The perplexing entity of rudimentary uterine horn

doi:10.4236/ojog.2011.14042

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Open Journal of Obstetrics and Gynecology, 2011, 1, 217-220

doi:10.4236/ojog.2011.14042 Published Online December 2011 (http://www.SciRP.org/journal/ojog/ OJOG

Published Online December 2011 in SciRes. http://www.scirp.org/journal/OJOG

The perplexing entity of rudimentary uterine horn

Ria Malik, A. G. Radhika, Alpana Singh, Gita Radhakrishnan, Rachna Aggarwal

Department of Obs and Gynae, University College of Medical Sciences (UCMS) and GTB Hospital, Delhi, India.

Email: [email protected]

Received 15 September 2011; revised 19 October 2011; accepted 5 November 2011.

ABSTRACT

A rudimentary uterine horn may be responsible for

intriguing presentations in different stages of life. The-

se presentations often masquerade commoner gynae-

cologic disorders resulting in diagnostic and manage-

ment challenges for the treating clinicians. Whereas

many of these anomalies may be discovered during

the investigative workup for cryptomenorrhoea, dys-

menorrhoea and infertility, due to lack of symptoms

especially in a parous woman, a large proportion re-

mains undiagnosed. We report here, two interesting

presen- tations of this benign entity resulting in sig-

nificant morbidity. The first case report describes

late activation of a rudimentary horn presenting as

chronic pelvic pain. The second patient presented

with a failed second trimester induction of labor

(abortion) for fetal demise. Her examination and in-

vestigations suggested an abdominal pregnancy, yet,

on laparotomy, a 15 week pregnancy within an acces-

sory uterine horn was discovered.

Keywords: Rudimentary Uterine Horn; Chronic Pelvic

Pain; Laproscopic Management; Abdominal Pregnancy;

Ruptured Rudimentary Horn

1. INTRODUCTION

Mullerian duct malformations repr esent a wide spectrum

of anomalies, resulting from varying degrees of defects

in the development and fusion of the mesonephric ducts.

We present here, two unusual presentations of unicorn-

nuate uterus wi t h ru di mentary horn.

2. CASE I

Ms S, 37 yrs, P3L3 presented in our out patient clinic

with complaints of dull achin g lower abdominal pain sin-

ce past 4 yrs. Recent history of dysmenorrhoea was no-

ted. Her many visits to different gynecologists and sur-

geons did not prove fruitful. No symptoms suggestive of

pelvic infection, bladder or bowel pathology were noted.

Her first two children were term normal vaginal delive-

ries, third was a caesarian section with tubal ligation for

placenta previa, five years ago. Her postoperative period

was uneventful and she was not informed of anything

unusual at discharge; no papers relevant to the surgery

were available when she presented to us. Her menstrual

cycles had been regular and no period of amenorrhea

was reported. General physical examination was unre-

markable except for a pfannensteil caesarean scar. Spe-

culum examination revealed normal cervix and vagina.

On vaginal examination, uterus anteverted multiparous

size. There was a 4 cm × 4 cm size, mobile, smooth and

slightly tender right adnexal mass. Transvaginal sono-

gramphy showed a well circumscribed thick walled

complex mass 4 × 3 cms in right adnexa with partially

echogenic contents. Left ovary was normal. Provisional

diagnosis of chronic pelvic inflammatory disease (PID)

or possibly an endometrioma was considered. Her persis-

tent symptoms despite treatment for PID mandated

laparoscopy. At laparoscopy, the right ovary was found

normal, the ligated right fallopian tube was seen arising

from a 3 c m × 3 cm smooth surfaced thick muscular mass

attached at the right uterine cornu. The right round liga-

ment was attached to this mass confirming it to be rudi-

mentary uterine horn. 4 ml of dark altered blood was

aspirated from this mass. The other uterine half was

normal in shape size and contour, left ovary and tube

(ligated) were normal (Figure 1).

3. CASE II

25 yr old Ms. B, primigravida was referred by a practi-

tioner for hysterotomy following failed attempt at se-

cond trimester termination of a nonviable pregnancy.

She was married for 10 years and was under treatment

for infertility since 7 years. The hysterosalpingogram arou-

sed the suspicion of a bicornuate uterus though it was

not confirmed laparoscopically. The present pregnancy

was a natural conception. Ultrasonography done in the

first trimester did not show anything abnormal. At 5

months into pregnancy, she had an episode of vaginal

bleeding, when ultrasound revealed fetal demise. Termi-

nation of pregnancy using vaginal and systemic prosta-

glandins was unsuccessfully attempted by the practitio-

ner, and patient referred to our centre for hysterotomy.

