{"paper_id":"1ffe6889-676f-4503-94cf-873d0be1ebdb","body_text":"Copyright © 2022 The Authors. This is an Open Access article distributed under the terms of the Creative \nCommons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, \nprovided the original work is properly cited.\n1 12 Airforce Hospital, Department of Laboratory Sciences, Gorakhpur, Uttar Pradesh, India\nDeciduosis in a cesarean scar\nToyaja Jadhav1 , Rohini Doshetty1 \nHow to cite: Jadhav T, Doshetty R. Deciduosis in a cesarean scar. Autops Case Rep [Internet]. 2022;12:e2021383. https://doi.org/10.4322/\nacr.2021.383\nClinical Case Report and Review\nABSTRACT\nDeciduosis is the presence of ectopic decidual tissue outside the uterus, pelvic, or abdominal organs usually associated with \npregnancy. Cutaneous deciduosis is a highly uncommon manifestation of deciduosis and most commonly is misdiagnosed \nas a primary malignancy or a metastatic deposit. Typically, it is detected incidentally during operative procedures. It has \nbeen rarely documented within a surgical scar; with the incidence of surgically proven deciduosis being approximately \n1.6%, and is often difficult to diagnose due to its rarity. Here, we present a case of deciduosis of cesarean scar in a \n34-year-old pregnant female. \nKeywords  \nCesarean Section; Embryo implantation; Cicatrix\nINTRODUCTION\nDeciduosis is clinically defined as the presence \nof ectopic decidual tissue in locations outside the \nuterus. It has been reported to occur in various pelvic \nand extra-pelvic sites. It is typically known to present \nduring pregnancy but has also been reported in non-\npregnant women. It is considered to be a benign lesion \nduring pregnancy, not associated with any obstetric \ncomplications. It does not have any pathological \nimpact on the mother as well as the fetus. It is usually \nasymptomatic and can remain undetected throughout \npregnancy. Total remission is generally known to occur \nin the postpartum period; however, some cases may \nrequire surgical intervention, especially those that often \nmimic a malignancy. 1\nCutaneous deciduosis is an uncommon \nmanifestation of cutaneous endometriosis. Cutaneous \nendometriosis usually occurs within the umbilical \nregion or in abdominal surgical scars, the latter typically \noccurring after cesarean section, appendectomy, or an \ninguinal hernia repair. Although deciduosis has been \nreported in numerous ectopic locations, most lesions \nare encountered within the cervix or ovary. 2\nTo date, only a few well-documented instances \nof cutaneous deciduosis have been reported, with very \nfew occurring within abdominal scars from previous \ncesarean sections. 2,3\nWe present a case of cutaneous deciduosis of \na cesarean scar diagnosed incidentally in a 34-year-\nold pregnant female, along with some review of the \navailable literature.\nMETHODOLOGY\nAll the case series and case reports, inclusive of \ntheir references, identified by extensively searching \nthe PubMed, Scopus, Medline and Google Scholar \n\nDeciduosis in a cesarean scar\n2-8 Autops Case Rep (São Paulo). 2022;12:e2021383\ndatabases utilizing the keywords “deciduosis”, “extra- \nabdominal deciduosis”, “cutaneous deciduosis” and \n“deciduosis in a cesarean scar” were read and included \nin this manuscript. A total of 13 cases of cutaneous \ndeciduosis have been reported in literature from 1982; \nof these, 8 of them have been known to occur in a \nscar of a previous cesarean section.\nCASE REPORT\nA 34-year-old pregnant female (G2P1L1A0) \npresented to this hospital at 38 weeks of gestation \nwith complaints of abdominal pain.\nHer previous pregnancy was six years ago, \nwhich had concluded in a healthy child with breech \npresentation, delivered through lower segment \ncesarean section (LSCS). She was a known case \nof hypothyroidism and was also suffering from \nGestational Diabetes Mellitus (GDM) in the current \npregnancy and was being managed for the same with \nTab Eltroxin 50ug, Inj Glargine, and Tab Metformin \n500mg, respectively.