{"paper_id":"b3a56623-7c41-4753-a271-98705d69248a","body_text":"Review began\n 10/10/2023 \nReview ended\n 10/23/2023 \nPublished\n 10/25/2023\n© Copyright \n2023\nPutta Nagarajan et al. This is an open\naccess article distributed under the terms of\nthe Creative Commons Attribution License\nCC-BY 4.0., which permits unrestricted use,\ndistribution, and reproduction in any\nmedium, provided the original author and\nsource are credited.\nScar Endometriosis vs Hemangioma: A Diagnostic\nConundrum\nHrithik Dakssesh Putta Nagarajan \n \n, \nKeerthivasan Selvanathan \n \n, \nVrijesh Gopalakrishnan \n \n, \nRam Vivek\nRamamoorthy \n \n, \nJeyachitra Gopalakrishnan \n1.\n Department of Dermatology, Madurai Medical College, Madurai, IND \n2.\n Department of General Surgery, Madurai\nMedical College, Madurai, IND \n3.\n Department of Obstetrics and Gynaecology, Nithilaa Nursing Home, Madurai, IND\nCorresponding author: \nHrithik Dakssesh Putta Nagarajan, \npnhrithik@gmail.com\nAbstract\nScar endometriosis refers to the presence of endometrial glands and stroma at the site of a scar.\nHemangiomas, on the other hand, are benign vascular tumors. In this case report, we unravel the clinical\nenigma around a patient who presented with a painful mass at the previous cesarean section scar site.\nInitially, we were confident that this was ectopic endometrium presenting as scar endometriosis. However,\nour journey took an unexpected turn when histopathological findings contradicted our clinical suspicions.\nHere, we delve into the intricate details of this captivating case, shedding light on the complexities of the\ndiagnosis we faced.\nCategories:\n Dermatology, Obstetrics/Gynecology, General Surgery\nKeywords:\n catamenial changes, skin, case report, excision and biopsy, cyclical abdominal pain, menstrual cycle,\nhemangioma, scar site endometriosis\nIntroduction\nEndometriosis is a gynecological disease, characterized by the development and presence of endometrial\ntissue in places outside of the uterine cavity. The most commonly affected areas include the ovaries (54.9%),\nfollowed by the posterior broad ligament (35.2%), anterior cul-de-sac (34.6%), posterior cul-de-sac (34.0%),\nand the uterosacral ligament (28.0%) \n[1]\n. Scar endometriosis is a relatively rare form of this condition, with a\nreported incidence of 0.08% among women who have had cesarean sections \n[2]\n. It typically occurs in the\nanterior abdominal wall, at the site of previous cesarean section scars.\nHemangiomas are benign overgrowths of blood vessels and endothelial cells, found either in the outer layers\nof the skin, deeper layers of the skin, internal organs, subcutaneous tissue, or sometimes in multiple\nlocations simultaneously. Hemangiomas are categorized as capillary or cavernous based on the size of the\nvascular channels. Capillary hemangiomas have small diameter vascular channels. On the other hand,\ncavernous hemangiomas have large diameter vascular channels \n[3]\n. While hemangiomas are commonly seen\nin infants, encountering them in adults, especially within post-cesarean section scars, is exceptionally rare.\nMany cases of hemangioma occur spontaneously without any identifiable cause, while others are associated\nwith factors such as injuries, burns, immobilization using casts, pregnancy, and the use of antiretroviral\ntherapy \n[4]\n.\nCase Presentation\nA 34-year-old South Asian female presented to the clinic with chief complaints of swelling and associated\npain in the right lower abdomen, specifically over the lateral aspect of the previous cesarean section scar on\nthe right side, which had been bothering her for the past 10 months. The patient has two healthy children\ndelivered via lower segment cesarean section (LSCS). The patient noticed the development of this swelling\nduring her second pregnancy, around the fifth month, over the LSCS scar of the first pregnancy (Figure \n1\n).\nInitially, it was suspected to be a keloid.\n1\n2\n1\n2\n3\n \n Open Access Case\nReport\n \nDOI:\n 10.7759/cureus.47674\nHow to cite this article\nPutta Nagarajan H, Selvanathan K, Gopalakrishnan V, et al. (October 25, 2023) Scar Endometriosis vs Hemangioma: A Diagnostic Conundrum.