The Journal
of P E D I A
Mareh 1979 T R ICS
387
Uterine malformation and fetal deformation Beyond the enhanced miscarriage and prematurit), associated with the structurally abnormal uterus, we have found 14 examples of fetal deformation secondary to uterine ma(]brmation with its consequent uterine constraint. In several instances the constellation of deformations had been misinterpreted as a multiple malformation disorder. One patient died of pulmonary hypoplasia, but all the survivors showed restoration toward normal form postnatally. Recognition of the basic' problem allows/'or surgical reconstruction of the uterus, which is generally accompanied by an improved outlook for subsequent pregnancies.
Marvin E. Miller, M.D., Peter M. Dunn, M.D., a n d D a v i d W . S m i t h , M . D . , * Seattle,
Wash., a n d Bristol, United K i n g d o m
THE STRUCTURALLY ABNORMAL UTERUS, which includes didelphys uterus, bicornuate uterus, septate uterus, and unicornuate uterus, has been appreciated as a cause o f several i m p o r t a n t reproductive p r o b l e m s which can interfere with normal pregnancy or delivery.' It can also be a cause o f congenital deformations, which is the major focus o f this study.
CASE REPORTS The impetus for this study grew out of experience with the following three families. Family 1, One of the authors (D. W. S.) was consulted for genetic counseling because a couple had an infant delivered at 34 weeks' gestation by cesarean section, who died shortly after birth from respiratory insufficiency with what was considered to be an unknown multiple malformation disorder. Birth weight was 1,980 gm and the length was 44.5 cm, both twenty-fifth percentile for age. The major features were severe edema of the limbs with misshapen and asymmetric skull and facies (Fig. 1). Chromoso-
From the Dysmorpholog~ Unit, Department of Pediatrics, and the Center for Inherited Diseases, University of Washington School of Medicine, and Department of Child Health, University of Bristol. Supported by Bureau of Community Health Services, Health Services Administration, Public Health Service, Department of Health, Education and Welfare, Project 913; National Institutes of Health, Grant No. HD 05961; Public Health Service, Grant No. GM 15253; and Gene Center Training Grant (P32-GM-07454-O), National Institutes of Health. *Reprint address: Department of Pediatrics RD-20, University of Washington School of Medicine. Seattle. WA 98195.
0022-3476/79/300387+04500.40/0 9 1979 The C. V. Mosby Co.
mal studies done on the infant and the parents were normal. At the time of the cesarean section, it was noted that the mother had a bicornuate uterus; the infant was in the right horn. The pattern of congenital anomalies as well as the respiratory distress was interpreted by us as being consistent with a deformation process resulting from the marked fetal constraint imposed by the bicornuate uterus. Family 2. A 1.350 gm (25%) white boy was delivered by cesarean section at 32 weeks' gestation to a 31-year-old gravida 2, para 1, abortus 1 mother. The mother had a completely septate uterus and the infant was wedged in the left side of the Uterus. The baby had a misshapen head, deformed ears, a small chest, and marked edema of the left arm (Fig. 2). All of the physical abnormalities resolved and these were attributed by us as being secondary to the constraint imposed on the baby by being confined to one side of the uterus. Family 3. A mother who had 11 prior spontaneous abortions gave birth to an infant at 34 weeks' gestation, following a pregnancy complicated by premature rupture of membranes. Because of a double-footling breech presentation, a cesarean section was done, and the infant was found in the left horn of a bicornuate uterus. The birth weight was 1,080 gm and length 38 cm, both below the third percentile for age. The baby had a tower-shaped skull with brachycephaly and a head circumference of only 26 cm. The palpebral fissures were downslanting and the nose was parrotshaped with a depressed nasal tip. There were mild fifth finger clinodactyly and bilateral simian creases. The infant developed severe respiratory distress immediately after birth, but otherwise did well. Because of the initial craniofacial appearance, the diagnosis of Crouzon syndrome was entertained. By three weeks of age the head was no longer tower-shaped, the sutures were normal and the nose and palpebral fissures had
Vol. 94, No. 3, pp. 387-390
388
Miller et al.
