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(2):45-49

Case Report

Bilateral retrograde ureteric passage of contrast medium during hysterosalpingography: A case report

Dinçer Sümer1,*, Muhammed Emin Öz2, Gülizar Özer2, Ali Yanik2

1Bitlis State Hospital, Bitlis, Turkey2Cumhuriyet University Faculty Of Medicine Department of Obstetrics and Gynecology, Sivas, Turkey

Abstract

A 30-year-old patient, has not been conceived for fifteen years, was evaluating for infertility via hysterosalpingography. In the course of exam-ination after inserting the canulla to the external cervical ostium, medium performed gently through cervical canal. Cervical canal, uter-ine cavity and bilaterally fallopian tubes distended by medium, respectively. Contrast medium poured onto peritoneum and as soon as pour-ing, suddenly, ascended bilaterally and synchoronously, enlarged and branched at the level of second lumbal vertebral silhuette which is indicating the kidneys. Bilateral linear, thin and sharp contoured images were ureters. We reported this unusual case whom intravenous-py-elogram, magnetic resonance imaging and intraoperative findings were inefficient for explaining of this retrograde passage of medium.

Key Words:

Retrograde, ureter, hysterosalpingography

Introduction

Article History:Received: 03/04/2015Accepted: 21/04/2015

*Correspondence: Dr. Dincer SumerAddress: Bitlis State Hospital, Bitlis, TurkeyTel: 05073432971E-mail: drdincersumer@gmail.com

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Hysterosalpingography (HSG) is the most commonly used technique for evaluating infertility and used to assess the anatomy of the uterus and the patency of the fallopian tubes [1]. Occasionally, during performing HSG contrast medium transudes into the venous and lymphatic canals and thereby to the systemic circulation and known as in-travasation. The presence of contrast within the bladder and ureters after a time later, indicates the clearance of me-dium from the vascular circulation [2]. Here we report an extraordinary passase of contrast into ureters and thereby kidneys, a retrograde cruise, in an infertile patient and has not associated with intravasation or vascular clearence.

Case Presentation

A 30-year-old, gravida 0, para 0 patient admitted to clinic for infertility for fifteen years. There was only tiroidectomy operation, and any other operation or instrumentation was not notable at her medical history. Uterus was found en-larged at pelvic examination. Transvaginal ultrasonography showed 60x55 mm sized hypoechogenity suggesting an intramural leiomyoma arising from posterolateral portio of uterus and normally ovaries with 6-7 antral folicules. There was not ascites or fluid at posterior cul de sac. She had reg-ular menstruation periods and serum follicle stimulating hormone (FSH), luteinising hormone (LH) and estradiol (E2) levels were with normal range at third day of cycle. A cervical Papanicolaou test taken and revealed normally. HSG was planned at early proliferative period of menstrual cycle. In the course of procedure at dorsal lithotomy po-sition, after inserting sterile speculum, cervix hold by one

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teeth tenaculum. The canulla inserted to the external cervi-cal ostium then contrast medium performed gently through the cervical canal. Hydrosoluble iodized contrast medium (Omnipaque; Nycomed, Amersham, UK) 20 mL was slow-ly administered with fluoroscopic guidance. Cervical canal and uterine cavity distended respectively at the scopy. Mini-mal filling defect at fundus was suggesting subseptus. Irreg-ular, reticular, round like contour appeared at right postero-lateral side of uterus, due to intravasation, pointed out the leiomyoma demonstrated by ultrasonography before simul-taneously passage of medium through the fallopian tubes bilaterally, slowly at left side, and pouring into the peritone-um. As soon as pouring, the hypoechoic image of contrast medium, ascended as a thin line originating from the lateral borders of fallopian tubes bilaterally, curved to the medial and became closer to the lumbal vertebras symmetrically, than enlarged and branched synchronously and terminat-ed at the level of first-second lumbal vertebra silhuette in few seconds. The hypoechogenity of contrast medium was distending cervical canal, uterus, fallopian tubes and was pointing out the leiomyoma, peritoneum and the unusu-al bilateral ascending image at the sametime (Figure 1).

Patient turned side to right decubitis position and sco-py studied again. The echogenity was beginning from posterior cul de sac, curving and ascending at anteri-or of uterus in the pelvis minor, and became closer to the silhuette of vertebras on pelvis major (Figure 2).

