Non-Communicating inactive rudimentary horn of the uterus presenting with dysmenorrhoea—a case report of successful laparoscopic excision

doi:10.4236/ojog.2011.14041

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Open Journal of Obstetrics and Gynecology, 2011, 1, 213-216 OJOG

doi:10.4236/ojog.2011.14041 Published Online December 2011 (http://www.SciRP.org/journal/ojog/).

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

Non-Communicating inactive rudimentary horn of the uterus

presenting with dysmenorrhoea—a case report of successful

laparoscopic excision

Rachana Dwivedi1, Keerthi Perera2, Padma Eedarapalli1

1The Royal Bournemouth and Christchurch Hospitals NHS Foundation Trust, Bournemouth, UK;

2Poole Hospital NHS Foundation Trust, Poole, UK.

Email: [email protected]

Received 7 September 2011; revised 17 October 2011; accepted 27 October 2011.

ABSTRACT

Uterine developmental abnormalities can cause both

obstetric and gynaecological complications. Non-func-

tioning rudimentary uterine horn is a rare cause of

dysmenorrhoea which usually starts after menarche.

Cases with lateral/inferior arrangement of blood su-

pply to the functioning rudimentary horn and lapa-

scopic removal have been described in literature. Ours

is a unique case of non-functioning rudimentary horn

with an unusual medial arrangement of vasculature

successfully treated by laparoscopic excision. We also

present a brief review of the literature. The patient

presented with irretractable dysmenorrhoea despite

hormonal manipulation and analgesics. Following the

diagnosis using laparoscopy and hysteroscopy and

MRI, the left sided non-communicating rudimentary

horn with inactive endometrium was removed lapa-

roscopically. Her symptoms were r esolved and this was

followed by successful pregnancies. Diagnosis of ru-

dimentary horn with inactive endometrium is diffi-

cult. Ultrasound is unreliable. MRI, 3D CT scan and

3D ultrasound are gaining popularity. Accurate, prior

identification of the type using laparoscopy and hys-

teroscopy if necessary is essential. This helps in sur-

gical planning. The laparoscopic approach is increa-

singly being used to resect these horns due to its sa-

fety and merits.

Keywords: Dysmenorrhoea; Rudimentary Horn; Lapa-

roscopic Excision

1. INTRODUCTION

Congenital uterine anomalies result from arrested de-

velopment of one or both of the Mullerian ducts and/or

their defective fusion. In 80% - 90% of cases, there is no

communicating channel between the two uterine cavi-

ties [1]. There are many cases reported with non-commu-

nicating rudimentary horn and the functioning endome-

trium associated with various complications such as dys-

menorrhoea, adenomyosis, ectopic pregnancy and haema-

tometra [2,3]. There are also several in literature, treated

with laparoscopic excision where the blood supp ly of the

rudimentary horn was either lateral or inferior [4]. How-

ever, to our knowledge, there are no case reports of uni-

cornuate uterus and a non-communicating rudimentary

horn with an inactive endometrium presenting with uni-

lateral dysmenorrhoea. Moreover, none so far have de-

scribed a medial arrangement of vasculature.

We report a case of an 18 year old girl who presented

with primary cyclic spasmodic dysmenorrhoea which was

relieved after laparoscopic excision of type 3 rudimen-

tary horn. The horn was non-communicating with a well

developed horn on the other side. The vessels were ar-

ranged medially and the endometrium was non-func-

tioning.

2. CASE REPORT

The nulliparous g irl was first referred to paediatrician s at

the age of 14 for left loin pain which settled in 2 days.

She presented a year later with similar pain which was

thought to be due to renal co lic. An Ultrasound scan and

an X-ray were unremarkable. Two years later she pre-

sented to gynaecologists for pelvic pain and cyclical left

sided dysmenorrhoea which did not respond to analge-

sics. Clinical examination elicited tenderness in the left

iliac fossa. Repeat ultrasounds and X-Rays were normal

and the pain settled on minipill.