\nHer obstetric examination revealed a uterine \nfundus height of 36 weeks with a breech presentation \nand a normal fetal heart rate (FHR). Her preliminary \nhematological as well as serological investigations were \nwithin normal clinical limits.\nObstetric ultrasound examination was also carried \nout, which revealed an adequate Amniotic Fluid Index \n(AFI) with the placenta placed anteriorly and a breech \nfetal presentation.\nHence, in view of the above clinical and \nultrasonographical findings, the patient was taken \nfor elective LSCS as a case of Antenatal Case (ANC) \nwith breech presentation with previous LSCS with \nGestational DM and hypothyroidism.\nA healthy infant was delivered. Additionally, \nintraoperatively, the scar of previous LSCS presented \nwith features of endometriosis along the left lateral \nmargin, which was excised clinically as endometrioma \nand sent for histopathological evaluation.\nGrossly, the sample presented as multiple \nfragmented tissue bits, with the largest measuring \napproximately 2cm and the smallest measuring \napproximately 1cm in its greatest dimension, \nrespectively.\nOn microscopic evaluation, hematoxylin and \neosin (H&E) stained sections revealed multiple nodules \ncomposed of decidualized stromal cells surrounding a \nfew slit-like endometrial glands with fibroblasts and \ncollagen (Figure 1).\nThese decidual cells were polygonal, with large \nnuclei, abundant homogenous eosinophilic cytoplasm \n(Figure 2), and associated with vacuolar degeneration \nin some places.\nOccasional dilated endometrial glands were also \nnoted, which showed eosinophilic secretions with \nadipose tissue present along the periphery ( Figure 3). \nNo features depicting atypia were noted.\nOn immunohistochemical evaluation, it was noted \nthat the decidual cells showed reactivity to PR receptors \nand CD10 antibody ( Figure 4).\nFigure 1. Photomicrograph of the lesion reveals \nmultiple nodules composed of decidualized stromal cells \nsurrounding a few slit-like endometrial glands admixed \nwith fibroblasts and collagen. The arrow highlights the \nslit-like endometrial glands (H&E, 40x).\nFigure 2. Photomicrograph of the lesion. High power \nview of the lesion showing the round to polygonal \nstructure of decidual cells. The arrow highlights slit-like \nendometrial glands (H&E;100x).\n\nJadhav T, Doshetty R\n3-8Autops Case Rep (São Paulo). 2022;12:e2021383\nThese histopathological features, along with \nthe clinical findings connoted with the diagnosis of \ndeciduosis of cesarean scar.\nDISCUSSION\nDeciduosis is clinically defined as the presence of \ndecidual tissue at sites other than the uterus.1 Walker4 \nfirst described it in 1887. Extrauterine decidual cell \ndeposition is most commonly seen in the ovaries, \ncervix, uterine serosa, and the lamina propria of \nthe fallopian tubes, while it is less commonly noted \nalong the appendix, omentum, diaphragm, liver, \nspleen, paraaortic-pelvic lymph nodes and renal \npelvis. Involvement of a previous surgical scar is \nuncommon. The incidence of a surgically proven \ncutaneous deciduosis is approximately 1.6%. 3 It is \ncommonly associated with pregnancy, as seen in our \ncase. However, it can also be seen in a non-pregnant \nstate. It is associated with a progesterone-secreting \nactive corpus luteum or the adrenal cortex in the non-\npregnant condition. Most patients are asymptomatic. \nHowever, some may present with features of hematuria \nor even obstructed ileus due to the involvement of \nvarious organs. 5 Our patient was asymptomatic.\nGrossly, deciduosis of cesarean scar may show \na varied presentation ranging from an individual \nFigure 3. Photomicrograph of occasional dilated endometrial glands containing eosinophilic secretions seen lying \namongst the decidual cells (H&E, 40x).\nFigure 4. A and B – decidual cells showing positive reaction for PR and CD10 respectively (400x).\n\nDeciduosis in a cesarean scar\n4-8 Autops Case Rep (São Paulo). 2022;12:e2021383\ngeographic pattern, nodular distribution or a polypoid \nappearance, which may often mimic a neoplasm.