fetal death.…”
Section: Discussionmentioning
confidence: 99%
“…A complete histopathologic evaluation of the placenta is emphasized, as most notable findings are observed microscopically. Leukocytosis with left-shifted hematopoietic elements and myeloid blasts can be seen within vessels of the umbilical cord, chorionic plate, and chorionic villi 12,16,17,[19][20][21][22] (Figure 2, A through D). These mononuclear blasts are characterized by large, lobulated nuclei with open chromatin, high nuclear to cytoplasmic ratio, prominent nucleoli, and associated apoptosis (Figure 2, E).…”
Section: Gross Pathology and Histopathologymentioning
confidence: 99%