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Szewczuk O, Szewczuk W, Walędziak M, Różańska-Walędziak A. Spontaneous regression of fetal pleural effusion in pregnancy complicated with Herpes simplex infection: Clinical presentation and literature review. Eur J Obstet Gynecol Reprod Biol 2024; 299:193-198. [PMID: 38885560 DOI: 10.1016/j.ejogrb.2024.06.024] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/21/2024] [Accepted: 06/11/2024] [Indexed: 06/20/2024]
Abstract
Fetal primary hydrothorax is a rare congenital anomaly with an estimated incidence of 1:10,000-15,000 pregnancies, with an unpredictable clinical course, ranging from spontaneous resolution to fetal death. A case of unilateral fetal pleural effusion was diagnosed at 35th week of gestation during a routine ultrasonographic fetal assessment in an uncomplicated pregnancy. A large echogenic collection of fluid was revealed in the right pleural cavity, together with atelectasis of the right lung, as well as displacement of heart and mediastinal structures to the left side of thorax. The patient was also diagnosed with polyhydramnios and there was a disproportion of heart ventricles volume. No other fetal structural abnormalities were detected and there were no symptoms of edema. Fetal biometrics was consistent with the gestational age. In echocardiography, fetal heart was structurally and functionally normal. Screening tests for congenital infections of the fetus were negative. Autoimmune fetal hydrops was excluded after laboratory tests. There was no parents' consent for the analysis of the karyotype. The patient presented clinical symptoms and was diagnosed with Herpes simplex virus infection and was treated with oral acyclovir. Serial fetal ultrasound exams showed gradual decrease in pleural fluid volume up to complete resolution in 38th week of pregnancy. Pregnancy was ended in the 38th week of gestation with a cesarean delivery of a healthy neonate. It is yet to be determined if there is a direct association between Herpes simplex virus infection in pregnancy and the risk of fetal pleural effusion. The incidence of fetal pleural effusion is low and the neonatal outcome difficult to be predicted. The optimum management of fetal pleural effusion should be subject to further studies to determine the best clinical practice.
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Affiliation(s)
- Oksana Szewczuk
- 2nd Department of Obstetrics and Gynecology, Medical University, Princess Anna Mazowiecka Hospital, Karowa st. 2, 00-315 Warsaw, Poland
| | - Wiktor Szewczuk
- Department of Pathology, Military Institute of Medicine-National Research Institute, Szaserow st. 128, 04-141 Warsaw, Poland
| | - Maciej Walędziak
- Department of General, Oncological, Metabolic and Thoracic Surgery, Military Institute of Medicine-National Research Institute, Szaserów 128 st., 04-141 Warsaw, Poland.
| | - Anna Różańska-Walędziak
- Department of Human Physiology and Pathophysiology, Faculty of Medicine, Collegium Medicum, Cardinal Stefan Wyszynski University in Warsaw, 01-938 Warsaw, Poland
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Dionysopoulou A, Pirih E, Macchiella D, Fruth A, Jahn-Eimermacher A, Kampmann C, Mildenberger E, Whybra C. The Cardiovascular Profile Score in Patients with Non-immune Hydrops Fetalis and Cardiac Anomalies - a Pilot Study. Reprod Sci 2023; 30:2805-2812. [PMID: 36988903 PMCID: PMC10480243 DOI: 10.1007/s43032-023-01216-w] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/06/2022] [Accepted: 03/09/2023] [Indexed: 03/30/2023]
Abstract
The purpose of this paper is to explore whether the cardiovascular profile score (CVPS) correlates with fetal outcome in patients with non-immune hydrops fetalis (NIHF) and cardiac anomalies. In this retrospective study, we included fetuses with NIHF and the suspicion of a cardiac anomaly in prenatal ultrasound. The CVPS was calculated using information obtained by fetal echocardiographic examination. Feto-neonatal mortality (FNM) was defined as intrauterine fetal demise or death in the first 6 months of life. We reviewed 98 patients, who were referred to the Department of Obstetrics and Gynecology of the Johannes Gutenberg University in Mainz with the diagnosis of NIHF between January 2007 and March 2021. By eighteen of them, the suspicion of a cardiac anomaly was raised. After exclusion of six pregnancies (one termination of pregnancy and five because of incomplete data), 12 cases were left for analysis. Mean gestational age at which the CVPS was calculated was 29 + 2 weeks. Two fetuses died in utero. Of the remaining ten hydropic fetuses, three newborns died in the neonatal period, and seven survived after a 6-month surveillance period. Median CVPS of all fetuses was 6 points. Surviving fetuses showed statistically significantly higher CVPS values (median 8 points) than fetuses with FNM (median 5 points, p value = 0.009). Our results point towards a positive association between CVPS and fetal outcome in fetuses with NIHF and cardiac anomalies. The CVPS appears to be a useful marker in the assessment of heart failure in utero.
