Antenatal treatment of chylothorax and cystic hygroma with OK-432 in nonimmune hydrops fetalis.
OBJECTIVE
To present our experience of using OK-432 in treating fetal cystic hygroma and chylothorax complicated with nonimmune hydrops fetalis.


METHODS
OK-432 (Picibanil) was injected into the fetal pleural cavity or fetal cystic hygroma.


RESULTS
Patient 1: A 23-year-old, gravida 2, para 1, was found to have a recurrent fetal chylothorax at GA 29 weeks. Serial amnioreduction and thoracocentesis was performed at GA 31, 32, 33, and 34 weeks. Intrapleural OK-432 injection was performed twice at GA 33 and 34 weeks. Cyanosis and respiratory distress were noted immediately after birth (GA 34 weeks). The baby expired despite of aggressive neonatal resuscitation. Patient 2: A 26-year-old, gravida 2, para 1, was found to have a cystic hygroma of her fetus at GA 17 weeks. Karyotype of the cystic fluid and the amniocytes were 46, XY. Fetal ascites developed at GA 22 weeks. OK-432 injection into the tumour was performed at GA 23 weeks. Stabilization of the cystic hygroma was noted throughout the pregnancy (about 3.5 cm in diameter). Serial fetal paracentesis and/or amnioreduction were performed. Karyotype of the ascites was again 46, XY. Maternal dietary modification with medium chain triglyceride was also prescribed. Chylothorax developed and the baby was born by cesareans at GA 32 weeks. Resolution of pleural effusion, ascites, and regression of cystic hygroma were noted since the 2nd day after birth. The baby had survived beyond 4 months of age at submission.


CONCLUSIONS
Combination of antenatal OK-432 injection, maternal dietary modification, serial thoracocentesis plus paracentesis, together with amnioreduction and tocolysis, appeared to contribute to the success of antenatal treatment. Fetal pulmonary expansion may determine the immediate neonatal survival.
Department of Medical Genetics, National Taiwan University Hospital, Taipei, Taiwan. mchen_cch@yahoo.com
Ming
M
Chen
Chih-Ping
CP
Chen
Jin-Chung
JC
Shih
Hung-Chieh
HC
Chou
Chia-Li
CL
Yu
Bao-Tyan
BT
Wang
Chang-Yao
CY
Hsieh
D000328:Adult; D000970:Antineoplastic Agents / Q000008:administration & dosage*; D002916:Chylothorax / Q000000981:diagnostic imaging / Q000188:drug therapy*; D005260:Female; D005315:Fetal Diseases / Q000000981:diagnostic imaging / Q000188:drug therapy; D006801:Humans; D015160:Hydrops Fetalis / Q000000981:diagnostic imaging / Q000188:drug therapy*; D007231:Infant, Newborn; D018191:Lymphangioma, Cystic / Q000000981:diagnostic imaging / Q000188:drug therapy*; D008223:Lymphoma / Q000000981:diagnostic imaging / Q000188:drug therapy*; D008297:Male; D010844:Picibanil / Q000008:administration & dosage*; D011247:Pregnancy; D016216:Ultrasonography, Prenatal
D000970:Antineoplastic Agents; D010844:Picibanil