K. MARGOLIS, M. HAHER AND M. SIDHU 513

normal within 1 month. The other patient (Case 1 ) when last seen had an ongoing pregnancy with no visible abnormality on ultrasound.

The chance of recurrence of a hydatidiform mole in a subsequent pregnancy is reported to be 1 % after one previous mole (8) and 20-25% after 2 previous moles. (6, 8). The increased risk persists even if the patient conceives with a different partner (8). The risk of persistent GTD if a second mole follows a CHM is 50% (8). Careful histological review of the placenta and repeated follow up postnatal beta HCG measurements are therefore essential.

Acknowledgements

The authors wish to thank Sue Healey, from Nuclear Genetics in the Department of Pathology at the Royal Brisbane Hospital, for doing the genetic analysis and Professor S.K. Khoo and Dr B Hill of the Department of Obstetrics and Gynaecology at the Royal Women's Hospital, for allowing them access to the clinical records of Case 2.

References

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Steller MA, Genest DR, Bemstein MR, Lage JM, Goldstein DP, Berkowitz RS. Natural history of twin pregnancy with complete hydatidiform mole and coexisting fetus. Obstet Gynecol 1994;

Miller D, Jackson R, Ehien T, McMurtrie E. Complete hydatidiform mole coexistent with a twin live fetus: clinical course of four cases with complete cytogenetic analysis. Gynecol Oncol 1993; 50: 119-123. Fishman DA, Padilla LA, Keh P, Cohen L, Frederiksen M, Lurain JR. Management of twin pregnancies consisting of complete hydatidiform mole and normal fetus. Obstet Gynecol 1998; 91: 546-550. Soto-Wright V, Bernstein M, Goldstein DP, Berkowitz RS. The changing presentation of complete molar pregnancy. Obstet Gynecol 1995; 86: 775-779. Vejerslev LO. Clinical management and diagnostic possibilities in hydatidiform mole with coexistent fetus (review). Obstet Gynecol SUIT 1991; 46: 577-588. Baergen RN, Kelly T, McGinniss MJ, Jones OW, Benirschke K. Complete hydatidiform mole with coexistent embryo. Hum Pathol 1996; 27: 731-734. Goldstein DP, Berkowitz RS. Current management of complete and partial molar pregnancy. J Reprod Med 1994; 39: 139-146. Berkowitz RS, Bernstein MR, Laborde 0, Goldstein DP. Subsequent pregnancy experience in patients with gestational trophoblastic disease. New England Trophoblastic Centre, 1965- 1992. J Reprod Med 1994; 39: 228-232

83: 35-42

Aust NZ J Obstet Gynaecol 1999; 39: 4: 513-515

Endometrial Ossification in Infertile Patients - Report of 3 Cases Harsh M ~ h a n ' . ~ MD, MNAMS, Ritambhra Nada2,3 MD, Praveen M0han~7~ MD DGO, R. S. Punia3 MD

and Sarla Malh~t ra ' .~ MD Departments of Patholog$ and Obstetrics and Gynaecology4, Government Medical College, Chandigarh, India

EDITORIAL COMMENT We accepted this paper for publication because endometrial bone formation is an interesting pathological condition and its relationship to infertility is of interest. Our only publication on this subject (A) was a case of a woman with secondary infertility who had had a previous termination. i n this case the woman became pregnant in the cycle following curettage and removal of the bony fragment in the uterus which was presumably retained fetal tissue. The present case reports are diferent and the authors have reviewed the other associations of endometrial bone formation reported in the literature including those associated with uterine tumours. N.B.

(A) Bellingham FR. Endometrial bone formation. Aust NZ J Obstet Gynaecol 1996; 36: 109- 1 10. 1. Professor and Head

2. Reader. 3. Senior Lecturer.

Address for correspondence: Dr. Harsh Mohan, House No. 1 112, GMC Campus, Sector 32-B Chandigarh - 160047, India.

s14 AUST AND NZ JOURNAL OF OBSTETRICS AND GYNAECOLOGY

Figure 1. Photomicrograph showing pieces of bone surrounded by sparse lymphomononuclear cell infiltrate beneath the endometrial lining (H and E X100).

Endometrial ossification is very rare in the human uterus (1-4). It has been documented in cases of prolonged use of oral contraceptives, intrauterine contraceptive device (IUCD), Asherman syndrome or with papillary adenocarcinoma of the uterus and mixed Mullerian tumours of uterus (5-7). It has been associated with chronic endometritis following retention of fetal parts (8-9) or seen in primary infertility due to chronic endometritis with osseous metaplasia (10). Clinical presentations may include abnormal vaginal bleeding or discharge, dysmenorrhea and secondary infertility (8-9, 11-12). It has rarely been associated with primary infertility (10). Here we present 3 cases, 2 of primary infertility and 1 of secondary infertility, where endometrial ossification was noted during routine histopathological examination.

