{"paper_id":"5338fb63-9a36-43df-91c8-f7557531bdb7","body_text":"C A S E R E P O R T Open Access\nHuge pyogenic cervical cyst with endometriosis,\ndeveloping 13 years after myomectomy at the\nlower uterine segment: a case report\nKatsutoshi Oda 1*, Yuji Ikeda 1,2, Daichi Maeda 3,4, Takahide Arimoto 1, Kei Kawana 1, Masashi Fukayama 5,\nYutaka Osuga 1 and Tomoyuki Fujii 1\nAbstract\nBackground: Surgical site infections are potential complications following open myomectomy. These infections\nusually develop immediately after the surgery, and are most often located in the myometrium. Pyogenic cervical\ncysts are rare and have not been previously reported to occur at the site of myomectomy.\nCase presentation: A 41-year-old nulligravida Japanese woman was referred to our hospital with a large cervical\ncyst (>15 cm in diameter). She had undergone a myomectomy 13 years previously, and the surgical site had extended\nto the endocervical gland. Standard blood tests did not show any evidence of inflammation. The patient underwent a\ntotal abdominal hysterectomy, which revealed that the cyst contained multiple components, including Escherichia coli,\nold blood, and evidence of endometriosis. A pathological review did not show malignant cells within the cyst. The\npyogenic cyst originated from the upper anterior cervix, which was one of the sites involved in the previous\nmyomectomy.\nConclusion: We reported a huge pyogenic cervical cyst exhibiting signs of endometriosis, in the vicinity of the\nuterine scar from the open myomectomy. The previous su rgery and endometriosis might have contributed to\nthe formation of this rare pyogenic cyst.\nKeywords: Cervical pyogenic cyst, Uterine fibroid, Myomectomy scar, Endometriosis\nBackground\nUterine fibroids are commonly observed, affecting >30%\nof women of reproductive age [1]. Fertility-sparing sur-\ngery (myomectomy) is one of the major treatment op-\ntions for younger patients [2], although potential risks\nare associated with this treatment. Myomectomy is asso-\nciated with a risk of recurrent uterine fibroids, pelvic ad-\nhesions, as well as complications at the incision site.\nFurthermore, myomectomy increases the risk of uterine\nrupture during labor, and commonly requires planned\ncesarean sections [3]. Unfortunately, post-cesarean sec-\ntion abscess formation has been reported at the site of\nuterine incision [4]. Moreover, intra-uterine infection\nmay develop before or after cesarean section, and the\nrisk of surgical site infection has been reported to in-\ncrease after cesarean section [5]. However, no studies\nhave reported the development of pyogenic cervical cysts,\nwithout signs of infection, after myomectomy. In the\npresent report, we describe a rare case of a pyogenic cer-\nvical cyst, further complicated by endometriosis, arising\n13 years after open myomectomy.\nCase presentation\nA 41-year-old nulligravida Japanese woman was referred\nto our hospital (Kawakita General Hospital, Tokyo, Japan)\ndue to the presence of a large cervical mass. Thirteen\nyears previously, she had undergone myomectomy for the\ntreatment of multiple uterine fibroids in the lower uterine\nsegment, including one weighing 1.4 kg. Penetration be-\nyond the endocervical gland had occurred during resec-\ntion, and the anterior endocervical canal had been opened\n* Correspondence: katsutoshi-tky@umin.ac.jp\n1Department of Obstetrics and Gynecology, The University of Tokyo, 7-3-1\nHongo, Bunkyo-ku, Tokyo 113-8655, Japan\nFull list of author information is available at the end of the article\n© 2014 Oda et al.; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative\nCommons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and\nreproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain\nDedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article,\nunless otherwise stated.