{"paper_id":"2582ad21-a64f-486e-a4ab-85bb8d8cc906","body_text":"Received\n 07/26/2023 \nReview began\n 08/03/2023 \nReview ended\n 08/14/2023 \nPublished\n 08/31/2023\n© Copyright \n2023\nBolgarina et al. This is an open access\narticle distributed under the terms of the\nCreative Commons Attribution License CC-\nBY 4.0., which permits unrestricted use,\ndistribution, and reproduction in any\nmedium, provided the original author and\nsource are credited.\nCervical and Vaginal Deciduosis: Insights on\nManagement and a Systematic Review of\nObservational Studies on Pregnancy\nComplications and Management Outcomes\n(Including Vaginal Birth)\nZoryana Bolgarina \n \n, \nHeet N. Desai \n \n, \nMithum Senaratne \n \n, \nShivling S. Swami \n \n, \nSoe Lwin Aye \n \n, \nYash\nTrivedi \n \n, \nAbeer O. Elshaikh \n1.\n Obstetrics & Gynecology, California Institute of Behavioral Neurosciences & Psychology, Fairfield, USA \n2.\n Internal\nMedicine, California Institute of Behavioral Neurosciences & Psychology, Fairfield, USA \n3.\n Internal Medicine,\nCalifornia Institute of Behavioral Neurosciences and Psychology, Fairfield, USA \n4.\n Internal Medicine/Family Medicine,\nCalifornia Institute of Behavioral Neurosciences & Psychology, Fairfield, USA\nCorresponding author: \nZoryana Bolgarina, \ndr.bolgarina@gmail.com\nAbstract\nIntroduction. Deciduosis is an ectopic transformation of connective tissue into decidual-like cells. This is\nthe first systematic review describing the clinical course, associated pregnancy complications, and\nmanagement outcomes of cervical and vaginal deciduosis.\nMethods. Our search covered worldwide observational studies published in English in five databases\n(PubMed, PubMed Central (PMC), Europe PMC, ScienceDirect, and Google Scholar) from inception to\nFebruary 24, 2023. We followed the Preferred Reporting Items for Systematic Reviews and Meta-Analysis\n(PRISMA) guidelines and critically appraised studies using CAse REport (CARE) and Joanna Briggs Institute\n(JBI) tools. Then, we extracted patient characteristics, clinical features, management-related information,\nand outcomes.\nResults. The selection process identified 15 studies describing 30 pregnancies. Macroscopic cervical and\nvaginal deciduosis presented as recurrent vaginal bleeding in over 16 of 24 women (57%). Differential\ndiagnoses included miscarriages, cervical pregnancy, placenta previa, and malignancy. Significant antenatal\nhemorrhages, preterm rupture of membranes, and preterm birth were the most frequent pregnancy\ncomplications. Only one of 27 electively performed procedures resulted in biopsy-induced uncontrolled\nvaginal bleeding (0.04%), suggesting the relative safety of the interventions. Lesion resection led to the\ncessation of recurrent symptoms in eight of eight patients (100%) compared to eight of 15 women (53%)\nunder observation management. All women with polypoid deciduosis over 1.5 cm entered labor and\ndelivered without complications.\nConclusions. We described the clinical course, pregnancy complications, diagnostic-related challenges,\nmanagement, and associated outcomes in women with macroscopic cervical and vaginal deciduosis. We\nsupported the analysis with the current state of the problem and discovered gaps for prospective studies.\nCategories:\n Obstetrics/Gynecology\nKeywords:\n antenatal bleeding, polypectomy, decidual polyp, decidual ectopy, systematic review\nIntroduction And Background\nDeciduosis, an extra-uterine transformation of connective tissue into decidual-resembling cells, mainly\noccurs during pregnancy. Microscopically, 70.2% of biopsies obtained during a cesarean section \n[1]\n and\n15.2%-34% of cervical cytological smears \n[2,3]\n, as well as up to 90% of cervical biopsies \n[4]\n, revealed decidual\ncells. The incidence of macroscopic lesions is unknown.\nDeciduosis is generally considered a benign reaction. However, it might lead to significant pregnancy\ncomplications and management challenges. Existing systematic reviews \n[5-7]\n listed deciduosis-associated\ncomplications, such as spontaneous hemoperitoneum in pregnancy, (peri)appendicitis, bowel perforation,\nendometriotic lesion decidualization, disruptions of such lesions (especially threatening when\napproximated to vulnerable areas like uterine vessels and ureters), urinary bladder or ovarian pseudotumor\nformation, hemothorax, catamenial pneumothorax, etc. However, to our best knowledge, no systematic\nreview has analyzed deciduosis confined to the lower genital tract location and its impact on pregnancy.\nMoving externally from the endocervix, deciduosis of the lower genital tract presents as polyp and ectopy,\nincluding its papillary, polypoid, and infiltrative forms \n[8]\n. Polyp and papillary ectopy arise from the stroma\n1\n2\n2\n2\n3\n2\n4\n \n Open Access Review\nArticle\n \nDOI:\n 10.7759/cureus.44479\nHow to cite this article\nBolgarina Z, Desai H N, Senaratne M, et al. (August 31, 2023) Cervical and Vaginal Deciduosis: Insights on Management and a Systematic Review\nof Observational Studies on Pregnancy Complications and Management Outcomes (Including Vaginal Birth). Cureus 15(8): e44479. \nDOI\n10.7759/cureus.44479\n\nunder the columnar epithelium and appear as multicolored \"beans\" and pale grape-like columnar epithelial\nvilli enlargement, respectively. Polypoid ectopy occurs under the columnar and squamous epithelium,\nfrequently includes a transformation zone, and presents as a friable, yellow-brown mass. Infiltrative ectopy\noriginates under the squamous epithelium as multiple small elevations. Lesion ulceration is common.