An increasing trend of iatrogenic scar endometriosis after lower segment cesarean section

Endometriosis; Scar; Cesarean; Iatrogenic Disease (Mesh terms) Nishat Fatema 1* ，Dil Anziz Begum 1 , Shirin Fatema 1 , A.K.M Arif Uddin Ahmed 2 1 Department of Obstetrics & Gynecology, Imperial Hospital Limited, Chattogram Bangladesh 2 Academic Coordinator Imperial Hospital Limited, Chattogram Bangladesh ABSTRACTIJCR : https://escipub.com/international-journal-of-case-reports/ 1 N ishat Fatema et al., IJCR, 2020 4:117IJ CR: https://escipub.com/international-journal-of-case-reports/ 2

The growth of functional endometrial tissues outside the uterine cavity is termed as endometriosis. It is a gynecological dis order depended on the sex hormone1. Endometriosis is categorized as intra-pelvic and extra-pelvic endometriosis. Intra -pelvic endometriosis is developed in different pelvic structures like ovaries, utero sacral ligaments, pelvic peritoneum, recto -uterine pouch, cervix, vagina, and round lig ament. Although extra- pelvic endometriosis is rare but may develop in gastrointestinal tract , respiratory tract, urinary tract, skin, brain, and abdominal wall2. In previous studies, the etiopathogenesis of scar endometriosis is described as, the direct implantation of endometrial tissues to the uterine scar, abdominal musculature or subcutaneous tissue during surgical procedures which eventually become active and spread under the influence of Oestrogen3. Abdominal wall endometriosis is seen at the surgical incision site following obstetric or gynecological surgeries, commonly observed in cesarean section scar4. During surgery, as the wall of the uterus is cut and opened, the endometrial cells may move to the pelvic cavity by amniotic fluid and may be transported to ectopic sites, such as on the skin, subcutaneous tissues, muscles of the abdomen, near the scar. Th en the implanted endometrial cells at the new site have the capability to proliferate due to a highly vascularized environment and then the hormonal effects (mainly for oestrogen) allowing the implanted endometrial tissues to grow and to form a mass which leads to clinical symptoms5. The causes of iatrogenic scar endometriosis (ISE) are mainly due to the longitudinal pattern of the abdominal vessels and the large dissection. During a Pfannenstiel incision , more capillaries are cut off than in a vertical incision, causing more blood loss. So the Pfannenstiel incision that is commonly used in the lower segment cesarean section (LSCS) is the most commonly reported type for the occurrence of ISE; The endometrial cells require an adequate blood supply to grow on the ectopic sites, thus the angiogenesis plays an impor tant role in the pathogenesis of endometriosis1. In previous literature, the incidence of abdominal wall endometriosis following the cesarean section is mentioned approximately 0.03% to 0.4%. The incidence is increased by up to 1.08% after hysterotomy2,4,6. Prevalence of iatrogenic scar endometriosis is raised due t o the recent increasing trend of cesarean section7. Patients with cesarean scar endometriosis may present following the several months to years after the surgical intervention. The provisional diagnosis can be made if there is a tender palpable mass seen overlying the cesarean scar which may be associated with the secretion of blood during menstruation4. Case: A 21-year old para 2 woman had a history of cesarean section 2.5 years back, presented to us with a painful swelling on the overlying skin of cesarean wound scar. She noticed the swelling around 1 month back which became tender during her last menstruation with blood stained discharge. Her previous ob stetric course was uneventful, at term she had cesarean section due to fetal distress. On local abdominal examination, a healthy Pfannenstiel scar was seen with a small mass measuring around 1.5x1.5 cm in the middle of the scar (Figure 1). On palpation , the mass was found to be mild tender, firm feeling with restricted mobility. USG and doppler examinations of the abdominal wall soft tissue revealed a heterogeneous hypoechoic mass with echogenic shadow and vascular signals inside the lesion (Figure 2). The lesions showed the involvement of some superficial fibers of the fascia. On the basis of the history, clinical and USG findings the provisional diagnosis was made scar endometriosis. We performed surgical excision of the suspected cesarean scar endometrioma with a 1 cm clear margin (Figure 3A & 3B) . Histopathological analysis revealed endometrial glands and stroma associated with IJ CR: https://escipub.com/international-journal-of-case-reports/ 3N ishat Fatema et al.,IJCR,2020 4:117 evidence of hemorrhage in the fibrous scar (Figure 4A & 4B). Following surgery, we kept her on the combined oral pill for 3 months as adjuvant therapy to avoid recurrence. No recurrence of scar endometriosis was observed during her follow up period. Figure 1: Cesarean section scar with a small mass on overlying skin at the midpoint of the scar Figure 2: Anterior abdominal wall showing an irregular hypoechoic lesion measuring 1.8x1.5cm and color doppler demonstrates Doppler signal inside the lesion

