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Intrauterine decompression of megalocystis during the second trimester of pregnancy
Volume Number
Communications
144 3
in brief
355
Bieber, F. R., and Redwine, F. 0.: Neural tube defects in two offspring of an affected female, Lancet 2:1202, 1980. Wald, N., Cuckle, M., Boreman, J., and Stirrat, G.: Small biparietal diameter of fetuses with spina bifida: Implication for antenatal screening, Br. J. Obstet. Gynaecol. 87:219, 1980. Hobbins, J. C., Campbell, S., Platt, L. D., Sabbagha, R., Bezjian, A., Miskin, M., Deter, R., Kremkau, F., Hansman, M., and Gough, D.: Proceedings of Third Scarborough Meeting on AFP. In Press.
Intrauterine decompression of megalocystis during the second trimester of pregnancy NANCY CHARLES
A.
GADZIALA, Y.
FREDERICK DOUGLAS
J.
M.D.
DOHERTY, KOZA.
*
J”‘. Fig. 1. Sagittal scan at 26 weeks’ gestation, markedly distended bladder(B) compressing
M.D.
KAWADA, J.
.
demonstrating the thorax.
M.D.
M.D.
Departments of Obsteh-ics and Gynecology and Radiology, Tufts University School of Medicine and the New England Medical Center Hospital, and the Department of Radiology, St. Margaret’s Hospital, Boston, Massachwetts
FETAL MEGALOCYSTIS has been diagnosed in utero by ultrasound with successful intrauterine decompression described in the third trimester.’ However, we know of no previous case where successful bladder decompression was performed during the second trimester with continuation of the pregnancy to term. The following case describes a successful bladder decompression in a 27-week fetus subsequently born with the prune-belly syndrome. A 23-year-old black woman, gravida 2, para 1, underwent routine ultrasound examination at 21 weeks’ gestation. A large intra-abdominal sonolucent area was noted and did not decompress during the course of the study. Because of problems with patient compliance, the study was not repeated until 27 weeks’ gestation, at which time a massive sonolucent area measuring 18 by 16 by 16 cm was demonstrated; the mass filled the fetal abdomen and compressed the thoracic cavity (Fig. 1). There was minimal fetal activity with occasional fetal heart decelerations to 80 bpm. The biparietal diameter was consistent with 26 weeks’ gestation. Oligohydramnios was also present, suggesting some type of urinary tract impairment. In view of the ultrasound findings and the threat to fetal viability, decompression of the bladder was performed with an 18gauge Amplatz needle under ultrasound guidance. Once the needle was positioned in the bladder, a polyethylene catheter was advanced into the cavity and the needle was removed. A total of 1,000 ml of clear, slightly yellow fluid was drained from the cavity. After the catheter was withdrawn, ultrasound scans revealed residual fluid in the collapsed bladder, mildly hydronephrotic kidneys, and fetal ascites presumably from intra-abdominal leakage of urine (Fig. 2). Analysis of the fluid
Reprint requests: Charles Y. Kawada, M.D., Department Obstetrics and Gynecology, Tufts New England Medical ter, 17 1 Harrison Ave., Boston, Massachusetts 02111. 0002-9378/82/190355+02$00.20/O
0
of Cen-
1982 The C. V. Mosby Co.
Fig. 2. Ultrasound sion. P, Placenta;
scan immediately after bladder decompresabd. wall, abdominal wall; and B, bladder.
showed a pH of 8, a specific gravity of 1.004, a few white blood cells, and a few epithelial cells. Tests for glucose, protein, and albumin were negative. On subsequent ultrasound examinations done 1 and 6 weeks after decompression, the bladder remained dilated but stable, measuring 5 by 7 by 5 cm. Ten weeks after drainage, at 37 weeks’ gestation, the bladder increased in size to 15 by 11 by 10 cm. Oligohydramnios was again noted. Amniocentesis was performed under ultrasound guidance in the suprapubic region, and 3 ml of clear fluid was obtained. The lecithin/ sphingomyelin (L/S) ratio could not be determined, suggesting possible pulmonary hypoplasia. The patient then went into spontaneous labor. The fetal bladder was again decompressed in an attempt to prevent abdominal dystocia, and 550 ml of clear fluid was obtained. A 3,140 gm baby boy was delivered spontaneously with Apgar scores of 1 and 5. The abdominal girth measured 45 cm. The prune-belly triad, including absence of abdominal wall musculature, undescended testes, and uretheral obstruction was present.2 Renal function was normal, and the baby showed no evidence of pulmonary hypoplasia. There were, however, other abnormalities consisting of meconium ileus with microcolon, hypoplasia of the biliary tract, and a fistula in the abdominal wall created by the initial tap.