\nCureus 15(10): e47674. \nDOI 10.7759/cureus.47674\n\nFIGURE\n 1: Image showing the presence of mass (arrow) in the lateral\naspect of the previous LSCS scar on the right side of the abdomen.\nLSCS: lower segment cesarean section.\nThe patient sought medical attention five months after the delivery of the second child, due to the\nemergence of pain in the swelling, coinciding with the resumption of her menstruation, which occurred\nthree months postpartum. During the initial three months following delivery, the patient did not experience\nany menstrual flow due to lactational amenorrhea.\nThe size of the swelling was noted to be equivalent to that of a soybean (0.7 cm x 0.4 cm) by the operating\nsurgeon during the delivery of the second baby. But then, it gradually increased in size to measure 3 cm x 2\ncm. Throughout the second pregnancy and the initial three months following delivery, the skin over the\nswelling was normal. However, with the onset of menstruation, the skin over the swelling began to peel\noff and was accompanied by serous discharge. The swelling also exhibited color changes in relation to the\nmenstrual cycle. It appeared with a bright reddish hue just before the onset of menses and turned pale\npinkish after menstruation. It is important to note that the patient did not complain of any bleeding from\nthe swelling.\nAdditionally, the patient noticed that the swelling became harder in consistency during menstruation,\ncompared to its firm state at other times. She complained that the intensity of the pain varied in accord with\nher menstrual cycle. The pain typically spiked during the time of menstrual blood flow, which usually lasts\nfor about five days in this patient. However, she also reported a period of gradual waxing of pain, for about\nthree days before the onset of menstrual flow, and a subsequent period of waning of pain, for about three\ndays after the passage of menses. The patient also reported experiencing intermittent pricking pain, that\n2023 Putta Nagarajan et al. Cureus 15(10): e47674. DOI 10.7759/cureus.47674\n2\n of \n6\n\nlasts for about five minutes per episode, over the swelling at times other than during menstruation.\nInvestigations\nUltrasound Imaging\nAfter the initial clinical workup was done, she was sent for an ultrasound imaging of the mass. The scan\nshowed a heteroechoic lesion, measuring 2.9 cm x 1.8 cm at the scar site in the subcutaneous plane. Color\nflow revealed rich vascularity.\nMagnetic Resonance Imaging\nFurther imaging studies were considered necessary. So a magnetic resonance imaging (MRI) of the abdomen\nand pelvis was taken. It showed a well-defined polypoidal, intensely enhancing soft tissue mass with\ninternal hemorrhagic foci and significant vascularity, in the lateral aspect of the postoperative scar site on\nthe right side measuring 3.2 cm x 2.2 cm x 1.8 cm in size, with adjacent skin thickening (Figure \n2\n).\nFIGURE\n 2: Magnetic resonance imaging (MRI) of the abdomen and\npelvis revealing the mass (arrow).\nIt is seen as an intensely enhancing soft tissue mass.\nTreatment\nAfter a proper workup, excision and biopsy of the swelling was planned. Owing to the easy excisability of the\nlesion, no trial of hormonal drug therapy was tried. The excision and biopsy was performed under local\nanesthesia and was uneventful without any complications. The excised mass (Figure \n3A\n) was sent for\nhistopathology. The incision site was then closed with intermittent polypropylene sutures (Figure \n3B\n). The\npatient was discharged, with advice to return in case of persistence of symptoms, and to return to the clinic\nafter two weeks for the removal of sutures. Nonsteroidal anti-inflammatory drugs (NSAIDs) were prescribed\nfor three days following surgery for the relief of postoperative pain.\n2023 Putta Nagarajan et al. Cureus 15(10): e47674. DOI 10.7759/cureus.47674\n3\n of \n6\n\nFIGURE\n 3: Image of the excised mass and postoperative image.\nA) Image of the excised mass with a centimeter (cm) scale for reference. B) Postoperative image showing the\nincision site closed with polypropylene sutures.