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Affiliation(s)
- Anna Dionysopoulou
- Department of Obstetrics and Gynecology, University Medical Center of the Johannes Gutenberg University Mainz, Langenbeckstrasse 1, 55131, Mainz, Germany.
| | - Etienne Pirih
- Department of Neonatology, University Medical Center of the Johannes Gutenberg University Mainz, Mainz, Germany
| | - Doris Macchiella
- Department of Obstetrics and Gynecology, University Medical Center of the Johannes Gutenberg University Mainz, Langenbeckstrasse 1, 55131, Mainz, Germany
| | - Anja Fruth
- Department of Obstetrics and Gynecology, University Medical Center of the Johannes Gutenberg University Mainz, Langenbeckstrasse 1, 55131, Mainz, Germany
| | - Antje Jahn-Eimermacher
- Department of Mathematics and Natural Sciences, University of Applied Sciences, Darmstadt, Germany
| | - Christoff Kampmann
- Department of Pediatric Cardiology, University Medical Center of the Johannes Gutenberg University Mainz, Mainz, Germany
| | - Eva Mildenberger
- Department of Neonatology, University Medical Center of the Johannes Gutenberg University Mainz, Mainz, Germany
| | - Catharina Whybra
- Department of Neonatology, University Medical Center of the Johannes Gutenberg University Mainz, Mainz, Germany
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Takita H, Matsuoka R, Goto M, Tokunaka M, Arakaki T, Nakamura M, Sekizawa A. Long-term outcome of cases of fetal pleural effusion: A study at a single perinatal center in Japan. JOURNAL OF CLINICAL ULTRASOUND : JCU 2022; 50:805-809. [PMID: 35394680 DOI: 10.1002/jcu.23196] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 09/01/2021] [Revised: 02/20/2022] [Accepted: 02/21/2022] [Indexed: 06/14/2023]
Abstract
PURPOSE To analyze the long-term prognosis of primary and secondary fetal pleural effusion (FPE). METHODS We investigated all cases of FPE in a single University hospital (2005-2020). Cases were classified as primary (cases with only pleural effusion) and secondary (cases with other abnormalities such as chromosomal abnormalities or fetal cardiac failure). We retrospectively reviewed the medical records from the time of diagnosis, to assess medical procedures performed, chromosomal test results, and clinical outcomes. RESULTS Among 18 027 deliveries, 17 FPEs were identified (primary FPE: 8, secondary FPE: 9). Most primary FPEs were diagnosed in the second trimester of pregnancy, while all secondary FPEs were diagnosed in the third trimester. Secondary FPE was often associated with chromosomal abnormalities, including trisomy 21. The prognosis of pleural effusion caused by trisomy 21 was relatively good, except for cases with TAM. Cases of secondary FPE without trisomy 21 were of cardiac origin, and the neonatal prognosis was poor. The short-term prognosis was better in the primary FPE group, but long-term follow-up identified conditions such as acute encephalitis with refractory, repetitive partial seizures, developmental delay and attention deficit hyperactivity disorder. CONCLUSION Fetal pleural effusion without the presence of chromosomal abnormalities or morphologies has a good short-term prognosis, but the long-term prognosis is poor. Thus, long-term follow-up is necessary for all cases of fetal pleural effusion.
