European Journal of Obstetrics & Gynecology and Reproductice Biology, 47 (1992) 135 139

135

0 1992 Elsevier Science Publishers B.V. All rights reserved 002%2243/92/$05.00 EUROBS 01428

The unicornuate

uterus: clinical implications

Peter F.J. Donderwinkel a, Joep P.J. Dijrr a and Wim N.P. Willemsen b a Department of Obstetrics and Gynecology, Westeinde Hospital, The Hague, The Netherlands and ’ Department of Obstetrics and Gynecology, Sint Radboud Unicersity Hospital, Nijmegen, The Netherlands

Accepted for publication 19 August 1992

Summary

The unicornuate uterus is associated with a poor reproductive outcome and many gynecological problems. We collected data from 45 women with a unicornuate uterus. We found a high abortion rate of 22% in the first trimester and 16% in the second trimester. Premature labor occurred in 18%. The prevalence of infertility and endometriosis in women with a unicornuate uterus was comparable to women without the anomaly. Unicornuate uterus; Obstetric outcome; Infertility; Endometriosis

Introduction The unicornuate uterus is a rare uterine anomaly resulting from failure of development of one Miillerian duct. The prevalence of congenital uterine malformations has been estimated at l-10% [l-3]. The unicornuate uterus is found in a small percentage of the uterine malformations, varying between 2.5 and 13.2% [4-61. The unicornuate uterus is associated with a poor reproductive outcome 171. A high abortion rate and low fetal survival rate have been reported [8,9]. Gynecological problems related to the hemiuterus are primary infertility, endometriosis and urinary tract anomalies. Since unicornuate uterus is a rare anomaly and

Correspondence to: Joep P.J. DGrr, M.D., Department of Obstetrics and Gynecology, Westeinde Hospital, P.O. Box 432, 2501 CK, The Hague, The Netherlands.

no simple screening technique exists a prospective study is hard to perform. Therefore the current knowledge depends on small retrospective studies or case reports. The best way to study the subject is to collect many data by retrospective file research of a large series [lo]. In this article we present a large series of 45 women with unicornuate uterus and try to clarify ‘the obstetric and gynecologic implications. Patients and Methods

Between 1981 and 1989, 4.5 patients with a unicomuate uterus were examined. In 35 cases the uterine anomaly was demonstrated by both hysterosalpingography (HSG) and laparoscopy, in 4 cases by HSG only. The remaining 6 patients were diagnosed by laparoscopy or by laparotomy. In 32 out of 45 patients with unicornuate uterus the primary complaint was infertility. Two other patients were presented for reversal of tubal ligation. Twice second-trimester abortion and once

136

premature delivery were the reason for examination of the uterus. Three of the 45 women visited the gynecologist for dysmenorrhoea. In the remaining 5 cases a unicornuate uterus was detected accidentally during laparoscopy or laparotomy. Infertility work-up included cycle analysis with BBT chart, mid-luteal serum progesterone estimations, and serum gonadotropin and steroid measurements if necessary, semen analysis, postcoital testing and HSG and/or laparoscopy with dye for tubal patency. Data were collected by retrospective file research. The mean age was 32.7 years, ranging from 18 to 39 years. The patients were categorized according to Buttram’s and Gibbon’s classification [ll]. Thirty-three patients had a unicornuate uterus of which 10 without a rudimentary horn (class II and IIb). In 2 cases these data were not available. In 34 patients the urinary tract was evaluated by intravenous pyelogram, and in 1 case by ultrasound examination. Results Obstetric outcome Twenty-three out of 45 patients had had a total of 47 pregnancies. Two pregnancies were terminated and excluded from the study. The remaining 45 pregnancies included ten first-trimester abortions, seven second-trimester abortions, eight premature deliveries and 20 term deliveries. All premature- and term-born infants survived. The second-trimester abortions could be attributed to cervical incompetence in one case and fetal death in two cases. In one patient with a twin pregnancy labor started at 23 weeks. The other three cases remained unexplained. A caesarean section was performed in 7 patients. In 9 pregnancies the fetus presented as breech (Table I).

