Huge pyogenic cervical cyst with endometriosis, developing 13 years after myomectomy at the lower uterine segment: a case report

Background: Surgical site infections are potential complications following open myomectomy. These infections usually develop immediately after the surgery, and are most often located in the myometrium. Pyogenic cervical cysts are rare and have not been previously reported to occur at the site of myomectomy. Case presentation: A 41-year-old nulligravida Japanese woman was referred to our hospital with a large cervical cyst (>15 cm in diameter). She had undergone a myomectomy 13 years previously, and the surgical site had extended to the endocervical gland. Standard blood tests did not show any evidence of inflammation. The patient underwent a total abdominal hysterectomy, which revealed that the cyst contained multiple components, including Escherichia coli, old blood, and evidence of endometriosis. A pathological review did not show malignant cells within the cyst. The pyogenic cyst originated from the upper anterior cervix, which was one of the sites involved in the previous myomectomy.

We reported a huge pyogenic cervical cyst exhibiting signs of endometriosis, in the vicinity of the uterine scar from the open myomectomy. The previous su rgery and endometriosis might have contributed to the formation of this rare pyogenic cyst.

Cervical pyogenic cyst, Uterine fibroid, Myomectomy scar, Endometriosis

Uterine fibroids are commonly observed, affecting >30% of women of reproductive age [1]. Fertility-sparing sur- gery (myomectomy) is one of the major treatment op- tions for younger patients [2], although potential risks are associated with this treatment. Myomectomy is asso- ciated with a risk of recurrent uterine fibroids, pelvic ad- hesions, as well as complications at the incision site. Furthermore, myomectomy increases the risk of uterine rupture during labor, and commonly requires planned cesarean sections [3]. Unfortunately, post-cesarean sec- tion abscess formation has been reported at the site of uterine incision [4]. Moreover, intra-uterine infection may develop before or after cesarean section, and the risk of surgical site infection has been reported to in- crease after cesarean section [5]. However, no studies have reported the development of pyogenic cervical cysts, without signs of infection, after myomectomy. In the present report, we describe a rare case of a pyogenic cer- vical cyst, further complicated by endometriosis, arising 13 years after open myomectomy. Case presentation A 41-year-old nulligravida Japanese woman was referred to our hospital (Kawakita General Hospital, Tokyo, Japan) due to the presence of a large cervical mass. Thirteen years previously, she had undergone myomectomy for the treatment of multiple uterine fibroids in the lower uterine segment, including one weighing 1.4 kg. Penetration be- yond the endocervical gland had occurred during resec- tion, and the anterior endocervical canal had been opened * Correspondence: [email protected] 1Department of Obstetrics and Gynecology, The University of Tokyo, 7-3-1 Hongo, Bunkyo-ku, Tokyo 113-8655, Japan Full list of author information is available at the end of the article © 2014 Oda et al.; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. 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. Oda et al. BMC Women's Health 2014, 14:104 http://www.biomedcentral.com/1472-6874/14/104 during the surgery. Her original follow-ups were approxi- mately once a year at another hospital, where cancer screening was performed using transvaginal ultrasonog- raphy and cytology of the cervix and endometrium. Eight years after the original surgery, she had undergone pelvic magnetic resonance imaging (MRI), which did not detect any abnormal masses in the uterine body or cervix. Twelve years after the original surgery, she underwent transvaginal ultrasonography, and no abnormal masses were detected. However, a pelvic examination, performed 13 years after her myomectomy, indicated marked enlargement of the cervix, which was suspected to be a cervical ma- lignancy. The patient had experienced no symptoms prior to the examination, and given the lack of observed progression, we were unable to estimate the period over which the mass developed. Transvaginal ultrasonography indicated that the mass contained multilocular, hypoe- choic lesions, with heterogeneous internal echogeni- city. Furthermore, MRI confirmed the presence of a multilocular, irregularly shaped cystic mass (overall diameter, >15 cm) in the upper anterior cervix (Figure 1A and B). Computed tomography (CT) did not show any evidence of enlarged lymph nodes, ascites, or a distant tumor. Blood tests did not show elevated levels of in- flammation or tumor markers. The patient ’sw h i t e blood cell count was 6,000/mL and C-reactive protein level was <0.3 mg/dL. Levels of CA125, CA19-9, and squamous cell carcinoma antigen (SCC) were 32.4 U/mL, Figure 1 Magnetic resonance imaging (MRI) of the pyogenic cervical cyst. (A) a sagittal T1-weighted and (B) a sagittal T2-weighted imaging revealed a large cystic mass at the anterior cervix. The mass was a complex multilocular cyst, consisting of at least two components. The cranial part of the cyst contained two high-intensity cystic masses on the T1-weighted image, and the caudal part contained two multilocular masses that demonstrated low-intensity on the T1-weighted image and high-intensity on the T2-weighted image. Figure 2 Macroscopic findings of the cut surface of the pyogenic cervical cyst. The distal part of the mass contained old blood, similar to that in an endometrial cyst, and the proximal part contained mucinous fluid, similar to a nabothian cyst or lobular endocervical glandular hyperplasia. Oda et al. BMC Women's Health 2014, 14:104 Page 2 of 4 http://www.biomedcentral.com/1472-6874/14/104 6.4 U/mL, and 1.4 U/mL, respectively. Results from cyto- logical examination of both the cervix and endometrium were negative for cancer. Therefore, we suspected that the mass was either a degenerated uterine fibroid, lobular endocervical glandu- lar hyperplasia, or cervical malignancy. We performed a total abdominal hysterectomy, and dense adhesions were observed in the pelvis, particularly around the cervical mass. Moreover, the cervix was markedly enlarged, given the mass in the anterior wall. However, no additional ab- scesses or ascites were detected in the abdomen. A macroscopic examination revealed that the cystic mass originated from the upper anterior cervix, which was one of the sites involved in the original myomectomy. The mass consisted of 2 components, one containing an abscess within the soft solid tissue, and another that was filled with old blood (similar to an endometrial cyst) (Figure 2). Following culture of the abscess tissue, Escherichia coli was identified as the causat ive bacteria. Microscopic examination revealed the coexistence of endometriosis, and an abscess was confirmed within the cyst, although malignant cells were not identified. An additional patho- logical review was performed at the University of Tokyo, which confirmed the absence of atypical or malignant cells. Thus, we diagnosed the mass as a pyogenic cyst, with endometriosis, which most likely occurred at the site of the uterine scar.