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Affiliation(s)
- Hiroko Takita
- Department of Obstetrics and Gynecology, Showa University School of Medicine, Tokyo, Japan
| | - Ryu Matsuoka
- Department of Obstetrics and Gynecology, Showa University School of Medicine, Tokyo, Japan
| | - Minako Goto
- Department of Obstetrics and Gynecology, Showa University School of Medicine, Tokyo, Japan
| | - Mayumi Tokunaka
- Department of Obstetrics and Gynecology, Showa University School of Medicine, Tokyo, Japan
| | - Tatsuya Arakaki
- Department of Obstetrics and Gynecology, Showa University School of Medicine, Tokyo, Japan
| | - Masamitsu Nakamura
- Department of Obstetrics and Gynecology, Showa University School of Medicine, Tokyo, Japan
| | - Akihiko Sekizawa
- Department of Obstetrics and Gynecology, Showa University School of Medicine, Tokyo, Japan
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Maranto M, Cigna V, Orlandi E, Cucinella G, Lo Verso C, Duca V, Picciotto F. Non-immune hydrops fetalis: Two case reports. World J Clin Cases 2021; 9:6531-6537. [PMID: 34435022 PMCID: PMC8362581 DOI: 10.12998/wjcc.v9.i22.6531] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/07/2021] [Revised: 05/06/2021] [Accepted: 06/04/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Fetal hydrops is a serious condition difficult to manage, often with a poor prognosis, and it is characterized by the collection of fluid in the extravascular compartments. Before 1968, the most frequent cause was the maternal-fetal Rh incompatibility. Today, 90% of the cases are non-immune hydrops fetalis. Multiple fetal anatomic and functional disorders can cause non-immune hydrops fetalis and the pathogenesis is incompletely understood. Etiology varies from viral infections to heart disease, chromosomal abnormalities, hematological and autoimmune causes.
CASE SUMMARY A 38-year-old pregnant woman has neck lymphoadenomegaly, fever, cough, tonsillar plaques at 14 wk of amenorrhea and a rash with widespread itching. At 27.5 wk a fetal ultrasound shows signs of severe anemia and hydrops. Cordocentesis is performed with confirmation of severe fetal anemia and subsequent fetal transfusion. The karyotype is 46, XX, array-comparative genome hybridization (CGH) negative, and infectious tests are not conclusive. In the following days there is a progressive improvement of the indirect signs of fetal anemia. At 33.6 wk, for relapse of severe fetal anemia, further fetal transfusions are necessary and an urgent cesarean section is performed. On the day 12 of life, for the detection of anemia, the newborn is subjected to transfusion of concentrated red blood cells and begins treatment with erythropoietin. Later there is a normalization of blood chemistry values and the baby does not need new transfusions. A 29-year-old pregnant woman, with Sjogren's syndrome and positive Anti-Ro/SSA antibodies, is subjected to serial fetal ecocardio for branch block. At 26.5 wk there is a finding of fetal ascites. Infectious disease tests on amniotic fluid are negative as well as quantitative fluorescent polymerase chain reaction, Array CGH. At cordocentesis Hb is 1.3 mmol/L, consequently fetal transfusion is performed. Also in this case, due to continuous episodes of relapse of fetal anemia with consequent transfusions, at 29.4 wk a cesarean section is performed. On day 9 of life, a treatment with erythropoietin is started in the newborn, but the baby needs three blood transfusions. The search for autoantibodies in the baby found SS-A Ro60 positive, SSA-Ro52 positive and SS-B negative. The hemoglobin values normalized after the disappearance of maternal autoantibodies.
CONCLUSION An attempt to determine the etiology of hydrops should be made at the time of diagnosis because the goal is to treat underlying cause, whenever possible. Even if the infectious examinations are not conclusive, but the pregnancy history is strongly suggestive of infection as in the first case, the infectious etiology must not be excluded. In the second case, instead, transplacental passage of maternal autoantibodies caused hydrops fetalis and severe anemia. Finally, obstetric management must be aimed at fetal support up to an optimal timing for delivery by evaluating risks and benefits to increase the chances of survival without sequelae.
