{"paper_id":"bbe3de7c-a886-47cf-ae53-7dd148c7c023","body_text":"Received\n 05/17/2024 \nReview began\n 05/28/2024 \nReview ended\n 07/29/2024 \nPublished\n 08/05/2024\n© Copyright \n2024\nZhang et al. This is an open access article\ndistributed under the terms of the Creative\nCommons Attribution License CC-BY 4.0.,\nwhich permits unrestricted use, distribution,\nand reproduction in any medium, provided\nthe original author and source are credited.\nDOI:\n 10.7759/cureus.66223\nAbdominal Wall Endometriosis: A Case Report\nand Literature Review of Pfannenstiel Incision\nEndometrioma\nNathan Zhang \n, \nSedona Robrahn \n, \nKatherine R. Thornburgh \n, \nJustin Moon \n, \nMuhammad K. Ather \n,\nColton P. Boney \n, \nJoel A. Yalowitz \n1.\n Medicine, Alabama College of Osteopathic Medicine, Dothan, USA \n2.\n Research, Alabama College of Osteopathic\nMedicine, Dothan, USA \n3.\n Radiology, Decatur Morgan Hospital, Decatur, USA\nCorresponding author: \nJoel A. Yalowitz, \njyalowit@gmail.com\nAbstract\nWe depict a unique case of a 39-year-old woman who presented to the emergency department with\ncomplaints of right upper quadrant pain. Work-up and a computed tomography (CT) scan revealed acute\ncholecystitis and the patient underwent laparoscopic cholecystectomy without complication. At this time, an\nincidental mass was discovered in the subcutaneous fat adjacent to the abdominal wall. The patient returned\nsix months later with progressive, cyclic abdominal pain since her last hospital admission. Initial admission\nlab work was within normal limits and a urine pregnancy test was negative. Physical exam revealed\ntenderness around her previous cesarean section scar. Repeat CT revealed an enlarging, spiculated mass\nadherent to the abdominal wall. After imaging confirmation, the patient underwent complete open surgical\nexcision for the removal of the mass. Post-surgical biopsy confirmed endometrial gland and stroma\nconsistent with abdominal wall endometrioma. The patient was discharged with adjuvant therapy and\nrecommended follow-up with the surgeon and her obstetrician-gynecologist. The radiological diagnosis,\nguidelines, and decision-making for initiating interventional treatment are discussed in this report. Our\npurpose in documenting this case is to present a rare diagnosis of an atypical location for an endometrioma\non the abdominal wall, in a patient with prior cesarean delivery. Although this patient was treated with open\nexcision, different interventional radiology treatments from radiofrequency ablation and focused ultrasound\nwere discussed. In doing so, we hope to contribute to the systematic literature review on surgical excision as\na treatment option for Pfannenstiel incision endometrioma.\nCategories:\n Obstetrics/Gynecology, Radiology, General Surgery\nKeywords:\n ct imaging, abdominal-wall endometriosis, endometriosis excision, scar site endometriosis, tumor\nimaging, ob-gyn, scar endometrioma, open excision, minimally invasive interventional radiology, general radiology\nIntroduction\nEndometriosis is a disease that affects around 10-15% of reproductive-age women and is defined as\nfunctional endometrial stroma and glands located outside of the uterus \n[1]\n. Symptoms include infertility,\nchronic pain, dysuria, dyschezia, dysmenorrhea, and dyspareunia \n[1]\n. The most common locations of\nendometriosis include the pelvis, specifically the ovaries; however, it can also involve the fallopian tubes,\nuterosacral ligaments, or surrounding peritoneum. Less common locations include the urinary bladder,\ngastrointestinal tract, or soft tissues, such as the cervix, vagina, vulva, and abdominal wall \n[2]\n. \nAlthough there are many theories about the cause of endometriosis, the most accepted is retrograde\nmenstruation during a menstrual period. Blood travels out of the fallopian tubes and into the pelvis, causing\nendometrial lesions including endometriomas. Endometriomas, known colloquially as “chocolate cysts”, are\nformed when ectopic endometrial tissue, glands, and stroma bleed in response to normal hormonal\nsignaling. This hormonal bleeding may result in a hematoma that is lined by fibrous tissue, forming a cystic\nlesion \n[3]\n. Endometriomas are associated with more severe disease and are present in 17-44% of women with\nendometriosis \n[3]\n. Endometriomas can occur in many locations, most commonly in the pelvic region.