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Novel treatment of a patient with secondary infertility due to retained fetal bone
FERTILITY AND STERILITY威 VOL. 79, NO. 4, APRIL 2003
CASE REPORTS
Copyright ©2003 American Society for Reproductive Medicine Published by Elsevier Science Inc. Printed on acid-free paper in U.S.A.
Novel treatment of a patient with secondary infertility due to retained fetal bone Kimberly Elford, M.D., and Paul Claman, M.D. Division of Reproductive Medicine, Department of Obstetrics and Gynecology, Ottawa Hospital, University of Ottawa, Ottawa, Ontario, Canada
Objective: To describe a simple and previously unreported treatment for retained fetal bone fragments as a cause of secondary infertility. Setting: Fertility center at a Canadian teaching hospital. Design: Case report. Patient(s): A 36-year-old woman with a 15-year history of secondary infertility. Intervention(s): A second dilation and curettage (D⫹C) performed under abdominal ultrasound guidance was performed where the curette could be directed for the removal of echogenic endometrial foci. Main Outcome Measure(s): Resolution of long-term infertility. Result(s): Spontaneous pregnancy 4 months after ultrasound-guided D⫹C and subsequent term delivery. Conclusion(s): If an echogenic area is discovered in the endometrium, it is now standard to look at the uterine cavity by hysteroscopy. However, if the hysteroscopy is normal, we suggest that a D⫹C with intraoperative abdominal ultrasound assistance be done to ensure that all the abnormal tissue is removed. (Fertil Steril威 2003; 79:1028 –30. ©2003 by American Society for Reproductive Medicine.) Key Words: Endometrial echo, retained fetal bone, secondary infertility, ultrasound
Over the past few decades, there have been a number of case reports of secondary infertility caused by retained fetal bone fragments. Many of these recent cases were discovered with hysteroscopic visualization. We present a case of a woman with a 15-year history of secondary infertility who had a completely normal hysteroscopy yet was found to have a bright echogenic endometrium on vaginal ultrasound examination. Received May 10, 2002; revised and accepted August 2, 2002. Reprint requests: Paul Claman, M.D., Ottawa Hospital Parkdale Clinic, 737 Parkdale Avenue, Suite 505, Ottawa, Ontario, Canada K1Y 4E9 (FAX: 613-761-4678; E-mail: pclaman@ottawahospital. on.ca). 0015-0282/03/$30.00 doi:10.1016/S0015-0282(02) 04919-1
1028
CASE REPORT A 36-year-old G1P0 presented for infertility treatment after 15 years of infertility with her current partner. She had previously conceived as a teenager with a different partner and had undergone a late first trimester therapeutic abortion. She had regular ovulatory menstrual cycles associated with mild dysmenorrhea. The investigations done included normal hysterosalpingogram, diagnosis of stage 1 endometriosis and treatment by laser laparoscopy, and
diagnosis of mild asthenoteratospermia on strict morphology semen analysis. The woman had previously been treated with three cycles of clomiphene citrate without success. After a discussion with the couple about their options, a decision was made to proceed with IVF and intracytoplasmic sperm injection for male factor infertility. A routine pelvic ultrasound done before the IVF cycle revealed two bright echogenic endometrial interfaces, each measuring approximately 8 mm in length. Hysteroscopy revealed a completely normal endometrial cavity, at which time curettings were taken. The grossly normal curettings demonstrated histologic evidence of multiple tiny bone fragments embedded in proliferative endometrium without evidence of endometritis. A repeat ultrasound was done postoperatively, which showed persistent bright echogenic areas at the endometrial interface.
FIGURE 1 The repeat vaginal ultrasound after the first D⫹C showing the uterus in sagittal view with two distinct echogenic areas measuring 8.5 and 6.8 mm in length at the endometrial interface.
Elford. Novel treatment for retained fetal bone. Fertil Steril 2003.
A second hysteroscopy with dilation and curettage (D⫹C) performed under transabdominal ultrasound guidance was arranged. The repeat hysteroscopy again showed a normal cavity and endometrium. Under ultrasound guidance, the posterior endometrial wall was curetted to clear away the echogenic material seen there. These curettings were seen to contain two fragments of bone on histologic examination. Four months after the ultrasound-guided D⫹C, she spontaneously conceived and delivered a healthy male infant at term.
