Cervical Endometriosis – Case Report and Review of Literature

1 „Prof. Dr. Panait Sarbu” Clinical Hospital of Obstetrics and Gynecology, Bucharest, Romania 2 „Elias” Emergency University Hospital, Bucharest, Romania 3 „Carol Davila” University of Medicine and Pharmacy, Bucharest, Romania Corresponding author: Octavian Enciu, Department of Surgery, Elias Emergency University Hospital, 17th Marasti Boulevard, 011461, Bucharest, Romania. E-mail: esoctavian@gmail.com Abstract


CASE REPORT
A 24-year-old Romanian woman was examined for a second opinion in the Gynecology Department of "Professor Panait Sîrbu Hospital". She was previously examined two months before and diagnosed with glandular cervical dysplasia (PAP smear that demonstrated AGCNOS). Biopsy demonstrated cervical endometriosis so she was referred to a specialized center.
During anamnesis it was noted that patient was nulliparous and suff ered dysmenorrhea 7-VAS, chronic pelvic pain 3-VAS, without dyspareunia. Th e patient was diagnosed with primary infertility. She had no previously surgical intervention and no family history of endometriosis.
On speculum examination 2 violet macular lesions, multiple 5 mm red-violet polypoid lesions at 6-9 o'clock on the vagina wall and 2 violet macular lesions on the cervix surface were observed ( Figure 1).
Bimanual pelvic examination revealed diminished uterine mobility and pain during mobilization, pain during examination of the thickened and retracted retro-cervical space. Th e posterior vaginal cul-de-sac was shortened.

INTRODUCTION
Endometriosis became a public health problem both due to the increasing number of aff ected patients and the severe quality of life impairment. Th e incidence of the disease has been estimated to 5-10% of adult women, with a rise in incidence with respect to the previous generations, aff ecting presently roughly 176 million women throughout the world 1 .
First reported by Fels in 1928, cervical endometriosis it was considered a rare localization of this disease but authors like Williams, Novak and Hoge have overturned this hypothesis, Williams having published a series of 111 cases by 1960 2,3 .
For a correct defi nition of this pathology, primary and secondary endometriosis need to be understood as distinct entities. Secondary endometriosis represents the extension of the disease from the pelvis, usually from the rectovaginal septum, and is more frequent. Primary cervical endometriosis is a rare entity and is defi ned as ectopic endometrial tissue at the level of the cervix 4 .
Siddal and Mack have established 4 criteria for primary endometriosis: 1-localization of endometrial tissue on the anterior lip of the cervix; 2-presence of endometrial tissue should be limited to the surface of the cervix and should not extend beyond the cervical squamous epithelium; 3-the absence of uterine, rectovaginal septum and vaginal wall endometriosis; 4-cessation of symptoms after removal of cervical endometrial lesions 5,6 .
Cervical procedures (punch biopsy, LEEP, conization) or intrapartum cervical laceration, are considered risk factors for cervical endometriosis. Due to the increased rates of these procedures in the last decades, a rise in incidence of cervical endometriosis should be expected 7 .
Th e diff erential diagnosis includes benign cervical lesions as polyps, myoma, endocervical glandular dysplasia, adenocarcinoma in situ and rarely invasive adenocarcinoma. Several cases have been reported where after radical surgery for abnormal PAP smear results, fi nal pathology demonstrated cervical endometriosis 10 . Colposcopy was unrevealing because the squamocolumnar junction was type III. During examination with acetic acid and Lugol no new lesions could be described. Transvaginal ultrasound was normal.
Th e patient had no digestive symptoms so pelvic MRI was not mandatory, even though pelvic MRI with intrarectal and intravaginal gel could have revealed the extension of the endometriotic lesions.
Following the general consensus for the benefi ts of surgery for endometriosis associated with pelvic pain, the patient was considered a candidate for laparoscopic surgery [11][12][13] .
During laparoscopic exploration the uterus and the ovary appeared normal so as did the peritoneum lining the ovarian fossae. Th e anterior aspect of the rectum was adherent to the uterus above the isthmus ( Figure  2).
Th e dissection began within normal anatomy with the identifi cation and dissection of the ureters in the iliac part and continued at the paracervical level. Th en the pararectal spaces were opened with the dissection of the uterosacral ligaments, having exposed the anterior and lateral parts of the rectum. Dissection continued in the space between the rectum and the cervix with the identifi cation and resection of a large endometriosis nodule. Endometriosis typically invades surrounding tissues and an ideal dissection plane most often can't be found to allow en-block resection, in our case the resection left endometriotic tissue both on the anterior aspect of the rectum, the cervix and the vagina. Th e remaining tissue on the rectum was removed by shaving with monopolar cautery. Th e tissue remaining on the vagina and cervix was clinically reevaluated by intraoperative vaginal examination and resection was decided. En-block resection of a 3/3 cm vaginal nodule followed with continuous suture of the vaginal wall and cervical shaving. Th e cervical canal was tutored with a stent in order to prevent stenosis (Figure 3-4). Bilateral tubal permeability test was positive ( Figure 5). Final pathology examination confi rmed cervical endometriosis ( Figure 6).
Th e postoperative course was uneventful, the pelvic drainage was removed in the second postoperative day and the cervical stent was removed after 14 days.
Th e patient completed the EHP-30 questionnaire both before and after the surgery at 6, 12 and 24 months 14 . Dysmenorrhea and dyspareunia were certifi ed using the VAS (visual analogue scale) 15 . Preoperative dysmenorrhea scored 7, dyspareunia 0 and 3 for chronic pelvic pain while during follow up at 2 years dysmenorrhea scored 2 and dyspareunia scored 0 and chronic pelvic pain 1.   Patients with secondary cervical endometriosis should be referred to a specialized center because the surgical treatment may imply a high degree of diffi culty. Extensive endometriosis with colorectal involvement may require a multidisciplinary team including a digestive surgeon 18 .

