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Serial transabdominal amnioinfusion in the management of gastroschisis with severe oligohydramnios
Serial Transabdominal Gastroschisis By Marc Dommergues,
Amnioinfusion in the Management With Severe Oligohydramnios
Yann Ansker, Marie C6cile Aubry, Jaques Mac Aleese, Claire Nihoul-F6kBt6, and Yves Dumez Paris, France
0 Two fetuses with gastroschisis diagnosed in utero (at 19 weeks’ gestation) had severe oligohydramnios at 30 to 31 weeks. Serial transabdominal amnioinfusions were performed to fill the amniotic cavity with saline, thereby avoiding the potential consequences of fetal exposure to severe oligohydramnios. In both cases, premature rupture of membranes occurred at 36 weeks, and the fetuses were delivered by cesarean section. There were minimal lesions of the extraabdominal bowel. After primary closure of the abdomen, the postoperative course was uneventful. These observations show that serial amnioinfusion is a feasible therapeutic approach for severe third-trimester oligohydramnios associated with gastroschisis. Copyright o 1996 by W.B. Saunders Company INDEX WORDS: Gastroschisis, sion, fetal therapy.
oligohydramnios,
amnioinfu-
ASTROSCHISIS can be complicated by oligohydramnios in up to 25% of pregnancies.’ In some G cases, oligohydramnios may be severe enough to put the fetus at risk for pulmonary hypoplasia, limb deformities,2x3 and fetal distress owing to cord compression. We present two cases in which severe oligohydramnios associated with fetal gastroschisis was treated in utero by serial transabdominal amnioinfusions of saline. This therapeutic approach was considered successful; both pregnancies could be prolonged to 36 weeks and resulted in the births of neonates free of complications of chronic exposure to severe oligohydramnios. In addition, bowel lesions were minimal in both cases. CASE REPORTS Case 1 A gravida 1, 21 year old was referred at 19 weeks, after gastroschisis had been diagnosed by routine ultrasonography. The fetal karyotype was normal 46,Xx, and no associated malformation was found by ultrasonography. Biparietal diameter and femur length were appropriate for gestational age. The gastroschisis (34 x 16 mm in diameter) contained only bowel. There was no significant dilatation of the small bowel, no intestinal mural thickness, and the intestinal content appeared transonic. Amniotic fluid volume was normal. The kidneys and bladder appeared normal and remained so throughout the pregnancy. At 27 weeks, cephalic and femur measurements were between the third and tenth percentile for gestational age. Intraabdominal as well as extraabdominal bowel loops were moderately dilated, but there was no mural thickness or modification of the bowel content. The diameter of the largest intestinal loop was 17 mm. Umbilical and uterine artery Doppler waveforms were normal. The oligohydramnios was moderate. JournalofPediatric
Surgery,
Vol31,
No 9 (September),
1996:
pp 1297-1299
Stephen
of
Lot-tat-Jacob,
At 30.5 weeks, severe oligohydramnios was diagnosed during a follow-up ultrasound examination. There was a single amniotic fluid pool of less than 1 cm. The fetal growth, umbilical Doppler result, and sonographic appearance of the bowel were similar to the previous findings. Transabdominal amnioinfusion of 500 mL of warm saline was performed under ultrasound guidance, using a 20-gauge needle inserted under local anesthesia, as described previously.4x5Weekly sonograms showed a progressive decrease in amniotic fluid volume, and amnioinfusion (of 350 mL) had to be repeated at 34 and 35 weeks (Table 1). At 36 weeks the amniotic fluid volume was normal ultrasonographically. One extraabdominal bowel loop was dilated (23 mm) and