DISCUSSION Over the past few decades there have been several case reports describing endometrial ossification. It is most commonly believed to be due to retained fetal bone fragments, but some cases may be due to metaplasia of mature endometrial stromal cells in response to chronic inflammation or trauma (1). Recently, it has been suggested that the incidence of this complication, after induced or spontaneous abortions, was underestimated in the literature (2). In the early 1990s, Melius et al. (3) found more than 50 cases in the literature, with 80% of them occurring after pregnancy. Most cases of endometrial ossification are discovered because of prolonged secondary infertility after a late therapeutic abortion. Dawood and Jarrett suggested that the bone fragments cause infertility by acting like intrauterine synechia or an intrauterine device, increasing endometrial prostaglandins and preventing implantation (4). Commenting on a similar case of infertility, Naftolin concluded in 1999 that “the bone remnants had been acting like an intrauterine contraceptive device and that a pregnancy following their removal is more than coincidence” (5). FERTILITY & STERILITY威
Unlike our patient, many women with retained fetal bone will have symptoms of menometrorrhagia, dysmenorrhea, vaginal discharge, pelvic pain, and spontaneous elimination of bony fragments in the menses in addition to their infertility (6). Lewis et al. measured menstrual blood volume and prostaglandin E2 concentrations before and after the removal of retained fetal bones in a woman with infertility and menorrhagia. He found the menstrual volume and total prostaglandin concentration decreased by 50% after the retained bone was removed (7). On reviewing the literature, the diagnosis of retained fetal bone was not discovered until hysterectomy in some patients. In others, it was discovered by a “gritty feeling” to the endometrium on D⫹C for abnormal bleeding and confirmed by pathology. In some cases, the fetal bone fragments were discovered on D⫹C after a filling defect was visualized on hysterosalpingogram. Bone fragments have been directly visualized protruding into the endometrial cavity on hysteroscopy in more recent cases. Occasionally, as in our case, an echogenic area in the endometrium on vaginal ultrasound may be the only clue to the presence of fetal bone fragments. A large secondary infertility case series by a Korean group (8) describes 10 women with infertility of 1–5 years’ duration after a therapeutic abortion. All of these women had a linear echogenic density discovered on vaginal ultrasound. After a D⫹C to remove the bone fragments (3 women required more then one curettage), all the women with open tubes conceived spontaneously and delivered healthy infants. Another recent case series with West African women (9) involved 11 women with secondary infertility lasting 2–15 years’ duration. All these women had a previous pregnancy termination performed between 10 and 26 weeks gestation. Diagnosis of retained fetal bone was made with transvaginal ultrasound in all cases. Hysteroscopy was then performed to remove the bone fragments, and 8 out of the 11 women subsequently conceived spontaneously. A blind D⫹C is sometimes diagnostically inaccurate and often ineffective as it may miss focal endometrial lesions. Chan (10) described how in his case the curettings from a blind D⫹C were normal before a hysteroscopy that visualized many retained fetal bone fragments. Although many recent case reports state that bone fragments can be directly visualized by hysteroscopy, that was not the situation that we encountered. We speculate that bone fragments held within the uterine cavity might stimulate myometrial contractions, which may further embed the fragments deep into the myometrium. Thus, spontaneous expulsion becomes less likely with passing time and normal endometrium can then overgrow the deeply embedded fragments. The best imaging technique that can readily visualize retained fetal bone is vaginal ultrasound. Any fetus that has attained at least 12 weeks gestation is capable of endochon1029
dral ossification. If that pregnancy is terminated or spontaneously aborted, any retained bone fragments might only be displayed on an ultrasound as bright echogenic areas with posterior shadowing. It is interesting to note that the presence of retained fetal bones may be more common in cases of uterine anomalies (3, 6). In these cases, care must be taken to understand the anomaly before instrumentation. This will minimize the risk of uterine perforation and will help to facilitate complete removal of the fragments. Wetzel (11) described a case of endometrial ossification in a septate uterus that was extremely difficult to evacuate and required a number of serial procedures.