TABLE

I

Pregnancy

outcome

in patients

(n = 45) with a unicornuate

uterus

45

Patients Conceived

23

Pregnancies

47

Legal abortions

51%

2

a

10

22% h

Second trim. abortion

7

16% h

Premature

8

First trimester

abortion

labour

Caesarean

44% h

8

section

Breech presentation

9

Cerclage

5 28

Live born babies a Excluded

18%b

20

Term delivery

from the study.

h Percentages

calculated

from 45 pregnancies.

rhoea later developed primary infertility. So, a total of 35 of the 45 patients (78%) was troubled by infertility. This work-up revealed one or more well-known causes in 30 patients, e.g., anovulation (6), tubal pathology (3), cervical hostility (I), suboptimal sperm quality (81, combined male and female factors (7) and endometriosis (5). Primary infertility remained unexplained in 2 women. One of them conceived spontaneously. No data on the infertility work-up could be traced in three patients. All secondary infertile and 11 primary infertile patients conceived. Eighteen primary infertile patients did not conceive. The most common causes of infertility amongst them were anovulation (4) and male subfertility (4). Endometriosis was found in 8 of the 45 patients during laparoscopy. Two of them had consulted their gynecologist for dysmenorrhoea.

TABLE Renal

II malformations

found in 35 examinations

tract in patients with a unicornuate

Gynecologic data In 32 of the 45 patients infertility was the main reason for evaluation of the uterus by hysterosalpingography and laparoscopy with dye test. Three patients initially presented for dysmenor-

%

n

Renal agenesis

uterus 5

Ectopic kidney

4

Hypoplastic

2

Total

pyelum

11

of the urinary

137

Shirodkar cerclage was performed. In two other patients a prophylactic cervical cerclage was performed. Wajntraub et al. [13] reported the beneficial effect of this prophylactic cervical cerclage for prevention of premature delivery in patients with unicornuate uterus. Craig [14] reported an increased incidence of cervical incompetence in patients with uterine malformations. Data on prophylactic cerclage in patients with unicornuate uteri are not available. Current literature [151 suggests that previous second trimester abortions or preterm deliveries constitute a basis for cervical cerclage. Patients with multiple pregnancies, a history of cervical surgery or other indications such as hemiuterus cannot expect any benefit from cerclage. The diminished muscle mass of the unicornuate uterus is considered to play an important part in second-trimester abortion and premature delivery [16,17]. Unfortunately, most studies showed incidences of abortion without specifying the trimester of abortion. Therefore our findings cannot be compared with data found in the literature. Precise data of large series are necessary to draw conclusions on the mechanism and therapy of secondtrimester abortion in patients with unicornuate uterus. As Table III indicates, the incidence of premature delivery and breech presentation is

Eleven of the 35 urinary tract examinations (31%) revealed abnormalities. This included 5 renal agenesis, 4 ectopic kidneys, and 2 hypoplasia of the renal pelvis (Table II). Discussion

Since unicornuate uterus is a rare anomaly and since no simple screening technique exists, prospective research cannot be done. Therefore the current knowledge depends on small retrospective studies or case reports. Obstetrics There are three etiological theories which explain fetal loss in patients with unicornuate uterus: an abnormal uterine blood flow, cervical incompetence and diminished muscle mass of the hemiuterus. The high incidence of first-trimester abortions may be due to a disturbance in the uterine blood flow caused by an absent or abnormal uterine or ovarian artery. This could also explain intrauterine growth retardation or stillbirth related to hemiuterus [9,11,121. In one patient the two consecutive second trimester abortions could be attributed to cervical incompetence. In two following pregnancies this patient delivered at 34 and 37 weeks after a

TABLE III Pregnancy outcome of unicornuate uterus compared with literature

Patients Conceived Pregnancy Horn pregnancy Abortion 1st + 2nd trim. Abortion 2nd trim. Premature labor (pert) Term deliveries (pert) Breech presentations

Buttram 1979

Andrews 1982

Heinonen 1982

Buttram 1983 a

Heinonen 1983 b

Heinonen 1983 ’

Fedele 1987

Present study 1992

19 9 14

5 5 14

13 10 1.5 _

31 ? 60 _ 29

20 1.5 35

19 14 26

19 13 29 1 17

?

?

10 (17%) 21(35%) ?

5 (14%) 12 (34%) 5

45 23 47 _ 17 c 7 8 (18%:) 29 (44%) 9

12

3 -

2 (14%) ?