\nStudies identified that pregnancy-associated changes in the cervix, including decidual cells or the Arias-\nStella reaction, are sometimes mistakenly recognized as atypical \n[9,10]\n. Colposcopic impressions can be\nmisleading while performing a biopsy during pregnancy might be risky because of the possibility of excessive\nbleeding and coincidental pregnancy complications \n[11]\n. Contrary to popular belief, it is most important to\nrule out the coexistence of malignancy in suspicious cases. \nAccumulated data suggest that cervical deciduosis increases the risks of late miscarriages and preterm birth\nbecause of a premature rupture of membranes \n[12-14]\n. Besides, large lesions in labor, especially those that\nlead to intrapartum bleeding, can raise additional challenges \n[15-16]\n.\nTherefore, this study aims to review the clinical course and management of pregnancies in patients with\ndeciduosis of the lower genital tract. We have included all the possible pregnancy complications and\nanalyzed the incidence of intervention-associated complications, symptoms, and lesion resolution according\nto the chosen management and, if lesions persisted, the size of the lesions and problems accompanying\nvaginal delivery. Summarizing these data, we describe the possible challenges, pregnancy complications,\nand management outcomes. Additionally, we discover knowledge gaps that might serve as a guide for\nsubsequent report descriptions.\nReview\nMethods\nSearch Strategy and Selection Process \nWe followed the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) guidelines\nreleased in 2020 to prepare a systematic review \n[17]\n. Our search strategy, using keywords such as deciduosis,\nectopic decidua, ectopic decidual reaction, and ectopic decidualization, was introduced in five databases\n(PubMed, PubMed Central (PMC), Europe PMC, ScienceDirect, and Google Scholar) and retrieved the\nreferences from inception to February 24, 2023. Complete queries and the search results are provided in\nTable \n1\n.\nDatabases\nQueries\nResults\nPubMed\nDeciduosis OR \"Ectopic decidua\" OR \"Ectopic decidual reaction\" OR \"Ectopic decidualization\" Filters: Case\nReports, Observational Study\n77\n a\nPMC\n((Deciduosis) OR \"Ectopic decidua\") OR \"Ectopic decidual reaction\") OR \"Ectopic decidualization\"\n159\nEurope PMC\n(\"Deciduosis\" OR \"Ectopic decidua\" OR \"Ectopic decidual reaction\" OR \"Ectopic decidualization\")\n215\nScienceDirect\n\"Deciduosis\" OR \"Ectopic decidua\" OR \"Ectopic decidual reaction\" OR \"Ectopic decidualization\"\n99\n b\nGoogle\nScholar\n\"Deciduosis\" OR \"Ectopic decidua\" OR \"Ectopic decidual reaction\" OR \"Ectopic decidualization\"\n980\nTABLE\n 1: The databases, search queries, and results\nThe initial search showed: \na\n139 results on PubMed (applied filters: case reports, observational study); \nb\n249 results on ScienceDirect (applied filters: case\nreports, research articles)\nPMC: PubMed Central\nFollowing the search, we uploaded the citations into the EndNote software (Clarivate, Philadelphia, PA,\nUSA) and shared them among the authors (ZB, HD, MS). Then, two reviewers (ZB and HD) independently\nproceeded with the identification and screening process. They were consulted by a third author (MS)\nwhenever disagreements arose. After removing the duplicates, non-English, and irrelevant records, full-text\narticles were retrieved and reviewed for eligibility criteria, as provided in Table \n2\n.\n2023 Bolgarina et al. Cureus 15(8): e44479. DOI 10.7759/cureus.44479\n2\n of \n13\n\nInclusion criteria\nExclusion criteria\n1. Population: Pregnant women with lower genital tract deciduosis.\n1. Associated ectopic or molar pregnancy\n2. Phenomenon of interest: Clinical course of the pregnancies and unique difficulties\n2. Type of studies: non-primary studies\n3. Outcomes: Pregnancy complications and management-related outcomes\n3. ‘Grey’ literature, including unpublished studies\n4. Types of studies: Case reports, case series, and case-control studies\n4. Animal studies\n5. Human studies\n \n6. Time: Published from the inception to February 24, 2023.\n \n7. Location: Worldwide\n \n 8. Language: Articles written in English\n \n 9. Free full-text articles\n \nTABLE\n 2: Full eligibility criteria\nBesides, checking reference lists retrieved three additional studies for the analysis. During the whole\nprocess, we did not use any automatic tools. The entire process is shown in Figure \n1\n.