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Rudimentary horn pregnancy associated with pre-eclampsia
367
Int. J. Gynecol. Obstet., 1989,30: 367-370 International Federation of Gynecolo8y and Obstetrics
Rudimentary horn pregnancy associated with pre-eclampsia J .O. Emembolu Department of Obstetricsand Gynaecology. Ahmadu Bell0 UniversityTeaching Hospital, (ABUTHZa), Zaria (Nigeria) (Received May 27th, 1988) (Revised and accepted November 22nd. 1988)
Abstract The unusual association of pre-eclampsia with a pregnancy in the rudimentary horn of the uterus in a multigravid patient is reported. The infrequent development of rudimentary horn pregnancy beyond the twentieth week of gestation may explain the rarity of this complication. The possible effect of rapid distension of the rudimentary horn with exposure of the myometrial surface to overwhelming fetal antigenic proteins may be of relevance in the development of pre-eclampsia in this case. Keywords: Rudimentary horn pregnancy; Uterine overdistension; Immunological overload; Pre-eclampsia. Introduction Pregnancy in the rudimentary horn of the uterus is a rare form of ectopic gestation, which usually manifests between the twelfth and twentieth week of gestation. The incidence is approximately 1 in 100,000 pregnancies [9], or 1 in 5000 to 15,000 ectopic pregnancies [6,11]. It usually manifests as an acute abdominal emergency from rupture W, 111, rarely proceeding to secondary abdominal pregnancy in 10% [9] and even more rarely achieving term with the delivery of a live baby, dead fetus or lithopedion [ 111. 0020-7292/89/$03.50 0 1989 International Federation of Gynecology and Obstetrics Published and Printed in Ireland
The rarity of this condition alone justifies its publication but this is more so when there is an associated complication such as preeclampsia as in this case. Case report Mrs. C.I. was a 25-year-old multigravida (Para 4”) whose last childbirth was in September, 1979. She was admitted from the antenatal booking clinic on June 18, 1985 at 26 weeks gestation with the complaints of severe headache, epigastric pains and visual disturbance of 1 h duration. Her last menstrual period was December 24, 1984. Physical examination revealed a young obese woman of height 1.55 m and weight 79.0 kg who was neither pale nor jaundiced. She had marked facial puffiness associated with generalised edema up to the abdomen and the fingers. Her pulse was 96 beats/min, blood pressure 200/130 mmHg but the heart sounds were normal. There was no abnormality at funduscopy. There was tenderness over the right hypochondrium but otherwise no abnormality was noted on abdominal examination. The uterus was consistent with a gestation of 24-26 weeks, the fetal parts were palpable and the fetal heart sounds heard with sonicaid. Vaginal examination revealed an unfavourable cervix with the ostium closed otherwise no pelvic abnormality was suspected. The results of investigations showed a hemoglobin of 16 g/dl, hematocrit 48%, Case Report
368
Emembolu
normal white cell and platelet counts. The urea and electrolytes were normal but the serum uric acid level was 0.54 mmol/l. The liver function tests and serum proteins were normal and her blood group was 0 rhesus positive. Urinalysis revealed significant albuminuria (500 mg/dl) but neither glycosuria nor acetonuria using Combur 9 Test (Boehringer Mannheim). A diagnosis of fulminating pre-eclampsia in a multiparous patient was made and she was commenced on lytic cocktail (pethidine HCl 100 mg, promethazine HCl 50 mg and chlorpromazine HCI 50 mg), infusion of hydrallazine HCl80 mg in 1 1 of normal saline solution titrated to control the blood pressure. Thereafter, induction of labor was commenced with escalating infusion of syntocinon (Sandoz) 20 international units in 1 1 of 5% dextrose solution commencing at 10 drops/min. The infusion rate was increased every 30 min to a maximum rate of 60 drops/ min. Depending on the uterine contractions, the dosage of syntocinon per liter of infusion was gradually increased up to 100 international units in a liter of solution. The review of progress revealed that the cervix remained long and uneffaced but had dilated to admit the index finger, though the uterine contents could not be felt. Meanwhile, the blood pressure was labile and ranged from 130/90 mmHg to 230/140 mmHg. Her symptoms had however abated under the induced sedation and she had not fitted. Her urine output, monitored following bladder catheterisation, over the preceding 24 h was 600 ml and albuminuria (500 mg/dl) was consistent. A bolus intravenous injection of 120 mg frusemide did not improve the urinary output. A decision to terminate the pregnancy by laparotomy was therefore made in view of these findings. At laparotomy, a congenitally malformed uterus consistent with a 24 weeks gestation and containing an intact gestational sac in its left rudimentary horn was observed (Fig. 1). Hysterotomy with delivery of the female fetus in its sac and placenta was easily achieved and Int J Gynecol Obstet 30
Diagrammatic representation of the rudimentary 1. horn pregnancy associated with fulminating pre-eclampsia.
