Survival of Hydrops Fetalis with and without Fetal Intervention
Abstract
1. Introduction
2. Materials and Methods
Statistical Analysis
3. Results
4. Discussion
5. Conclusions
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Conflicts of Interest
References
- Bellini, C.; Donarini, G.; Paladini, D.; Calevo, M.G.; Bellini, T.; Ramenghi, L.A.; Hennekam, R.C. Etiology of non-immune hydrops fetalis: An update. Am. J. Med. Genet. Part A 2015, 167, 1082–1088. [Google Scholar] [CrossRef] [PubMed]
- Ota, S.; Sahara, J.; Mabuchi, A.; Yamamoto, R.; Ishii, K.; Mitsuda, N. Perinatal and one-year outcomes of non-immune hydrops fetalis by etiology and age at diagnosis. J. Obstet. Gynaecol. Res. 2016, 42, 385–391. [Google Scholar] [CrossRef] [PubMed]
- Akkermans, J.; Peeters, S.H.; Klumper, F.J.; Lopriore, E.; Middeldorp, J.M.; Oepkes, D. Twenty-Five Years of Fetoscopic Laser Coagulation in Twin-Twin Transfusion Syndrome: A Systematic Review. Fetal Diagn. Ther. 2015, 38, 241–253. [Google Scholar] [CrossRef] [PubMed]
- Yang, Y.-S.; Ma, G.-C.; Shih, J.-C.; Chen, C.-P.; Chou, C.-H.; Yeh, K.-T.; Kuo, S.-J.; Chen, T.-H.; Hwu, W.-L.; Lee, T.-H.; et al. Experimental treatment of bilateral fetal chylothorax using in-utero pleurodesis. Ultrasound Obstet. Gynecol. 2012, 39, 56–62. [Google Scholar] [CrossRef]
- Huang, H.-R.; Tsay, P.-K.; Chiang, M.-C.; Lien, R.; Chou, Y.-H. Prognostic Factors and Clinical Features in Liveborn Neonates with Hydrops Fetalis. Am. J. Perinatol. 2007, 24, 33–38. [Google Scholar] [CrossRef]
- Lee, C.-J.; Tsao, P.-N.; Chen, C.-Y.; Hsieh, W.-S.; Liou, J.-Y.; Chou, H.-C. Prenatal therapy improves the survival of premature infants with congenital chylothorax. Pediatr. Neonatol. 2016, 57, 127–132. [Google Scholar] [CrossRef]
- Wu, W.-J.; Shih, J.-C.; Sago, H.; Chen, M. Complete resolution of hydrops by placement of double basket catheter in a case of macrocystic type multilocular pulmonary sequestration. Taiwan J. Obstet. Gynecol. 2017, 56, 402–405. [Google Scholar] [CrossRef]
- Lin, P.-H.; Wu, H.-H.; Tsai, H.-D.; Hsieh, C.T.-C. Successful treatment of atrial flutter by repeated intraperitoneal and intra-amniotic injections of amiodarone in a fetus with hydrops. Taiwan J. Obstet. Gynecol. 2016, 55, 434–436. [Google Scholar] [CrossRef]
- Adzick, N.S.; Harrison, M.R.; Crombleholme, T.M.; Flake, A.W.; Howell, L.J. Fetal lung lesions: Management and outcome. Am. J. Obstet. Gynecol. 1998, 179, 884–889. [Google Scholar] [CrossRef]
- Nassr, A.A.; Ness, A.; Hosseinzadeh, P.; Salmanian, B.; Espinoza, J.; Berger, V.; Werner, E.; Erfani, H.; Welty, S.; Bateni, Z.H.; et al. Outcome and Treatment of Antenatally Diagnosed Nonimmune Hydrops Fetalis. Fetal Diagn. Ther. 2017, 43, 123–128. [Google Scholar] [CrossRef]
- Gleason, C.; Juul, S. Avery’s Disease of Newborn, 10th ed.; Patricia Tannian: Philadelphia, PA, USA, 2018. [Google Scholar]
- Gozar, L.; Gabor-Miklosi, D.; Toganel, R.; Fagarasan, A.; Gozar, H.; Toma, D.; Cerghit-Paler, A. Fetal Tachyarrhythmia Management from Digoxin to Amiodarone—A Review. J. Clin. Med. 2022, 11, 804. [Google Scholar] [CrossRef] [PubMed]
- Kim, S.A.; Lee, S.M.; Hong, J.-S.; Lee, J.; Park, C.-W.; Kim, B.J.; Park, K.H.; Park, J.S.; Jun, J.K. Ultrasonographic severity scoring of non-immune hydrops: A predictor of perinatal mortality. J. Périnat. Med. 2015, 43, 53–59. [Google Scholar] [CrossRef] [PubMed]
