Corioni, Serena

Cozzolino, Mauro

Fambrini, Massimiliano

Guaschino, Secondo

Maggio, Luana

Sorbi, Flavia

English

Florence Research

Endometriosis-Related Hemoperitoneum in Pregnancy: ADiagnosis to Keep in MindMauro Cozzolino, MD, Serena Corioni, MD, Luana Maggio, MD, Flavia Sorbi, MD, Secondo Guaschino, MD,Massimiliano Fambrini, MDDepartment of Biomedical, Experimental, and Clinical Sciences–Division of Obstetrics and Gynecology, University of Florence, Florence, ItalyBackground: Endometriosis is an important gynecologic clinical entity, pathologically defined by the ectopic presence ofendometrium and frequently associated with pelvic pain, that affects approximately 10% of females of reproductive age. A rarebut severe complication of endometriosis during pregnancy is spontaneous hemoperitoneum in pregnancy (SHiP), severeintraabdominal bleeding that can be life threatening.Case Report: We present the case of a patient with SHiP at 29 weeks of pregnancy. A supraumbilical midline laparotomy wasperformed, and pelvic exploration revealed a lacerated and bleeding right ovary. Right annessiectomy was performed, and acesarean section was performed because hemostasis was not achievable. A healthy baby was born, and hemostasis was finallyachieved.Conclusion: We believe that in gravid females with a history of endometriosis, severe abdominal pain, and a reduction ofhemoglobin, physicians should always suspect SHiP. Because SHiP is a life-threatening condition for both the mother and thebaby, a prompt diagnosis must lead to prompt treatment.Keywords: Cesarean section, endometriosis, hemoperitoneum, neonatology, pregnancy, premature birthAddress correspondence to Mauro Cozzolino, MD, Department of Biomedical, Experimental, and Clinical Sciences–Division of Obstetrics andGynecology, University of Florence, Largo Brambilla 3, 50134, Florence, Italy. Tel: (þ39) 055-794-6219. Email: maurocoz@yahoo.itINTRODUCTIONEndometriosis is an important gynecologic clinical entity,pathologically defined by the ectopic presence of endome-trium and clinically associated with pelvic pain. A rare butsevere complication of endometriosis during pregnancy isspontaneous hemoperitoneum in pregnancy (SHiP), life-threatening intraabdominal bleeding.Endometriosis represents one of the main risk factors forSHiP because of the rupture of utero-ovarian vessels orbleeding from endometriosis implants. Because an in-creased number of patients with severe endometriosisachieve pregnancy thanks to assisted reproduction tech-niques, physicians should be aware of endometriosis-related SHiP as a cause of acute abdomen during the thirdtrimester of pregnancy. We report the case of a patient withsevere abdominal pain caused by SHiP.CASE REPORTA 33-year-old gravida 3 para 1 patient was admitted to thehospital for abdominal pain at week 29 of pregnancy. Hermedical history was significant for appendectomy at 15years of age and endometriosis. She underwent laparos-copy in 2007 and 2008 for coagulation and excision ofsuperficial peritoneal endometriosis.The patient’s main symptom was a severe stabbing pelvicpain that started after intercourse. Examination revealed agravid abdomen of 29 weeks, remarkable for a diffusetenderness that worsened at the right superior quadrantwith some guarding but no rebound and positive bowelsounds. The pain was steady and diffuse. She denieddysuria, constipation, and diarrhea but reported nausea andvomiting.A fetal assessment confirmed the presence of a normalsingleton pregnancy with a regular left-located placenta anda normal amniotic fluid volume. Tocography was negative,and the cervical length was 35 mm. Complete blood count,metabolic profile, and urinalysis were performed. Thepatient’s vital signs were normal: blood pressure of 120/70mmHg, pulse of 109 beats per minute, and respiratory rateof 15 breaths per minute. Her laboratory results showedhemoglobin of 8.8 g/dL with 18.8 3 109/L leukocytes.Metabolic hepatic panel and urinalysis were negative.As the patient’s pain and history were suggestive foracute cholecystitis, acute pyelonephritis, or gastroenteritis,an abdominal and pelvic ultrasound was performed thatrevealed free fluid in the abdomen; the liver, spleen,kidneys, and cholecyst were regular. Within 7 hours ofadmission, the patient gradually complained of worsening262 The Ochsner JournalThe Ochsner Journal 15:262–264, 2015 Academic Division of Ochsner Clinic FoundationCASE REPORTpain. Steroid therapy with betamethasone was administeredfor fetal lung maturation.Abdominal and pelvic magnetic resonance imaging wasperformed but was hampered by the presence of the graviduterus. Imaging was only able to detect a large quantity