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CASE REPORT  
Year : 2020  |  Volume : 63  |  Issue : 2  |  Page : 295-297
Endometrial and cervical osseous metaplasia with extramedullary haematopoiesis presenting as amenorrhea: Report of three cases


Department of Pathology, All India Institute of Medical Sciences, Patna, India

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Date of Web Publication18-Apr-2020
 

   Abstract 


Endometrial and cervical osseous metaplasia (OM) is a rare phenomenon seen mostly in reproductive age group. Patients generally present with complaints of secondary infertility. Although few cases of endometrial OM have been described in literature, to the best of our knowledge, only six cases of OM in cervix have been reported so far. We report three cases of endometrial OM with extramedullary hematopoiesis, presented with complaints of amenorrhea and secondary infertility. In addition, one of the patients also had cervical OM.

Keywords: Cervix, endometrium, extramedullary hematopoiesis, osseous metaplasia

How to cite this article:
Jamal I, Gupta RK, Raj S, Bhadani PP. Endometrial and cervical osseous metaplasia with extramedullary haematopoiesis presenting as amenorrhea: Report of three cases. Indian J Pathol Microbiol 2020;63:295-7

How to cite this URL:
Jamal I, Gupta RK, Raj S, Bhadani PP. Endometrial and cervical osseous metaplasia with extramedullary haematopoiesis presenting as amenorrhea: Report of three cases. Indian J Pathol Microbiol [serial online] 2020 [cited 2020 May 30];63:295-7. Available from: http://www.ijpmonline.org/text.asp?2020/63/2/295/282700





   Introduction Top


Osseous metaplasia (OM) is defined as the presence of heterotopic normal bone tissue at unusual site. Overall reported incidence of uterine OM is about 0.3 per 1000 women.[1] Endometrial and cervical OM is a rare complication, it usually occurs after abortion, leading to secondary infertility. The etiopathogenesis of OM is controversial and many theories have been proposed to describe this phenomenon. More than 80% of the reported cases occur after pregnancy. Most recent and widely accepted theory is metaplasia of the endometrial stromal cells, usually fibroblasts, which change into osteoblasts and thus produce bone in the endometrium.[2] About 100 cases of endometrial OM have been reported in the world literature. The gold standard treatment is hysteroscopy, followed by complete curettage. Most of the patients regain their fertility after hysteroscopic removal of bony spicules. We present three cases of endometrial ossification with hematopoiesis, one with concomitant cervical OM with review of literature.


   Case Details Top


Case 1

A 26-year-old female G3P2 presented with amenorrhea and secondary infertility since 5 months. She also had a history of previous abortion at 4 months of gestation 3 years ago after which an intrauterine contraceptive device (IUCD, Copper-T) was inserted. Later, she developed per vaginal foul smelling discharge, which led to removal of Copper-T 2 years back. Ultrasonography (USG) revealed presence of multiple bony chips at lower uterine segment extending into endocervical region, largest measuring 9.5 mm [Figure 1]. Mantoux test and tuberculosis (TB) polymerase chain reaction (PCR) were negative. No intraepithelial lesion/malignancy seen in conventional Papanicolaou test (PAP) smear examination. Hysteroscopy with curettage of the bony tissue was done.
Figure 1: Ultrasound image showing multiple hyerechoic shadows (arrows) both in the endometrial and endocervical cavity

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Case 2

A 35-year-old woman G4P3 presented for evaluation of secondary amenorrhoea since 5 months. She also had a history of medical termination of pregnancy, followed by dilatation and evacuation 6 months ago. Her Mantoux test was positive (12 × 12 mm) but TB PCR was negative. USG revealed presence of multiple bony chips in the endometrial cavity. Hysteroscopic curettage of the bony tissue was performed.

Case 3

A 38-year-old woman G3P1 presented for evaluation of secondary amenorrohea and lower abdomen pain since 1 year. She gave history of induced abortion by medicines 5 years back but no dilatation and evacuation or curettage was done that time. Hysteroscopic curettage was done and sent for histopathology.

Pathological findings

Grossly samples from all three patients were comprised of multiple irregular grayish white to soft tissue pieces along with elongated bony chips.

Microscopically, all three cases showed multiple irregular lamellar and woven bony spicules with scattered endometrial glands in proliferative phase [Figure 2]a. In addition, Case 1 also revealed involvement of the cervix [Figure 2]b. Extramedullary hematopoiesis including erythroid, granulocytic, as well as megakaryocytic lineage was delineated in case 1 and 2 [Figure 2]c and [Figure 2]d. No granuloma, chorionic villi, or trophoblastic tissues were noted.
Figure 2: Histopathological images showing (a) multiple bony trabeculae with scattered endometrial glands (arrow) (HE ×100), (b) cervical osseous metaplasia with part of ectocervix (HE ×100), (c) low power (HE ×100), and (d) high power (HE ×400), showing extramedullary hematopoietic elements with arrow pointing a megakaryocyte. (Inset showing a megakaryocyte)

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   Discussion Top


Endometrial OM is an uncommon entity. Around 100 cases have been reported in world literature, including nine from India. Most of the cases present with secondary infertility and prior history of abortion. Other sites of ossification are cervix, vagina, and ovary.

