{"paper_id":"dcb450b5-4fbf-490b-8aa2-ff722d9ff36c","body_text":"Introduction\n!\nBleeding events, especially in the subpartum and\npostpartum periods, together with complications\ndue to thromboembolisms and gestosis, are the\nmain causes of maternal morbidity and mortality\n[1–3].\nExtrauterine, intra-abdominal causes for pre- or,\nrespectively, postpartum bleeding are very rare\noccurrences with only very few typical factors\n[4]. Besides injuries due to accidents, ruptured\naneurysms and haemorrhagic deciduosis are con-\nsidered to be the main causes.\nDeciduosis – the presence of ectopic decidua – is a\nfrequently encountered constellation and occurs\nin the course of almost all pregnancies (85 –\n100 %). However, as a rule deciduosis remains\nasymptomatic and its detection is usually a coin-\ncidental finding on histology [5 –6]. Life-threaten-\ning complications such as massive intra-abdomi-\nnal bleeding are an absolute rarity.\nWe report on two cases of symptomatic, intra-ab-\ndominal bleeding due to a histologically con-\nfirmed deciduosis. On the basis of these two cases\nwe discuss the rare clinical picture of symptomat-\nic – in part life-threatening for mother and child –\nhaemorrhagic deciduosis in the prepartum and,\nhere reported for the first time, also in the post-\npartum period and illustrate the problems of\nmaking the diagnosis.\nCase Reports\n!\nCase 1: Massive intra-abdominal bleeding\ndue to erosion of the left pelvis wall\nby deciduosis\nThe 36-year-old primigravida presented in the\n25 + 2 week of pregnancy at the University Hospi-\ntal in Oldenburg with the diagnosis of an acute\nabdomen. On admission the patient exhibited\nubiquitous abdominal pain, a marked peritonism\nand, on sonography, copious free intra-abdominal\nfluids. The haemoglobin value on admission was\n8.5 g/dl but dropped within a short time to 5.2 g/\ndl. Induction of foetal pulmonary maturation\ncould no longer be initiated due to the fulminant\ncourse. After clinical exclusion of appendicitis the\nAbstract\n!\nThe term “deciduosis” is used to describe the se-\nvere pregnancy-associated occurrence of ectopic\ndecidua with a usually asymptomatic course. We\nreport on two cases of massive maternal intra-ab-\ndominal bleeding due to such symptomatic\nchanges. The complications arose at different\ntime points for the two cases: prepartum (26th\nweek of pregnancy) or, respectively, – reported\nhere for the first time – seven days postpartum.\nAs well as differential diagnostic aspects we de-\nscribe the management of the disease and its pos-\nsible effects on subsequent pregnancies.\nZusammenfassung\n!\nUnter dem Begriff „Deziduose“ wird das schwan-\ngerschaftsassoziierte Auftreten ektoper Dezidua\nverstanden, welches zumeist asymptomatisch\nverläuft. Berichtet wird über 2 Fälle mütterlicher\nintraabdomineller Massenblutungen aufgrund\ndieser hier symptomatischen Veränderung. Zu\nden Komplikationen kam es zu verschiedenen\nZeitpunkten in den jeweiligen Fällen: präpartal\n(26. Schwangerschaftswoche) bzw. – erstmals\nüberhaupt publiziert – 7 Tage postpartal. Neben\ndifferenzialdiagnostischen Aspekten werden das\nManagement der Erkrankung und mögliche Aus-\nwirkungen auf mögliche Folgegraviditäten dis-\nkutiert.\nSevere Maternal Pre- and Postpartum\nIntra-Abdominal Bleeding due to Deciduosis\nSchwere prä- und postpartale intraabdominale maternale Blutungen\naufgrund einer Deziduose\nAuthors D. W. Lüdders 1, R.-P. Henke2, M. Saba 1, L. Raddatz 1, A. Soliman 1,E .M a l i k1\nAffiliations 1 Department of Obstetrics and Gynaecology, University Hospital Oldenburg, Oldenburg\n2 University Hospital Oldenburg, Institute for Pathology, Oldenburg\nKey words\nl\" pregnancy\nl\" intra‑abdominal bleeding\nl\" deciduosis\nl\" childbed\nSchlüsselwörter\nl\" Schwangerschaft\nl\" intraabdominale Blutung\nl\" Deziduose\nl\" Wochenbett\nreceived 31. 12. 