\nHowever, endometriomas can appear atypically in extra-pelvic locations. They are most commonly located\non the ovaries but have also been found in the bowel as well as in prior surgical incisions \n[3]\n. \nOne such example of an atypical location of endometrioma is the Pfannenstiel incision scar tissue. The\nPfannenstiel incision is the preferred approach for cesarean delivery and is a slightly curved incision located\napproximately 2-3 cm above the symphysis pubis \n[4]\n. When endometrial tissue seeding has taken place at the\nsite of a surgical scar, it is then termed as “incisional endometriosis” \n[5]\n. While rare, the occurrence of\nendometriosis in a Pfannenstiel incision post-cesarean section has been reported with multiple studies\nindicating an infrequent incidence of no more than 1% of all patients who have undergone a cesarean\nsection \n[4,5]\n. One proposed mechanism suggests that incisional endometriosis could be primarily due to the\nsurgical displacement of tissue accentuated by normal physiologic changes in the post-partum period. These\ninclude the hyper-estrogen state following obstetrical delivery and the vasogenic and irritative changes from\nvascular growth factors, inflammatory cytokines, and weakened cellular immunity \n[6]\n. Clinical suspicion of\n1\n2\n1\n1\n1\n1\n3\n \nOpen Access Case Report\nPublished via Alabama College of\nOsteopathic Medicine Research\nHow to cite this article\nZhang N, Robrahn S, Thornburgh K R, et al. (August 05, 2024) Abdominal Wall Endometriosis: A Case Report and Literature Review of\nPfannenstiel Incision Endometrioma. Cureus 16(8): e66223. \nDOI 10.7759/cureus.66223\n\nPfannenstiel endometrioma increases with dysmenorrhea, a cyclic, abdominopelvic pain along with an\nassociated superficial mass. However, this diagnosis is difficult to make clinically as the symptomatology\noverlaps with many other common gastrointestinal and obstetric pathologies.\nFor the best imaging method, experts agree that initial abdominal ultrasound (AUS) is preferred with\nsequential computed tomography (CT), or magnetic resonance imaging (MRI) depending on the sonographic\narchitectural findings \n[7,8]\n. While imaging is the current mainstay of guiding clinical management,\ndefinitive diagnosis and treatment occur through excisional biopsy and histological analysis, which often\ndoes not occur due to the relatively low incidence and often, asymptomatic presentation of smaller\nendometriomas \n[5,7]\n. In this report, we explore a case of a young woman with Pfannenstiel endometrioma\nto better understand the role of imaging in atypical endometrioma diagnosis and treatment.\nCase Presentation\nA 39-year-old gravida 2, para 2 female presented to the hospital with progressively worsening right upper\nquadrant abdominal pain since yesterday evening after dinner. She had extreme difficulty sleeping and was\nunable to keep down her food. She had attempted to control the pain with over-the-counter analgesics but\nultimately decided to visit the Emergency Department (ED) after she developed a low-grade fever and an\nepisode of vomiting later the next morning. She had a history of well-controlled migraine headaches, major\ndepressive disorder, Bipolar I, and attention deficit hyperactivity disorder. She had no significant surgical\nhistory aside from one cesarean section via Pfannenstiel incision and a laparoscopic gastric bypass. She was\nseveral months postpartum and had no difficulty with postpartum care. She has no history of illicit drug use\nand is a non-smoker and non-drinker. Home medications included propranolol, bupropion, lamotrigine, and\ndextroamphetamine-amphetamine (Adderall©) for her psychiatric comorbidities. She denied any recent\nillness, upper respiratory symptoms, dietary changes, or sick contacts. The reports that her diet consisted\nprimarily of processed food and her body mass index (BMI) was 34.3. The patient was later diagnosed with\nacute cholecystitis and the surgeon on call was able to remove the gallbladder laparoscopically with no\ncomplications. As part of the surgical workup, a computed tomography (CT) scan revealed a well-\ncircumscribed, soft tissue mass near the inferior rectus abdominis muscle (Figures \n1\n, \n2\n).