\nFIGURE\n 1: PRISMA flow diagram on the selection of studies\nPRISMA: Preferred Reporting Items for Systematic Reviews and Meta-Analysis; PMC: PubMed Central\nData Collection and Synthesis\nThe same reviewers extracted data to a Microsoft Excel (Microsoft Corporation, Redmond, WA, USA) file\nindependently according to the previously chosen variables and outcomes, which were as follows: (1)\npatients' characteristics included age, parity, preexisting history of cervical pathology and its treatment, and\nthe results of cervical cancer screening; (2) clinical features included location, form, and size of the lower\ngenital tract deciduosis, course of previous pregnancies, clinical presentation while establishing the\ndiagnosis, and differential diagnosis; (3) management-related parameters: weeks of gestation (WG), results\nof the cytology and colposcopy and associated challenges, interventions (biopsy, polypectomy or removal of\npolypoid masses, or their combinations), and postpartum follow-up results within eight weeks; and (4)\noutcomes: (a) pregnancy complications, such as significant hemorrhages, pregnancy loss, intraamniotic\n2023 Bolgarina et al. Cureus 15(8): e44479. DOI 10.7759/cureus.44479\n3\n of \n13\n\ninfection (IAI) and/or preterm rupture of the membranes (PROM), preterm birth, cesarean section, etc.; and\n(b) management-associated results (intervention-associated complications, symptom resolution or\npersistence according to the chosen management, spontaneous regression of lesions during observation,\nand, if lesions persisted, their size and vaginal birth outcomes).\nWe made certain assumptions whenever information could not be recovered or clarified. If the authors\nreported multiple elevations on the cervix, we suspected an \ninfiltrative form\n of deciduosis \n[18,19]\n. When the\nauthors described the lesion as a single ectocervical or vaginal \"mass,\" we interpreted it as a \npolypoid form\n of\nectopy \n[20-22]\n. We recognized the case of having a decidual polyp during the previous pregnancy as\na \npolyp \n[23]\n. In one case, it was hard to differentiate a polyp from a potentially papillary form of deciduosis\nor expulsed fragments of the decidua described as the endocervical \"grayish membranes\" \n[24]\n. If\n vaginal\nbleeding\n was not followed by any emergent workup, we considered it insignificant. Additional workup to rule\nout differential nosologies and concerns mentioned in the discussion sections of the articles contributed to\nthe \ndifferential diagnosis\n analysis. We also recalculated the patient’s \nage \nbased on the provided chronology\n[25]\n and the \nterm of gestation\n based on the last menstrual period day or, if unavailable, the confinement date\nor size of uterus enlargement \n[22,23]\n. The \n\"reassuring\" result of cytology\n was interpreted as normal \n[19]\n. Given\nthe diverse terms of gestation and circumstances of revealing the pathology, we \ncategorized pregnancies as\nmanaged expectantly\n if: (1) there were no planned interventions \n[24,26]\n; (2) patients underwent urgent or\nemergent procedures \n[27,28]\n; (3) unprovoked pregnancy complications led to pregnancy termination \n[20,25]\n;\nor (4) patients presented initially after 37WG \n[15,16,21,23]\n. When reports included a scheduled remote\npostpartum follow-up for cervix re-evaluation within eight weeks, we suspected \nlesion persistence\n \n[19,22,28]\n.\nWe excluded underreported and unrestored information from the corresponding analysis, marking them as\n\"not reported\" or \"not applicable.\" The extracted data is organized in Tables \n3\n-\n5\n. \nAuthor, Year\n(Country)\nAge\nParity\nPreexisting cervical\npathology/treatment and cancer\nscreening\nLocation\nForm (size)\nSymptoms at the time of\nrevealing the pathology\nPreexisted\npregnancy\ncourse\nDifferential diagnosis\nOh et al., 2022\n(Korea) \n[20]\n33\n0\nNR\nUpper third of the\nvagina\nPolypoid (RV DIE\n6.1×4.1 cm)\nRecurrent VB\nRecurrent VB\nVaginal cancer, DIE\nmalignization\nBatkoska et al., 2022\n(Slovenia) \n[25]\n27\n0\nCIN3/LLETZ 5Y prior\nCervix\nPolyp (2.2×1 cm)\nVB\nNR\nNone\n28\n0\nCIN3/LLETZ 6Y prior\nCervix\nPolyp (4×1 cm)\nMalodorous VD\nNR\nNone\n29\n0\nCIN3/LLETZ 7Y prior\nCervix\nPolyp (4×2 cm)\nResolved VB\nNR\nNone\nVerma et al., 2022\n(India) \n[18]\n28\n2\nNR\nCervix\nInfiltrative (0.5-to-1.5\ncm)\nResolved VB\nNR\nCervical cancer\nButtery et al., 2021\n(Australia) \n[16]\n22\n0\nNR\nCervix\nPolypoid (large)\nNR\nNR\nPlacenta previa\nMangla et al., 2021\n(India) \n[27]\n28\n0\nNR\nCervix\nPolyp (2×2 cm)\nRecurrent VB\nRecurrent VB\nNone\nXiaoyin et al., 2018\n(China) \n[26]\n32\n1\nNR\nCervix\nPolyp (0.8 cm)\nVB\nNR\nNR\n25\n0\nNR\nCervix\nInfiltrative/Polypoid (1\ncm)\nVB\nNR\nNR\n27\n0\nNR\nCervix\nUlcerated + Polyp\nVB\nNR\nNR\n37\n1\nNR\nCervix\nPolyp (2 cm)\nRecurrent VD/VB\nRecurrent\nVD/VB\nNR\n28\n0\nNR\nCervix\nInfiltrative/Polypoid (2\ncm)\nVB\nNR\nNR\n22\n0\nNR\nCervix\nPapillary + Polyp\nRecurrent VD/VB\nRecurrent\nVD/VB\nNR\n37\n0\nNR\nCervix\nPolypoid (1.5 cm)\nVD/VB\nNR\nNR\n29\n0\nNR\nCervix\nInfiltrative + Polyp (2\ncm)\nRecurrent VB\nRecurrent VB\nNR\n27\n0\nNR\nCervix\nUlcerated/Polypoid\n(1.5 cm)\nRecurrent VB\nRecurrent VB\nNR\n26\n0\nNR\nCervix\nPolyp (1 cm)\nVB\nNR\nNR\n2023 Bolgarina et al. Cureus 15(8): e44479. DOI 10.7759/cureus.44479\n4\n of \n13\n\n23\n0\nNR\nCervix\nPapillary/Polypoid (2\ncm)\nRecurrent VB\nRecurrent VB\nNR\nvan Diepen et al.,\n2015 (Netherlands)\n[19]\n34\n0\nCIN3/LLETZ 1Y prior; Cytology:\nNormal 0.5Y and 1Y prior\nCervix\nInfiltrative (0.5-to-1.5\ncm)\nVB\nNR\nPolyps, cervical adenoma\nOladipo et al., 2005\n(UK) \n[28]\n28\n0\nInsignificant history; Cytology:\nNormal 1Y prior\nCervix\nPolypoid (4×4 cm)\nVB of “300 ml”\n“Uneventful”\nPlacenta previa or\nabruption\nGornall et al., 2000\n(UK) \n[15]\n23\n0\nCytology: Normal 2Y prior\nCervix\nPolypoid (8 cm)\nPROM\n“Offensive” VD\nCervical