- Yeang, C.-H.; Ma, G.-C.; Shih, J.-C.; Yang, Y.-S.; Chen, C.-P.; Chang, S.-P.; Wu, S.-H.; Liu, C.-S.; Kuo, S.-J.; Chou, H.-C.; et al. Genome-Wide Gene Expression Analysis Implicates the Immune Response and Lymphangiogenesis in the Pathogenesis of Fetal Chylothorax. PLoS ONE 2012, 7, e34901. [Google Scholar] [CrossRef] [PubMed]
- Bellini, C.; Hennekam, R.C.; Fulcheri, E.; Rutigliani, M.; Morcaldi, G.; Boccardo, F.M.; Bonioli, E. Etiology of nonimmune hydrops fetalis: A systematic review. Am. J. Med. Genet. Part A 2009, 149, 844–851. [Google Scholar] [CrossRef] [PubMed]
- Ker, C.-R.; Kuo, Y.-L.; Chan, T.-F. A survey of current use, dilemma and outlook of antenatal ultrasonography in Taiwan. Taiwan J. Obstet. Gynecol. 2019, 58, 820–826. [Google Scholar] [CrossRef] [PubMed]
- Moreno, C.A.; Kanazawa, T.; Barini, R.; Nomura, M.L.; Andrade, K.C.; Gomes, C.P.; Heinrich, J.K.; Giugliani, R.; Burin, M.; Cavalcanti, D.P. Non-immune hydrops fetalis: A prospective study of 53 cases. Am. J. Med. Genet. Part A 2013, 161, 3078–3086. [Google Scholar] [CrossRef] [PubMed]
- Turgal, M.; Ozyuncu, O.; Boyraz, G.; Yazicioglu, A.; Sinan Beksac, M. Non-immune hydrops fetalis as a diagnostic and survival problems: What do we tell the parents? J. Perinat. Med. 2015, 43, 353–358. [Google Scholar] [CrossRef]
- Abrams, M.E.; Meredith, K.S.; Kinnard, P.; Clark, R.H. Hydrops Fetalis: A Retrospective Review of Cases Reported to a Large National Database and Identification of Risk Factors Associated with Death. Pediatrics 2007, 120, 84–89. [Google Scholar] [CrossRef]
- Deurloo, K.L.; Devlieger, R.; Lopriore, E.; Klumper, F.J.; Oepkes, D. Isolated fetal hydrothorax with hydrops: A systematic review of prenatal treatment options. Prenat. Diagn. 2007, 27, 893–899. [Google Scholar] [CrossRef]
- Kessel, I.; Makhoul, I.R.; Sujov, P. Congenital hypothyroidism and nonimmune hydrops fetalis: Associated? Pediatrics 1999, 103, e9. [Google Scholar] [CrossRef]
- Yanai, N.; Shveiky, D. Fetal hydrops, associated with maternal propylthiouracil exposure, reversed by intrauterine therapy. Ultrasound Obstet. Gynecol. 2004, 23, 198–201. [Google Scholar] [CrossRef] [PubMed]
- Su, W.-J.; Ni, Y.-H.; Liu, D.-P.; Chiou, L.-S.; Cheng, W.-Y.; Wu, J.-S.J.; Lu, C.-Y. Low Seroprevalence of Parvovirus B19 in Taiwanese Children and Young Adults. Pediatr. Neonatol. 2010, 51, 265–268. [Google Scholar] [CrossRef][Green Version]
- Phibbs, R.H.; Johnson, P.; Tooley, W.H. Cardiorespiratory status of erythroblastotic newborn infants. II. Blood volume, hematocrit, and serum albumin concentration in relation to hydrops fetalis. Pediatrics 1974, 53, 13–23. [Google Scholar] [CrossRef] [PubMed]
Etiology and Prognosis | Prognosis | Fetal Intervention | ||
---|---|---|---|---|
Survival (N = 30) | Died (N = 12) | Yes (N = 22) | No (N = 20) | |
Idiopathic disorder | 5 | 5 | 4 | 6 |
Cardiovascular disorder | ||||
Cardiac failure | 2 | 1 | 1 | |
Prominent Eustachian valve | 1 | 1 | ||
Interatrial aneurysm | 1 | 1 | ||
Ectopic atrial rhythm | 1 | 1 | ||
Renal disorder | ||||
Congenital nephrotic syndrome | 1 | 1 | ||
Maternal or placenta condition | ||||
Placenta chorioangioma | 1 | 1 | ||
Vascular accidents | ||||
Twin–twin transfusion | 1 | 1 | ||
Nervous system lesions | ||||
Vein of Galen malformation | 1 | 1 | ||
Hemolytic anemias | ||||
Rh incompatibility with anti-E + c | 1 | 1 | ||
Maternal-fetal transfusion | 1 | 1 | ||
Infections | ||||
Congenital hepatitis | 1 | 1 | ||
Gastrointestinal conditions | ||||
Meconium peritonitis | 4 | 3 | 1 | |
Lymphatic abnormalities | ||||
Congenital chylothorax | 4 | 1 | 3 | 2 |
