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Spontaneous viable pregnancies in cervical and rectal endometriosis: a report of two cases
Spontaneous viable pregnancies in cervical and rectal endometriosis: a report of two cases Asha Latha Ganesh, M.B.B.S.,a and Baidyanath Chakravarty, M.D.b a
School of Medical Science and Technology, Indian Institute of Technology, Kharagpur, and Medicine, Salt Lake, Kolkata, India
b
Institute of Reproductive
Objective: To report two cases of advanced pelvic endometriosis, both with deep rectal involvement and one with extensive cervical involvement appearing like a carcinoma, where both patients achieved spontaneous pregnancy and delivered viable babies. Design: Case report. Setting: Institute of Reproductive Medicine. Patient(s): Two nulliparous patients, one with extensive cervical and rectal and other with deep rectal endometriosis and primary infertility. Intervention(s): Medical management. Main Outcome Measure(s): Description and treatment of two patients with advanced pelvic endometriosis and pregnancy. Result(s): Delivery of viable babies in both cases. Conclusion: Prolonged medical treatment may have helped to arrest the progression of the disease by restoring anatomic proximity leading to spontaneous pregnancy in both cases. (Fertil Steril威 2007;87:697.e1– 4. ©2007 by American Society for Reproductive Medicine.) Key Words: Cervical endometriosis, advanced pelvic endometriosis, polypoidal mass, pregnancy
Endometriosis is a condition in which ectopic endometrial glands and stroma are found outside the uterus (1). Most endometrial deposits are found in the pelvis (ovaries, peritoneum, uterosacral ligaments, pouch of Douglas, and rectovaginal septum). Extrapelvic deposits, including those in the umbilicus and diaphragm, are rare (2).
involvement appearing like a carcinoma, where both patients achieved spontaneous pregnancy and delivered viable babies, are reported. Approval was obtained for both the cases from the institutional review board before surgical intervention.
The incidence of endometriosis is reported as 4%–17% of all women during their reproductive age. Endometriosis of the colon and rectum, though rare, comprises 3%–34% of these endometriosis cases (3). A colposcopic examination series reports the incidence of cervical endometriosis to be 0.11%–2.4% (4).
CASE 1 A 28-year-old nulliparous woman, married for 6 years, was seen at our clinic with rectal bleeding and persistent mucous discharge. She had regular menstrual cycles initially after attaining menarche at the age of 13 years; 7 years later she started to have irregular cycles. Her chief complaint of rectal bleeding was of 2 years duration. She was treated earlier with progesterone but was not relieved of her symptoms.
The most commonly reported symptoms of endometriosis are dysmenorrhea (60%– 80%), pelvic pain (30%–50%), and infertility (30%– 40%). Diagnosis is based on clinical and histologic findings. The conservative medical line of treatment aims at the arrest of growth and activity of ectopic endometrial implants. The second line of management would be surgical resection of as much of the endometriotic tissue as possible. A combination of the two therapies may also be used, if necessary. Assisted reproductive techniques may be used in patients with infertility. Two rare cases of advanced pelvic endometriosis, both with deep rectal involvement and one with extensive cervical Received for publication February 18, 2006; revised and accepted June 3, 2006. Reprint requests: Baidyanath Chakravarty, M.D., Institute of Reproductive Medicine, HB36/A/3, Sector III, Salt Lake, Kolkata, India 700106 (FAX: 91-33-2337-6290; E-mail: [email protected], [email protected]).
0015-0282/07/$32.00 doi:10.1016/j.fertnstert.2006.06.061
Vaginal examination revealed firm consistency of the cervix and an indurated posterior lip, extending to the upper third of the vaginal vault, more on the posterior vaginal wall. The cervix appeared to be fixed posteriorly with the anterior rectal wall. The uterus was bulky with restricted mobility. On speculum examination, a fungating growth (3 ⫻ 2.5 cm) with polypoidal projection involving the posterior lip of the cervix was seen (Fig. 1). On speculum insertion, bleeding from the lesion was noticed. On rectal examination, parametrium was found to be indurated extending up to the lateral pelvic wall on both sides. Rectal mucosa was indurated, ulcerated, and fixed to the growth. The tip of the examining finger was stained with blood. On the basis of these clinical findings, stage IV carcinoma of the cervix was diagnosed provisionally.
