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      A Case of Alloimmune Thrombocytopenia, Hemorrhagic Anemia-Induced Fetal Hydrops, Maternal Mirror Syndrome, and Human Chorionic Gonadotropin–Induced Thyrotoxicosis

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          Abstract

          Fetal/neonatal alloimmune thrombocytopenia (FNAIT) can be a cause of severe fetal thrombocytopenia, with the common presentation being intracranial hemorrhage in the fetus, usually in the third trimester. A very unusual case of fetal anemia progressed to hydrops. This was further complicated by maternal Mirror syndrome and human chorionic gonadotropin–induced thyrotoxicosis. Without knowledge of etiology, and possibly due to associated cardiac dysfunction, fetal transfusion resulted in fetal demise. Subsequent testing revealed FNAIT as the cause of severe hemorrhagic anemia. In cases with fetal anemia without presence of red blood cell antibodies, FNAIT must be ruled out as a cause prior to performing fetal transfusion. Fetal heart may adapt differently to acute hemorrhagic anemia compared with a more subacute hemolytic anemia.

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          Noninvasive diagnosis by Doppler ultrasonography of fetal anemia due to maternal red-cell alloimmunization. Collaborative Group for Doppler Assessment of the Blood Velocity in Anemic Fetuses.

          Meredith Berry, F. Rahman, R. Vazquez Gomez … (2000)
          Invasive techniques such as amniocentesis and cordocentesis are used for diagnosis and treatment in fetuses at risk for anemia due to maternal red-cell alloimmunization. The purpose of our study was to determine the value of noninvasive measurements of the velocity of blood flow in the fetal middle cerebral artery for the diagnosis of fetal anemia. We measured the hemoglobin concentration in blood obtained by cordocentesis and also the peak velocity of systolic blood flow in the middle cerebral artery in 111 fetuses at risk for anemia due to maternal red-cell alloimmunization. Peak systolic velocity was measured by Doppler velocimetry. To identify the fetuses with anemia, the hemoglobin values of those at risk were compared with the values in 265 normal fetuses. Fetal hemoglobin concentrations increased with increasing gestational age in the 265 normal fetuses. Among the 111 fetuses at risk for anemia, 41 fetuses did not have anemia; 35 had mild anemia; 4 had moderate anemia; and 31, including 12 with hydrops, had severe anemia. The sensitivity of an increased peak velocity of systolic blood flow in the middle cerebral artery for the prediction of moderate or severe anemia was 100 percent either in the presence or in the absence of hydrops (95 percent confidence interval, 86 to 100 percent for the 23 fetuses without hydrops), with a false positive rate of 12 percent. In fetuses without hydrops that are at risk because of maternal red-cell alloimmunization, moderate and severe anemia can be detected noninvasively by Doppler ultrasonography on the basis of an increase in the peak velocity of systolic blood flow in the middle cerebral artery.
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            Serum human chorionic gonadotropin concentrations greater than 400,000 IU/L are invariably associated with suppressed serum thyrotropin concentrations.

            Christopher Lockwood, Ann M. Gronowski, David Grenache (2009)
            During pregnancy, when human chorionic gonadotropin (hCG) concentrations are highest, there is a transient suppression of serum thyrotropin (TSH). In normal pregnancy, TSH concentrations generally remain within nonpregnant reference intervals; however, in some patients TSH is suppressed. Here we sought to extend previous studies to examine the relationship between very high serum concentrations of hCG (>200,000 IU/L) and the thyroid hormones TSH and free thyroxine (FT(4)). The objective of this study was to determine: 1) if there is an hCG concentration above which TSH concentrations are suppressed ( 200,000 IU/L were identified, and TSH and FT(4) concentrations were measured. Medical records were reviewed for clinical information. Thirty-seven percent of subjects had hyperemesis gravidarum (HG) and 19% had gestational trophoblastic disease (GTD). TSH was suppressed ( 200,000 IU/L and 100% of specimens with hCG concentrations >400,000 IU/L. FT(4) concentrations were elevated above the reference interval (1.8 ng/dL) in 32% of specimens with hCG concentrations >200,000 IU/L and in 80% of specimens with hCG concentrations >400,000 IU/L. Only four subjects had documented signs of hyperthyroidism. Women with GTD had a median hCG concentration twofold higher than women with HG and a median TSH concentration one half that of women with HG. 1) At hCG concentrations >400,000 IU/L, TSH is consistently suppressed; 2) serum FT(4) and TSH respond to changes in serum hCG concentrations; and 3) most patients with hCG concentrations >200,000 IU/L lack overt hyperthyroid symptoms.
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              Intracranial hemorrhage in alloimmune thrombocytopenia: stratified management to prevent recurrence in the subsequent affected fetus.

              James Bussel, Richard L Berkowitz, Crystal Hung … (2010)
              We sought to prevent intracranial hemorrhage (ICH) through antenatal management of alloimmune thrombocytopenia. A total of 33 women (37 pregnancies) with alloimmune thrombocytopenia and ICH in a previous child were stratified according to the timing of the previous child's ICH: extremely high risk (HR) (n = 8) had ICH 100,000/mL, and 1 was grade I. The other 2 ICHs were unequivocal treatment failures; both were grade III-IV and resulted in fetal demise. These findings demonstrate the success of stratified treatment in these HR patients, which tailored interventions according to the timing of the sibling's ICH. Copyright (c) 2010 Mosby, Inc. All rights reserved.
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                Author and article information

                Journal
                Journal ID (nlm-ta): AJP Rep
                Journal ID (iso-abbrev): AJP Rep
                Title: AJP Reports
                Publisher: Thieme Medical Publishers (333 Seventh Avenue, New York, NY 10001, USA. )
                ISSN (Print): 2157-6998
                ISSN (Electronic): 2157-7005
                Publication date (Electronic): 25 January 2013
                Publication date (Print): May 2013
                Volume: 3
                Issue: 1
                Pages: 41-44
                Affiliations
                [1 ]Division of Maternal-Fetal Medicine Royal Alexandra Hospital, University of Alberta, Edmonton, Alberta, Canada
                [2 ]Department of Pathology and Laboratory Medicine, Royal Alexandra Hospital, University of Alberta, Edmonton, Alberta, Canada
                [3 ]Division of Pediatric Cardiology/Fetal and Neonatal Cardiology Program, Royal Alexandra Hospital, University of Alberta, Edmonton, Alberta, Canada
                Author notes
                Address for correspondence Venu Jain, MD, PhD Assistant Professor, 5S131 Lois Hole Hospital, Royal Alexandra Hospital 10240 Kingsway Avenue, Edmonton, Alberta T5H 3V9Canada venu.jain@ 123456albertahealthservices.ca
                Article
                Publisher ID: 03041
                DOI: 10.1055/s-0032-1331379
                PMC ID: 3699162
                PubMed ID: 23943709
                SO-VID: eb9151ba-d7b2-4ca1-a6fe-8975c2f4ec3f
                Copyright © © Thieme Medical Publishers
                History
                Date received : 08 August 2012
                Date accepted : 11 August 2012
                Categories
                Subject: Article

                Keywords: fnait,nait,antiplatelet antibodies,hpa 1a,intrauterine transfusion,intraperitoneal transfusion
                Data availability:
                Keywords: fnait, nait, antiplatelet antibodies, hpa 1a, intrauterine transfusion, intraperitoneal transfusion

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