{"paper_id":"fc5ac43e-1564-4879-90e0-f5d3e325c9a7","body_text":"Open Access, Volume 4 \nEndometrial ossification: Unusual cause of chronic pelvic pain \nin low-resource settings\nCase Report\nwww.jcimcr.org\nJournal of\nClinical Images and Medical Case Reports\nReceived: Jun 09, 2023\nAccepted: Jun 26, 2023\nPublished: Jul 03, 2023 \nArchived: www.jcimcr.org\nCopyright: © Matovelo D (2023).\nDOI: www.doi.org/10.52768/2766-7820/2482\n*Corresponding Author: Dismas Matovelo \nCatholic University of Health and Allied Sciences \n(CUHAS), Department of Obstetrics & Gynecology, \nMwanza, Tanzania. \nEmail: magonza@bugando.ac.tz\nISSN 2766-7820\nIntroduction\nEndometrial ossification is a rare condition affecting repro -\nductive aged women characterized by presence of mature or \nimmature bone tissue in the endometrium. The incidence of \nendometrial ossification is estimated to be 3/10,000 cases and \noften results from termination or spontaneous abortion that \nleads to either persistence of fetal bone or may follow true os -\nseous metaplasia of endometrial tissue [1,2].\nThe most common presenting symptoms are secondary in -\nfertility in more than 80% of cases and rarely heavy menstrual \nbleeding and chronic pelvic pain [3]. There are controversies in \nthe mechanism of endometrial ossification however, the widely \naccepted theory is the metaplasia of the stromal cells into os -\nteoblastic cells that produce mature bone [4,5].\nHysteroscopy is the gold standard for diagnosis and treat -\nment for endometrial ossification, with histopathological exam-\nAbstract\nIntroduction: Endometrial ossification is a rare condition in which \nits actual etiology and pathogenesis is controversial and debatable \nhowever, the condition is usually related to secondary infertility after \nabortion and endometritis.\nCase presentation: A 32-year-old para 2 living 1 lady who presented \nwith long-standing intermittent sharp pelvic pain for seven years. She \nwas a self-referral to BMC gynecology outpatient unit due to chronic \npelvic pain. No prior history of intrauterine copper device insertion. \nUpon evaluation, she had normal ovulatory cycles. A pelvic x-ray could \nnot show any spine deformities or any pelvic abnormalities. During \ntransabdominal pelvic ultrasound imaging, a well-defined thick linear \nhyperechogenic structure with acoustic shadowing was seen in the en-\ndometrial cavity measuring 2 x 3 cm aligned to endometrial strips.\nUltrasound guided sharp dilatation & curettage (D&C) was per -\nformed by using ovum forceps in which we successfully removed a tu-\nbular structure measuring 2.3 by 3.4 cm with sharp ends deep in the \nendometrium. Histopathological analysis revealed trabecula and extra-\ncellular matrix osteocytes in keeping with endometrial ossification. \nConclusion: Endometrial ossification can be effectively treated with \nultrasound guided dilation and curettage as an alternative to hyster -\noscopy. \nKeywords: Endometrial ossification; Chronic pelvic pain; Ultra -\nsound-guided dilatation; Curettage.\nSamweli Ndulila1; Oscar Ottoman2; Richard Kiritta1; Edgar Ndaboine1; Simplice Harusha4; Rajab Kidenda3; Dismas Matovelo1*\n1Catholic University of Health and Allied Sciences (CUHAS), Department of Obstetrics & Gynecology, Mwanza, Tanzania.\n2Catholic University of Health and Allied Sciences (CUHAS), Department of Pathology, Mwanza, Tanzania.\n3Catholic University of Health and Allied Sciences (CUHAS), Department of Radiology and Imaging Mwanza, Tanzania.\n4Sengerema Designated District Hospital, Department of Obstetrics & Gynecology, Mwanza, Tanzania.\n\nwww.jcimcr.org                Page 2\nCitation: Ndulila S, Ottoman O, Kiritta R, Ndaboine E, Matovelo D, et al. Endometrial ossification: Unusual cause of chronic \npelvic pain in low-resource settings. J Clin Images Med Case Rep. 2023; 4(7): 2482.\nination as an adjunct diagnostic aid to hysteroscopy [3,6]. How-\never, in resource scarce setting, ultrasonography examination \noffers an alternative to hysteroscopy. Furthermore, the majority \nof cases resume their fertility sooner once the tissue had been \nremoved. Moreover, saline infusion hysterography with Dop -\npler studies may be performed to evaluate the stratum basalis \nand spinosum if there is a chance of fertility [7]. Here, we report \na case of endometrial ossification in a woman presenting with \nchronic pelvic pain.\nCase presentation\nA 32-year-old woman, para 2 living 1 complained of lower \nabdominal pain for seven years. The pain was severe and came \nand went at regular intervals. Symptoms began two weeks af -\nter termination of her 3rd pregnancy at 29 weeks’ gestation in \n2015. Without any regular pattern, the pain intensified with ac-\ntivity and decreased with rest. She was kept on multiple anal -\ngesics, but she remained in excruciating agony for quite some \ntime.