Gynecologic Oncology Reports 2 (2012) 121–123

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Gynecologic Oncology Reports

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Case Report

Adjuvant intravaginal brachytherapy for uterus didelphys with synchronousendometrial adenocarcinomas and unfavourable vaginal topography

Lucinda Morris ⁎, Mark John Stevens, Susan Valmadre, Judith Martland, Tony LeeDepartment of Radiation Oncology, Northern Sydney Cancer Centre, Royal North Shore Hospital, St Leonards, NSW 2065, Australia

a r t i c l e i n f o

Article history:Received 16 May 2012Accepted 10 July 2012Available online 20 July 2012

Keywords:Uterus didelphysEndometrial cancerIntravaginal brachytherapyIntravaginal moulage brachytherapyMouldMoulage

⁎ Corresponding author at: Northern SydneySt Leonards, NSW, 2065, Australia. Fax: +61 99

E-mail address: Lucinda.Morris@swahs.he

2211-338X/$ – see front matter. Crown Copydoi:10.1016/j.gynor.2012.07.001

Introduction

Adjuvant intravaginal brachytherapy (IVBT) is an evidenced-based strategy that significantly reduces the long-term rate of post-hysterectomy vaginal mucosal relapse in women with intermediateand high-risk endometrial cancer (EC) (Nout et al., 2010). Due tothe physical properties of brachytherapy, optimal IVBT dose deliveryis dependent on close conformance of the vaginal applicator withthe mucosal target tissue. Internationally (Nout et al., 2010; Small etal., 2005) the standard IVBT applicator is a multi-diameter rigid round-topped segmented or solid plastic cylinder with a central metal tandemcatheter. A recent study however, has questioned the anatomic suitabil-ity of the rigid cylinder system in all post-hysterectomy EC patients andsuggested that IVBT be customized to the final vaginal vault topography(Miller et al., 2010).Weused this approach in anunusual case of bilateralEC in a patient with a congenital Mullerian tract anomaly (uterusdidelphys; UD).

Case history

In May 2010, a 78-year-old nulliparous woman (BMI 36 kg.m−2)with known UD and previous vaginal septoplasty was investigatedfor post-menopausal bleeding. Speculum examination revealed bilat-eral cervices with an intervening partial septum within the proximalvagina. Abdominal ultrasound showed complete reproductive tractduplication without renal dysgenesis. Fractional curettage confirmed

Cancer Centre, Royal North Shore Hospital,266833.alth.nsw.gov.au (L. Morris).

right © 2012 Published by Elsevier Inc. All rig

International Federation of Obstetrics and Gynaecology (FIGO) welldifferentiated (G1) endometrioid adenocarcinoma in each hemi-uterus and subsequent surgical staging (Fig. 1) demonstrated deeplyinfiltrating Grade 1 endometrioid adenocarcinoma of the right hemi-uterus (12 mm of 20 mm thick myometrium) with lymph-vascularspace invasion (FIGO 2008 Stage IB). No residual EC was found withinthe hypoplastic left uterus (final FIGO Stage IA without invasion).Lymph node sampling was not considered due to the low histologicalgrade at curettage. Adjuvant IVBT was recommended based on herhigh intermediate-risk status (i.e. age >70 years, deepmyometrial in-filtration and lymph-vascular space invasion) (Nout et al., 2010; Kunoset al., 2011).

Fig. 1. Uterus didelphys (UD): In vivo (top) and ex vivo (bottom). Note larger righthemi-uterus (*), normal paired fallopian tube and ovary, and presence of the recto-vesicalligament between the hemi-uteri. The latter is a pathognomonic finding in UD.

hts reserved.

Fig. 2. Final IVBTmould constructed from vaginal alginate impression. Note noncylindricalconfiguration, asymmetric proximal expansion, andmulti-cathererHDRB loading capacity.