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Affiliation(s)
- Marianna Maranto
- Fetal Medicine and Prenatal Diagnosis Unit, Villa Sofia Cervello Hospitals, Palermo 90146, Italy
| | - Valentina Cigna
- Fetal Medicine and Prenatal Diagnosis Unit, Villa Sofia Cervello Hospitals, Palermo 90146, Italy
| | - Emanuela Orlandi
- Fetal Medicine and Prenatal Diagnosis Unit, Villa Sofia Cervello Hospitals, Palermo 90146, Italy
| | - Gaspare Cucinella
- Health Promotion, Maternal and Infant Care Unit, Internal Medicine and Medical Specialties “G. D’Alessandro”, University of Palermo, Palermo 90100, Italy
| | - Clelia Lo Verso
- Neonatology and Neonatal Intensive Care Unit, Civico Di Cristina Benfratelli Hospital, Palermo 90100, Italy
| | - Vincenzo Duca
- Neonatology and Neonatal Intensive Care Unit, Ingrassia Hospital, Palermo 90100, Italy
| | - Francesco Picciotto
- Fetal Medicine and Prenatal Diagnosis Unit, Villa Sofia Cervello Hospitals, Palermo 90146, Italy
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Late-onset fetal bilateral pleural effusions associated with Down syndrome. Taiwan J Obstet Gynecol 2018; 57:133-136. [DOI: 10.1016/j.tjog.2018.01.001] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 01/10/2018] [Indexed: 11/21/2022] Open
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Cao L, Du Y, Wang L. Fetal pleural effusion and Down syndrome. Intractable Rare Dis Res 2017; 6:158-162. [PMID: 28944136 PMCID: PMC5608924 DOI: 10.5582/irdr.2017.01028] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/10/2017] [Revised: 05/22/2017] [Accepted: 05/27/2017] [Indexed: 01/23/2023] Open
Abstract
Fetal pleural effusion is a rare abnormality that results from accumulation of fluid in the chest cavity. It can be classified as primary fetal hydrothorax and secondary fetal hydrothorax. The underlying causes of pleural effusion are still unknown, and the current treatment strategies are mainly based on symptoms. The prognosis of fetal pleural effusion varies significantly, ranging from spontaneous resolution to perinatal death. Recent advances in prenatal diagnostic methods and treatment such as thoracoamniotic shunting have significantly improved the survival rates for patients with or without hydrops.
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Affiliation(s)
- Li Cao
- Ultrasound Department, Obstetrics and Gynecology Hospital of Fudan University, Shanghai, China
| | - Yan Du
- Office of Clinical Epidemiology, Obstetrics and Gynecology Hospital of Fudan University, Shanghai, China
| | - Ling Wang
- Laboratory for Reproductive Immunology, Hospital & Institute of Obstetrics and Gynecology, IBS, Fudan University Shanghai Medical College, Shanghai, China
- The Academy of Integrative Medicine of Fudan University, Shanghai, China
- Shanghai Key Laboratory of Female Reproductive Endocrine-related Diseases, Shanghai, China
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Abstract
Nonimmune hydrops fetalis (NIHF) is a condition in which excess fluid has accumulated in the fetal interstitial spaces as a result of one or more nonimmune factors. A plethora of maternal, placental, and fetal disease processes have been associated with NIHF. Knowledge of the various etiologies of NIHF and how the disease process affects fluid homeostasis is important for planning patient care and counseling families of patients diagnosed with nonimmune hydrops fetalis. This article discusses the mechanisms governing fluid distribution in the extracellular spaces, examines the various etiologies associated with NIHF, and describes the pathogenesis of NIHF for each etiologic category.
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Abstract
Hydrops fetalis is a condition in which there is an excess of total body fluid, primarily within the fetal interstitial spaces. Etymologically, hydrops fetalis is a Latin term meaning "edema of the fetus." In addition to generalized edema, the fetus has at least one of the following: ascites, pericardial effusion, pleural effusion(s), and an abnormally thick (>6 cm) placenta. Hydrops is classified as nonimmune hydrops fetalis (NIHF) when it occurs without evidence of isoimmunization.
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Czernik C, Proquitté H, Metze B, Bührer C. Hydrops fetalis – has there been a change in diagnostic spectrum and mortality? J Matern Fetal Neonatal Med 2010; 24:258-63. [DOI: 10.3109/14767058.2010.483522] [Citation(s) in RCA: 22] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/30/2023]
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Bellini C, Hennekam RCM, Boccardo F, Campisi C, Serra G, Bonioli E. Nonimmune idiopathic hydrops fetalis and congenital lymphatic dysplasia. Am J Med Genet A 2006; 140:678-84. [PMID: 16502426 DOI: 10.1002/ajmg.a.31100] [Citation(s) in RCA: 18] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/30/2023]
Abstract
Six newborns that presented at birth with nonimmune hydrops fetalis and for whom no cause could be found were investigated for the presence of lymphatic dysplasia. Careful analysis led to findings of some degree of lymphatic dysplasia in all patients. This suggests that lymphatic dysplasia may represent at least part of the causes that are responsible for the "idiopathic" form of nonimmune hydrops fetalis. Carefully searching for lymphatic dysplasia in these patients, and if indicated in their relatives, as well as establishing the exact nature of the lymphatic dysplasia must be carried out so as to provide proper genetic counseling to families with nonimmune hydrops.