\nFIGURE\n 1: Axial CT abdomen view of the incidental 1.8 cm nodule. The\nnodule is well-circumscribed with homogeneous soft-tissue density and\nmargins.\n \nPublished via Alabama College of\nOsteopathic Medicine Research\n2024 Zhang et al. Cureus 16(8): e66223. DOI 10.7759/cureus.66223\n2\n of \n9\n\nFIGURE\n 2: Sagittal CT abdomen view of the incidental 1.8 cm nodule.\nThe nodule is well-circumscribed with homogeneous soft-tissue density\nand margins.\nThrough shared decision-making, the patient agreed to defer any intervention for the nodule and to proceed\nwith watchful waiting. This decision was made jointly due to the location and small size (<2 cm) of the\nnodule, as well as the lack of lower quadrant abdominal pain or mass-effect symptoms.\nFive months later, the patient returned to the hospital with another episode of abdominal pain. The patient\ndenied any significant trauma, sick contacts, or dietary changes since she was last seen in the hospital\nmonths prior. She stated the pain had been dull and progressive, and poorly localized with diffuse\ndiscomfort around the right lower quadrant. She also stated that the pain had worsened recently, and often\noccurred during her menses and was worried about permanent complications to her future fertility. On\nfurther history, the patient denied any menorrhagia, melena, or hematochezia and stated her menses\noccurred monthly and usually lasted 4-5 days with moderate flow. Initial lab work included a complete blood\ncount (CBC), comprehensive metabolic panel (CMP), and quantitative beta-hCG to assess for ectopic\npregnancy. Admission blood work and vital signs were grossly within normal limits (Table \n1\n). \n \nPublished via Alabama College of\nOsteopathic Medicine Research\n2024 Zhang et al. Cureus 16(8): e66223. DOI 10.7759/cureus.66223\n3\n of \n9\n\nLab\nPatient value\nNormal value\nWhite blood cells\n7.7 x 10\n9\n/L\n4.5-11 x 10\n9\n/L\nRed blood Cells\n4.5 million/mm\n3\n3.5-5.5 million/mm\n3\nPlatelets\n238,000 /mm\n3\n150,000 - 400,000 /mm\n3\nNeutrophils\n61.0%\n54-62%\nEosinophils\n2.6%\n1-3%\nLymphocytes\n27.9%\n25-33%\nBUN\n13 mg/dl\n7-18 mg/dl\nCreatinine\n0.7 mg/dl\n0.6-1.2 mg/dl\nHemoglobin\n13.0 g/dl\n12-16 g/dl\nHematocrit\n40.8%\n36 - 46%\nAlanine aminotransferase (ALT)\n27 U/mL\n10-40 U/L\nAspartate aminotransferase (AST)\n18 U/L\n12-38 U/L\nAlkaline phosphatase\n57 U/L\n25-100 U/L\nAlbumin\n4.4 g/dl\n3.5-5.5 g/dl\nBilirubin, Total\n0.8 mg/dl\n0.1-1.0 mg/dl\nBeta-human chorionic gonadotropin  (β-hCG)\n<3.4 IU/L\n<5 IU/L for  non-pregnant \nTABLE\n 1: Admission lab work obtained on return hospital visit including complete blood count,\ncomprehensive metabolic panel, liver function tests, and beta-human chorionic gonadotropin\nBUN: Blood urea nitrogen\nOn physical exam, the lower mid-abdomen was tender upon palpation near the site of the prior cesarean scar\ntissue. An abdominal CT was ordered to assess for a significant tumor mass-effect or possible deep abscess.\nImaging revealed a cystic, soft-tissue mass in the subcutaneous tissue just posterior and deep to the\nabdominal muscles (Figures \n3\n, \n4\n).\n \nPublished via Alabama College of\nOsteopathic Medicine Research\n2024 Zhang et al. Cureus 16(8): e66223. DOI 10.7759/cureus.66223\n4\n of \n9\n\nFIGURE\n 3: Axial CT abdomen view of the enlarging cystic mass that is\nnow 6.7cm x 3.6cm. The nodule has irregular margins with mild\nspiculations and is directly adherent to the fibrotic, Pfannenstiel scar\ntissue.\n \nPublished via Alabama College of\nOsteopathic Medicine Research\n2024 Zhang et al. Cureus 16(8): e66223. DOI 10.7759/cureus.66223\n5\n of \n9\n\nFIGURE\n 4: Sagittal CT abdomen view of the enlarging cystic mass that\nis now 6.7cm x 3.6cm. The nodule has irregular margins with mild\nspiculations and is directly adherent to the fibrotic, Pfannenstiel scar\ntissue.