Introduction

Endometriosis is a pathological condition characterised by the presence of endometrial glands and stroma outside the uterus(1). It can occur in multiple sites such as ovary, pouch of Douglas, abdominal wall, gastro intestinal tract, peritoneum, breast, nose, lungs and previous surgical scars(2). Most common clinical presentation is cyclical pain, abdominal pain, dyspareunia, dysmenorrhoea, infertility followed by pain during urination and low back pain(3). It can occur in reproductive age groups, seen in 3-10% of young females(4). Endometriosis can mimic various gynaecological, gastrointestinal, peritoneal lesions and pose diagnostic and therapeutic challenges. Therefore, we are presenting this case series of endometriosis with varying clinical presentation, anatomic sites and synchronous pathologies to enhance clinicians' ability to recognize the diverse faces of endometriosis and tailor management strategies accordingly.

Case Reports

Case 1:

Thirty year female P1L1 presented with abdominal swelling and pain for 2 years. Ultrasonogram revealed tuboovarian mass and subacute appendicitis. Patient underwent excision of abdominal wall swelling, salpingoophrectomy and appendicectomy. Histopathological examination of tubovarian mass showed endometriotic cyst with calcified parasite of Enterobius vermicularis and organised abscess. HPE of appendix and abdominal wall showed Enterobius vermicularis of appendix and endometriosis respectively.

Case 2:

Thirty year female P2L2 presented with cyclical abdominal pain for 5 years. Ultrasonogram revealed Right tuboovarian mass. Patient underwent salpingo-oophrectomy and appendicectomy. Histopathological examination revealed endometriosis of right fallopian tube and ovary along with salpingitis isthmic nodosa and reactive lymphoid hyperplasia of appendix.

Case 3:

Thirty seven year female P1L1 presented with abdominal pain on and off for 3 years. Ultrasonogram revealed ovarian cystic lesion measuring 4x 3 cm and cervical fibroid. Possibility of benign ovarian cystic lesion in ovary and cervical fibroid was considered. Patient underwent total abdominal hysterectomy with salpingo oophorectomy. Biopsy report revealed endometriotic ovarian cyst, fallopian tube endometriosis, salpingitis isthmic nodosa and cervical leiomyoma with endometriosis and giant cell reaction.

Case 4:

Thirty year old female P2L2 presented with swelling over LSCS scar for 4 years. Patient underwent wide excision of swelling. Histopathological examination revealed scar endometriosis with giant cell reaction.

Case 5:

Twenty four year old female nulligravida presented with abdominal distension and pain for 2 years. Ultrasonogram revealed bilateral benign ovarian cyst. Histopathological examination revealed bilateral endometriotic ovarian cyst and reactive lymphoid hyperplasia of appendix.

Case 6:

Twenty nine year old female presented with pain and swelling over LSCS scar for 3 years. Patient underwent wide excision of swelling. Histopathological examination revealed scar endometriosis.

Figure 1: a) Gross of Endometriotic cyst with organised abscess b) Gross of Scar endometriosis – fibrous tissue with tiny haemorrhagic cystic spaces.

Figure 2: Histopathological images a) Calcified Enterobius vermicularis in ovarian cyst with organised abscess b) Endometrial glands and stroma within ovarian cyst c) Collection of hemosiderin laden macrophages d) Cut section of Enterobius Vermicularis in appendix specimen received along with ovarian cyst. e) Appendix showing reactive lymphoid hyperplasia.

Discussion:

Endometriosis is characterized by the occurrence of endometrial tissue outside the uterus, which has the capability to react to hormonal signals from the ovaries(5). Typical presentation of endometriosis includes cyclical abdominal pain, dysmenorrhoea and chronic infertility. Most commonly, endometriosis occurs in the fallopian tubes, ovaries and scar site. Less common sites are cervix, GIT, peritoneum, nose and lung.

