■ LETTER TO THE EDITOR ■ ACUTE ABDOMEN CAUSED BY HEMATOMETRA AND HEMATOSALPINX FOUR MONTHS FOLLOWING RIGHT OOPHORECTOMY IN A TEENAGER WITH RIGHT PERITUBAL ADHESIONS, UNICORNUATE UTERUS AND A RIGHT NONCOMMUNICATING RUDIMENTARY HORN Chih-Yu Lin1, Chih-Ping Chen1,2, Hung-Hung Lin1, Shu-Chin Chien3, Chin-Yuan Hsu1* 1Department of Obstetrics & Gynecology, Mackay Memorial Hospital, Taipei, 2Department of Biotechnology and Bioinformatics, Asia University, and 3Department of Medical Genetics and Obstetrics & Gynecology, China Medical University Hospital, Taichung, Taiwan. Unicornuate uterus with a rudimentary horn is a rare and voluntary guarding of the right lower quadrant. uterine malformation resulting from the arrest of one of Her leukocyte count was 11,000/cm3, the urinary preg- the müllerian ducts during embryologic development nancy test was negative, and plain abdominal X-ray [1]. The incidence of this congenital uterine anomaly showed moderate bowel air accumulation in the right in the female population is estimated to be about lower quadrant. According to her statement, she denied 0.00001% [2]. An unicornuate uterus with a noncom- any systemic disease since childhood. However, she municating horn and functional endometrium constitute underwent a laparoscopic right oophorectomy at a a distinct subgroup with clinical repercussion, because local clinic 4 months previously because of an ovarian it can cause hematometra and endometriosis leading tumor, but she was not informed of any genital tract to progressive dysmenorrhea, lower abdominal pain, anomalies at that time. She had been suffering from infertility, and obstetrical complications, such as rupture dysmenorrhea since her menarche at the age of 13, but of pregnant rudimentary horn and ectopic pregnancy her symptom worsened after the surgical procedure. [1,3,4]. Unfortunately, this type of uterine anomaly is The appearance of her external genitalia was unremark- usually unrecognized until these complications develop. able, with normal pubic hair and an intact hymen. Here, we report a case of a unicornuate uterus with A transabdominal ultrasound scan suggested a bicor- noncommunicating functional horn complicated with nuate uterus with a homogeneous hypoechoic sand- hematometra and hematosalpinx, in which severe dys- like lesion at the right side of the cavity, consistent with menorrhea and an acute abdominal pain developed after hematometra (Figure 1). A computed tomography scan an oophorectomy. An iatrogenic etiology is discussed. revealed an absent right kidney, a right adnexal cyst, and A 17-year-old nulligravida woman presented to our a bicornuate uterus with a right hematometra (Figure emergency department with severe right lower abdom- 2A). Pelvic examination under anesthesia showed a inal pain. Intermittent right lower abdominal pain single cervix deviated to the left side without vaginal was noted for about 3 weeks and became persistent. anomaly. Uterine sounding showed no connection Progressive dysmenorrhea was also noted for about between cervix and the right adnexal mass. The patient 4 months. Physical examination revealed diffuse ten- underwent laparotomy that demonstrated a left uni- derness of the lower abdomen with rebounding pain cornuate uterus with cervix and a noncommunicating right rudimentary horn attached to the unicornuate uterus at the base, dilated by a large hematometra *Correspondence to: Dr Chin-Yuan Hsu, Department of Obstetrics with chocolate-like content (Figures 2B, 2C and 3). and Gynecology, Mackay Memorial Hospital, 92, Chung-Shan Right hematosalpinx and multiple endometriotic North Road, Section 2, Taipei 104, Taiwan E-mail: [email protected] implants were found in the peritoneum of the Douglas Accepted: July 9, 2007 pouch. Removal of the rudimentary horn, ipsilateral 456 Taiwan J Obstet Gynecol • December 2007 • Vol 46 • No 4 Acute Abdomen and Unicornuate Uterus with Rudimentary Horn salpingectomy, and electrocauterization of endometri- endometrium, hematometra and hematosalpinx. Six osis were performed. The postoperative course was months after the surgery, the patient continued to have uneventful, and the patient was discharged on the third regular menstrual cycles without dysmenorrhea. postoperative day. Histology was consistent with a non- Patients with a noncommunicating uterine horn and communicating rudimentary horn with a functional a functional endometrium are at risk for developing several complications, such as progressive dysmenor- rhea, acute and chronic pelvic pain, infertility, tubal ectopic pregnancy, and rupture of the pregnant rudi- mentary horn [1,3,5,6]. If this uterine anomaly is unrecognized or misdiagnosed during a previous surgi- cal procedure, an obstruction of the retrograde out- flow of the