\nShe had her first vaginal birth successfully ten years back in \n2010, she then conceived again in 2014 in which it ended up \nwith spontaneous abortion at 2 months without any adverse \nevent reported thereafter. Prior to her third pregnancy, she con-\ntinued to experience regular menstrual cycles, normal volume \nand length, her cervical cancer screening was negative, and she \nhad never experienced symptoms suggestive of sexual trans -\nmitted infections.\nHer ongoing suffering led to bouts of chronic tension and \nworry, as well as social withdrawal and marital problems, all of \nwhich contributed to divorce in 2019. In 2018 and 2019, she \nvisited several different medical facilities and had two uterine \nevacuations, but the pain continued despite these procedures. \nIn 2021, she went to three further Primary Healthcare (PHC) \nfacilities and underwent a number of ultrasounds, all of which \nproduced results that were inconclusive. After some time, she \nmade the decision to check in at our medical center with above \nhistory.\nClinical examination revealed a young woman who appeared \nstressed but well-kept and well-nourished.  She was not pale, \nher blood pressure was 111/65 mmHg, pulse rate 72 beats per \nminute, respiratory rate 18 cycles per minute and temperature \nof 36 Celsius, and normal oxygen saturation.\nHer cardiovascular and respiratory findings were normal. \nHer abdomen was normal in contour and shape, with a tender \nhypogastrium, with no palpable mass.  Her uterus appeared \nslightly bulky on bimanual examination with a positive cervical \nexcitation test however on visual inspection vulva, vaginal and \ncervix were normal.\nInvestigations\nShe had serum βhCG of 0.3 miu/ml, her full blood count \nwas within normal reference, Hemoglobin level was 12.4 g/dl, \nWBC 4 x 10 9 Platelets count 234 x 10 3 and normal white blood \ncell differentials. She underwent trans-abdominal pelvic ultra -\nsound, which revealed a well-defined thick linear hyperecho -\ngenic structure with acoustic shadowing in the endometrial cav-\nity measuring 2 x 3 cm (Figure 1). Her pelvic x-ray was normal. \nTransabdominal ultrasound findings \nManagement and follow-up: She had been scheduled for a \ndilatation and curettage (D&C), and with the help of grayscale \n2D ultrasound guidance, a tubular structure that had been \ndeeply adhered to the endometrium was evacuated. The tissue \nbiopsy was sent to histopathology for analysis, and the patient \nwas released the next day in excellent condition.\nFigure 1: Showing a linear hyper echogenic structure in the endo -\nmetrial cavity measuring 2x3 cm occupying the internal os and mid \ncavity of the endometrium shown by orange arrow.\nHistopathological findings\nGross findings: Showed multiple hemorrhagic tissue biopsy \nfrom endometrial curettage measured (2 x 1) cm mixed with \nhard calcified bone fragments measured 2 cm long as shown in \nfigure 2 below.\nMicroscopic findings: Section showing fragments composed \nof endometrial gland and its stroma accumulated with area of \nbone trabecular some with calcification as shown in figures \n3and 4 below.  \nDiscussion \nEndometria ossification is an occasional phenomenon most-\nly occurring following termination or spontaneous abortion \nin more than 80% of the cases reported [2]. About 100 cases \nworldwide have been documented. Its presentation varies but \nthe frequent complaint is secondary infertility and heavy men -\nstrual bleeding, and chronic pelvic pain [3,5].\nFigure 2: Showing the gross appearance of the bone tissue from \nthe submitted sample of the endometrial curettage.\n\n\nwww.jcimcr.org                Page 3\nFigure 3: Hematoxylin and Eosin section showing the area of bone \ntrabecular on endometrial glands and its stroma pointed by red \narrow (x10Hpf).\nFigure 4: Hematoxylin and Eosin section showing the area of bone \ntrabecular infiltrated within the endometrial stroma pointed by \nred arrow (x10Hpf).\nControversies exists in the mechanism of endometrial ossi -\nfication; the widely accepted mechanism is metaplasia of the \nstromal cells into osteoblastic cells that produce mature bone. \nThe endometrial ossification can be confused with an intra-\nuterine copper devices making difficult to diagnose [5,8].\nOur patient presented with history of chronic pelvic pain for \nseven years with multiple uterine evacuation without conclu -\nsive results. However, prior to this, she had termination of an \nadvanced pregnancy. This patient had termination of pregnan -\ncy at 29 weeks followed by 2 episodes of uterine evacuations.  \nUpon review of literature, the major risk factor for endometrial \nossification is the termination of pregnancy at least ≥ 3 months \neither spontaneous or surgical termination [3,9].