\nMicroscopically, decidual cells are commonly \nfound to be associated with endometrial tissue present \nover a scar. The lesions may frequently present as small \ncell groups or single-cell clusters, and uncommonly, \nthey are in the form of widespread-diffuse deciduosis \nthat completely occupies the scar along with the \nunderlying adipose stroma. Our case presented with \nmicroscopic features depicting the latter. Decidual cells \nare generally large and polygonal, with homogeneous, \neosinophilic cytoplasm associated with varying degrees \nof vacuolar degeneration. Decidual cell vacuolization \nis related to the duration of the pregnancy. Stroma \nmay also show myxoid deposit due to vacuole rupture \nif the decidual cell cytoplasmic vacuolar degeneration \nis over 50%. 5 Our case did present some degree of \nvacuolar degeneration; however, myxoid changes \nwere not noted.\nThe pathogenesis of ectopic decidual reactions \nis not yet fully understood. It is still not completely \ndeciphered whether it is a physiological reaction or \na pathological process. It is said to be the result of \nthe exaggerated response of the endometrium to \nprogesterone during pregnancy. 5\nZaytsev and Taxy 6 have suggested two related \ntheories. The most commonly accepted theory \nis the metaplasia of the sub-celomic pluripotent \nmesenchymal cells with the effect of progesterone. The \nfact that the lesion resolves once the hormonal stimulus \nends supports this theory. Another theory is the “de \nnovo” development of decidual cells. Endometriotic \nfoci undergo marked stromal decidualization with \nthe effect of progesterone during pregnancy, \nwhich resembles ectopic decidua. It is, therefore, \nnecessary to differentiate deciduosis from decidualized \nendometriosis clinicopathologically. The presence of \nclinical symptoms at the beginning of the menstrual \nperiod and the presence of endometriotic foci in other \nareas is important for cases with a clinical picture \nof endometriosis. 5 Our patient did not present with \nany clinical features or a history confirming previous \nendometriosis. Histologically, the diffuse distribution of \nthe lesion, edema of the decidualized stroma, old and \nnew hemorrhagic foci, presence of pseudoxanthoma \ncells and fibrosis signifying endometrial gland atrophy \nand Arias-Stella reaction are important diagnostic \nfeatures of decidual transformation of endometriotic \nfoci in pregnancy, i.e. decidualized endometriosis.5 Our \ncase showed very scant endometrial tissue admixed \nwith decidual cells, with the absence of the other \nabove-mentioned features, which confirms pregnancy-\nrelated ectopic decidua of a cesarean scar.\nHistopathologically, it is important to differentiate \ndeciduosis comprising decidual cells showing varying \ndegrees of atypia with foci of hemorrhagic necrosis \nfrom deciduoid variant of malignant mesothelioma and \nmetastatic malignant melanoma. Ectopic decidual tissue \ncontaining myxoid stroma and vacuolated decidual \ncells must also be differentiated from metastatic signet \nring cell carcinoma. The clinical history of the patient, \nthe lack of active mitosis in decidual cells along with \nthe CD10 and Progesterone Receptor (PR) positivity \nwith non-reactivity to keratin (CK), WT1, calretinin, and \nHBME-1 antibody on immunohistochemistry support \ndeciduosis.5,7 Table 1 depicts the differences between \nthe above-mentioned entities.\nDeciduosis of cesarean scar also needs to be \ndistinguished from a recently introduced entity called \nDeciduoma. Deciduoma is a manifestation of ectopic \ndeciduosis; however, it is a large lesion with abundant \nvascularity and has a high potential for hemorrhagic \ncomplications. 1\nAdditionally, our patient was a known case \nof hypothyroidism and GDM. The literature has \nshown an increased predisposition to endometriosis \ndevelopment in women suffering from hypothyroidism. \nA study conducted by Peyneau et al.25 showed altered \nmetabolism of thyroid hormones in-vitro and also \nconfirmed the aggravating role of thyroid hormones \nin endometriosis. Although GDM is associated with \nplacental vasculopathy, 26 there is no literature citing \nthe association of GDM with deciduosis.