8

? 2 (14%) 4 (29%) 1

7

? 3 (20%) 5 (33%) 4

8

8

? 5 (29%) 3 (12%) 5

2 3 (10%) 8 (28%) 6

a Buttram presented in his report a literature study not his own material. b The data of Heinonen included one woman with unicornuate uterus who had had nine term pregnancies with spontaneous deliveries. The figures in the sixth column are recalculated with exclusion of this patient. ’ This number does not include two legal abortions.

138 TABLE IV Pregnancy outcome of unicornuate uterus compared with the Dutch National Obstetrics Registration (LVR) 1988

Second trimester Premature delivery Term delivery Postmature delivery Unknown

LVR (n = 60942)

Unicornuate a (n = 35)

1.5% 11.5% 80.2% 6.3% 0.5%

20.5% 20.5% 59%

a The first trimester abortions are excluded from this comparison, because the LVR registration starts from 16 weeks amenorrhoea.

comparable with data in the literature. Six times a caesarean delivery was performed due to premature labor with breech presentation. The Dutch National Obstetrics Registration (LVR) collects data from 90% of all hospital deliveries occurring in The Netherlands. Registration starts at 16 weeks. In 1988, 60,942 deliveries were registered. Data are available revealing the incidence of second-trimester abortion, premature delivery, breech presentation and caesarean section in a large population. Although our study represents only a group of patients with unicornuate uteri who needed medical care for gynecological or obstetrical problems, comparison of our data with data from the LVR gives the impression that the obstetric outcome of the unicornuate uterus is poor (Table IV). As yet the correct obstetric care has to be defined. Gynecology An infertility work-up includes a hysterosalpin-

gography and laparoscopy and therefore reveals a uterine anomaly if present. This suggests that uterine abnormalities can cause infertility. Heinonen et al. [18] stated that uterine anomalies are rarely associated with primary infertility. In our 35 patients with infertility problems, we found no other additional causes in only two patients. Our results therefore confirm the view that non-uterine factors play a larger part in infertility than do gross uterine anomalies. A high incidence of endometriosis is associated with the unicornuate uterus 119,201. Many

etiologic theories have been proposed. Retrograde menstruation [21] and metaplastic conversion of omnipotential mesothelium to functional endometrium [22] have been hypothesized to cause endometriosis. An increase in retrograde menstruation can be expected in uterine anomalies; especially, the unicornuate uterus with noncommunicating cavitary horn [21]. The true prevalence of endometrioses in the general population is unknown. Many women with endometriosis are asymptomatic and therefore unaware of its presence. The prevalence of endometriosis as diagnosed by laparoscopy varies depending on the indication for the laparoscopy. The prevalence was 10-B% in patients undergoing tubal sterilization if special attention was paid to diagnose endometriosis. In patients with chronic pelvic pain it was found in 8.5-47% and in women requesting reversal of sterilization in 18% [23]. In our study 8 out of 45 (18%) patients had endometriosis discovered during laparoscopy or laparotomy. Only two women with endometriosis complained of dysmenorrhoea. It is often very difficult to assess whether there is a communication between the endometrial cavity and the rudimentary horn. Ultrasound may be helpful in the future, but its contribution to evaluation of uterine anomalies is still to be defined [24]. Recently magnetic resonance imaging appeared to be a good but expensive technique for investigation of the unicornuate uterus and the rudimentary horn [2_5].For these reasons a division in cavitary or non-cavitary horns could not be made in the present study. Urinary tract anomalies are frequently associated ‘with uterine malformations [6]. In 35 out of 45 patients (78%) intravenous urograms were performed. Eleven (31%) showed abnormalities. Heinonen et al. 1161found a higher incidence of uropoietical anomalies. The most frequently found abnormality is an absent or ectopic kidney contralateral to the side of the uterine horn. References 1 Fenton AN, Singh BP, Pregnancy associated with congenital abnormalities of female reproductive tract. Am J Obstet Gynecol 1952;63:744-755.