R. Malik et al. / Open Journal of Obstetrics and Gynecology 1 (2011) 217-220

218

Figure 1. Laproscopic view of rudimentary horn with normal

uterus, causing CPP in Case 1Laparoscopic hemi hysterectomy

of the rudimentary horn was performed (Figure 2). The patient

had an uneventful post operative period. Histopathological ex-

amination confirmed uterine tissue. The patient remains sy-

mptom free till date.

Figure 2. Altered blood which was aspirated from the horn and

the resected rudimentary horn.

On examination, her vitals were stable; there was a pal-

pable abdominal mass arising form pelvis corresponding

to 14 wks gravid uterus. Speculum examination demon-

strated a single centrally placed cervix. On vaginal exa-

mination, cervix uneffaced, uterus normal sized deviated

to left side. A mobile nontender cystic mass 8 cm × 8 cm

was felt on the right side. Ultrasound revealed a normal

sized empty uterus having endometrial thickness of 5

mm. A gestational sac with a dead fetus of 15 wks was

noted to the right and anterior to uterus (Figure 3).

Both ovaries were normal. Provisional diagnosis of se-

condary abdominal pregnancy was considered. MRI re-

ported similar findings (Figure 4), further confirming the

diagnosis of secondary abdominal pregnancy.

Figure 3. Ultrasound of Case 2 showing single fetus in a mus-

cular structure outside the normal uterus.

Figure 4. MRI showing findings suggestive of abdominal

pregnancy.

An exploratory laparotomy was performed. At lapro-

tomy, the uterus, left tube and ovar y were normal. There

was a right sided 8 cm × 8 cm muscular structure, at-

tached to uterus by a fibrous band; the right tube was

seen arising from this mass. Round ligament of right side

was seen arising lateral to this mass, thereby confirming

it to be a uterine horn. The right ovary was normal. This

rudimentary horn was excised with the fetus in situ. Cut

section of the mass showed a muscular wall with a dead

fetus inside. Post operative recovery of the patient was

uneventful. Definitive con firmation of uterine tissue was

R. Malik et al. / Open Journal of Obstetrics and Gynecology 1 (2011) 217-220 219

done by histo p at hology.

4. DISCUSSION

The exact incidence of mullerian abnormalities is not

known because of normal fertility outcome in majority

of these women. Estimates range from 1% - 3.5%, with

higher percentage prevalence in infertile women (6.3%)

[1]. Unicornuate uterus is categorized in Class II of the

American Fertility Society classification (1988) of mul-

lerian anomalies. It accounts for 2.4% - 13% of all mul-

lerian anomalies [2,3]. Since fertility outcome is not

usually affected, the clinical presentation may be highly

variable; ranging from an apparently trivial dysmenor-

rhoea in an adolescent girl to intractable vague pelvic

pain in a parous woman. The presence of normal men-

strual flow further blankets the possibility of a uterine

anomaly. As high as 78% of rudimentary horns have

been known to presen t in the third decade of life o r later,

most often as acute emergencies resulting from rupture

of horn pregnancy [4]. Unless specifically sought, ultra-

sound done for other gynecologic disorders often fails to

pick up such malformations. In a review of 266 rudi-

mentary horn presen tations, sensitivity of two dimensio-

nal USG as a diagnostic tool was shown to be only 26%

[4]. A novel method of prerup ture diag nosis of rudiment-

tary horn pregnancy has been suggested by Buntugu et

al. The method involves placement of a Foley’s catheter

into uterine cavity with bulb inflated. An abdominal ul-

trasound with full bladder is then performed. Although

the method is claimed to offer better sensitivity over

conventional sonography, more studies are needed be-

fore it can be recommended for routine use [5]. Recent

studies by Ghi et al, found 3-dimensional transvaginal

ultrasonography to be extremely accurate in the diagno-

sis of uterine anomalies. They found a concordance be-

tween endoscopy and 3D ultrasound in 52 of 54 cases [6].

However, MRI where available, still remains the gold

standard for confirmation of mullerian anomalies.

Chronic pelvic pain has been reported in 15% - 20%

of women aged 18 yrs - 50 yrs [7,8,9] and is an indica-

tion for at least 40% of all gynecologic laparoscopies

[10]. Whereas 8.5% of adolescents undergoing laparo-

scopy for pelvic pain have some form of uterine mal-

formation [11], mullerian anomalies as a cause for

chronic pelvic pain in a parous woman is rarely reported

[12].