\nThe following day, the patient was admitted to the inpatient floor, Interventional radiology was consulted\ndue to the abnormal CT findings on the day prior. Although there was high clinical suspicion of incisional\nendometriosis, hormonal contraception treatment, usually the first line for symptomatic relief of\nendometriosis, was deferred for multiple reasons. Open laparotomy, when compared to both laparoscopic\nsurgery and contraception, was preferred due to the large size and well-localized seeding into the\nsuperficial, subcutaneous tissue. The patient's above-average BMI and large size of the endometrioma in\nparticular made it difficult for an ultrasonographic ablation approach. As a result, the Interventional\nRadiologist discussed with General Surgery who then opted for open surgical intervention. This decision was\ndeemed more appropriate than symptomatic or conservative management (combined contraceptives) due to\nher ongoing attempts to conceive and her history of migraines. For similar reasons, marginal excision of the\nendometrioma was also chosen over definitive hysterectomy to preserve future fertility. These decisions\nwere reinforced and supported by current radiologic and obstetric literature, discussed below.\nDuring the excisional biopsy, a standard cautery-dissection 15-blade was used for the well-defined, palpable\nmass. No significant spillage or bleeding occurred, and complete, circumferential excision was performed to\nseparate the mass from the underlying fat and muscle. Samples of the resected mass were then sent to\nsurgical pathology which described scanty smooth muscle stroma and active glandular tissue definitive for\ntypical endometriosis. The patient was then discharged with minimal complications. As part of the post-\noperative care, the patient was given Ketorolac and other non-steroidal anti-inflammatory drugs (NSAIDs)\nfor pain control and discharged later that week. The patient was informed of possible recurrence and\nstandard post-operative suppressive hormonal therapy but remained firm on her desire for immediate\npregnancy. She was instructed to avoid sexual intercourse but was assured that her fertility would return to\nnormal function within the next two months. Her two-week follow-up was without complications, and she is\n \nPublished via Alabama College of\nOsteopathic Medicine Research\n2024 Zhang et al. Cureus 16(8): e66223. DOI 10.7759/cureus.66223\n6\n of \n9\n\nscheduled to follow up with her OB/GYN later this year and has not since reported any abdominal symptoms\nor pain.\nDiscussion\nAWE includes cesarean scar endometriosis (CSE), a type of incisional endometriosis. Incisional\nendometriosis also has occurred during other gynecologic procedures, such as hysterectomies, episiotomies,\nsalpingostomies, and laparoscopic surgeries \n[5]\n. CSE has a reported incidence of 0.03-0.45% and is best\nexplained by the direct inoculation theory, where endometrial tissue is transferred directly into the incision\nduring surgery \n[9]\n. The hypothesized mechanism has also been proposed to include irritative and vasogenic\nchanges following delivery \n[6]\n. Symptoms of CSE include a palpable abdominal mass, dysmenorrhea, and\ncyclical pain. The duration between cesarean section and the onset of symptoms was previously found to be\na mean of 28 months with a standard deviation of 25 months \n[9]\n. Our patient experienced an onset of\nsymptoms several months postpartum, which is less than the mean onset of symptoms reported in some\nstudies \n[1,8,10]\n. One possibility is the association of obesity with more advanced endometriosis due to a\nstrong association from elevated estrogen levels in excessive adipose tissue as endometriosis is an estrogen-\ndependent disease \n[10]\n. It is highly likely that the patient's clinical obesity (BMI of 34) contributed to her\nrapid mass progression and earlier onset of symptoms. Counseling the patient about lifestyle modifications\nfor weight loss may have reduced progression of the disease. \nOne important shift in recent years is the increase in the incidence of AWE seen due to higher rates of\nelective cesarean sections and gynecological laparoscopic surgeries in recent decades \n[4,5,7]\n. There is\nongoing debate in the literature and it is well documented that this upward trend contributes to current\nstandards and indications for elective cesarean treatment. The Pfannenstiel incision, the preferred approach\nto cesarean section, is