CONCLUSION Our case stresses the need to perform endovaginal ultrasound in all women who present with secondary infertility who had a previous pregnancy that progressed beyond 11 weeks gestation and then ended in spontaneous or therapeutic abortion. It may not be adequate to just visualize the endometrium by hysteroscopy as some deeper endometrial pathology may be missed. If a bright echogenic area is found in the endometrium on ultrasound, we suggest that the abnormal tissue be removed by D⫹C under abdominal ultrasound guidance to ensure it is
1030 Elford and Claman
entirely removed at a single surgery. The simplicity of the treatment and the good postoperative prognosis justify an accurate and complete ultrasound examination of the endometrium in all women with secondary infertility who have a history of abortion. Not only will an ultrasound identify unusual endometrial pathology, it may identify uterine anomalies that may have been previously missed. References 1. Roth E, Taylor H. Heterotopic cartilage in the uterus. Obstet Gynecol 1966;27:838 –44. 2. Radestad A, Flam F. Intrauterine retention of fetal bones after abortion. Acta Obstet Gynecol Scand 1995;74:662–4. 3. Melius F, Julian T, Nagel T. Prolonged retention of intrauterine bones. Obstet Gynecol 1991;78:919 –21. 4. Dawood Y, Jarrett J. Prolonged intrauterine retention of fetal bones after abortion causing infertility. Am J Obstet Gynecol 1982;143: 715–7. 5. Naftolin N. A bone of contention: an unusual case of secondary infertility. Br J Obs Gyn 1999;106:1098 –9. 6. Chervenak F, Amin H, Neuwirth R. Symptomatic intrauterine retention of fetal bones. Obstet Gynecol 1982;59:58S–61S. 7. Lewis V, Khan-Dawood F, King M, Beckmann C, Dawood MY. Retention of fetal bone increases menstrual prostaglandins. Obstet Gynecol 1990;75:561–3. 8. Moon HS, Park YH, Kwon HY, Hong SH, Kim SK. Iatrogenic secondary infertility caused by residual intrauterine fetal bone after midtrimester abortion. Am J Obstet Gynecol 1997;176:369 –70. 9. Graham O, Cheng L, Parsons J. The ultrasound diagnosis of retained fetal bones in West African patients complaining of infertility. Br J Obstet Gynecol 2000;107:122–4. 10. Chan N. Intrauterine retention of fetal bone. Aust NZ Obstet Gynaecol 1996;36:368 –71. 11. Wetzels LC, Essed GG, de Haan J, van de Kar AJ, Willebrand D. Endometrial ossification: unilateral manifestation in a septate uterus. Gynecol Obstet Invest 1982;14:47–55.
Novel treatment for retained fetal bone
Vol. 79, No. 4, April 2003
CASE REPORTS
Copyright ©2003 American Society for Reproductive Medicine Published by Elsevier Science Inc. Printed on acid-free paper in U.S.A.
Novel treatment of a patient with secondary infertility due to retained fetal bone Kimberly Elford, M.D., and Paul Claman, M.D. Division of Reproductive Medicine, Department of Obstetrics and Gynecology, Ottawa Hospital, University of Ottawa, Ottawa, Ontario, Canada
Objective: To describe a simple and previously unreported treatment for retained fetal bone fragments as a cause of secondary infertility. Setting: Fertility center at a Canadian teaching hospital. Design: Case report. Patient(s): A 36-year-old woman with a 15-year history of secondary infertility. Intervention(s): A second dilation and curettage (D⫹C) performed under abdominal ultrasound guidance was performed where the curette could be directed for the removal of echogenic endometrial foci. Main Outcome Measure(s): Resolution of long-term infertility. Result(s): Spontaneous pregnancy 4 months after ultrasound-guided D⫹C and subsequent term delivery. Conclusion(s): If an echogenic area is discovered in the endometrium, it is now standard to look at the uterine cavity by hysteroscopy. However, if the hysteroscopy is normal, we suggest that a D⫹C with intraoperative abdominal ultrasound assistance be done to ensure that all the abnormal tissue is removed. (Fertil Steril威 2003; 79:1028 –30. ©2003 by American Society for Reproductive Medicine.) Key Words: Endometrial echo, retained fetal bone, secondary infertility, ultrasound
Over the past few decades, there have been a number of case reports of secondary infertility caused by retained fetal bone fragments. Many of these recent cases were discovered with hysteroscopic visualization. We present a case of a woman with a 15-year history of secondary infertility who had a completely normal hysteroscopy yet was found to have a bright echogenic endometrium on vaginal ultrasound examination. Received May 10, 2002; revised and accepted August 2, 2002. Reprint requests: Paul Claman, M.D., Ottawa Hospital Parkdale Clinic, 737 Parkdale Avenue, Suite 505, Ottawa, Ontario, Canada K1Y 4E9 (FAX: 613-761-4678; E-mail: pclaman@ottawahospital. on.ca). 0015-0282/03/$30.00 doi:10.1016/S0015-0282(02) 04919-1
1028
CASE REPORT A 36-year-old G1P0 presented for infertility treatment after 15 years of infertility with her current partner. She had previously conceived as a teenager with a different partner and had undergone a late first trimester therapeutic abortion. She had regular ovulatory menstrual cycles associated with mild dysmenorrhea. The investigations done included normal hysterosalpingogram, diagnosis of stage 1 endometriosis and treatment by laser laparoscopy, and
diagnosis of mild asthenoteratospermia on strict morphology semen analysis. The woman had previously been treated with three cycles of clomiphene citrate without success. After a discussion with the couple about their options, a decision was made to proceed with IVF and intracytoplasmic sperm injection for male factor infertility. A routine pelvic ultrasound done before the IVF cycle revealed two bright echogenic endometrial interfaces, each measuring approximately 8 mm in length. Hysteroscopy revealed a completely normal endometrial cavity, at which time curettings were taken. The grossly normal curettings demonstrated histologic evidence of multiple tiny bone fragments embedded in proliferative endometrium without evidence of endometritis. A repeat ultrasound was done postoperatively, which showed persistent bright echogenic areas at the endometrial interface.
