Case Reports in Women’s Health 31 (2021) e00335 Contents lists available at ScienceDirect Case Reports in Women's Health journal homepage: www.elsevier.com/locate/crwh Pelvic inflammatory disease presenting 16 months after vaginal hysterectomy: A case report and literature review Eloy Cabello-Garcia a, Elena Ferriols-Pérez a, b, *, Berta Urpí-Tosar a, Mireia González-Comadran a, b a Obstetrics and Gynecology Department, Consorci Parc de Salut MAR, Universitat Autònoma de Barcelona, Barcelona, Passeig marítim 25-29 (08003), Spain b GRI-BCN, Barcelona Infertility Research Group, IMIM, Institut Hospital del Mar d’Investigacions Mèdiques, Barcelona, Dr. Aiguader Street, 88 (08003), Spain A R T I C L E I N F O A B S T R A C T Keywords: Pelvic inflammatory disease after hysterectomy is rare and the underlying route of infection is highly hetero­ Hysterectomy geneous. We report the case of a 52-year-old woman with a history of vaginal hysterectomy for uterine prolapse Pelvic inflammatory disease admitted to the emergency department with acute abdominal pain and fever. Vaginal discharge and pelvic Tubo-ovarian abscess tenderness were evident in the clinical examination. Ultrasound and computed tomography scans showed a cystic pelvic mass in contact with the vaginal cuff, suggesting the diagnosis of pelvic inflammatory disease. Laparoscopic examination revealed a bilateral tubo-ovarian abscess firmly attached and fistulized to the vaginal cuff, and after tubal removal and antibiotic coverage the patient had an optimal recovery. We performed a re­ view of the case reports published on this subject, and concluded that pelvic inflammatory disease should not be excluded in patients with a history of hysterectomy when symptoms and findings are compatible. 1. Introduction (in cases of appendicitis or diverticulitis). Rarely, PID can develop in patients who have previously undergone Pelvic inflammatory disease (PID) is an infection of the female upper hysterectomy. Indeed, some authors have reported cases of tubo-ovarian genital tract that affects 8–10% of sexually active women of reproduc­ abscess (TOA) from 8 months up to 16 years after the surgery [8–15]. tive age [1,2]. It is caused by the ascension of pathogenic bacteria to the This case report presents the development of PID in a patient who had uterus and the adnexa, although the mechanisms involved remain un­ had a vaginal hysterectomy 16 months earlier. The literature on this clear [3]. PID has a wide range of clinical presentations, mostly subject is reviewed in the Discussion. depending on the pathogen involved [4,5]. More than 85% of cases of acute PID (≤30 days of duration) are associated with bacterial vaginosis- 2. Case Presentation related microbes and/or sexually transmitted pathogens, while less than 15% of the cases are due to respiratory or enteric pathogens that have A 52-year-old postmenopausal, multiparous woman presented to the colonized the vaginal canal [3]. Subclinical PID is twice as common and emergency room relating a 2-day history of lower abdominal pain re­ is caused by similar pathogens as acute PID [6]. fractory to oral analgesia, and fever up to 39 ◦ C. The patient reported no The cause of chronic PID (> 30 days) is more commonly associated nausea, vomiting, diarrhea or urinary symptoms, but described white with actynomices species or even pathogens that arrive through the vaginal discharge in the previous hours. bloodstream (Mycoplasma tuberculosis) [7]. Nevertheless, it is frequent to The most relevant medical history was a vaginal hysterectomy due to find polymicrobial colonies in the bacterial cultures, commonly pelvic organ prolapse 16 months before this presentation. The patient including anaerobic bacteria [1,2]. Apart from the ascending route, it was initially evaluated in the general surgery department and the has been reported that the microorganisms can reach the genital tract abdominal