122 L. Morris et al. / Gynecologic Oncology Reports 2 (2012) 121–123

Vaginal examination 6 weeks post-hysterectomyhowever, revealedgross disturbance of proximal vaginal topographywith deep involutionof the central vault due to duplicated vaginas and attempted resectionof the septal remnant. At the time of brachytherapy assessment,standard “best-fit” cylinder IVBT was seen to be anatomically non-conformal with the vaginal mucosa on pelvic CT scan. A vaginal mouldwas thus constructed using the Institut Gustave Roussey “moulage”technique (Albano et al., 2008) (Fig. 2). Briefly, the moulage methoduses a liquid alginate paste to capture a precise impression of surfacetopography of the vaginal vault and shaft. With the patient in the dorsallithotomy position, alginate paste was injected to fill the vagina underlight pressure. The paste solidifies within minutes forming an alginate“caste” which was extracted using a modification of the GustaveRoussey gauze strip system (we used corrugated plastic tape) andconverted to a personalized acrylic mould into which multiple brachy-therapy source catheters were positioned and fixated with wax. In ourpatient excellent mould-to-mucosa conformance was finally achievedusing this anatomical customizing system. Before treatment in vivoCT modelling of her vaginal mucosal surface dose (45-mm length)with the Oncentra Brachytherapy Planning System (v3.3; NucletronCorporation, Veenendaal, Netherlands) determined an optimal multi-

Fig. 3. Screen shot from Oncentra brachytherapy planning system (BPS; Nucletron B.V., Veender and rectum (with contrast). Excellent mould-to-mucosa conformity in trans-axial (top)D2cc per fraction (279 cGy) and bladder D2cc (400 cGy.)

catheter source loadingpattern (Fig. 3). Rectal, bladder, and vaginalmu-cosal volumetric exposureswere recorded. Two insertions in 8 days de-livered a vaginal surface dose of 3000 cGy in 6 fractions.

At 20 months post-IVBT, the patient remains disease-free withnormal vaginal cytology and no vaginal, rectal, or bladder toxicity.

Discussion

Uterus didelphys (UD) exists within a spectrum of congenitalanomalies in which there is complete non-fusion of the Mulleriantracts resulting in parallel duplication of the female reproductive or-gans (uterine corpus, cervix, and vagina) sometimes with renal dys-genesis. The incidence of UD is 0.1% to 0.5% (Grimbizis and Campo,2010) and is usually asymptomatic until fertility or obstetric issuesoccur. Longitudinal studies have shown that a didelphic uterus perse does not however predispose to malignancy (Heinonen, 2009) .Atotal of 9 cases of unilateral EC have been reported in the English lit-erature from 1962 to 2009 (Heinonen, 2009; Dane et al., 2009).

Synchronous endometrial cancers (EC) within both hemi-uteri areexceedingly rare having been described in only 3 previous case reports.However, in contrast to our case, all of the latter women were pre-menopausal and only one had high-risk EC (Fanfani et al., 2006;Kunos et al., 2011; Bhalla et al., 2005) which required adjuvant therapy.

This patient underwent exploratory laparotomy and had resectionof her smaller hemi-uterus and ipsilateral adnexa (tubo-ovarianmetastases) before proceeding to neo-adjuvant brachytherapy. Theremoval of one hemi-uterus allowed conventional intra-uterinetandem and vaginal colpostat low dose-rate brachytherapy. This to-gether with supplemental external radiation provided good dosimet-ric coverage of the vagina without the compromise imposed by heroriginal anatomy (Fanfani et al., 2006).

The current didelphic patient with synchronous bilateral EC is thefirst in which the fidelity of adjuvant intra-vaginal brachytherapy hadbeen prospectively questioned and for whom a customized multi-channel vaginal mould was successfully utilised. Our ability to con-struct and dosimetrically model to a wide range of vaginal topogra-phies using the moulage technique has prompted a “first-fraction”CT simulation protocol in all EC patients requiring adjuvant highdose-rate brachytherapy at our institution. Quality management ofthis clinical pathway with pelvic CT is inexpensive (approximately

endaal, Netherlands). Note vaginal mould in central pelvis between catheterized blad-, coronal (bottom) and sagittal (right) planes. DVH depicting CTV D90 (500 cGy), rectal

123L. Morris et al. / Gynecologic Oncology Reports 2 (2012) 121–123

US dollar 200 per case). From this experience (unpublished) we havefound significant post-hysterectomy vaginal cylinder to mucosa non-conformance in over 30% of women subject to pelvic CT before their ini-tial vaginal brachytherapy fraction. It is posited that this relatively highproportion is associatedwith the increasing use of laparoscopic surgicaltechniques to close the vagina and was not a factor in our current pa-tient with challenges imposed by her congenital anomaly and bilateralhemi-uterine cancers.

Conflict of interest statementThe authors declare that there are no conflicts of interest.

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