FIGURE 1 The repeat vaginal ultrasound after the first D⫹C showing the uterus in sagittal view with two distinct echogenic areas measuring 8.5 and 6.8 mm in length at the endometrial interface.
Elford. Novel treatment for retained fetal bone. Fertil Steril 2003.
A second hysteroscopy with dilation and curettage (D⫹C) performed under transabdominal ultrasound guidance was arranged. The repeat hysteroscopy again showed a normal cavity and endometrium. Under ultrasound guidance, the posterior endometrial wall was curetted to clear away the echogenic material seen there. These curettings were seen to contain two fragments of bone on histologic examination. Four months after the ultrasound-guided D⫹C, she spontaneously conceived and delivered a healthy male infant at term.
DISCUSSION Over the past few decades there have been several case reports describing endometrial ossification. It is most commonly believed to be due to retained fetal bone fragments, but some cases may be due to metaplasia of mature endometrial stromal cells in response to chronic inflammation or trauma (1). Recently, it has been suggested that the incidence of this complication, after induced or spontaneous abortions, was underestimated in the literature (2). In the early 1990s, Melius et al. (3) found more than 50 cases in the literature, with 80% of them occurring after pregnancy. Most cases of endometrial ossification are discovered because of prolonged secondary infertility after a late therapeutic abortion. Dawood and Jarrett suggested that the bone fragments cause infertility by acting like intrauterine synechia or an intrauterine device, increasing endometrial prostaglandins and preventing implantation (4). Commenting on a similar case of infertility, Naftolin concluded in 1999 that “the bone remnants had been acting like an intrauterine contraceptive device and that a pregnancy following their removal is more than coincidence” (5). FERTILITY & STERILITY威
Unlike our patient, many women with retained fetal bone will have symptoms of menometrorrhagia, dysmenorrhea, vaginal discharge, pelvic pain, and spontaneous elimination of bony fragments in the menses in addition to their infertility (6). Lewis et al. measured menstrual blood volume and prostaglandin E2 concentrations before and after the removal of retained fetal bones in a woman with infertility and menorrhagia. He found the menstrual volume and total prostaglandin concentration decreased by 50% after the retained bone was removed (7). On reviewing the literature, the diagnosis of retained fetal bone was not discovered until hysterectomy in some patients. In others, it was discovered by a “gritty feeling” to the endometrium on D⫹C for abnormal bleeding and confirmed by pathology. In some cases, the fetal bone fragments were discovered on D⫹C after a filling defect was visualized on hysterosalpingogram. Bone fragments have been directly visualized protruding into the endometrial cavity on hysteroscopy in more recent cases. Occasionally, as in our case, an echogenic area in the endometrium on vaginal ultrasound may be the only clue to the presence of fetal bone fragments. A large secondary infertility case series by a Korean group (8) describes 10 women with infertility of 1–5 years’ duration after a therapeutic abortion. All of these women had a linear echogenic density discovered on vaginal ultrasound. After a D⫹C to remove the bone fragments (3 women required more then one curettage), all the women with open tubes conceived spontaneously and delivered healthy infants. Another recent case series with West African women (9) involved 11 women with secondary infertility lasting 2–15 years’ duration. All these women had a previous pregnancy termination performed between 10 and 26 weeks gestation. Diagnosis of retained fetal bone was made with transvaginal ultrasound in all cases. Hysteroscopy was then performed to remove the bone fragments, and 8 out of the 11 women subsequently conceived spontaneously. A blind D⫹C is sometimes diagnostically inaccurate