examination revealed low abdominal pain, with voluntary through the bloodstream (especially in tuberculosis) or by direct contact guarding but no rebound tenderness. * Corresponding author at: Obstetrics and Gynecology Department, Consorci Parc de Salut Mar, Universitat Autònoma de Barcelona, Passeig marítim 25-29 (08003), Barcelona, Spain. E-mail addresses: ecabello@psmar.cat (E. Cabello-Garcia), eferriols@psmar.cat (E. Ferriols-Pérez), burpi@psmar.cat (B. Urpí-Tosar), mgonzalezcomadran@ psmar.cat (M. González-Comadran). https://doi.org/10.1016/j.crwh.2021.e00335 Received 31 May 2021; Received in revised form 4 June 2021; Accepted 8 June 2021 Available online 10 June 2021 2214-9112/© 2021 Published by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). E. Cabello-Garcia et al. Case Reports in Women’s Health 31 (2021) e00335 by laparoscopy and the postoperative recovery was uneventful. The patient was discharged from the hospital with oral antibiotics. At the follow-up visit, 6 weeks after discharge, the patient was full recov­ ered. Pathologic examination showed chronic and acute salpingitis, and both blood and abscess cultures were negative. 3. Discussion The development of a TOA in patients after hysterectomy is rare. In order to gain a deeper understanding of the clinical case, a review of the published literature was performed. An exhaustive electronic search was performed in PUBMED and only 8 case reports were iden­ tified regarding the development of a PID in patients with a hysterec­ Fig. 2. Left tubo-ovarian abscess measuring 30 × 40 mm. tomy at least 4 weeks prior to attendance for PID [8–15][Table 1]. All patients from the articles reviewed were women of reproductive age (the age range was 28 to 49 years) at the time of the diagnosis of the PID. Five of them (62.5%) had a undergone abdominal hysterectomy [5,8,10,11,14], while the remaining three (37.5%) had undergone vaginal hysterectomy [9,12,13]. The average time between the hyster­ ectomy and the onset of the symptoms was highly heterogeneous, with one case reported within the first year of the surgery [14], 4 cases be­ tween 2 and 6 years [8–10,12], and 2 cases after 12 and 16 years from the hysterectomy [11,15]. Abdominal pain and fever were the most consistent symptoms, as well as blood tests that repeatedly revealed leucocytosis in all the patients except for one [12]. Information regarding the antibiotic regimen and surgical treatment was provided in only 5 cases [10–12,14,15], which consisted on the removal of the fal­ lopian tubes, and in half of these cases additionally the adnexa. Cultures were reported positive in 4 cases [9–12], although 2 authors did not provide this information. The postoperative recovery was uneventful in 4 cases [10–12,15], Fig. 1. Right tubo-ovarian abscess measuring 40 × 70 mm. and only one author reported a pelvic abscess diagnosed three weeks after bilateral salpingectomy, although the suspected mechanism for PID was bacterial translocation from the gastrointestinal tract [14]. In this regard, the mechanism of infection was unknown in most cases, Laboratory test revealed white blood cell count within the normal although various mechanisms have been proposed, including an range (leykocytes 8.380/mL with 70% neutrophils), although acute ascending route through a tubo-vaginal fistula, intestinal translocation phase reactants were increased (reactive C protein 19.9 mg/dL and and a haematogenous route [8,10,14,15]. procalcitonin levels 0.065 ng/mL). The clinical case presented herein was comparable to the previous Appendicitis and diverticulitis were both excluded from the diag­ cases reviewed in the literature, in terms of both symptoms at onset and nosis after performing an abdominal ultrasound and a computed to­ findings through imaging and laboratory tests. The mechanism of mography (CT) scan. However, the CT scan revealed an elongated 76 × infection is still