Abstract
Endometriosis is a common and painful condition. We present a case of a 33-year-old woman who had delivered triplets after in vitro fertilisation (IVF) for male factor infertility. She did not have any clinical features suggestive of endometriosis before the IVF treatment. The patient presented 7 years after conception, with premensural and postmenstrual abdominal pain and intense pain on defecation. The patient was diagnosed to have endometriosis in the tract of transvaginal ovum pick up in the right pouch of Douglas and right distal uterosacral ligament. The patient underwent excision of a nodule of endometriosis. The patient is asymptomatic 1 month after surgery. Histopathology analysis revealed features suggestive of endometriosis. The needle ovum pick up tract must have implanted the endometriotic tissue near the pararectal tissue. This is an unreported and late complication of IVF treatment.
Keywords: obstetrics and gynaecology, surgery, reproductive medicine
Background
Endometriosis is a pathological entity characterised by the abnormal growth of endometrial tissue in ectopic sites, outside the endometrial cavity. Positions in the body where endometrial tissue can be found in the context of endometriosis include the ovaries, the rectovaginal fascia and the uterosacral ligament, the peritoneal cavity, the urinary bladder, the fallopian tubes, the colorectal tract and even in the lungs.1 Endometrial glands and stroma can also be found in incisional scars, although in rare cases.2
In vitro fertilisation (IVF) has become the treatment of choice for many cases of infertility.3 Such common use of IVF has promoted the ongoing development of methods to be used in each stage of IVF.4 Oocyte pickup (OPU) is one of the most important stages and since the first description of follicular aspiration under transvaginal ultrasound guidance in the early 1980s, it has gained superiority because of its simplicity and because it is a successful method.5 6 However, it is sometimes associated with complications including puncture of blood vessels, perforation of the bowel, bleeding from the vagina vault puncture and pelvic infection. Pelvic inflammatory disease and subsequent pelvic abscess formation are rare complications of oocyte retrieval, which usually fail the procedure.7
Occult endometriosis tissue may implant in the tract of transvaginal OPU, which may manifest with uncommon clinical features.
Case presentation
A 33-year-old woman presented with severe pelvic pain which started 4–5 days before menses and ended 3–4 days after menses. She also reported of intense pelvic pain on defecation.
After getting married at 21 years of age, the patient and her husband could not conceive for 3 years of unprotected sexual intercourse. The couple was diagnosed to have male factor infertility, the cause being severe oligospermia (sperm count 6 million/mL). The patient underwent IVF treatment. During the OPU, due to the anteverted uterine position, the transvaginal ultrasound-guided OPU was technically challenging and uterine puncture was suspected. Yet the patient successfully conceived and delivered triplets by undergoing a Caesarian section. The patient had no history suggestive of endometriosis before IVF therapy. This treatment was 7 years before the current presentation.
On per-speculum examination cervix and vagina were healthy. The per-vaginal examination was very painful. The uterus was found to be bulky and anteverted. Bilateral fornices were clear. There was severe cervical motion tenderness. A nodular, cord-like, tender structure was felt posteriorly in the right lower part of the uterus going towards the rectum.
On the per-rectal digital examination, the tender nodular structure was palpated on the anterior wall.
Investigations
On clinical examination, diagnosis of endometriosis in the tract of OPU was made. Ultrasonography of the pelvis and abdomen was suggestive of the bulky uterus. There were no other significant findings.
Cancer Antigen 125 (CA 125) was within normal limits.
CT scan of the abdomen and pelvis was suggestive of a bulky cervix. Sclerosis of the ileum adjacent to the sacroiliac joints on both sides suggestive of osteitis condensates ili. MRI of the abdomen and pelvis revealed a sclerotic tract around the anterior right pararectal tissue (figure 1)
Figure 1.
MRI of abdomen and pelvis revealing sclerotic tract around the anterior right para-rectal tissue.
Treatment
The patient underwent excision of a nodule of endometriosis. After the initial laparoscopic evaluation, it was observed that the ovaries were bilaterally normal. There was a nodule of 2×3×1 cm in the pouch of Douglas, which was appearing adhered to the rectum and the right vaginal wall. There was no evidence of endometriotic lesions anywhere else in the pelvis. No evidence of any superficial or deep infiltrates or pelvic adhesions, which is usually seen in cases of endometriomas.
