A Delayed Therapy of Dysmenorrhea Due to Noncommunicating Rudimentary Horn and Unicornuate Uterus: Case Report and Review of the Literature
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J Cases Obstet Gynecol, 2016;3(3):80-82 J o u r n a l o f C a s e s i n Obs tetrics & G ynecology 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 Department 2Medicana IVF Center, Obstetrics and Gynecology Department 3Baskent University Hospital, Perinatology Department 4Novafertil IVF Center, Urology Department 5Selcuk University, Pathology Department Abstract Although 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 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- ysmenorrhea is defined as difficult menstrual flow or D formation. The frequency of uterine congenital anomalies painful menstruation. Dysmenorrhea is a common com- is 1/200 to 1/600 and 1/100000 for rudimentary horn [4]. plaint of women especially adolescent girls are affected. A non-communicating rudimentary horn usually has inac- The prevelance of dysmenorrhea is about 25% which its tive endometrium but can be associated with complications possible cause is the spasmic contraction of uterus [1]. It from menarche to menopause such as endometriosis, pri- can be divided in to 2 broad categories; primary (occuring mary infertility, hematometra, anomalies of the urinary sys- in the absence of pelvic pathology) and secondary (resulting tem and obstetrical problems such as malpresentation, ha- from organic diseases). Pharmacotherapy especially non- bitual abortus, ectopic pregnancies and premature birth [5]. steroidal anti inflammatory drugs (NSAID) are the most A middle aged women who has severe and intractible dys- effective treatment choices in primary dysmenorrhea [2]. menorrhea with noncommunicating rudimentary horn is There are some organic causes of secondary dysmenorrhea. reported here and discussed in the light of the literature. One of them is the congenital mullerian malformation of the uterus as bicornuate uterus or subseptate uterus [3]. Abnor- Case Presentation Article History: Received: 08/02/2016 Accepted: 09/05/2016 A 37-year-old woman with gravida:2 who had 2 vag- inal deliveries attended to gynecology department of our *Correspondence: Ali Sami Gurbuz Address: Novafertil IVF Center, Konya Turkey hospital with complaints of dysmenorrhea, severe cy- E-mail: [email protected] clic pelvic pain from menarche. The pelvic pain was Phone: +90 533 5530442 80 Journal of Cases in Obstetrics & Gynecology J o u r n a l o f C a s e s i n Obs tetrics & G ynecology typically felt over the right lower abdomen and used to start before the menstrual cycle and lasted during men- Figure 2. 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. Figure 1. Hysterosalpingography of the patient which demonstrates unicornuat uterus 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- Transvaginal ultrasonographic view of the rudi- dimentary horn and main functional endometrial cavity, he- mentary horn containing endometrial stripe matometra and maybe hematosalpinx would be observed [6]. Other mechanism of dysmenorrhea in unicornuate uter- Laparoscopy was planned but because of the adhesions us with rudimentary horn is the endometriosis. These pa- between intestines and rudimentary horn, laparotomy was tients usually attend to gynecology or emergency depart- performed for not to cause intestinal injury. No endome- ments with severe pain or pelvic mass [7]. Agarwal et al. triosis was observed in the abdomen. On laparotomy, the reported a 18-year-old woman who developed endome- right –sided rudimentary horn was excised. Cystoscopy triosis and had severe pelvic pain. Generally it is consid- was performed by urology doctor which revealed non-vi- ered to become the adolescent girl’s pathology but here sualization of the right ureteric orifice with normal left ure- we presented a 37-year-old woman with this abnormality teric orifice and bladder mucosa. The postoperative period with two vaginal deliveries. Borah et al. presented again was uneventful and the patient was discharged on the 2nd an adolescent girl with primary dysmenorrhea with similar day of operation. Histopathologic evaluation of the spec- mullerian abnormality [8]. The management of these two imen revealed a rudimentary horn with active endometri- cases were excision of the rudimentary horn. The wom- al and myometrial tissue (Figure 3). The dysmenorrhea en with primary dysmenorrhea are not evaluated in detail complaint was not observed during the follow-up period. 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 81 www.jcasesobstetgynecol.com July 2016 Gurbuz et al. with infertility, recurrent pregnancy loss, preterm deliv- ery and ectopic pregnancy. There may be coexisting uri- Figure 3. 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. 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