She presented again to gynaecologists with cyclical left

sided dysmenorrhoea despite minipill, tricycling com-

bined pill and d epot provera. On laparoscopy, the uterine

fundus appeared broad. The right tube and ovary were

normal. The left tube had a small 2 cm sized swelling

near the isthmic end but not continuous with the fundus

R. Dwivedi et al. / Open Journal of Obstetrics and Gynecology 1 (2011) 213-216

214

(Figure1). This was confirmed with methylene blue dye

test. There was no evidence of endometriosis. A rudi-

mentary uterine horn was suspected. She subsequently

had a hysteroscopy which confirmed the fully developed

right horn with a single right sided tubal ostium. Mag-

netic resonance imaging (MRI) further confirmed this

finding and detected a 1 cm sized non-communicating

rudimentary horn with endometrial lining on the left side

(Figure 2). Renal ultrasound excluded any urinary tract

abnormality. She was offered excision of the left horn

and fallopian tube laparoscopically. This was carried out

successfully through 3 ports using tripolar scissors. The

arterial supply coursed medial to the rudimentary horn

(Figure 3). The left hemi uterus was only loosely atta-

ched to the right sided well developed horn. The left round

and utero-ovarian ligaments were diathermied and tran-

sected. After identification of the ipsilateral ureter, the

uterine vessels on the medial side of this horn (to the left

of the right horn) was then similarly cauterised and cut

(Figure 4). The patient recovered well from the opera-

tion and was discharged home within 24 hours. Histolo-

gy showed a normal fallopian tube, and the rudimentary

uterine structure measured 35 mm× 20 mm × 15 mm

with a lumen lined by inactive endometrium.

Figure 1. Laparoscopy demonstrating the rudimentary left ute-

rine horn attached to the left fallopian tube.

Figure 2. MRI Images of Noncommunicating rudimentary horn.

Figure 3. Laparoscopic excision of the left rudimentary horn

and fallopian tube demonstrating the medial arrangement of

vasculature.

Figure 4. Excised rudimentary horn.

Although her left sided cyclical dysmenorrhoea reso-

lved postoperatively, she continued to have mild cyclical

central dysmenorrhoea. She was content to manage this

with minipill. Subsequently she had successful pregnan-

cy with an emergency caesarean section for obstetr ic rea-

sons and is currently pregnant with her second child (at

the time of submission of this paper).

3. DISCUSSION

The incidence of Mullerian anomalies in the reproduc-

tive age group varies from 0.50% - 017%. The incidence

of unicornuate uter us is 2. 5% - 13.2% [ 5] . The fre quency

of rudimentary horn is extremely rare at 1:100,000. In

80% - 90% cases there is no communication with the

other horn. Rudimentary horn could be either firmly atta-

ched to the unicornuate uterus or separated by a loose

band of tissue. The American Society for Reproductive

Medicine (ASRM) [6] divides this into 4 subtypes: Fig-

ure 1 unicornuate uterus with a communicating rudi-

mentary horn; Figure 2 and 3 with a non-communicat-

ing horn, with or without cavity and in Figure 4, an iso-

R. Dwivedi et al. / Open Journal of Obstetrics and Gynecology 1 (2011) 213-216 215

lated unicornuate uterus.

Women with obstructive uterine anomalies usually pre-

sent with chronic cyclic or continuous pelvic pain. The

presentation can be delayed with increasing severity of

dysmenorrhoea, with each subsequent menstrual period

as a common feature. The symptoms may be severe e-

nough to interfere with quality of life and may not res-

pond to medication. Unlike in obstructive uterine ano-

malies where symptoms are caused by the accumulation

of blood in the rudimentary horn, disordered arrange-

ment of uterine musculature and abnormal contraction

are thought to be the cause in cases such as ours with

inactive endometrium [7]. Unequal development of Mu-

llerian system gives rise pain on one side of the pelvis.

Torsion of the rudimentary horn has also been reported.

Other complications include endometriosis and primary

infertility. Anomalies of the urinary system are usually

associated with uterine developmental abnormalities due

to their close embryological origins. Endometriosis re-

sults from retrograde menstruation from the functioning

endometrial cavity through the ipsilateral fallopian tube.

Obstetric complications such as rupture uterus, recurrent

miscarriages, premature labour and malpresentation can

also occur [8].

A high index of suspicion for Mullerian duct abnor-

mality is therefore needed in teenagers with dysmenor-

rhoea to prevent delays in diagnosis [9]. Ultrasonogra-

phy including hysterosonography is not specific in non-

functioning uterine horns as the detail of adnexa is in-

sufficient. In a recent review of the literature, the sensi-

tivity of ultrasonography even in cases with functioning

horns was only 26%. MRI is much more accurate and

detected all cases in these series and is considered to be

the gold standard [10]. 3D CT scanning has been used in

some cases. 3D ultrasound also may also have a future

role. An early recourse to laparoscopy combined with

hysteroscopy is required to confirm the diagnosis most

cases.