Cesarean scar endometriosis is an uncommon disorder developed due to iatrogenic implantation of endometrial tissues in the cesarean incision site. The implantation theory is not sufficient to explain the pathogenesis of ISE completely. The development of ISE may be related to the hereditary predisposition. Although The occurrence of ISE is commonly reported in Pfannenstiel type incision , the relationship between the CS incision type and the pathogenesis of ISE is not well described in literature1. Surgical scar endometriosis is commonly seen in abdominal skin and subcutaneous tissues in comparison to muscle and fascia. In our case, we found that the endometrial tissues spread up to some superficial fibers of the facia. Other than the cesarean section, the scar endometriosis is observed after the gynecological surgeries also which include hysterectomy, tubal ligation, laparotomy for ectopic pregnancy, salpingectomy and episiotomy3. The patients with cesarean scar endometriosis typically presented with a painful mass or lump on the abdominal wall or at the cesarean scar site. The mass is becoming more tender and sometime may associate with bleeding during menstruation8. Our patient also reported to us with the typical presentation. N ishat Fatema et al., IJCR, 2020 4:117IJ CR: https://escipub.com/international-journal-of-case-reports/ 4 Figure: 3 A & 3B Gross photograph showing wide excision of the mass Figure 4: Histopathology A: 10X view of endometriosis showing endometrial glands and stroma (surrounding purple area) ; B: 40x close up view of the endometri al glands with endometrial stroma associated blood 3A 3B A B IJ CR: https://escipub.com/international-journal-of-case-reports/ 5N ishat Fatema et al.,IJCR,2020 4:117 The diagnosis of scar endometriomas can be challenging. These lesions need to be distinguished from a number of other lesions 9. The differential diagnosis of ISE may include other causes of abdominal wall mass like desmoid tumor, hematoma, fibrosis, suture granuloma, keloid scar, nodular fasciitis, and incisional hernia3,7. After the preliminary clinical diagnosis of ISE, the further evaluation of the mass should be done by various imaging techniques like USG, CT or MRI. Imaging techniques are nonspecific in terms of final diagnosis, which is only achieved in 20-50% of cases, probably due to low clinical suspicion due to the non-specific nature of the symptoms and their late development10. But by these investigation tools the location of the mass, size, volume and local extension can be determined which may be helpful for further management plan11,12. USG and MRI are the most accepted methods for the diagnosis of ISE. The typical USG features of SE includes a hypoechoic echogenic texture with internal scattered hyperechoic echoes8,11. MRI is considered the superior imaging modality for differentiating the glandular and fibrous components of the implanted endometrial tissue in an endometriotic lesion, and to exclude other differential diagnoses, as well as helpful in preoperative management plan 7. We didn’t perform MRI for our patient because our clinical suspicion and ultrasound finding was obvious for endometriosis. In some studies authors suggested that FNAC can be the option for the definitive diagnosis of scar endometriosis prior to surgical intervention. On the contrary, in other studies the authors didn’t encourage to use this technique as a diagnostic tool because with this procedure there is an increased risk of de-novo endometriotic implants at the puncture site, as well as organ injury if the diagnosis is uncertain6,8,11. Wide local surgical excision with 1 cm negative margin is the most recommended treatment modality for the scar endometriosis . To repair the large defect after the excision polypropylene mesh should be used to reinforce the defect for the prevention of incisional hernia2,8. In previous studies authors have suggested that postoperative adjuvant therapy with GNRHa, OCP or aromata se inhibitor may be useful in symptomatic relief and recurrence of endometriosis7. Following surgery, we started combined oral pill for our patient and continued till 3 months. Histopathological analysis of the excised specimen is required for t he Final diagnosis of ISE. The p resence of endometrial tissue can be confirmed if two out of the three following histopathological features are present , endometrial-like glands, spindled endometrial stroma or hemosiderin pigment either within macrophages or in the stroma, Histopathological examination reveals a mixture of different sized glandular structures that are capable of undergoing cyclical changes and. epithelial cells lining the glands can range from cuboidal to columnar with relatively normal cytomorphology and are surrounded by a mucinous and edematous stroma7. Regarding the prevention of Iatrogenic Scar endometriosis, in some studies authors have proposed a variety of recommendations on the basis of the implantation hypothesis. These recommendations include meticulous irrigation and pelvic lavage with saline before the closure of abdominal wall, to use separate sutures, needles, gloves and sponges during uterine and the abdominal wound closure and closure of the parietal and visceral peritoneum13,14.

