J o u r n a l o f C a s e s i nObs te trics & G ynecology

J Cases Obstet Gynecol, 2016;3(3):80-82

Case Report

A delayed therapy of dysmenorrhea due to noncommunicating rudimentary horn and unicornuate uterus: Case report and review of the literature

Ali Sami Gurbuz1,*, Necati Ozcimen2, Emel Ebru Ozcimen3, Ahmet Salvarcı4, Serdar Altunay5

1Novafertil IVF Center, Obstetrics and Gynecology Department2Medicana IVF Center, Obstetrics and Gynecology Department3Baskent University Hospital, Perinatology Department4Novafertil IVF Center, Urology Department5Selcuk University, Pathology Department

AbstractAlthough asymptomatic cases naturally occur, congenital mullerian abnormalities can present with infertility, menstrual irregulari-ty and recurrent pregnancy loss. Women with normal reproductive outcomes and recurrent pregnancy loss exhibit approximately 2-4% and 5-10% incidence of various congenital mullerian abnormalities. Following septate uterus, bicornuate uterus and arcuate uterus, unicor-nuat uterus is the fourth most frequent congenital uterine abnormality seen in a combined population of infertile and fertile women. Uni-cornuate uterus with a non-communicating functional rudimentary horn is a type of mullerian anomaly and is a rare cause of dysmen-orrhea. Here, we presented a case of this kind of abnormality and discussed the management strategies in the light of the literature.

Key Words:Dysmenorrhea, mullerian anomaly, unicornuate uterus, rudimentary horn

Introduction

Article History:Received: 08/02/2016Accepted: 09/05/2016

*Correspondence: Ali Sami GurbuzAddress: Novafertil IVF Center, Konya TurkeyE-mail: alisamigurbuz@hotmail.comPhone: +90 533 5530442

Journal of Cases in Obstetrics & Gynecology80

Dysmenorrhea is defined as difficult menstrual flow or painful menstruation. Dysmenorrhea is a common com-plaint of women especially adolescent girls are affected. The prevelance of dysmenorrhea is about 25% which its possible cause is the spasmic contraction of uterus [1]. It can be divided in to 2 broad categories; primary (occuring in the absence of pelvic pathology) and secondary (resulting from organic diseases). Pharmacotherapy especially non- steroidal anti inflammatory drugs (NSAID) are the most effective treatment choices in primary dysmenorrhea [2].There are some organic causes of secondary dysmenorrhea. One of them is the congenital mullerian malformation of the uterus as bicornuate uterus or subseptate uterus [3]. Abnor-

mal fusion of the mullerian ducts results from anatomical changes in the female genital system. Unicornuate uterus with a rudimentary horn is a rare type of mullerian duct mal-formation. The frequency of uterine congenital anomalies is 1/200 to 1/600 and 1/100000 for rudimentary horn [4].A non-communicating rudimentary horn usually has inac-tive endometrium but can be associated with complications from menarche to menopause such as endometriosis, pri-mary infertility, hematometra, anomalies of the urinary sys-tem and obstetrical problems such as malpresentation, ha-bitual abortus, ectopic pregnancies and premature birth [5].A middle aged women who has severe and intractible dys-menorrhea with noncommunicating rudimentary horn is reported here and discussed in the light of the literature.

Case Presentation

A 37-year-old woman with gravida:2 who had 2 vag-inal deliveries attended to gynecology department of our hospital with complaints of dysmenorrhea, severe cy-clic pelvic pain from menarche. The pelvic pain was

J o u r n a l o f C a s e s i nObs te trics & G ynecology

81www.jcasesobstetgynecol.com July 2016

typically felt over the right lower abdomen and used to start before the menstrual cycle and lasted during men-strual cycle. She used various analgesics for long years.On examination, no abnormality was detected on system-ic examination. Transvaginal ultrasonography showed us unicornuate uterus with non-communicating rudimentary horn. In the cavity of rudimentary horn, active endometri-um echogenicity was observed. To confirm the diagnosis, magnetic resonance imaging (MRI) was performed. Intra-venous pyelography also confirmed right renal agenesis.

Laparoscopy was planned but because of the adhesions between intestines and rudimentary horn, laparotomy was performed for not to cause intestinal injury. No endome-triosis was observed in the abdomen. On laparotomy, the right –sided rudimentary horn was excised. Cystoscopy was performed by urology doctor which revealed non-vi-sualization of the right ureteric orifice with normal left ure-teric orifice and bladder mucosa. The postoperative period was uneventful and the patient was discharged on the 2nd day of operation. Histopathologic evaluation of the spec-imen revealed a rudimentary horn with active endometri-al and myometrial tissue (Figure 3). The dysmenorrhea complaint was not observed during the follow-up period.

Discussion

Unicornuate uteus with non-communicating rudimentary horn is rare form of mullerian abnormalities which is also an important cause of dysmenorrhea. It causes cyclic pain due to intracavitary retentation of menstrual effluent and retro-grade menstrual flow. If there was no relationship between ru-dimentary horn and main functional endometrial cavity, he-matometra and maybe hematosalpinx would be observed [6].Other mechanism of dysmenorrhea in unicornuate uter-us with rudimentary horn is the endometriosis. These pa-tients usually attend to gynecology or emergency depart-ments with severe pain or pelvic mass [7]. Agarwal et al. reported a 18-year-old woman who developed endome-triosis and had severe pelvic pain. Generally it is consid-ered to become the adolescent girl’s pathology but here we presented a 37-year-old woman with this abnormality with two vaginal deliveries. Borah et al. presented again an adolescent girl with primary dysmenorrhea with similar mullerian abnormality [8]. The management of these two cases were excision of the rudimentary horn. The wom-en with primary dysmenorrhea are not evaluated in detail during routine examination [2,8]. But a good transvaginal ultrasonography is enough for diagnosis but pelvic mag-netic resonance imaging (MRI) is good for confirmation of the diagnosis. This mullerian abnormality may be related

Figure 1.

Transvaginal ultrasonographic view of the rudi-mentary horn containing endometrial stripe

Figure 2.

Hysterosalpingography of the patient which demonstrates unicornuat uterus

with infertility, recurrent pregnancy loss, preterm deliv-ery and ectopic pregnancy. There may be coexisting uri-nary tract abnormalities like unilateral renal agenesis [8,9]. In this case drug resistant primary dysmenorrhea and uni-lateral renal agenesis were observed. Two term uneventful pregnancies were delivered vaginally. In the literature the laparoscopic excision of the rudimentary horn is offered for possible severe complications [3,10]. But as in our case se-vere adeshions can make laparoscopy difficult. If a woman complains about primary dysmenorrhea from menarche and presents with unilateral pelvic mass a mullerian anomaly should be remembered and shoud be evaluated carefully.

AcknowledgementNone

Declaration of InterestNone

Journal of Cases in Obstetrics & Gynecology82

Gurbuz et al.

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Figure 3.

Histopathological evaluation of the rudimentary horn specimen