\nThere was a delay in making of the diagnosis to this patient \nfrom lower health facilities due to lack of pathologists in her \nresidential area and lack of clinician awareness of this condi -\ntion [5]. On other hand our patient presented with unique and \nunusual activity-related pain which is an occasional symptom in \nendometrial ossification [10]. Infertility was not a concern for \nher because she had early been divorced. In several cases, di -\nagnosis of endometrial ossification can be delayed due to the \nconfusion with other diagnoses such as the Intrauterine Copper \nDevices (IUCDs), foreign bodies, calcified submucous fibroids, \nendometrial tuberculosis, Asherman’s syndrome, and rarities \nsuch as heterotopic bone and uterine malignant mixed Mulle -\nrian tumor and furthermore due to the fact that in most cases is \nsilent unless it present with either infertility or abnormal uter -\nine bleeding. In majority of cases, diagnosis of the endometrial \nossification requires a high degree of suspicion, expertise and \ntechnology [5,11].\nEndometrial ossification is best simultaneously diagnosed \nand treated with office hysteroscopy. Endometrium hypertro -\nphy like white coral is usually visualized during hysteroscopy \n[3]. Hysteroscope offers the best modality in both diagnosing \nand removing the bone fragment [3,5,12], however, in resource \nlimited setting like ours due to lack of hysteroscope we opted \ninitially to perform D&C using an ovum forceps, but the bone \ncould not be felt. Transvaginal ultrasound which is available in \nlimited resource settings is an excellent alternative, with a high \nsensitivity and specificity for endometrial pathology, as applied \nto our patient [1].\nAfter the removal of the bone from the endometrial cav -\nity, the patient reported disappearance of the pelvic pains she \nhad. The pain relief, resumption of fertility and less likelihood of \nrecurrence after treatment has made it to be among diseases \nwith good prognosis [3,5]. Furthermore, there is no role of hys-\nterectomy in the management of endometrial ossification since \nmost cases present in the reproductive age group and with in -\nfertility. Hysterectomy may be an option in women within peri-\nmenopausal group and have completed family [5]. \nOur case was different as the blind ovum forceps use was not \nsuccessful, we improvised with ultra-sound guided with slight \ncurette and successful removed the bone piece. On our side, we \ndeclare that this is the first time we encountered the case, and \nwe present ultrasound guided D&C as an alternative to office \nhysteroscopy [3,13].\nConclusion\nUltrasound guided dilatation and curettage can be as effec -\ntive as office hysteroscopy in treating endometrial ossification \nin low-resource settings.\nDeclarations\nPatient’s perspective: The care provided was timely with full \nexplanation of the diagnosis and prognosis and with a follow-up \nplan explained.\nAcknowledgments: We are humbly grateful for the support \nand encouragement given by both Obstetrics and Gynecology \ndepartment at Bugando Medical Centre and Catholic University \nof Health & Allied Sciences (CUHAS).\nTimeline: The patient was admitted after the clinical work -\nout, emergency surgery and other management were immedi -\nately performed to the patient. Preparation and completion of \nthe case took 1 month, including follow up and after obtaining \nconsent.\nAuthor’s contribution: SN and DM played equal roles in eval-\nuating the patient before surgery, performed surgery and pre -\npared the initial drafts of this case report. OO did the histopath-\nological studies of the sample and reviewed several draft of this \nmanuscript, RK, SH and EN followed the patient post-surgery \nand later reviewed the final draft of the manuscript. RK did a \npelvic ultrasound and later reviewed several drafts of the manu-\nscripts. All authors read and approved the final manuscript.\nFunding: The cost of care offered to this patient was partly \ncovered by the patient and some was waived by the hospital \nadministration. The cost of preparing this manuscript and pub-\nlication was covered by the authors and the Catholic University \nof Health and Allied Sciences (CUHAS).\nConsent for publication: Written informed consent was ob-\n\nwww.jcimcr.org                Page 4\ntained from the patient for publication of this case and any ac -\ncompanying images. A copy of the written consent is available \nfor review by the Editor-in-Chief of this journal. Additionally, \nconsent was sought and granted by the Catholic University of \nHealth and Allied Sciences Directorate of Research and Publica-\ntion to publish this work. A copy of the clearance document is \nalso available for review by the Editor-in-Chief of this journal.\nCompeting interests: The authors declare that they have no \ncompeting interests..\nReferences\n1. Poddar P , Chavan K, Saraogi RM, Yadav P . 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