Fig.
bilateral tubal ligation performed by the modified Pomeroy technique. There was no abnormality of the fetus and placenta at inspection. When probed, the rudimentary horn did not communicate with the normally formed and enlarged right uterus and cervix. The patient’s subsequent recovery was rapid and unremarkable. Her blood pressure stabilised to 100/600 mmHg within 24 h of delivery and antihypertensive therapy was discontinued by 48 h. There was marked diuersia of 2.4-4.6 1 of urine over the subsequent 96 h while the intravenous injection of 80 mg frusemide was discontinued after the second dose. The electrolyte level showed slight derrangement but the uric acid level was 0.22 mmol/l and normal by the third postoperative day. She, however, developed puerperal psychosis with agitation and confusion on the seventh post-partum day. This responded rapidly to chlorpromazine HCl 100 mg intramuscularly eight hourly for 4 days. She was discharged with a blood pressure of 100/70 mmHg and hematocrit of 35%. The urea and electrolyte levels, serum uric acid and liver function tests were within normal range. The hysterosalpingogram (Fig. 2) per-
Rudimentaryhorn pregnancy and pre-eclampsia
Fig. 2. tion.
369
HSG shows normal right unicornuate uterus associated with al xdimentary horn. The tube terminates at the point of
formed at the follow-up clinic shows the normal uterine horn with the tube revealed only for a short distance to the point of the tubal ligation. The left-sided rudimentary horn was not visualised. Discussion Pregnancy in a rudimentary horn is a rarity and its natural history compels that complication arise early with termination of the pregnancy by the twentieth week of gestation in most instances [6,9,11]. As such, complication associated with late gestations of pregnancy are either not observed or are underreported in the literature. Furthermore, severe pre-eclampsia is uncommon in the multigravid patients and may be related to underlying renal or vascular pathology, and low socio-economic status. Its occurrence in the second trimester of pregnancy may be associated with molar pregnancy. Rhesus isoimmunisation or multiple gestation. Pre-eclampsia has been reported in association with extra-uterine pregnancies [l] but not
with advanced pregnancy in the rudimentary horn. This of course is related to the infrequent continuation of this pregnancy into the third trimester. This presentation in a multigravid previously normotensive patient furthermore raises questions as to the validity of the various theories of the disease. The present confusion regarding its etiology prompted Jeffcoate [7] to coin the phrase “a disease of theories” in relation to preeclampsia/eclampsia. The distension theory of Sophian, supported by others [3,4,5,14] suggests that isometric tension in the myometrium induced utero-renal reflux leading to various degrees of renal cortical ischemia resulting in salt albuminuria and hypertension. retention, However, this phenomenon was not easily explained in the instances of extra-uterine pregnancy associated with pre-eclampsia [1,2]. Redman et al. [13] presupposed that maternal recognition of fetal antigens generates a protective response that is imperfectly developed in pre-eclampsia. The phenomenon was confirmed by Jenkins et al. [8] and Platt et al. [12] implicating reduced incomCase Report
370
Emembolu
patibility and immune response to fetal antigens in pre-eclampsia/eclampsia. Mitchison [lo] had earlier explained that multiple pregnancy, hydatidiform mole, hydrops fetalis and triploidy with relatively large placental mass may cause overwhelming antigenic challenge via the mechanism of immunological overload with consequent impairment of immune responsiveness and therefore preeclampsia. The rapid and unequal stretch between the placental and maternal surfaces against uterine resistance [2] may be the operational factor in this case which has caused greater disruption of the endometrial barrier exposing vast areas of unprotected myometrium of the rudimentary horn to the antigenic placental tissue. Such a situation may therefore lead to failure of maternal immune response and hence the occurrence of severe pre-eclampsia in association with a second trimester rudimentary horn pregnancy in this multigravida. References Anderton KJ, Duncan SLB, Lunt RL: Advanced abdominal pregnancy with severe pre-eclampsia. Br J Obstet Gynaecol83: 90, 1976. Benjamin F, Graig CJ: Uterine distension and preeclamptic toxaemia. J Obstet Gynaecol Br Commonw 68: 827, 1961. Browne FJ: Aetiology of pre-eclamptic toxaemia and eclampsia. Lancet i: 115, 1958.