unknown, as all cultures came back negative. The use of 33 mm predominantly cystic mass in the right iliac region, suggesting a antibiotics prior to the surgery may explain the negative results in the right TOA. culture obtained from the TOA. The patient was then evaluated by the gynecology department. The From the surgical findings, it can be hypothesised that the mecha­ pelvic examination revealed vaginal cuff tenderness and purulent nism of infection might have been through a tubo-vaginal fistula, as the discharge, but no sign of dehiscence. The transvaginal sonography right fallopian tube adhered to the vaginal cuff. Nevertheless, the fistula findings were compatible with bilateral TOA, and there was no evidence might have also been secondary to the inflammatory process, instead of of free fluid in the pouch of Douglas that would suggest ruptured TOA the initial cause. Hematogenous colonization, although unlikely, could Fig. 1, Fig. 2]. not be excluded. Intravenous antibiotic treatment was initiated for PID, with cefex­ ime, doxycycline and metronidazole. There was a clear reduction in pain 4. Conclusions and the subsequent laboratory results showed a decrease in inflamma­ tory markers. However, the patient’s temperature persisted around A hysterectomy represents the anatomic interruption of the 37–38 ◦ C despite antibiotic treatment and, therefore, exploratory lapa­ communication between the upper and the lower female genital tract. roscopic surgery was performed. During the surgical procedure a bilat­ This article emphasises the importance of considering the diagnosis of eral TOA was confirmed. The right tube measured approximately 6 × 8 PID when symptoms suggest so, even if the patient has previously un­ cm, was firmly attached to the vaginal cuff and fistulized to the right dergone hysterectomy, especially in women among reproductive age. margin, while the left tube measured 3 × 4 cm and was attached only to the pelvic peritoneum. Both fallopian tubes were successfully removed 2 E. Cabello-Garcia et al. Case Reports in Women’s Health 31 (2021) e00335 Table 1 Cases published in the literature of pelvic inflammatory disease after hysterectomy. Author(s) Canas et al. Fletcher et al. Lau et al. Rivlin et al. Tohya et al. Mendez et al. Mosholt et al. Hueston Age (years) 38 38 28 33 49 39 42 NR Parity Terciparous Quintiparous NR Primiparous Secundiparous Primiparous NR NR Medical history Cholecystectomy Type II diabetis NR Previous STD Cesarian section Cesarian section, NR NR Tubal ligation (vaginal tubal ligation trichomona) Time between 12 years 8 months 15 months 2 years 16 years 4 years 2 years 6 years hysterectomy and the event Type of TAH TAH VH VH STAH TAH VH TAH hysterectomy Reason for Cervical dysplasia Leiomyoma NR Cervical Uterine rupture Leiomoma Uterine NR hysterectomy carcinoma in prolapse situ Symptoms Abdominal pain, Abdominal Abdominal Abdominal pain, Abdominal pain Abdominal pain Abdominal NR chills, nausea and pain, chills, pain and nausea and and chills pain, fever vomiting nausea and fever vomiting and pelvic vomiting mass Number of 12.200 22.500 22.700 9.200 12.200 17.400 NR NR Leukocytes in blood (/mm3) Antibiotic Gentamicin Gentamicin NR Cefotetan Clarythromycin Ampicilin, NR NR regime Metronidazole Clindamycin Metronidazole Cefepime Gentamicin Doxycycline Clindamycin Surgical Bilateral Bilateral NR Right Bilateral Bilateral NR NR Treatment salpingectomy salpingectomy adnexectomy adnexectomy adnexectomy Cultures Negative (abscess Negative NR Neisseria Streptococcus Streptococcus Chlamydia NR and vaginal cuff) gonorrhoeae milleri and intermedius trachomatis (vaginal Bacteroides (abscess) (peritoneal discharge) ovaltus (abscess) fluid) Route of Unknown - Unknown- Unknown Confirmed tubo- Ascending route Unknown- Falopian tube Unknown- post- infection possible tubo- possible vaginal fistula intestinal prolapse operative vaginal fistula intestinal