Ruptured ectopic pregnancy in non-communicating right rudimentary horn: A case report

Ruptured ectopic pregnancy in non-communicating right rudimentary

horn: A case report

Case Report

Ruptured ectopic pregnancy innon-communicating right rudimentaryhorn: A case report

Ahmed S. Elagwany*, Hisham H. Elgamal, Tamer M. Abdeldayem

Department of Obstetrics and Gynecology, Alexandria University, Egypt

a r t i c l e i n f o

Article history:

Received 19 August 2013

Accepted 4 October 2013

Available online xxx

Keywords:

Acute abdomen

Hemoperitoneum

Rudimentary horn

Rupture

Ultrasound

a b s t r a c t

Rudimentary horn is a developmental anomaly of the uterus, and pregnancy in a non-

communicating rudimentary horn is very difficult to diagnose before it ruptures. As the

fetus enlarges in the rudimentary horn, the chances of rupture in the first or second

trimester are increased. Catastrophic hemorrhage results in increased maternal and

perinatal mortality and morbidity. To date, management of such cases remains a challenge

due to diagnostic dilemma. Expertise in ultrasonography and early resort to surgical

management are lifesaving in such cases. A case of undiagnosed rudimentary horn preg-

nancy presented to our department in shock with features of acute abdomen, and the

diagnosis was confirmed at laparotomy that revealed ruptured rudimentary horn preg-

nancy. And excision of the accessory horn was done.

Copyright ª 2013, Indraprastha Medical Corporation Ltd. All rights reserved.

1. Introduction

Congenital uterine malformations are the result of abnormal

Mullerian duct development, fusion, canalization and septal

resorption. The prevalence of congenital uterine anomalies

among fertilewomen is reported as 1:200e1:600.Where there is

atresia of one of the Mullerian duct, there is a unicornuate

uterus with a single tube. Its true incidence is unknown and is

approximately estimatedat a rate of about 1 in250.Unicornuate

uterusmay also have a rudimentary horn. Rudimentary horn is

one of the rarest congenital uterine anomalies. The prevalence

of unicornuate uterus with rudimentary horn is even rarer i.e.

1:100,000.1 The rudimentary horn may consist of a functional

cavity, or itmaybe a small solid lumpof theuterinemusclewith

no functional endometrium. In a series of 366 cases with rudi-

mentary horn, non-communicating horn accounted for 92% of

cases, and renal anomaly was found in 36% cases. Unilateral

renal agenesis was found in 38% cases in another series.2

Provided there is no obstruction to menstrual flow, these

uterine anomalies present few problems in the absence of

pregnancy. Pregnancy in a rudimentary horn is rare. The

incidence of rudimentary horn pregnancy is difficult to

calculate. Frequently quoted figures are between 1 per 76,000

and 1 per 140,000 pregnancies.3

A case of pregnancy occurring in a rudimentary horn with

consequent rupture is reported.

2. Case report

An unusual case of a 25-year-old woman, G1P0, with a preg-

nancy of 18 weeks, presented to the emergency unit of our

* Corresponding author. El-Shatby Maternity Hospital, Alexandria University, Alexandria, Egypt. Tel.: þ20 1228254247.E-mail address: [email protected] (A.S. Elagwany).

Available online at www.sciencedirect.com

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a p o l l o m e d i c i n e x x x ( 2 0 1 3 ) 1e4

Please cite this article in press as: Elagwany AS, et al., Ruptured ectopic pregnancy in non-communicating right rudimentaryhorn: A case report, Apollo Medicine (2013), http://dx.doi.org/10.1016/j.apme.2013.10.002

0976-0016/$ e see front matter Copyright ª 2013, Indraprastha Medical Corporation Ltd. All rights reserved.http://dx.doi.org/10.1016/j.apme.2013.10.002

department with acute abdominal pain and hypovolemic

shock. The patient did not seek antenatal care before admis-

sion. Pregnancy was diagnosed by urine pregnancy test after a

missed period for one week. Ultrasound was not done till

admission .The patient was illiterate and living in a rural area

far away from any health facility. She was not complaining

so she did not seek medical advice. No other significant his-

tory was noted.

On examination, the patient was drowsy, in agony and in a

nearly shock state with pale, cold, clammy extremities, a

thready pulse of 120 beats/minute and arterial blood pressure

of 70/40 mmHg.

Abdominal examination revealed distention and tenderness

all over. On vaginal examination, mild vaginal bleeding was

noted. A speculum examination did not reveal any cervical or

vaginalpathology.Thecervicaloswas tightlyclosed.Thepatient

was resuscitatedwith intravenous fluids and blood transfusion.