Int J Gynecol Obstet30
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9 10 11 12
13
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Corner GW, Csapo A: Action of ovarian hormones on uterine muscle (Ciba Foundation Lecture). Br Med J I: 687,1953. Green HN, Hopewell JD, Threlfell CJ: Plasma pentose levels in pre-eclampsia and their aetiological significance. BrMedJ2:571,1951. Holden R, Hart P: First trimester rudimentary horn pregnancy: pre-rupture USG diagnosis. Obstet Gynecol 6J(3 Suppl): 565,1983. Jeffcoate TNA: Pre-eclampsiaIeclampsia: disease of theories. Proc R Sot Med 59: 397,1966. Jenkins DM, Need JA, Scott JS, Morris H, Pepper M: Human leucocyte antigens and mixed lymphocyte reaction in severe pm-eclampsia. Br Med J 1: 542,197s. Johansen K: Pregnancy in rudimentary horn. Obstet Gynecol34: 805,1969. Mitchison NA: Induction of immunological paralysis in two zones of dosage. Proc R Sot BiolJ6J: 275,1945. O’Learly JL, O’Leary JA: Rudimentary horn pregnancy. Obstet Gynecol22: 371,1%8. Platt R, Stewart AE, Emery EW: The aetiology, incidence and heredity of pre-eclamptic toxaemia of pregnancy. Lancet i: 552.1958. Redman CWG, Bodmer WF, Bodmer JF, Beilin LJ, Bonnar J: HLA antigens in severe pre-eclampsia. Lancet iii: 397, 1978. Reynolds SRM: Conditions of blood flow in gravid uterus. Am J Obstet Gynecol59: 529, 1950.
Address for reprints: J.O. Emenbolu Department of Obstetrics and Gynaecology Abmadu Be110University Teaching Hospital Zaria, Nigeria
Int. J. Gynecol. Obstet., 1989,30: 367-370 International Federation of Gynecolo8y and Obstetrics
Rudimentary horn pregnancy associated with pre-eclampsia J .O. Emembolu Department of Obstetricsand Gynaecology. Ahmadu Bell0 UniversityTeaching Hospital, (ABUTHZa), Zaria (Nigeria) (Received May 27th, 1988) (Revised and accepted November 22nd. 1988)
Abstract The unusual association of pre-eclampsia with a pregnancy in the rudimentary horn of the uterus in a multigravid patient is reported. The infrequent development of rudimentary horn pregnancy beyond the twentieth week of gestation may explain the rarity of this complication. The possible effect of rapid distension of the rudimentary horn with exposure of the myometrial surface to overwhelming fetal antigenic proteins may be of relevance in the development of pre-eclampsia in this case. Keywords: Rudimentary horn pregnancy; Uterine overdistension; Immunological overload; Pre-eclampsia. Introduction Pregnancy in the rudimentary horn of the uterus is a rare form of ectopic gestation, which usually manifests between the twelfth and twentieth week of gestation. The incidence is approximately 1 in 100,000 pregnancies [9], or 1 in 5000 to 15,000 ectopic pregnancies [6,11]. It usually manifests as an acute abdominal emergency from rupture W, 111, rarely proceeding to secondary abdominal pregnancy in 10% [9] and even more rarely achieving term with the delivery of a live baby, dead fetus or lithopedion [ 111. 0020-7292/89/$03.50 0 1989 International Federation of Gynecology and Obstetrics Published and Printed in Ireland
The rarity of this condition alone justifies its publication but this is more so when there is an associated complication such as preeclampsia as in this case. Case report Mrs. C.I. was a 25-year-old multigravida (Para 4”) whose last childbirth was in September, 1979. She was admitted from the antenatal booking clinic on June 18, 1985 at 26 weeks gestation with the complaints of severe headache, epigastric pains and visual disturbance of 1 h duration. Her last menstrual period was December 24, 1984. Physical examination revealed a young obese woman of height 1.55 m and weight 79.0 kg who was neither pale nor jaundiced. She had marked facial puffiness associated with generalised edema up to the abdomen and the fingers. Her pulse was 96 beats/min, blood pressure 200/130 mmHg but the heart sounds were normal. There was no abnormality at funduscopy. There was tenderness over the right hypochondrium but otherwise no abnormality was noted on abdominal examination. The uterus was consistent with a gestation of 24-26 weeks, the fetal parts were palpable and the fetal heart sounds heard with sonicaid. Vaginal examination revealed an unfavourable cervix with the ostium closed otherwise no pelvic abnormality was suspected. The results of investigations showed a hemoglobin of 16 g/dl, hematocrit 48%, Case Report