offree fluid in the abdomen and pelvis. In a few hours, thepatient’s hemoglobin dropped to 7.6 g/dL.We decided to perform surgery because of the patient’sworsening clinical features and the diagnosis of acuteabdomen and SHiP. The patient underwent a supraumbil-ical midline laparotomy under general anesthesia.Almost 1,500 mL of blood was aspirated from theperitoneal cavity. Pelvic exploration revealed a bluish coloron the posterior right side of the uterus. The right fallopiantube and ovary had adhesive bands with the right uterinewall. Active bleeding from the lacerated right ovary and fromthe right-posterior surface of the uterus was observed. Rightannessiectomy was performed with an interrupted 2-0VICRYL (Ethicon US, LLC) single-layer suture to repair theexcision (Figure).Because hemostasis was not achievable with the graviduterus, a cesarean section was performed. A male baby wasextracted who weighed 1.390 kg and whose Apgar scoreswere 4 and 7 at the first and fifth minute, respectively. Afterclosing the cesarean breach, the uterus reduced its volume,and the bleeding at the site of annessiectomy lessened withfew bleeding points left. Hemostasis was achieved withelectrocoagulation and a second layer of 2-0 VICRYLinterrupted suture.After careful confirmation of hemostasis, the abdominalwall was closed. Intraoperatively, the patient received 2units of red blood cells and 2 units of fresh frozen plasma.She recovered well and was discharged on postoperativeday 5. The infant had an uneventful course in neonatalintensive care and was discharged 1 month after birth.DISCUSSIONEndometriosis is a common gynecologic condition thataffects approximately 10% of females of reproductive age. Itis a chronic inflammatory disease defined as the presenceof endometrial-like tissue outside the uterine cavity. Endo-metriosis generally involves the peritoneum, ovaries, andrectovaginal septum but may also involve other abdominalor extraabdominal sites. Clinically, the symptoms includedysmenorrhea, pelvic pain, deep dyspareunia, and infertil-ity.1-3Endometriosis is associated with an increased risk ofearly pregnancy complications, such as ectopic pregnancyand miscarriage, but the literature contains few reports ofcomplications linked to severe endometriosis during thethird trimester of pregnancy.2 A rare but potentiallycatastrophic complication occurring in advanced pregnancyis hemoperitoneum as a result of spontaneous bleedingfrom blood vessels.1-4During pregnancy, hemoperitoneum caused by sponta-neous rupture of uterine vessels is an extremely rarecondition but is almost always a life-threatening complica-tion associated with maternal and perinatal mortality.2,5Published series report maternal mortality ranging from3.6%-49%.2 Maternal mortality associated with SHiPdropped dramatically during the second half of the 20thcentury to approximately 4%, but fetal mortality remainshigh (approximately 31%), with 44% of the deaths attribut-able to maternal shock.1,6 The diagnosis of this condition isdifficult and rarely reached without surgery. Laparotomy isoften needed to manage these patients. Typical presentingsymptoms are sudden intense abdominal pain and signs ofhypovolemic shock; a marked drop of hemoglobin level is afrequent finding. The pitfall of SHiP is differential diagnosis.Placental abruption is the most common preoperativemisdiagnosis. Other differential diagnoses include uterinerupture; abdominal pregnancy; ruptured appendix; severepreeclampsia; HELLP (hemolysis, elevated liver enzymes,low platelet count) syndrome; acute fatty liver disease;chorioamnionitis; and hepatic or splenic rupture.2Approximately 25 cases of SHiP have been described inthe literature during the past 20 years. This condition hasbeen described out of labor (61%), during labor (18%), andin the early postpartum period (21%).6,7 Twenty-ninepercent of out-of-labor SHiP cases have been reported tooccur before 33 weeks of gestation, 39% of cases occurredbetween 33-37 weeks of gestation, and 32% of casesoccurred at term. Endometriosis is recognized as the majorrisk factor for SHiP.1,2Hemoperitoneum in pregnant women with endometriosiscan be caused by spontaneous rupture of utero-ovarianvessels or bleeding endometriosis implants. The spontane-ous endometriosis-linked rupture of utero-ovarian vesselsmay be attributed to 3 factors: chronic inflammation causedby endometriosis may cause utero-ovarian vessel leakage;2adhesions may create tension on these vessels as theuterus becomes enlarged in pregnancy;1 decidualization ofendometrial foci during pregnancy may cause utero-ovarianvessel perforation.2,3,6 Moreover, venous pressure in theutero-ovarian circulation increases