and often ineffective as it may miss focal endometrial lesions. Chan (10) described how in his case the curettings from a blind D⫹C were normal before a hysteroscopy that visualized many retained fetal bone fragments. Although many recent case reports state that bone fragments can be directly visualized by hysteroscopy, that was not the situation that we encountered. We speculate that bone fragments held within the uterine cavity might stimulate myometrial contractions, which may further embed the fragments deep into the myometrium. Thus, spontaneous expulsion becomes less likely with passing time and normal endometrium can then overgrow the deeply embedded fragments. The best imaging technique that can readily visualize retained fetal bone is vaginal ultrasound. Any fetus that has attained at least 12 weeks gestation is capable of endochon1029
dral ossification. If that pregnancy is terminated or spontaneously aborted, any retained bone fragments might only be displayed on an ultrasound as bright echogenic areas with posterior shadowing. It is interesting to note that the presence of retained fetal bones may be more common in cases of uterine anomalies (3, 6). In these cases, care must be taken to understand the anomaly before instrumentation. This will minimize the risk of uterine perforation and will help to facilitate complete removal of the fragments. Wetzel (11) described a case of endometrial ossification in a septate uterus that was extremely difficult to evacuate and required a number of serial procedures.
CONCLUSION Our case stresses the need to perform endovaginal ultrasound in all women who present with secondary infertility who had a previous pregnancy that progressed beyond 11 weeks gestation and then ended in spontaneous or therapeutic abortion. It may not be adequate to just visualize the endometrium by hysteroscopy as some deeper endometrial pathology may be missed. If a bright echogenic area is found in the endometrium on ultrasound, we suggest that the abnormal tissue be removed by D⫹C under abdominal ultrasound guidance to ensure it is
1030 Elford and Claman
entirely removed at a single surgery. The simplicity of the treatment and the good postoperative prognosis justify an accurate and complete ultrasound examination of the endometrium in all women with secondary infertility who have a history of abortion. Not only will an ultrasound identify unusual endometrial pathology, it may identify uterine anomalies that may have been previously missed. References 1. Roth E, Taylor H. Heterotopic cartilage in the uterus. Obstet Gynecol 1966;27:838 –44. 2. Radestad A, Flam F. Intrauterine retention of fetal bones after abortion. Acta Obstet Gynecol Scand 1995;74:662–4. 3. Melius F, Julian T, Nagel T. Prolonged retention of intrauterine bones. Obstet Gynecol 1991;78:919 –21. 4. Dawood Y, Jarrett J. Prolonged intrauterine retention of fetal bones after abortion causing infertility. Am J Obstet Gynecol 1982;143: 715–7. 5. Naftolin N. A bone of contention: an unusual case of secondary infertility. Br J Obs Gyn 1999;106:1098 –9. 6. Chervenak F, Amin H, Neuwirth R. Symptomatic intrauterine retention of fetal bones. Obstet Gynecol 1982;59:58S–61S. 7. Lewis V, Khan-Dawood F, King M, Beckmann C, Dawood MY. Retention of fetal bone increases menstrual prostaglandins. Obstet Gynecol 1990;75:561–3. 8. Moon HS, Park YH, Kwon HY, Hong SH, Kim SK. Iatrogenic secondary infertility caused by residual intrauterine fetal bone after midtrimester abortion. Am J Obstet Gynecol 1997;176:369 –70. 9. Graham O, Cheng L, Parsons J. The ultrasound diagnosis of retained fetal bones in West African patients complaining of infertility. Br J Obstet Gynecol 2000;107:122–4. 10. Chan N. Intrauterine retention of fetal bone. Aust NZ Obstet Gynaecol 1996;36:368 –71. 11. Wetzels LC, Essed GG, de Haan J, van de Kar AJ, Willebrand D. Endometrial ossification: unilateral manifestation in a septate uterus. Gynecol Obstet Invest 1982;14:47–55.
Novel treatment for retained fetal bone
Vol. 79, No. 4, April 2003