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At least 19 recordsLinked to original sources
PubMed2004

Hemophagocytic lymphohistiocytosis presenting with nonimmune hydrops fetalis.

Hydrops fetalis is a condition of diverse etiologies. An association between hemophagocytic lymphohistiocytosis (HLH) and hydrops fetalis has not been reported in the English literature. We describe an intrauterine case of HLH in an infant who presented with hydrops fetalis at 32 weeks gestation. We suggest that HLH should be considered in the differential diagnosis of nonimmune hydrops fetalis, especially in the presence of cytopenias.

Adult↗
PubMed1998

Etiology and outcome of hydrops fetalis.

Hydrops fetalis is a morbid condition caused by a wide variety of diseases. Although treatment for some underlying pathologies is becoming available, the prognosis remains poor. To analyze the etiology and outcome of hydrops fetalis, we reviewed the records of 79 fetuses encountered at the National Taiwan University Hospital between January 1993 and October 1996. The mean +/- standard deviation gestational age at presentation was 24.9 +/- 6.3 weeks. One case was due to RhE incompatibility. Seventy-eight fetuses were diagnosed as having nonimmune hydrops fetalis; this was idiopathic in 20 fetuses. In the other 58, the causes and associated conditions were classified into eight groups: hematologic (25), cardiovascular (15), chromosomal (5), gastrointestinal (4), cystic hygroma (3), pulmonary (2), infection (2), and miscellaneous (3). One fetus with cystic hygroma also had a chromosomal abnormality (Turner's syndrome). The most common cause of nonimmune hydrops fetalis was homozygous alpha-thalassemia (31%). Twenty-nine fetuses were lost to follow-up after initial evaluation and 50 fetuses were delivered at our hospital. Of these, there were eight cases of antepartum death, 24 cases of intrapartum death, and 18 live births. Seventeen of the live-born infants were treated at our neonatal intensive care unit, of whom only seven survived.

Adult↗
PubMed1989

Chylothorax in Down's syndrome associated with hydrops fetalis.

Hydrops fetalis is an unusual manifestation of Down's syndrome and diagnosis of Down's syndrome in the hydropic newborn can be difficult as the clinical signs are masked by severe oedema. The baby may also be seriously ill and die soon after birth, before any investigative procedures could be done. Down's syndrome should be considered in foetuses or babies presented with pleural effusion and/or ascites.

Chylothorax↗
PubMed1999

Nonimmune hydrops fetalis.

Hydrops fetalis is a relatively rare phenomenon that presents itself in the delivery room in an extremely acute manner. Prompt resuscitation, an understanding of the condition and its presentation, and immediate treatment can make the difference between life and death in these cases. The focus of this article is on nonimmune hydrops, the type of hydrops seen in the clinical setting. Maternal and fetal pathophysiology, current theories, diagnostic evaluations, neonatal pathophysiology, clinical manifestations, treatment options, pertinent research, and the needs of the family are discussed.

Delivery, Obstetric↗
PubMed1995

Pathophysiology of hydrops fetalis.

Hydrops fetalis occurs when the rate of interstitial fluid production by capillary ultrafiltration exceeds the rate of interstitial fluid return to the circulation via lymphatic vessels. Developmental differences in the microcirculation and lymphatic system of the fetus, as compared with mature subjects, renders the fetus susceptible to interstitial fluid accumulation. These differences include greater capillary permeability, more compliant interstitial compartment, and greater influence of venous pressures on lymphatic return. The balance between interstitial fluid production and removal is most commonly disrupted as a consequence of homeostatic mechanisms serving to preserve adequate systemic delivery of metabolic substrate when cardiocirculatory function is impaired. The pathophysiology of two conditions of impaired cardiocirculatory function, atrial tachycardia and severe anemia, serve as examples of the mechanisms by which these homeostatic mechanisms perturb the balance of interstitial fluid movement.

Capillary Permeability↗
PubMed1995

[Non-immunological hydrops fetalis].

Hydrops foetalis is an uncommon condition, occurring in 1/1400-1/4000 of all pregnancies. The possible etiologies to the disease are numerous and up to now the prognosis has been very dull, with a mortality-rate of 40-90%. A successfully treated case story is presented. It is concluded, that it is essential from the very beginning to treat the child aggressively to restore normal respiratory and circulatory function. When this is performed the child will have a good chance of surviving and developing normally.

Female↗
PubMed1989

A case of recurrent infantile polycystic kidney associated with hydrops fetalis.