cancer  \nArmenia et al., 1964\n(USA) \n[23]\n22\n2\nUnremarkable cervix at 24WG\nCervix\nPolypoid\nNone\nUnremarkable\nPrimary reticulum cell\nsarcoma\n22\n3\nPrevious pregnancy: 1-cm decidual\npolyp\nCervix\nPolyp (6 cm)\nVB\nVB\n“Undifferentiated tumor of\nreticuloendothelial origin”\nOrr et al., 1961\n(Northern Ireland) \n[29]\n26\n1\n“Erosion”/Diathermy\nCervix + Upper\nthird of the\nvagina\nPolypoid (large)\nRecurrent VB\nRecurrent VB\nPlacenta previa, carcinoma\nMathie, 1957 (UK) \n[30]\n39\n2\nNR\nUpper third of the\nvagina\nPolypoid (large)\nNone\n“Uneventful”\nCervical, then vaginal\ncarcinoma\nSpivack, 1949 (USA)\n[24]\n31\n1\nNR\nCervix\nInfiltrative + \"grayish\nmembranes\" (1.5 cm)\nWorsened leukorrhea\nLeukorrhea\nMiscarriage, ectopic\npregnancy\nLapan, 1949 (USA)\n[22]\n33\n0\n“Normal”\nCervix + Upper\nthird of the\nvagina\nPolypoid (large)\nProfuse VB\nVB worsening\nCervical cancer\n35\n1\nNR\nUpper third of the\nvagina\nPolypoid (large)\nNone\nNR\nVaginal cancer\n31\n1\n“Normal”\nCervix\nPolypoid (large)\nNone\nNR\nAnaplastic cancer\nKlein et al., 1946\n(USA) \n[21]\n23\n0\n12WG: No polyps, \"erosions\", or\ngrowths\nCervix\nPolypoid (2 cm)\nVB (“cupful”)\n\"Uneventful\"\nPlacenta previa, cervical\ncancer\nTABLE\n 3: Data extraction of the clinical characteristics\nNR: not reported; RV: rectovaginal; DIE: deep infiltrative endometriosis; VB: vaginal bleeding; CIN3: cervical intraepithelial neoplasia (grade 3); LLETZ:\nlarge loop excision of the transformation zone; Y: year(s); VD: vaginal discharge; PROM: preterm rupture of membranes; WG: weeks of gestation\nAuthor, Year\n(Country)\nWG\na\nCytology\nColposcopy\nBiopsy(ies)\nEctomy of\nlesions\nPostpartum\nfollow-up within\n8 weeks\nOh et al., 2022\n(Korea) \n[20]\n34WG\nNR\nNR\nN/A (Active VB)\nNone\nNR\nBatkoska et al.,\n2022 (Slovenia)\n[25]\n6WG\nNR\nPolyp\nNone\nPolypectomy\nN/A (Removed)\n10WG\nNR\nNR\nNone\nNone\nN/A (Removed vs.\nRegressed)\n7WG\nNR\nNR\nNone\nPolypectomy\nN/A (Removed)\nVerma et al., 2022\n(India) \n[18]\n32WG\nNR\nNR\nBiopsy\nNone\nSpeculum:\nNormal; Cytology:\nNILM\nButtery et al., 2021\n(Australia) \n[16]\n41WG\nNR\nNR\nNone (Intraoperative)\nNone\nNR\nMangla et al., 2021\n(India) \n[27]\n20WG\nNR\nNR\nNone\nUrgent\npolypectomy\nN/A (Removed)\n2023 Bolgarina et al. Cureus 15(8): e44479. DOI 10.7759/cureus.44479\n5\n of \n13\n\nat 24WG\nXiaoyin et al., 2018\n(China) \n[26]\n8WG\nNILM\nPolyp\nNone\nNone\nN/A (Regressed)\n21WG\nNILM\nEctopy\nBiopsy\nPolypectomy\nb\nat 24WG\nN/A (Removed)\n19WG\nNILM\nPolyp/Ectopy\nBiopsy\nNone\nN/A (Regressed)\n10WG\nNILM\nPolyp\nBiopsy\nPolypectomy\nat 12WG\nN/A (Removed)\n15WG\nNILM\nEctopy\nBiopsy\nNone\nNR\n16WG\nNILM\nPolyp/Ectopy\nBiopsy\nPolypectomy\nb\nat 19WG\nN/A (Removed)\n7WG\nNILM\nEctopy\nBiopsy\nPolypectomy\nb\nat 12WG\nN/A (Removed)\n23WG\nNILM\nPolyp/Ectopy\nBiopsy\nPolypectomy\nb\nat 27WG\nN/A (Removed)\n32WG\nNILM\nEctopy\nBiopsy\nNone\nNR\n8WG\nNILM\nPolyp\nNone\nNone\nN/A (Regressed)\n28WG\nNILM\nEctopy\nBiopsy\nPolypectomy\nb\nat 31WG\nN/A (Removed)\nvan Diepen et al.,\n2015\n(Netherlands) \n[19]\n11WG\nNormal  \nAcetic acid: \"no abnormalities\"\nColposcopy-guided\nbiopsy at 25WG\nNone\nSpeculum: Almost\nresolved;\nCytology: Normal\nOladipo et al.,\n2005 (UK) \n[28]\n28WG\nNR\nUnsatisfactory Acetic acid:\n“grayish-white”, fine\npunctuations, \"atypical vessels\"\nColposcopy-guided,\nmultiple biopsies\n(Urgent settings)\nNone\nColposcopy:\nNormal\nGornall et al., 2000\n(UK) \n[15]\n38WG\nNR\nNR\nBiopsies\nNone\nSpeculum:\nNormal;\nColposcopy:\nNormal\nArmenia et al.,\n1964 (USA) \n[23]\n39WG\nNR\nN/A (Old study)\nNR\nNone\nHysterectomy:\n0.1×0.8 cm nodule\n15WG\nNR\nN/A (Old study)\nBiopsy; Biopsies at\n17WG\nNone\nCold knife\nconization: normal\nOrr et al., 1961\n(Northern Ireland)\n[29]\n36WG\nNR\nN/A (Old study)\nBiopsy\nNone\nSpeculum: Almost\nresolved\nMathie, 1957 (UK)\n[30]\n28WG\nNR\nN/A (Old study)\nBiopsy\nNone\nSpeculum: Normal\nSpivack, 1949\n(USA) \n[24]\n10WG\nNR\nN/A (Old study)\nNone (pseudobiopsy\nc\n)\nNone\nSpeculum:\n\"Inflamed and\neroded”\nLapan, 1949\n(USA) \n[22]\n12WG\nNR\nN/A (Old study)\nBiopsy\nNone\nSpeculum:\n\"Erosion”; Biopsy:\nDiffuse\nadenomatosis\n12WG\nNR\nN/A (Old study)\nBiopsy\nNone\nSpeculum: Normal\n˂12WG\nNR\nN/A (Old study)\nBiopsy\nNone\nSpeculum: Normal\nKlein et al., 1946\n(USA) \n[21]\n41WG\nNR\nN/A (Old study)\nNone\nNone\nSpeculum:\n“Erosion”; Biopsy:\nNormal\n2023 Bolgarina et al. Cureus 15(8): e44479. DOI 10.7759/cureus.44479\n6\n of \n13\n\nTABLE\n 4: Data extraction of the management results\nWG: weeks of gestation; NR: not reported; N/A: not applicable; VB: vaginal bleeding; NILM: negative for intraepithelial lesion or malignancy; cm:\ncentimeter(s)\na\nThe column \"WG\" refers to the initial revealing of pathology. The term of gestation for delayed or additional procedures was mentioned additionally in the\ncorresponding fields.\nb\nThe term polypectomy included polypectomy and/or removal of the ectopic mass.\nc\nThe term pseudobiopsy means gentle tissue removal without obtaining a baseline layer.