Congenital lymphatic dysplasia | 7 | 6 | 1 | |
Pulmonary conditions | ||||
Cystic adenomatoid malformation of the lung | 1 | 1 | 2 | 0 |
Congenital hypothyroidism | 2 | 2 |
Without Fetal Intervention | With Fetal Intervention | ||||
---|---|---|---|---|---|
N | Median | N | Median | p-Value | |
Length of stay (days) | 20 | 12.5 (1–91) | 22 | 35 (1–120) | 0.293 |
Gestational age (weeks) | 20 | 32.5 (25–38) | 22 | 33.0 (24–36) | 0.469 |
GA [a] at diagnosis (weeks) | 16 | 30.5 (20–38) | 21 | 28.0 (20–33) | 0.025 |
Birth body weight (gm) | 20 | 2481.0 (840–3670) | 22 | 2152.0 (690–3598) | 0.257 |
Apgar score at 1 min | 20 | 6.5 (1–9) | 22 | 5.0 (1–9) | 0.268 |
Apgar score at 5 min | 20 | 8.0 (1–10) | 22 | 7.0 (2–9) | 0.580 |
Albumin (g/dL) Hb (g/dL) | 20 18 | 2.1 (1.0–3.2) 13.3 (1.0–18.5) | 22 21 | 1.7 (1–3) 15.1 (2.1–19.7) | 0.283 0.015 |
Survivors | Death | ||||
---|---|---|---|---|---|
N | Median | N | Median | p-Value | |
Length of stay (days) | 30 | 36.5 (4–120) | 12 | 1.0 (1–46) | <0.001 |
Gestational age (weeks) | 30 | 33.0 (28–38) | 12 | 29.0 (24–35) | 0.002 |
Birth body weight (gm) | 30 | 2499.0 (1510–3670) | 12 | 1738.0 (690–3008) | 0.006 |
Apgar score at 1 min | 30 | 6.5 (1–9) | 12 | 2.0 (1–8) | <0.001 |
Apgar score at 5 min | 30 | 8.0 (5–10) | 12 | 5.0 (1–8) | <0.001 |
GA [a] at diagnosis (weeks) | 26 | 30.5 (20–38) | 11 | 27.0 (20–32) | 0.022 |
albumin (g/dL) | 30 | 2.2 (1.3–3.2) | 12 | 1.1 (1–1.5) | <0.001 |
1st ph. | 29 | 7.3 (6.9–7.4) | 10 | 6.9 (6.9–7.4) | 0.001 |
Parameter | Univariate Analysis (Crude) | Multiple Analysis (Adjusted) | ||||||
---|---|---|---|---|---|---|---|---|
Odds Ratio | 95% CI | p-Value | Odds Ratio | 95% CI | p-Value | |||
Gestational age | 0.650 | 0.486 | 0.869 | 0.004 | ||||
Birth body weight | 0.998 | 0.997 | 0.999 | 0.006 | ||||
A/S (1) [a] | 0.474 | 0.306 | 0.734 | 0.001 | 0.513 | 0.282 | 0.934 | 0.029 |
A/S (5) [b] | 0.407 | 0.230 | 0.722 | 0.002 | ||||
GA [c] at diagnosis | 0.859 | 0.740 | 0.998 | 0.047 | ||||
Albumin (g/dL) | 0.000 | 0.000 | 0.129 | 0.017 | ||||
1st pH | 0.000 | 0.000 | 0.015 | 0.001 | 0.000 | 0.000 | 0.238 | 0.026 |
PIH [d] (Yes vs. No) | 7.000 | 1.078 | 45.437 | 0.041 | 97.655 | 1.159 | 8227.646 | 0.043 |
Antenatal Intervention | Number |
---|---|
basket shunt | 2 |
pleurodesis + basket shunt | 1 |
thoracentesis + pleurodesis | 2 |
thoracentesis + ascites tapping | 2 |
ascites tapping | 6 |
thoracentesis | 8 |
intrauterine injection of amiodarone | 1 |
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Huang, Y.-Y.; Chang, Y.-J.; Chen, L.-J.; Lee, C.-H.; Chen, H.-N.; Chen, J.-Y.; Chen, M.; Hsiao, C.-C. Survival of Hydrops Fetalis with and without Fetal Intervention. Children 2022, 9, 530. https://doi.org/10.3390/children9040530
Huang Y-Y, Chang Y-J, Chen L-J, Lee C-H, Chen H-N, Chen J-Y, Chen M, Hsiao C-C. Survival of Hydrops Fetalis with and without Fetal Intervention. Children. 2022; 9(4):530. https://doi.org/10.3390/children9040530
Chicago/Turabian StyleHuang, Yu-Yun, Yu-Jun Chang, Lih-Ju Chen, Cheng-Han Lee, Hsiao-Neng Chen, Jia-Yuh Chen, Ming Chen, and Chien-Chou Hsiao. 2022. "Survival of Hydrops Fetalis with and without Fetal Intervention" Children 9, no. 4: 530. https://doi.org/10.3390/children9040530
APA StyleHuang, Y.-Y., Chang, Y.-J., Chen, L.-J., Lee, C.-H., Chen, H.-N., Chen, J.-Y., Chen, M., & Hsiao, C.-C. (2022). Survival of Hydrops Fetalis with and without Fetal Intervention. Children, 9(4), 530. https://doi.org/10.3390/children9040530