Acta Obstet Gynecol Scand 5 4 497-498, 1975
PREVENTION OF PREMATURE DELIVERY IN A UNICORNUATE UTERUS BY CERVICAL CERCLAGE G. Wajntraub, A. Milwidsky and D. Weiss From the Department of Obstetrics and Gynaecology (Head: Zvi Palti, M.D.) General Jewish Hospital “Shaare ZedeK’, Jerusalem, Israel
Absrracr. A 26-year-old woman had two premature deliveries when 6 months pregnant. Neither produced a living child. A hysterosalpingogram was done, confirming a diagnosis of unicornuate uterus. When the patient became pregnant again, a third and fourth time, cervical cerclage was done in both cases. Pregnancies were terminated by cesarean section because of breech presentation and malformation of the uterus and in each case a healthy living child was delivered. In our view, in order to prevent premature deliveries, when a diagnosis of uterus malformations has been confirmed, a cerclage should be performed.
Due to the widespread use of the hysterosalpingography and laparoscopy, congenital anomalies of the genital tract can be discovered with greater frequency than before (9). The true uterus unicornis or hemiuterus is a comparatively rare condition. An incidence of only 0.3 percent was found by Baker ( I ) in the cases that he reported. Uterine abnormalities with the exception of infantile and hypoplastic types have an incidence of 1 :420 in a series of 13000 cases analysed by Gemmel (3). Munro-Kerr (6) stated that pregnancy is rare in the unicornuate uterus. An admirable classification of developmental anomalies of the uterus has been given by Hunter (4) and our case appears to fall in the category of “uterus unicornis” “unicorpus” and “unicollis”. Demarez (2) and Schattenberg (8) reported 32 cases upto 1940 of true unicornuate uterus. Since that time, to our knowledge, only 18 further cases have been reported in the literature. CASEREPORT P. G. (ref. no. 216.211) a 26-year-old married woman, attended our Outpatient Clinic having a history of two previous mid-trimester abortions, when 6-months preg-
nant. As a result she had no living children. A hysterosalpingogram was made and this revealed a long, narrow uterus, with only one Fallopian tube, on the right side, which we diagnosed as a hemiuterus. An intravenous pyelography was later made and showed normal kidneys. Since we know that this kind of malformation can cause premature deliveries, we advised the patient that when she next became pregnant we hoped to be able to help her by doing a cervical cerclage at the beginning of the 4th month. Shortly after the patient became pregnant and the cerclage was performed. In the 9th month, the fetus was lying as a breech presentation and therefore it was decided to deliver her by cesarean section. The section was performed in another hospital since our patient, while visiting her mother there, appeared to be in labour. The patient was delivered of a healthy female child, the first living after 3 pregnancies. After two years she conceived again and at the end of the 4th month we again did a cervical cerclage. The antenatal period was uneventful and the pregnancy grew normally, till the 38th week. At that time she was admitted to our department with a view to repeat elective cesarean section, since there was again a breech presentation. The examinations revealed a well-healed midline subumbilical surgical scar. By palpation we found the uterus to be the size of a 38-week pregnancy with a breech presentation. The fetal heart-rate was normal. Vaginal examination showed that the cervix was high, deviated to the right and softened. The internal 0s did not admit the tip of a finger. X-ray confirmed the breech presentation and showed that the lower femoral epiphyseal ossification centre has appeared, which verified that the fetus was mature. Consequently we considered this the right time to perform a lower segment cesarean section after prior removal of the cervical cerclage. The uterus appeared twisted to the right. The operation was completed and a living female infant was delivered which weighed 2 850 g. The placenta was removed but one cotyledon was adherent high on the anterior wall so we did a curettage with a macrocurette. Further examination of the abdomen after closure of the uterus showed no trace of the left uterine vessels. The uterus appeared to be unicornuate, with one normal tube, and broad, round and ovarian ligaments on the right side only. At the left inquiAGIO Obsrer Gynecol Sccind 54 (1975)
498
Prevention of premature delivery
Fig. 1 . Hysterosalpingogram showing a unicornuate uterus with a single patent right tube.
nal ring we found the second part of a small fibrous uterus, 4 cm long, and at its left corner there was a long but very narrow left ovary, 5 . 0 ~ 0 . 3cm in length, with a short tube and a welldeveloped fimbrial end and round ligament. There was no connection to the right uterus. No abnormalities of any other abdominal organ were found. Post-operative progress was good. The infant showed no congenital abnormalities.
COMMENT Only 12 cases are mentioned in the literature we were able to study, in which there was a total absence of one ovary, Fallopian tube and broad ligament. It is remarkable that in 3 of these cases this occurred on the left side and in 9 cases on the right side. I n I 1 other cases the second ovary was ectopic and located above the pelvic brim. The ovary in general was usually longer and narrower on the abnormal side. When the Fallopian tube was present, it was almost always a short portion of the fimbriated end. The round ligament usually existed as a small and poorly developed structure attached to the cervical portion of the uterus. The broad ligament was also absent on the defective side although occasionally a small part of it could be observed. The ovarian ligament was found to extend down toward or into the internal inguinal ring.
