FERTILITY AND STERILITY
Vol. 58, No.2, August 1992
Copyright © 1992 The American Fertility Society
Printed on acid-free paper in U.S.A.
Pseudomyxoma peritonei associated with secondary infertility
Holly Ann Hales, B.S. C. Matthew Peterson, M.D.* Christopher J. Jolles, M.D.
Michael W. Varner, M.D. William R. Keye, M.D.t
Division of Reproductive Endocrinology, Department of Obstetrics and Gynecology, University of Utah School of Medicine, Salt Lake City, Utah
Pseudomyxoma peritonei is a rare condition in which mucus-secreting epithelium coats peritoneal surfaces, resulting in progressive gelatinous mucinous ascites and ultimately bowel obstruction. This condition is usually associated with spontaneous rupture of a mucinous cystandenoma or cystadenocarcinoma of appendiceal or ovarian origin. In this report, we describe a woman with secondary infertility that we attribute to pseudomyxoma peritonei because of a ruptured appendiceal mucocele. Fertility was restored by resection of the primary tumor. CASE REPORT
A 33-year-old, gravida 2, with two full term deliveries presented in December of 1988 with infertility of 3 years' duration. She noted severe pelvic pain at ovulation that radiated down her legs and alternating bouts of constipation and diarrhea. Her symptoms progressed gradually over 2 years. Her basal body temperature charts were ovulatory, and semen analysis and postcoital tests were normal. A laparoscopy was performed as part of an infertility evaluation in January of 1989, revealing a moderate amount of green, gelatinous material in the pelvis that appeared to be coming from the fallopian tubes. This material was found in the tubes, Received January 20, 1992; revised and accepted March 25, 1992. * Reprint requests: C. Matthew Peterson, M.D., Division of Reproductive Endocrinology, Department of Obstetrics and Gynecology, University of Utah School of Medicine, 50 North Medical Drive, Salt Lake City, Utah 84132. t Present address: Beaumont Fertility Center, Royal Oak, Michigan. Vol. 58, No.2, August 1992
cul-de-sac, and around the ovaries. Cultures of the fluid were negative. The appendix was not seen. Initially, this condition was thought to be associated with a pelvic infection, and she was given a course of metronidazole and vibramycin for 10 days. The patient was lost to follow-up for 18 months. A second laparoscopy was repeated in August of 1991 to further evaluate the original finding. This was followed by a laparotomy revealing approximately two liters of gelatinous greenish/yellow fluid within the peritoneal cavity, consistent with pseudomyxoma peritonei. The peritoneal surfaces were red, thickened, and injected. An 8-cm diameter ruptured appendiceal mucocele was identified and a right hemicolectomy performed. Twenty-five to 30 cm of the right colon, including the cecum, and appendix were removed along with the terminal 30 cm of ileum. A primary anastomosis was performed. The uterus and fallopian tubes were normal in appearance, and both ovaries were normal in size and contour. They were not resected. Approximately 6 months later, after four spontaneous cycles in which attempts were made at conception, she conceived and had an uncomplicated pregnancy concluding at 39 weeks with a spontaneous vaginal delivery of a healthy female infant. At 3 months postpartum (18 months after bowel resection), there was no clinical evidence of recurrence. DISCUSSION
Pseudomyxoma peritonei is a disease of the reproductive and postmenopausal years. It is a rare entity that usually arises from ruptured mucinous Hales et al.
Communications-in-brief
425
l
neoplasms of the ovary 'or appendix. Viable mucin-secreting cells may implant on the peritoneal or omental surfaces with proliferation and mucous secretion resulting in mucinous ascites (1). Two cases of infertility resulting from pseudomyxoma peritonei have been reported (2); however, this is the first report managed successfully with restoration of fertility. Surgical resection is the only known effective therapy for this condition. Generally speaking, such therapy requires a hysterectomy, bilateral salpingooopharectomy, appendectomy, omentectomy, and resection of peritoneal tumor masses, resulting in permanent infertility (3). In this case, the reproductive organs were not resected and the ascites resolved. The patient was able to conceive spontaneously. We attribute her infertility to the peritoneal inflammation and mechanical inhibition of sperm -oocyte interaction caused by the mucinous ascites.
426
Hales et al.
Communications-in-brief
SUMMARY
In summary, we report a case of secondary infertility attributed to pseudomyxoma peritonei caused by ruptured appendiceal mucocele. Resection of the tumor and visible mucinous ascites resulted in spontaneous conception. We hypothesize secondary infertility was caused by significant peritoneal inflammation and inhibition of sperm-oocyte interaction from the ascites. Key Words: Pseudomyxoma peritonei, infertility, mucinous tumor. REFERENCES L Limber GK, King RE, Silverberg SG. Pseudomyxoma peritoneL a report of 10 cases. Ann Surg 1973;178:587-93. 2. Sugarbaker PH, Kern K, Lack E. Malignant pseudo myxoma peritonei of colonic origin: natural history and presentation of a curative approach to treatment. Dis Colon Rectum 1987;30:772-9. 3. Lee HL, Agha FP, Weatherbee L, Boland CR. Pseudomyxoma peritonei. J Clin Gastroenterol 1986;8:312-6.
