Koerten et al.
around the inferior retention sutures. Cortisone was decreased from 37 to 25 mg/day. On day 11 she had hematemesis with a blood pressure of 30/0 mm Hg. Nasogastric irrigation and transfusion were unable to stabilize the patient and an antrectomy (Bill roth II procedure) was performed for anterior and posterior gastric ulcers, both penetrating through the wall, with a bleeding gastroduodenal artery. She received 24 units of blood and 7 units of fresh-frozen plasma. Her postoperative course was complicated by pulmonary edema, adult respiratory distress syndrome, recurrent bleeding and fistulization from the anastomosis site, pneumonia with Candida and Klebsiella organisms, and oxygen toxicity of the lungs. Cardiopulmonary arrest and death ensued. Follow-up of the baby revealed a grade 4 intraventricular hemorrhage and bronchopulmonary dysplasia 6 months later.
Comment This is the thirty-third case report and the first report of death in a patient with Cushing's syndrome in pregnancy. A review of the literature in Table I reveals that maternal complications are common. Seven of 16 (47%) patients with adrenal adenoma developed pulmonary edema, and every patient whose pregnancy progressed
March. 1986 Am J Obstet Gyneco1
to the first trimester experienced hypertension. Maternal complications were much less common in patients with adrenal hyperplasia, with only one of 12 patients developing hypertension. There was a high incidence of prematurity with only 13% term births. This may be due to 17-hydroxylase induction in high-cortisol states, described by Liggins. 2 Early diagnosis and prompt treatment are of vital importance. Prompt operation or pituitary irradiation during pregnancy yielded five term births, two premature infants, and one spontaneous abortion, giving a 62.5% term birth rate. Medical treatment was attempted by Gormley with metyrapone. This treatment appears to be an especially viable alternative in poor surgical candidates or in stabilizing patients prior to operation. Prophylactic cimetidine may also be advisable because of the high incidence of gastric ulcers in these patients. REFERENCES I. Hardy JO. Hardy's textbook of surgery. Philadelphia: JB Lippincott, 1983:422. 2. Liggins GC. Initiation of spontaneous labor. Clin Obstet Gynecol 1983;26:47-54. A complete list of references is available on request from Dr. Koerten.
Amniotic fluid ferning in early gestation Max Borten, M.D., and Emanuel A. Friedman, M.D., Sc.D. Boston, Massachusetts Ferning of amniotic fluid was studied in early pregnancy. Arborization was demonstrated during the interval of 6';, to 16 weeks by flame-drying. Air-drying yielded a spectrum of crystallization from none prior to 8 weeks to full ferning by 16 weeks. Technical differences are stressed. (AM J 0asTET GVNECOL 1986;154:628-30.)
Key words: Amniotic fluid, ferning, crystallization, early gestation
Premature rupture of membranes during the second trimester of pregnancy confronts the obstetrician and the patient with a dilemma. The high incidence of fetal pulmonary hypoplasia, deformations from amniotic bands, and maternal infection requires serious in-depth discussion with the patient about whether to interrupt the gestation or risk the consequences of continuing
From the Department of Obstetrics and Gynecology, The Charles A. Dana Biomedical Research Institute, Beth Israel Hospital, Harvard Medical School. Received for publication August 29, 1985; accepted September 3, 1985. Reprint requests: Max Borten, M.D., Department of Obstetrics and Gynecology, Beth Israel Hospital, 330 Brookline Ave., Boston, MA 02215.
628
the pregnancy. Before distressing the patient with such weighty decisions, one has a positive obligation to confirm that the integrity of the amniotic sac has indeed been breached. Following Papanicolaou'S original observation of crystallization (arborization) of dried cervical mucus, this phenomenon was reproduced in most body fluids. Its occurrence is related to the presence of proteins and electrolytes (sodium chloride in particular) in the solution being investigated. Arborization has also been demonstrated in amniotic fluid. It is believed to reflect the relative concentrations of solutes. This characteristic has been used to differentiate amniotic fluid from urine and thus help make a diagnosis of ruptured membranes.'
Amniotic fluid ferning in early gestation
Volume 154 Number 3
629
Fig. 1. Ferning of amniotic fluid at various gestational ages, air-drying (left) versus flame-drying (right): a and b, 6 1/2 weeks; c and d, 12 weeks; e andf, 16 weeks. In air-dried specimens, the degree of arborization increases with gestational age with no ferning seen at 6 1/2 weeks (a), early ferning at 12 weeks (c) and fully developed ferning at 16 weeks (e). All flame-dried specimens of fluid showed unmistakable arborization from 61f2 weeks (b) to 16 weeks (j). (Contrast phase photomicrographs. x 16; except c, x 40.)