\nOda et al. BMC Women's Health 2014, 14:104\nhttp://www.biomedcentral.com/1472-6874/14/104\n\nduring the surgery. Her original follow-ups were approxi-\nmately once a year at another hospital, where cancer\nscreening was performed using transvaginal ultrasonog-\nraphy and cytology of the cervix and endometrium. Eight\nyears after the original surgery, she had undergone pelvic\nmagnetic resonance imaging (MRI), which did not detect\nany abnormal masses in the uterine body or cervix.\nTwelve years after the original surgery, she underwent\ntransvaginal ultrasonography, and no abnormal masses\nwere detected.\nHowever, a pelvic examination, performed 13 years\nafter her myomectomy, indicated marked enlargement\nof the cervix, which was suspected to be a cervical ma-\nlignancy. The patient had experienced no symptoms\nprior to the examination, and given the lack of observed\nprogression, we were unable to estimate the period over\nwhich the mass developed. Transvaginal ultrasonography\nindicated that the mass contained multilocular, hypoe-\nchoic lesions, with heterogeneous internal echogeni-\ncity. Furthermore, MRI confirmed the presence of a\nmultilocular, irregularly shaped cystic mass (overall\ndiameter, >15 cm) in the upper anterior cervix (Figure 1A\nand B). Computed tomography (CT) did not show any\nevidence of enlarged lymph nodes, ascites, or a distant\ntumor. Blood tests did not show elevated levels of in-\nflammation or tumor markers. The patient ’sw h i t e\nblood cell count was 6,000/mL and C-reactive protein\nlevel was <0.3 mg/dL. Levels of CA125, CA19-9, and\nsquamous cell carcinoma antigen (SCC) were 32.4 U/mL,\nFigure 1 Magnetic resonance imaging (MRI) of the pyogenic cervical cyst. (A) a sagittal T1-weighted and (B) a sagittal T2-weighted imaging\nrevealed a large cystic mass at the anterior cervix. The mass was a complex multilocular cyst, consisting of at least two components. The cranial\npart of the cyst contained two high-intensity cystic masses on the T1-weighted image, and the caudal part contained two multilocular masses\nthat demonstrated low-intensity on the T1-weighted image and high-intensity on the T2-weighted image.\nFigure 2 Macroscopic findings of the cut surface of the\npyogenic cervical cyst. The distal part of the mass contained old\nblood, similar to that in an endometrial cyst, and the proximal part\ncontained mucinous fluid, similar to a nabothian cyst or lobular\nendocervical glandular hyperplasia.\nOda et al. BMC Women's Health 2014, 14:104 Page 2 of 4\nhttp://www.biomedcentral.com/1472-6874/14/104\n\n6.4 U/mL, and 1.4 U/mL, respectively. Results from cyto-\nlogical examination of both the cervix and endometrium\nwere negative for cancer.\nTherefore, we suspected that the mass was either a\ndegenerated uterine fibroid, lobular endocervical glandu-\nlar hyperplasia, or cervical malignancy. We performed a\ntotal abdominal hysterectomy, and dense adhesions were\nobserved in the pelvis, particularly around the cervical\nmass. Moreover, the cervix was markedly enlarged, given\nthe mass in the anterior wall. However, no additional ab-\nscesses or ascites were detected in the abdomen. A\nmacroscopic examination revealed that the cystic mass\noriginated from the upper anterior cervix, which was\none of the sites involved in the original myomectomy.\nThe mass consisted of 2 components, one containing an\nabscess within the soft solid tissue, and another that was\nfilled with old blood (similar to an endometrial cyst)\n(Figure 2).\nFollowing culture of the abscess tissue, Escherichia coli\nwas identified as the causat ive bacteria. Microscopic\nexamination revealed the coexistence of endometriosis,\nand an abscess was confirmed within the cyst, although\nmalignant cells were not identified. An additional patho-\nlogical review was performed at the University of Tokyo,\nwhich confirmed the absence of atypical or malignant\ncells. Thus, we diagnosed the mass as a pyogenic cyst,\nwith endometriosis, which most likely occurred at the site\nof the uterine scar.\nDiscussion\nSurgical site infections are common complications of\nmyomectomy, and the development of infection should\nbe carefully examined, particularly in patients where the\nscar extends to the uterine cavity [6-8]. However, post-\noperative infections are typically early-onset, and the eti-\nology of late-onset infections is not well understood.