{"paper_id":"bae720f1-406b-4d50-8fe8-e853af980c41","body_text":"INTERNATIONAL JOURNAL OF SURGICAL CASE REPORTS | ISSN 2674-4171  \n  \n \nAvailable online at www.sciencerepository.org  \n \nScience Repository \n \n \n \n \n \n*Correspondence to: Maria del Pilar Marín Sánchez, Servicio de Obstetricia y Ginecología, Hospital Clínico Universitario Virgen de la Arrixaca, El Palmar, Ctra. \nMadrid-Cartagena, s/n, 30120, Murcia, Carretera Cartagena, Murcia, s/n, 30170 El Palmar, Spain; E -mail: Ibsyna@hotmail.com \n© 2023 Maria del Pilar Marín Sánchez. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits \nunrestricted use, distribution, and reproduction in any medium, provided the orig inal author and source are credited. Hosting by Science Repository.  \nhttp://dx.doi.org/10.31487/j.IJSCR.2023.01.03  \nResearch Article \nIntravascular Involvement in Adenomyosis: An Endometriosis Dissemination \nPathway? \nJ. Pérez Buendía, M.P. Marín Sánchez *, A.R. Guijarro Campillo, J.R. Pérez Vidal, I. Ñiguez Sevilla, E. Iborra Laca l, \nM.C. Carrascosa Romero and A. Nieto Díaz  \nServicio de Obstetricia y Ginecología, Hospital Clínico Universitario Virgen de la Arrixaca, El Palmar, Murcia, Spain \nA R T I C L E  I N F O \nArticle history: \nReceived: 14 February, 2023 \nAccepted: 28 February, 2023 \nPublished: 7 April, 2023 \nKeywords: \nUterus \nadenomyosis \nvascular involvement \n A B S T R A C T \nIntravascular adenomyosis is an entity described for years in the literature, but its clinical -pathologic \ncorrelation hasn´t been studied in detail.  \nObjective: To study the prevalence of adenomyosis including intravascular involvement, as well as \navailable literature review about this pathology.  \nMaterial and Methods: A retrospective observational study has been conducted in our hospital analysing \nhysterectomies specimens from January to December 2020.  \nResults: 447 hysterectomies specimens have been analysed, 68 with adenomyosis and just one with \nintravascular involvement. \nConclusion: Intravascular involvement adenomyosis is a rare entity but with potential etiopa thogenic \nconsequence. \n \n                                                        © 2023 Maria del Pilar Marín Sánchez. Hosting by Science Repository . \nIntroduction \n \nAdenomyosis is a benign uterus condition, defined as the presence of \nectopic endometrial glands and stroma within  the myometrium. \nPrevalence report is around 1 to 70%. This wide range show the lack of \nconsensus on standard diagnostic criteria , both imaging  studies and \nhistopathology analysis [1]. Number of diagnostic strategies are being \ndeveloped in the last few years, specially focus on imaging studies like \ntwo or three dimensional transvaginal ultrasound and magnetic \nresonance imaging. In the same way, it aims to set universal standards \naccording to imaging studies findings. \n \nIt´s an ordinary condition, but its etiology and natural history remains \nunknown now. There are four pathogenesis theories proposed: the first \none (most widely accepted) explains a m yometrial invasion from \nendometrial tissue. It remains unknown the motivation of this irruption, \nhowever it might be related to prior pregnancies (angiogenesis and \ntrophoblast invasion), surgeries or immunological abnormal activity \namong endometrial-myometrial junction (endometrial tissue break -in \nmyometrium during regeneration, healing process and re-epithelization) \n[2]. Another second theory claims that adenomyosis is the result of \nembryonic pluripotent Müllerian  remnants. This belief come from \nadenomyosis finding out of myometrial thickness (recto-vaginal septum) \nin a Rokitansky -Kuster-Hauser syndrome patient, thus, without \nfunctional endometrium [3]. \n \nAn additional one hypothesis states that