Surgical site infections are common complications of myomectomy, and the development of infection should be carefully examined, particularly in patients where the scar extends to the uterine cavity [6-8]. However, post- operative infections are typically early-onset, and the eti- ology of late-onset infections is not well understood. One previous study has described how the vagina can act as a bacterial reservoir during the fecal-vaginal course of transmission in extraintestinal infections [9], which could hypothetically explain the late onset in the present case. However, we are uncertain when or how the infection developed, which precludes us from con- cluding that the pyogenic cyst was caused by late-onset infection secondary to the myomectomy. Another possi- bility is that the endometriosis was significantly associ- ated with the infection, although it is surprising that no symptoms or inflammation markers were observed at the time of our initial diagnosis. This suggests that the infection did not occur immediately, and that the myo- mectomy scar was associated with the formation of endo- metriosis. Alternatively, a cervical endometriotic cyst might have progressed to pyogenic granuloma, or a gigantic cer- vical abscess might have developed de novo within the endometriosis. Patients should be carefully monitored for development of pyogenic cervical cysts; this case may indi- cate the currently unknown etiology of these cysts.

We have reported the rare case of a pyogenic cervical cyst, containing signs of endometriosis and E. coli infection, in the vicinity of the uterine scar from previous open myo- mectomy. The previous surgery might be associated with the endometriosis at the site of infection, and endometri- osis and/or infection might have contributed to the forma- tion of the cyst. Consent The patient gave written consent for the case report to be published. Abbreviations CT: Computed tomography; MRI: Magnetic resonance imaging; SCC: Squamous cell carcinoma antigen. Competing interests The authors declare that they have no competing interests. Authors’ contributions KO and YI treated the case and wrote the manuscript. DM and MF diagnosed the case pathologically. TA, KK, YO, and TF contributed to the diagnosis, obtained informed consent, and determined the management of the case. All authors read and approved the final manuscript. Acknowledgments We thank Tsuneki Nagasaka, Iwao Hosoya, and Hirofumi Haraguchi for their support. This work was supported by the Grant-in-Aid for Scientific Research (C), grant number 26462515 (to K Oda) and Grant-in-Aid for Research Activity Start-up, grant number 25893229 (to Y Ikeda) from the Ministry of Education, Culture, Sports, Science, and Technology of Japan. Author details 1Department of Obstetrics and Gynecology, The University of Tokyo, 7-3-1 Hongo, Bunkyo-ku, Tokyo 113-8655, Japan. 2Department of Obstetrics and Gynecology, Kawakita General Hospital, 1-7-3 Asagayakita, Suginami, Tokyo 166-0001, Japan. 3Department of Pathology, Kawakita General Hospital, 1-7-3 Asagayakita, Suginami, Tokyo 166-0001, Japan. 4Department of Pathology, Akita University Hospital, 44-2 Azahasunuma Hiroomote, Akita City 010-8502, Japan. 5Department of Pathology, The University of Tokyo, 7-3-1 Hongo, Bunkyo-ku, Tokyo 113-8655, Japan. Received: 11 May 2014 Accepted: 31 August 2014 Published: 3 September 2014

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