CONCLUSION
Th e diff erential diagnosis between primary and secondary cervical endometriosis is very important because the therapeutic management is diff erent. Th e main concern is the undertreatment of secondary endometriosis, only prolonging these patients' angst.
Laparoscopic surgery for endometriosis is proven to improve the patients' quality of life, diminishing the symptoms and increases the likelihood of obtaining a spontaneous pregnancy.
Th e presented case illustrates the benefi ts of surgery for secondary cervical endometriosis with marker improvement in quality of life proven with EHP-30 up to 2 years after surgery.

Compliance with ethics requirements:
Th e authors declare no confl ict of interest regarding this article. Th e authors declare that all the procedures and experiments of this study respect the ethical standards in the Helsinki Declaration of 1975, as revised in 2008(5), as well as the national law. Informed consent was obtained from all the patients included in the study.

DISCUSSION
Th e peculiarity of the cases resides in the fact that the patient had none of the symptoms most frequently associated with cervical endometriosis and although she had dysmenorrhea, dyspareunia was absent. Th e endometriotic lesions involved the rectovaginal septum and mobilization of the cervix provoked pain. For the less acquainted physician, the endometriotic lesions on the cervix may represent the sole lesions when in fact these are the tip of the iceberg. In this case, the ERAD approach would have been suboptimal.
Th e patient does not present none of the risk factors for cervical endometriosis (natural labor with secondary impairment of the cervix, cervical procedures -punch biopsy, ERAD, conization).
Another remarkable aspect of the presented case is that the lesions were limited to the rectovaginal space, cervix and vagina, with no involvement of the ovary and the peritoneum lining in the ovarian fossae. If endometrioma would have been found on ultrasound the diagnosis would have been ready made.
Cervical endometriosis may mimic cervical glandular anomalies with false positive PAP smear results. Th ese results are explained by cytomorphological alterations under hormonal variations during the menstrual cycle 16 .
Patients with asymptomatic primary cervical endometriosis do not require treatment, the wait and see approach being feasible. Symptomatic cases diagnosed with primary cervical endometriosis have indication for ERAD. Local destructive techniques like diathermy have high recurrence rates 17 . Cryotherapy and laser therapy have insuffi cient data reported.