\nAuthor, Year\n(Country)\nPregnancy\ncomplications\nPregnancy course and immediate postpartum period\nVaginal birth:\ndelivered/complication\n(peripartum size of the lesions)\nPlanned procedure:\nUneventful/Complication\nObservation (including\nbiopsy)/Polypectomy: Symptom(s)\nresolution or recurrence\nRegression of lesions\n(if no ectomy was\nperformed)\nOh et al., 2022\n(Korea) \n[20]\nVB/Shock,\nUterine scar,\nPreterm birth\nN/A (Emergent settings)\nObservation: Recurrent VB\nN/A (Emergent C-\nsection)\nN/A (Emergent C-section)\nBatkoska et al.,\n2022 (Slovenia)\n[25]\nN/A (Remote\nmissed abortion)\nPolypectomy:\nUneventful\nPolypectomy: VB resolved\nN/A (Removed)\nN/A (Missed abortion)\nIAI/PROM, Late\npregnancy loss\nN/A (None)\nObservation: Recurrent VB and\nUTI\nN/A (Unknown:\nRemoved vs.\nRegressed)\nN/A (Late pregnancy loss)\nN/A\n(Polypectomy)\nPolypectomy:\nUneventful\nPolypectomy: VB resolved\nN/A (Removed)\nN/A (Removed)\nVerma et al.,\n2022 (India) \n[18]\nUterine scar,\nPreterm birth\nBiopsy: Uncontrolled VB\nObservation: NR\nN/A (Urgent C-\nsection)\nN/A (Urgent C-section)\nButtery et al.,\n2021 (Australia)\n[16]\nUterine scar\nN/A (Intraoperative)\nObservation: NR\nPersisted\nEarly stage of labor complicated by\nVB/fetal distress (entire posterior lip\nof the cervix)\nMangla et al.,\n2021 (India) \n[27]\nVB/Shock  \nN/A (Urgent\npolypectomy)\nObservation: Recurrent VB; Urgent\npolypectomy: NR\nN/A (Removed)\nN/A (Removed)\nXiaoyin et al.,\n2018 (China)\n[26]\nNR\nN/A (None)\nObservation: NR\nRegressed at 12WG\nN/A (Regressed)\nNR\nBiopsy: Uneventful;\nPolypectomy\na\n:\nUneventful\nPolypectomy\na\n: Recurrent VB\nresolution\nN/A (Removed)\nN/A (Removed)\nNR\nBiopsy: Uneventful\nObservation: Recurrent VB\nRegressed at 35WG\nN/A (Regressed)\nNR\nBiopsy: Uneventful;\nPolypectomy:\nUneventful\nPolypectomy: Recurrent VB\nresolution\nN/A (Removed)\nN/A (Removed)\nNR\nBiopsy: Uneventful\nObservation: Recurrent VB\nPersisted\nNR\nNR\nBiopsy: Uneventful;\nPolypectomy\na\n:\nUneventful\nPolypectomy\na\n: Recurrent VB\nresolution\nN/A (Removed)\nN/A (Removed)\nNR\nBiopsy: Uneventful;\nPolypectomy\na\n:\nUneventful\nPolypectomy\na\n: Recurrent VB\nresolution\nN/A (Removed)\nN/A (Removed)\nNR\nBiopsy: Uneventful;\nPolypectomy\na\n:\nUneventful\nPolypectomy\na\n: Recurrent VB\nresolution\nN/A (Removed)\nN/A (Removed)\n2023 Bolgarina et al. Cureus 15(8): e44479. DOI 10.7759/cureus.44479\n7\n of \n13\n\nNR\nBiopsy: Uneventful\nObservation: Recurrent VB\nPersisted\nNR\nNR\nN/A (None)\nObservation: NR\nRegressed at 20WG\nN/A (Regressed)\nNR\nBiopsy: Uneventful;\nPolypectomy\na\n:\nUneventful\nPolypectomy\na\n: Recurrent VB\nresolution\nN/A (Removed)\nN/A (Removed)\nvan Diepen et al.,\n2015\n(Netherlands)\n[19]\nN/A (C-section\ndue to failure to\nprogress)\nBiopsy: Uneventful\nObservation: NR\nPersisted\nN/A (Failure to progress)\nOladipo et al.,\n2005 (UK) \n[28]\nSevere VB\nN/A (Urgent settings)\nObservation: \"Without further\ncomplications\"\nPersisted\nDelivered\nGornall et al.,\n2000 (UK) \n[15]\nIAI/PROM,\nUterine scar\nBiopsies: Uneventful\nObservation: “Offensive” VD\nPersisted\nN/A (Emergent C-section)\nArmenia et al.,\n1964 (USA) \n[23]\nNone\nNR\nObservation: \"Uneventful\"\nPersisted\nDelivered (1.5×0.6×0.4 cm)\nNone\n15WG Biopsy:\nUneventful; 17WG\nBiopsies: Uneventful\nObservation: NR\nPersisted\nDelivered (2 cm)\nOrr et al., 1961\n(Northern\nIreland) \n[29]\nN/A (None)\nBiopsies: Uneventful\nObservation: Recurrent VB\nPersisted\nDelivered (large)\nMathie, 1957\n(UK) \n[30]\n“Mild toxemia”\nBiopsies: Uneventful\nObservation: Provoked VB\nPersisted\nIOL followed by mild VB (large)\nSpivack, 1949\n(USA) \n[24]\nN/A (None)\nN/A (Pseudobiopsy\nb\n)\nObservation: Recurrent\nVB/Persisted leucorrhea\nNR\nNR vs. Regressed?\nLapan, 1949\n(USA) \n[22]\nN/A (None)\nBiopsies: Uneventful\nObservation: Provoked profuse VB\nPersisted\nDelivered\nN/A (None)\nBiopsies: Uneventful\nObservation: \"Uneventful\"\nNR\nNR vs. Regressed?\nN/A (None)\nBiopsies: Uneventful\nObservation: NR\nPersisted\nDelivered\nKlein et al., 1946\n(USA) \n[21]\nProfuse VB\nEarly postpartum\nbiopsy: VB\nObservation: \"Uneventful\"\nPersisted\nDelivered (2 cm)\nTABLE\n 5: Data extraction of the outcomes\nVB: vaginal bleeding; N/A: not applicable; C-section: caesarean section; IAI: intraamniotic infection; PROM: preterm rupture of membranes; UTI: urinary\ntract infection(s); NR: not reported; WG: weeks of gestation; VD: vaginal discharge; cm: centimeter(s); IOL: induction of labor\na\nThe term polypectomy included polypectomy and/or removal of the ectopic mass.\nb\nThe term pseudobiopsy means gentle tissue removal without obtaining a baseline layer.\nAfter completing the data extraction, two reviewers (ZB and HD) critically appraised the studies\nindependently by using the CAse REport (CARE) assessment tool for case reports \n[31]\n and the Joanna Briggs\nInstitute (JBI) critical appraisal tools for case series \n[32]\n. The third reviewer (MS) resolved doubts and\ndisagreements. The studies that scored 70% or higher contributed to our systematic review. This threshold\nwas lowered to 60% for the studies published before 2013, as we did not want to lose clinically relevant\ninformation due to differences in the requirements of reporting cases.\nWe summarized clinically relevant qualitative variables by frequencies and presented them as percentages\nsupported by a numerator and denominator. Continuous variables were presented as the mean and range in\na normal data distribution and the median and interquartile range in a non-normal data distribution.