Case I

A 34-year-old female, married for 10 years was unable to conceive. Scanty endometrial curettings were obtained during routine &agnostic endometrial biopsy. Histopathological examination revealed a fragment of ossified bone below the endometrial lining accompanied with lymphomononuclear cells and a few endocervical glands. No granulomas were present (figure 1) .

Case 2

A 19-year-old female, married for 2 years had irregular bleeding per vaginam for 1 month. There was no history of preceding amenorrhoea. Endometrial curettage yielded gritty uterine curettings which on histopathological examination showed bone and cartilage along with fibrinous material. There were no chorionic villi, granulomas or endometrial glands.

Case 3

A 30-year-old female married for 10 years had had a female baby by Caesarean section. There was no

history of abortion or use of intrauterine device or oral contraceptives. She presented with secondary infertility for 7 years. The endometrial curettings were irregular, soft to gritty. On histopathological examination, the endometrial tissue showed ossification, calcification, haemorrhage and a few endometrial glands in the secretory phase. There was no significant inflammatory response and staining for tubercle bacilli was negative.

DISCUSSION

Endometrial biopsy evaluation constitutes an important part of the complex investigative work-up of infertility. Endometrial biopsy could reflect indirectly, ovarian dysfunction or can be employed for detecting primary endometrial lesions. Chronic endometritis can be caused by microorganisms e.g. Mycobacterium tuberculosis, mycoplasma, chlamydia or it can be nonspecific endometritis. Nonspecific endometritis is due to ascending infection from the lower genital tract or follows abortion or delivery due to retained decidua or fetal parts and may result from mechanical obstruction to menstrual flow due to cervical or isthmic lesions (€49, 13). It has been rarely associated with sterility (8-10, 14). Nonspecific endometritis can follow retention of fetal bones with secondary infertility (8-9) or can cause meta- plastic endometrial ossification with primary infer- tility (10) as in 2 of our cases. Probably operative trauma was responsible for metaplastic ossification in Case 3 in which the woman had secondary infertility as her first baby was delivered by Caesarean section.

In addition to occurrence with chronic endometritis, endometrial ossification can occur due to intake of contraceptive pills, prolonged use of an intrauterine contraceptive device (IUCD), Asherman syndrome, papillary adenocarcinoma of the endometrium and malignant mixed mesoder- mal tumour of the endometrium (1-5).

Heterotopic osseous tissue formation in the endometrium is very rare; we encountered these 3 cases out of 285 specimens of endometrial biopsy submitted in cases of infertility for histopathological evaluation over a period of 2 years. These biopsy specimens were received from the twin tertiary-care hospitals attached to the Institution (225 bed GMCH and 500 bed General Hospital) where the combined annual delivery rate is approximately 12,500 and cases of infertility constitute 14% of the outpatient attendance in the Gynaecology and Obstetric Department.

Histogenesis of endometrical ossification has been speculated upon differently by various authors. It may be due to prolonged intake of calcium and vitamin D, chronic inflammation as a result of retention of fetal parts in incomplete abortion, or due to foreign body in

PAUL A. MOWN ET ALL 515

the uterus in the form of an IUCD (1, 5, 6). In our cases the patient had secondary infertility for 7 years after Caesarean section and the endometrial ossification could have been due to operative trauma.

Patients with endometrial ossification can present with irregular bleeding, vaginal discharge, dysmenon-hea or secondary infertility (8, 9, l l ) . It is rarely documented in patients with primary infertility, including a woman who had ossification in 1 side of a septate uterus (10). Endometrial ossification can be treated by repeated dilatation and curettage, hysterectomy or by hysteroscopic removal (1 1, 12). In 2 of our cases the women had primary infertility while in the third the secondary infertility was not due to retention of fetal bones as her baby was delivered by Caesarean section. However, none of the 3 women reported here have conceived yet during follow-up for 9 months, 1 year 4 months, and 2 years, respectively.

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10. Wetze l sk , Essed GG, AeHaan J, Van de Kar AJ, Willibrand D. Endometrial ossification: unilateral manifestation in a septate uterus. Gynecol Obstet Invest 1982; 14: 47-55.

11.Tome A, Jau P, Pagano R, Sanchez I, Ordi J, Vanrell JA. Endometrial ossification successfully treated by hysteroscopic resection. Eur J Obstet Gynecol Reprod Biol 1996; 66: 75-77.

12.Bellingham FR. Endometrial bone formation. Aust NZ J Obstet Gynaecol 1996; 36: 109-110.

13.Honore LH. Pathology of Infertility. In Haines and Taylor Obstetrical and Gynaecological Pathology, 3rd ed. London: Churchill Livingstone (ed H. Fox), 1987,718-817.

14.Czernobilsky B. Endometritis and infertility. Fertil Steril 1978; 30: 119-130.