Fertility and Sterility姞 Vol. 87, No. 3, March 2007 697.e1 Copyright ©2007 American Society for Reproductive Medicine, Published by Elsevier Inc.
FIGURE 1 Speculum examination showing a fungating growth (3 ⫻ 2.5 cm) with polypoidal projection involving posterior lip of cervix in case 1.
but periodic episodes of heavy bleeding continued. Surgical castration by bilateral oophorectomy was considered because of inadequate response and inability of the patient to afford prolonged GnRH-agonist therapy. Because of severe rectal involvement, hysterectomy was not attempted as it would involve a posterior exenteration type of surgery with permanent colostomy. On laparotomy, the pelvis was found to be frozen. The uterus, tubes, and ovaries were not visualized on either side. With further dissection, a part of the anterior wall of the uterus and both adnexa could be identified. The left ovary had a small chocolate cyst, and the right ovary was normal. Ovaries on both sides and part of the body of the uterus (at the level of the round ligament) that could be visualized were removed. After surgery, the wound healed with secondary intention and without major complications. Subsequently her hemoglobin level improved, and no further vaginal or rectal bleeding was reported. CASE 2 A 29-year-old nulliparous woman, married for 5 years, was referred to our institute as a case of advanced endometriosis
Ganesh. Endometriosis with spontaneous pregnancy. Fertil Steril 2007.
Hemoglobin was found to be 8.4 gm/dL. Ultrasonographic examination of the lower abdomen and pelvis revealed a bulky uterus with an ill-defined space-occupying lesion involving the cervix and extending into the vagina. Ovaries on both sides were normal.
FIGURE 2 Polypoidal growth removed in case 1 and examined histopathologically. Findings showing cervical squamous epithelium at top and stroma below with several endometrial glands.
Initially, hematinics were given; examination under anesthesia confirmed the speculum findings. The polypoidal growth was removed with use of sponge forceps and sent for histopathologic examination, which confirmed endometriosis (Fig. 2). The patient was given danazol for 5 months, but bleeding could not be controlled adequately. The polypoidal growth recurred and was removed vaginally. Subsequently, danazol was combined with GnRH-agonist, after which bleeding stopped. Both drugs were withdrawn after 4 months. She had a normal cycle after 1 month, following which she became amenorrheic. Urine pregnancy test was positive. She did not attend our antenatal clinic regularly, as advised. She had an uneventful antenatal period and later came to our clinic at 32 weeks with preterm onset of labor. She delivered normally a live female baby weighing 2.2 kg, without any cervical tear or postpartum hemorrhage. Six months after delivery, she reported with continuous rectal and vaginal bleeding. Her hemoglobin was 7.8 gm/dL, and recurrence of similar polypoidal growth with dense rectal involvement was observed. Gonadotropin-releasing hormone-agonist and hematinics were given for 9 months, 697.e2 Ganesh and Chakravarty
Ganesh. Endometriosis with spontaneous pregnancy. Fertil Steril 2007.
Endometriosis with spontaneous pregnancy
Vol. 87, No. 3, March 2007
with pelvirectal sinus. She had severe pain in the lower abdomen, irregular bleeding from the vagina, rectal bleeding and mucous discharge, and primary infertility of 5 years’ duration. Throughout her menstrual history, after menarche onset at the age of 12 years (1978), she had had dysmenorrhea and intermenstrual bleeding. Her medical and surgical history is significant and is briefly reviewed. She had recurrent episodes of acute abdomen for which laparotomy was performed on four occasions between 1978 and 1997. The indications and findings of laparotomy, histologic examination reports, line of management after the surgeries, and other relevant events are summarized chronologically. The first laparotomy (1978) was performed 6 months after menarche for acute pain in the lower abdomen. The pelvic cavity was found to be filled with chocolate-colored fluid mixed with pus. Her left ovary was enlarged and adherent to the pouch of Douglas. Her left ovary was partially resected and diagnosed as an infected chocolate cyst of the left ovary. The histologic report was not available. Later for 6 years she had had intermittently severe dysmenorrhea and rectal discharge for which she was treated with progesterones and danazol periodically for temporary relief. A second laparotomy (1989) was performed for acute abdomen as an emergency. On laparotomy, the abdomen and