Further assessment with transabdominal ultrasound

showed a free floating nonviable fetus of 18weeks gestation in

abdominal cavity with anhydramnios and moderate abdom-

inal collection surrounding. Absent visual continuity of myo-

metrial tissue surrounding the fetus was noted. The placenta

was intrauterine, lying posterior with no retroplacental clots

seen. Abdominal tapping with spinal needle revealed non

clotted dark bloody collection.

In view of the previous data, a provisional diagnosis of a

ruptured uterus with a differential diagnosis of an abdominal

pregnancy was made.

An emergency laparotomy was performed immediately

after resuscitation. Intraoperative findings revealed a uni-

cornuate uterus with a ruptured pregnancy in a right rudi-

mentaryhorn.Also, adead fetusfloating in theperitoneal cavity

with blood clots surrounding was noted. The placenta was still

attached to the cavity of the rudimentary horn (Figs. 1 and 2).

It was, however, difficult to visualize the uterine septum.

No communication from the horn to the contralateral uterine

cavity was seen. The uterus, lying separate in the pelvis, was

soft in consistency, globular and enlarged to a size consistent

with tenweeks. The right fallopian tube and ovary were found

healthy and were attached normally to the unicornuate

uterus.

Excision of the rudimentary horn and the right fallopian

tube with conservation of the right ovary was done together

with the nonviable fetus and the placenta. All the pedicles

were secured with good hemostasis. A total volume of nearly

2 L of hemoperitoneum blood was also removed. Both the

ureters and kidneys were normal. Due to the significant blood

loss throughout the operation, the patient was given a

massive blood transfusion of four units and two units of

plasma transfusion.

The excised specimen was sent for histopathological ex-

amination, which was reported as (sections from the uterine

horn show areas of hemorrhage and necrosis. Section from

the fallopian tube was morphologically normal. Sections of

the placenta showfibrosed chorionic villi with syncytial knots.

No villitis was seen). Histopathology confirmed the diagnoses

(ruptured pregnant uterine rudimentary horn).

Post-operatively, hemoglobin level was 9.8 g/dl. Patient had

a smooth postoperative recovery. The patient was counseled

for contraception with oral pills for one year. The patient was

discharged healthy two days after admission.

Follow-up appointment was planned for intravenous uro-

gram to rule out any associated renal anomalies. No associ-

ated renal anomaly was diagnosed. The patient was

Fig. 1 e A nonviable fetus with placenta attached to ruptured right rudimentary horn.



scheduled for a hysterosalpingogram 6 weeks post-

operatively before any further decisions on future pregnancy.

3. Discussion

The incidence of uterine anomalies ranges between 0.5% and

2%. Unicornuate uterus (Type II) can further be classified as

those having an endometrial cavity and those with no endo-

metrial cavity, and 90% of the cases are those with an endo-

metrial cavity. A further division can be between the

communicating and non-communicating types. Identification

of unicornuate uterus is almost always accidental, as it is

usually asymptomatic until reproductive age. Initial presen-

tation often takes place with a recurrent first trimester

miscarriage (5e10%), a second trimester loss (25%), or during

an infertility work-out.4

Mariceau and Vassal published the first description of a

rudimentary horn pregnancy in 1669, and 600 cases have since

been described.5 Pregnancies occur in both communicating

and non-communicating horns in proportion to their relative

incidence and are equally likely to rupture. Neonatalmortality

is very high as most cases are emergency laparotomies after

uterine rupture at premature gestational age. Maternal mor-

tality is low (0.5%) but morbidity is very high in view of

massive blood loss and morbidly adherent placentation.2,6

In cases with successful pregnancy, the risks of recurrent

early trimester loss andmid-trimester loss are increased, with

preterm delivery and abnormal fetal lie contributing factors.

Pregnancy in a rudimentary horn is a form of ectopic

pregnancy and it can cause mortality or severe morbidity. It

can occur in both communicating and non-communicating

types. In the case of a non-communicating rudimentary

horn like our patient had, it is postulated that the fertilization

was possibly due to transperitoneal migration of the sperm.7

The outcomes of pregnancy in rudimentary horns are

ectopic pregnancy (2.7%), first trimester loss 24.3%, second

trimester abortion 9.7%, preterm delivery 20.1%, intrauterine

demise (10.5%) and 49.9% live birth up to 28e30 weeks of

gestation. Uterine rupture in the second trimester is usually a

life-threatening condition resulting from a much thinner myo-

metrial part of the rudimentary horn, with the non-functional

endometrium leading to placenta adherence anomalies.8

The prerupture diagnosis of pregnancy in rudimentary

horn has drastically reduced maternal mortality.7 But the

sensitivity of ultrasound to detect prerupture rudimentary

horn pregnancy is very poor (30%),8 probably because of rarity

of the diagnosis and non-familiarity of the radiologists about

this potentially lethal condition. Early diagnosis before

rupture can be managed laparoscopically.9

Prerupture diagnosis of rudimentary horn pregnancy is

challenging. A careful ultrasound in the first trimester with a

high index suspicion, one should be able to make a diagnosis

of pregnancy in the rudimentary horn and sensitivity de-

creases as the pregnancy advances.10 In such cases magnetic

resonance imaging (MRI) is very useful not only in confirming

the diagnosis, it also helps to plan the surgery.11 Tubal preg-

nancy, cornual pregnancy, and abdominal pregnancy are

common sonographic and clinical misdiagnosis. It is very

difficult to establish diagnosis in second trimester due to lack

of definitive clinical criteria.