Fertility and Sterility
Vol. 58, No.2, August 1992
Copyright © 1992 The American Fertility Society
Printed on acid-free paper in U.S.A.
Pseudomyxoma peritonei associated with secondary infertility
Holly Ann Hales, B.S. C. Matthew Peterson, M.D.* Christopher J. Jolles, M.D.
Michael W. Varner, M.D. William R. Keye, M.D.t
Division of Reproductive Endocrinology, Department of Obstetrics and Gynecology, University of Utah School of Medicine, Salt Lake City, Utah
Pseudomyxoma peritonei is a rare condition in which mucus-secreting epithelium coats peritoneal surfaces, resulting in progressive gelatinous mucinous ascites and ultimately bowel obstruction. This condition is usually associated with spontaneous rupture of a mucinous cystandenoma or cystadenocarcinoma of appendiceal or ovarian origin. In this report, we describe a woman with secondary infertility that we attribute to pseudomyxoma peritonei because of a ruptured appendiceal mucocele. Fertility was restored by resection of the primary tumor. CASE REPORT
A 33-year-old, gravida 2, with two full term deliveries presented in December of 1988 with infertility of 3 years' duration. She noted severe pelvic pain at ovulation that radiated down her legs and alternating bouts of constipation and diarrhea. Her symptoms progressed gradually over 2 years. Her basal body temperature charts were ovulatory, and semen analysis and postcoital tests were normal. A laparoscopy was performed as part of an infertility evaluation in January of 1989, revealing a moderate amount of green, gelatinous material in the pelvis that appeared to be coming from the fallopian tubes. This material was found in the tubes, Received January 20, 1992; revised and accepted March 25, 1992. * Reprint requests: C. Matthew Peterson, M.D., Division of Reproductive Endocrinology, Department of Obstetrics and Gynecology, University of Utah School of Medicine, 50 North Medical Drive, Salt Lake City, Utah 84132. t Present address: Beaumont Fertility Center, Royal Oak, Michigan. Vol. 58, No.2, August 1992
cul-de-sac, and around the ovaries. Cultures of the fluid were negative. The appendix was not seen. Initially, this condition was thought to be associated with a pelvic infection, and she was given a course of metronidazole and vibramycin for 10 days. The patient was lost to follow-up for 18 months. A second laparoscopy was repeated in August of 1991 to further evaluate the original finding. This was followed by a laparotomy revealing approximately two liters of gelatinous greenish/yellow fluid within the peritoneal cavity, consistent with pseudomyxoma peritonei. The peritoneal surfaces were red, thickened, and injected. An 8-cm diameter ruptured appendiceal mucocele was identified and a right hemicolectomy performed. Twenty-five to 30 cm of the right colon, including the cecum, and appendix were removed along with the terminal 30 cm of ileum. A primary anastomosis was performed. The uterus and fallopian tubes were normal in appearance, and both ovaries were normal in size and contour. They were not resected. Approximately 6 months later, after four spontaneous cycles in which attempts were made at conception, she conceived and had an uncomplicated pregnancy concluding at 39 weeks with a spontaneous vaginal delivery of a healthy female infant. At 3 months postpartum (18 months after bowel resection), there was no clinical evidence of recurrence. DISCUSSION
Pseudomyxoma peritonei is a disease of the reproductive and postmenopausal years. It is a rare entity that usually arises from ruptured mucinous Hales et al.
Communications-in-brief
425
l
neoplasms of the ovary 'or appendix. Viable mucin-secreting cells may implant on the peritoneal or omental surfaces with proliferation and mucous secretion resulting in mucinous ascites (1). Two cases of infertility resulting from pseudomyxoma peritonei have been reported (2); however, this is the first report managed successfully with restoration of fertility. Surgical resection is the only known effective therapy for this condition. Generally speaking, such therapy requires a hysterectomy, bilateral salpingooopharectomy, appendectomy, omentectomy, and resection of peritoneal tumor masses, resulting in permanent infertility (3). In this case, the reproductive organs were not resected and the ascites resolved. The patient was able to conceive spontaneously. We attribute her infertility to the peritoneal inflammation and mechanical inhibition of sperm -oocyte interaction caused by the mucinous ascites.
426
Hales et al.
Communications-in-brief
SUMMARY
In summary, we report a case of secondary infertility attributed to pseudomyxoma peritonei caused by ruptured appendiceal mucocele. Resection of the tumor and visible mucinous ascites resulted in spontaneous conception. We hypothesize secondary infertility was caused by significant peritoneal inflammation and inhibition of sperm-oocyte interaction from the ascites. Key Words: Pseudomyxoma peritonei, infertility, mucinous tumor. REFERENCES L Limber GK, King RE, Silverberg SG. Pseudomyxoma peritoneL a report of 10 cases. Ann Surg 1973;178:587-93. 2. Sugarbaker PH, Kern K, Lack E. Malignant pseudo myxoma peritonei of colonic origin: natural history and presentation of a curative approach to treatment. Dis Colon Rectum 1987;30:772-9. 3. Lee HL, Agha FP, Weatherbee L, Boland CR. Pseudomyxoma peritonei. J Clin Gastroenterol 1986;8:312-6.
Fertility and Sterility