pelvic organs were found to be plastered. The abdomen was closed without any interference. Antitubercular drugs were started empirically; after 4 months jaundice developed, and the antitubercular drugs were stopped temporarily. After treatment of jaundice, antitubercular drugs were continued. The patient, however, continued to have dysmenorrhea, irregular bleeding, and rectal discharge. Ultrasonographic examination revealed a cystic mass of 10 ⫻ 12 cm arising from the left adnexal region, filling the pelvis and extending to the umbilical region. The right ovary was also enlarged (4 ⫻ 5 cm). The uterus was bulky with heterogeneous echotexture. A third elective laparotomy (1990) was performed after 1 year for removal of the cyst. In spite of the adhesions, the cyst could be removed, but its origin could not be identified. The histologic report of the cyst wall showed spindle-shaped cells dispersed haphazardly and infiltrated by round cells. Colonoscopy was performed for persistent rectal discharge. A central depression with a tiny opening on the anterior rectal wall was seen. Histopathologic examination of the rectal biopsy specimen revealed an increase in lymphocytic mononuclear cells and neutrophil infiltration in the lamina propria. After 1 year she had another attack of acute abdominal pain. Ultrasonographic examination revealed bilateral chocolate cyst with pelvic collection and rectal fistula. The pelvic collection was drained partly through posterior colpotomy and partly through rectal sinus under general anesthesia. For 3 years she made frequent visits to gynecologists, and each time ultrasonography showed bilateral chocolate cyst with features of extensive pelvic adhesions. She received progesterones and danazol intermittently. Fertility and Sterility姞
A fourth emergency laparotomy (1997) was performed for acute abdominal pain. Partial resection of the cyst was performed, and danazol was prescribed for another 6 months. She finally was referred to our clinic. On examination, her general condition was fair with moderate pallor. Vaginal examination revealed a uterus that was fixed to surrounding structures and could not be felt as a separate structure. Lumps were felt in both the fornices. Ultrasonography revealed a bulky uterus with an irregular outline. Both ovaries showed chocolate cysts (5 ⫻ 5 cm and 3.5 ⫻ 2.5 cm) that were adherent to the enlarged uterus. Gonadotropin-releasing hormone-agonist for four cycles was advised. This therapy provided considerable symptomatic relief and appreciable reduction in the size of the cysts, after which the patient expressed her desire to conceive. In vitro fertilization– embryo transfer was performed for five cycles during the period from 1998 to 2001. Biochemical pregnancies were achieved in three cycles. While she was being prepared for repeat IVF, she conceived spontaneously. The antenatal period was complicated and required intensive monitoring. She had unilateral massive swelling of the left lower limb, probably of lymphatic origin. Color Doppler was performed to exclude deep venous thrombosis. She further had urinary retention and incontinence, which was treated by continuous catheterization. Ultrasonography revealed mild dilatation of the left ureter and hydronephrosis. Rectal discharge and incontinence of stool persisted. Pregnancy-induced hypertension (borderline) and a mild rise in blood urea and creatinine levels were encountered. She had preterm onset of labor at 35 weeks. Emergency cesarean section had to be performed through an extraperitoneal approach. Because of dense adhesions the peritoneal cavity could not be opened. A healthy female baby weighing 1.8 kg was delivered, and the patient is now symptom free. DISCUSSION Deep pelvic endometriosis, which is often associated with peritoneal and ovarian lesions, may involve the uterosacral ligaments, the pouch of Douglas, the vagina, the rectum, and occasionally the bladder (5). The main area of the gastrointestinal tract affected by deep pelvic endometriosis is the rectosigmoid colon in 3%–37 % of cases (6). Few recent reports are found of cervical endometriosis; this may be attributed to the common belief that the cervix uteri is a rare localization for endometriosis (4). Cervical endometriosis presenting with a uterus-like mass originating from the cervix (7) and a polypoid mass in the cervix (8) have been reported earlier. Hysterectomy is generally the treatment of choice in such cases. An attempt of hysterectomy, however, was not a suitable solution in the present cases. The clinical appearance of cervical endometriosis cases may be misleading as in case 1. The patient presented to us 697.e3