Total duration up to this moment starting from per-forming the medium though the cervical canal was approximately ten seconds. Half an hour lat-er the hypoechogenity of medium demonstrated at the distending bladder compatable with vasculer clearence (Figure 3). The procedure was terminated and further in-vestigation planned electively. Intravenous-pyelogram and voiding cystography performed for evaluating the urinary system and graphies showed kidneys at their well-known location, ureters and bladder, without duplication or leak-age of medium (Figure 4). Reflux was not determined at

Figure 1.

Contrast medium presenting at uterine cavity, fallo-pian tubes and bilateral ureters at the same time

Figure 2.

Right lateral decubitis graphy showing contrast medium ascending at posterolateral abdominal wall

voiding cystography. There was only 6x6x7 cm heteroge-nous hypointence mass as pathologic finding at right pos-terolateral side of uterus at Magnetic Resonance Imaging. Endometrial sampling revealed proliferative endometrium.

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Diagnostic hysteroscopy and myomectomy via laparato-my planned for assessment of endometrial cavity and sus-picious filling defect of subseptus and enucluation of the posterolateral located leiomyoma causing enlargement and distortion of uterus and for restoration of the uterine anat-omy but patient was lost for follow- up. Approximately two years later, patient preparated for operation. During hysteroscopy cervical canal and left tubal ostium seemed normally, right tubal ostium was deplaced and endometrial cavity was distortioned by external compression. Intramu-ral leiomyoma, 8x8cm sized at posterolateral side of uter-us, found at laparotomy. Fallopian tubes were adherent to posterior cul de sac but ovaries were normally. There was not any visible endometriotic implants or ascitic fluid.

Figure 3.

Distending bladder 30 minutes after performing medium competible with retardation of vascular clearence mecha-nism

Figure 4.

Normally Intravenous-pyelogram of patient

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Methilene blue was applied from cervix, for chromo-per-tubation, but it was inefficacious. Although myomectomy, adhesiolysis and distal salpingoplasty established, minimal dye hardly passed through fallopian tubes. Biopsy has been taken from peritoneum for pathological examination. A drain with negative pressure placed at posterior cul de sac and op-eration was terminated. Postoperative at third hour the color of urine was bluish-green. There was 130 mililiter serous liquid in drain at morning after. Creatinine level of incoming analised and it was similar with blood serum. Any medical or surgical complication has occured and patient discharged at postoperative third day. Final pathologic examination revealed endosalpingiosis and degenerating leiomyoma. Discussion

HSG is the only radiologic procedure routinely performed in the initial evaluation of the infertile woman and used to assess the anatomy of the uterus and the patency of the fal-lopian tubes [1]. The venous and lymphatic intravasation in uterine and adnexal vessels is acomplicated disorder which occurs due to progressive destruction and ulceration of en-dometrium. The most important cause of intravasation is the transudation of contrast medium into the venous and lymphatic canals through unprotected vessels. The pres-ence of contrast within the bladder and ureters indicates the clearance of contrast from the vascular circulation so it get times after the process [2]. In this unusual case contrast medium distended and pointed out cervical canal, uterine cavity, leiomyoma, fallopian tubes respectively. Meanwhile the medium poured into the posterior cul de sac and as-cended, branched and terminated at the level of kidneys. The medium was presented at uterine cavity, leiomyoma, fallopian tubes, posterior cul de sac, ascending thin line ‘ureters’ and kidneys at the sametime. The duration was only few seconds up to this moment. The retrograde cruise of contrast, which is not compatable with pelvic veins, and occurance in a few seconds rules out the vascular clearence mechanism and confirms the passage of medium into the ureters directly from peritoneum. The hypoechogenity of medium was seen as distending the bladder half an hour later which was compatable with retardation of the vas-