356
Communications
in brief
October Am. J. Obstet.
Megalocystis has been associated with pulmonary hypoplasia secondary either to extrinsic pressure on the fetal thorax or to the oligohydramnios itself. The compression of the abdomen and thorax was so severe in this case that it was felt to be incompatible with life. Decompression of the bladder apparently was lifesaving, and the presence of the fistulous tract allowed enough continued drainage for the fetus to reach 37 weeks’ gestation without repeat intervention. A recent article described the use of an indwelling suprapubic catheter that was placed into the fetus, which would minimize the need for repeat intervention.’ The inability to determine the L/S ratio is an interesting finding in that it did not reflect the pulmonary status of the baby. There is a strong possibility that the maternal bladder may have been inadvertently tapped. No amniotic fluid was present during labor or delivery to confirm these results. Although vaginal delivery has been advised with the prune-belly syndrome, abdominal dystocia was encountered in this case, where the abdominal girth was proportionately much larger than the fetal head. Most of the reported cases of prune-belly syndrome secondary to megalocystis have had poor outcomes, although some infants have done well after correction of the genitourinary anomalies.’ This baby is presently doing well at 6 months of age. In addition to the lack of abdominal musculature, he has an elevated bilirubin level secondary to biliary atresia and also has a suprapubic cystotomy. Present plans are to readmit him at 1 year of age to correct the genitourinary anomalies. How aggressively one should manage a case such as this is difficult to determine. Our experience demonstrates the feasibility of intrauterine intervention in the second trimester with continuation of the pregnancy until term. REFERENCES
1. Golbus, M. S., Harrison, M. R., Filly, R. A., et al.: In utero treatment of urinary tract obstruction, AM. J. OBSTET. GYNECOL.
142:383,
1982.
2. Eagle, J. F., and Barrett, G. S.: Congenital deficiency of abdominal musculature with associated genito-urinary abnormalities: A syndrome, Pediatrics 6:721, 1950.
Intrauterine fetal tachypnea ROBERTO ROMERO, M.D. FRANK A. CHERVENAK, M.D. RICHARD L. BERKOWITZ, M.D. JOHN C. HOBBINS, M.D. Department of Obstetrics and Gynecology, Yale University School of Medicine, New Haven, Connecticut Reprint requests: Roberto Romero, M.D., Yale University School of Medicine, Department of Obstetrics and Gynecology, 333 Cedar St., P. 0. Box 3333, New Haven, Connecticut 06510. 000%9378/82/190356+02$00.20/O
0
1982 The C.V.Mosby
Co
1, 1982 Gynecol.
RECENT DEVELOPMENTS in real-time gray-scale ultrasonography have made possible the observation and study of fetal breathing movements. Research has been directed toward fetal breathing movements as an indicator of fetal well-being. The normal rate of fetal breathing has been defined by Manning and associates’ to be 12 to 60 breaths per minute. Much attention has been directed toward decreased fetal breathing movement as a predictor of fetal compromise. However, observations of abnormally increased fetal breathing movements have been limited to two previously reported cases.‘, 2 Two further cases of intrauterine fetal tachypnea are presented and clinical implications are discussed. Case I. A 20-year-old, gravida 2, para I-O-O- 1, Class A diabetic woman was admitted at 36 weeks of gestation after a nonreactive nonstress test and an equivocal contraction stress test.