2014\nrevised 8. 2. 2015\naccepted 10. 2. 2015\nBibliography\nDOI http://dx.doi.org/\n10.1055/s-0035-1545876\nGeburtsh Frauenheilk 2015; 75:\n259–262 © Georg Thieme\nVerlag KG Stuttgart · New York ·\nISSN 0016‑5751\nCorrespondence\nPD Dr. Dörte W. Lüdders\nUniversity Hospital Oldenburg\nDepartment of Obstetrics\nand Gynaecology\nRahel-Strauss-Straße 10\n26133 Oldenburg\nDoerte.luedders@gmx.de\n259\nLüdders DW et al. Severe Maternal Pre- … Geburtsh Frauenheilk 2015; 75: 259 –262\nCase Report\nDeutschsprachige\nZusatzinformationen\nonline abrufbar unter:\nwww.thieme-connect.de/\nejournals/toc/gebfra\n\n\ndiagnosis was unclarified abdominal bleeding as indication for a\nlaparotomy with longitudinal incision.\nIntraoperatively intraperitoneal bleeding from an eroded artery\nin the region of the left pelvic wall was seen. Several groups of le-\nsions typical for deciduosis were seen in the vicinity of this pelvic\nwall and the left ovary so that the tentative diagnosis of preg-\nnancy-associated deciduosis was made intraoperatively. The in-\nflamed areas were removed and a subtle haemostasis initiated.\nAfter attaining complete stoppage of the bleeding, a sterile vagi-\nnal sonography was performed intraoperatively with visu-\nalisation of a normal-frequency foetal cardiac function and un-\naffected anterior wall placenta. Transabdominal sonography was\nperformed immediately postoperatively and gave the same unre-\nmarkable result. The patient was transferred to the intensive care\nunit for observation. In the course of a control examination four\nhours postoperatively a highly pathological CTG pattern (bird\nwings) was observed and led to the indication for an emergency\nCaesarean section. As expected for the gestational age, an ex-\ntremely immature female baby was delivered (820 g, Apgar 3/4/\n7, NapH: 7.19 BE: − 7 mmol/l) and immediately transferred to\nthe neonatal intensive care ward.\nAfter a short postoperative monitoring on the ICU the mother\nwas transferred to the maternity ward. Altogether, four erythro-\ncyte concentrates were administered during the course.\nAfter an initially stable period, on the fourth day of life the baby\nsuffered from massive intracranial bleeding in both hemispheres\nwith a lethal result.\nHistological work-up of the maternal samples taken intraopera-\ntively confirmed the tentative diagnosis of deciduosis ( l\n\" Figs. 1\nand 2).\nFig. 1 Connective tissue transformed by deciduo-\nsis from the vicinity of the ovary, some characteris-\ntic deciduosis nests are stained (haematoxylin and\neosin).\nFig. 2 Large bodied deciduosis cells with broad\ncytoplasm (arrow) and uniformly contoured nuclei\nare seen (haematoxylin/eosin).\n260\nLüdders DW et al. Severe Maternal Pre- … Geburtsh Frauenheilk 2015; 75: 259 –262\nGebFra Science\n\n\nCase 2: Intra-abdominal bleeding from areas\nof deciduosis in the vicinity of the uterine border\non the seventh postoperative day after primary\nCaesarean section for total placenta praevia totalis\nin the 35 + 1 week\nA 39-year-old women (II gravida 0 para) presented in the 35 + 1\nweek of pregnancy for a planned Caesarean section due to the ex-\nistence of a placenta praevia totalis and transverse foetal position.\nA conspicuous intraoperative finding was that the uterus was\ntwisted by 120°. The left tube was displaced caudally in the vicin-\nity of the uterine cervix. It was not possible to untwist the preg-\nnant uterus and so an oblique incision was made into the accessi-\nble part of the uterus. Delivery of the baby was free of problems.\nAfter removal of the placenta, the uterus could easily be twisted\nback into its normal anatomic position. Inspection of the adnexa\nand uterine cervix revealed small endometrial lesions. The fur-\nther surgical course was unremarkable.