Nonimmune hydrops fetalis is becoming a predominant form of fetal hydrops due to the declining incidence of immune hydrops fetalis triggered by Rh isoimmunization. Infantile polycystic kidney appeared to be related to hydrops fetalis whether it is causal or merely coincidental and may represent another entry to differential diagnoses. Infantile polycystic kidney was diagnosed by an elevated maternal serum alpha-fetoprotein (AFP) value coupled with an ultrasonographic abnormality scanned as a multicystic mass with ascites in the fetal abdomen antenatally. This study presents a case of infantile polycystic kidney that resulted in a stillborn baby with hydrops fetalis and extensive placental calcification; it was the first case in Korea in which nonimmune hydrops fetalis was associated with infantile polycystic kidney in consecutive siblings by autosomal recessive inheritance in one family. In addition, this paper comprehensively reviews the incidence, etiology, prenatal diagnosis and proper management of nonimmune hydrops fetalis.

Adult↗
PubMed1987

Percutaneous ultrasound-guided fetal blood sampling in the management of nonimmune hydrops fetalis.

Nonimmune hydrops fetalis can be caused by fetal chromosomal, hematologic, cardiac, or infectious conditions. A fetal blood sample obtained from percutaneous ultrasound-guided fetal blood sampling can offer vital information about the underlying cause of nonimmune hydrops fetalis, and perinatal management can be planned accordingly. Ten cases of nonimmune hydrops fetalis were investigated with percutaneous ultrasound-guided fetal blood sampling and subsequent fetal blood analysis. Nine were proved to be Bart's hemoglobin hydrops fetalis, with 78.2% to 99% of Bart's hemoglobin in the fetal blood. Terminations of pregnancy were undertaken immediately, resulting in a lowered maternal morbidity rate. Free cord loops or an intra-abdominal portion of the dilated umbilical vein can be used for percutaneous fetal blood sampling in nonimmune hydrops fetalis when the placental cord insertion is difficult to visualize. Percutaneous ultrasound-guided fetal blood sampling and subsequent fetal blood analysis will eventually play a key role in the management of the puzzling nonimmune hydrops fetalis if the safety of ultrasound-guided fetal blood sampling can be verified by further clinical studies.

Adult↗
PubMed1995

Fetal blood sampling in the assessment of acute nonimmune hydrops fetalis.

Nonimmune hydrops fetalis can be associated with many fetal conditions. A fetal blood sample obtained from ultrasound-guided cordocentesis can offer vital information about the underlying cause of the nonimmune hydrops fetalis, and perinatal management can be planned accordingly. We attempted to evaluate a case of nonimmune hydrops fetalis conveying poor perinatal outcome through serial fetal blood sampling. Although we could establish no apparent reason for this acute case, fetal blood sampling in addition to ultrasonographic screening can play a key role in the management of the 'perplexing' nonimmune hydrops fetalis.

Adult↗
PubMed1994

Brain lesions of fetal onset in encephalopathic infants with nonimmune hydrops fetalis.

Nonimmune hydrops fetalis (NIHF) comprised 79% (45/57) of all infants with hydrops fetalis at our institution over a 6-year period. Thirty-seven infants with NIHF were liveborn. One or more electroencephalograms were performed on 40% of liveborn infants (15/37); the majority (87%) were moderately to markedly abnormal, including burst suppression, lack of background, multifocal sharp waves, excessive discontinuity, and disorganization reflecting significant neonatal encephalopathies. Postmortem neuropathologic examinations were performed in 86% of infants with NIHF who died or were stillborn, 81% of whom demonstrated intrauterine brain insults including microcalcifications, cerebral and/or cerebellar hypoplasia, microcephaly, encephalomalacia, cavitary lesions, astrocytosis, polymicrogyria, and severe neuronal loss. Cranial ultrasonography failed to document the diverse pathologic lesions that were later noted on postmortem examination. Ten infants survived the neonatal period, but 6 were neurologically abnormal at the time of discharge. Infants with NIHF are at risk for antepartum brain injury, and electroencephalographic abnormalities reflect in part a fetal brain disorder. A prospective clinical study is needed to fully assess the prevalence, incidence, spectrum of central nervous system involvement, contribution of intrapartum and neonatal stress, and long-term outcome in surviving infants with NIHF.

Brain↗
PubMed1991

The diagnosis and treatment of the nonimmune hydrops fetalis.

Fetal hydrops is associated with two distinct pathophysiologic situations. The isoimmune hydrops fetalis is a well understood disorder, and as the result of medical advances and prophylactic therapy its frequency is diminishing. The nonimmune hydrops fetalis is a poorly understood disease with a bad prognosis. The two disorders can be differentiated with the indirect Coombs test. In both cases the ultrasound examination plays an important role in the diagnosis, prognosis and management. Examination of the fetal blood sample gives recently a possibility to approach the disease. In NIHF the examination of fetal blood sample would give a relatively quick and effective diagnosis but its value for the treatment is limited. Although with the present technology it is impossible to diagnose all cases of NIHF, the early recognizing, the careful and step by step investigation, the active perinatologic management mostly can show the etiology and can help the perinatal team at the treatment of the disease.