\nVery few cases of cutaneous deciduosis have been \nreported in literature to date. Table 2 summarizes the \ncases of cutaneous deciduosis reported in literature.27-35\nThe mainstay of treatment of scar deciduosis \nis surgical excision if it fails to undergo complete \nremission in the post-partum period. The patients are \nknown to completely recover following excision of the \nlesion, and recurrence, if present, is very rare.\nScar deciduosis is usually an uncomplicated \nevent with an asymptomatic course. Complications, \nif present, are rare and may manifest in the form of \n\nJadhav T, Doshetty R\n5-8Autops Case Rep (São Paulo). 2022;12:e2021383\nTable 2. Cases of cutaneous deciduosis reported in literature to date\nRef. No of \ncases Age Site AE Symptoms\nincreased  \nduring  \npregnancy\nIHC studies Treatment Follow – up\n27 01 30 CS - None NR NR Excision on CS NR\n28 01 25 CS -\npainful \nnodule, 1 year \npreviously.\nYes\nVimentin + \nα1antitrypsin+ \nKeratin -ve\nDanazol until \npregnancy Anti-\ninflammatory \ntherapy Excision \non CS\nAW\n29 01 40 U +\nUmbilical \nnodule Cyclic \nenlargement\n- NR Excision on CS Recurrence \nafter excision\n2\n02 21 V NR\nVulvar nodule, \nNoted during \npregnancy\nYes Vimentin +, \nKi67 + PAS + Excision NR\n27 U _\nUmbilical \nnodule during \ncurrent \npregnancy\nYes NR Excision NR\n30 01 24 CS NR Lesion noted \n2 years before Yes CD10 +, ER –, \nCalretinin + NR NR\n31 01 36 CS NR Noted 2 years \nbefore -\nCK8+, hPL +, \nCD10+, EMA 2, \nPLAP 2, CK 5/6 \n-, calretinin -.\nExcision AW\nAE = abdominal Endometriosis; AW = Alive and Well; CS = cesarean scar; NR = Not Reported; U = umbilicus; V = vulva; \nP = Perineum; AAW = Anterior Abdominal wall; CK = cytokeratin; PAS = Periodic Acid - Schiff; EMA = epithelial membrane \nantigen; ER = estrogen receptor; PR = progesterone receptor; PLAP = placental alkaline phosphatase; hPL = Human \nPlacental Lactogen; MNF 116 = cytokeratin MNF116; Ki67= Labile, non-histone nuclear protein expressed in G1, S, g2 \nand M phase of cell cycle and then rapidly catabolized at the end of M phase, and hence, not detectable in G0 and early \ng1 phase cells; hence utilized as a marker of cell proliferation; Rt = right.\nTable 1. Differences between Deciduosis, Deciduoid variant of Malignant Mesothelioma, Metastatic Malignant \nMelanoma and Signet Cell Carcinoma\nFeatures Deciduosis\nDeciduoid variant \nof malignant  \nmesothelioma\nMetastatic  \nmalignant  \nmelanoma\nSignet cell  \ncarcinoma\nCell of origin8-11 Mesenchymal stem \ncells Mesoderm Neural crest cells Epithelium\nMost common site \naffected12-15 Ovaries Peritoneum Lung Liver\nGender affected2,16-18 Females Males Males Males\nAge group \nmost commonly \naffected2,10,13,19,20\nReproductive age (20-\n40 years) 5th – 6th decade 5th decade and older 6th decade\nMorphology2,11,19,21,22\nDecidualized stromal \ncells are polygonal, \noval to spindle shaped \ncells with large \nnuclei and abundant \neosinophilic cytoplasm\nMalignant dyscohesive \nlarge epithelioid \ncells, eosinophilic \ncytoplasm, large \nround nuclei\nLarge epithelioid \nor spindle shaped, \nmixed cytological \nmorphology, macro \nnucleoli\nSignet ring cells with \nintracellular and \nextracellular mucin\nImmunohistochemical \nfeatures2,11,12,21,22\nVimentin, ER, PR, \nDesmin, CD 30 and \nCD 10 positivity\nCytokeratin (CK) \nMNF116, HBME-1 and \nCalretinin positivity\nS100, HMB-45 \npositivity\nCK20, CDX2, MUC2, \nMUC5AC positivity, \nvariable MUC1 \npositivity\nAssociation with \noccupational \nexposure23,24\nNot associated Occupational exposure \nto asbestos Not associated Not associated\nER = estrogen receptor; PR = progesterone receptor.\n\nDeciduosis in a cesarean scar\n6-8 Autops Case Rep (São Paulo). 2022;12:e2021383\nrupture of the scar, with or without uterine rupture, or \nsecondary infection of the lesion resulting in sepsis.36,37\nCONCLUSION\nScar deciduosis is an uncommon but possible \nmanifestation of cutaneous endometriosis and \nshould always be considered in an appropriate clinical \nsetting. Although it may often mimic a neoplasm, the \nhistopathological features of decidual cells along with \nthe utilization of appropriate immunohistochemical \ntechniques help to establish the diagnosis and rule out \nother neoplastic mimics of deciduosis.