Past obstetric history revealed a spontaneous vaginal delivery after a term gestation during which the patient was diagnosed as having gestational diabetes. Past medical and surgical history was not remarkable. Family history revealed the patient’s grandmother to have diabetes. During this pregnancy results of a 3-hour glucose tolerance test performed at 27 weeks of gestation were normal. Plasma glucose values were as follows: fasting, 96 mg/dl; 1 hour, 144 mgidl; 2 hours, 90 mg/dl; and 3 hours, 119 mg/dl. Because of glycosuria in a random urine sample and the previous history of gestational diabetes, a repeat 3-hour glucose tolerance test was performed at 35 weeks; plasma glucose values were as follows: fasting, 100 mg/dl; 1 hour, 226 mg/dl; 2 hours, 250 mg/dl; and 3 hours, 199 mg/dl. A test for hemoglobin A,C performed at this time showed a value of 9.0% (normal value, 4% to 8%). The patient was placed on a 2,200 calorie American Diabetic Association diet. A nonstress test was nonreactive and a contraction stress test was equivocal. Because of the gestational diabetes at 36 weeks of gestation and an equivocal contraction stress test, an amniocentesis for determination of the lecithin/sphingomyelin (L/S) ratio was performed. The following day the L/S ratio was 5.0, a repeat
contraction stress test was positive, and the decision was made to induce labor. Four hours after the beginning of the induction, there was difficulty in obtaining an adequate fetal heart rate tracing with the external monitor. With Doppler ultrasound, the nursing staff detected unusual frequency changes in the fetal heart rate. A real-time linear array transducer was applied, and intermittent episodes of fetal breathing movements at a rate of 80 to 90 per minute were detected. Breathing movements were shallow. The fetal heart rate was 140 bpm. The plasma glucose level at this time was 160 mg/dl. Artificial rupture of the membranes was performed. An internal fetal heart rate tracing revealed moderate cord patterns and occasional late decelerations. A scalp pH was 7.26. Subsequently, the late decelerations recurred, and a scalp sample pH was 7.13. An emergency cesarean section was performed. A female infant, with Apgar scores of 5 and 7, was delivered: the umbilical cord pH was 7.20. The respiratory rate of the neonate was 50 bpm. The results of a Dextrostix test dropped to 25 mgidl, but the infant responded rapidly to oral feeding. Ca~r 2. A 29-year old, gravida 3, para l-0-1-1, Class B diabetic woman was transferred to Yale-New Haven Hospital at
Communications
144 3
in brief
355
Bieber, F. R., and Redwine, F. 0.: Neural tube defects in two offspring of an affected female, Lancet 2:1202, 1980. Wald, N., Cuckle, M., Boreman, J., and Stirrat, G.: Small biparietal diameter of fetuses with spina bifida: Implication for antenatal screening, Br. J. Obstet. Gynaecol. 87:219, 1980. Hobbins, J. C., Campbell, S., Platt, L. D., Sabbagha, R., Bezjian, A., Miskin, M., Deter, R., Kremkau, F., Hansman, M., and Gough, D.: Proceedings of Third Scarborough Meeting on AFP. In Press.
Intrauterine decompression of megalocystis during the second trimester of pregnancy NANCY CHARLES
A.
GADZIALA, Y.
FREDERICK DOUGLAS
J.
M.D.
DOHERTY, KOZA.
*
J”‘. Fig. 1. Sagittal scan at 26 weeks’ gestation, markedly distended bladder(B) compressing
M.D.
KAWADA, J.
.
demonstrating the thorax.
M.D.
M.D.