Humans↗
PubMed1992

Intrauterine brain damage in nonimmune hydrops fetalis.

Nonimmune hydrops fetalis has been described in a large number of pathological conditions, but brain aspect has been poorly explored. We report the neuropathological findings in a series of 38 fetuses and neonates with anasarca of various origin. Fourteen fetal cerebral ultrasonograms were available; 8 presented some abnormalities. On brain examination, 23 cases showed hypoxic-ischemic lesions. The white matter was the main site of damage that consisted in classical leucomalacia or other features such as: astrocytic glial reaction, microcalcifications and microthromboses either as isolated finding or in association. Anoxic neuronal damage was much less frequent. Anemia, hypoprotidemia and cardiac failure with hypotension, which often occur in hydrops fetalis, may account for brain perfusion failure and hypoxic-ischemic changes.

Brain↗
PubMed1999

Congenital hypothyroidism and nonimmune hydrops fetalis: associated?

Hydrops fetalis (HF) consists of an abnormal accumulation of fluid in two or more fetal compartments, including ascites, pleural effusion, pericardial effusion, and skin edema. Almost all observed cases of HF are of the nonimmune type, the causes of which remain undetermined in 15% of patients. We report a newborn infant with nonimmune hydrops fetalis (NIHF) and congenital hypothyroidism. The infant's mother was healthy and there were no malformations of the placenta or umbilical cord. The infant did not show any structural abnormalities of his central nervous, cardiovascular, gastrointestinal, or urinary tract systems, and there was no evidence of anemia, infectious disease, or inborn error of metabolism. An immune-based process was unlikely, because the blood group of the mother and infant was A-positive and results of an indirect Coombs test in the mother and a direct Coombs test in the infant were negative. The patient's condition gradually improved with mechanical ventilation, repeated thoracocentesis, and total parenteral nutrition. By day 5 of age the skin edema, pericardial effusion, and ascites disappeared, but accumulation of significant amounts of chylous pleural fluid persisted. Because of lethargy, FT4 and thyroid-stimulating hormone levels were obtained and showed hypothyroidism. Thyroid hormone supplementation was then started, and within 4 days the infant became more vigorous and was weaned from mechanical ventilation. After 7 days, the chylothorax resolved completely as the serum thyroxine level normalized. No reaccumulation of pleural effusion was noticed. The infant started to gain weight and was discharged from the hospital at 35 days of age. A possible pathophysiologic association between congenital hypothyroidism and NIHF is discussed. NIHF may be caused by lymphatic congestion attributable to an impairment of lymphatic flow and a delayed return of lymph to the vascular compartment. There could be a possibility that because of thyroid hormone deficiency in this patient, there was reduced adrenergic stimulation of the lymphatic system. This could result in a sluggish flow of the lymph with engorgement of the lymphatic system, leakage of lymph into the pleura and the interstitial spaces, and the production of chylothorax with NIHF. Animal studies demonstrate a direct relationship between lymph flow rate or lung liquid clearance and adrenergic receptor activity in the lymphatic system. These observations support our hypothesis that deficient adrenergic activity in congenital hypothyroidism might lead to chylothorax with NIHF in the fetus. We speculate that thyroid hormone may play a role in the regulation of adrenergic receptors in the lymphatic system and lungs, thus modulating both the lymphatic flow rate and lung liquid clearance, and facilitating the resolution of chylothorax. Examination of thyroid functions should be included in the investigation of fetuses and neonates with NIHF of an obscure origin.

Congenital Hypothyroidism↗
PubMed2002

Retrospective analysis of 17 liveborn neonates with hydrops fetalis.

