Progressively enlarged Caesarean section scar endometriosis during pregnancy: case report and literature review

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This paper reports a 36-year-old pregnant woman with prior Caesarean section who developed abdominal wall scar endometriosis near the incision that initially measured 1 cm in 2018, then progressively enlarged with multifocal spread during pregnancy and reached a cystic mass of about 10 × 6 cm by 36 weeks with prominent internal vascularity; imaging raised concern for malignancy. The lesion was excised during elective Caesarean delivery after preoperative ultrasound and MRI assessed invasion depth, and en bloc resection was performed with primary wound closure without mesh; histopathology confirmed endometriosis with decidual-like stromal changes, glandular atrophy, and progesterone receptor positivity with estrogen receptor negativity, while pelvic exploration found no concurrent pelvic endometriosis. The authors note limitations typical of a single case plus literature context, with only sparse prior reports and uncertainty about generalizability of management during pregnancy. This paper is centrally about endometriosis—specifically progressively enlarged Caesarean section scar (abdominal wall) endometriosis during pregnancy.

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Discussion

Abdominal wall endometriosis is a rare form of endometriosis, with a reported incidence of 0.03% to 0.4%.1 Most cases occur near a CS scar. Previous studies have described changes in pelvic endometriosis during pregnancy,2 3 but there is limited literature describing the progression and management of AWE during pregnancy. In this case, the patient experienced significant enlargement of scar endometriosis during pregnancy, indicating that AWE, similar to pelvic endometriotic lesions, may also gradually enlarge during gestation. The first-line treatment for AWE is surgical excision of the mass, which is typically performed in the non-pregnant period. As the lesion is often closely related to a previous CS scar, simultaneous excision may significantly increase the complexity of CS. In this case, the lesion was dissected along its margin during CS. Our retrospective study demonstrated that among 367 patients who underwent margin-based excision, the recurrence rate was 3.3%,4 comparable to or lower than rates reported in previous studies.5 6 This suggests that excision along the lesion margin is sufficient and does not increase the risk of recurrence. After thorough assessment, excision during CS may be considered if the lesion is small and demonstrates minimal depth of invasion. Given the potential for lesion enlargement and the challenges of abdominal wall reconstruction during pregnancy, timely surgical intervention is particularly advisable for patients planning future pregnancies. Preoperative evaluation is also crucial. Our previous research classified patients into three types according to the depth of lesion invasion.4 This patient could be clinically classified as Type I AWE. Given the minimal muscular invasion and stretching of the abdominal wall due to the gravid uterus, primary closure without mesh placement was feasible. This highlights the importance of comprehensive preoperative assessment to guide the choice of surgical approach and anticipate operative complexity. Decidualisation of endometriosis can occur under the influence of high progesterone levels.7 Since Pellegrini8 first described this condition in 1982, only nine cases of decidualised AWE have been reported.2 8 9 10 11 12 13 14 15 In these cases, the mean age was 28.6 ± 5.8 years, and the mean latency from first CS to mass presentation was 29.6 ± 24.2 months. The depth of invasion in these patients differs from that in non-pregnant patients. Our previous research4 showed that in non-pregnant patients, AWE lesions most commonly invade the rectus abdominis muscle. In contrast, existing literature indicates that in pregnant patients, lesions were confined to the subcutaneous and adipose layers, with only one case showing invasion of the rectus abdominis. Enlargement during pregnancy was reported in two patients, with the maximum diameter reaching 3 cm.9 10 Six patients underwent lesion excision during CS,2 8 10 11 12 13 and one underwent elective surgery after CS.14 In our case, the patient experienced rapid enlargement of the AWE during pregnancy and imaging raised a suspicion of malignancy. This suggests that decidualised AWE during pregnancy may present with features mimicking malignancy. Histopathological examination in our patient revealed a positive progesterone receptor expression and absence of oestrogen receptor expression, indicating that the intrinsic biological characteristics of the lesion may have contributed to its abnormal growth. Limited treatment options during pregnancy pose additional risks to maternal and fetal safety. Therefore, in patients with AWE who wish to conceive in the future, surgery should ideally be performed before pregnancy. Author contributions Concept or design: Y Wu, J Leng, Y Dai. Acquisition of data: Y Wu, J Shi, X Li.. Analysis or interpretation of data: Y Li.. Drafting of the manuscript: Y Wu.. Critical revision of the manuscript for important intellectual content: J Leng, Y Dai. Acquisition of data: Y Wu, J Shi, X Li.. Analysis or interpretation of data: Y Li.. Drafting of the manuscript: Y Wu.. Critical revision of the manuscript for important intellectual content: J Leng, Y Dai. All authors had full access to the data, contributed to the study, approved the final version for publication, and take responsibility for its accuracy and integrity. Conflicts of interest All authors have disclosed no conflicts of interest. Funding/support This study was supported by the National Key Research and Development Program Project (Ref No.: 2022YFC2704000) and the National Natural Science Foundation of China (Ref No.82071628). The funders had no role in the study design, data collection/analysis/interpretation, or manuscript preparation. Ethics approval This study was approved by the Institutional Review Board of Peking Union Medical College Hospital, China (Ref No.: K23N4099). Informed consent was obtained from the patient for all treatments and procedures, and for publication of the case report with the accompanying clinical images.