rudimentary horn may lead to surgical complications. To date, at least six cases of unicornuate uterus with noncommunicating functional horn complicated with dysmenorrhea and abdominal pain following pelvic operations, such as tubal sterilization (two cases), cesarean section (two cases) and unilateral adnexec- tomy (two cases), have been reported (Table) [7–11]. The reported intervals between the first surgical proce- Figure 1. Transabdominal ultrasound shows a 4.3 × 3.0 cm dure and acute symptoms requiring immediate surgery homogenous content inside the rudimentary horn that is varied from 5 months to 8 years. The Table shows that consistent with hematometra (H). three of the six cases had unilateral renal agenesis and A C RT H LT H U *** * U LO * *** * * ** * *** * ** * B LT Figure 2. (A) Computed tomography scan reveals two uter- H U ine cavities with fluid accumulation in the right side. (B) Gross image of the right rudimentary horn and left unicor- nuate uterus during laparotomy. (C) An illustration of the uterine anomaly with right hematosalpinx (arrowhead), right peritubal adhesions (arrow) and multiple endometriosis foci over the peritoneal surface (*). H = hematometra; LO = right ovary; LT = left tube; RT = right tube; U = unicornuate uterus. Taiwan J Obstet Gynecol • December 2007 • Vol 46 • No 4 457 C.Y. Lin,etal C.Y. phy, computerized tomography resonancemagnetic and phy, ultrasonography,intravenousas such studies, pyelogra- pleteexaminationpelvic complementaryand imaging includingdetailedtive work-ups history taking, com- Jayasinghe etal[6]suggested thatthorough preopera- surgicalcompletesmall a or work-up field [7,10]. because oftheinexperience ofthesurgeons, thelackof noncommunicating functionalhorncanbedifficult diagnosisofaunicornuate uterus witha However, future complicationsandtorelieve symptoms[5,6,9]. lateral tubeisrecommended upondiagnosistoavoid retrograde menstruation. the rightadnexa andthecomplete obstructiontothe factors suchassevere postoperative adhesionsaround in ourcasewaslikelybecauseofmultipleiatrogenic ment ofhematometra andtheprevious oophorectomy The short interval of4monthsbetweenthedevelop- men developed4monthsafterinitial operation. the metriosis, andsevere dysmenorrhea andanacute abdo- hematosalpinx, rightrenal agenesisandpelvicendo- a rightrudimentary hornwithhematometra, right Inthepresent case, there were menstruation [1,3,5]. ciated withendometriosisasaresult oftheretrograde functional nicating in more than halfofthecases[6,12]. agenesis willpredict anipsilateral mülleriananomaly renal aberrations, andthepresence ofunilateral renal Müllerian ductanomaliesare usuallyassociated with endometrial activitywithintherudimentary horn[6]. be dependentonthedegree ofobstructionthe mentary ofintervals horn.Variation hasbeennoted to All caseshadagoodoutcomeafter removal oftherudi- all, except one,were associated withendometriosis. (H) andrighttubewithhematosalpinx(RT). Figure 3. 458 Early removal oftherudimentary hornanditsipsi- RT Gross oftherightrudimentary appearance horn horns havebeennoted tobeasso- H Noncommu- Table. Reported cases with unicornuate uterus, noncommunicating functional horn, dysmenorrhea, and abdominal pain associated with previous surgical procedures Outcome following Müllerian duct Associated Unilateral renal Author Age (yr) Previous surgery Interval Symptom removal of Taiwan JObstetGynecol Taiwan abnormalities endometriosis agenesis rudimentary horn Robischon et al [7] 27 Unicornuate uterus + right Tubal sterilization 5 mo Dysmenorrhea No No Good noncommunicating horn Chang et al [8] 30 Unicornuate uterus + right Cesarean section 8 mo Dysmenorrhea and progressive Yes Yes Good noncommunicating horn abdominal pain • December 2007 Kriplani et al [9] 24 Unicornuate uterus + right Cesarean section 2 yr Dysmenorrhea and progressive Yes No Good noncommunicating horn abdominal pain Lee et al [10] 39 Unicornuate uterus + left Tubal sterilization 6 yr Dysmenorrhea Yes No Good noncommunicating horn • + Vol 46 Vol Tanaka et al [11] 32 Unicornuate uterus left Left adnexectomy 3 yr Dysmenorrhea Yes Yes Good noncommunicating horn • + No 4 Present case 17 Unicornuate uterus right Right oophorectomy 4 mo Dysmenorrhea and progressive Yes Yes Good noncommunicating horn abdominal pain Acute Abdomen and Unicornuate Uterus with Rudimentary Horn imaging, are mandatory. In doubtful cases, laparoscopy 6. Jayasinghe Y, Rane A, Stalewski H, Grover S. The presentation in combination with hysteroscopy, a tubal dye study, and early diagnosis of the rudimentary uterine horn. Obstet and an intraoperative ultrasonography are useful Gynecol 2005;105:1456–67. 7. Robischon K, Baram D, Phipps W. Presentation of a müller- [7,11,13]. ian anomaly with outflow obstruction after tubal ligation. In