The Journal of Pediatrics March 1979
a normal alignment. Thus, the physical abnormalities of prenatal onset were no longer present and the situation was interpreted as having been a transient constellation of deformations secondary to intrauterine constraint in one horn of a bicornuate uterus. METHODS This initial experience led us to seek further knowledge about the impact of the structurally abnormal uterus on the developing fetus. The obstetric histories of women with structural uterine abnormalities were evaluated at the University Hospital in Seattle. Eight women with uterine malformations were ascertained; they had a total of ten live-born offspring, of whom three had deformations of mild-moderate severity which required no medical intervention. Three additional such patients were referred to the Dysmorphology Unit for evaluation of congenital anomalies in the child. Finally, five examples were contributed by one of the authors (P. M. D.) from his comprehensive deformation study of approximately 4,500 consecutive newborn babies.'-' Including the aforementioned three case reports, and exclusive of the literature, a total of 14 patients were thus ascertained with deformations born of women with a structurally abnormal uterus. Fig. 1. This premature infant (Patient 1) was delivered by cesarean section from a mother with a bicornuate uterus and died shortly after birth from respiratory distress. Note the severe edema of the left extremities and the markedly deformed head.
Fig. 2. This premature infant (Patient 2) was delivered by cesarean section and was noted to be wedged into one side of a completely septate uterus. Note the abnormally shaped head at birth with a prominent and bulging forehead and micrognathia. Restoration to a more normal configuration occurred by 3~/2 months.
RESULTS
AND COMMENTS
It is estimated that 1 to 2% of women have a structural uterine abnormality? although the majority of these cannot be diagnosed on routine pelvic examination and may only be suspected after repeated problems with pregnancy, alerting an astute physician to the possibility of this diagnosis? Table 1 sets forth the relative excess frequency of the observed problems which have been noted in the obstetric histories of women with such a malformed uterus. ~ Deformation, as defined by one of the authors (P. M. D.), is the consequence of extrinsic biomechanical factors, in this instance external uterine constraint. 5 The pliable, growing fetal tissues are molded in response to the aberrant constraint. This type of abnormality in morphogenesis should be distinguished from a malformation, in which there is an intrinsic problem within one or more developing tissues of the fetus. ~ The structurally abnormal uterus can be a potential "home of constraint" for the developing fetus for at least two reasons. (1) The simple geometry of sueh a uterus may provide a significantly smaller intrauterine volume within which the developing fetus can grow. (2) The structurally abnormal uterus is often deficient in uterine musculature, thus making it less capable of expanding to accommodate the fetus. :~
Volume 94 Number 3
The findings in the combined group of 14 infants with deformations born to women with a uterine malformation are shown in Table II. The number of different deformations per patient ranged from one to five, with an average of three. The patterns of deformation observed are similar to those found in other instances of uterine constraint. Almost two-thirds of the patients had unusual molding of the head, which ranged from abnormal overlapping of the sutures to one which initially mimicked the Crouzon syndrome in appearance. Facial deformations occurred in three-quarters of the patients, the most common being mandibular asymmetry. The next most common facial deformation involved the ear, and consisted of either an overfolding of the helix or a flattening of the auricle. Limb deformations were present in 46% of the patients, and included both joint contractures and edema of the limbs. The edema was interpreted as being secondary to proximal obstruction to lymph flow due to compression. Pulmonary hypoplasia was found or implied in two of the 14 infants and was considered the probable cause of death in one of these infants. As emphasized by Thomas and Smith ~ intrauterine compressive forces can limit lung growth and result in pulmonary hypoplasia with secondary respiratory distress at birth. Although most deformations are usually benign and resolve with time, pulmonary hypoplasia is a potentially life-threatening complication. Neuropraxia was found in one infant as a result of facial compression with transient seventh nerve palsy. In two of the 14 cases, there was a history of amniotic fluid leakage, and in each infant a "Potter facies" was noted, as well as other deformations. Undoubtedly the oligohydramnios in these instances added to the severity of constraint. Whether the structurally abnormal uterus predisposed to premature rupture of membranes with chronic amniotic fluid leakage in these two instances is not clear. Another factor which may enhance the impact of uterine constraint and may contribute to the severity of deformations is the high frequency of abnormal fetal positioning in the malformed uterus. At least three previous reports of women with structural uterine abnormalities have made mention of offspring with congenital abnormalities, although the direct deforming role of the uterine constraint has not been emphasized. ~-'~ It is important that these women be recognized, since those who have a poor reproductive history may have a uterine anomaly that is amenable to surgical correction (metroplasty). Metroplasty has been shown to increase the chance of carrying a healthy newborn infant to term without complications in women with certain structural abnormalities of the uterus who have had poor reproductive performances. Buttram et al H