\nWe did not develop the protocol and register the systematic review at the International Prospective Register\nof Systematic Reviews (PROSPERO) due to the short timeline for its completion. The protocol was\nsubstituted with data extraction tables in Microsoft Excel and subsequent analysis.\n2023 Bolgarina et al. Cureus 15(8): e44479. DOI 10.7759/cureus.44479\n8\n of \n13\n\nResults\nOur search strategy identified 1745 records. After removing the duplicates (n = 443), foreign language\narticles (n = 162), and, partially, irrelevant reports whenever filters could be applied (n = 212), 928 records\nunderwent screening. The inability to retrieve four reports limited our assessment for eligibility to only 24\nrecords; of those, we excluded nine studies due to the wrong population (n = 5) \n[33-37]\n, wrong publication\ntype (n = 1) \n[8]\n, and incompleteness of the presented data for the analysis (n = 3) \n[14,38,39]\n. Finally, 15 of 17\nstudies scored from 63% to 100% during the critical appraisal process and were subject to analysis \n[15,16,18-\n30]\n; 14 case reports, and one case series describing 30 cases.\nPatients' Characteristics\nThe mean patient age was 28 years (± 4.86). Nulliparous women represented the majority - 18 of 30 women\n(60%). Among preexisting pathologies, there was a history of cervical intraepithelial neoplasia (grade 3)\ntreated with loop electrosurgical excision of the transformation (four cases), cervical \"erosion\" treated with\ndiathermy (one case), and recurrent decidual polyps (three cases).\nClinical Presentation\nLesions were localized at the cervix (83%, 25 of 30 reports), the upper third of the vagina (10%, three of 30\nreports), or both locations (7%, two of 30 cases). Among the 22 reported cases of ectopies (73%), 40%\naccounted for polypoid (12 cases), 10% were infiltrative (three cases), and 23% were coexisting forms (seven\ncases).\nClinically, deciduosis presented an accidental finding only in four of 29 patients (14%). The rest of the\npatients complained of leukorrhea or infectious vaginal discharge (21%, six of 29 women) and/or vaginal\nbleeding, which was the chief complaint in 22 of 29 women (76%) and had a recurrent character in more\nthan 16 of 24 patients (57%) based on the previous and subsequent course of the pregnancies.\nDifferential diagnoses included threatened abortion and ectopic pregnancy (one case), cervical adenoma\n(one patient), placenta previa (four cases), and cervical or vaginal malignancy (11 cases), including one\npatient with suspicious rectovaginal deep infiltrative endometriosis malignization.\nManagement\nDeciduosis was revealed before 24WG in 18 of 30 pregnancies (60%). All reported cytological findings were\nnormal. Only one study documented the details of colposcopy. According to that study, the possible\nchallenges are incomplete visualization of the transformation zone, \"grayish-white\" epithelium after\napplying acetic acid, and \"atypical vessels.\"\nEleven of the patients were managed expectantly (37%). The rest of the pregnant women underwent a total\nof 27 planned procedures: before 24WG (12 cases), during 24-to-33 6/7WG (seven cases), and after 34WG\n(eight cases). In the eight weeks postpartum, five of 12 patients (42%) had residual findings, including\n\"erosions\" and diffuse adenomatosis.\nOutcomes\nStudies reported the following complications of the pregnancies: significant antenatal bleeding (four\nstudies), late abortion (one case), IAI and/or PROM (two cases), preterm birth (two cases), and operative\ndelivery with uterine scar formation (four patients).\nAmong the 27 scheduled procedures, only one case of uncontrolled vaginal bleeding occurred at 32WG\n(0.04%).\nObservation, including pregnancies managed expectantly or biopsied, was followed by recurrent symptoms\n(vaginal bleeding, urinary tract infections, vaginal discharge) or uneventfully (none, single, or provoked\nepisode) in eight (53%) and seven (47%) women, respectively. Recurrent vaginal bleeding resolved in all\neight cases (100%) of performed polyp or polypoid mass ectomies.\nSpontaneous regression of lesions during observation happened in 20% (three of 16 patients) at 12WG,\n20WG, and 35WG. All of these lesions were polyps, including one case of ulcerated ectopy.\nNine studies reported lesion persistence until vaginal birth. Almost all of these women had large polypoid\nlesions during their pregnancies. Among them, six women entered labor with the reported size of the lesions\nranging from 1.5-2 cm to \"large.\" None of the cases were complicated with significant intrapartum\nhemorrhage, although one study reported fetal distress of unknown etiology in the early stage of labor.\n2023 Bolgarina et al. Cureus 15(8): e44479. DOI 10.7759/cureus.44479\n9\n of \n13\n\nDiscussion\nDeciduosis of the lower genital tract was observed in women of 22 to 39 years of age, appearing on the\ncervix (83%) and/or vaginal fornices. Unfortunately, most case reports did not report the prior history of\ncervical or vaginal pathologies, limiting the opportunity to suggest the recurrence nature of the pathology,\nalthough decidual polyps demonstrated the tendency \n[23,25]\n.