Various hypotheses have been given to explain the pathogenesis of endometrial ossification such as hypercalcemia, hypervitaminosis D, hyperphosphatemia, chronic endometritis, pyometra, TB persistent hormonal stimulation, and osteogenesis promoted by retained fetal or necrotic tissue.

Perino et al. suggested chronic renal failure that is a known cause of abnormal calcium–phosphorous metabolism with metastatic calcifications could be the reason behind endometrial OM in their case.[2]

In most of the cases, patients belong to reproductive age group with a history of first trimester abortion. The indexed cases also had prior history of abortion. Time interval between previous abortion and detection of ossification can vary from 8 weeks to 23 years.[2],[3] In our study, the average time interval was 1.75 years. Shimzu and Nakayama described endometrial ossification in a 62-year-old postmenopausal woman who had a history of abortion 37 years ago. Sometimes patient with endometrial OM may have complaints of menstrual irregularities like menometrorrhagia and polymenorrhagia.[3] Both our cases presented with secondary amenorrhoea and infertility.

OM of cervix is caused by several factors such as injury to cervix by surgical trauma like loop electrosurgical excision procedure for severe dysplasia or by recurrent infection that cause mesenchymal cells to undergo OM.

Sabatini et al. reported cervical intraepithelial neoplasia (CIN) stage II in their case and explained that cervical OM could be a result of the inflammatory and traumatic processes caused by cervical infections, CIN, and cervical biopsy.

Elkattan et al. described a case of cervical OM in a nulligravida woman presented with primary infertility ruling out evidence of active infection, dysplasia, and metabolic disorders.[4]

In our study, the patient with cervical OM showed no intraepithelial lesion/malignancy in conventional PAP smear examination.

There are six reported cases of cervical OM in the literature with concomitant endometrial OM in two of the patients. In all the cases of cervical OM, endometrial cavity should be examined for concomitant involvement.[5]

Clinicians should be aware of OM and consider this entity in the differential diagnosis of patients with uncertain obstetrical history who present with radiological image resembling an IUCD. In one of our case, patient had a history of Copper-T insertion after abortion, following which she developed both endometrial and cervical OM. Chronic irritation and inflammation because of Copper-T might be the triggering factor for development of OM in the above patient.

Ultrasound examination is very crucial in the diagnosis of patients with OM, which characteristically shows a hyperechogenic material mimicking foreign body in the endometrial cavity. This should be further confirmed by hysteroscopic biopsy and histopathological examination.

Various types of mesenchymal metaplasia such as smooth muscle, osseous, cartilaginous, fatty change, foam cell change, and presence of glial tissue are described. Before classifying the heterologous tissue as benign, the pathologists should exclude the possibility component of malignant mixed mullerian tumor. In our case, no such tumor elements were noted.[6]

Genital TB should also be ruled out as it is a common cause of infertility in developing countries like India and can lead to subsequent calcification in the uterine cavity. In one of our cases, Mantoux test was positive; however, no epithelioid cell granuloma, necrosis, or acid fast bacilli was demonstrated and TB PCR was also negative. In none of our case, there was any metabolic disorder like hypervitaminosis D, hypercalcemia, or hyperphosphatemia.

Although both the cases in our study had a prior history of abortion, but no chorionic villi, trophoblastic or decidual tissues or fetal parts were microscopically identified.

To conclude, endometrial and cervical OM are conditions frequently overlooked and misdiagnosed and hence it is important to recognize non-neoplastic nature of this entity to avoid misinterpretation as malignant mixed mullerian tumor. It also resembles IUCD on USG having same contraceptive effect as that of IUCD. Hence, clinicians, radiologists, and pathologists should be aware of this condition to avoid an erroneous diagnosis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Sabatini L, Rainey AJ, Tenuwara W, Webb JB. Osseous metaplasia of cervical epithelium. BJOG 2001;108:333-34.  Back to cited text no. 1
    
2.
Perino A, Mangione D, Svelato A, Forlani F, Gargano F, Incandela D, et al. Chronic renal failure and endometrial osseous metaplasia: A hypothetical pathway. Acta Obstet Gynecol Scand 2013;92:111-19.  Back to cited text no. 2
    
3.
Shimizu M, Nakayama M. Endometrial ossification in a postmenopausal women. J Clin Pathol 1997;50:171-2.  Back to cited text no. 3
    
4.
Elkattan E, Abdelbadei M, Abdelmoaty H, Ali E, Samir D, Kheidr H. Osseous metaplasia of the cervix: A rare transformation. J Turk Ger Gynecol Assoc 2015;16:58-9.  Back to cited text no. 4
    
5.
Palat I, Sahin O, Yildrim G, Karaman E, Erim A, Tekirdag A. Osseous metaplasia of the cervix and endometrium: A case of secondary infertility. Fertil Steril 2011;95:2434.e1-4.  Back to cited text no. 5
    
6.
Umashankar T, Patted S, Hadingund R. Endometrial osseous metaplasia: Clinicopathological study of a case and literature review. J Hum Reprod Sci 2010;3:102-4.  Back to cited text no. 6
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Correspondence Address:
Rakesh K Gupta
Department of Pathology and Laboratory Medicine, Academic Block, First Floor, All India Institute of Medical Sciences, Patna - 801 505
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_574_18

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