Resection not only offers symptomatic relief in cases

unresponsive to medication, it also prevents the possibi-

lity of pregnancy in the rudimentary horn due to trans-

peritoneal migration of sperm or fertilised ovum. Accu-

rate diagnosis of the anomaly is required prior to exci-

sion to decide on the precise surgical approach as in so-

me cases the two horns are firmly attached. This requires

difficult dissection to develop a plane between the hemi

uteri. If the other tube is normal, the ipsilateral fallopian

tube should be removed to prevent ectopic pregnancy. If

the other tube is damaged, microsurgical transposition of

the ipsilateral tube may need to be considered [11]. Al-

though laparotomy was used traditionally, there are seve-

ral publications of the laparoscopic removal of the rudi-

mentary horn [10]. This follows a similar trend in other

types of gynaecological surgery. Most cases are amena-

ble to laparoscopic management and this is preferred to

prevent adhesions and damage to the myometrium of the

well developed horn. Rudimentary horn attached to the

unicornuate uterus by a band of tissue, as in our case, is

relatively easy to dissect. Minimally invasive, with well

established safety profile, laparoscopic approach has other

advantages such as shorter hospital stay and quicker re-

turn to activities [12]. Aberrant blood vessels and uro-

logical anomalies increase the risk of bleeding and ina-

dvertent injury. Review of MRI scans with radiologists

prior to surgery allows selected cases to be referred to

advanced laparoscopic surgeons.

REFERENCES

[1] Dadhwal, V., Mittal, S., Kumar, S. and Barua, A. (2000)

Hematometra in postmenarchal adolescent girls: A report

of two cases. Gynecologic and Obstetric Investigation,

50, 67-69. doi:10.1159/000010284

[2] Chakravarthi, S. and Chin, K. (2003) Rudimentary uter-

ine horn: Management of a diagnostic enigma. Acta Ob-

stetricia et Gynecologica Scandinavica, 82, 1153-1154.

doi:10.1046/j.1600-0412.2003.00234.x

[3] Goel, P., Aggarwal, A., Devi, K., Takkar, N., Saha, P.K.

and Huria, A. (2005) Unicornuate with non-communi-

cating rudimentary horn different clinical presentations.

The Journal of Obstetrics & Ganecology of India, 55,

155-158.

[4] Falcone, T., Gidwani, G., Paraiso, M., Beverly, C. and

Goldberg, J. (1997) Anatomical variation in rudimentary

horns of a unicornuate uterus: Implications for laparo-

scopic surgery. Human Reproduction, 12, 263-265.

doi:10.1093/humrep/12.2.263

[5] Green, L.K. and Harris, R.E. (1976) Uterine anomalies:

Frequency of diagnosis and associated obstetric compli-

cations. Obstetrics Gynecology, 47, 427-429.

[6] Buttram, V.C. Jr. and Gibbons, W.E. (1979) Mullerian

anomalies: A proposed classification (an analysis of 144

cases). Fertility and Sterility, 32, 40-46.

[7] Jeffcoate, N. (2006) Malformation and maldevelopments

of the genital tract: In principles of gynaecology. The 4th

Edition, Butterworth & Co Ltd, London, 138.

[8] Atmaca, R., Germen, A.T., Burak, F. and Kafkasli, A.

(2005) Acute abdomen in a case with noncommunicating

rudimentary horn and unicornuate uterus. Journal of the

Society of Laparoendoscpic Surgeons, 9, 235-237.

[9] Badawy, S.Z.A. and Montgomery, S. (2009) Severe pri-

mary dysmenorrhoea due to functioning noncommunica-

timg rudimentary horn. Journal of Gynecologic Surgery,

25, 23-27. doi:10.1089/gyn.2009.B-02330

[10] Spitzer, R.F., Kives, S. and Allen, L.M. (2009) Case se-

ries of laparoscopically resected noncommunicating

functional uterine horns. Journal of Pediatric and Ado-

lescent Gynecology, 22, E23-E28.

doi:10.1016/j.jpag.2007.12.001

[11] Gold Berg, J.M. and Friedman, C.I. (1988) Microsurgical

fallopian tube transposition with subsequent term preg-

nancy. Fertility and Sterility, 50, 661-662.

R. Dwivedi et al. / Open Journal of Obstetrics and Gynecology 1 (2011) 213-216

216

[12] Liatsikos, S.A., Tsikouras, P., Souftas, V., Ammari, A.,

Prassopoulos, P., Maroulis, G. and Liberis, V. (2010) Di-

agnosis and laparoscopic management of a rudimentary

uterine horn in a teenage girl, presenting with hema-

tometra and severe endometriosis: Our experience and

review of literature. Minimally Invasive Therapy and Al-

lied Technologies, 19, 241-247.

doi:10.3109/13645701003644491