\nHistopathology\nThe excised mass was sent in for histopathological studies. The cut surface of the mass was noted to be gray-\nbrown in color. A thorough study of sections from the sample, under the microscope, showed\nfibrocollagenous tissue with dilated proliferating blood vessels, areas of hemorrhage and inflammatory cell\ninfiltrates (Figure \n4\n). Endometrial glands and stroma were nowhere to be found, and the other features were\nsuggestive of a capillary hemangioma. So, a definitive diagnosis of hemangioma was made.\nFIGURE\n 4: Histopathology of the excised mass showing\nfibrocollagenous tissue enclosing numerous proliferating blood vessels\nlined by prominent endothelial cells surrounded by neutrophils and\nlymphocytes.\nFollow-Up\nAfter two weeks, the patient reported back to the clinic for removal of sutures. The scar site was healthy with\n2023 Putta Nagarajan et al. Cureus 15(10): e47674. DOI 10.7759/cureus.47674\n4\n of \n6\n\nno discharge. No residual signs were noted, and the sutures were removed. Telephonic follow-up after one\nmonth from the date of the procedure was done. The patient was informed of no residual symptoms.\nDiscussion\nScar endometriosis typically presents with localized cyclical pain, often occurring in conjunction with\nmenstruation. The pain is characterized as sharp, localized discomfort that intensifies during menstruation,\nreflecting the influence of hormones on endometrial tissue. Other manifestations include a palpable mass or\nswelling at the cesarean scar site which may present with tenderness \n[5,6]\n. The elicited history of cesarean\nsection and correlation of symptoms with the menstrual cycle were pivotal in our case and led to a\nprovisional diagnosis of scar endometriosis.\nThe clinical presentation of hemangioma depends on its location and depth within the body. The majority\npresent as a single localized cutaneous mass. Hemangiomas most commonly occur on the head and neck\nregion, accounting for 60% of cases. Following that, 25% of the cases are seen on the trunk, as in this case.\nAlthough 25% is a significant share, all hemangiomas are commonly observed only in infants and are rare to\nbe seen in adults. Hemangiomas can be superficial, deep, or mixed with components of both superficial and\ndeep layers. Superficial lesions involve the superficial dermis and are raised, lobulated, and bright red. Deep\nhemangiomas, also called subcutaneous hemangiomas, arise from the reticular dermis and/or the subcutis\nlayer and appear as bluish-hued nodule or plaque. Mixed hemangiomas present with features of both\nlocations \n[7]\n. The presentation of hemangioma tends to be consistent and unaffected by the menstrual cycle.\nHemangiomas may sometimes present with pain. However, it is exceedingly rare for superficial\nhemangiomas to be painful, especially when the surface is intact. Furthermore, it is even rarer for the pain to\nfollow a cyclical pattern. Only hemangiomas with ulceration and spinal hemangiomas are commonly known\nto cause pain. There is very little literature providing evidence for hemangiomas presenting with such\ncyclical features. In the case report by Ortiz-Rey et al. \n[8]\n, evidence for catamenial changes with the color,\nsize, and intensity of pain of a similar lesion is given. They also faced the same dilemma as we faced and\ninitially thought it was a case of cutaneous endometriosis and had the lesion excised. In that case report,\ndetails of a hemangioma on the lower abdomen of a woman taking oral contraceptives are given. In that\npatient, the catamenial nature of these changes was precisely elicited, as the menstrual cycle was under\ncontrol with the use of monophasic contraceptive pills \n[8]\n.\nAlthough there are a few more similar clinical evidence in medical literature \n[9-12]\n, to our knowledge, this is\nthe first case in which this type of lesion presented over the scar from previous LSCS in a post-cesarean\nsection female, causing utmost confusion in the diagnosis as these features were very typical of a case of\nscar endometriosis. These clinical pieces of evidence, in the form of case reports, support the possibility of\nsex steroid influence on vascular lesions. In most of these cases, the results of immunohistochemistry for\nestrogen and progesterone receptors came back negative. But estrogen by itself, without acting on the\nreceptors, can cause venodilation leading to changes in the lesion as observed in our patient \n[13]\n. So the\ncatamenial fluctuations in the levels of estrogen may be responsible for the catamenial changes in these\nvascular lesions.