adenomyosis would be the \nconsequence of endometrial basal invag ination throughout \nintramyometrial linfatic system. Finally, there is the assumption that this \npathology might be arise from bone marrow stem cells. It is not clear \nwhy adenomyosis appears in some women and not in others to date, \nthough, it has been noted ultrastructural differences at smooth muscle \ncells level in adenomyosis uterus compared to normal [4]. \n \nMoreover, it has been recognized several risk factors that would increase \nthe incidence, among which is hiperestrogenic environment (early \nmenarche, bod y mass index, Tamoxifen previous treatment, oral \ncontraceptives, …). As well it has been associated with parity and \nprevious uterine surgeries. On the other hand, it seems to be that women \nsmoking would have lower risk [5]. As for the treatment, histerectomy \n\nIntravascular Involvement in Adenomyosis: An Endometriosis Dissemination Pathway?               2 \n \nInt Jour Surg Case Rep doi:10.31487/j.IJSCR.2023.01.03     Volume 5(1): 2-3 \nis the currently definitive management, although there are various \nalternatives that can mitigate the symptoms. We provide medical \ntherapies on one side, non -steroidal anti -inflammatory drug, \ncontraceptive oral pills, gestagenics, and even Danazol or Gonadotropin-\nreleasing hormone agonist (GnRH). Similarly, there are several surgical \ntechniques among which are endometrial ablation, hysteroscopy, uterine \narterial embolization. \n \nSettle on the best treatment in each case is complex, because of \nheterogeneous symptoms and others gynaecological conditions \nassociated to adenomyosis, like endometriosis or myomas. From what, \nthe decision will be based on factors like age, sympton severity, \nreproductive desire and comorbidities. Focusing on involvement \nintravascular in adenomyosis, there aren´t many recent publications \nabout it, for what the limited data is available. Intravascular involvement \nadenomyosis has been found in a series of hysterectomies in 12.4% of \nthis adenomyosis uterus [6].  \n \nMaterials and Methods \n \nFor this review, anatomopathological reports from every hysterectomies \nprocedure at University Hospital Virgen Arrixaca were revised from \nJanuary to December 2020. It is therefore a retrospective observational \nstudy. The appearance or not of adenomyosis and, the presence or not of \nintravascular involvement in adenomyosis were the variables included. \nWithin the adenomyosis group, the age, uterine weight and concomitant \nendometriosis and/or oncological diseases have been analysed too.  \n \nResults \n \n447 hysterectomies procedures in our center. 68 (15.1%) have been \ndiagnosed like adenomyosis by pathologists. The mean age of these \nwomen was 51.57 years, and the mean uterine weight was 200.77 grams. \nEndometriosis was present in 29.41% (20) of adenomyosis, and 25 \nhysterectomies cases had been made in an oncological context (16 of \nwhom have been for endometrial adenocarcinoma, which represents the \n23.53% of adenomyosis). \n \nIn only one surgical specimen was detected intravascular involvement, \ninform like “perivascular / in travascular zones placement with growth \ntowards vascular lumen, always been endotelized in their surfaces \n(immunohistochemical techniques confirmed)” (Figure 1). “In other \nways, active endometrial mucosa had been identified within \nintramyometrial veins, su rrounded by red blood cells and plasma” \n(Figure 2). \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \nFigure 1: Adenomyosis with intravascular involvement. Focus covered by endometrium. (CD31 marker). \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \nFigure 2: Adenomyosis with intravascular involvement. Hematoxylin-eosin stain. \n \n\nIntravascular Involvement in Adenomyosis: An Endometriosis Dissemination Pathway?               