The frequency of the ISE is raised because of the increasing trend of lower segment cesarean section in modern obstetric practice 1. CS might be a great risk factor for the development of scar endometriosis due to higher exposure of endometrial cells to the subcutaneous tissue IJ CR: https://escipub.com/international-journal-of-case-reports/ 6N ishat Fatema et al.,IJCR,2020 4:117 during the procedure 15. Prevention of decidual cell contamination to the superficial abdominal layers may reduce the occurrence of ISE16. Early diagnosis of ISE and timely surgical intervention and excision of endometrioma is crucial because failure to perform the necessary treatment, or any delay in performing the treatment may have a detrimental effect on the quality of life of the patient8.

1. Zhang P, Sun Y, Zhang C, et al. Cesarean scar endometriosis: Presentation of 198 cases and literature review. BMC Womens Health . 2019;19(1). doi:10.1186/s12905-019-0711-8 2. Tatli F, Gozeneli O, Uyanikoglu H, et al. The clinical characteristics and surgical approach of scar endometriosis: A case series of 14 women. Bosn J Basic Med Sci . 2018;18(3):275-278. doi:10.17305/bjbms.2018.2659 3. Sedhain N, Dangal G, Karki A, et al. Caesarean Scar Endometriosis. J Nepal Health Res Counc . 2018;15(3):292-294. doi:10.3126/jnhrc.v15i3.18859 4. Khanani K. Scar Endometriosis: An Entity Not to Be Forgotten . Vol 67.; 2017. https://ecommons.aku.edu/pakistan_fhs_mc_radi ol. Accessed November 14, 2019. 5. Alnafisah F, Dawa SK, Alalfy S. Skin Endometriosis at the Caesarean Section Scar: A Case Report and Review of the Literature. Cureus. January 2018. doi:10.7759/cureus.2063 6. Khachani I, Filali Adib A, Bezad R. Cesarean Scar Endometriosis: An Uncommon Surgical Complication on the Rise? Case Report and Literature Review. Case Rep Obstet Gynecol . 2017;2017:1-4. doi:10.1155/2017/8062924 7. Kocher M, Hardie A, Schaefer A, McLaren T, Kovacs M. Cesarean-section scar endometrioma: A case report and review of the literature. J Radiol Case Rep . 2017;11(12):8-15. doi:10.3941/jrcr.v11i12.3178 8. Yıldırım D, Tatar C, Doğan O, et al. Post - Cesarean scar endometriosis. Turk Jinekoloji ve Obstet Dern Derg . 2018;15(1):33 -38. doi:10.4274/tjod.90922 9. Francica G, Scarano F. Delayed diagnosis is associated with changes in the cl inical and ultrasound features of subcutaneous endometriosis near cesarean section scars. J Ultrasound. 2009;12(3):101 -106. doi:10.1016/j.jus.2009.05.004 10. Garcí a-Gavilá n M del C, Ferná ndez -Pé rez F, Hinojosa-Guadix J, Gonzá lez -Bá rcenas ML. Rectus abdominal e ndometriosis on cesarean scar. Gastroenterol y Hepatol (English Ed . 2016;39(5):341-343. doi:10.1016/j.gastre.2016.04.001 11. Goel P, Devi L, Tandon R, Saha PK, Dalal A. Scar endometriosis - A series of six patients. Int J Surg. 2011;9(1):39-40. doi:10.1016/j.ijsu.2010.08.003 12. Nigam J, Omhare A, Sharma A. Fine -needle aspiration cytology of a cesarean scar endometriosis. Tzu Chi Med J . 2017;29(4):232 - 234. doi:10.4103/tcmj.tcmj_37_17 13. Akdemir A, Akman L, Yavuzsen HT, Zekioglu O. Clinical features of patients with endometriosis on the cesarean scar. Kaohsiung J Med Sci . 2014;30(10):541-543. doi:10.1016/j.kjms.2013.12.001 14. Ding DC, Hsu S. Scar endometriosis at the site of cesarean section. Taiwan J Obstet Gynecol . 2006;45(3):247-249. doi:10.1016/S1028- 4559(09)60234-5 15. Nominato NS, Prates LFVS, Lauar I, Morais J, Maia L, Geber S. Caesarean section greatly increases risk of scar endometriosis. Eur J Obstet Gynecol Reprod Biol . 2010;152(1):83 -85. doi:10.1016/j.ejogrb.2010.05.001 16. Reddi Rani P, Soundararaghavan S, Rajaram P. Endometriosis in abdominal scars - review of 27 cases. Int J Gynecol Obstet. 1991;36(3):215-218. doi:10.1016/0020-7292(91)90716-I