When the ovarian vessels were traced, they were found to arise from the aorta, or to enter the vena cava. In all cases reported, external genitalia were normal. Menses were either normal or irregular and painful. Philipp (7) also commented that the right side is frequently more developed than the left, as in our case. From the X-ray, a so-called “hemiuterus” can also be seen, whereas in the case of a uterus bicornis or duplex, where the rudimentary horn does not fill up, it cannot be seen. The explanation for the higher percentage of breech presentation is that the spindle shape of the uterus unicornis appears to favour this position, as in both pregnancies we describe ( 5 , 10). The new case we are here presenting is of special interest due to the fact that two living children were delivered by cesarean section after two premature deliveries without a living child, before the cervical cerclage was performed.
REFERENCES I . Baker, W. S., Jr, Roy, R. L., Bancroft. C. E., McGaughey, H., Dickman, P. N. & Tucker, C. W.: Am J Obstet Gynecol66: 580, 1940. 2. Demarez, R., Herbert, J. & Ziskind, Y.: Ann Anat Pathol (Paris) 15: 1054, 1938. 3. Gemmel, 4. A.: British Eiiqclop. of Medical Practice, vol. 12. Butterworth, London, 1939. 4. Hunter, W.: J Obstet GynecolBr Emp57:721, 1950. 5 . Kayser, A.: cited by H. Finkbeiner: Das Rontgenbild des Entwicklungsgestorten Uterus. G . Thieme, Leipzig, 1951. 6. Munro-Kerr, J. & Moir, J. C.: Operative Obstetrics. Ballitre, Tindall and Cox, London, 1956. 7. Philipp, F.: cited by H. Finkbeiner: Das Rontgenbild des Entwicklungsgestorten Uterus. G . Thieme, Leipzig, 1951. 8. Schattenberg, H. J. & Ziskind, Y.: Amer J Obstet Gynecol40: 298, 1940. 9. Titus, P.: Management of Obstetric Difficulties. Edition IV. C. V. Mosby Company, St. Louis, Missouri, 1950. 10. Wajntraub, G.: Bull SOC Roy Belg Gynec Obstet 36: 125, 1966.
Submitted for publication May 8 , 1974
G. Wajntraub General Jewish Hospital “Shaare Zedek” Jerusalem Israel
PREVENTION OF PREMATURE DELIVERY IN A UNICORNUATE UTERUS BY CERVICAL CERCLAGE G. Wajntraub, A. Milwidsky and D. Weiss From the Department of Obstetrics and Gynaecology (Head: Zvi Palti, M.D.) General Jewish Hospital “Shaare ZedeK’, Jerusalem, Israel
Absrracr. A 26-year-old woman had two premature deliveries when 6 months pregnant. Neither produced a living child. A hysterosalpingogram was done, confirming a diagnosis of unicornuate uterus. When the patient became pregnant again, a third and fourth time, cervical cerclage was done in both cases. Pregnancies were terminated by cesarean section because of breech presentation and malformation of the uterus and in each case a healthy living child was delivered. In our view, in order to prevent premature deliveries, when a diagnosis of uterus malformations has been confirmed, a cerclage should be performed.
Due to the widespread use of the hysterosalpingography and laparoscopy, congenital anomalies of the genital tract can be discovered with greater frequency than before (9). The true uterus unicornis or hemiuterus is a comparatively rare condition. An incidence of only 0.3 percent was found by Baker ( I ) in the cases that he reported. Uterine abnormalities with the exception of infantile and hypoplastic types have an incidence of 1 :420 in a series of 13000 cases analysed by Gemmel (3). Munro-Kerr (6) stated that pregnancy is rare in the unicornuate uterus. An admirable classification of developmental anomalies of the uterus has been given by Hunter (4) and our case appears to fall in the category of “uterus unicornis” “unicorpus” and “unicollis”. Demarez (2) and Schattenberg (8) reported 32 cases upto 1940 of true unicornuate uterus. Since that time, to our knowledge, only 18 further cases have been reported in the literature. CASEREPORT P. G. (ref. no. 216.211) a 26-year-old married woman, attended our Outpatient Clinic having a history of two previous mid-trimester abortions, when 6-months preg-
nant. As a result she had no living children. A hysterosalpingogram was made and this revealed a long, narrow uterus, with only one Fallopian tube, on the right side, which we diagnosed as a hemiuterus. An intravenous pyelography was later made and showed normal kidneys. Since we know that this kind of malformation can cause premature deliveries, we advised the patient that when she next became pregnant we hoped to be able to help her by doing a cervical cerclage at the beginning of the 4th month. Shortly after the patient became pregnant and the cerclage was performed. In the 9th month, the fetus was lying as a breech presentation and therefore it was decided to deliver her by cesarean section. The section was performed in another hospital since our patient, while visiting her mother there, appeared to be in labour. The patient was delivered of a healthy female child, the first living after 3 pregnancies. After two years