Simultaneous osseous metaplasia nodules of the submucosa and mesosalpinx after first trimester abortion: a case report
European Journal of Medical Research volume 18, Article number: 47 (2013)
Here, we report a case of simultaneous osseous metaplasia nodules of the submucosa and mesosalpinx after a first trimester abortion.
A 36-year-old woman presented to the Women’s Hospital, School of Medicine, Zhejiang University with vaginal bleeding and infertility resulting from osseous metaplasia nodules of the submucosa and mesosalpinx after a first trimester abortion. Diagnostic and operative hysteroscopy and laparoscope procedures were performed. The osseous metaplasia nodules disappeared after hysteroscopy and laparoscope interventions; 2 weeks postoperatively, the patient underwent a transvaginal ultrasound examination and the abnormal ultrasound appearance had resolved.
Osseous metaplasia nodules in the submucosa and mesosalpinx can be a rare cause of vaginal bleeding and infertility. Autologous tissue, not persistent heterologous tissue, may be the true reason for metaplasia. Treatment is by ultrasound-guided curettage or by hysteroscopic and laparoscope removal.
Osseous metaplasia occurs in approximately 3 in every 10,000 women. A history of abortion, either spontaneous or therapeutic, is the hallmark of this condition. The etiology and pathogenesis are controversial. Retained fetal bones and inflammatory response following the abortion are thought to be the reasons for endometrial ossification.
Bony fragments in the uterus occur after second trimester termination of pregnancy[1–3]. Very rarely, bony fragments can form following first trimester loss. The clinical presentation of osseous metaplasia can be pelvic pain, vaginal bleeding, menstrual irregularities, dyspareunia, and secondary infertility[2, 3, 5]. Osseous metaplasia is easily diagnosed by ultrasound examination, revealing an echogenic band, and diagnosis can subsequently be confirmed by hysteroscopy[1, 2, 6]. In this report, we present a rare case with osseous metaplasia involving the intrauterine submucosa and mesosalpinx after the first trimester loss.
A 36-year-old woman was admitted to our hospital with a complaint of vaginal bleeding and infertility. Her medical history revealed she had regular menstrual cycles with no history of endocrine abnormalities. She had undergone four pregnancy terminations: one at 12 weeks’ gestation 12 years ago, one at 20 weeks’ gestation 9 years ago, one at 12 weeks’ gestation 6 years ago and one at 6 weeks’ gestation 2 years ago. The abortions were all electively terminated by dilatation and curettage. A pelvic examination revealed a normal size and normal adnexa. The results of laboratory investigations, which included a blood count, urine analysis, serum calcium, phosphorus, endocrine hormone levels and her partner’s spermiogram, were within normal limits.
During the tests, the transvaginal ultrasonography displayed two strong echogenic stripes with attenuation in the uterine submucosa and mesosalpinx (Figure 1). Two separate bony nodules were seen on three-dimensional multislice computed tomography (MSCT-3D) (Figure 2). Diagnostic hysteroscopic and laparoscope examinations performed in the operating room also revealed two separate bony nodules in the submucosa and mesosalpinx (Figure 3). A biopsy was obtained and the histology established the diagnosis of osseous metaplasia of the submucosa and mesosalpinx (Figure 4).
At 2 weeks postoperatively, the patient underwent a transvaginal ultrasound examination; the abnormal ultrasound appearance had resolved.
The ossification of the endometrium is a rare benign disease[6–8]. In the present case, the patient had a history of four terminations of pregnancy and osseous metaplasia nodules were found on transvaginal ultrasonography after the latest first trimester termination. First trimester abortions are an unusual cause of fetal bones, though this has been reported in a case previously. The period of time between abortion and diagnosis of the disease can range from 8 weeks to 23 years[6, 9]. In our case, the time interval was 2 years.
Ossification has also been reported in the cervix, the ovary, and the vagina[2, 10–12]. To the best of our knowledge, this is the first reported case of osseous metaplasia nodules of both the submucosa and mesosalpinx. Persistent heterologous tissue, such as retained fetal bone, has been suggested as the etiology of osseous metaplasia of the endometrium in most reports[1–3]. However, it is unlikely that the endometrial bone is of fetal origin because there was no fetal tissue found in the biopsy material studied after the first trimester abortion, and the biopsy also showed minimal or no tissue reaction. Thus, we predict that the osseous metaplasia may have a mullerian origin, arising in the myoendometrial transitional zone.
Studies have shown that chronic endometritis can stimulate the proliferation of mesenchymal cells that have inherent metaplasia properties and can differentiate into chondroblasts or osteoblasts. Recently, Cayuela et al. used DNA analysis to show that endometrial ossification is not of fetal origin, but derived from the patient’s own tissue. Additionally, our patient has undergone four curettage surgeries that may have resulted in chronic pelvic inflammation, causing the patient’s own tissue to differentiate into chondroblasts or osteoblasts. Therefore, persistent heterologous tissue may not be the cause of osseous metaplasia nodules of the mesosalpinx.
