Nonimmune Hydrops Fetalis Diagnosed during the Second Half of Pregnancy in Southern China

Liao, Can; Wei, Jiaxue; Li, Qiuming; Li, Jian; Li, Lixian; Li, Dong-Zhi

doi:10.1159/000100796

Cited by 59 publications

(47 citation statements)

References 31 publications

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“…Thailand is an endemic area for thalassemia [15], and therefore our finding of 21.8% of HF resulting from Hb Bart is similar to that of series from other countries in the same part of the world, such as Taiwan with 31% [16] and Southeast China with 55.1% [17]. These higher numbers may also reflect insufficient patient access to antenatal thalassemia-screening programs in regions where the disease is prevalent.…”

Section: Discussionsupporting

confidence: 85%

Hydrops Fetalis in the Stillborn: A Series from the Central Region of Thailand

Taweevisit

Thorner

2010

Pediatr Dev Pathol

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The purpose of this study was to analyze the cause of hydrops fetalis (HF) among fetal deaths in the central region of Thailand. Autopsy reports diagnosed as HF from 1999 to 2008 at King Chulalongkorn Memorial Hospital were retrieved, and the pathologic findings, clinical information, fetal ultrasonographic studies, and laboratory investigations were reviewed. There were 78 stillborn autopsies during this 10-year period; the mean gestational age was 28 weeks. The causes of fetal hydrops were identified in 88.5%; no cases of immune hydrops were detected. Anemia was the predominant cause of HF (n = 33; 42.2%): related to homozygous α-thalassemia (n = 17; 21.8%), twin-twin transfusion syndrome (n = 8; 10.2%), hemoglobin H (n = 3; 3.8%), lung hemorrhage (n = 1; 1.3%), adrenal hemorrhage (n = 1; 1.3%), and 3 cases of unspecified etiology (3.8%). Other causes of high-output failure included mass lesions resulting in vascular shunting (n = 2; 2.6%) and 1 case each (1.3% each) of maternal diabetes mellitus, intestinal lymphangiectasia, and Beckwith-Wiedemann syndrome. Causes resulting in low-output cardiac failure were congenital heart disease (n = 16; 20.5%) and thoracic space-occupying lesions (n = 7; 9%). The remaining causes included fetal infection (n = 5; 6.4%), congenital abnormalities suggestive of a chromosomal or genetic basis (n = 2; 2.6%), and 1 case (1.3%) of placental vascular thrombosis. Nine cases (11.5%) had no identifiable cause. Thus, the most common cause of HF in this series was homozygous α-thalassemia, reflecting the geographic location of this series.

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Section: Discussionsupporting

confidence: 85%

Hydrops Fetalis in the Stillborn: A Series from the Central Region of Thailand

Taweevisit

Thorner

2010

Pediatr Dev Pathol

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“…They account for the majority of cases of hydrops fetalis in couples in Southern China (1). Although carriers of single a gene deletions most often do not present with any clinical or hematological findings, these deletions are also clinically important in the context of compound heterozygosity with the double-gene deletions (--/-a), which produces Hb H (b4) disease, a condition that might not be as benign as previously thought (2).…”

mentioning

confidence: 99%

Compound Heterozygosity for HKααand anin CisDeletion of DoubleαGenes Presents asα-Thalassemia Trait

et al. 2015

Hemoglobin

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The HKαα (Hong Kongαα) allele is an unusual rearrangement of the α-globin gene cluster containing both the -α(3.7) (rightward) and ααα(anti 4.2) crossover deletion/insertion. During our thalassemia screening program, we identified 10 adult individuals and two newborns who were confirmed to be compound heterozygotes for HKαα and the Southeast Asian deletion (- -(SEA)). Their hematological data showed a typical α-thalassemia (α-thal) trait. The routine gap-polymerase chain reaction (gap-PCR) based assay revealed the presence of -α(3.7), - -(SEA) and normal α2 alleles in the α-globin gene clusters. These confusing findings indicated the existence of more complex derivative alleles produced possibly by repeated unequal crossover of recombinant alleles between α-globin gene clusters. A two-round nested PCR strategy confirmed the diagnosis of HKαα. Considering the large-scale population screening in the thalassemia-prevalent regions in China, the current diagnostic strategy might need to be modified accordingly. The detection of HKαα would improve accuracy in genetic counseling, especially in couples where one partner was a - -(SEA) carrier and the other carries a -α(3.7) deletion identified by routine gap-PCR methods.

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“…Cardiovascular (23.3%), idiopathic (20.1%) causes and genetic disorders (9.3%) are the most common vehicles leading to edema. In northern China, however, the most common NIHF cause is an alphatalesemia, representing more than 50% of the causes of edema [8].…”

Section: Introductionmentioning

confidence: 99%

Troponin T as a marker of postnatal state of newborn with non-immune hydrops fetalis

Sobczuk¹,

Jakubowski²,

Draga-Zapora³

et al. 2017

Clin Obstet Gynecol Reprod Med

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Aim of the study: The aim of the study is to analyze correlation between level of troponin T in fetal blood with NIHF and the course of pregnancy.Material and method: The analysis of troponin T level in fetuses blood was conducted in 22 cases of cordocentesis carried out at the Department of Gynecology, Fertility and Fetal Therapy between 2014-2016. There were 15 cordocentesis performed in cases of NIHF undergoing intrauterine therapy and 7 cordocentesis performed in cases of other pathology that have been assigned to the control group.Results: In the study group there were 9 results obtained over a standard 0.308 ng/ml and 6 results below this value. In the control group 5 results were normal and 2 were above norm. In the group of fetuses with NIHF, 89% of cases with elevated levels of troponin T were unsuccessful, while in the group with normal levels 83% of the cases had a successful course. In the control group 100% of normal levels of troponin T had a successful course, and there were 2 cases of elevated levels of troponin T in 1 newborn rated at 3 points in the Apgar score because of respiratory disorders. Conclusions:Based on the analysis, it appears that the level of troponin T in fetal blood can be a marker providing information on the prognosis and, therefore, the level of cTnT in fetal blood should be measured to determine the degree of heart failure in conjunction with the echocardiography study.

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Nonimmune Hydrops Fetalis Diagnosed during the Second Half of Pregnancy in Southern China

Cited by 59 publications

References 31 publications

Hydrops Fetalis in the Stillborn: A Series from the Central Region of Thailand

Hydrops Fetalis in the Stillborn: A Series from the Central Region of Thailand

Compound Heterozygosity for HKααand anin CisDeletion of DoubleαGenes Presents asα-Thalassemia Trait

Troponin T as a marker of postnatal state of newborn with non-immune hydrops fetalis

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