368
Emembolu
normal white cell and platelet counts. The urea and electrolytes were normal but the serum uric acid level was 0.54 mmol/l. The liver function tests and serum proteins were normal and her blood group was 0 rhesus positive. Urinalysis revealed significant albuminuria (500 mg/dl) but neither glycosuria nor acetonuria using Combur 9 Test (Boehringer Mannheim). A diagnosis of fulminating pre-eclampsia in a multiparous patient was made and she was commenced on lytic cocktail (pethidine HCl 100 mg, promethazine HCl 50 mg and chlorpromazine HCI 50 mg), infusion of hydrallazine HCl80 mg in 1 1 of normal saline solution titrated to control the blood pressure. Thereafter, induction of labor was commenced with escalating infusion of syntocinon (Sandoz) 20 international units in 1 1 of 5% dextrose solution commencing at 10 drops/min. The infusion rate was increased every 30 min to a maximum rate of 60 drops/ min. Depending on the uterine contractions, the dosage of syntocinon per liter of infusion was gradually increased up to 100 international units in a liter of solution. The review of progress revealed that the cervix remained long and uneffaced but had dilated to admit the index finger, though the uterine contents could not be felt. Meanwhile, the blood pressure was labile and ranged from 130/90 mmHg to 230/140 mmHg. Her symptoms had however abated under the induced sedation and she had not fitted. Her urine output, monitored following bladder catheterisation, over the preceding 24 h was 600 ml and albuminuria (500 mg/dl) was consistent. A bolus intravenous injection of 120 mg frusemide did not improve the urinary output. A decision to terminate the pregnancy by laparotomy was therefore made in view of these findings. At laparotomy, a congenitally malformed uterus consistent with a 24 weeks gestation and containing an intact gestational sac in its left rudimentary horn was observed (Fig. 1). Hysterotomy with delivery of the female fetus in its sac and placenta was easily achieved and Int J Gynecol Obstet 30
Diagrammatic representation of the rudimentary 1. horn pregnancy associated with fulminating pre-eclampsia.
Fig.
bilateral tubal ligation performed by the modified Pomeroy technique. There was no abnormality of the fetus and placenta at inspection. When probed, the rudimentary horn did not communicate with the normally formed and enlarged right uterus and cervix. The patient’s subsequent recovery was rapid and unremarkable. Her blood pressure stabilised to 100/600 mmHg within 24 h of delivery and antihypertensive therapy was discontinued by 48 h. There was marked diuersia of 2.4-4.6 1 of urine over the subsequent 96 h while the intravenous injection of 80 mg frusemide was discontinued after the second dose. The electrolyte level showed slight derrangement but the uric acid level was 0.22 mmol/l and normal by the third postoperative day. She, however, developed puerperal psychosis with agitation and confusion on the seventh post-partum day. This responded rapidly to chlorpromazine HCl 100 mg intramuscularly eight hourly for 4 days. She was discharged with a blood pressure of 100/70 mmHg and hematocrit of 35%. The urea and electrolyte levels, serum uric acid and liver function tests were within normal range. The hysterosalpingogram (Fig. 2) per-
Rudimentaryhorn pregnancy and pre-eclampsia
Fig. 2. tion.