Elias et aU reported crystallization of amniotic fluid in 24 of 25 samples obtained at 16 to 18 weeks of gestation. We sought to evaluate if arborization of amniotic fluid is present in gestations of < 16 weeks' duration. For this purpose, we obtained amniotic fluid from 10 patients in early pregnancy. The earliest amniotic fluid sample was obtained from an unruptured tubal gestation of approximately 6V2 weeks' duration (46 days from last normal menstrual period). Specimens from gestations ranging from 8 to 14 weeks in duration were procured by aspiration at the time of pregnancy interruption. Amniotic fluid corresponding to the fifteenth and sixteenth weeks of pregnancy was sampled at the time of genetic amniocentesis. The amniotic fluid specimen was spread over two glass slides. One specimen was smeared in a thin layer and allowed to air-dry before being examined microscopically under medium-power magnification (x 16). The second specimen was dripped onto a slide and flame-dried (but not boiled); it was then evaluated microscopically, as before. Crystallization (ferning) was
seen in all specimens that were rapidly heat-dried (Fig. 1, b, d, and}). When amniotic fluid was allowed to airdry spontaneously, however, the appearance of arborization was inconsistent. There was a gradation seen with advancing gestation. None was seen in the 6V2week fluid (Fig. 1, a). There were a few clusters of ferning first seen at 8 weeks; this was somewhat more fully developed by 12 weeks (Fig. 1, c). Abundant ferning appeared in all second-trimester specimens (Fig. 1, e). Failure of amniotic fluid from early pregnancy to arborize when allowed to air-dry may be misleading. Crystallization of the same specimen when flame-dried suggests that a thicker layer of fluid containing more proteins and electrolytes allows this phenomenon to occur. Air-drying requires a relatively thin layer of amniotic fluid. The absence of ferning may, therefore, result from the smaller absolute amount of solutes present. When thick drops of amniotic fluid were allowed to air-dry (a long, tedious process), comparable ferning did occur.
Borten and Friedman
Ferning of amniotic fluid obtained at 6 1/2 weeks of gestation suggests that crystallization is a property of amniotic fluid throughout pregnancy. Its failure to appear in some specimens may be an artifact relating to the preparation method. It is, therefore, recommended that a drop of the solution to be examined should be flame-dried for evaluation of ferning.
March, 1986 Am J Obstet Gynecol
REFERENCES I. Kovacs D. Crystallization test for the diagnosis of ruptured membranes. AM] OBSTET GYNECOL 1962;83:1257. 2. Elias S, Martin AO, Patel VA, et al. Analysis for amniotic fluid crystallization in second-trimester amniocentesis. AM ] OBSTET GYNECOL 1979;133:401.
The prenatal diagnosis of Robin anomalad Gianluigi PHu, M.D., Roberto Romero, M.D., E. Albert Reece, M.D., Phillipe Jeanty, M.D., and John C. Hobbins, M.D. New Haven, Connecticut The Robin anomalad was diagnosed by the sonographic detection of polyhydramnios and fetal micrognathia in a patient at risk because of a previously affected child. Ultrasound in the second trimester failed to demonstrate any facial anomaly, but mandibular hypoplasia was clearly documented in the third trimester. The antenatal diagnosis allowed immediate neonatal assistance to prevent glossoptosis-induced respiratory failure. (AM J OSSTET GYNECOL 1986;154:630-2.)
Key words: Robin anomalad, prenatal diagnosis, ultrasound, fetus The Robin anomalad is characterized by the association of micrognathia and glossoptosis. It is frequently accompanied by deformity of the palate, which may take the form of a postalveolar cleft or a high, arched palate. I The Robin anomalad is a neonatal emergency because the tongue may obstruct the airways and lead to suffocation. Antenatal recognition would permit the neonatologist to be present in the delivery room and to provide immediate care to the infant. The purpose of this communication is to report the prenatal diagnosis of the Robin anomalad.
Case report The patient, a 31-year-old white woman, gravida 2, para 1, was referred for sonographic examination at 23 weeks of gestation because of a previous child affected with the Robin anomalad. The infant had required respiratory assistance and tube feeding at birth, and operation was performed for correction of a posterior cleft palate. Sonography demonstrated a singleton infant whose biometric parameters were consistent From the Section of Maternal-Fetal Medicine, Department of Obstetrics and Gynecology, Yale University School of Medicine. Received for publication August 7,1985; accepted August 15,1985. Reprint requests: Gianluigi Pilu, M.D., Cattedra Fisiopatologia Prenatale, Istitulo Clinica Oslelrica Ginecologica II, Via Massarenti 13,40138 Bologna, Italy.
630
with assigned gestational age (biparietal diameter, 5.7 cm; femur length, 3.9 cm; humerus, 3.7 cm; outer orbital dimension, 3.7 cm; tibial length, 3.4 cm; ulna, 3.4 cm). A careful examination did not reveal any visceral abnormalities. A midsagittal scan of the fetal face was obtained and is shown in Fig. 1. This scan was interpreted as normal. Coronal scans of the face demonstrated the integrity of the anterior maxilla (Fig. 2). As the accuracy of ultrasound in the detection of the Robin anomalad had not been tested, the patient was told that the sonogram was normal but the sonologist could not exclude the presence of such an anomaly. Another scan was obtained at 35 weeks. At that time polyhydramnios was noted and a midsagittal scan showed striking micrognathia (Fig. 3). The polyhydramnios worsened in the following 3 weeks. The patient went into spontaneous labor and was delivered of a female infant weighing 3600 gm with the typical features of the Robin anomalad, including a postalveolar cleft of the palate (Fig. 4). The infant required intubation in the delivery room; subsequently tube feeding was necessary. The neonatal course was otherwise unremarkable, and the infant will undergo surgical repair of the posterior cleft palate.
Comment Recent advances III ultrasound imaging have permitted examination of the fetal face and the antenatal