contained echogenic material. There was no mural thickening of the extraabdominal bowel. Diastolic flow was identified in the extraabdominal mesenteric artery. Fetal measurements corresponded to the tenth percentile for gestational age, and umbilical Doppler findings were normal. Premature rupture of membranes occurred at 36 weeks. Two days later, cesarean section was performed because of an unripe cervix and ruptured membranes. A 2,450-g girl was delivered and immediately transferred to the surgical unit. Her 5-minute Apgar score was 10. The exteriorized bowel was normal morphologically, and there was no bowel thickening or fibrous peel. Primary closure was performed on day 1. Oral nutrition was begun on day 22. Parenteral nutrition was discontinued on day 34. The baby was discharged on day 54, with normal intestinal transit and appropriate growth (weight, 3,100 g). Case 2 A 23-year-old primigravida was referred at 19 weeks’ gestation because gastroschisis had been suspected during routine ultrasound examination. The fetal karyotype was normal 46,XY, and no associated malformation was found by ultrasonography. Biparietal diameter and femur length were appropriate for gestational age. The gastroschisis contained only bowel. There was no significant bowel distension, no mural thickness, and the intestinal content appeared transonic. Amniotic fluid volume was normal. The kidneys and bladder appeared normal and remained so throughout the pregnancy. At 27.5 weeks, cephalic and femur measurements were between the tenth and twenty-fifth percentile for gestational age. The bowel was not significantly dilated. The maximal diameter of the intestinal loops was 6 mm, without mural thickness or modification of the bowel content. Umbilical artery Doppler waveforms were normal. There was moderate oligohydramnios. At 31.5 weeks, severe oligohydramnios was diagnosed, with a single amniotic fluid pool of less than 1 cm. Cephalic and femur measurements were between the third and twenty-fifth percentile,
From the Matemite’ Port Royal-Baudeloque, the Institut de Pu&iculture, and the Service de Chirurgie Infantile, HGpital Necker Enfants Malades, Paris, France. Address reprint requests to Mare Dommergues, MD, Mater&k Port Royal-Baudeloque, 123 Boulevard Port Royal, 75014 Paris, France. Copyright o 1996 by W B. Saunders Company 0022-3468/9613109-0026$03.00/O 1297
DOMMERGUES
1298
Table
1. Amniotic
Fluid
Volume
and
Amounts
of Amnioinfusion
ET AL
count for the increased rate of fetal distress.’ Oligohydramnios is documented in up to 25% of cases, but it usually is moderate and therefore has no major Case 1 30.5
Age (wk)
AF Index (cm)
Amnioinfusion
(mL)
TRANSABDOMINAL
AMNIOINFUSION
AND
GASTROSCHISIS
1299
natural amniotic fluid and its replacement by saline would create a favorable condition for the gastroschisis. In the cases reported herein, this was achieved during 4 to 5 weeks, and the absence of significant bowel peeling could be a secondary end point for determining the success of the amnioinfusions. In addition, although no definitive conclusion can be drawn from the fact that the sonographic appearance of the extraabdominal bowel in case 2 improved after the second amnioinfusion, this might be interpreted
as sonographic evidence of the potential beneficial effect of amnioinfusion on fetal bowel. In conclusion, our experience is too limited to suggest that amnioinfusion could be performed in the presence of a normal amniotic compartment in order to protect bowel by fluid exchange. Such a hypothesis would require testing through experimental animal studies. However, our results indicate that serial amnioinfusion may be beneficial in selected cases of gastroschisis associated with severe oligohydramnios.
