A L J O A T U N R I N R A E L P Original Article P L E R A Perinatal Journal 2019;27(3):183–188 I N N R A U T A L J O ©2019 Perinatal Medicine Foundation Perinatal outcomes in the fetuses diagnosed with hydrops fetalis and isolated pleural effusion Reyhan Ayaz1 İD , Oya Demirci2 İD 1Department of Perinatology, Faculty of Medicine, Istanbul Medeniyet University, Istanbul, Turkey 2Department of Perinatology, Zeynep Kamil Maternity and Pediatric Training and Research Hospital, University of Health Sciences, Istanbul, Turkey Abstract Özet: Hidrops fetalis ve izole plevral effüzyon saptanan fetüslerde perinatal sonuçlar Objective: Our study aims to evaluate our clinical experience on and Amaç: Klini¤imize baflvuran primer plevral effüzyon ve hidrops postnatal outcomes of the cases with primary pleural effusion and geliflen olgularda klinik deneyimimiz ve postnatal sonuçlar›n de- hydrops who admitted to our clinic. ¤erlendirilmesidir. Methods: In our study, the cases found to have fetal pleural effusion Yöntem: Çal›flmada Ocak 2017 – Ekim 2019 tarihleri aras›nda with or without hydrops finding in the second and third trimester ikinci ve üçüncü dönemde hidrops bulgusu olan veya olmayan fe- between January 2017 and October 2019 were evaluated retrospec- tal plevral effüzyon saptanan olgular retrospektif olarak incelendi. tively. The patients were examined in terms of the blood types, mid- Hastalar kan gruplar›, orta serebral arter pik sistolik velositesi ve dle cerebral artery peak systolic velocity and lymphocyte levels in plevral effüzyon s›v›s›nda lenfosit düzeyi yönünden incelendi ve their pleural effusion fluid, and their detailed fetal anatomic screen- hastalar›n ayr›nt›l› fetal anatomik taramas›, fetal ekokardiyografi- ing, fetal echocardiography, karyotyping examination, and TORCH si, karyotip incelemesi ve maternal kanda TORCH tetkikleri ya- screening in the maternal blood were carried out. p›ld›. Results: Twelve patients who underwent thoracocentesis and 2 Bulgular: Çal›flmaya torakosentez uygulanan 12, uygulanmayan 2 patients who did not undergo thoracocentesis were included in the hasta al›nd›. 6 hasta hidrops fetalis, 8 hasta izole plevral effüzyon study. It was found that 6 patients had hydrops and 8 patients had iso- olarak de¤erlendirildi. ‹zole olgular›n 1 tanesinde postnatal diyaf- lated pleural effusion. Postnatal diaphragmatic hernia was detected in ram hernisi saptand› ve primer onar›ld›. 2 hasta hariç 12 hastaya 1 of the isolated cases, and it was repaired primarily. Thoracocentesis torakosentez yap›ld›. Birinci torakosentez sonras› 6 hastada effüz- was carried out in 12 patients except 2 patients. When the effusion yon tekrar› olmas› üzerine 2 hastaya do¤uma yak›n ifllem tekrar› relapsed in 6 patients after the first thoracocentesis, the procedure gerçeklefltirildi. ‹fllem tekrar› yap›lan bebeklerde postnatal entü- was repeated in 2 patients when the labor was close. While postnatal basyon gerekmezken, yap›lmayan 4 hasta entübe edildi. 2. ifllem s›- intubation was not necessary in the newborns that underwent the ras›nda bir fetüste fetal distres geliflmesi üzerine acil sezaryen ile repeated procedure, 4 patients who did not undergo were intubated. do¤um yapt›r›ld›. Tekrarlayan plevral effüzyonu olan 6 hastaya Emergency cesarean section was performed upon the development of postnatal toraks tüpü tak›ld›. Hidrops olan ve plevral s›v›daki len- fetal distress in a fetus during the second procedure. Postnatal thorax fosit oran› %1–2 olan bir fetüs intrauterin, iki bebek postnatal dö- tube was placed to 6 patients who had recurrent pleural effusion. One nemde kaybedildi. Ortalama 9 ayl›k izlem sürecinde 11 hastada fetus which had hydrops and whose lymphocyte rate was 1–2% in the komplikasyon geliflmedi. pleural fluid died in the intrauterine period and two newborns died in Sonuç: Fetal plevral effüzyon varl›¤›nda hidrops fetalis olmas›, the postnatal period. No complication developed in 11 patients dur- plevral s›v›da lenfosit oran›n düflük saptanmas› prognozu olumsuz ing the follow-up period which took about 9 months. etkilemektedir. Hidropsla birlikte olan ya da olmayan plevral ef- Conclusion: Having hydrops fetalis in the presence of fetal pleural füzyon varl›¤›nda torakosentez uygulanmas› ve do¤uma yak›n tek- effusion and detecting low lymphocyte rate in the pleural fluid affect rarlanmas› perinatal sonuçlar üzerine olumlu etkisi olmaktad›r. ‹n- the prognosis negatively. Performing thoracocentesis in the presence trauterin fetal tedavi uygun olgularda ailelere önerilmeli ve do¤u- of pleural effusion together with or without hydrops and repeating the mun 3. basamakta yap›lmas› planlanmal›d›r. procedure when the labor is close have a positive impact on the peri- natal outcomes. The families should be