Uterine malformation and fetal deformation
3 89
Table I. Gestational features of the structurally abnormal uterus (1)
No. of times greater than controls
Feature Spontaneous abortion Prematurity Malpresentation Breech Transverse lie Retained placenta Cesarean section Stillbirths Uterine rupture Uterine dysfunction Fetal deformations*
3 4 6 25 15 8 Increased Increased Increased 15
*Present study. Table 11. 14 infants with deformation problems born of women with a malformed uterus
Deformation Skull Face Ear Nose Mandible Pulmonary hypoplasia Neuropraxia Limb Clubfoot Misaligned hips Misaligned knees Edema Dorsiflexion, feet Talipes equinovarus
I
No. 9 10 4 2 5 2 1 7 2 2 1 3 2 1
report a 20% fetal survival rate in pregnancies of women with septate uteri; the survival rate was 88% following metroplasty. Strassman ~reports a similar success rate with metroplasty in women with structurally abnormal uteri, and states that the fecundity of such women approaches normal following metroplasty. It has been estimated that 25% of women with a bicornuate uterus will have a serious reproductive problem and no living children. 1~ Since 1 or 2% of all women have structural uterine abnormalities of some type, these may be an important cause of fetal wastage, poor reproductive performance, complications of pregnancy, and, as this study suggests, deformations in the newborn infant. At least three babies with deformations were noted from a total of ten live-born infants from eight mothers ascertained in this study because of a structural uterine
390
Miller et al.
abnormality. Although this is a small number, the 30% incidence of deformations in this group of infants is significantly higher than the 2% incidence of congenital deformations found in a general population of newborn infants. ~ Thus unusual physical features and problems in infants born to women with a structurally abnormal uterus should be suspected of being deformations rather than malformations. As illustrated by the three infants in the case reports, deformations can be confused with malformation disorders. The recognition of the fundamental nature of the problem is important in providing a prognosis, since the natural history of a deformation problem is usually restoration toward normal form and function once released from the causative constraint, whereas in a malformation the features are often irreversible. Distinction between malformation and deformation is also important in genetic counseling. W h e n the situation is that of a deformation from a structural uterine abnormality, the recurrence risk is that for deformational problems. If indicated, surgical correction of the uterus should be considered to increase the likelihood of having a normal child. The authors are indebted to the following individuals for their help in the contribution of case material: Dr. Peter Bowen of Edmonton University, Edmonton, Canada, for Family 1; Dr. Morton Stenchever of the University of Washington School of Medicine, and Dr. Larry Jung of the University of Utah for Family 2; Dr. Kenneth Lyons Jones of the University of California Medical School for Family 3; Dr. Youn of BronxLebanon Hospital, New York for informing us of another case.
The Journal of Pediatrics March 1979
REFERENCES
1. Zabriskie JR: Pregnancy and the malformed uterus, West .I Obstet Gynecol 70:293, 1962. 2. Dunn PM: The influence of the intrauterine environment in the causation of congenital postural deformities, with special reference to congenital dislocation of the hip (thesis for M~D. degree), University of Cambridge, 1969. 3. Strassman EO: Fertility and unification of double uterus, Fertil Steril 17:165, 1966. 4. Jones WS: Obstetric significance of female genital anomalies, Obstet Gynecol 10:113, 1957. 5. Dunn PM: Congenital postural deformities, Br Med Bull 32:71, 1976. 6. Clarren SK, and Smith DW: Congenital deformities, Pediatr Clin North Am 24:665, 1977. 7. Thomas JT, and Smith DW: Oligohydramnios, cause of the nonrenal features of Potter's syndrome, including pulmonary hypoplasia, J PEDIATR84:811, 1974. 8. Findley P: Pregnancy in uterus didelphys, Am J Obstet Gynecol 12:318, 1926. 9. Baker WS, Roy RL, Bancroft CE, McGaughey H, Dickman FN, and Tucker GW: Congenital anomalies of the uterus associated with pregnancy: An analysis of 118 cases from the literature with a report of 9 additional cases, Am J Obstet Gynecol 66:580, 1953. 10. Wilson DC, and Harris GH: Congenital abnormalities of the uterus and associated malformations, Br J Obstet Gynecol 68:841, 1961. 11. Buttran VC, Zarotti L, Acosta AA, Vanderheyden JS, Besch PK, and Franklin RR: Surgical correction of the septate uterus, Fertil Steril 25:323, 1974. 12. Jones HW, and Jones GES: Double uterus as an etiological factor in repeated abortion: Indications for surgical repair, Am J Obstet Gynecol 65:325, 1953.
of P E D I A
Mareh 1979 T R ICS
387
Uterine malformation and fetal deformation Beyond the enhanced miscarriage and prematurit), associated with the structurally abnormal uterus, we have found 14 examples of fetal deformation secondary to uterine ma(]brmation with its consequent uterine constraint. In several instances the constellation of deformations had been misinterpreted as a multiple malformation disorder. One patient died of pulmonary hypoplasia, but all the survivors showed restoration toward normal form postnatally. Recognition of the basic' problem allows/'or surgical reconstruction of the uterus, which is generally accompanied by an improved outlook for subsequent pregnancies.