a low, transverse incision located approximately 2-3 cm above the symphysis pubis. It\nhas succeeded as the new standard over vertical incision due to cosmetic reasons, reduced postoperative\npain, reduced risk of incisional hernia, and easier repair \n[11]\n. Alternatively, the Pfannenstiel incision has\nwider surgical dissection planes which may be more easily inoculated with endometrial tissue \n[12]\n. Many\nmethods have been proposed to reduce CSE in Pfannenstiel incisions, including thorough irrigation and\ncareful instrumentation \n[9]\n. Despite recommendations, it is possible that these methods were not\ncompletely utilized in our patient in the past, resulting in a higher risk and subsequent CSE in our patient.\nIn addition to a cesarean section, other risk factors for incisional endometriosis include heavy menstrual\nflow and alcohol consumption \n[13]\n which our patient did not report. As the prior cesarean section was a\nmajor factor and due to the location of the endometrioma, future consideration of proper surgical\ntechniques may reduce the seeding of endometrial tissue into the incision and CSE risk. This may not be as\nconcerning in smaller endometriomas or those treated appropriately with high-intensity focused ultrasound\n(HIFU) ablation.\nSuspected CSE is typically evaluated initially with ultrasound, followed by CT or MRI \n[5,6]\n. Due to the\npatient’s initial presentation of right upper quadrant pain from acute cholecystitis, this mass was\nincidentally discovered with CT upon surgical workup without a prior ultrasound of the lower abdomen.\nOfficial diagnosis can only be made with histopathological findings, by surgical excision of the mass or fine\nneedle aspiration \n[6]\n. HIFU and surgical excision have both been shown to be safe and effective treatments\nfor AWE. There are no differences in complication rate, recurrence of AWE, and pain levels on follow-up\nbetween the two treatments, although more research is needed comparing the two \n[14]\n. As mentioned\nabove, there is a possibility that the size limitation and smaller endometriomas treated with HIFU can\nreduce some of the complication burden or risk seen with larger, excised endometriomas. Among other\nbenefits, HIFU has been shown to result in shorter hospital stays and reported post-operative pain.\nAdditionally, there is a cosmetic benefit for women who are concerned about residual scars as HIFU does not\ncause any abdominal incisions \n[15]\n. Although the benefits of HIFU are many, one of the biggest shortcomings\nis size limitations with a resectable mass having a diameter below 3-4 cm as mentioned previously \n[16]\n.\nWhile HIFU may be appropriate for small-to-moderate-sized masses, this patient’s larger mass exceeding\n6cm, leaves surgical excision as a more preferential treatment of choice. Progestins, combined oral\ncontraceptive pills, and NSAIDs are first-line treatments for endometriosis-associated pain and symptomatic\nmanagement \n[17]\n. Hysterectomy is a last-line treatment and is appropriate for patients who do not desire\nfuture fertility and have not responded to more conservative management \n[17]\n. Our patient also desired\nimmediate, future fertility so contraceptive management or hysterectomy was deferred in this case.\nLastly, it is important to note that endometriosis has been shown to undergo a rare, malignant\ntransformation in as few as 0.7-1.5% of all endometrioma cases, with 79% of these neoplastic\ntransformations occurring in the ovaries \n[18]\n. CSE was the most common site for malignant transformation\nof AWE, with an incidence of 0.3-1%. If left untreated, subsequent malignant transformation archetypes\ninclude clear cell cancer followed by endometrioid adenocarcinoma \n[18]\n. It is therefore important to be\nproactive in treating symptomatic endometriosis, especially in atypical locations such as adjacent to the\nabdominal wall. Some studies report malignant Pfannenstiel endometrioma transformations to have\nsignificant mortality, nearly upwards of 43% if left untreated \n[7]\n.