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Affiliation(s)
- Carlo Bellini
- Servizio di Patologia Neonatale, Dipartimento di Pediatria (DIPE), Università di Genova, Istituto G. Gaslini, Genova, Italia.
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Alkalay AL, Galvis S, Ferry DA, Simmons CF, Krueger RC. Hemodynamic changes in anemic premature infants: are we allowing the hematocrits to fall too low? Pediatrics 2003; 112:838-45. [PMID: 14523175 DOI: 10.1542/peds.112.4.838] [Citation(s) in RCA: 46] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/24/2022] Open
Abstract
OBJECTIVE Currently, many nurseries allow hematocrits to fall to <21% in apparently "stable" premature infants before considering a blood transfusion. We evaluated clinical changes and hemodynamic changes by echocardiogram in "stable" anemic premature infants before, during, and after transfusion. METHODS "Stable" premature infants (< or =32 weeks' gestation) who were to receive transfusions (2 aliquots of 10 mL/kg packed red blood cells, 12 hours apart) were eligible for prospective enrollment. Cardiac function by echocardiography and vital signs were measured 4 times: 1 to 3 hours before and 2 to 4 hours after the initial aliquot and 4 to 7 hours and 27 to 34 hours after the second aliquot. Infants were grouped prospectively according to pretransfusion hematocrit ranges for analysis: < or =21% (low), 22% to 26% (mid), and > or =27% (high). RESULTS Thirty-two infants were enrolled. No differences were observed between the groups in sex, birth weight, postconceptional age, or postnatal weight at enrollment. Before transfusion, low- and mid-range groups had higher left ventricular end systolic and diastolic diameters, in comparison with high range. Low range had increased stroke volume in comparison with the high-range group. These changes persisted after transfusion. Mean diastolic blood pressure rose and peak velocity in the aorta fell in the low-range group after transfusion. Pretransfusion hematocrit was correlated with but poorly predictive of echocardiographic measurements. Infants with inappropriate weight gain had increased ventricular end diastolic diameters, consistent with congestive heart failure. CONCLUSIONS Apparently "stable" anemic premature infants may be in a clinically unrecognized high cardiac output state, and some echocardiographic measurements do not improve within 48 hours after transfusion. The benefits of transfusion practices guided by measures of cardiac function should be evaluated.
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Affiliation(s)
- Arie L Alkalay
- Division of Neonatology, Department of Pediatrics, Ahmanson Pediatric Center, University of California at Los Angeles School of Medicine, Los Angeles, California 90048, USA.
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Mascaretti RS, Falcão MC, Silva AM, Vaz FAC, Leone CR. Characterization of newborns with nonimmune hydrops fetalis admitted to a neonatal intensive care unit. REVISTA DO HOSPITAL DAS CLINICAS 2003; 58:125-32. [PMID: 12894308 DOI: 10.1590/s0041-87812003000300001] [Citation(s) in RCA: 18] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/21/2022]
Abstract
PURPOSE To determine the incidence and characteristics of nonimmune hydrops fetalis in the newborn population. METHOD A retrospective study of the period between 1996 and 2000, including all newborns with a prenatal or early neonatal diagnosis of nonimmune hydrops fetalis, based on clinical history, physical examination, and laboratory evaluation. The following were analyzed: prenatal follow-up, delivery type, gender, birth weight, gestational age, presence of perinatal asphyxia, nutritional classification, etiopathic diagnosis, length of hospital stay, mortality, and age at death. RESULTS A total of 47 newborns with hydrops fetalis (0.42% of live births), 18 (38.3%) with the immune form and 29 (61.7%) with the nonimmune form, were selected for study. The incidence of nonimmune hydrops fetalis was 1 per 414 neonates. Data was obtained from 21 newborns, with the following characteristics: 19 (90.5%) were suspected from prenatal diagnosis, 18 (85.7%) were born by cesarean delivery, 15 (71.4%) were female, and 10 (47.6%) were asphyxiated. The average weight was 2665.9 g, and the average gestational age was 35 3/7 weeks; 14 (66.6%) were preterm; 18 (85.0 %) appropriate delivery time; and 3 (14.3%) were large for gestational age. The etiopathic diagnosis was determined for 62%, which included cardiovascular (19.0%), infectious (9.5%), placental (4.8%), hematologic (4.7%), genitourinary (4.8%), and tumoral causes (4.8%), and there was a combination of causes in 9.5%. The etiology was classified as idiopathic in 38%. The length of hospital stay was 26.6 +/- 23.6 days, and the mortality rate was 52.4%. CONCLUSIONS The establishment of a suitable etiopathic diagnosis associated with prenatal detection of nonimmune hydrops fetalis can be an important step in reducing the neonatal mortality rate from this condition.
