{"paper_id":"7a304e65-8afa-42b0-83f0-b7f18fc636ad","body_text":"~ 26 ~ \n \nISSN Print: 2664-8393 \nISSN Online: 2664-8407 \nIJGS 2024; 6(1): 26-31 \nwww.gynaecologyjournal.net \nReceived: 22-12-2023 \nAccepted: 28-01-2024 \n \nDr. Sandhyasri Panda  \nDepartment of Obstetrics and \nGynecology, Maharajha’s \nInstitute of Medical Sciences, \nVizainagaram, India \n \nDr. Apurva Ganeshprakash \nGupta \nDepartment of Obstetrics and \nGynecology, Maharajha’s \nInstitute of Medical Sciences, \nVizainagaram, India \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \nCorresponding Author: \nDr. Sandhyasri Panda  \nDepartment of Obstetrics and \nGynecology, Maharajha’s \nInstitute of Medical Sciences, \nVizainagaram, India \n \nA series of caesarean scar endometriosis with \nliterature review \n \nDr. Sandhyasri Panda and Dr. Apurva Ganeshprakash Gupta \n \nDOI: https://doi.org/10.33545/26648393.2024.v6.i1a.28 \n \nAbstract \nIntroduction: Scar endometriosis is a rare form of endometriosis that usually develops in the scar after \nobstetric or gynaecological surgeries. With the increasing caesarean section rates, the frequency of \ncaesarean scar endometriosis (CSE) is expected to rise. The symptoms and signs of scar endometriosis \nmay be ambiguous and hence remains under diagnosed and under reported. \nAim and Objectives: To analyse the cases of CSE and to establish surgery as the mode of treatment. \nMaterials and Methods: This study involves review of records of nine cases with a clinical diagnosis \nof caesarean scar endometriosis. Seven of them received surgical management while two chose medical \nmanagement. Diagnosis was made by clinico-imaging and cyto-histopathology as applicable. \nResults: The mean age of patients is 30.5 years. All subjects had history of caesarean section; they \npresented with cyclical pain at the scar and dysmenorrhea (100%). Five (55.6%) subjects had a \npalpable lump. Seven/nine subjects received wide local excision whereas two opted medical therapy. \nTwo subjects underwent hysterectomy, one in view of recurrence and the other a multiparous women \nwith concurrent AUB. Three subjects were given postoperative medical management in view of margin \npositive histology. \nConclusion: The medical therapy was aimed the symptoms and to suppress the lesion; preferred in \nsmaller endometriomas, desirous of future child bearing and in margin positive postoperative cases. \nHowever, surgical excision, which is both diagnostic and curative, remains the most effective treatment \nfor scar endometriosis.  \n \nKeywords: Caesarean scar endometriosis, wide local excision, medical management \n \nIntroduction \nEndometriosis is defined as the presence or growth of functional ectopic endometrial tissue \n[1]. Ectopic tissue responds to ovarian hormonal stimulation and tends to proliferate when \nstimulated by cyclic estrogens, thus, appearing ''to menstruate'' [2]. It affects 89 million \nwomen of reproductive age worldwide, approximately 5 to 10% of all women [3]. The ovary \nand pelvic peritoneum are the two most common pelvic locations for endometriotic tissue. \nThe gastrointestinal tract, urinary tract, and respiratory system are extra-pelvic sites [4-6]. One \nof the rare type of extrapelvic endometriosis is abdominal wall endometriosis and its \nincidence varies from 0.03 to 3.5% [7]. AWE can be superficial (affecting only subcutaneous \ntissue above the fascia), intermediate (infiltrating the rectus sheath), or deep (affecting rectus \nmuscles) depending on the layers involved [8]. \nScar endometriosis refers to the development of abdominal wall endometriosis (AWE) at the \nsurgical incision site following obstetric or gynecological surgeries [9]. The majority of scar \nendometriosis cases have been reported following procedures such as cesarean section, \nhysterotomy, hysterectomy, episiotomy, tubal ligations, and a few following myomectomy, \nappendicectomy, in the laparoscopic trocar tract, and amniocentesis needle tract [10, 11] . \nCaesarean Scar Endometriosis (CSE) is the most commonly reported form of AWE [12].  \nThe actual incidence is difficult to determine because it is under diagnosed and under \nreported. Meyer reported the first case in 1903(13). Scar endometriosis incidence is reported \nto be 0.03-0.4% after caesarean section [7], and 1.1% after mid-trimester abortion [10]. The \nprevalence of concomitant pelvic endometriosis and scar endometriosis has been estimated to \nbe 14.3-26% [14]. \nScar endometriosis is expected to become more common as Caesarean section rates increase \n[15]. \nInternational Journal of Gynaecology Sciences  2024;  6(1): 26-31 \n \n\n\n \n~ 27 ~ \nInternational Journal of Gynaecology Sciences https://www.gynaecologyjournal.net \n \nSome refer to AWE as the \"iatrogenic\" subtype of \nendometriosis; however, the mechanism remains \nundetermined [16]. Various theories have been proposed \nregarding the development of scar endometriosis. The direct \nimplantation theory states that during surgery, endometrial \ncells scattered around the abdominal wall proliferate or \ncause metaplasia of the surrounding fascia due to hormones. \nThe second theory proposes that endometrial cells spread \nvia lymphatic or hematogenous routes [10]. The third theory \nholds that pluripotent mesenchymal cells differentiate to \nform endometriosis [17]. Scar endometriosis may present \nwith ambiguous symptoms and sign [9]. Patients typically \nreport a triad of complaints including cyclic pain and \nswelling at the incision site with history of obstetric and \ngynaecological surgery [7]. A clinical examination may \nreveal a painful and palpable nodule with maximum \ntenderness over the scar during menstruation. Only half of \nthem have classic symptoms and can be misdiagnosed as an \nincision hernia, abscess, hematoma, lipoma, carcinoma \n(primary), granuloma, lymphoma, neuroma, cyst, abscess, or \nsoft tissue sarcoma [18]. This causes a significant delay in \ndiagnosing and initiating treatment. It is hypothesized that \nobesity-related suboptimal closure of uterine incision or \nabdominal layers contributes to the development of AWE. \nPelvic endometriosis increases the risk of developing AWE \n[15]. \nUltrasound aids in the diagnosis of subcutaneous lesions by \ndetecting the presence of a hypoechoic mass, which may \noccasionally reveal internal hyperechoic areas [19]. It also \nhelps to rule out other possibilities, such as an abdominal \nwall hernia. Doppler may reveal increased vascularity. An \nMRI is required to determine the extent of the lesion beyond \nthe subcutaneous plane and to identify any intra abdominal \nextension. FNAC may help distinguish between other \ndiagnoses and metastatic disease, but it may be harmful in \nthe case of abdominal wall hernia. FNAC helps to rule out \nmalignant transformation of abdominal wall endometriosis \n[20]. Ectopic endometrial glands with cellular stroma, \nextravasated erythrocytes, and inflammatory infiltrate are \ncommon histological findings. However, fibrosis in the \nendometriotic tissue may lead to an inconclusive diagnosis \n[13]. \nMalignant change in abdominal wall endometriosis is \nextremely rare, with an estimated incidence of 0.3% to 1% \n[21]. Advanced age, postmenopausal status, and lesion \ndiameter of more than 9 cm are all risk factors. Malignancy \nshould be suspected in cases of multiple recurrences, failure \nto respond to treatment, and sudden rapid growth. Clear cell \ncarcinoma is the most common histological subtype [15]. \n Medical treatment with combined oral contraceptives \n(COCs), progestins, androgens, or gonadotropin-releasing \nhormone (GnRH) analogues temporarily relieves symptoms, \nbut they recur after the therapy is discontinued [18]. Surgery \nis frequently required for both treatment and definitive \ndiagnosis, with a 95% success rate [15]. \nThe purpose of this study is to examine the clinical aspects \nof caesarean scar endometriosis, to correlate clinico-imaging \nwith histopathology findings and to establish wide local \nexcision as the treatment of choice. \n \nMaterials and Methods \nIn this study, prospectively maintained case profiles of nine \ncases between 2015 and 2021 with a clinical diagnosis of \ncaesarean site scar endometriosis were reviewed. Seven of \nthem received surgical management, which included a wide \nlocal excision, while two were given medical management. \nA detailed history about symptoms at presentation, \nindication and the stage of labor when caesarean delivery \nwas conducted, examination findings, ultrasonography \nfindings, and surgical notes describing the lesion were \nrecorded. Any information about postoperative medical \nmanagement was obtained. CSE is confirmed by the \npresence of endometrial tissue, focal hemorrhage, \nhemosiderin deposits