\nConclusions\n \nPublished via Alabama College of\nOsteopathic Medicine Research\n2024 Zhang et al. Cureus 16(8): e66223. DOI 10.7759/cureus.66223\n7\n of \n9\n\nOne of the goals of our case report is to highlight the presentation of Pfannenstiel endometrioma and\ndecision making that indicates early intervention. Although rare, Pfannenstiel endometrioma should be\nconsidered a differential diagnosis for a patient with a previous cesarean section presenting with cyclical\nabdominal pain and a palpable tenderness along the incision site. CSE may also be asymptomatic, as seen in\nthis patient upon the incidental discovery of the endometrioma. Due to this, shared decision making\nbetween the patient and physician is necessary to determine preference for treatment. In this case, the\npatient was managed conservatively for several months until her symptoms worsened, as surgical\nmanagement was not performed after an initial incidental finding on CT imaging. While no official\nguidelines have been established for management of cesarean section endometriosis, it is important to be\nfamiliar with conservative and surgical options for patients.\nOne alternative way to approach CSE is to increase efforts in reducing risk factors for more advanced\nendometriosis disease progression, such as obesity. As it is well established, contraceptives and NSAIDs may\nbe helpful for symptomatic management. However, removal of the endometrioma, i.e. HIFU or surgical\nexcision are both options for definitive management. Definitive management may not only prevent the\ncommon symptoms of chronic pain, discomfort, and dysmenorrhea but also lower the risk of malignant\ntransformation. This should be taken into consideration when deciding to surgically treat a patient with\nPfannenstiel endometriosis, however, it is noted that malignant transformation is rare and occurs more\nfrequently in other locations. Importantly, preventing and reducing risk for the development of Pfannenstiel\nendometriosis starts at the initial cesarean section procedure, by using surgical techniques to minimize\nseeding into the incision. Lastly, the upward trend of elective cesarean sections poses an additional\nchallenge for radiographers and clinicians toward the already, highly debated management guidelines of\natypical endometriomas. By introducing this case, we hope to contribute to additional and to guide clinician\ndecision-making to proceed or withhold interventions on endometriomas, incidental or atypical.\nAdditional Information\nAuthor Contributions\nAll authors have reviewed the final version to be published and agreed to be accountable for all aspects of the\nwork.\nConcept and design:\n  \nNathan Zhang, Sedona Robrahn, Katherine R. Thornburgh, Justin Moon\nAcquisition, analysis, or interpretation of data:\n  \nNathan Zhang, Sedona Robrahn, Katherine R.\nThornburgh, Justin Moon, Muhammad K. Ather, Colton P. Boney, Joel A. Yalowitz\nDrafting of the manuscript:\n  \nNathan Zhang, Sedona Robrahn\nCritical review of the manuscript for important intellectual content:\n  \nNathan Zhang, Sedona Robrahn,\nKatherine R. Thornburgh, Justin Moon, Muhammad K. Ather, Colton P. Boney, Joel A. Yalowitz\nSupervision:\n  \nNathan Zhang, Joel A. Yalowitz\nDisclosures\nHuman subjects:\n Consent was obtained or waived by all participants in this study. \nConflicts of interest:\n In\ncompliance with the ICMJE uniform disclosure form, all authors declare the following: \nPayment/services\ninfo:\n All authors have declared that no financial support was received from any organization for the\nsubmitted work. \nFinancial relationships:\n All authors have declared that they have no financial\nrelationships at present or within the previous three years with any organizations that might have an\ninterest in the submitted work. \nOther relationships:\n All authors have declared that there are no other\nrelationships or activities that could appear to have influenced the submitted work.\nReferences\n1\n. \nParasar P, Ozcan P, Terry KL: \nEndometriosis: epidemiology, diagnosis and clinical management\n. 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Eur J Obstet Gynecol Reprod Biol. 2021,\n264:363-7. \n10.1016/j.ejogrb.2021.08.006\n \nPublished via Alabama College of\nOsteopathic Medicine Research\n2024 Zhang et al. Cureus 16(8): e66223. DOI 10.7759/cureus.66223\n9\n of \n9","source_license":"CC0","license_restricted":false}