\nACKNOWLEDGEMENTS\nThe authors would like to thank Lt Col (Dr) \nManoj Gopal Madakshira, Assistant Professor, \nDept of Pathology, Command Hospital (Central \nCommand), Lucknow for helping us with the \nimmunohistochemical evaluation of this case.\nREFERENCES\n1. Dasani M, Lee HJ, Rijhsinghani A. Deciduoma, a large \nintrauterine mass of deciduosis. AJP Rep. 2019;9(4):e337-\n40 . http://dx.doi.org/10.1055/s-0039-1697647 . \nPMid:31737406.\n2. Fair KP, Patterson JW, Murphy RJ, Rudd RJ. Cutaneous \ndeciduosis.  J Am Acad Dermatol . 2000 ;43(1):102-\n7. http://dx.doi.org/10.1067/mjd.2000.100962 . \nPMid:10863233.\n3. Kute K, Swami S, Narwade S, Badlani K. Deciduosis \nin a cesarean scar: a case report.  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No of \ncases Age Site AE Symptoms\nincreased  \nduring  \npregnancy\nIHC studies Treatment Follow – up\n32\n02\n31 Rt P NR\nNodule noted \nby the patient \nafter an \nuneventful \nvaginal \ndelivery\nNR\nCD10 diffuse \ncytoplasmic +, \nVimentin +, \nPan CK and CK \n8/18 -, ER weak \n+, PR strong +, \nPAS +, Colloidal \nIron stain +.\nExcision NR\n26 CS +\nTender, solid, \nenlarging \nmass in \nsuprapubic \narea, superior \nto the \ncesarean \nincision site. \nMass cyclical \nthrobbing \nwith the \nmenstrual \ncycle.\nNR\nCD 10 + \nVimentin + \nER weak + PR \nstrong +\nPartial excision \nduring CS, \nfollowed by \nexcision of \nthe remnant \ntissue by \nPanniculectomy \n06 weeks later.\nNR\n33 01 34 CS NR NR NR NR Excision on CS NR\n34 01 34 CS NR\nNodule at \nprevious \ncesarean scar\nNR NR Excision on CS NR\n35 01 30 AAW - None NR NR Excision on CS NR\n3 01 37 CS NR NR NR NR Excision on CS NR\nThis \ncase 01 34 CS NR NR NR NR Excision during \nCS AW\nAE = abdominal Endometriosis; AW = Alive and Well; CS = cesarean scar; NR = Not Reported; U = umbilicus; V = vulva; \nP = Perineum; AAW = Anterior Abdominal wall; CK = cytokeratin; PAS = Periodic Acid - Schiff; EMA = epithelial membrane \nantigen; ER = estrogen receptor; PR = progesterone receptor; PLAP = placental alkaline phosphatase; hPL = Human \nPlacental Lactogen; MNF 116 = cytokeratin MNF116; Ki67= Labile, non-histone nuclear protein expressed in G1, S, g2 \nand M phase of cell cycle and then rapidly catabolized at the end of M phase, and hence, not detectable in G0 and early \ng1 phase cells; hence utilized as a marker of cell proliferation; Rt = right.\n\nJadhav T, Doshetty R\n7-8Autops Case Rep (São Paulo). 2022;12:e2021383\n4. 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A systematic \nreview on endometriosis during pregnancy: diagnosis, \nmisdiagnosis, complications and outcomes. Hum Reprod \nUpdate. 2016;22(1):70-103. http://dx.doi.org/10.1093/\nhumupd/dmv045. PMid:26450609.\n37. Sholapurkar SL, Sharp NC, Hirschowitz L. Life-threatening \nuterine haemorrhage six weeks after Cesarean section \ndue to uterine scar endometriosis: case report and review \nof literature. Aust N Z J Obstet Gynaecol. 2005;45(3):256-\n8. http://dx.doi.org/10.1111/j.1479-828X.2005.00401.x. \nPMid:15904459.\nThis study carried out at the Department of Laboratory Sciences, 12 Airforce Hospital, Gorakhpur, Uttar Pradesh, \nIndia.\nAuthors’ contributions: Toyaja Jadhav was responsible for data collection and manuscript preparation. Rohini \nDoshetty was responsible for manuscript review.\nEthics statement:  The authors hereby state that an informed consent authorizing data publication was taken \nfrom the patients. The manuscript has been drafted as per the Ethics Committee rules and has also been cleared \nby the institutional Ethics Committee.\nConflict of interest:  None.\nFinancial support:  None.\nSubmitted on: March 11th, 2022 \nAccepted on: April 24th, 2022\nCorrespondence  \nToyaja Jadhav  \n12 Airforce Hospital, Department of Laboratory Sciences  \nAkash Vihar, Gorakhpur, 273002, Uttar Pradesh, India  \nPhone: (+91) 9930310808  \ntoyajadhav.21@gmail.com","source_license":"CC0","license_restricted":false}