Departments of Obsteh-ics and Gynecology and Radiology, Tufts University School of Medicine and the New England Medical Center Hospital, and the Department of Radiology, St. Margaret’s Hospital, Boston, Massachwetts
FETAL MEGALOCYSTIS has been diagnosed in utero by ultrasound with successful intrauterine decompression described in the third trimester.’ However, we know of no previous case where successful bladder decompression was performed during the second trimester with continuation of the pregnancy to term. The following case describes a successful bladder decompression in a 27-week fetus subsequently born with the prune-belly syndrome. A 23-year-old black woman, gravida 2, para 1, underwent routine ultrasound examination at 21 weeks’ gestation. A large intra-abdominal sonolucent area was noted and did not decompress during the course of the study. Because of problems with patient compliance, the study was not repeated until 27 weeks’ gestation, at which time a massive sonolucent area measuring 18 by 16 by 16 cm was demonstrated; the mass filled the fetal abdomen and compressed the thoracic cavity (Fig. 1). There was minimal fetal activity with occasional fetal heart decelerations to 80 bpm. The biparietal diameter was consistent with 26 weeks’ gestation. Oligohydramnios was also present, suggesting some type of urinary tract impairment. In view of the ultrasound findings and the threat to fetal viability, decompression of the bladder was performed with an 18gauge Amplatz needle under ultrasound guidance. Once the needle was positioned in the bladder, a polyethylene catheter was advanced into the cavity and the needle was removed. A total of 1,000 ml of clear, slightly yellow fluid was drained from the cavity. After the catheter was withdrawn, ultrasound scans revealed residual fluid in the collapsed bladder, mildly hydronephrotic kidneys, and fetal ascites presumably from intra-abdominal leakage of urine (Fig. 2). Analysis of the fluid
Reprint requests: Charles Y. Kawada, M.D., Department Obstetrics and Gynecology, Tufts New England Medical ter, 17 1 Harrison Ave., Boston, Massachusetts 02111. 0002-9378/82/190355+02$00.20/O
0
of Cen-
1982 The C. V. Mosby Co.
Fig. 2. Ultrasound sion. P, Placenta;
scan immediately after bladder decompresabd. wall, abdominal wall; and B, bladder.
showed a pH of 8, a specific gravity of 1.004, a few white blood cells, and a few epithelial cells. Tests for glucose, protein, and albumin were negative. On subsequent ultrasound examinations done 1 and 6 weeks after decompression, the bladder remained dilated but stable, measuring 5 by 7 by 5 cm. Ten weeks after drainage, at 37 weeks’ gestation, the bladder increased in size to 15 by 11 by 10 cm. Oligohydramnios was again noted. Amniocentesis was performed under ultrasound guidance in the suprapubic region, and 3 ml of clear fluid was obtained. The lecithin/ sphingomyelin (L/S) ratio could not be determined, suggesting possible pulmonary hypoplasia. The patient then went into spontaneous labor. The fetal bladder was again decompressed in an attempt to prevent abdominal dystocia, and 550 ml of clear fluid was obtained. A 3,140 gm baby boy was delivered spontaneously with Apgar scores of 1 and 5. The abdominal girth measured 45 cm. The prune-belly triad, including absence of abdominal wall musculature, undescended testes, and uretheral obstruction was present.2 Renal function was normal, and the baby showed no evidence of pulmonary hypoplasia. There were, however, other abnormalities consisting of meconium ileus with microcolon, hypoplasia of the biliary tract, and a fistula in the abdominal wall created by the initial tap.
356
Communications
in brief
October Am. J. Obstet.
Megalocystis has been associated with pulmonary hypoplasia secondary either to extrinsic pressure on the fetal thorax or to the oligohydramnios itself. The compression of the abdomen and thorax was so severe in this case that it was felt to be incompatible with life. Decompression of the bladder apparently was lifesaving, and the presence of the fistulous tract allowed enough continued drainage for the fetus to reach 37 weeks’ gestation without repeat intervention. A recent article described the use of an indwelling suprapubic catheter that was placed into the fetus, which would minimize the need for repeat intervention.’ The inability to determine the L/S ratio is an interesting finding in that it did not reflect the pulmonary status of the baby. There is a strong possibility that the maternal bladder may have been inadvertently tapped. No amniotic fluid was present during labor or delivery to confirm these results. Although vaginal delivery has been advised with the prune-belly syndrome, abdominal dystocia was encountered in this case, where the abdominal girth was proportionately much larger than the fetal head. Most of the reported cases of prune-belly syndrome secondary to megalocystis have had poor outcomes, although some infants have done well after correction of the genitourinary anomalies.’ This baby is presently doing well at 6 months of age. In addition to the lack of abdominal musculature, he has an elevated bilirubin level secondary to biliary atresia and also has a suprapubic cystotomy. Present plans are to readmit him at 1 year of age to correct the genitourinary anomalies. How aggressively one should manage a case such as this is difficult to determine. Our experience demonstrates the feasibility of intrauterine intervention in the second trimester with continuation of the pregnancy until term. REFERENCES
1. Golbus, M. S., Harrison, M. R., Filly, R. A., et al.: In utero treatment of urinary tract obstruction, AM. J. OBSTET. GYNECOL.