\nWe noted that macroscopic cervix and/or vaginal fornice deciduosis was most commonly symptomatic,\npresenting as recurrent painless vaginal bleeding in more than half of the patients. Three pregnancies with\nlesions exceeding 2 cm in diameter were complicated by spontaneous antenatal hemorrhage of over 250 ml\nbetween 24WG and 34WG \n[20,27-28]\n. According to the literature, the lesions start regression around 25WG\nand, in 60%-70%, disappear completely by 38WG \n[8]\n. Because of this, it seems reasonable to diagnose the\ncondition early, while ruling out cancer and locating the placenta, and consider respiratory distress\nsyndrome prevention, especially if a biopsy is unavoidable during the late term of gestation.\nReviewing a previous history of cervical dysplasia, its treatment, and subsequent surveillance, including\ncervical cytology obtained in the first trimester of pregnancy, might help establish the diagnosis while\nminimizing the risks associated with cervical biopsy. For example, in one study \n[19]\n, a patient underwent\ntreatment for cervical intraepithelial neoplasia (grade 3) with subsequent normal cytological results before\nconception. She developed an infiltrative form of deciduosis, followed by reassuring cytological plus\ncolposcopy surveillance until 25WG when the biopsy was performed because of the growth of the lesions.\nThis case highlights the importance of cervical cancer screening before 20WG \n[40]\n when the lesions do not\nobscure the transformation zone and awareness of the pathology (in this case, the infiltrative form does not\ninclude the transformation zone and is multifocal form, therefore can allow avoiding a biopsy). Moreover,\nthe absence of a high-grade pattern, surrounding foci of lower-grade abnormalities, and intensive necrosis\nof the lesions during colposcopy might help to avoid biopsy if local guidelines do not require it, regardless of\nthe dense whitening of the lesions. However, it is the polypoid form that seems to be the most problematic\none from a differential perspective and the most reported form of ectopy (58%), as it includes a\ntransformation zone. In addition to taking a history and performing the mentioned diagnostics, this form of\ndeciduosis presents as confusingly friable compared to relatively solid cancer. The less frequently reported\nform of ectopy was the papillary one, which may be the less recognizable form of deciduosis, as likely shown\nin an example \n[24]\n. Still, in this case, expulsed fragments of the decidua are a suitable explanation too.\nPrevious studies noted that performing a biopsy of the cervix during pregnancy is generally safe in terms of\nthe occurrence of significant bleeding or pregnancy complications \n[40-42]\n. We obtained similar findings\nbased on 27 electively performed procedures before 24WG (44%), during 24-to-33 6/7WG (26%), and after\n34WG (30%). Our analysis also reflects the relative safety of biopsy decidual lesions that might be more\nprone to hemorrhages due to their friability, frequent association with chronic inflammation, and\nsusceptibility to necrosis \n[24]\n, especially with pregnancy progression \n[8]\n. For instance, the included studies\nreported severe antepartum hemorrhage \n[20,27,28]\n, profuse bleeding provoked by gynecologic evaluation\n[21]\n, and biopsy-induced uncontrolled bleeding \n[18]\n in women between 24-41 WG. These lead to two\nconclusions: (1) a cervical biopsy or stiff brush procedure is needed if there are any doubts regarding cervical\ncancer \n[11]\n, and (2) respiratory distress prophylaxis may be considered if the procedure is planned after\n24WG. Lately, remote studies have indicated that misleading results might lead to unnecessary procedures\nsuch as cervical conization during pregnancy \n[39]\n, postpartum hysterectomy, and cold knife conization \n[23]\n.\nThe authors cite that cervical and/or vaginal deciduosis does not require special pregnancy management\nunless complications develop. However, there are a few considerations. First, a retrospective cohort study of\n550 pregnant women with cervical polyps noted that 45.45% of cases accounted for decidualized ones, which\nled to a greater frequency of pregnancy complications than non-decidualized polyps (28.1% vs. 6.1%) \n[43]\n.\nThus, managing decidualized polyps in certain circumstances can be distinct from those without changes.\nSecond, decidual polyp or polypoid mass ectomies might be helpful in resolving recurrent symptoms,\nthough it is uncertain who might benefit from the procedure and whether resection reduces the risk of\npregnancy complications. Perhaps pregnant women over 11WG with a polyp width greater than 11 mm,\nrecurrent vaginal bleeding (in our opinion, recurrent inflammatory discharges in the setting of either\nmulticolored or fragile lesions), and visualized roots could be good candidates \n[25,44-46]\n. At the same time,\none study noted that spontaneous regression of the lesions, which is common, did not reduce the risk of\ncervical insufficiency and pregnancy loss \n[13]\n. Again, the authors did not evaluate these outcomes based on\nthe histological type of the polyps. Lastly, one case report showed a 6-to-2 cm polyp size reduction in two\nmonths on local treatment with Neosporin \n[23]\n. Although a natural regression process can explain it, similar\noptions could be considered when the risk of symptomatic lesion removal is high (e.g., non-visualized roots)\nand infected and edematous lesions are suspected.