REFERENCES 1. Crawford RAF, Ryan G, Wright VM, et al: The importance of serial biophysical assessment of fetal wellbeing in gastroschisis. Br J Obstet Gynaecol99:899-902,1992 2. Thibeault D, Beatty E, Hall R, et al: Neonatal pulmonary hypoplasia with premature rupture of fetal membranes and oligohydramnios. J Pediatr 107:273-277, 1985 3. Chamberlain PF, Manning FA, Morrison L, et al: The relationship of marginal and decreased amniotic fluid to perinatal outcome. Am J Obstet Gynecol 150:245-249,1984 4. Mandelbrot L, Dommergues M, Dumez Y: Prepartum transabdominal amnioinfusion for severe oligohydramnios. Acta Obstet Gynecol Stand 71:124-125,1992 5. Mandelbrot L, Verspick E, Dommergues M, et al: Transabdominal amnioinfusion for the management of nonlaboring postdates with severe oligohydramnios. Fetal Diagn Ther 8:412-417, 1993 6. Blanc CE, Wesley JR, DiPietro MA, et al: Gastrointestinal complications of laparoschisis. AJR 144:589-591, 1985 7. Swartz K, Harrison M, Campbell T, et al: Selective management of gastroschisis. Ann Surg 203:214-218,1986 8. Sipes S, Weiner C, Sipes I, et al: Gastroschisis and omphalocle: Does either antenatal diagnosis or route of delivery make a difference in perinatal outcome? Obstet Gynecol76:195-199, 1990 9. Fries MH, Filly RA, Callen PW, et al: Growth retardation in prenatally diagnosed cases of gastroschisis. J Ultrasound Med 12:583-588, 1993
10. Sakala EP, Erhard LN, White JJ: Elective cesarean section improves outcomes of neonates with gastroschisis. Am J Obstet Gynecol169:1050-1053,1993 11. Sapin E, Lewin F, Baron JM, et al: Prenatal diagnosis and management of gastroschisis. Pediatr Surg Int 8:31-33,1993 12. Moore T: The role of labor in gastroschisis bowel thickening and prevention by elective pre-term and pre-labor cesarean section. Pediatr Surg Int 7:256-259,199Z 13. Tibboel D, McNee M, Azmy A, et al: Developmental aspects of gastroschisis. J Pediatr Surg 21:865-869, 1986 14. Langer JC, Longaker M, Cromblehome T, et al: Etiology of intestinal damage in gastroschisis, I: Effects of amniotic fluid exposure and bowel constriction in a fetal lamb model. J Pediatr Surg 24:992-997,1989 15. Langer JC, Bell JG, Castillo R. et al: Etiology of intestinal damage in gastroschisis, II: Timing and reversibility of histological changes, mucosal function, and contractility. J Pediatr Surg25:11221126,199O 16. Kluck P, Tibboel D, van der Kamp A, et al: The effect of fetal urine on the development of the bowel in gastroschisis. J Pediatr Surg 18:47-50,1983 17. Aktug T, Erdag G, Kargi A, et al: Amnio-allantoic exchange for the prevention of intestinal damage in gastroschisis: An experimental study on chick embryos. J Pediatr Surg 30:384-387, 1995
Amnioinfusion in the Management With Severe Oligohydramnios
Yann Ansker, Marie C6cile Aubry, Jaques Mac Aleese, Claire Nihoul-F6kBt6, and Yves Dumez Paris, France
0 Two fetuses with gastroschisis diagnosed in utero (at 19 weeks’ gestation) had severe oligohydramnios at 30 to 31 weeks. Serial transabdominal amnioinfusions were performed to fill the amniotic cavity with saline, thereby avoiding the potential consequences of fetal exposure to severe oligohydramnios. In both cases, premature rupture of membranes occurred at 36 weeks, and the fetuses were delivered by cesarean section. There were minimal lesions of the extraabdominal bowel. After primary closure of the abdomen, the postoperative course was uneventful. These observations show that serial amnioinfusion is a feasible therapeutic approach for severe third-trimester oligohydramnios associated with gastroschisis. Copyright o 1996 by W.B. Saunders Company INDEX WORDS: Gastroschisis, sion, fetal therapy.