with rectal and vaginal bleeding, which are rare symptoms of cervical endometriosis, and the clinical findings were suggestive more of stage IV carcinoma of the cervix. She had no traumatizing procedures to the cervix in the past. Histopathologic report confirmed endometriosis. In case 2, though speculative, diagnosis of endometriosis was corroborated by the patient’s response to GnRH-agonist therapy. Interestingly, in both these cases, the uterus was not enlarged abnormally by adenomyosis. The size of the uterus may have been reduced by GnRH-agonist, thereby enhancing its receptivity. Prolonged medical treatment may have helped to arrest the progression of the disease by restoring anatomic proximity leading to spontaneous pregnancy in both cases. Once the women were pregnant, availability of a large amount of endogenous progesterone could have been a key factor for continuation of pregnancy. In case 1, the patient had recurrence of symptoms after delivery, and surgical castration was found suitable. In case 2, the postdelivery symptom-free state may be attributed either due to prolonged suppression of the hypothalamic–
697.e4 Ganesh and Chakravarty
pituitary– ovarian axis or due to persistently elevated levels of progesterone during pregnancy. REFERENCES 1. Clement MD. Diseases of the peritoneum (including endometriosis). In: Kurman RJ, ed. Blaustein’s pathology of the female genital tract. Vol. 1. New York: Springer-Verlag, 2002:729 – 89. 2. Farquhar CM. Extracts from the “clinical evidence.” Endometriosis. Br Med J 2000;320:1449 –52. 3. Graham B, Mazier WP. Diagnosis and management of endometriosis of the colon and rectum. Dis Colon Rectum 1988;31:952– 6. 4. Ata B, Ates U, Usta T, Attar E. Cervical endometriosis, a case presenting with intractable spotting. MedGenMed 2005;7:64. 5. Koninckx PR, Oosterlynck D, D’Hooghe T, Meuleman C. Deeply infiltrating endometriosis is a disease whereas mild endometriosis could be considered a non-disease. Ann N Y Acad Sci 1994;734:333–341. 6. Roseau G, Dumontier I, Palazzo L, Chapron C, Dousset B, Chaussade S, et al. Rectosigmoid endometriosis: endoscopic ultrasound features and clinical implications. Endoscopy 2000;32:525–30. 7. Fukunaga M. Uterus-like mass in the uterine cervix: superficial cervical endometriosis with florid smooth muscle metaplasia? Virchows Arch 2001;438:302–5. 8. Kano H, Kanda H. Cervical endometriosis presented as a polypoid mass of portio cervix uteri. J Obstet Gynaecol 2003;23:81–92.
Endometriosis with spontaneous pregnancy
Vol. 87, No. 3, March 2007
School of Medical Science and Technology, Indian Institute of Technology, Kharagpur, and Medicine, Salt Lake, Kolkata, India
b
Institute of Reproductive
Objective: To report two cases of advanced pelvic endometriosis, both with deep rectal involvement and one with extensive cervical involvement appearing like a carcinoma, where both patients achieved spontaneous pregnancy and delivered viable babies. Design: Case report. Setting: Institute of Reproductive Medicine. Patient(s): Two nulliparous patients, one with extensive cervical and rectal and other with deep rectal endometriosis and primary infertility. Intervention(s): Medical management. Main Outcome Measure(s): Description and treatment of two patients with advanced pelvic endometriosis and pregnancy. Result(s): Delivery of viable babies in both cases. Conclusion: Prolonged medical treatment may have helped to arrest the progression of the disease by restoring anatomic proximity leading to spontaneous pregnancy in both cases. (Fertil Steril威 2007;87:697.e1– 4. ©2007 by American Society for Reproductive Medicine.) Key Words: Cervical endometriosis, advanced pelvic endometriosis, polypoidal mass, pregnancy
Endometriosis is a condition in which ectopic endometrial glands and stroma are found outside the uterus (1). Most endometrial deposits are found in the pelvis (ovaries, peritoneum, uterosacral ligaments, pouch of Douglas, and rectovaginal septum). Extrapelvic deposits, including those in the umbilicus and diaphragm, are rare (2).
involvement appearing like a carcinoma, where both patients achieved spontaneous pregnancy and delivered viable babies, are reported. Approval was obtained for both the cases from the institutional review board before surgical intervention.
The incidence of endometriosis is reported as 4%–17% of all women during their reproductive age. Endometriosis of the colon and rectum, though rare, comprises 3%–34% of these endometriosis cases (3). A colposcopic examination series reports the incidence of cervical endometriosis to be 0.11%–2.4% (4).
