Progesterone-Responsive Vaginal Leiomyoma and Hyperprogesteronemia Due to Ovarian Luteoma in an Older Bitch L
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Ferré-Dolcet et al. BMC Veterinary Research (2020) 16:284 https://doi.org/10.1186/s12917-020-02507-z CASE REPORT Open Access Progesterone-responsive vaginal leiomyoma and hyperprogesteronemia due to ovarian luteoma in an older bitch L. Ferré-Dolcet* , S. Romagnoli, T. Banzato, L. Cavicchioli, R. Di Maggio, A. Cattai, M. Berlanda, M. Schrank and A. Mollo Abstract Background: This is the first report about a vaginal leiomyoma concomitant with an ovarian luteoma in a bitch. Case presentation: A 11-year-old intact female Labrador retriever was referred because of anuria, constipation and protrusion of a vaginal mass through the vulvar commissure. The bitch had high serum progesterone concentration (4.94 ng/ml). Because of the possibility of progesterone responsiveness causing further increase of the vaginal mass and since the bitch was a poor surgical candidate a 10 mg/kg aglepristone treatment was started SC on referral day 1. A computerized tomography showed a 12.7 × 6.5 × 8.3 cm mass causing urethral and rectal compression, ureteral dilation and hydronephrosis. A vaginal leiomyoma was diagnosed on histology. As serum progesterone concentration kept increasing despite aglepristone treatment, a 0.02 ng/mL twice daily IM alfaprostol treatment was started on day 18. As neither treatment showed remission of clinical signs or luteolysis, ovariohysterectomy was performed on referral day 35. Multiple corpora lutea were found on both ovaries. On histology a luteoma was diagnosed on the left ovary. P4 levels were undetectable 7 days after surgery. Recovery was uneventful and 12 weeks after surgery tomography showed a reduction of 86.7% of the vaginal mass. The bitch has been in good health and able to urinate without any complication ever since. Conclusions: This case demonstrates the importance of identifying progesterone related conditions as well as the importance of judiciously using a combined medical and surgical approach. Keywords: Leiomyoma, Aglepristone, Prostaglandin, Luteoma Background considered to be non-invasive, slow-growing, smooth- Several reproductive conditions have been described to be muscle derived masses which do not cause metastasis in induced in bitches by ovarian steroidal hormones such as other organs [6, 7]. Ovarian tumors are reported to be un- mammary, vaginal and vulvar tumors, vaginal prolapse or common in dogs [8–11]. Sex cord-stromal tumors have the cystic endometrial hyperplasia-pyometra complex [1, the ability to be hormonally active [12]. 2]. Tumors affecting the canine vaginal vestibule or vulva Surgical excision of vaginal masses through episiotomy are rare, accounting for 2.4–3% of all tumors occurring in has been described as the treatment of choice [13–15]. the dog and being mostly benign mesenchymal tumors However, because of a) the old age of these patients and such as leiomyomas or fibromas [3–5]. These tumors are the risk performing extensive episiotomy and b) the fact that the growth of these masses may be stimulated by ovarian steroids, the use of steroidal hormones receptor * Correspondence: [email protected] Department of Animal Medicine, Production and Health, University of Padua, antagonists or surgical ovariectomy have been reported Padua, Italy © The Author(s). 2020 Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data. Ferré-Dolcet et al. BMC Veterinary Research (2020) 16:284 Page 2 of 7 as successful treatments [16, 17]. Aglepristone (AGLE) Abdominal ultrasonography showed a normal appearance (Alizin®, Virbac) is a synthetic antiprogestogen marketed of the uterine wall and mucosa and lack of fluid in the as an abortifacient in bitches in Europe and Australia uterine lumen; several antral follicles of < 0.7 cm diameter and its off-label use for progesterone-related canine con- were detected in both ovaries (Fig. 2a). Ultrasonography ditions has been reported [18]. Alfaprostol is a synthetic of the perineal area showed the mass to be originating prostaglandin F2apha (PGF2α) compound marketed for from the cranial vagina and invading the vestibulum (Fig. use in food animals and reported to be able to induce 2b). Complete blood count revealed mild leukocytosis luteolysis also in bitches [19]. (22.10 × 103/μl); biochemistry was unremarkable. Serum progesterone (P4) concentration measured with immuno- Case presentation fluorometry (AIA 369 TOSOH, FUTURLAB s.r.l., Padova, A 11-year old intact Labrador Retriever bitch was referred Italia) was 4.94 ng/mL