Marvin E. Miller, M.D., Peter M. Dunn, M.D., a n d D a v i d W . S m i t h , M . D . , * Seattle,
Wash., a n d Bristol, United K i n g d o m
THE STRUCTURALLY ABNORMAL UTERUS, which includes didelphys uterus, bicornuate uterus, septate uterus, and unicornuate uterus, has been appreciated as a cause o f several i m p o r t a n t reproductive p r o b l e m s which can interfere with normal pregnancy or delivery.' It can also be a cause o f congenital deformations, which is the major focus o f this study.
CASE REPORTS The impetus for this study grew out of experience with the following three families. Family 1, One of the authors (D. W. S.) was consulted for genetic counseling because a couple had an infant delivered at 34 weeks' gestation by cesarean section, who died shortly after birth from respiratory insufficiency with what was considered to be an unknown multiple malformation disorder. Birth weight was 1,980 gm and the length was 44.5 cm, both twenty-fifth percentile for age. The major features were severe edema of the limbs with misshapen and asymmetric skull and facies (Fig. 1). Chromoso-
From the Dysmorpholog~ Unit, Department of Pediatrics, and the Center for Inherited Diseases, University of Washington School of Medicine, and Department of Child Health, University of Bristol. Supported by Bureau of Community Health Services, Health Services Administration, Public Health Service, Department of Health, Education and Welfare, Project 913; National Institutes of Health, Grant No. HD 05961; Public Health Service, Grant No. GM 15253; and Gene Center Training Grant (P32-GM-07454-O), National Institutes of Health. *Reprint address: Department of Pediatrics RD-20, University of Washington School of Medicine. Seattle. WA 98195.
0022-3476/79/300387+04500.40/0 9 1979 The C. V. Mosby Co.
mal studies done on the infant and the parents were normal. At the time of the cesarean section, it was noted that the mother had a bicornuate uterus; the infant was in the right horn. The pattern of congenital anomalies as well as the respiratory distress was interpreted by us as being consistent with a deformation process resulting from the marked fetal constraint imposed by the bicornuate uterus. Family 2. A 1.350 gm (25%) white boy was delivered by cesarean section at 32 weeks' gestation to a 31-year-old gravida 2, para 1, abortus 1 mother. The mother had a completely septate uterus and the infant was wedged in the left side of the Uterus. The baby had a misshapen head, deformed ears, a small chest, and marked edema of the left arm (Fig. 2). All of the physical abnormalities resolved and these were attributed by us as being secondary to the constraint imposed on the baby by being confined to one side of the uterus. Family 3. A mother who had 11 prior spontaneous abortions gave birth to an infant at 34 weeks' gestation, following a pregnancy complicated by premature rupture of membranes. Because of a double-footling breech presentation, a cesarean section was done, and the infant was found in the left horn of a bicornuate uterus. The birth weight was 1,080 gm and length 38 cm, both below the third percentile for age. The baby had a tower-shaped skull with brachycephaly and a head circumference of only 26 cm. The palpebral fissures were downslanting and the nose was parrotshaped with a depressed nasal tip. There were mild fifth finger clinodactyly and bilateral simian creases. The infant developed severe respiratory distress immediately after birth, but otherwise did well. Because of the initial craniofacial appearance, the diagnosis of Crouzon syndrome was entertained. By three weeks of age the head was no longer tower-shaped, the sutures were normal and the nose and palpebral fissures had
Vol. 94, No. 3, pp. 387-390
388
Miller et al.