CASE 1 A 28-year-old nulliparous woman, married for 6 years, was seen at our clinic with rectal bleeding and persistent mucous discharge. She had regular menstrual cycles initially after attaining menarche at the age of 13 years; 7 years later she started to have irregular cycles. Her chief complaint of rectal bleeding was of 2 years duration. She was treated earlier with progesterone but was not relieved of her symptoms.
The most commonly reported symptoms of endometriosis are dysmenorrhea (60%– 80%), pelvic pain (30%–50%), and infertility (30%– 40%). Diagnosis is based on clinical and histologic findings. The conservative medical line of treatment aims at the arrest of growth and activity of ectopic endometrial implants. The second line of management would be surgical resection of as much of the endometriotic tissue as possible. A combination of the two therapies may also be used, if necessary. Assisted reproductive techniques may be used in patients with infertility. Two rare cases of advanced pelvic endometriosis, both with deep rectal involvement and one with extensive cervical Received for publication February 18, 2006; revised and accepted June 3, 2006. Reprint requests: Baidyanath Chakravarty, M.D., Institute of Reproductive Medicine, HB36/A/3, Sector III, Salt Lake, Kolkata, India 700106 (FAX: 91-33-2337-6290; E-mail: [email protected], [email protected]).
0015-0282/07/$32.00 doi:10.1016/j.fertnstert.2006.06.061
Vaginal examination revealed firm consistency of the cervix and an indurated posterior lip, extending to the upper third of the vaginal vault, more on the posterior vaginal wall. The cervix appeared to be fixed posteriorly with the anterior rectal wall. The uterus was bulky with restricted mobility. On speculum examination, a fungating growth (3 ⫻ 2.5 cm) with polypoidal projection involving the posterior lip of the cervix was seen (Fig. 1). On speculum insertion, bleeding from the lesion was noticed. On rectal examination, parametrium was found to be indurated extending up to the lateral pelvic wall on both sides. Rectal mucosa was indurated, ulcerated, and fixed to the growth. The tip of the examining finger was stained with blood. On the basis of these clinical findings, stage IV carcinoma of the cervix was diagnosed provisionally.
Fertility and Sterility姞 Vol. 87, No. 3, March 2007 697.e1 Copyright ©2007 American Society for Reproductive Medicine, Published by Elsevier Inc.
FIGURE 1 Speculum examination showing a fungating growth (3 ⫻ 2.5 cm) with polypoidal projection involving posterior lip of cervix in case 1.
but periodic episodes of heavy bleeding continued. Surgical castration by bilateral oophorectomy was considered because of inadequate response and inability of the patient to afford prolonged GnRH-agonist therapy. Because of severe rectal involvement, hysterectomy was not attempted as it would involve a posterior exenteration type of surgery with permanent colostomy. On laparotomy, the pelvis was found to be frozen. The uterus, tubes, and ovaries were not visualized on either side. With further dissection, a part of the anterior wall of the uterus and both adnexa could be identified. The left ovary had a small chocolate cyst, and the right ovary was normal. Ovaries on both sides and part of the body of the uterus (at the level of the round ligament) that could be visualized were removed. After surgery, the wound healed with secondary intention and without major complications. Subsequently her hemoglobin level improved, and no further vaginal or rectal bleeding was reported. CASE 2 A 29-year-old nulliparous woman, married for 5 years, was referred to our institute as a case of advanced endometriosis
Ganesh. Endometriosis with spontaneous pregnancy. Fertil Steril 2007.
Hemoglobin was found to be 8.4 gm/dL. Ultrasonographic examination of the lower abdomen and pelvis revealed a bulky uterus with an ill-defined space-occupying lesion involving the cervix and extending into the vagina. Ovaries on both sides were normal.
FIGURE 2 Polypoidal growth removed in case 1 and examined histopathologically. Findings showing cervical squamous epithelium at top and stroma below with several endometrial glands.