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Affiliation(s)
- Renata Suman Mascaretti
- Departament and Experimental Research Unit, Children's Institute, Hospital das Clínicas, Faculty of Medicine, University of São Paulo, São Paulo, SP, Brazil
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Abstract
Although most NIH fetuses have poor prognosis, it is imperative to attempt to identify the underlying cause to present parents with the best estimate for prognosis and future outcomes. Identification of a disorder with a good prognosis may prevent unnecessary termination of pregnancy and give a chance for therapy. Identification of a disorder with poor prognosis may still provide information that may be unavailable after delivery regarding this and future pregnancies.
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Affiliation(s)
- R Bukowski
- Division of Maternal Fetal Medicine, Department of Obstetrics and Gynecology, University of Texas Medical Branch, Galveston, Texas, USA
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15
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Abstract
Hydrops fetalis is a morbid condition caused by a wide variety of fetal, placental, and maternal diseases. Mortality is high and depends on the gestational age at the time of occurrence and underlying etiology. Although the condition was described more than 300 years ago, recent advances in obstetric ultrasound, prenatal diagnostics have made it possible to differentiate various etiologies involved. It is also possible to treat some of these fetuses prenatally. In utero medical and surgical therapy is presently done in some centers. However, the majority of cases diagnosed remain untreatable. Early diagnosis of untreatable cases allows parents to make informed choices about subsequent management. Recent advances are covered in this review.
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Affiliation(s)
- I Forouzan
- Department of Obstetrics and Gynecology, University of Pennsylvania Medical Center, Philadelphia, USA
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Rejjal AR, Rahbeeni Z, al-Zahrani AF. Prognostic factors and prenatal management in non immune hydrops fetalis are still a dilemma. J Perinat Med 1996; 24:461-6. [PMID: 8950726 DOI: 10.1515/jpme.1996.24.5.461] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/03/2023]
Abstract
Seventeen cases of non-immune hydrops fetalis (NIHF) were diagnosed prenatally at King Faisal Specialist Hospital and Research Centre, Riyadh, Saudi Arabia over a period of 15 years (1979-1994). In nine patients (53%) a possible underlying mechanism was suspected. Of the six patients who survived beyond the first year of life, four had normal neurological and development follow-up. Family history was positive for NIHF in five cases (29%): two of these had a history of four siblings each who had been diagnosed with NIHF. All patients had prenatal ascites and subcutaneous oedema diagnosed by ultrasound. All five patients who had prenatal ascites, pericardial and pleural effusion died, while 9 of 11 (82%) patients who had prenatal pleural effusion and ascites also succumbed. Four of five (80%) patients with congenital anomalies died. One patient required intrauterine blood transfusion because of fetal anemia with subsequent partial resolution of the hydrops. Two patients received digitalis transplacentally for treatment of congestive heart failure secondary to congenital heart disease without response. We conclude that the presence of prenatal pericardial and pleural effusion or congenital anomalies carries a very poor prognosis in patients with NIHF.