oligohydramnios,
amnioinfu-
ASTROSCHISIS can be complicated by oligohydramnios in up to 25% of pregnancies.’ In some G cases, oligohydramnios may be severe enough to put the fetus at risk for pulmonary hypoplasia, limb deformities,2x3 and fetal distress owing to cord compression. We present two cases in which severe oligohydramnios associated with fetal gastroschisis was treated in utero by serial transabdominal amnioinfusions of saline. This therapeutic approach was considered successful; both pregnancies could be prolonged to 36 weeks and resulted in the births of neonates free of complications of chronic exposure to severe oligohydramnios. In addition, bowel lesions were minimal in both cases. CASE REPORTS Case 1 A gravida 1, 21 year old was referred at 19 weeks, after gastroschisis had been diagnosed by routine ultrasonography. The fetal karyotype was normal 46,Xx, and no associated malformation was found by ultrasonography. Biparietal diameter and femur length were appropriate for gestational age. The gastroschisis (34 x 16 mm in diameter) contained only bowel. There was no significant dilatation of the small bowel, no intestinal mural thickness, and the intestinal content appeared transonic. Amniotic fluid volume was normal. The kidneys and bladder appeared normal and remained so throughout the pregnancy. At 27 weeks, cephalic and femur measurements were between the third and tenth percentile for gestational age. Intraabdominal as well as extraabdominal bowel loops were moderately dilated, but there was no mural thickness or modification of the bowel content. The diameter of the largest intestinal loop was 17 mm. Umbilical and uterine artery Doppler waveforms were normal. The oligohydramnios was moderate. JournalofPediatric
Surgery,
Vol31,
No 9 (September),
1996:
pp 1297-1299
Stephen
of
Lot-tat-Jacob,
At 30.5 weeks, severe oligohydramnios was diagnosed during a follow-up ultrasound examination. There was a single amniotic fluid pool of less than 1 cm. The fetal growth, umbilical Doppler result, and sonographic appearance of the bowel were similar to the previous findings. Transabdominal amnioinfusion of 500 mL of warm saline was performed under ultrasound guidance, using a 20-gauge needle inserted under local anesthesia, as described previously.4x5Weekly sonograms showed a progressive decrease in amniotic fluid volume, and amnioinfusion (of 350 mL) had to be repeated at 34 and 35 weeks (Table 1). At 36 weeks the amniotic fluid volume was normal ultrasonographically. One extraabdominal bowel loop was dilated (23 mm) and contained echogenic material. There was no mural thickening of the extraabdominal bowel. Diastolic flow was identified in the extraabdominal mesenteric artery. Fetal measurements corresponded to the tenth percentile for gestational age, and umbilical Doppler findings were normal. Premature rupture of membranes occurred at 36 weeks. Two days later, cesarean section was performed because of an unripe cervix and ruptured membranes. A 2,450-g girl was delivered and immediately transferred to the surgical unit. Her 5-minute Apgar score was 10. The exteriorized bowel was normal morphologically, and there was no bowel thickening or fibrous peel. Primary closure was performed on day 1. Oral nutrition was begun on day 22. Parenteral nutrition was discontinued on day 34. The baby was discharged on day 54, with normal intestinal transit and appropriate growth (weight, 3,100 g). Case 2 A 23-year-old primigravida was referred at 19 weeks’ gestation because gastroschisis had been suspected during routine ultrasound examination. The fetal karyotype was normal 46,XY, and no associated malformation was found by ultrasonography. Biparietal diameter and femur length were appropriate for gestational age. The gastroschisis contained only bowel. There was no significant bowel distension, no mural thickness, and the intestinal content appeared transonic. Amniotic fluid volume was normal. The kidneys and bladder appeared normal and remained so throughout the pregnancy. At 27.5 weeks, cephalic and femur measurements were between the tenth and twenty-fifth percentile for gestational age. The bowel was not significantly dilated. The maximal diameter of the intestinal loops was 6 mm, without mural thickness or modification of the bowel content. Umbilical artery Doppler waveforms were normal. There was moderate oligohydramnios. At 31.5 weeks, severe oligohydramnios was diagnosed, with a single amniotic fluid pool of less than 1 cm. Cephalic and femur measurements were between the third and twenty-fifth percentile,
From the Matemite’ Port Royal-Baudeloque, the Institut de Pu&iculture, and the Service de Chirurgie Infantile, HGpital Necker Enfants Malades, Paris, France. Address reprint requests to Mare Dommergues, MD, Mater&k Port Royal-Baudeloque, 123 Boulevard Port Royal, 75014 Paris, France. Copyright o 1996 by W B. Saunders Company 0022-3468/9613109-0026$03.00/O 1297
DOMMERGUES
1298
Table
1. Amniotic
Fluid
Volume
and
Amounts
of Amnioinfusion
ET AL
count for the increased rate of fetal distress.’ Oligohydramnios is documented in up to 25% of cases, but it usually is moderate and therefore has no major Case 1 30.5
Age (wk)
AF Index (cm)
Amnioinfusion
(mL)
TRANSABDOMINAL
AMNIOINFUSION
AND
GASTROSCHISIS
1299
natural amniotic fluid and its replacement by saline would create a favorable condition for the gastroschisis. In the cases reported herein, this was achieved during 4 to 5 weeks, and the absence of significant bowel peeling could be a secondary end point for determining the success of the amnioinfusions. In addition, although no definitive conclusion can be drawn from the fact that the sonographic appearance of the extraabdominal bowel in case 2 improved after the second amnioinfusion, this might be interpreted
as sonographic evidence of the potential beneficial effect of amnioinfusion on fetal bowel. In conclusion, our experience is too limited to suggest that amnioinfusion could be performed in the presence of a normal amniotic compartment in order to protect bowel by fluid exchange. Such a hypothesis would require testing through experimental animal studies. However, our results indicate that serial amnioinfusion may be beneficial in selected cases of gastroschisis associated with severe oligohydramnios.