142:383,
1982.
2. Eagle, J. F., and Barrett, G. S.: Congenital deficiency of abdominal musculature with associated genito-urinary abnormalities: A syndrome, Pediatrics 6:721, 1950.
Intrauterine fetal tachypnea ROBERTO ROMERO, M.D. FRANK A. CHERVENAK, M.D. RICHARD L. BERKOWITZ, M.D. JOHN C. HOBBINS, M.D. Department of Obstetrics and Gynecology, Yale University School of Medicine, New Haven, Connecticut Reprint requests: Roberto Romero, M.D., Yale University School of Medicine, Department of Obstetrics and Gynecology, 333 Cedar St., P. 0. Box 3333, New Haven, Connecticut 06510. 000%9378/82/190356+02$00.20/O
0
1982 The C.V.Mosby
Co
1, 1982 Gynecol.
RECENT DEVELOPMENTS in real-time gray-scale ultrasonography have made possible the observation and study of fetal breathing movements. Research has been directed toward fetal breathing movements as an indicator of fetal well-being. The normal rate of fetal breathing has been defined by Manning and associates’ to be 12 to 60 breaths per minute. Much attention has been directed toward decreased fetal breathing movement as a predictor of fetal compromise. However, observations of abnormally increased fetal breathing movements have been limited to two previously reported cases.‘, 2 Two further cases of intrauterine fetal tachypnea are presented and clinical implications are discussed. Case I. A 20-year-old, gravida 2, para I-O-O- 1, Class A diabetic woman was admitted at 36 weeks of gestation after a nonreactive nonstress test and an equivocal contraction stress test.
Past obstetric history revealed a spontaneous vaginal delivery after a term gestation during which the patient was diagnosed as having gestational diabetes. Past medical and surgical history was not remarkable. Family history revealed the patient’s grandmother to have diabetes. During this pregnancy results of a 3-hour glucose tolerance test performed at 27 weeks of gestation were normal. Plasma glucose values were as follows: fasting, 96 mg/dl; 1 hour, 144 mgidl; 2 hours, 90 mg/dl; and 3 hours, 119 mg/dl. Because of glycosuria in a random urine sample and the previous history of gestational diabetes, a repeat 3-hour glucose tolerance test was performed at 35 weeks; plasma glucose values were as follows: fasting, 100 mg/dl; 1 hour, 226 mg/dl; 2 hours, 250 mg/dl; and 3 hours, 199 mg/dl. A test for hemoglobin A,C performed at this time showed a value of 9.0% (normal value, 4% to 8%). The patient was placed on a 2,200 calorie American Diabetic Association diet. A nonstress test was nonreactive and a contraction stress test was equivocal. Because of the gestational diabetes at 36 weeks of gestation and an equivocal contraction stress test, an amniocentesis for determination of the lecithin/sphingomyelin (L/S) ratio was performed. The following day the L/S ratio was 5.0, a repeat
contraction stress test was positive, and the decision was made to induce labor. Four hours after the beginning of the induction, there was difficulty in obtaining an adequate fetal heart rate tracing with the external monitor. With Doppler ultrasound, the nursing staff detected unusual frequency changes in the fetal heart rate. A real-time linear array transducer was applied, and intermittent episodes of fetal breathing movements at a rate of 80 to 90 per minute were detected. Breathing movements were shallow. The fetal heart rate was 140 bpm. The plasma glucose level at this time was 160 mg/dl. Artificial rupture of the membranes was performed. An internal fetal heart rate tracing revealed moderate cord patterns and occasional late decelerations. A scalp pH was 7.26. Subsequently, the late decelerations recurred, and a scalp sample pH was 7.13. An emergency cesarean section was performed. A female infant, with Apgar scores of 5 and 7, was delivered: the umbilical cord pH was 7.20. The respiratory rate of the neonate was 50 bpm. The results of a Dextrostix test dropped to 25 mgidl, but the infant responded rapidly to oral feeding. Ca~r 2. A 29-year old, gravida 3, para l-0-1-1, Class B diabetic woman was transferred to Yale-New Haven Hospital at