3 \n \nInt Jour Surg Case Rep doi:10.31487/j.IJSCR.2023.01.03     Volume 5(1): 3-3 \nDiscussion \n \nIn 447 hysterectomies reviewed, we recognize 15.21% of adenomyotic \nuterus. Lower prevalence compared other reported series, since average \nfrequency was over 20 and 30% [1]. Adenomyosis and endometriosis \nwere considered part of the same entity until 1920s, but after define them \nlike different pathologies, several studies have enquired about the \nrelationship between them [7]. 29.41 % of adenomyosis ha ve \nendometriosis associated (adenomyosis intravascular involvement \nincluded), higher percentage compared with other studies reported, as \nthat of Di Donato et al., which discuss a 21.28% of correlation among \nthese two entities [7].  \n \nVascular involvement can lead to a malign misdiagnosed, this is why \npathologist knowledge of this entity is required. It might be mistaken for \nendometrial stromal sarcoma, which is distinguished by absence of a \nmacroscopic mass (unless an adenomyoma), as well as typical uterine \nadenomyosis appearance. In addition, stromal cells look atrophic in \nadenomyosis, in contrast to sarcoma expansive growth pattern which \ncould spreads outside uterus [6]. Conversely, there is another entity, \nintravascular leiomyomatosis, which is smooth musc le tumoral cells \nmass growing into uterine venous system. It comes along with a \nleiomyoma and, sometimes with adenomyosis. Smooth muscle cells \npresence would be the key of differential diagnosis with adenomyosis \nwith intravascular involvement [8]. \n \nDespite of have just diagnosed only an adenomyosis with intravascular \ninvolvement, it should be considered. It may not have clinical \nsignificance, but it might be speculated that intravascular tissue could \nshatter from vessel wall giving the meaning of brain and lung \nendometriosis, places where the two common histopathological \nendometriosis theories (retrograde menstruation theory and coelomic \nmetaplasia theory) cannot justify the endometrial tissue presence [6]. \nIntravascular growth could involve a potential way of disease \ndissemination. \nConflicts of Interest \n \nNone. \nREFERENCES \n \n1. Struble J, Reid S, Bedaiwy MA (2016) Adenomyosis: A Clinical \nReview of a Challenging Gynecologic Condition. J Minim Invasive \nGynecol 23: 164-185. [Crossref] \n2. Benagiano G, Habiba M, Brosens I (2012) The pathophysiology of \nuterine adenomyosis: an update. Fertil Steril 98: 572-579. [Crossref] \n3. Enatsu A, Harada T, Yoshida S, Iwabe T, Terakawa N (2000) \nAdenomyosis in a patient with the Rokitansky -Kuster-Hauser \nsyndrome. Fertil Steril 73: 862-863. [Crossref] \n4. Mehasseb MK, Bell SC, Brown L, Pringle JH, Habiba M (2011) \nPhenotypic characterisation of the inner and outer myometrium in \nnormal and adenomyotic uteri. Gynecol Obstet Invest  71: 217 -224. \n[Crossref] \n5. Parazzini F, Vercellini P, Panazza S, Chatenoud L, Oldani S et al. \n(1997) Risk factors for adenomyosis. Hum Reprod  12: 1275 -1279. \n[Crossref] \n6. Meenakshi M, McCluggage WG (2010) Vascular involvement i n \nadenomyosis: report of a large series of a common phenomenon with \nobservations on the pathogenesis of adenomyosis. Int J Gynecol Pathol \n29: 117-121. [Crossref] \n7. Di Donato N, Montanari G, Benfenati A, Leonardi D, Bertoldo V et al. \n(2014) Prevalence of adenomyosis in women undergoing surgery for \nendometriosis. Eur J Obstet Gynecol Reprod Biol  181: 289 -293. \n[Crossref] \n8. Hirschowitz L, Mayall FG, G anesan R, McCluggage WG (2013) \nIntravascular adenomyomatosis: expanding the morphologic spectrum \nof intravascular leiomyomatosis. Am J Surg Pathol  37: 1395 -1400. \n[Crossref]","source_license":"CC0","license_restricted":false}