The Journal of Pediatrics March 1979
a normal alignment. Thus, the physical abnormalities of prenatal onset were no longer present and the situation was interpreted as having been a transient constellation of deformations secondary to intrauterine constraint in one horn of a bicornuate uterus. METHODS This initial experience led us to seek further knowledge about the impact of the structurally abnormal uterus on the developing fetus. The obstetric histories of women with structural uterine abnormalities were evaluated at the University Hospital in Seattle. Eight women with uterine malformations were ascertained; they had a total of ten live-born offspring, of whom three had deformations of mild-moderate severity which required no medical intervention. Three additional such patients were referred to the Dysmorphology Unit for evaluation of congenital anomalies in the child. Finally, five examples were contributed by one of the authors (P. M. D.) from his comprehensive deformation study of approximately 4,500 consecutive newborn babies.'-' Including the aforementioned three case reports, and exclusive of the literature, a total of 14 patients were thus ascertained with deformations born of women with a structurally abnormal uterus. Fig. 1. This premature infant (Patient 1) was delivered by cesarean section from a mother with a bicornuate uterus and died shortly after birth from respiratory distress. Note the severe edema of the left extremities and the markedly deformed head.
Fig. 2. This premature infant (Patient 2) was delivered by cesarean section and was noted to be wedged into one side of a completely septate uterus. Note the abnormally shaped head at birth with a prominent and bulging forehead and micrognathia. Restoration to a more normal configuration occurred by 3~/2 months.
RESULTS
AND COMMENTS
It is estimated that 1 to 2% of women have a structural uterine abnormality? although the majority of these cannot be diagnosed on routine pelvic examination and may only be suspected after repeated problems with pregnancy, alerting an astute physician to the possibility of this diagnosis? Table 1 sets forth the relative excess frequency of the observed problems which have been noted in the obstetric histories of women with such a malformed uterus. ~ Deformation, as defined by one of the authors (P. M. D.), is the consequence of extrinsic biomechanical factors, in this instance external uterine constraint. 5 The pliable, growing fetal tissues are molded in response to the aberrant constraint. This type of abnormality in morphogenesis should be distinguished from a malformation, in which there is an intrinsic problem within one or more developing tissues of the fetus. ~ The structurally abnormal uterus can be a potential "home of constraint" for the developing fetus for at least two reasons. (1) The simple geometry of sueh a uterus may provide a significantly smaller intrauterine volume within which the developing fetus can grow. (2) The structurally abnormal uterus is often deficient in uterine musculature, thus making it less capable of expanding to accommodate the fetus. :~
Volume 94 Number 3
The findings in the combined group of 14 infants with deformations born to women with a uterine malformation are shown in Table II. The number of different deformations per patient ranged from one to five, with an average of three. The patterns of deformation observed are similar to those found in other instances of uterine constraint. Almost two-thirds of the patients had unusual molding of the head, which ranged from abnormal overlapping of the sutures to one which initially mimicked the Crouzon syndrome in appearance. Facial deformations occurred in three-quarters of the patients, the most common being mandibular asymmetry. The next most common facial deformation involved the ear, and consisted of either an overfolding of the helix or a flattening of the auricle. Limb deformations were present in 46% of the patients, and included both joint contractures and edema of the limbs. The edema was interpreted as being secondary to proximal obstruction to lymph flow due to compression. Pulmonary hypoplasia was found or implied in two of the 14 infants and was considered the probable cause of death in one of these infants. As emphasized by Thomas and Smith ~ intrauterine compressive forces can limit lung growth and result in pulmonary hypoplasia with secondary respiratory distress at birth. Although most deformations are usually benign and resolve with time, pulmonary hypoplasia is a potentially life-threatening complication. Neuropraxia was found in one infant as a result of facial compression with transient seventh nerve palsy. In two of the 14 cases, there was a history of amniotic fluid leakage, and in each infant a "Potter facies" was noted, as well as other deformations. Undoubtedly the oligohydramnios in these instances added to the severity of constraint. Whether the structurally abnormal uterus predisposed to premature rupture of membranes with chronic amniotic fluid leakage in these two instances is not clear. Another factor which may enhance the impact of uterine constraint and may contribute to the severity of deformations is the high frequency of abnormal fetal positioning in the malformed uterus. At least three previous reports of women with structural uterine abnormalities have made mention of offspring with congenital abnormalities, although the direct deforming role of the uterine constraint has not been emphasized. ~-'~ It is important that these women be recognized, since those who have a poor reproductive history may have a uterine anomaly that is amenable to surgical correction (metroplasty). Metroplasty has been shown to increase the chance of carrying a healthy newborn infant to term without complications in women with certain structural abnormalities of the uterus who have had poor reproductive performances. Buttram et al H
