{"paper_id":"7c780d58-fa83-460c-9e74-98a0e2e4bffb","body_text":"Case Report\n135\nCopyright© 2025 The Author(s). Published by Galenos Publishing House on behalf of National Society of Gynecology and Obstetrics. This is an open \naccess article under the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 (CC BY-NC-ND) International License.\nAnat J Obstet Gynecol Res 2025;2(3):135-137\nIntramyometrial Ectopic Pregnancy in a Patient with Adenomyosis: \nA Rare Case Report\n Sefa Arlıer,  Sibel Öncel Yavuz\nUniversity of Health Sciences Turkey , Adana City Training and Research Hospital, Clinic of Obstetrics and Gynecology , Adana, Turkey\nIntramyometrial ectopic pregnancy (IMP) is an exceptionally rare form of ectopic gestation, representing <1% of cases. It \nposes diagnostic challenges as it often mimics leiomyoma or interstitial pregnancy on imaging. A 42-year-old woman with a \nhistory of adenomyosis and previous salpingectomy was admitted with acute abdomen and hypovolemic shock. Preoperative \nlaboratory tests revealed hemoglobin (Hb): 9.2 g/dL, hematocrit: 28%, white blood cell: 12,300/mm³, platelet: 210,000/mm³, \nand beta-human chorionic gonadotropin: 65,000 mIU/mL. After emergency laparotomy for ruptured intramyometrial pregnancy , \nthe postoperative Hb level was 7.4 g/dL; however, this value may have been relatively overestimated due to preoperative \nhemoconcentration. Transvaginal ultrasonography revealed a gestational sac embedded in the uterine fundal myometrium \nwith fetal cardiac activity . Emergency laparotomy revealed a ruptured intramyometrial gestation with massive hemoperitoneum. \nEstimated intraoperative blood loss was 1800 mL, requiring transfusion of three units of red blood cells and two units of \nfresh frozen plasma. Total abdominal hysterectomy and salpingectomy were performed. Histopathological analysis confirmed \nintramyometrial pregnancy associated with adenomyosis. This case highlights the importance of considering IMP in the \ndifferential diagnosis of abnormal uterine masses, especially in patients with prior uterine surgery or adenomyosis. Early \nrecognition and prompt surgical intervention are important to prevent catastrophic and potentially life-threatening hemorrhage.\nKeywords: Intramyometrial pregnancy , ectopic pregnancy , adenomyosis, hysterectomy\nABSTRACT\nINTRODUCTION\nEctopic pregnancy constitutes approximately 1-2% of \nall pregnancies, with the vast majority localized in the \nfallopian tubes. Intramyometrial ectopic pregnancy (IMP), \ndefined as implantation of the gestational sac entirely \nwithin the myometrium without communication with the \nuterine cavity or fallopian tubes, is exceedingly rare (<1%). \nThe pathogenesis, frequency, and natural history of \nintramural pregnancy, a rare ectopic pregnancy, are not well \nunderstood. Treatment varies based on symptom severity, \npregnancy location, viability, and stage at diagnosis, with \nno consensus on ultrasound criteria for identification. 1 \nThe clinical challenge of IMP lies in its diagnostic difficulty, \nsince it can easily be mistaken for degenerating fibroid \nor interstitial pregnancy. Literature reports fewer than 100 \ncases worldwide, underlining the rarity of the condition. 2-4 \nEtiological factors include uterine trauma (dilatation and \ncurettage, cesarean section, myomectomy), assisted \nreproductive techniques, adenomyosis (as an abnormal \nniche for implantation), and pelvic inflammatory \ndisease.2,5,6 Adenomyosis itself is associated with infertility, \ndysmenorrhea, menorrhagia, and may alter implantation \nthrough ectopic endometrial tissue within the myometrium. 6 \nDiagnostic modalities such as transvaginal ultrasonography \n(TVUS) and magnetic resonance imaging (MRI) are very \nhelpful for differential diagnosis, as they allow distinction of \nIMP from fibroid degeneration or interstitial pregnancy. 2,7 \nTreatment options vary from conservative management \n(methotrexate, laparoscopic excision, hysteroscopic \nevaluation) to radical interventions such as emergency \nhysterectomy in hemodynamically unstable patients. 