translocation vs complication vs translocation hematogenous. hematogenous. Recovery Uneventful Pelvic abscess at NR Uneventful Uneventful Uneventful NR NR 3rd week NR, not reported; STAH, sub-total abdominal hysterectomy; TAH, total abdominal hysterectomy; VH, vaginal hysterectomy. Contributors Patient Consent Eloy Cabello-Garcia, the primary author, was a physician involved in The patient described in this case study provided written informed patient care, drafted the initial manuscript and revised and approved the consent. final submission. Elena Ferriols-Pérez, the corresponding author, was a senior physi­ cian involved in patient care, and revised the manuscript and approved Provenance and Peer Review the final submission. Berta Urpí-Tosar was a physician involved in patient care, performed This case report was peer reviewed. the literature review, revised the manuscript and approved the final submission. References Mireia González-Comadran was a senior physician involved in pa­ [1] I. Simms, J.M. Stephenson, Pelvic inflammatory disease epidemiology: What do we tient care, and the primary surgeon in this case, and revised the manu­ know and what do we need to know? Sex. Transm. Infect. 76 (2000). script and approved the final submission. [2] C.L. Haggerty, R.B. Ness, Epidemiology, pathogenesis and treatment of pelvic inflammatory disease, Expert Rev. Anti-Infect. Ther. 4 (2006) 235–247. [3] J. Ravel, I. Moreno, C. Simón, Bacterial vaginosis and its association with Conflict of Interest infertility, endometritis, and pelvic inflammatory disease, Am. J. Obstet. Gynecol. 224 (2021). The authors declare that they have no conflict of interest regarding [4] E. Cicinelli, D. De Ziegler, R. Nicoletti, G. Colafiglio, N. Saliani, L. Resta, et al., the publication of this case report. Chronic endometritis: correlation among hysteroscopic, histologic, and bacteriologic findings in a prospective trial with 2190 consecutive office hysteroscopies, Fertil. Steril. 89 (3) (2008). Funding [5] R.C. Brunham, S.L. Gottileb, J. Paavonen, Pelvic inflammatory disease, N. Engl. J. Med. 372 (21) (2015) 2039–2048. [6] K. Holmes, P. Sparling, W. Stamm, P. Piot, J. Wasserheit, L. Corey, et al., Sexually No funding from an external source supported the publication of this Transmitted Diseases, 4th ed., The McGraw-Hill Companies, New York, 2007, case report. pp. 344–345. [7] D.G. Tsevat, H.C. Wiesenfeld, C. Parks, J.F. Peipert, Sexually transmitted diseases and infertility, Am. J. Obstet. Gynecol. 216 (1) (2017) 1–9. Available from, htt ps://doi.org/10.1016/j.ajog.2016.08.008. [8] W.J. Hueston, A case of tubo-ovarian abscess 6 years after hysterectomy, J. Ky Med. Assoc. 90 (3) (1992) 114–116. 3 E. Cabello-Garcia et al. Case Reports in Women’s Health 31 (2021) e00335 [9] K.S.S. Mosholt, T.A. Filtenborg, Fallopian tube prolapse and Chlamydia peritonitis [13] M. Lau, C.A. Cross, P. Berens, K. Cunningham, J. Mahnke, V. Ali, et al., Ovarian after vaginal hysterectomy, Ugeskr. Laeger 174 (3) (2012). abscess 15 months after vaginal hysterectomy: A case report, J Reprod Med Obstet [10] L.E. Mendez, S.M. Bhoola, I.R. Horowitz, Bilateral tubo-ovarian abscesses four Gynecol 42 (10) (1997). years after total abdominal hysterectomy, Infect. Dis. Obstet. Gynecol. 6 (3) (1998) [14] J.L. Fletcher, T.E. Nolan, M.J. Milam, Late tubo-ovarian abscess following 138–140. abdominal hysterectomy, J. Fam. Pract. 33 (2) (1991). [11] T. Tohya, T. Yoshimura, C. Onoda, Tubo-ovarian abscess occurring 16 years after [15] A.M. Canas, B. Holloran-Schwartz, T. Myles, Tuboovarian abscess 12 years after supracervical hysterectomy, Infect. Dis. Obstet. Gynecol. 11 (3) (2003). total abdominal hysterectomy, Obstet. Gynecol. 104 (5) (2004) 1039–1041. [12] M.E. Rivlin, Tubovaginal fistula after vaginal hysterectomy complicated by a tubo- ovarian abscess and diffuse peritonitis, Obstet. Gynecol. 94 (5 SUPPL. 1) (1999 Nov) 858. 4