recommended intrauterine fetal treatment for the appropriate cases and the labor should be planned in a tertiary center. Keywords: Pleural effusion, hydrops, thoracocentesis. Anahtar sözcükler: Plevral effüzyon, hidrops, torakosentez. Correspondence: Reyhan Ayaz, MD. Department of Perinatology, Faculty of Medicine, Istanbul Medeniyet University, Istanbul, Turkey. e-mail: [email protected] / Received: December 7, 2019; Accepted: December 31, 2019 Please cite this article as: Ayaz R, Demirci O. Perinatal outcomes in the fetuses diagnosed with hydrops fetalis and isolated pleural effusion. Perinatal Journal 2019;27(3):183–188. doi:10.2399/prn.19.0273010 ORCID ID: R. Ayaz 0000-0001-5261-1792; O. Demirci 0000-0001-5578-4437 Ayaz R, Demirci O Introduction trimesters in the Perinatology Clinics of Zeynep Kamil Maternity and Pediatric Training and Research Hospital Fetal primary pleural effusion (PE) is the accumulation of the University of Health Sciences and Medeniyet of fluid in the pleural cavity, and it is called as hydrotho- University between January 2017 and October 2019 were rax in the antenatal period and chylothorax in the post- included in the study. The data of the newborns during natal period. It is a rare condition with an incidence of [1] pregnancy and postnatal period were accessed from the 1/15,000. Its clinical course varies, and it may regress hospital database retrospectively. Hydrops, was consid- spontaneously or result in fetal death. Its optimal treat- [2] ered as the fluid accumulation, coexisting with the pleu- ment method is unclear. ral effusion, in the multiple areas such as pericardium, Primary hydrothorax develops after the leakage of subcutaneous regions and abdomen. The patients were lymphatic fluid between parietal and visceral pleural mem- examined and checked in the antenatal period for blood brane as a result of the increased intrathoracic pressure, types, detailed fetal anatomic screening, middle cerebral and it can be unilateral or bilateral. Most of the cases are artery peak systolic velocity (PSV) to rule out the fetal displayed ultrasonographically as anechoic fluid collection [3] anemia by Doppler ultrasonography, fetal echocardiogra- in the pleural area in the third trimester. It may be a part phy, lymphocyte level in the pleural effusion fluid, and of the non-immune hydrops fetalis. The pleural effusion CMV, Parvovirus, Toxoplasma, Syphilis, Rubella, Herpes developing in association with infections, fetal anemia, simplex immunoglobulin M and immunoglobulin G. chromosomal diseases, fetal malformations and intratho- Conventional karyotyping was carried out but array racic masses (cystic adenomatoid malformation, diaphrag- CGH was not evaluated. The data on the maternal age, matic hernia, etc.) are defined as the secondary pleural gravida, parity, week of gestation during treatment, lym- [4,5] effusion. Depending on the severity of the pleural effu- phocyte rate in the pleural fluid, delivery week, and tho- sion, the esophagus undergoes pressure and congestive racocentesis number were obtained from the hospital heart failure develops, and secondary polyhydramnios, dis- records. The birth weight, Apgar scores, the need for [6–11] ruption in the venous return and hydrops develop. postnatal thorax tube, sex, delivery type, the period The development of pleural effusion in the early weeks between thoracocentesis and labor, hospitalization period of gestation, presence of hydrops findings and preterm at the newborn intensive care unit and intubation period labor are associated with the poor prognosis.[1,6] The prog- were evaluated. Thoracocentesis procedure was carried nosis changes in the secondary PE depending on the out on the fetuses only meeting following criteria: (1) the underlying cause. It was reported that performing cases having pleural effusion previously and progressing drainage in the cases with hydrops, severe pleural effusion, to hydrops, and which were thought that the reason of mediastinal shift and polyhydramnios improves the peri- hydrops was the isolated pleural effusion, (2) the presence natal outcomes.[12–14] It is known that neonatal mortality of fluid in more than 50% of the rib cage causing repul- increases in the cases, who do not undergo any interven- sion in the mediastinum without hydrops, (3) the cases tion, due to pulmonary immaturity and preterm labor.[15] developing polyhydramnios quickly (8 cm above the The treatment options are controversial as it is a rare find- deepest vertical pocket or 25 cm above the total amniot- ing, and therefore the methods such as thoracocentesis, ic fluid index), (4) the cases without any structural anom- thoracoamniotic shunt, and pleurodesis by OK-432 can aly in other organs or systems, and (4) the fetuses with be used for