{"paper_id":"113e3f64-d872-415a-9b05-80275f4d842a","body_text":"www.eCERM.org  Copyright © 2014. THE KOREAN SOCIETY FOR REPRODUCTIVE MEDICINE\n174\nCASE REPORT\nhttp://dx.doi.org/10.5653/cerm.2014.41.4.174\npISSN 2233-8233 · eISSN 2233-8241\nClin Exp Reprod Med 2014;41(4):174-177\nUterine infarction in a patient with uterine \nadenomyosis following biochemical pregnancy\nJae-Yeon Lee1,2, Kyu-Ri Hwang1,2, Kyu-Hee Won1,2, Da-Yong Lee1,2, Hye-Won Jeon1,2, Min-Hwan Moon3\n1Department of Obstetrics and Gynecology, SMG-SNU Boramae Medical Center, Seoul; 2Department of Obstetrics and Gynecology, Seoul National \nUniversity College of Medicine, Seoul; 3Department of Radiology, SMG-SNU Boramae Medical Center, Seoul, Korea\nAdenomyosis is a common gynecological disorder characterized by the presence of endometrial glands and stroma deep within the myome-\ntrium associated with myometrial hypertrophy and hyperplasia. Focal uterine infarction after IVF-ET in a patient with adenomyosis following \nbiochemical pregnancy has not been previously reported, although it occurs after uterine artery embolization in order to control symptoms \ncaused by fibroids or adenomyosis. We report a case of a nulliparous woman who had uterine adenomyosis presenting with fever, pelvic pain \nand biochemical abortion after undergoing an IVF-ET procedure and the detection of a slightly elevated serum hCG. Focal uterine infarction \nwas suspected after a pelvic magnetic resonance imaging demonstrated preserved myometrium between the endometrial cavity and inner \nmargin of the necrotic myometrium. This case demonstrates that focal uterine infarction should be considered in the differential diagnosis of \nacute abdominal pain, vaginal bleeding and infectious signs in women experiencing biochemical abortion after an IVF-ET procedure.\nKeywords: Adenomyosis; Infarction; In vitro fertilization \nIntroduction\nUterine adenomyosis is a common gynecologic disorder character-\nized by the presence and growth of heterotopic endometrial or en-\ndometrium-like structures in the myometrium, with adjacent smooth \nmuscle hyperplasia and can lead to dysmenorrhea and infertility [1]. \nThe ectopic endometrial tissue induces hypertrophy and hyperplasia \nof the surrounding myometrium, resulting in diffuse globular enlarge-\nment of the uterus analogous to the concentric enlargement of the \npregnant uterus. The presenting symptoms include a soft and dif-\nfusely enlarged uterus with menorrhagia (40% ± 50%), dysmenor-\nrhea (10% ± 30%), metrorrhagia (10% ± 12%), dyspareunia and dys-\nchezia [2]. Typically the symptoms start one week prior to menstrua-\ntion. The advised treatment for severe adenomyosis is total hysterec-\ntomy, but for patients wishing to preserve their uterus, a minimally \ninvasive alternative procedure, uterine artery embolization (UAE), \ncan be performed. UAE is a nonsurgical alternative for patients with \nmenorrhagia, symptomatic adenomyosis, or symptomatic uterine fi-\nbroids [3,4]. \nAlthough uterine infarction is a relatively common occurrence after \nUAE for the treatment of fibroids or adenomyosis [5], uterine infarc-\ntion after IVF-ET in a patient with adenomyosis is very rare. To our \nknowledge, this is the first report of uterine infarction after IVF-ET in \na patient with uterine adenomyosis in Korea.\nCase report\nA 31-year-old nulliparous woman was admitted to hospital for pel-\nvic pain. She had suffered severe dysmenorrhea, menorrhagia, and \npelvic pain due to severe adenomyosis which was diagnosed follow-\ning a pelvic magnetic resonance imaging (MRI) and clinical examina-\ntion three years earlier. Her pelvic MRI showed a 9.07 × 6.89 cm, dif-\nReceived: Jun 13, 2014 ∙ Revised: Nov 12, 2014 ∙ Accepted: Nov 14, 2014\nCorresponding author:  Kyu-Ri Hwang\nDepartment of Obstetrics and Gynecology, Seoul National University College of \nMedicine; SMG-SNU Boramae Medical Center, 20 Boramae-ro 5 gil, Dongjak-gu, \nSeoul 156-707, Korea \nTel: +82-2-870-2344  Fax: +82-2-831-2826  E-mail: orangemd@snu.ac.kr\nThis is an Open Access article distributed under the terms of the Creative Commons Attribution \nNon-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits \nunrestricted non-commercial use, distribution, and reproduction in any medium, provided the \noriginal work is properly cited.\n\nwww.eCERM.org\nJY Lee et al.     