\nOne previous study has described how the vagina can\nact as a bacterial reservoir during the fecal-vaginal\ncourse of transmission in extraintestinal infections [9],\nwhich could hypothetically explain the late onset in the\npresent case. However, we are uncertain when or how\nthe infection developed, which precludes us from con-\ncluding that the pyogenic cyst was caused by late-onset\ninfection secondary to the myomectomy. Another possi-\nbility is that the endometriosis was significantly associ-\nated with the infection, although it is surprising that no\nsymptoms or inflammation markers were observed at\nthe time of our initial diagnosis. This suggests that the\ninfection did not occur immediately, and that the myo-\nmectomy scar was associated with the formation of endo-\nmetriosis. Alternatively, a cervical endometriotic cyst might\nhave progressed to pyogenic granuloma, or a gigantic cer-\nvical abscess might have developed de novo within the\nendometriosis. Patients should be carefully monitored for\ndevelopment of pyogenic cervical cysts; this case may indi-\ncate the currently unknown etiology of these cysts.\nConclusions\nWe have reported the rare case of a pyogenic cervical cyst,\ncontaining signs of endometriosis and E. coli infection, in\nthe vicinity of the uterine scar from previous open myo-\nmectomy. The previous surgery might be associated with\nthe endometriosis at the site of infection, and endometri-\nosis and/or infection might have contributed to the forma-\ntion of the cyst.\nConsent\nThe patient gave written consent for the case report to\nbe published.\nAbbreviations\nCT: Computed tomography; MRI: Magnetic resonance imaging;\nSCC: Squamous cell carcinoma antigen.\nCompeting interests\nThe authors declare that they have no competing interests.\nAuthors’ contributions\nKO and YI treated the case and wrote the manuscript. DM and MF\ndiagnosed the case pathologically. TA, KK, YO, and TF contributed to the\ndiagnosis, obtained informed consent, and determined the management of\nthe case. All authors read and approved the final manuscript.\nAcknowledgments\nWe thank Tsuneki Nagasaka, Iwao Hosoya, and Hirofumi Haraguchi for their\nsupport. This work was supported by the Grant-in-Aid for Scientific Research\n(C), grant number 26462515 (to K Oda) and Grant-in-Aid for Research Activity\nStart-up, grant number 25893229 (to Y Ikeda) from the Ministry of Education,\nCulture, Sports, Science, and Technology of Japan.\nAuthor details\n1Department of Obstetrics and Gynecology, The University of Tokyo, 7-3-1\nHongo, Bunkyo-ku, Tokyo 113-8655, Japan. 2Department of Obstetrics and\nGynecology, Kawakita General Hospital, 1-7-3 Asagayakita, Suginami, Tokyo\n166-0001, Japan. 3Department of Pathology, Kawakita General Hospital, 1-7-3\nAsagayakita, Suginami, Tokyo 166-0001, Japan. 4Department of Pathology,\nAkita University Hospital, 44-2 Azahasunuma Hiroomote, Akita City 010-8502,\nJapan.\n5Department of Pathology, The University of Tokyo, 7-3-1 Hongo,\nBunkyo-ku, Tokyo 113-8655, Japan.\nReceived: 11 May 2014 Accepted: 31 August 2014\nPublished: 3 September 2014\nReferences\n1. 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Fertil Steril 2006, 85:36–39.\nOda et al. BMC Women's Health 2014, 14:104 Page 3 of 4\nhttp://www.biomedcentral.com/1472-6874/14/104\n\n8. Rivlin ME, Patel RB, Carroll CS, Morrison JC: Diagnostic imaging in uterine\nincisional necrosis/dehiscence complicating cesarean section. J Reprod\nMed 2005, 50:928–932.\n9. Obata-Yasuoka M, Ba-Thein W, Tsukamoto T, Yoshikawa H, Hayashi H:\nVaginal Escherichia coli share common virulence factor profiles, serotypes\nand phylogeny with other extraintestinal E. coli. Microbiology 2002,\n148: 2745–2752.\ndoi:10.1186/1472-6874-14-104\nCite this article as: Oda et al. : Huge pyogenic cervical cyst with\nendometriosis, developing 13 years after myomectomy at the lower\nuterine segment: a case report. 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