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Affiliation(s)
- A R Rejjal
- Department of Pediatrics, King Faisal Specialist Hospital and Research Centre, Riyadh, Kingdom of Saudi Arabia
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17
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Abstract
As the incidence of Rh-isoimmunization has been decreasing with the availability of Rh immune globulin, the proportion of cases of fetal hydrops from nonimmune causes has increased. Evaluation of the fetus with hydrops requires an integrated approach, beginning with targeted ultrasound evaluation and potentially including maternal and fetal blood testing and other invasive testing. Because the list of conditions that may cause hydrops is long and continues to grow, it is often difficult to make a precise diagnosis; however, through a systematic approach, one may frequently narrow the etiology to a category of disorders and determine whether any interventions are available that are likely to be helpful in improving the outlook for the fetus.
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Affiliation(s)
- D C Jones
- Department of Obstetrics & Gynecology, Yale University School of Medicine, New Haven, CT 06520, USA
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18
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Rice HE, Estes JM, Hedrick MH, Bealer JF, Harrison MR, Adzick NS. Congenital cystic adenomatoid malformation: a sheep model of fetal hydrops. J Pediatr Surg 1994; 29:692-6. [PMID: 8035286 DOI: 10.1016/0022-3468(94)90743-9] [Citation(s) in RCA: 58] [Impact Index Per Article: 1.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/28/2023]
Abstract
Congenital cystic adenomatoid malformation (CCAM) can be diagnosed in utero. Nonimmune hydrops associated with CCAM is a predictor of fetal demise. Fetuses with prenatally diagnosed large CCAM tumors and hydrops have undergone successful in utero resection. An animal model is needed to understand the pathophysiology of CCAM and hydrops. To create a model of CCAM and hydrops, the authors implanted an intrathoracic tissue expander in six fetal sheep at 120 days' gestation. The inflatable tissue expander was implanted in the right side of the chest, and arterial, venous, intrathoracic, and intraamniotic pressure catheters were placed. Each day, the expander was inflated with 25 to 50 mL of saline (maximum, 150 mL), ultrasound examination was performed, and all pressure measurements were taken. In all six fetuses, hydrops developed after expander inflation. Expander inflation correlated with an increase in central venous pressure (CVP) (4 +/- 2 mm Hg v 16 +/- 2 mm Hg [mean +/- SD]; P < .05). To simulate in utero CCAM resection, the expander was deflated in four hydropic sheep, resulting in return of the CVP to near baseline and resolution of hydrops. Silicone vascular casts of two postmortem sheep demonstrated lateral displacement and compression of the vena cavae by the expander. The authors successfully created a model of CCAM and hydrops by inflating an intrathoracic tissue expander in fetal sheep. Based on this model, hydrops associated with CCAM results from obstruction of cardiac venous return and central venous hypertension. This pathophysiology is reversed by expander deflation, which simulates in utero CCAM resection.(ABSTRACT TRUNCATED AT 250 WORDS)
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Affiliation(s)
- H E Rice
- Department of Surgery, University of California, San Francisco 94143
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Sheikh AU, Ernest JM, O'Shea M. Long-term outcome in fetal hydrops from parvovirus B19 infection. Am J Obstet Gynecol 1992; 167:337-41. [PMID: 1497035 DOI: 10.1016/s0002-9378(11)91411-2] [Citation(s) in RCA: 63] [Impact Index Per Article: 1.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/27/2022]
Abstract
Parvovirus B19 infection in the fetus is associated with anemia and hydrops and can result in fetal death. Fetal transfusion has been used in an attempt to improve outcome; however, it is associated with its own perinatal morbidity. We report two cases of fetal parvovirus B19 infection that were confirmed by polymerase chain reaction for parvovirus B19 deoxyribonucleic acid in umbilical cord blood. Ultrasonographic signs of compromise were observed at 30 and 24 weeks of gestation. Both fetuses were hydropic and one fetus was also anemic. Serial sonograms demonstrated that the hydrops resolved spontaneously over 3 to 5 weeks after diagnosis. One infant was delivered at 32 weeks of gestation as a result of idiopathic preterm labor. The other infant was delivered at term. Both infants appeared relatively normal at birth and have developed normally in the first year of life. Thus fetal hydrops in association with parvovirus B19 infection does not always lead to poor long-term outcome. A conservative approach without in utero therapy may be appropriate for the management of some of these fetuses.
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Affiliation(s)
- A U Sheikh
- Department of Obstetrics and Gynecology, Bowman Gray School of Medicine, Wake Forest University, Winston-Salem, NC 27157-1066
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