References

1. Carsote M, Terzea DC, Valea A, Gheorghisan-Galateanu AA. Abdominal wall endometriosis (a narrative review). Int J Med Sci 2020;17:536-42. Crossref 2. D’Agostino C, Surico D, Monga G, Palicelli A. Pregnancy-related decidualization of subcutaneous endometriosis occurring in a post–Caesarean section scar: case study and review of the literature. Pathol Res Pract 2019;215:828-31. Crossref 3. Sachdeva G, Divyashree PS, Shailaja N. A non-classical presentation of scar endometriosis during pregnancy: case report and review of literature. JBRA Assist Reprod 2022;26:563-66. Crossref 4. Wu Y, Dai Y, Zhang J, et al. The clinical features and long-term surgical outcomes of different types of abdominal wall endometriosis. Arch Gynecol Obstet 2023;307:163-8. Crossref 5. Marras S, Pluchino N, Petignat P, et al. Abdominal wall endometriosis: an 11-year retrospective observational cohort study. Eur J Obstet Gynecol Reprod Biol X 2019;4:100096. Crossref 6. Horton JD, Dezee KJ, Ahnfeldt EP, Wagner M. Abdominal wall endometriosis: a surgeon’s perspective and review of 445 cases. Am J Surg 2008;196:207-12. Crossref 7. Frühauf F, Fanta M, Burgetová A, Fischerová D. Endometriosis in pregnancy—diagnostics and management. Ceska Gynekol 2019;84:61-7. 8. Pellegrini AE. Cutaneous decidualized endometriosis. A pseudomalignancy. Am J Dermatopathol 1982;4:171-4. Crossref 9. El-Gohary YM, Garcia MT, Ganjei-Azar P. Decidualized endometrioma diagnosed by fine needle aspiration cytology: a case report with immunocytochemical confirmation. Diagn Cytopathol 2009;37:373-6. Crossref 10. Nogales FF, Martin F, Linares J, Naranjo R, Concha A. Myxoid change in decidualized scar endometriosis mimicking malignancy. J Cutan Pathol 1993;20:87-91. Crossref 11. Kavari A, Samizadeh B, Maghbool M. Pregnancy-related decidualization in post–Cesarean section scar endometriosis: a case report. Int J Surg Case Rep 2024;124:110274. Crossref 12. Natale KE, Royer MC, Rush WL, Lupton GP. Cutaneous deciduosis: a report of two cases of an unusual pseudomalignancy. J Cutan Pathol 2012;39:777-80. Crossref 13. Günal A, Keskin U, Deveci G, Deveci MS, Yenen MC, Dede M. Tumor-like myxoid change in decidualized scar endometriosis of pregnancy: a case report and review of literature. Turk J Pathol 2009;25:49-52. Crossref 14. Val-Bernal JF, Val D, Gómez-Aguado F, Corcuera MT, Garijo MF. Hypodermal decidualized endometrioma with aberrant cytokeratin expression. A lesion mimicking malignancy. Am J Dermatopathol 2011;33:e58-62. Crossref 15. Berardo M, Valente P, Powers C. Cytodiagnosis and comparison of nondecidualized and decidualized endometriosis of the abdominal wall. A report of two cases. Acta Cytol 1992;36:957-62.

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