\nOn the 7th postoperative day the patient developed very severe\npain in the vicinity of her left flank. After exclusion of an ascend-\ning urinary tract infection and renal congestion, sonography\ndemonstrated increasing amounts of free intra-abdominal fluids.\nIn addition, a progressive decrease in haemoglobin from 10.1 g/dl\nto 5.8 g/dl within a period of 13 hours was noted, so that – on the\nassumption of intra-abdominal bleeding – revision by way of an\ninfraumbilical longitudinal laparotomy was undertaken. The sus-\npicion of massive bleeding was confirmed intraoperatively. The\nbleeding site was identified as a conspicuous area in the region\nof the left dorsal uterine wall that was macroscopically compati-\nble with deciduosis. A tissue sample was taken, followed by\nstaunching of the bleeding; in addition two erythrocyte concen-\ntrates were administered perioperatively. Histological work-up\nof the tissue sample confirmed the tentative diagnosis of decid-\nuosis.\nThe subsequent postoperative course was unremarkable. The pa-\ntient was released home seven days after the revision operation.\nDiscussion\n!\nDeciduosis is a mostly pregnancy associated, large-cell stromal\nreaction which can occur as ectopic deciduosis in the sera and\nmucosa of many different organs [7–8]. It can, however, also arise\nfrom a secondary decidual transformation of pre-existing extra-\ngenital endometrial lesions [9 –11].\nIt only very rarely occurs in the physiological cycle events outside\nof pregnancy, as it is regulated by the action of ovarian and\nplacental hormones, in particular, the permanently high proges-\nterone level in pregnancy. If deciduosis occurs outside of preg-\nnancy, then above all the progesterone produced in the adrenal\ngland is responsible for it [5]. During pregnancy the decidua are\nformed from endometrial stromal cells under the influence of the\nrespective hormones and thus represent a physiological reaction\nto the progesterone stimulation caused by pregnancy. The ear-\nliest reported occurrence of deciduosis was observed on the ova-\nry in the 9th week of pregnancy [7]. Regression of deciduosis be-\ngins towards the end of the pregnancy and generally occurs over\na period of 4 to 6 weeks [5]. Deciduosis-like changes have, how-\never, also been described during a postpartum period of up to\nseveral months [12]. This postpartum regression possibly ex-\nplains, as described in our case, the acute postpartum haemor-\nrhagic deciduosis. In this case it is possible that an insufficiently\nadvanced regression could have triggered the late massive decid-\nual bleeding. On the other hand another possible explanation for\nthe massive bleeding could also be hormone withdrawal caused\nby the end of the pregnancy. Both are hypotheses that have not\nbeen previously reported in the literature and that also cannot\nbe clarified for our cases. Even so, this is the first report to de-\nscribe a severe decidual bleeding in childbed.\nHowever, as a general rule, deciduosis remains asymptomatic\nand its detection is usually an incidental finding on histology of\nperitoneal biopsies taken during Caesarean sections. Also in most\ncases it does not have any clinical consequences. The patho-\ngenesis of deciduosis has not yet been completely clarified.\nThere are various hypotheses but none of them have been un-\nequivocally clarified. On the one hand it is discussed that a decid-\nuosis can develop on the basis of a pre-existing endometriosis.\nOn the other hand a de novo reaction of pluripotent submesothe-\nlial stromal cells has been proposed as the cause of deciduosis\n[13].\nFrom the embryological point of view, intraembryonal coelom\nepithelium is the source tissue of not only endometrial stroma\nbut also of submesothelial stroma. Intraembryonal coelom epi-\nthelium is, in addition, the source tissue of serosa of the pleura\nand pericardium as well as of the peritoneal cavity. This can be\ndeduced from the widely differing localisations of deciduosis le-\nsions that can become apparent through the corresponding local-\nisation-dependent clinical symptoms [4, 13].