Uterine malformation and fetal deformation
3 89
Table I. Gestational features of the structurally abnormal uterus (1)
No. of times greater than controls
Feature Spontaneous abortion Prematurity Malpresentation Breech Transverse lie Retained placenta Cesarean section Stillbirths Uterine rupture Uterine dysfunction Fetal deformations*
3 4 6 25 15 8 Increased Increased Increased 15
*Present study. Table 11. 14 infants with deformation problems born of women with a malformed uterus
Deformation Skull Face Ear Nose Mandible Pulmonary hypoplasia Neuropraxia Limb Clubfoot Misaligned hips Misaligned knees Edema Dorsiflexion, feet Talipes equinovarus
I
No. 9 10 4 2 5 2 1 7 2 2 1 3 2 1
report a 20% fetal survival rate in pregnancies of women with septate uteri; the survival rate was 88% following metroplasty. Strassman ~reports a similar success rate with metroplasty in women with structurally abnormal uteri, and states that the fecundity of such women approaches normal following metroplasty. It has been estimated that 25% of women with a bicornuate uterus will have a serious reproductive problem and no living children. 1~ Since 1 or 2% of all women have structural uterine abnormalities of some type, these may be an important cause of fetal wastage, poor reproductive performance, complications of pregnancy, and, as this study suggests, deformations in the newborn infant. At least three babies with deformations were noted from a total of ten live-born infants from eight mothers ascertained in this study because of a structural uterine
390
Miller et al.
abnormality. Although this is a small number, the 30% incidence of deformations in this group of infants is significantly higher than the 2% incidence of congenital deformations found in a general population of newborn infants. ~ Thus unusual physical features and problems in infants born to women with a structurally abnormal uterus should be suspected of being deformations rather than malformations. As illustrated by the three infants in the case reports, deformations can be confused with malformation disorders. The recognition of the fundamental nature of the problem is important in providing a prognosis, since the natural history of a deformation problem is usually restoration toward normal form and function once released from the causative constraint, whereas in a malformation the features are often irreversible. Distinction between malformation and deformation is also important in genetic counseling. W h e n the situation is that of a deformation from a structural uterine abnormality, the recurrence risk is that for deformational problems. If indicated, surgical correction of the uterus should be considered to increase the likelihood of having a normal child. The authors are indebted to the following individuals for their help in the contribution of case material: Dr. Peter Bowen of Edmonton University, Edmonton, Canada, for Family 1; Dr. Morton Stenchever of the University of Washington School of Medicine, and Dr. Larry Jung of the University of Utah for Family 2; Dr. Kenneth Lyons Jones of the University of California Medical School for Family 3; Dr. Youn of BronxLebanon Hospital, New York for informing us of another case.
The Journal of Pediatrics March 1979
REFERENCES
1. Zabriskie JR: Pregnancy and the malformed uterus, West .I Obstet Gynecol 70:293, 1962. 2. Dunn PM: The influence of the intrauterine environment in the causation of congenital postural deformities, with special reference to congenital dislocation of the hip (thesis for M~D. degree), University of Cambridge, 1969. 3. Strassman EO: Fertility and unification of double uterus, Fertil Steril 17:165, 1966. 4. Jones WS: Obstetric significance of female genital anomalies, Obstet Gynecol 10:113, 1957. 5. Dunn PM: Congenital postural deformities, Br Med Bull 32:71, 1976. 6. Clarren SK, and Smith DW: Congenital deformities, Pediatr Clin North Am 24:665, 1977. 7. Thomas JT, and Smith DW: Oligohydramnios, cause of the nonrenal features of Potter's syndrome, including pulmonary hypoplasia, J PEDIATR84:811, 1974. 8. Findley P: Pregnancy in uterus didelphys, Am J Obstet Gynecol 12:318, 1926. 9. Baker WS, Roy RL, Bancroft CE, McGaughey H, Dickman FN, and Tucker GW: Congenital anomalies of the uterus associated with pregnancy: An analysis of 118 cases from the literature with a report of 9 additional cases, Am J Obstet Gynecol 66:580, 1953. 10. Wilson DC, and Harris GH: Congenital abnormalities of the uterus and associated malformations, Br J Obstet Gynecol 68:841, 1961. 11. Buttran VC, Zarotti L, Acosta AA, Vanderheyden JS, Besch PK, and Franklin RR: Surgical correction of the septate uterus, Fertil Steril 25:323, 1974. 12. Jones HW, and Jones GES: Double uterus as an etiological factor in repeated abortion: Indications for surgical repair, Am J Obstet Gynecol 65:325, 1953.