3,5 \nAddress for Correspondence: Sefa Arlıer, University of Health Sciences Turkey , Adana City Training and Research Hospital, Clinic of Obstetrics and \nGynecology , Adana, Turkey \nE-mail: sefaarlier@gmail.com ORCID ID: orcid.org/0000-0002-0019-8403\nReceived: 17.08.2025 Accepted: 25.08.2025 Publication Date: 26.01.2026\nCite this article as: Arlıer S, Öncel Yavuz S. intramyometrial ectopic pregnancy in a patient with adenomyosis: a rare case report. Anat J Obstet Gynecol \nRes. 2025;2(3):135-137\nDOI: 10.4274/anajog.galenos.2025.28199\n\nAnat J Obstet Gynecol Res 2025;2(3):135-137Arlıer and Öncel Yavuz. Intramyometrial Ectopic Pregnancy with Adenomyosis\n136\nThe objective of this study was to contribute to the scarce \nliterature and highlight the diagnostic and therapeutic \nchallenge of IMP associated with adenomyosis, complicated \nby rupture and life-threatening hemorrhage. \nCASE REPORT\nA 42-year-old multiparous woman (G6P4Y4) with a prior \nright salpingectomy due to ectopic pregnancy and a \nhistory of adenomyosis presented to the emergency \ndepartment with acute abdominal pain, hypotension, \nand tachycardia. The diagnosis of adenomyosis had \npreviously been made by TVUS due to menorrhagia and \ndysmenorrhea. She had missed her menstrual period and \nreported progressive abdominal distension and weakness.  \nOn admission, vital signs were: blood pressure 100/72 \nmmHg, heart rate 100/min, respiratory rate 21/min, \nand temperature 37.2 °C. Abdominal examination \nrevealed diffuse tenderness, guarding, and rebound.  \nPreoperative laboratory findings were: hemoglobin \n9.2 g/dL; hematocrit 28%; white blood cell count \n12,300/mm³; platelet count 210,000/mm ³; and beta-\nhuman chorionic gonadotropin ( β-hCG) 65,000 mIU/\nmL. Renal and liver function tests were normal.  \nImaging by TVUS showed a gestational sac in the uterine \nfundus, measuring approximately 46 mm, corresponding \nto 11+3 weeks, with positive fetal cardiac activity Figure 1. \nFree fluid with clots (~500 cc) was noted in the Pouch of \nDouglas  and in the perisplenic and perihepatic regions. MRI \nwas not performed due to the emergency clinical condition.  \nEmergency laparotomy revealed 5 × 5 cm ruptured \nintramyometrial pregnancy localized to the fundus with massive \nhemoperitoneum Figures 2. Right fallopian tube was absent, \nleft adnexa were normal. Estimated blood loss was 1800 mL. \nTotal abdominal hysterectomy and left salpingectomy were \nperformed, and three units of packed red blood cells plus two \nunits of fresh frozen plasma were transfused intraoperatively.  \nSubsequent histopathological examination confirmed \nIMP with necrotic decidual tissue and chorionic villi \ninfiltrating the myometrium, alongside adenomyotic foci.  \nThe patient was monitored in the intensive care unit for \n24 hours, recovered uneventfully, and was discharged on \npostoperative day five in a stable condition. Written informed \nconsent was obtained, and institutional University of Health \nSciences Turkey, Adana City Training and Research Hospital \nEthics Committee approval was secured for publication of this \nreport (approval number: 629, date: 10.07.2025).\nDISCUSSION\nIMP remains one of the rarest and most diagnostically \nchallenging types of ectopic gestation, with <1% incidence \nand fewer than 100 reported cases worldwide. 1,2 Because \nof its rarity, it is frequently misdiagnosed as fibroid \ndegeneration, interstitial pregnancy, or adenomyosis. 3,5,7 \nThe precise etiology of IMP is not fully understood. Well-\nestablished risk factors include uterine trauma from prior \nprocedures such as dilatation and curettage, cesarean \nsection, or myomectomy. 1,2 Assisted reproductive \ntechnologies, particularly in vitro fertilization and \nintrauterine insemination, have also been associated with \nintramyometrial implantation. 4 Pelvic inflammatory disease \nand perimetrial inflammation have also been implicated. 2,5 \nAdenomyosis has recently been proposed as a possible \npredisposing factor Shi et al.6 suggested that endometrial tissue \nlocated within adenomyotic foci can undergo decidualization \nin response to estrogen and progesterone, thereby creating \nan abnormal receptive environment for blastocyst implantation \ndeep within the myometrium. 6 Similarly, Aburayyan et al. 2 \nhighlighted adenomyosis as one of the plausible mechanisms \npredisposing to intramyometrial pregnancy. In the presented \npatient, histopathological examination confirmed the \ncoexistence of adenomyosis and intramyometrial pregnancy. \nThis observation supports the hypothesis that adenomyotic \nfoci may provide a niche for abnormal implantation. This case \nmay therefore represent one of the first histopathologically \nconfirmed reports of adenomyosis as an etiological factor in IMP . \nDiagnostic challenges of IMP are as follows. Clinical \npresentation is nonspecific. Amenorrhea, abdominal pain, \nand abnormal uterine bleeding are common but overlap \nwith other types of ectopic pregnancy. 