Uterine infarction after IVF-ET\n175\nfusely enlarged uterus without fibroids. The patient had undergone \nthree unsuccessful cycles of frozen-thawed embryo transfer at an ex-\nternal infertility clinic, due to primary infertility, during the period \nfrom 4–7 months before hospital admission. The patient was admit-\nted to hospital for treatment of ovarian hyperstimulation syndrome \n(OHSS) arising after controlled ovarian hyperstimulation about four \nmonths before the initiation of embryo transfer. She had been diag-\nnosed with hyperthyroidism and treated with propylthiouracil from \nSeptember 2012 onward. A thyroid function test on January 2013 in-\ndicated a euthyroid state: triiodothyronine 166 ng/dL (range, 80–200 \nng/dL), thyroid stimulating hormone 0.17 µIU/mL (range, 0.4–4.1 µIU/\nmL), free thyroxine 1.21 ng/dL (range, 0.80–1.90 mg/dL). The patient \nunderwent another cycle of IVF at a local clinic in February 2013, dur-\ning which she received follitropin-α 150 IU from menstrual cycle day \n3 to day 12 and chorionic gonadotropin 250 µg at menstrual cycle \nday 13. Ovum pick-up was performed about four weeks before she \npresent to the Emergency Department and embryo transfer three \ndays later. The patient received intramural progesterone 50 mg and \nwas prescribed a vaginal progesterone gel (Crinone) for daily use for \n11 days after the embryo transfer.\nThe patient presented to hospital with vaginal bleeding, fever and \nabdominal pain on eleven days after embryo transfer, and received \nantibiotic treatment followed by supportive care at a local clinic for \nthree days. She visited our outpatient clinic due to vaginal bleeding \nand lower abdominal pain on sixteen days after embryo transfer and \nwas admitted to hospital. C-reactive protein (CRP) was elevated to \n7.52 mg/dL and intravenous antibiotic treatment was given. Bio-\nchemical abortion was diagnosed on nineteen days following em-\nbryo transfer after serum β-hCG decreased from 161 to 63 mlU/mL in \ntwo days. The patient was discharged after symptom improvement, \nbut presented with vaginal bleeding and lower abdominal pain, at a \nlocal clinic two days later, where she received hydration. The last day \nof that month, she presented to the emergency department with \nhigh fever (39°C) and, lower abdominal pain. Peripheral blood cell \nanalysis indicated a: white blood cell count of 17.5 × 10³/μL, an ele-\nvated CRP of 24.3 mg/dL, a hemoglobin level of 7.6 g/dL and a he-\nmatocrit level of 24.5%. Renal, liver and clotting function tests were \nwithin normal limits. Blood culture on admission showed a negative \nresult. \nComputed tomography at the Emergency Department revealed a \nwedge-shaped low attenuation area in the anterior uterine corpus. \nThe radiology department recommended a further contrast-enhanced \npelvic MRI study. Focal uterine infarction was suspected, and the pel-\nvic MRI revealed a wedge-shaped, irregularly marginated, non-en-\nhancing area in the anterior uterine wall and the presence of pre-\nserved myometrium between the endometrial cavity and the inner \nmargin of the necrotic myometrium (Figure 1). These findings were \nsuggestive of uterine infarction without uterine perforation. Pelvic \nMRI also revealed peritoneal inflammation. \nAntibiotic infusion and red blood cell transfusion were administered. \nBlood markers investigated to establish the cause of infarction, in-\ncluding antiphospholipid immunoglobulin G (IgG) and IgM, protein \nC activation, protein S, and plasminogen, were all within normal \nranges. Five days after hospital admission, pelvic pain improved and \nCRP decreased to 6.08 mg/dL, and the patient was discharged. Two \nmonths later, pelvic MRI showed interval regression of the previously \nFigure 1. Magnetic resonance imaging of the pelvis in the (A) axial and (B) sagittal planes demonstrated a wedge-shaped, irregularly margin-\nated, non-enhancing area (arrows) in the anterior uterine wall. \nA B\n\n http://dx.doi.org/10.5653/cerm.2014.41.4.174\n Clin Exp Reprod Med 2014;41(4):174-177\n176\nnoted possible focal uterine infarction site with a remarkably im-\nproved blood flow (Figure 2). The patient still had dysmenorrhea and \nmenorrhagia but abdominal pain had improved significantly. \nDiscussion\nAdenomyosis uteri is a pathological entity characterized by the pres-\nence of endometrial glands and stroma embedded within the myo-\nmetrium, but without apparent contact with the endo-myometrial \njunction [6]. Uterine adenomyosis is relatively frequent, affecting 1% \nof females; its diagnosis is more often made in multiparous patients, \nin their fourth and fifth decade of life, and for this reason, infertility is \nnot frequently concurrent with adenomyosis. However, given the \ntrend for first pregnancies to be delayed until women are in their \nthirties or forties, adenomyosis uteri becoming a more frequently \nconsidered diagnosis [2]. Pregnancy resulting from assisted repro-\nductive technology (ART) is possible in patients with ademoyosis; \nhowever, spontaneous abortion, including biochemical abortion, still \noccurs in these patients. \nUterine infarction after IVF-ET in a patient with adenomyosis has not \npreviously been reported. Uterine infarction has been associated \nwith UAE for symptom control of fibroids or adenomyosis [7]. The ob-\njective of UAE is to initiate tumor infarction resulting in substantial re-\nduction of the uterine and tumor volumes [8].