Tsafrir et al. proposed the following criteria for ultrasono-

graphic diagnosis: 1. a pseudo pattern of an asymmetrical

bicornuate uterus, 2. absent visual continuity tissue sur-

rounding the gestation sac and the uterine cervix, and 3. the

presence of myometrial tissue surrounding the gestation sac.8

Three-dimensional ultrasound imaging and MRI are useful

tools in the improvement of diagnostic accuracy, guiding both

counseling and surgical planning.12

The traditional and established treatment for rudimentary

horn pregnancy is surgical removal of the pregnant horn even

in unruptured case to prevent rupture and recurrent rudi-

mentary horn pregnancy.13,14

Laparoscopic excision of the rudimentary horn pregnancy

prior to rupture has been done successfully since last two de-

cades.10,11 Renal anomalies are found in 36% of cases; hence it is

mandatory to assess these women prior to surgery and if

required better to doureterolysis before the excision of thehorn.

Medical management with methotrexate during early preg-

nancy in the rudimentary horn has also been used

successfully.14

In our patient, the diagnosis of ectopic pregnancy was only

established when she went into hypovolemic shock at 18

weeks. The non-communicating rudimentary horn rupture

was only confirmed intraoperatively and removal of the

rudimentary horn and ipsilateral fallopian tube was done.

This was to reduce the risk of the patient having another

ectopic pregnancy in the future.

This case further raises the question of whether routine

excision of rudimentary horn be undertaken in women with

Fig. 2 e Site of perforation in right rudimentary horn.

a p o l l o m e d i c i n e x x x ( 2 0 1 3 ) 1e4 3


unicornuate uterus as a prophylaxis to prevent such catas-

trophes. In our opinion, routine laparoscopic excision of

rudimentary horn with ipsilateral fallopian tube should be

offered to these women and those refusing should be

adequately counseled regarding the potential complications

and if pregnancy occurs in rudimentary horn first trimester

laparoscopic excision should be done.

4. Conclusion

Rudimentary horn pregnancy is a rare complication which

carries grave risk to the mother. More than 90% of the cases

present in second trimester with intraperitoneal hemorrhage

due to rupture of the horn. Ideally, history of repeated abor-

tions must be investigated before conception to exclude

Mullerian duct malformation. In addition, an intravenous

pyelogram is indicated because of high incidence of associ-

ated urinary anomalies in the presence of genital anomalies.

Diagnosis prior to rupture should be the concern in early

pregnancy with either ultrasound or MRI to prevent life-

threatening complications. The recent trend is to do laparo-

scopic excision of the rudimentary horn; laparotomy is still an

option when the patient is in shock.

Conflicts of interest

All authors have none to declare.

r e f e r e n c e s

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2. Jayasinghe Y, Rane A, Stalewski H. The presentation and earlydiagnosis of the rudimentary uterine horn. Obstet Gynecol.2005;105:1456e1467.

3. Acien P. Incidence of Mullerian defects in fertile and infertilewomen. Hum Reprod. 1997;12:1372e1376.

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10. Sharma D, Usha MG, Gaikwad R. Laparoscopic resection ofunruptured rudimentary horn pregnancy. Int J ReprodContracept Obstet Gynecol. 2013;2:95e98.

11. Edelman AB, Jensen JT, Lee DM. Successful medical abortionof a pregnancy within a noncommunicating rudimentaryuterine horn. Am J Obstet Gynecol. 2003;189:886e887.

12. Nahum G, Stanislaw H, McMohan C. Preventing ectopicpregnancies: how often does transperitoneal transmigrationof sperm occur in effecting human pregnancy? Br J ObstetGynecol. 2004;111:706e714.

13. Reichman D, Laufer MR, Robinson BK. Pregnancy outcomes inunicornuate uteri: a review. Fertil Steril. 2009;91:1886e1894.

14. Sunilkumar KS, Yaliwal LV, Amarnath A. Rupturedrudimentary horn of the unicornuate uterus at 16 weeks ofpregnancy: a case report. Int J Reprod Contracept Obstet Gynecol.2013;2:248e250.



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Ruptured ectopic pregnancy in non-communicating right rudimentary horn: A case report

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Transcript of Ruptured ectopic pregnancy in non-communicating right rudimentary horn: A case report