she conceived again and at the end of the 4th month we again did a cervical cerclage. The antenatal period was uneventful and the pregnancy grew normally, till the 38th week. At that time she was admitted to our department with a view to repeat elective cesarean section, since there was again a breech presentation. The examinations revealed a well-healed midline subumbilical surgical scar. By palpation we found the uterus to be the size of a 38-week pregnancy with a breech presentation. The fetal heart-rate was normal. Vaginal examination showed that the cervix was high, deviated to the right and softened. The internal 0s did not admit the tip of a finger. X-ray confirmed the breech presentation and showed that the lower femoral epiphyseal ossification centre has appeared, which verified that the fetus was mature. Consequently we considered this the right time to perform a lower segment cesarean section after prior removal of the cervical cerclage. The uterus appeared twisted to the right. The operation was completed and a living female infant was delivered which weighed 2 850 g. The placenta was removed but one cotyledon was adherent high on the anterior wall so we did a curettage with a macrocurette. Further examination of the abdomen after closure of the uterus showed no trace of the left uterine vessels. The uterus appeared to be unicornuate, with one normal tube, and broad, round and ovarian ligaments on the right side only. At the left inquiAGIO Obsrer Gynecol Sccind 54 (1975)
498
Prevention of premature delivery
Fig. 1 . Hysterosalpingogram showing a unicornuate uterus with a single patent right tube.
nal ring we found the second part of a small fibrous uterus, 4 cm long, and at its left corner there was a long but very narrow left ovary, 5 . 0 ~ 0 . 3cm in length, with a short tube and a welldeveloped fimbrial end and round ligament. There was no connection to the right uterus. No abnormalities of any other abdominal organ were found. Post-operative progress was good. The infant showed no congenital abnormalities.
COMMENT Only 12 cases are mentioned in the literature we were able to study, in which there was a total absence of one ovary, Fallopian tube and broad ligament. It is remarkable that in 3 of these cases this occurred on the left side and in 9 cases on the right side. I n I 1 other cases the second ovary was ectopic and located above the pelvic brim. The ovary in general was usually longer and narrower on the abnormal side. When the Fallopian tube was present, it was almost always a short portion of the fimbriated end. The round ligament usually existed as a small and poorly developed structure attached to the cervical portion of the uterus. The broad ligament was also absent on the defective side although occasionally a small part of it could be observed. The ovarian ligament was found to extend down toward or into the internal inguinal ring.
When the ovarian vessels were traced, they were found to arise from the aorta, or to enter the vena cava. In all cases reported, external genitalia were normal. Menses were either normal or irregular and painful. Philipp (7) also commented that the right side is frequently more developed than the left, as in our case. From the X-ray, a so-called “hemiuterus” can also be seen, whereas in the case of a uterus bicornis or duplex, where the rudimentary horn does not fill up, it cannot be seen. The explanation for the higher percentage of breech presentation is that the spindle shape of the uterus unicornis appears to favour this position, as in both pregnancies we describe ( 5 , 10). The new case we are here presenting is of special interest due to the fact that two living children were delivered by cesarean section after two premature deliveries without a living child, before the cervical cerclage was performed.
REFERENCES I . Baker, W. S., Jr, Roy, R. L., Bancroft. C. E., McGaughey, H., Dickman, P. N. & Tucker, C. W.: Am J Obstet Gynecol66: 580, 1940. 2. Demarez, R., Herbert, J. & Ziskind, Y.: Ann Anat Pathol (Paris) 15: 1054, 1938. 3. Gemmel, 4. A.: British Eiiqclop. of Medical Practice, vol. 12. Butterworth, London, 1939. 4. Hunter, W.: J Obstet GynecolBr Emp57:721, 1950. 5 . Kayser, A.: cited by H. Finkbeiner: Das Rontgenbild des Entwicklungsgestorten Uterus. G . Thieme, Leipzig, 1951. 6. Munro-Kerr, J. & Moir, J. C.: Operative Obstetrics. Ballitre, Tindall and Cox, London, 1956. 7. Philipp, F.: cited by H. Finkbeiner: Das Rontgenbild des Entwicklungsgestorten Uterus. G . Thieme, Leipzig, 1951. 8. Schattenberg, H. J. & Ziskind, Y.: Amer J Obstet Gynecol40: 298, 1940. 9. Titus, P.: Management of Obstetric Difficulties. Edition IV. C. V. Mosby Company, St. Louis, Missouri, 1950. 10. Wajntraub, G.: Bull SOC Roy Belg Gynec Obstet 36: 125, 1966.
Submitted for publication May 8 , 1974
G. Wajntraub General Jewish Hospital “Shaare Zedek” Jerusalem Israel