\nIt is possible to speculate on the potential mechanisms of occurrence \nof uterine infarction. First, when undergoing IVF-ET, the risk of throm-\nbosis is increased, as in the cases of pregnancy and hyperthyroidism, \nwith which the patient also presented [9,10]. The precise mechanisms \nby which the OHSS and exogenous hormonal stimulation used in ART \ninduce thromboembolic events are still uncertain. However, vascular \nendothelial growth factor secreted during OHSS, high estradiol con-\ncentrations, and blood hyperviscosity play a prominent part in induc-\ning a prothrombotic state [11]. Hansen et al. [12] reported that the \noverall venous thrombosis incidence rate during IVF pregnancies was \nthree times higher than in reference pregnancies. An elevated CRP \ncan activate the coagulation system, following which microthrombo-\nsis formation, in the uterine myometrium, could cause necrosis of the \nmyometrium and lead to focal uterine infarction. Second, the disrup-\ntion of the endometrial–myometrial interface also disrupts the orga-\nnization of myometrial muscle fibers and may also disrupt normal \ncontractility of the subendometrium [2]. Uterine adenomyosis with \nabnormal contractility and the subsequent abortion caused massive \nvaginal bleeding which may have led to uterine ischemia, and the re-\nsulting uterine infarction. Third, thyroid dysfunction modifies the bal-\nance between coagulation and fibrosis. Clinical hyperthyroidism is \ngenerally accompanied by a hypercoagulable state, which may lead \nto microthrombosis of the uterine myometrium [13]. Last, fever and \nthe elevated CRP suggested that infection could have complicated \nthe ischemic necrosis after uterine infarction [14]. \nThe patient we describe here presented with abnormal vaginal bleed-\ning and pelvic pain. Uterine sarcoma also presents with such symp-\ntoms. However, there were no clinical features such as rapid tumor \nFigure 2. Two months after treatment, magnetic resonance imaging of the pelvis in the (A) axial and (B) sagittal planes demonstrated a regres-\nsion of the previously noted possible focal uterine infarction on the background of the uterine adenomyosis.\nA B\n\nwww.eCERM.org\nJY Lee et al.     Uterine infarction after IVF-ET\n177\ngrowth, extrauterine extension or metastases [15,16], leading to the \nconclusion that the possibility of uterine sarcoma was very small. A \nfollow-up MRI three months later showed only traces of uterine in-\nfarction lesion and adenomyosis. \nIn conclusion, we experienced a rare case of uterine adenomyosis \nthat developed into uterine infarction after IVF-ET and biochemical \npregnancy. This case demonstrates that uterine infarction should be \nconsidered in the differential diagnosis of acute abdominal pain, vagi-\nnal bleeding and infectious signs in women with biochemical abor-\ntion after IVF-ET, with a history of adenomyosis and hyperthyroidism. \nThe insertion of a hormonal intrauterine device could be considered \nanother option for treating the symptoms of adenomyosis in cases \npresenting with focal uterine infarction dysmenorrhea and menor-\nrhagia. In addition, the surgical management of focal uterine infarc-\ntion, may be considered in the management of uterine infarction af-\nter IVF-ET in patients with uterine adenomyosis if future fertility is not \ndesired. Finally, it is important to note that the previous infarction site \nmight be a potential risk site for uterine rupture in a future pregnancy.\nConflict of interest \nNo potential conflict of interest relevant to this article was reported. \nReferences\n1. Valentini AL, Speca S, Gui B, Soglia G, Micco M, Bonomo L. Ade-\nnomyosis: from the sign to the diagnosis. Imaging, diagnostic \npitfalls and differential diagnosis: a pictorial review. Radiol Med \n2011;116:1267-87.\n2. Devlieger R, D’Hooghe T, Timmerman D. Uterine adenomyosis in \nthe infertility clinic. Hum Reprod Update 2003;9:139-47.\n3. Bradley LD. Uterine fibroid embolization: a viable alternative to \nhysterectomy. Am J Obstet Gynecol 2009;201:127-35.\n4. Kim MD, Kim S, Kim NK, Lee MH, Ahn EH, Kim HJ, et al. Long-term \nresults of uterine artery embolization for symptomatic adeno-\nmyosis. AJR Am J Roentgenol 2007;188:176-81.\n5. Nijenhuis RJ, Smeets AJ, Morpurgo M, Boekkooi PF , Reuwer PJ, \nSmink M, et al. 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Perri T, Korach J, Sadetzki S, Oberman B, Fridman E, Ben-Baruch G. \nUterine leiomyosarcoma: does the primary surgical procedure \nmatter? Int J Gynecol Cancer 2009;19:257-60.\n16. D’Angelo E, Prat J. Uterine sarcomas: a review. Gynecol Oncol \n2010;116:131-9.","source_license":"CC0","license_restricted":false}