and/or fibrosis on histopathology \nreports. Patients were followed up for two years. \n \nResults \nThe mean age of patients is 30.5 years ranging between 26 \nto 36 years. Six subjects are para 2 with a history of two \nprevious LSCS, while the remaining three are para1 with \none previous LSCS. The mean duration of symptoms is 10 \nmonths, with a range of 6 months to 24 months. All nine \nsubjects reported cyclical pain at the scar site (100%) along \nwith dysmenorrhea (100%). Five subjects (55.6%) had \npalpable solitary lump at the scar site. Clinically \ninconspicuous abdominal wall endometriomas in four \nsubjects were later supported by ultrasonography. Itching \nwas an accompanying complaint in two of them (22.22%). \nThe mean interval between caesarean section and diagnosis \nof abdominal wall endometriosis is 7.8 years, with a range \nof 3 to 14 years. Six subjects had a subumbilical midline \nscar (66.66%), while the other three subjects had \npfannensteil scar (33.33%). The majority of the pathology \noccurred on the right side (88.88%) of the incision followed \nby left side (11.11%). All subjects underwent \nultrasonography to aid in the diagnosis. It revealed the \npresence of a hypoechoic lesion with heterogenic spots or \ncystic lesions in the anterior abdominal wall. The mean \nlump size based on USG is 4.81 +/- 1.1 cm and \nintraoperatively is 4.43 +/- 1.1 cm, with fibrosis of adjacent \ntissue. Amongst the operated cases, there are four cases of \nsuperficial scar endometriosis (57.71%), which affects only \nthe skin and subcutaneous layers, and three intermediate \ncases that involve the rectus sheath (42.85%) as well, while \nnone had deep CSE i.e. none showed involvement of the \nrectus muscle or peritoneum (Table 1). Hence, it indicates \nthe endometrioma of scar invades from superficial to deep \nlayers. None of the subjects demonstrated signs of \nconcurrent pelvic endometriosis. FNAC was performed in 3 \nout of 9 subjects, with one yielding inconclusive results and \nthe other two revealing the presence of endometrial glands \nand stroma, thus confirming the diagnosis. As first-line \nmanagement, seven of nine subjects underwent wide local \nexcision with a 1cm free margin (Figure 1). In light of \nextensive intraoperative fibrosis and margin positive \nhistology reports, three patients received postoperative \nDMPA (depot medroxyprogesterone acetate) 150mg single \ndose, followed by continuous dienogest 2mg OD orally or \nCOC for three months in two of them respectively. These \nthree did not show any relapse hence confirming cure. A \n26year old, P1L1A1 conceived after 2 year of excision but \nhad a spontaneous abortion at third month of gestation. One \ncase underwent WLE along with TAH + BSO as first line \nmanagement due to concurrent complaints of AUB and \ncompletion of childbearing (Figure 2). Only one subject \n(No. 6) experienced recurrence after 10 months of wide \nlocal excision. The subject also had an incisional hernia. As \na result, a repeat excision was performed, along with a total \n\n \n~ 28 ~ \nInternational Journal of Gynaecology Sciences https://www.gynaecologyjournal.net \n \nabdominal hysterectomy and incisional hernia repair with \nmesh. The most likely cause of recurrence in this subject \ncould be due to incomplete excision of an extensive fibrosis, \nobesity with BMI (>35kg/m 2); she also presented with one \nand half years of ammenorhea, and resumed menstruation \nimmediately following the primary excision of the mass. \nTwo subjects with relatively small endometriomas denied \nsurgical treatment. One subject was treated with continuous \nCOC for three cycles and the other was given only \nsymptomatic treatment with NSAIDS based on surgeon \npreference. Both reported symptom relief during two year \nfollow-up period. \nHistopathological diagnosis is confirmed by the presence of \nendometrial glands and stroma, focal hemorrhage, \nhemosiderin deposits and/or fibrosis as shown in figure 3. \nHence, clinico-imaging diagnosis of CSE was proved to be \n100% accurate by histopathology reporting in all of the \nsurgically treated subjects.  \n \nTable 1: Caesarean scar endometriosis (CSE): Demographic details, symptoms and management \n \nSubject \nno. \nAge \n(in \nyears) \nParity No. of \nLSCS \nLCB \n(in \nyears) \nC/F \nType \nof \nscar \nSide \nSize of \nmass \n(in cm) \nUSG \nIntraop \nFNAC Location Preoperative \ndiagnosis \nManagement \n1st line Follow up Cyclical \npain Lump \n1 26 P1L1 \nA1 1 3 + + SUM Rt 5X5 6x5 - Subcutaneous CSE WLE \nDMPA f/b \ndienogest \nfor 3cycles \n2 28 P1L1 1 8 + - SUM Rt 2.6x2 3x4 - Subcutaneous \n+ RS CSE WLE - \n3 28 P2L2 2 8 + - SUM Rt 3x3 3.5x \n2.5 Inconclusive Subcutaneous \n+ RS CSE WLE \nDMPA f/b \nCOC for \n3cycles \n4 35 P2L2 2 6 + + Pf Rt 6.6x5 6x5 - Subcutaneous \n+ RS CSE WLE DMPA \n5 28 P1L1 1 3 + + SUM Rt 4x5 5x5 \nEndometrial \nglands and \nstroma + \nSubcutaneous CSE WLE - \n6 32 P2L3 2 13 + + SUM Lt 5x5 6x6 \nEndometrial \nglands and \nstroma + \nSubcutaneous CSE WLE - \n + + - Lt 3.5x \n3.2 4x3 - Subcutaneous \n+ RS \nRecurr-ence \nafter 10 \nmonths postop \nDMPA \n150mg \nx 2doses \nWLE \n+ TAH + \nIncisional \nhernia mesh \nrepair \n7 36 P2L2 2 14 + - SUM Rt 4.4x5 5x5 - Subcutaneous CSE \nWLE \n+ TAH \n+ BSO \n- \n8 30 P2l2 2 5 + - Pf Rt 1.2x \n1.2 - - - CSE COC x \n3cycles \nSymptom \nrelief + \n9 32 P2L2 2 7 + + Pf Rt 2.9x \n1.2 - - - CSE NSAIDS Symptom \nrelief + \nC/F: Clinical features; SUM: Subumbilical midline; Pf: Pfannensteil; Rt: Right; Lt: Left; RS: Rectus sheath; CSE: Caesarean s car \nendometriosis; WLE: Wide local excision; DMPA: Depot Medroxyprogesterone acetate; COC: Combined oral contraceptive; TAH: Total \nabdominal hysterectomy; BSO: Bilateral salphigoopherectomy  \n \n \n \nFig 1: [a] Surgical exploration showing margins and extension of caesarean scar site endometrioma and [b] postoperative imaging of \nresected mass in respective subjects, with skin excision in case 6 \n\n\n \n~ 29 ~ \nInternational Journal of Gynaecology Sciences https://www.gynaecologyjournal.net \n \n \n \nFig 2: [a] TAH with BSO specimen [b] excised mass from abdominal wall layers \n \n \n \nFig 3: Hematoxylin and eosin (H&E) stained section of excised tissue displaying endometrial glands (arrow head) and stroma (arrow) in [a], \n[b], and [c], with fibrosis (star) in [b] and hemorrhage (star) in [c]. Figure [d] shows proliferating capillaries (arrow head) and hemosedrin-\nladen macrophages (arrow) \n \nDiscussion \nCSE presents in reproductive age with mean age in our \nstudy being 30.5years while that in the study by Faik Tatli et \nal was 32.71 +/- 8.61 years, Piriyev et al as 36 +/- 5.4 years, \nSumathy et al was 35.19 +/- 6.72 years [22-24]. \nIt is important to note that a higher incidence is reported \nfollowing early hysterotomy (end of second or beginning of \nthird trimester), as early decidua appear to have more \npluripotential capabilities, which can lead to increased \ncellular replication and endometriosis [25]. In theory, \npregnancy, an altered immune response, and a caesarean \nsection could increase the risk of developing endometriosis \n[26]. \nBased on our observation and literature review it can be \nhypothesized that decidua of early gestational age and at the \nsame time elective caesarean or a caesarean section \nperformed at early stage of labor has higher incidence of \nCSE; one of the contributing factor might be elective \ncaesarean at earlier gestational age, as there is an increasing \ntrend of caesarean section at maternal request. Some authors \nemphasized that having two caesarean sections did not \nincrease the risk of being diagnosed with endometriosis \nwhen compared to having only one [26]. \nVellido-Costelo et al . reported that there appear to be no \nlink between pelvic and scar endometriosis development. In \ntheir study, 14% of patients had associated pelvic \nendometriosis, which is the incidence in the general \npopulation [27]. \nStudies by D.Poudel et al and Sumathy et al demonstrated \npresence of the endometrioma more to the left side on \nincisions unlike our study that showed more involvement on \nthe right side [1, 24] . This right sided dominance was \nsupported by Akbulut S et al [28]. \nIn a series of 12 patients by Franciaca et al, USG and color \nDoppler significantly contributed to the correct preoperative \ndiagnosis, and the authors suggest that sonographic and \ncolor Doppler, when combined with clinical data, may \nsignificantly contribute to the preoperative diagnosis [29]. \nOlder studies published a reporting in which FNAC was not \ndiagnostic in any of the cases who underwent the procedure \nand use of this technique is debatable, as some authors have \nwarned about the increased risk of producing new \nendometriotic implants at the puncture site, as