cular clearence mechanism. This unusual periton-ureteral passage may be a cause of infertility via discomposing the peritoneal habitus and may misdiagnosed as intravasation. Patient was given doksisiklin medication for infection profilaxis due to intravasation via leiomyoma [3]. Al-though embolism due to oil soluble contrast medium has been reported there was not any reports about em-bolism due to water soluble media so anticoagulant use for such indication is not clear [4]. Patient was not given anticoagulant medication with subsequent data.Deep infiltrating endometriosis (DIE) usu-ally involves the uterosacral ligaments, therectovaginal space, the upper third of the posterior vaginal wall, the bowel, and the urinary tract. Chapron et al. report-ed ureteric DIE at 2.1% of their 426 patients suffering from pelvic pain [5]. There was not narrowing segments at ureter-ic image at intravenous-pyelogram and visible endometriot-ic implants on peritoneum at laparatomy. Also peritoneal biopsies did not revealed histological proved endometriosis.Endosalpingiosis refers to the presence of ectopic tub-al-type ciliated glandular epithelium which resembles the normal endosalpinx [6]. Lesions of endosalpingiosis have been found to involve pelvic parietal and visceral peritone-um, omentum , pelvic lymph nodes, the appendix, intestine, uterine corpus skin and urinary bladder [7,8]. Li et al. re-ported ureteric involvement of endosalpingiosis [9]. Clem-ent et al. reported transmural involvement of the uterine cervix and lower uterine segment and contiguous corpus in the two cases with uterine involvement [10]. The capability of transmural involvement at endosalpingiosis lesions sug-gests potential role at passage of medium but unilateral pas-sage, confirmed by patency of ureters at IVP and analysis of incoming of abdominal drain, refutates this suggestion. Lymphatic stomatas are small openings of lymphatic cap-illaries on the free surface of the mesothelium. The perito-neal cavity is connected with lymphatic system via these small openings, which have the function of active absorp-tion. The ultrastructure of the lymphatic stomata and their absorption from the body cavities are important clinically, such as ascites elimination, neoplasm metastasis, and in-flammatory reaction [11]. Lymphatic stomata may be as-sociated with peritoneal absorption but it is insufficient for explanation of passage of medium into the ureters.

References

1. Stovall DW. The role of hysterosalpin-gography in the evaluation of infertil-ity. Am Fam Physician. 1997;55:621-282. Ahmadi F, Zafarani F, Shahrzad GS. Hystero-salpingographic Appearances of Female Genital Tract Tuberculosis: Part II: Uterus. Int J Fertil Ster-il. 2014 Apr;8(1):13-20. Epub 2014 Mar 9. Review3. Lindequist S, Justesen P, Larsen C, Rasmus-sen F. Diagnostic quality and complications of hysterosalpingography: oil- versus water-sol-uble contrast media--a randomized prospec-tive study. Radiology. 1991 Apr;179(1):69-74.4. Uzun O, Findik S, Danaci M, Katar D, Erkan L. Pulmonary and cerebral oil embolism after hysterosalpingography with oil soluble con-

trast medium. Respirology. 2004 Mar;9(1):134-65. Chapron C, Chopin N, Borghese B, Foulot H, Dousset B, Vacher-Lavenu MC,et al. Deeply infiltrating endo-metriosis: pathogenetic implications of the anatomi-cal distribution. Hum Reprod. 2006 Jul;21(7):1839-456. Heinig J, Gottschalk I, Cirkel U, Diallo R. Endosalpingio-sis—an underestimated cause of chronic pelvic pain or an accidental finding? A retrospective study of 16 cas-es. Eur J Obstet Gynaecol Reprod Biol 2002;103:75–8.).7. Hesseling M, de Wilde R. Endosalpingiosis in lapa-roscopy. J Am Assoc Gynaecol Laparosc 2000;7:215–8.8. Maniar KP, Kalir TL, Palese MA, Unger PD. En-dosalpingiosis of the urinary bladder: a case of probable implantative origin with characteri-zation of benign Fallopian tube immunohisto-

chemistry. Int J Surg Pathol. 2010 Oct;18(5):381-39. Li WM, Yang SF, Lin HC, Juan HC, Wu WJ, Huang CH,et al. Müllerianosis of ureter: a rare cause of hy-dronephrosis. Urology. 2007 Jun;69(6):1208.e9-1110. Clement PB, Young RH. Florid cystic en-dosalpingiosis with tumor-like manifestations: a report of four cases including the first report-ed cases of transmural endosalpingiosis of the uterus. Am J Surg Pathol. 1999 Feb;23(2):166-7511. Wang ZB, Li M, Li JC. Recent advanc-es in the research of lymphatic stomata. Anat Rec (Hoboken). 2010 May;293(5):754-61

The location, origin and cruise of contrast medium sug-gests the ureteric association, enlarging and branching at terminal, like renal pelvis supported this suggestion. There was not abnormality at imaging studies about uri-nary system and any pathologic findings except leiomy-oma at laparatomy. Creatinine level of drain incoming rules out fistulas. As a result we suggest the contrast me-dium passed through peritoneum into ureters and there-by kidneys via retrograde cruise but we couldn’t ex-plain the occurance of this situation with our knowledge.

AcknowledgementNone

Conflict of Interest StatementThe authors declare no conflict of interest

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