Initially, hematinics were given; examination under anesthesia confirmed the speculum findings. The polypoidal growth was removed with use of sponge forceps and sent for histopathologic examination, which confirmed endometriosis (Fig. 2). The patient was given danazol for 5 months, but bleeding could not be controlled adequately. The polypoidal growth recurred and was removed vaginally. Subsequently, danazol was combined with GnRH-agonist, after which bleeding stopped. Both drugs were withdrawn after 4 months. She had a normal cycle after 1 month, following which she became amenorrheic. Urine pregnancy test was positive. She did not attend our antenatal clinic regularly, as advised. She had an uneventful antenatal period and later came to our clinic at 32 weeks with preterm onset of labor. She delivered normally a live female baby weighing 2.2 kg, without any cervical tear or postpartum hemorrhage. Six months after delivery, she reported with continuous rectal and vaginal bleeding. Her hemoglobin was 7.8 gm/dL, and recurrence of similar polypoidal growth with dense rectal involvement was observed. Gonadotropin-releasing hormone-agonist and hematinics were given for 9 months, 697.e2 Ganesh and Chakravarty
Ganesh. Endometriosis with spontaneous pregnancy. Fertil Steril 2007.
Endometriosis with spontaneous pregnancy
Vol. 87, No. 3, March 2007
with pelvirectal sinus. She had severe pain in the lower abdomen, irregular bleeding from the vagina, rectal bleeding and mucous discharge, and primary infertility of 5 years’ duration. Throughout her menstrual history, after menarche onset at the age of 12 years (1978), she had had dysmenorrhea and intermenstrual bleeding. Her medical and surgical history is significant and is briefly reviewed. She had recurrent episodes of acute abdomen for which laparotomy was performed on four occasions between 1978 and 1997. The indications and findings of laparotomy, histologic examination reports, line of management after the surgeries, and other relevant events are summarized chronologically. The first laparotomy (1978) was performed 6 months after menarche for acute pain in the lower abdomen. The pelvic cavity was found to be filled with chocolate-colored fluid mixed with pus. Her left ovary was enlarged and adherent to the pouch of Douglas. Her left ovary was partially resected and diagnosed as an infected chocolate cyst of the left ovary. The histologic report was not available. Later for 6 years she had had intermittently severe dysmenorrhea and rectal discharge for which she was treated with progesterones and danazol periodically for temporary relief. A second laparotomy (1989) was performed for acute abdomen as an emergency. On laparotomy, the abdomen and pelvic organs were found to be plastered. The abdomen was closed without any interference. Antitubercular drugs were started empirically; after 4 months jaundice developed, and the antitubercular drugs were stopped temporarily. After treatment of jaundice, antitubercular drugs were continued. The patient, however, continued to have dysmenorrhea, irregular bleeding, and rectal discharge. Ultrasonographic examination revealed a cystic mass of 10 ⫻ 12 cm arising from the left adnexal region, filling the pelvis and extending to the umbilical region. The right ovary was also enlarged (4 ⫻ 5 cm). The uterus was bulky with heterogeneous echotexture. A third elective laparotomy (1990) was performed after 1 year for removal of the cyst. In spite of the adhesions, the cyst could be removed, but its origin could not be identified. The histologic report of the cyst wall showed spindle-shaped cells dispersed haphazardly and infiltrated by round cells. Colonoscopy was performed for persistent rectal discharge. A central depression with a tiny opening on the anterior rectal wall was seen. Histopathologic examination of the rectal biopsy specimen revealed an increase in lymphocytic mononuclear cells and neutrophil infiltration in the lamina propria. After 1 year she had another attack of acute abdominal pain. Ultrasonographic examination revealed bilateral chocolate cyst with pelvic collection and rectal fistula. The pelvic collection was drained partly through posterior colpotomy and partly through rectal sinus under general anesthesia. For 3 years she made frequent visits to gynecologists, and each time ultrasonography showed bilateral chocolate cyst with features of extensive pelvic adhesions. She received progesterones and danazol intermittently. Fertility and Sterility姞