well as \nvisceral injury if the diagnosis is uncertain [25]. Whereas \nmore recent studies by Sujaya Mazumder et al. and Katwal \net al  based on their experiences have argued that it is a \nquick, cost-effective, and accurate diagnostic tool to include \nin patients' management [13, 30]  because of the practice of \nultrasound guided FNAC. They supported the use of this \ntechnique to provide a tissue diagnosis prior to surgery and \nis also helpful in cases where the origin of the mass is \nuncertain. Vellido-Costelo et al. found that 52% of patients \nhad a FNAC diagnosis prior to surgery, and one of them was \ndiagnosed with cancer using this method, which led to a \ndifferent therapeutic management [27].  \n\n\n \n~ 30 ~ \nInternational Journal of Gynaecology Sciences https://www.gynaecologyjournal.net \n \n Ucar et al. reported no recurrences over a follow-up period \nof 12-60 months [31]. Horton et al. in a series of 445 cases \nfound a 4.3% recurrence rate [32], Zhang and Liu reported a \n7.8% recurrence over an average of 20 (±16) months in a \nseries of 151 cases [33] while we had 14.28% (1/7 cases) \nrecurrence rate over 24 months follow up period. \nMedical treatment with progestogens, combined oral \ncontraceptives, and danazol is ineffective in treatment of \nCSE and only provides partial relief from symptoms. \nRecently, there has been a report of the use of a \ngonadotrophin agonist, but only with immediate \nimprovement in symptoms and no change in lesion size. \nThese patients must be monitored because of the possibility \nof recurrence, which necessitates excision [3]. Surgical \nresection of the scar endometrioma remains the primary \ntreatment option, even if the disease recurs. Because of the \npotential for recurrence (4.3% after surgery) [32] and \nmalignant degeneration (0.3-1%) (21) of this condition, a \nlocal wide excision with at least a 1 cm resection margin is \nat present regarded as the best clinical practice. However, no \nstudies have so far evaluated whether the surgical margin \nwidth affects the probability of recurrence rate. \nTo avoid endometrial inoculation after a caesarean section, \nit's important to clean the abdominal wound thoroughly, \nespecially at the corners and on the surgeon's side which has \nbeen described in the older studies as well. It has also been \nsuggested that at the end of surgery, particularly on the \nuterus and tubes, the abdominal wall wound be thoroughly \ncleaned by giving peritoneal wash or irrigated vigorously \nwith a high jet solution before closure [1, 2, 34) . Other studies \nsuggested that the absence of closure of the parietal and \nvisceral peritoneum could significantly increase the risk of \nendometriosis in the skin incision scar [35]. Finally, \ninstrument and needle replacement when suturing more \nsuperficial abdominal layers to avoid iatrogenic inoculation \nof endometrial cells is recommended [7, 34]. \n \nConclusion \nDespite the high probability of CSE due to rapidly \nincreasing rate of abdominal birth and cut short technique of \nuterine and abdominal closure, still the diagnosis of scar \nendometriosis requires a high level of suspicion. It should be \nconsidered in all reproductive-age women with prior history \nof uterine surgery. They commonly present with cyclical \npain and lump at the scar site. Clinico-imaging diagnosis \nwas found highly specific as they correlated on \nhistopathology. Medical therapy was aimed at both \nsymptom relief and lesion suppression, with a preference in \nsmall endometriomas, in those who wish further child \nbearing and in margin positive postoperative cases. \nHowever, surgical excision, which is both diagnostic and \ncurative, remains the most effective treatment for scar \nendometriosis. \n \nAcknowledgments \nThere is no funding received for the study. We acknowledge \nMIMS management and the academic cell of the institute \nfor approving the study. We acknowledge the faculties and \npostgraduates of the department, the department of \nPathology for their cooperation. We are thankful to the \npatients for their confidence and consent; we declare no \nconflict of interest related to this work. \n \n \nReferences \n1. Poudel D, Acharya K, Dahal S, Adhikari A. A case of \nscar endometriosis in cesarean scar: A rare case report. \nInt J Surg Case Rep. 2023 Jan 1;102:107852. \n2. 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