Various theories have been proposed for the pathology of endometriosis. Among them the retrograde flow of endometrial tissue fragments/cells and protein-rich fluid through the fallopian tubes into the pelvic area during menstruation is widely regarded as the primary explanation for the formation of endometriotic lesions within the peritoneal cavity. However, this mechanism alone is not adequate, given that nearly all women experience retrograde menstruation. Other proposed origins include celomic metaplasia, as well as lymphatic and vascular metastasis(6).

None of the literature has described about the incidence of synchronous pathologies of endometriosis. Many literatures have stated that chronic inflammation is the etiological factor for endometriosis. As stated in the literature we have encountered salpingitis isthmic nodosa as the most common synchronous pathologies along with endometriosis. This proves that chronic inflammation acting as the risk factor for endometriosis.

Enterobius vermicularisis is a helminthic infection that most commonly affects the intestine. Enterobius vermicularis infection of female genital tract is rare. Enterobiasis is most commonly asymptomatic. It occurs after ingestion of eggs by self inoculation. Larva hatches in the small intestine. Male worm dies during copulation whereas gravid female worm migrate to the perianal area to lay eggs at night which produces perianal itching. Retroinfection to caecum can also occur. Rarely from anus Enterobius can migrate to the vagina then to the uterus, fallopian tube, ovary and peritoneum producing Enterobius infection of female genital tract(7). Synchronous pathology of endometriosis with enterobiasis in ovary has not yet been reported so far. Enterobius infection in the ovary often presents clinically as a tubo-ovarian mass, with symptoms related to infertility and conditions that mimic appendicitis. Consequently, many patients undergo an appendicectomy for acute appendicitis. In our case series, we observed reactive lymphoid hyperplasia of the appendix and resolving appendicitis in appendectomy specimens. These findings were noted alongside specimens from the female genital tract exhibiting endometriosis.

Conclusion:

Endometriosis is a significant gynecological issue affecting women of reproductive age, often leading to complications such as infertility. Chronic inflammation is a significant etiological factor, as evidenced by the frequent co-occurrence of conditions such as salpingitis isthmic nodosa. Additionally, we present a rare case of synchronous endometriosis and Enterobius vermicularis infection in the ovary, highlighting the need for clinicians to consider diverse etiologies and pathologies in diagnosis and treatment planning. This case series highlights the importance of recognizing the varied presentations and potential complications associated with endometriosis to optimize patient outcomes.

References:

1. Parasar P, Ozcan P, Terry KL. Endometriosis: Epidemiology, Diagnosis and Clinical Management. Curr Obstet Gynecol Rep. 2017 Mar;6(1):34–41.
2. Kavitha T, Shanthi N. Endometriosis externa: A rare presentation. Indian Journal of Obstetrics and Gynecology Research. 2022;9(4):578–81.
3. Galle PC. Clinical Presentation and Diagnosis of Endometriosis. Obstetrics and Gynecology Clinics of North America. 1989 Mar 1;16(1):29–42.
4. Hooda R, Jaglan A, Sirrohiwal D, Chaudhry M. Post-surgical endometriosis with varying scenarios: A retrospective study. Int J Clin Obstet Gynaecol. 2020 Mar 1;4(2):368–71.
5. Paramythiotis D, Karlafti E, Tsomidis I, Iraklis G, Malliou P, Karakatsanis A, et al. Abdominal wall endometriosis: a case report. The Pan African Medical Journal [Internet]. 2022 Mar 10 [cited 2024 May 3];41(193). Available from: https://www.panafrican-med-journal.com/content/article/41/193/full
6. Horne AW, Missmer SA. Pathophysiology, diagnosis, and management of endometriosis. BMJ. 2022 Nov 14;e070750.
7. Rajesh H, Kuppusamy B, Venkataswamy C, Ganesan N. Enterobius vermicularis Infection of the Uterine Endometrium in an Infertile Female. J Obstet Gynaecol India. 2020 Feb;70(1):89–91.