\nIn its clinical spectrum of symptoms, deciduosis may rather ex-\nhibit unspecific changes in varying degrees of severity. The range\nof clinical symptoms can thus vary from shortage of breath, hae-\nmoptyses, pneumothorax, ileus symptomatics, progressive anae-\nmia and increasing infection parameters through to the most se-\nvere and massive intra-abdominal bleeding with acute abdomen.\nHowever, these life-threatening complications are very rare and\nonly sporadic cases have been described in the literature [9, 11,\n13–15].\nMassive intra-abdominal bleeding can occur as a result of erosion\nof arterial vessels in the vicinity of the uterus and lesser pelvis by\ndeciduosis lesions, especially in the 2nd and 3rd trimesters of\npregnancy [4]. It is of decisive importance to recognise and re-\nsolve the bleeding complication, for just this reason a differential\nalgorithm for more rapid diagnosis and therapy is urgently\nneeded.\nMaternal bleeding complications are principally divided into\nsubpartum and postpartum types: the main causes of subpartum\nbleeding complications are placenta praevia bleeding, a prema-\nture placental abruption, vasa praevia bleeding, and bleeding\ndue to velamentous cord insertion as well as uterine rupture.\nNowadays these entities are very simple to diagnose clinically\nand by sonography and thus therapy can be initiated in good\ntime. With a proportion of 80 %, uterine atony plays a major role\nin postpartum bleeding that can usually also be quickly recog-\nnised and handled by a targeted and structured management\nprogramme [16].\nThe difficulty for the obstetrician in cases of extrauterine, intra-\nabdominal bleeding is the lack of direct vision of this complica-\ntion so that under certain circumstances much time is lost before\nthe – possibly life-saving – surgical intervention can be initiated.\nFor this reason it is recommended to include bleeding due to de-\nciduosis not only in the prepartum but also in postpartum period\nin the diagnostic considerations.\nOperative management by means of laparotomy – or in selected\ncases by laparoscopy – to staunch the bleeding is the therapy of\nchoice in emergency situations, whereby it must also be men-\n261\nLüdders DW et al. Severe Maternal Pre- … Geburtsh Frauenheilk 2015; 75: 259 –262\nCase Report\n\n\ntioned that as yet no clinical cases of surgical management by\nlaparoscopy have been described in the literature. Because of\nthe urgent need for action in such cases of extrauterine bleeding,\na close cooperation, especially with the surgical and anaesthesio-\nlogical colleagues, is highly necessary. Of course, the prevention\nof foetal complications is also of essential importance so that in\nthe case of a surgical intervention during the pregnancy, appro-\npriate neonatal care options, adequate for the week of pregnancy,\nmust also be available.\nIn most cases the intraoperative tentative diagnosis is made\nunder emergency conditions and must then later be confirmed\nby histology or immunohistochemistry.\nA topic for further discussion is whether or not the complete re-\nmoval and restructuring of all deciduosis lesions after postpar-\ntum regression is a reasonable and recommendable therapy op-\ntion after haemorrhagic deciduosis for the reduction of renewed\ncomplications in subsequent pregnancies. This has not yet been\nclarified. However, it can be assumed that especially those pa-\ntients who have suffered from deciduosis complications due to\nextragenital endometrial lesions during a pregnancy should be\nadvised to undergo surgical removal of the endometriosis.\nAccordingly, a subsequent detailed pathological work-up should\nbe performed on all removed tissue samples relevant for the di-\nagnosis in order to provide possible conclusions for postpartum\ntherapy in subsequent pregnancies.