\nIn labor, macroscopic polypoid lesions, especially large ones (e.g., covering the entire posterior lip or an 8-\ncm mass), are inflamed, and those that are initially revealed in the peripartum period concern providers\nregarding intrapartum bleeding and its differential diagnosis \n[15,16]\n. However, at least six old studies with\nthe size of lesions between 1.5 and large cm were not complicated by significant intrapartum hemorrhage. In\none report, the 2-cm polypoid lesion remained \"silent\" even in the early postpartum period until biopsy \n[21]\n.\nFinally, accelerated regression of cervical deciduosis is to be expected shortly after delivery and complete\n2023 Bolgarina et al. Cureus 15(8): e44479. DOI 10.7759/cureus.44479\n10\n of \n13\n\nlesion resolution by six weeks after delivery. According to our analysis, some residual findings are possible.\nLimitations\nOur systematic review has a few limitations. There is a risk of publication bias associated with reporting only\nrecognized and successfully managed cases. Of note, older studies reported mismanagement issues more\nopenly. Although improved antenatal management and advanced diagnostic opportunities might explain it,\nthe rarity of macroscopic lesions, the lesions' ability to appear at a later term of gestation, and uncertainty\nin managing some situations perplex providers \n[16,18]\n. We analyzed only studies published in English, which\ncould lead to losing important information. Besides, some studies mainly described cases around the episode\nof active management, missing the information essential for a thorough evaluation and, consequently,\nlimiting current knowledge on the diagnostic pitfalls that can be addressed in prospective case reports. We\nalso recovered or clarified some missed and descriptive data (discussed in the Materials and Methods\nsection) that unlikely impacted our results significantly. Of course, considering the rarity of the condition,\nwe could include only studies with a lower grade of evidence. However, we covered an important segment\nuseful for providers and prospective studies.\nConclusions\nWe described the clinical course, all pregnancy complications, and management-associated outcomes in\npregnant women with macroscopic cervical and/or vaginal deciduosis. Most frequently, it presented as\nrecurrent vaginal bleeding episodes that can be substantial after 24WG, requiring the exclusion of placenta\nprevia, cervical pregnancy, and malignancy, as well as consideration of respiratory distress syndrome\nprophylaxis (for instance, in cases of large lesions, planned procedures, and successfully treated\nhemorrhagic episodes). Pregnancy complications included significant antenatal hemorrhages, PROM,\nuncontrolled bleeding-associated late abortion or preterm birth, and operative delivery. Performing cervical\ncancer screening before 20WG, recognizing different forms of deciduosis, and confusing the friability of the\nlesions can minimize the risks related to performing a biopsy, which seems avoidable in most situations.\nHowever, a biopsy is necessary if there is any doubt regarding cancer (or guideline requirements), and it\nseems safe to biopsy decidualized lesions, especially before 24WG. Conservative management of the cases is\ncommonly accepted, while the benefits and indications of the excision of the lesion require further\nevaluation. There are no reports of associated significant intrapartum hemorrhages in women with polypoid\nectopies over 1.5 cm, which is a safe delivery route, although it might be individualized in some settings.\nThis review can serve as a standardized guideline for reporting all pertinent information in future case\nreports, filling the knowledge gap on diagnostic and management challenges in the context of current\ntechnological advancements and prenatal care standards. It can also be a quick source of information in the\nsearch for similar cases. There is a need to report cases of management concerning decidualized lesions in\nlabor, regardless of their successful and complicated resolution. An interesting direction is investigating the\ndebatable problem of managing cervical polyps in the presence or absence of decidualized changes.\nAdditional Information\nDisclosures\nConflicts of interest:\n In compliance with the ICMJE uniform disclosure form, all authors declare the\nfollowing: \nPayment/services info:\n All authors have declared that no financial support was received from\nany organization for the submitted work. \nFinancial relationships:\n All authors have declared that they have\nno financial relationships at present or within the previous three years with any organizations that might\nhave an interest in the submitted work. \nOther relationships:\n All authors have declared that there are no\nother relationships or activities that could appear to have influenced the submitted work.\nReferences\n1\n. \nKim HJ, Kim MJ, Song JY, Kim MR, Kim YT: \nThe prevalence of deciduosis in fertile women during cesarean\ndelivery\n. 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Early Hum Dev. 2017,\n104:33-7. \n10.1016/j.earlhumdev.2016.11.007\n2023 Bolgarina et al. Cureus 15(8): e44479. DOI 10.7759/cureus.44479\n13\n of \n13","source_license":"CC0","license_restricted":false}