We were intrigued by the late p resentation and missed

diagnosis of existence of this horn in our patient who

had had a normal reproductive outcome including a ce-

sarean section. The patient had denied any prior history

of pelvic pain or dysmenorrhoea, suggesting recent acti-

vation of disease. Possible explanation for the late onset

of symptoms in this case could be tubal ligation inciting

accumulation of menstrual blood in the horn, thereby

precipitating pelvic pain. This phenomenon has also been

reported by Fugimoto et al [13]. Rudimentary horn as a

cause of pelvic mass and pain has even been reported

after vaginal hysterectomy in a 41 yr old lady [14]. The-

se reports suggest possible role of various “triggers”

which cause activation and presentation of such rudi-

mentary horns late in reproductive life. The diagnosis

had been completely missed at cesarean section probably

due to the small size of the horn as compared to the term

gravid uterus and the obvious altered anatomic relations.

The commonly accepted treatment for this entity is

excision through laparotomy or laparoscopy. Other treat-

ment options include endometrial ablation of accessory

horn through hysteroscopic approach; with reported sy-

mptom free period of up to 3 yrs [15]. Hysteroscopic

drainage of hematometra in a noncommunicating acces-

sory horn by using electrocautery to create a communi-

cation between the horns has al so been descr i bed [16].

Pregnancy in rudimentary horn has been noted once in

1225 ectopic pregnancies [17] and once in 76000 total

pregnancies [18]. The most significant threat is the risk

of rupture; which is estimated to be about 50% and most

commonly occurs in second trimester [19]. Various signs

and symptoms pointing to presence of a uterine anomaly

have been described, including past history of dysme-

norrhoea, pregnancy along with a freely mobile tumor,

passage of a decidual cast, absent tenderness on exami-

nation (unlike a tubal ectopic) [20] etc. But still, only a

very small percentage of these rudimentary horn preg-

nancies are accurately diagnosed pre-operatively. While

excision of this “ectopic” pregnancy along with the ac-

cessory horn remains the most common treatment mo-

dality, combined medical and surgical treatment has also

been tried. This involves fetal intracardiac potassium

chloride and intraplacental methotrexate, followed 6 wks

later by laparoscopic resection of the horn [19]. Such

combined medical, surgical modality has the advantage

of reducing the vascularity, thereby decreasing operative

blood loss [19]. Additional studies are needed before the-

se treatment modalities are widely accepted.

5. RECOMMENDATIONS

The authors would like to recommend that clinicians be

sensitive to the presence of mullerian anomalies in pa-

tients with otherwise normal gynecologic and reproduce-

tive histories. The diagnosis should be considered in

unusual presentations of pelvic pain or early pregnancy.

Although relatively insensitive, the routine ultrasound

may prove helpful if clinicians bear in mind these dif-

ferential diagnoses. Emergency staff should be alert to

the possibility of such malformations in patients pre-

senting with acute abdomen, suspected ruptured ectopic

R. Malik et al. / Open Journal of Obstetrics and Gynecology 1 (2011) 217-220

220

or even failed induction with adequate doses of prosta-

glandins. A thorough inspection of the pelvis should be

performed at the time of any operative procedure. Pro-

phylactic resection of a noncommunicating uterine horn

should be considered in patients with incidental disco-

very on laprotomy to prevent endometriosis and possible

future horn pre gnancy.

[10] Howard, F.M. (1993) The role of laparoscopy in chronic

pelvic pain: Promise and pitfalls. Obstetrical and Gyne-

cological Survey, 48, 357-387.

doi:10.1097/00006254-199306000-00001

[11] Vercellini, P., Fedele, L., Arcaini, L., et al. (1989)

Laparoscopy in the diagnosis of chronic pelvic pain in

adolescent women. Journal of Reproductive Medicine, 34,

827.

[12] Howard, F.M. (2003) The role of laparoscopy in chronic

pelvic pain patient. Clinical Obstetrics and Gynecology,

46, 749-766. doi:10.1097/00003081-200312000-00004

6. CONFLICT OF INTEREST

We declare that we have no conflict of interests. [13] Fugimoto, V.Y., Klein, N.A. and Miller, P.B. (1998) Late

onset hematome tra and he matosalpi nx in a woman wit h a

noncommunicating uterine horn: A case report. Journal

of Reproductive Medicine, 43, 465-467.

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OJOG