369
HSG shows normal right unicornuate uterus associated with al xdimentary horn. The tube terminates at the point of
formed at the follow-up clinic shows the normal uterine horn with the tube revealed only for a short distance to the point of the tubal ligation. The left-sided rudimentary horn was not visualised. Discussion Pregnancy in a rudimentary horn is a rarity and its natural history compels that complication arise early with termination of the pregnancy by the twentieth week of gestation in most instances [6,9,11]. As such, complication associated with late gestations of pregnancy are either not observed or are underreported in the literature. Furthermore, severe pre-eclampsia is uncommon in the multigravid patients and may be related to underlying renal or vascular pathology, and low socio-economic status. Its occurrence in the second trimester of pregnancy may be associated with molar pregnancy. Rhesus isoimmunisation or multiple gestation. Pre-eclampsia has been reported in association with extra-uterine pregnancies [l] but not
with advanced pregnancy in the rudimentary horn. This of course is related to the infrequent continuation of this pregnancy into the third trimester. This presentation in a multigravid previously normotensive patient furthermore raises questions as to the validity of the various theories of the disease. The present confusion regarding its etiology prompted Jeffcoate [7] to coin the phrase “a disease of theories” in relation to preeclampsia/eclampsia. The distension theory of Sophian, supported by others [3,4,5,14] suggests that isometric tension in the myometrium induced utero-renal reflux leading to various degrees of renal cortical ischemia resulting in salt albuminuria and hypertension. retention, However, this phenomenon was not easily explained in the instances of extra-uterine pregnancy associated with pre-eclampsia [1,2]. Redman et al. [13] presupposed that maternal recognition of fetal antigens generates a protective response that is imperfectly developed in pre-eclampsia. The phenomenon was confirmed by Jenkins et al. [8] and Platt et al. [12] implicating reduced incomCase Report
370
Emembolu
patibility and immune response to fetal antigens in pre-eclampsia/eclampsia. Mitchison [lo] had earlier explained that multiple pregnancy, hydatidiform mole, hydrops fetalis and triploidy with relatively large placental mass may cause overwhelming antigenic challenge via the mechanism of immunological overload with consequent impairment of immune responsiveness and therefore preeclampsia. The rapid and unequal stretch between the placental and maternal surfaces against uterine resistance [2] may be the operational factor in this case which has caused greater disruption of the endometrial barrier exposing vast areas of unprotected myometrium of the rudimentary horn to the antigenic placental tissue. Such a situation may therefore lead to failure of maternal immune response and hence the occurrence of severe pre-eclampsia in association with a second trimester rudimentary horn pregnancy in this multigravida. References Anderton KJ, Duncan SLB, Lunt RL: Advanced abdominal pregnancy with severe pre-eclampsia. Br J Obstet Gynaecol83: 90, 1976. Benjamin F, Graig CJ: Uterine distension and preeclamptic toxaemia. J Obstet Gynaecol Br Commonw 68: 827, 1961. Browne FJ: Aetiology of pre-eclamptic toxaemia and eclampsia. Lancet i: 115, 1958.
Int J Gynecol Obstet30
4
5
6
7 8
9 10 11 12
13
14
Corner GW, Csapo A: Action of ovarian hormones on uterine muscle (Ciba Foundation Lecture). Br Med J I: 687,1953. Green HN, Hopewell JD, Threlfell CJ: Plasma pentose levels in pre-eclampsia and their aetiological significance. BrMedJ2:571,1951. Holden R, Hart P: First trimester rudimentary horn pregnancy: pre-rupture USG diagnosis. Obstet Gynecol 6J(3 Suppl): 565,1983. Jeffcoate TNA: Pre-eclampsiaIeclampsia: disease of theories. Proc R Sot Med 59: 397,1966. Jenkins DM, Need JA, Scott JS, Morris H, Pepper M: Human leucocyte antigens and mixed lymphocyte reaction in severe pm-eclampsia. Br Med J 1: 542,197s. Johansen K: Pregnancy in rudimentary horn. Obstet Gynecol34: 805,1969. Mitchison NA: Induction of immunological paralysis in two zones of dosage. Proc R Sot BiolJ6J: 275,1945. O’Learly JL, O’Leary JA: Rudimentary horn pregnancy. Obstet Gynecol22: 371,1%8. Platt R, Stewart AE, Emery EW: The aetiology, incidence and heredity of pre-eclamptic toxaemia of pregnancy. Lancet i: 552.1958. Redman CWG, Bodmer WF, Bodmer JF, Beilin LJ, Bonnar J: HLA antigens in severe pre-eclampsia. Lancet iii: 397, 1978. Reynolds SRM: Conditions of blood flow in gravid uterus. Am J Obstet Gynecol59: 529, 1950.
Address for reprints: J.O. Emenbolu Department of Obstetrics and Gynaecology Abmadu Be110University Teaching Hospital Zaria, Nigeria