A fourth emergency laparotomy (1997) was performed for acute abdominal pain. Partial resection of the cyst was performed, and danazol was prescribed for another 6 months. She finally was referred to our clinic. On examination, her general condition was fair with moderate pallor. Vaginal examination revealed a uterus that was fixed to surrounding structures and could not be felt as a separate structure. Lumps were felt in both the fornices. Ultrasonography revealed a bulky uterus with an irregular outline. Both ovaries showed chocolate cysts (5 ⫻ 5 cm and 3.5 ⫻ 2.5 cm) that were adherent to the enlarged uterus. Gonadotropin-releasing hormone-agonist for four cycles was advised. This therapy provided considerable symptomatic relief and appreciable reduction in the size of the cysts, after which the patient expressed her desire to conceive. In vitro fertilization– embryo transfer was performed for five cycles during the period from 1998 to 2001. Biochemical pregnancies were achieved in three cycles. While she was being prepared for repeat IVF, she conceived spontaneously. The antenatal period was complicated and required intensive monitoring. She had unilateral massive swelling of the left lower limb, probably of lymphatic origin. Color Doppler was performed to exclude deep venous thrombosis. She further had urinary retention and incontinence, which was treated by continuous catheterization. Ultrasonography revealed mild dilatation of the left ureter and hydronephrosis. Rectal discharge and incontinence of stool persisted. Pregnancy-induced hypertension (borderline) and a mild rise in blood urea and creatinine levels were encountered. She had preterm onset of labor at 35 weeks. Emergency cesarean section had to be performed through an extraperitoneal approach. Because of dense adhesions the peritoneal cavity could not be opened. A healthy female baby weighing 1.8 kg was delivered, and the patient is now symptom free. DISCUSSION Deep pelvic endometriosis, which is often associated with peritoneal and ovarian lesions, may involve the uterosacral ligaments, the pouch of Douglas, the vagina, the rectum, and occasionally the bladder (5). The main area of the gastrointestinal tract affected by deep pelvic endometriosis is the rectosigmoid colon in 3%–37 % of cases (6). Few recent reports are found of cervical endometriosis; this may be attributed to the common belief that the cervix uteri is a rare localization for endometriosis (4). Cervical endometriosis presenting with a uterus-like mass originating from the cervix (7) and a polypoid mass in the cervix (8) have been reported earlier. Hysterectomy is generally the treatment of choice in such cases. An attempt of hysterectomy, however, was not a suitable solution in the present cases. The clinical appearance of cervical endometriosis cases may be misleading as in case 1. The patient presented to us 697.e3
with rectal and vaginal bleeding, which are rare symptoms of cervical endometriosis, and the clinical findings were suggestive more of stage IV carcinoma of the cervix. She had no traumatizing procedures to the cervix in the past. Histopathologic report confirmed endometriosis. In case 2, though speculative, diagnosis of endometriosis was corroborated by the patient’s response to GnRH-agonist therapy. Interestingly, in both these cases, the uterus was not enlarged abnormally by adenomyosis. The size of the uterus may have been reduced by GnRH-agonist, thereby enhancing its receptivity. Prolonged medical treatment may have helped to arrest the progression of the disease by restoring anatomic proximity leading to spontaneous pregnancy in both cases. Once the women were pregnant, availability of a large amount of endogenous progesterone could have been a key factor for continuation of pregnancy. In case 1, the patient had recurrence of symptoms after delivery, and surgical castration was found suitable. In case 2, the postdelivery symptom-free state may be attributed either due to prolonged suppression of the hypothalamic–
697.e4 Ganesh and Chakravarty
pituitary– ovarian axis or due to persistently elevated levels of progesterone during pregnancy. REFERENCES 1. Clement MD. Diseases of the peritoneum (including endometriosis). In: Kurman RJ, ed. Blaustein’s pathology of the female genital tract. Vol. 1. New York: Springer-Verlag, 2002:729 – 89. 2. Farquhar CM. Extracts from the “clinical evidence.” Endometriosis. Br Med J 2000;320:1449 –52. 3. Graham B, Mazier WP. Diagnosis and management of endometriosis of the colon and rectum. Dis Colon Rectum 1988;31:952– 6. 4. Ata B, Ates U, Usta T, Attar E. Cervical endometriosis, a case presenting with intractable spotting. MedGenMed 2005;7:64. 5. Koninckx PR, Oosterlynck D, D’Hooghe T, Meuleman C. Deeply infiltrating endometriosis is a disease whereas mild endometriosis could be considered a non-disease. Ann N Y Acad Sci 1994;734:333–341. 6. Roseau G, Dumontier I, Palazzo L, Chapron C, Dousset B, Chaussade S, et al. Rectosigmoid endometriosis: endoscopic ultrasound features and clinical implications. Endoscopy 2000;32:525–30. 7. Fukunaga M. Uterus-like mass in the uterine cervix: superficial cervical endometriosis with florid smooth muscle metaplasia? Virchows Arch 2001;438:302–5. 8. Kano H, Kanda H. Cervical endometriosis presented as a polypoid mass of portio cervix uteri. J Obstet Gynaecol 2003;23:81–92.
Endometriosis with spontaneous pregnancy
Vol. 87, No. 3, March 2007