3,4 Serum β-hCG \nlevels are variable and have variously been reported \nas elevated, normal, or declining, thus making them \nFigure 1.  Transvaginal ultrasonographic appearance of an \nintramyometrial ectopic pregnancy located in the uterine fundus\nFigure 2. Gross intraoperative view of the ruptured intramyometrial \npregnancy specimen following emergency hysterectomy\n\n\nAnat J Obstet Gynecol Res 2025;2(3):135-137Arlıer and Öncel Yavuz. Intramyometrial Ectopic Pregnancy with Adenomyosis\n137\nunreliable.1,6 Ultrasonography may show a gestational sac \nembedded in the myometrium with an empty cavity, but \nmisdiagnosis as leiomyoma is frequent.1,5,7 Three-dimensional \nultrasonography and MRI improve accuracy, but intraoperative \nfindings and histopathology remain the gold standard.  \nManagement of IMP depends on hemodynamic stability, \ngestational age, and desire for future fertility. Conservative \noptions include methotrexate therapy and laparoscopic excision \nwith uterine repair.4,6 In hemodynamically unstable patients, as \nin the present case, emergency hysterectomy is life-saving. 3 \nPrognosis for IMP is also variable. Fertility-preserving \napproaches may be possible in selected and stable patients \nbut risk persistent ectopic tissue. Hysterectomy, although \ndefinitive, ensures survival. Early suspicion, individualized \ntreatment, and awareness of adenomyosis as a potential \nfactor may improve outcomes.\nCONCLUSION\nIMP is a rare but potentially life-threatening condition. The \npresented case demonstrates that adenomyosis may provide \na physiological niche for abnormal implantation, representing \na possible new etiological risk factor for IMP . Early diagnosis \nwith TVUS/MRI, awareness of differential diagnoses, and timely \nsurgical intervention are essential to reduce morbidity and \nmortality. The present case adds novel evidence to the literature \nand highlights the importance of considering adenomyosis in \nthe pathogenesis of intramyometrial pregnancy.\nEthics\nEthics Committee Approval: The study was approved by the \nUniversity of Health Sciences Turkey, Adana City Training and \nResearch Hospital Ethics Committee (approval number: 629, \ndate: 10.07.2025). \nInformed Consent: Written informed consent was provided \nby the patient.\nFootnotes\nAuthorship Contributions\nSurgical and Medical Practices: S.A., S.Ö.Y ., Concept: S.A., \nS.Ö.Y ., Design: S.A., S.Ö.Y ., Data Collection or Processing: \nS.A., S.Ö.Y ., Analysis or Interpretation: S.A., S.Ö.Y ., Literature \nSearch: S.A., Writing: S.A., S.Ö.Y .\nConflict of Interest:  No conflict of interest was declared by \nthe authors.\nFinancial Disclosure:   The authors declared that this study \nreceived no financial support. \nREFERENCES\n1. Memtsa M, Jamil A, Sebire N, Jauniaux E, Jurkovic D. Diagnosis \nand management of intramural ectopic pregnancy. Ultrasound \nObstet Gynecol. 2013;42(3): 359-362.\n2. Aburayyan ANMI, Zuhour OMB, Alsalameh BKS, Sabboh M, \nJobran AWM. Intramural pregnancy managed by conservative \nlaparoscopic resection:a case report. Case Rep Womens Health. \n2024;44:e00653.\n3. Yang WX, Wang TT , Zhao S. Persistence of ectopic pregnancy after \nintramural ectopic pregnancy surgery: a case report. Int J Surg \nCase Rep. 2025;126:110623.\n4. Yamamoto K, Takiuchi T , Kiso K, et al. Intramural pregnancy after \nintrauterine insemination in a nulligravid patient without previous \nuterine trauma, complicated by idiopathic thrombocytopenic \npurpura: a case report. Case Rep Womens Health. 2025;45:e00684.\n5. Chen YC, Huang CY . Successful laparoscopic management of a \nrare intramural ectopic pregnancy. J Minim Invasive Gynecol. \n2025:S1553-4650(25)00135-9.\n6. Shi J, Wu Y , Li X, et al. Effects of localization of uterine \nadenomyosis on clinical features and pregnancy outcome. Sci Rep. \n2023;13(1):14714.\n7. Memtsa M, Jamil A, Sebire N, Jauniaux E, Jurkovic D. Diagnosis \nand management of intramural ectopic pregnancy. Ultrasound \nObstet Gynecol. 2013;42(3):359-362.","source_license":"CC0","license_restricted":false}