Due to the technical challenges, the surgeon decided to convert the surgery into open. After a midline skin incision, the peritoneal cavity was exposed and the cord-like nodular endometriotic tissue was carefully dissected away from the rectum and vaginal wall. With digital palpation, the assistant surgeon made sure that the rectum and vagina were not perforated. The nodule was excised and sent for histopathology analysis. There was no other focus of endometriosis elsewhere. There was no iatrogenic injury to the rectum and vagina.
Outcome and follow-up
The patient had an uneventful recovery. The patient was put on dienogest and gonadotropin-releasing hormone (GnRH) agonists postoperatively for 4–6 months to prevent recurrence of endometriosis. The patient has had a symptom-free interval of 8 months and does not report of pelvic pain or pain on defecation. The histopathology analysis revealed the resected nodule to be of endometriosis (figure 2)
Figure 2.
Magnified image of histopathological analysis of the resected tissue depicting features of endometriosis.
Discussion
Endometriosis, the presence of functional endometrium outside the uterine cavity, is a common disease, causing abdominal pain, dysmenorrhea, dyspareunia and infertility in about 10% of the female population.1 Besides the metaplastic transformation of endometrial and peritoneal mesothelial cells, the transplantation, implantation and growth of exfoliated menstrual debris on the peritoneal and ovarian surfaces are the widely accepted mechanisms of endometriosis.2 3 Transplantation and implantation theory of Miccini et al 2 and the coelomic metaplasia theory of Wikland et al 5 are the theories proposed for the development of endometriosis. Infertility due to endometriosis can be treated by IVF treatment. IVF and associated techniques leading to endometriosis are yet unreported in the literature.
The patient did not have any symptoms of endometriosis before the presentation.
The accidental puncture site on the uterus at the time of ovum pick up may have created a track for the implantation of endometriotic tissue. Intraoperatively both the ovaries were normal in appearance. Laparoscopic examination of the pelvis revealed no evidence of endometriosis elsewhere. Considering the above-mentioned facts, the probability of the patient harbouring endometriosis before the IVF treatment is very low. Laparoscopy has a good negative predictive value for endometriosis.8 The absence of endometriotic lesions elsewhere in the pelvis and the anatomical location of the endometriosis tissue in this patient have made the authors propose that the transvaginal oocyte retrieval may have implanted endometriotic tissue in the rectovaginal pouch.
The use of dienogest and GnRH agonist has been shown to decrease the postoperative recurrence of endometriosis.9
This is an unreported and late complication of the transvaginal OPU procedure, which is usually very safe. With our review of the literature, we could not find any case report reporting such a complication.
Patient’s perspective.
The disease was very painful. I am happy to have had the surgery done with the doctors who had helped us conceive many years ago. I feel much better. There is no pain since the past 2 months. I feel much more comfortable than before. I deeply thank the doctors. Patient herself (translated from Marathi)
Learning points.
Endometriosis can develop in the tract of transvaginal oocyte pick up tract.
Endometriosis in the tract of oocyte pickup can manifest in unusual additional clinical features such as painful defecation and pelvic pain.
Surgical excision, although challenging, can be offered successfully in these patients.
Dienogest and gonadotropin-releasing hormone agonists can help prevent the recurrence after surgical resection.
Acknowledgments
We acknowledge the constant support and encouragement of Dr. Hemant Bhausaheb Deshpande, founder and the head of Deshpande Hospital.
Footnotes
Twitter: @swanitdeshpande
Contributors: SwarikaHD: significant contribution in conceptualisation and drafting the primary manuscript, completed thorough review of the literature. SwanitHD: edited the manuscript, significant contribution in editing the language and grammar, revised the manuscript critically for intellectual content. PHD: headed the patient management team, treating physician, expert opinion in the field of infertility. AP: operating surgeon, substantial contribution in the final revision of the manuscript.
Funding: The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Competing interests: None declared.
Provenance and peer review: Not commissioned; externally peer reviewed.
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