\nUntil now the recurrence risk in a subsequent pregnancy is not\nknown because as yet no systematic investigations on subse-\nquent pregnancies of the affected women have been published.\nAn appropriate registry could help to answer open questions.\nConclusion and Perspectives\n!\nLife-threatening complications that arise through massive intra-\nabdominal bleeding in pregnancy must be treated immediately\nafter recognition by an interdisciplinary team according to the\ngenerally valid clinical recommendations. Massive haemorrhagic\ndeciduosis must always be considered in the differential diagno-\nsis. Therapy of choice is the complete removal of the bleeding le-\nsions. It is still questionable if the postpartum remediation of en-\ndometriosis offers any benefits for subsequent pregnancies.\nConflict of Interest\n!\nNone.\nReferences\n1 Crombach G. Operative Behandlung schwergradiger postpartualer Blu-\ntungen. Gynäkologe 2000; 33: 286 –297\n2 Crombach G, Mosny D, Peisker U et al. Erfahrungen mit der B-Lynch-\nNahttechnik bei schwergradiger Blutung infolge postpartualer Uterus-\natonie. Geburtsh Frauenheilk 2001; 61: 15 –19\n3 Sroka M, Franke M. Späte atone Uterusnachblutung – Stärkste vaginale\nBlutung drei Wochen postpartum – junge Frau droht zu verbluten!\nNotfallmedizin 2003; 29: 419 –421\n4 Schulze G, Chuvashkin D, Riedel H. Schwere antepartuale Blutung in der\n26 + 1. Schwangerschaftswoche infolge der Ruptur der A. ovarica si-\nnistra bei dezidualer Transformation. Geburtsh Frauenheilk 2005; 65:\n881–884\n5 Büttner A, Bassler R, Theele C. Pregnancy associated ectopic decidua\n(deciduosis) of the greater omentum. Pathol Res Pract 1993; 189:\n352–359\n6 Malpica A, Deavers MT, Shahab I. Gross deciduosis peritonei obstruct-\ning labor: a case report and review of the literature. Int J Gynecol Pa-\nthol 2002; 21: 273 –275\n7 Kommos F, Pfisterer J, Peters F et al. Ektope Dezidualreaktion – mög-\nlicher Anlass zur kolposkopischen, histologischen und intraoperativen\nFehldiagnose. Geburtsh Frauenheilk 1998; 58: 446 –450\n8 DeClerck BK, Post MD, Wisell JA. Cutaneous decidualized endometriosis\nin a nonpregnant female: a potential pseudomalignancy. Am\nJ Dermatopathol 2012; 34: 541 –543\n9 Bohlmann MK, Brueggmann D, Schief W et al. Deziduose und Endomet-\nriose der Appendix als Ursache einer akuten abakteriellen Appendizi-\ntis. Geburtsh Frauenheilk 2007; 67: 61 –65\n10 Massi D, Susini T, Paglierani M et al. Pregnancy-associated ectopic de-\ncidua. Acta Obstet Gynecol Scand 1995; 74: 568 –571\n11 Georges B, Piltz S, Ochsenkühn T et al. Extrauterine Deziduose mit post-\npartaler Blutung und fulminanter Gerinnungsstörung. Z Geburtshilfe\nNeonatol 2003; 207 – PO_14_10\n12 Urbanczyk K, Hajduk A, Stachura J. Pregnancy associated diffuse malig-\nnant fibrous mesothelioma of peritoneum. Pol J Pathol 1996; 47: 233 –\n237\n13 Janssen P, Herrmanns B, Schelling M et al. Die Deziduose – eine seltene\nlebensbedrohliche Komplikation in der Schwangerschaft. Geburtsh\nFrauenheilk 2006; 66: 774 –777\n14 Heidegger H, Hümpfner A, Hugo R et al. Peritoneal deciduosis: cause for\nmechanical ileus in pregnancy. Geburtsh Frauenheilk 1991; 51: 307 –\n309\n15 Kondoh E, Shimizu M, Kakui K et al. Deciduosis can cause remarkable\nleukocytosis and obscure abdominal pain. J Obstet Gynaecol Res\n2012; 38: 1376 –1378\n16 PPH-Konsensus-Gruppe (D\n‑A‑CH). Postpartale Blutung – Handlungs-\nalgorithmus nach vaginaler Geburt oder in der postoperativen Über-\nwachungsphase nach Sectio caesarea. 2011. Online: http://www.sgar-\nssar.ch/fileadmin/user_upload/user_saoa/PPH_Alg2g.pdf; last access:\n29.03.2015\n262\nLüdders DW et al. Severe Maternal Pre- … Geburtsh Frauenheilk 2015; 75: 259 –262\nGebFra Science","source_license":"CC0","license_restricted":false}