BACKGROUND: Hydrops fetalis (HF) is a condition with a high mortality rate. The cause may be due to a variety of underlying diseases. In the majority of cases, death occurs antepartum and intrapartum. For those that are born alive, it is difficult to survive. The purpose of this study was to analyze the clinical manifestations, etiologies and outcomes of liveborn babies with hydrops fetalis. METHODS: From October 1995 through May 2001, 17 liveborn neonates that presented with HF were admitted to our neonate intensive care unit (NICU). We were retrospectively reviewed their records. Clinical data including gestational age (GA) at diagnosis and birth, birth weight, Apgar score, maternal and fetal presentations, laboratory data, etiology and outcome were retrospectively collected and analyzed. RESULTS: The mean GA at diagnosis was 30.5 weeks and the mean GA at birth was 33.8 weeks. The male to female ratio was 8:9. Most cases presented with ascites (12/17) and cardiomegaly (8/17). The most common problem faced by the liveborn HF neonates was cardiovascular anomalies (7/17). Seven of these liveborn HF neonates survived. The overall mortality rate of HF in this review was 59%. In comparison with survival cases, those that died were diagnosed earlier, had lower Apgar scores, had more severe acidosis, and had pericardial effusion. CONCLUSION: Recent advances in prenatal ultrasonographic examinations have made early detection of fetal hydrops possible. The mortality rate of these liveborn hydropic neonates without receiving prenatal therapy was high. More effort in prenatal intervention is needed in order to decrease the mortality rate and improve the outcome of neonates with HF.

Female↗
PubMed1993

Umbilical pressure measurement in the evaluation of nonimmune hydrops fetalis.

OBJECTIVE: Nonimmune hydrops fetalis continues to have a perinatal mortality rate > 50%. Although many abnormalities are associated with nonimmune hydrops fetalis, the direct mechanism by which the hydrops occurs is often obscure, even after delivery. There are at least three possible mechanisms for hydrops: heart failure (whether primary or a secondary effect of obstructed venous return), lymphatic malformation, and liver or peritoneal disease. The development of safe access to the fetal circulation by cordocentesis allows for the measurement of the umbilical venous pressure, which is closely related to the fetal central venous pressure. The premise that nonimmune hydrops fetalis of cardiac origin could be distinguished from that of noncardiac origin was examined by measuring the umbilical venous pressure. STUDY DESIGN: Umbilical venous pressure was measured during indicated diagnostic cordocentesis in three groups of fetuses: 20 with nonimmune hydrops fetalis, four with a cardiac malformation but without nonimmune hydrops fetalis, and eight with immune hydrops (fetal hemolytic disease). In 16 of 20 fetuses with nonimmune hydrops fetalis the serum total protein and albumin concentrations were also measured. RESULTS: Presumed inadequate cardiac output, as indicated by an elevated umbilical venous pressure, was the mechanism of nonimmune hydrops fetalis in 13 of 20 (65%). The pathologic condition included arrhythmia, cardiothoracic abnormalities, severe polycythemia and hyperviscosity, viral infection, and severe anemia. Successful antenatal treatment normalized the umbilical venous pressure. Nonimmune hydrops fetalis secondary to noncardiac mechanisms did not progress in severity and was not amenable to antenatal therapy. Hypoproteinemia and hypoalbuminemia were found in only six of 16 cases and were similarly distributed between cardiac and noncardiac mechanisms. CONCLUSIONS: This is the first report where the measurement of umbilical venous pressure was applied to the evaluation of nonimmune hydrops fetalis. Cardiac dysfunction was the most common mechanism causing hydrops. The finding of a normal umbilical venous pressure greatly reduces the likelihood that the heart is the cause of the hydrops, even when there is a coexistent heart malformation. This immediate information allows the practitioner either to focus on therapeutic interventions that might lower the umbilical venous pressure or to look for noncardiac causes for the hydrops.

Anemia↗
PubMed1999

Nonimmune hydrops fetalis.

Nonimmune hydrops fetalis (NIHF) is a term that defines an edematous fetus that does not have erythroblastosis fetalis from isoimmunization. All of the other reasons that a fetus develops NIHF are included in this category. The causes of NIHF range from diseases to structural anomalies to genetic abnormalities. The mere presence of NIHF is a poor prognostic indicator, even with the etiology unknown. Some causes of NIHF are treatable with intrauterine therapies. These currently include cordocentesis to administer intrauterine transfusions or medications and fetal surgery. Prenatal testing to establish an etiology or follow the progress of the fetus is extensive, expensive, and stressful. The mortality rate remains high. Education, counseling, and support of the patient and family are the work of the prenatal period. Delivery room management of the infant with NIHF requires a resuscitation team with a specific plan of care to maximize chances of survival.

Cordocentesis↗
PubMed1997

Subchorial hematoma: a probable cause of reversible nonimmune hydrops fetalis.

Nonimmune hydrops fetails diagnosed at 21 weeks' gestation with profound ascites, hydrothorax, and pericardial effusion receded gradually with regression of a subchorial placental lucencies immediately below the umbilical cord insertion. Careful inspection of the delivered placenta revealed that there was a yellowish lesion of fibrin deposits below the cord insertion site, which resulted from the absorption of hematoma. A subchorial placental hematoma, which detected as a subchorial placental lucencies by ultrasonography, can be a cause of reversible nonimmune hydrops fetalis.

Adult↗