REFERENCES 1. Crawford RAF, Ryan G, Wright VM, et al: The importance of serial biophysical assessment of fetal wellbeing in gastroschisis. Br J Obstet Gynaecol99:899-902,1992 2. Thibeault D, Beatty E, Hall R, et al: Neonatal pulmonary hypoplasia with premature rupture of fetal membranes and oligohydramnios. J Pediatr 107:273-277, 1985 3. Chamberlain PF, Manning FA, Morrison L, et al: The relationship of marginal and decreased amniotic fluid to perinatal outcome. Am J Obstet Gynecol 150:245-249,1984 4. Mandelbrot L, Dommergues M, Dumez Y: Prepartum transabdominal amnioinfusion for severe oligohydramnios. Acta Obstet Gynecol Stand 71:124-125,1992 5. Mandelbrot L, Verspick E, Dommergues M, et al: Transabdominal amnioinfusion for the management of nonlaboring postdates with severe oligohydramnios. Fetal Diagn Ther 8:412-417, 1993 6. Blanc CE, Wesley JR, DiPietro MA, et al: Gastrointestinal complications of laparoschisis. AJR 144:589-591, 1985 7. Swartz K, Harrison M, Campbell T, et al: Selective management of gastroschisis. Ann Surg 203:214-218,1986 8. Sipes S, Weiner C, Sipes I, et al: Gastroschisis and omphalocle: Does either antenatal diagnosis or route of delivery make a difference in perinatal outcome? Obstet Gynecol76:195-199, 1990 9. Fries MH, Filly RA, Callen PW, et al: Growth retardation in prenatally diagnosed cases of gastroschisis. J Ultrasound Med 12:583-588, 1993
10. Sakala EP, Erhard LN, White JJ: Elective cesarean section improves outcomes of neonates with gastroschisis. Am J Obstet Gynecol169:1050-1053,1993 11. Sapin E, Lewin F, Baron JM, et al: Prenatal diagnosis and management of gastroschisis. Pediatr Surg Int 8:31-33,1993 12. Moore T: The role of labor in gastroschisis bowel thickening and prevention by elective pre-term and pre-labor cesarean section. Pediatr Surg Int 7:256-259,199Z 13. Tibboel D, McNee M, Azmy A, et al: Developmental aspects of gastroschisis. J Pediatr Surg 21:865-869, 1986 14. Langer JC, Longaker M, Cromblehome T, et al: Etiology of intestinal damage in gastroschisis, I: Effects of amniotic fluid exposure and bowel constriction in a fetal lamb model. J Pediatr Surg 24:992-997,1989 15. Langer JC, Bell JG, Castillo R. et al: Etiology of intestinal damage in gastroschisis, II: Timing and reversibility of histological changes, mucosal function, and contractility. J Pediatr Surg25:11221126,199O 16. Kluck P, Tibboel D, van der Kamp A, et al: The effect of fetal urine on the development of the bowel in gastroschisis. J Pediatr Surg 18:47-50,1983 17. Aktug T, Erdag G, Kargi A, et al: Amnio-allantoic exchange for the prevention of intestinal damage in gastroschisis: An experimental study on chick embryos. J Pediatr Surg 30:384-387, 1995