during pregnancy. Thevenous pressure can be accentuated by physical effort,such as muscular activity, coughing, defecation, coitus, orthe pushing phase of labor.2 Our patient reported inter-course as the trigger of abdominal pain. Although in a fewcases the origin of bleeding remains unknown even duringlaparotomy, arteries and superficial veins or varicosities onthe posterior surface of the uterus or parametria are oftenthe site of bleeding.7Figure. Attempt to repair the excision after bleeding fromthe lacerated right ovary and from the right-posteriorsurface of the uterus was observed.Cozzolino, MVolume 15, Number 3, Fall 2015 263The 2009 study by Brosens et al reviewed all cases ofSHiP described since 1987.7 The authors noticed that SHiPwas associated with endometriosis in >50% of cases; thediagnosis of endometriosis was unknown until then inalmost half of the affected women. Biopsy samples of thesite of bleeding during pregnancy were obtained in 5 casesthat revealed endometriosis, characterized by prominentvascularization and decidualization of the lesion. In ourpatient, the diagnosis of endometriosis was already known,and the pathologic examination of the specimen confirmedthe presence of decidualized ectopic endometrial tissue.Several aspects of treating endometriosis remain contro-versial: whether surgical treatment for endometriosis shouldbe adopted not only to improve fertility but also to preventsignificant pregnancy complications such as SHiP; whethera tailored prenatal follow-up for pregnant women withendometriosis should be implemented and how it shouldbe performed (obstetrics and gynecology department visit,ultrasound, magnetic resonance imaging); and whethercesarean section should be performed routinely to preventirreversible fetal damage when laparotomy is required.2Lesions caused by severe endometriosis can lead tosignificant abdominal bleeding during the third trimester ofpregnancy.2 Bleeding from endometrial implants is anestablished cause of acute hemoperitoneum in pregnancy,and physicians should keep it in mind in cases of abdominalpain during pregnancy.1 Prompt intervention may berequired in such cases, and an expert surgical team shouldbe involved with adequate availability of blood products.2CONCLUSIONWe believe that in gravid females with a history ofendometriosis, severe abdominal pain, and a reduction ofhemoglobin, physicians should always suspect SHiP.Because SHiP is a life-threatening condition for both themother and the baby, a prompt diagnosis must lead toprompt treatment. A greater awareness of SHiP and itsassociated risk factors, such as pelvic endometriosis, mayfacilitate the diagnosis of this condition and expedite theintervention to improve maternal and fetal outcomes.ACKNOWLEDGMENTSThe authors have no financial or proprietary interest in thesubject matter of this article.REFERENCES1. Brosens I, Brosens JJ, Fusi L, Al-Sabbagh M, Kuroda K, BenagianoG. Risks of adverse pregnancy outcome in endometriosis. FertilSteril. 2012 Jul;98(1):30-35. doi: 10.1016/j.fertnstert.2012.02.024.2. Katorza E, Soriano D, Stockheim D, et al. Severe intraabdominalbleeding caused by endometriotic lesions during the thirdtrimester of pregnancy. Am J Obstet Gynecol. 2007 Nov;197(5):501.e1-e4.3. Stephansson O, Kieler H, Granath F, Falconer H. Endometriosis,assisted reproduction technology, and risk of adverse pregnancyoutcome. Hum Reprod. 2009 Sep;24(9):2341-2347. doi: 10.1093/humrep/dep186.4. Falconer H. Pregnancy outcomes in women with endometriosis.Semin Reprod Med. 2013 Mar;31(2):178-182. doi: 10.1055/s-0032-1333484.5. Aziz U, Kulkarni A, Lazic D, Cullimore JE. Spontaneous rupture ofthe uterine vessels in pregnancy. Obstet Gynecol. 2004 May;103(5Pt 2):1089-1091. Review. Erratum in: Obstet Gynecol. 2005 Jan;105(1):222.6. Passos F, Calhaz-Jorge C, Graça LM. Endometriosis is a possiblerisk factor for spontaneous hemoperitoneum in the thirdtrimester of pregnancy. Fertil Steril. 2008 Jan;89(1):251-252.7. Brosens IA, Fusi L, Brosens JJ. Endometriosis is a risk factor forspontaneous hemoperitoneum during pregnancy. Fertil Steril.2009 Oct;92(4):1243-1245. doi: 10.1016/j.fertnstert.2009.03.091.This article meets the Accreditation Council for Graduate Medical Education and the American Board of MedicalSpecialties Maintenance of Certification competencies for Patient Care and Medical Knowledge.Endometriosis-Related Hemoperitoneum264 The Ochsner Journal

Endometriosis-Related Hemoperitoneum in Pregnancy: A Diagnosis to Keep in Mind

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Endometriosis-Related Hemoperitoneum in Pregnancy: A Diagnosis to Keep in Mind

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