139 2 Cooper JM, Jouch RM, Rigberg HS. The incidence of intrauterine abnormalities found at hysteroscopy in patients undergoing elective hysteroscopic sterilization. J Reprod Med 1983;28:659-661. 3 Taylor PJ, Leader A, George RE. Combined laparoscopy and hysteroscopy in the investigation of infertility. In: Siegler AM, Lindemann JJ, eds. Hysteroscopy: Principles and Practice. Philadelphia: J.B. Lippincott, 1984;207-210. 4 Green LK, Harris RE. Uterine anomalies: frequency of diagnosis and associated obstetric complications. Obstet Gynecol 1976;47:427-429. 5 Ashton D, Amin HK, Richart RM, Neuwirt RS. The incidence of asymptomatic uterine anomalies in women undergoing transcervical tubal sterilization. Obstet Gynecol 1988;72:28-30. 6 Buttram VC, Gibbons WE. Miillerian anomalies: a proposed classification. Fertil Steril 1979;32:40-46. 7 Jones WS. Obstetric significance of female genital anomalies. Obstet Gynecol 1957;10:113-127. 8 Heinonen PK, Saarikoski S, Pystynen PP. Reproductive performance of women with uterine anomalies. Acta Obstet Gynecol Stand 1982;61:157-162. 9 Fedele L, Zamberletti D, Vercellini P, Dorta M, Candiani GV. Reproductive performance of women with unicornuate uterus. Fertil Steril 1987;47:416-419. 10 Editorial Comment to: Fedele L. Zamberletti D. Vercellini P. Dorta M. Candiani GB. Reproductive performance of women with unicornuate uterus. Fertil Steril 1987;47:416419; in: Obstet Gynecol Survey 1987;42:710-711. 11 Buttram VC. Miillerian anomalies and their management. Fertil Steril 1983;40:159-163. 12 Andrews MC, Jones HW. Impaired reproductive performance of the unicornuate uterus: Intrauterine growth retardation, infertility, and recurrent abortion in five cases. Am J Obstet Gynecol 1982;144:173-176. 13 Wajntraub G, Milwidsky A, Weiss D. Prevention of premature delivery in a unicornuate uterus by cervical cerclage. Acta Obstet Gynecol Stand 1975;54:497-498.

14 Craig CJT. Congenital abnormalities of the uterus and foetal wastage. S Afr Med J 1973;47:2000-2005. 15 Grant A. Cervical cerclage to prolong pregnancy. In: Chalmers I, Enkin M, Keirse MJNC, eds. Effective care in pregnancy and childbirth, vol. 1, part 5, chapter 40. Oxford University Press 1989; 633-646. 16 Heinonen PK. Clinical implications of the unicornuate uterus with rudimentary horn. Int J Gynaecol Obstet 1983;21:145-150. 17 Semmens JP. Congenital defects of the reproductive tract: clinical implications. Contemp Obstet Gynecol 1975;5:95102. I8 Heinonen PK, Pystynen PP. Primary infertility and uterine anomalies. Fertil Steril 1983;40:311-316. 19 Semmens JP. Congenital anomalies of the female genital tract. Functional classification based on review of 56 personal cases and 500 reported cases. Obstet Gynecol 1962;19:328-350. 20 Rolen AC, Choquette AJ, Semmens JP. Rudimentary uterine horn: Obstetric and gynecologic implications. Obstet Gynecol 1966;27:806-813. 21 Olive DL, Henderson DY. Endometriosis and Mullerian anomalies. Obstet Gynecol 1987;69:412-415. 22 Sanfilippo JS, Wakim NG, Schikler KN, Yussman MA. Endometriosis in association with uterine anomaly. Am J Obstet Gynecol 1986;154:39-43. 23 Dunselman GAJ. Endometriosis: clinical and experimental aspects. Thesis, State University of Limburg, Maastricht, The Netherlands, 1988. 24 Fedele L, Dorta M, Vercellini P, Brioschi D, Candiani GB. Ultrasound in the diagnosis of subclasses of unicomuate uterus. Obstet Gynecol 1988;71:274-277. 25 Carrington BM, Hricak H, Nuruddin RN, Secaf ES, Laros RK, Hill EC. Miillerian duct anomalies: MR imaging evaluation. Radiology 1990;176:715-720.

The unicornuate uterus: clinical implications.

The unicornuate uterus is associated with a poor reproductive outcome and many gynecological problems. We collected data from 45 women with a unicornu...
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