demonstrating the presence of lu- to the Veterinary Teaching Hospital of the University of teal tissue. Because of the progressive increase in mass size Padova (Italy) because of a vaginal mass prolapsing from after the onset of proestrus and considering the potential the vulva (Fig. 1). The bitch had a history of regular cyc- presence of P4 receptors within the mass [17] a 10 mg/kg ling every 8 months, had never been pregnant, her vaccin- dose of the P4 receptor antagonist AGLE was adminis- ation and heartworm prevention programs were current tered subcutaneously on day 1 and repeated the following and had had no other health problem until shortly after day. Such treatment has been shown to reduce the size of the onset of her most recent proestrus (1 week prior to re- a vaginal leyomioma by 50% in an older crossbreed bitch ferral) when she started to become anuric and constipated. allowing for an easier surgical removal [17]. Antibiotic Because of constipation, her diet was changed to a com- therapy with enrofloxacin (5 mg/kg) for 7 days was pre- mercial fiber diet. Presence of perineal swelling, an abun- scribed. On day 8 after presentation vaginal edema was dant bloody and muco-purulent vaginal discharge and a slightly decreased in volume and the mass was not pro- mass protruding from the vulva were first noticed 3 days truding from the vulva; however, it was still causing swell- after onset of proestrus. On clinical exam on the day of re- ing of the perineum and on vaginal palpation its ferral (day 1) the bitch was alert and normally responsive consistency was unchanged. The owner reported that con- and her clinical parameters (pulse, respiration and rectal stipation had improved starting from 48 h after the second temperature, skin and subcutis and palpable lymph nodes) AGLE injection, but the bitch was still anuric and with an were normal. Her perineal area appeared enlarged and a abundant purulent vulvar discharge. On hematology and rounded mass was protruding from the vulvar labia, cov- biochemistry the only abnormality was leukocytosis ered by pink translucent vaginal mucosa. On digital palpa- (37.98 × 103/μl) while all other parameters (including kid- tion the mass had a very hard consistency and it was ney function) were normal. P4 concentrations had in- impossible to insert the operator’s finger beyond the vesti- creased to 22.4 ng/mL. On abdominal ultrasonography the bulum due to the mass occupying the entire vestibular reproductive system showed no change while dilatation of lumen. Vaginal cytology was performed and stained with the urinary bladder and ureters was observed which was Diff Quick (RAL diagnostics, Milan, Italy) showing a 1:1 causing hydronephrosis. A Foley catheter was inserted rate of intermediate and keratinized cells with abundant into the urethra and the bladder was emptied. A 10 mg/kg neutrophils and a small percentage of red blood cells. AGLE dose was administered and a combination of Fig. 1 Large ovoid-shaped vaginal mass prolapsing from the vulvar commissure of an 11-year old intact Labrador bitch Ferré-Dolcet et al. BMC Veterinary Research (2020) 16:284 Page 3 of 7 Fig. 2 a Ultrasonography of the right ovary presenting preantral follicles of 0,7 cm. b Ultrasonography of the perineal area presenting the vaginal mass encapsulated in the cranial vagina enrofloxacin and amoxicillin-clavulanic acid (5 mg and 15 bilateral pyelectasis was evident (Fig. 3a-b). Regional mg/kg, respectively) twice daily was prescribed for another lymph nodes were unremarkable. In addition to CT, an week to replace enrofloxacin as empiric treatment until ultrasound guided transcutaneous needle biopsy was per- urinary results would be available. On day 11 a total body formed using a 20 G X 100 mm (ZAMAR s.r.l., Mantova, computed tomography (CT) was performed under intra- Italy). Pressure controlled mechanical ventilation and main venous anaesthesia in order to evaluate size and position cardiovascular and respiratory variables monitoring were of the mass and determine whether other internal organs guaranteed throughout both procedures. Histopathology were affected. After premedication with 0.03 mg/kg ace- revealed a thin outer layer of mesenchymal fibrous tissue promazine (Fatro, Italy) and 0.1 mg/kg methadone with a cellular population represented by spindle elements, (Dechra, The Netherlands) intramuscularly, propofol 1% lacking defined margins with an eosinophilic cytoplasm. (Merial, Italy) was administered using a target controlled Histological patterns were compatible with a diagnosis of infusion system to induce and maintain anesthesia [20].