Interestingly, osseous metaplasia nodules, not multiple small and hard bony spicules, of the submucosa and mesosalpinx formed simultaneously in present case. Leiomyoma with osseous differentiation and mature teratoma can mimic osseous metaplasia nodules. However, our patient’s osseous metaplasia nodules contained neither mature smooth muscle areas nor epithelial components, which are the characteristics of uterine leiomyoma with osseous differentiation and mature teratoma, respectively.
The diagnosis is suspected at ultrasound, MSCT-3D and confirmed by hysteroscopy, laparoscope and histopathologic examination. Treatment is by ultrasound guided curettage or by hysteroscopic and histopathologic removal. Autologous tissue, not persistent heterologous tissue, is resulting in a true metaplasia mostly.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
Zhou Feng and Qin Jiale are the joint first authors.
Coccia ME, Becattini C, Bracco GL, Scarselli G: Ultrasound-guided hysteroscopic management of endometrial osseous metaplasia. Ultrasound Obstet Gynecol 1996, 8: 134–136. 10.1046/j.1469-0705.1996.08020134.x
Bhatia NN, Hoshiko MG: Uterine osseous metaplasia. Obstet Gynecol 1982, 60: 256–259.
Polat I, Sahin O, Yildirim G, Karaman E, Erim A, Tekirdag AI: Osseous metaplasia of the cervix and endometrium: a case of secondary infertility. Fertil Steril 2011, 95: 2434-e4. e1–e4
Singla A, Gupta B, Guleria K: First trimester abortion: a rare cause of intrauterine bony spicules. Case Rep Obstet Gynecol 2012, 2012: 701021.
Akbulut M, Ege CB, Kelten EC, Soysal ME: A probable secondary infertility due to osseous metaplasia of the endometrium. Arch Gynecol Obstet 2008, 277(6):563–565. 10.1007/s00404-007-0515-1
Rosa-e-Silva JC, Barcelos ID, Navarro PA, Rosa-e-Silva AC, Nogueira AA, Ferriani RA: Osseous metaplasia of the endometrium associated with infertility: a case report and review of the literature. J Med Case Reports 2009, 3: 7427. 10.4076/1752-1947-3-7427
Perino A, Mangione D, Svelato A, Forlani F, Gargano F, Incandela D, Coppola MA, Venezia R: Chronic renal failure and endometrial osseous metaplasia: a hypothetical pathway. Acta Obstet Gynecol Scand 2013, 92(1):118–119. 10.1111/j.1600-0412.2012.01487.x
Damiani GR, Seghezzi U, Gaetani M, Riva D, Tartagni M, Loverro G: Intrauterine bone fragments and endometrial plaque of osseous metaplasia. Acta Obstet Gynecol Scand 2012, 91(8):1001. 10.1111/j.1600-0412.2012.01388.x
Basu M, Mammen C, Owen E: Bony fragments in the uterus: an association with secondary subfertility. Ultrasound Obstet Gynecol 2003, 22: 402–406. 10.1002/uog.221
Bedaiwy MA, Goldberg JM, Biscotti CV: Recurrent osseous metaplasia of the cervix after loop electrosurgical excision. Obstet Gynecol 2001, 98: 968–970. 10.1016/S0029-7844(01)01486-7
Campo S, Campo V, Zannoni GF, Gambadauro P: Simultaneous ovarian and endometrial osseous metaplasia: a case report. J Reprod Med 2007, 52: 241–242.
Landim FM, Tavares JM, de Melo Braga DN, da Silva JE Jr, Bastos Filho JB, Feitosa RG: Vaginal osseous metaplasia. Arch Gynecol Obstet 2009, 279: 381–384. 10.1007/s00404-008-0706-4
Umashankar T, Patted S, Handigund RS: Endometrial osseous metaplasia: clinicopathological study of a case and literature review. J Hum Reprod Sci 2010, 3(2):102–104. 10.4103/0974-1208.69329
Shroff CP, Kudterkar NG, Badhwar VR: Endometrial ossification–report of three cases with literature review. Indian J Pathol Microbiol 1985, 28: 71–74.
Cayuela E, Perez-Medina T, Vilanova J, Alejo M, Canadas P: True osseous metaplasia of the endometrium: the bone is not from a fetus. Fertil Steril 2009, 91: 1293.
We thank Medjaden Bioscience Limited for providing medical writing services.
The authors state they have no competing interests.
All authors made substantial contributions to conception and design, or acquisition of data, or analysis and interpretation of data; ZF and HL involved in drafting the manuscript and revising it critically for important intellectual content; HL gave final approval to the version to be published. All authors read and approved the final manuscript.
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Feng, Z., Jiale, Q., Xiaofei, Z. et al. Simultaneous osseous metaplasia nodules of the submucosa and mesosalpinx after first trimester abortion: a case report. Eur J Med Res 18, 47 (2013). https://doi.org/10.1186/2047-783X-18-47