\nConclusions\nOur case highlights the diagnostic challenges we faced in differentiating scar endometriosis from\nhemangioma. While scar endometriosis was our initial suspicion due to its clinical presentation,\nhistopathological examination of the excised tissue was crucial for confirming the diagnosis as hemangioma.\nAlthough rare, it is vital to keep hemangioma as a probable differential in our minds when faced with similar\nclinical situations. The importance of accurate diagnosis in cases like these cannot be emphasized enough.\nMisdiagnosis can lead to inappropriate treatments, such as hormonal therapies commonly used for\nendometriosis, being administered for cases of hemangiomas, which would be ineffective in treating them.\nIn contrast, a precise diagnosis enables the implementation of the most appropriate treatment plan. Further\nresearch is needed to better understand the clinical and histological characteristics of these conditions,\nfacilitating early and accurate diagnosis and improving patient outcomes.\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  \nHrithik Dakssesh Putta Nagarajan\nAcquisition, analysis, or interpretation of data:\n  \nHrithik Dakssesh Putta Nagarajan, Keerthivasan\nSelvanathan, Ram Vivek Ramamoorthy, Vrijesh Gopalakrishnan, Jeyachitra Gopalakrishnan\nDrafting of the manuscript:\n  \nHrithik Dakssesh Putta Nagarajan, Keerthivasan Selvanathan\n2023 Putta Nagarajan et al. Cureus 15(10): e47674. DOI 10.7759/cureus.47674\n5\n of \n6\n\nCritical review of the manuscript for important intellectual content:\n  \nHrithik Dakssesh Putta\nNagarajan, Ram Vivek Ramamoorthy, Vrijesh Gopalakrishnan, Jeyachitra Gopalakrishnan\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. \nJenkins S, Olive DL, Haney AF: \nEndometriosis: pathogenetic implications of the anatomic distribution\n.\nObstet Gynecol. 1986, 67:335-8.\n2\n. \nMinaglia S, Mishell DR Jr, Ballard CA: \nIncisional endometriomas after cesarean section: a case series\n. J\nReprod Med. 2007, 52:630-4.\n3\n. \nGeorge A, Mani V, Noufal A: \nUpdate on the classification of hemangioma\n. J Oral Maxillofac Pathol. 2014,\n18:S117-20. \n10.4103/0973-029X.141321\n4\n. \nHunt SJ, Santa Cruz DJ: \nVascular tumors of the skin: a selective review\n. Semin Diagn Pathol. 2004, 21:166-\n218. \n10.1053/j.semdp.2005.01.001\n5\n. \nNepali R, Upadhyaya Kafle S, Pradhan T, Dhamala JN: \nScar endometriosis: a rare cause of abdominal pain\n.\nDermatopathology (Basel). 2022, 9:158-63. \n10.3390/dermatopathology9020020\n6\n. \nTatli F, Gozeneli O, Uyanikoglu H, et al.: \nThe clinical characteristics and surgical approach of scar\nendometriosis: a case series of 14 women\n. Bosn J Basic Med Sci. 2018, 18:275-8. \n10.17305/bjbms.2018.2659\n7\n. \nChamli A, Aggarwal P, Jamil RT, Litaiem N: \nHemangioma\n. StatPearls [Internet]. StatPearls Publishing,\nTreasure Island, FL; 2023.\n8\n. \nOrtiz-Rey JA, González-Ruiz A, San Miguel P, Alvarez C, Iglesias B, Antón I: \nHobnail haemangioma\nassociated with the menstrual cycle\n. J Eur Acad Dermatol Venereol. 2005, 19:367-9. \n10.1111/j.1468-\n3083.2004.01168.x\n9\n. \nMorganroth GS, Tigelaar RE, Longley BJ, Luck LE, Leffell DJ: \nTargetoid hemangioma associated with\npregnancy and the menstrual cycle\n. J Am Acad Dermatol. 1995, 32:282-4. \n10.1016/0190-9622(95)90149-3\n10\n. \nCarlson JA, Daulat S, Goodheart HP: \nTargetoid hemosiderotic hemangioma: a dynamic tumor: report of 3\ncases with episodic and cyclic changes and comparison with solitary angiokeratomas\n. J Am Acad Dermatol.\n1999, 41:215-24. \n10.1016/s0190-9622(99)70052-6\n11\n. \nLazarou G, Goldberg MI: \nVulvar arteriovenous hemangioma. a case report\n. J Reprod Med. 2000, 45:439-41.\n12\n. \nFernandez-Flores A, Manjon AJ, Campo F: \nClinical changes in \"true\" hobnail hemangioma during\nmenstruation\n. Bratisl Lek Listy. 2008, 109:141-3.\n13\n. \nMendelsohn ME: \nGenomic and nongenomic effects of estrogen in the vasculature\n. Am J Cardiol. 2002,\n90:3F-6F. \n10.1016/s0002-9149(02)02418-9\n2023 Putta Nagarajan et al. Cureus 15(10): e47674. DOI 10.7759/cureus.47674\n6\n of \n6","source_license":"CC0","license_restricted":false}