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Tuboovarian autoamputation and infertility*
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FERTILITY AND STERILITY Copyright 0 1984 The American Fertility Society
Vol. 42, No.6, December 1984 Printed in U.SA.
Tuboovarian auto amputation and infertility*
Yoram Beyth, M,D, t Elchanan Bar-On, M,D,:j: Department of Obstetrics and Gynecology, Hadassah University Hospital, Jerusalem, Israel
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Torsion of uterine adnexa may be the cause of acute abdomen in children as well as in adult females. However, silent torsion of ovary and tube followed by autoamputation of these organs is a distinct, although rare, gynecologic problem. The first case report of an asymptomatic separation of an adnexa was described by Sebastian et al. 1 in 1973, and since then there have been three additional reports 2- 4 in the literature. We would like to describe two women in whom the absence of ovary and tube were incidentally diagnosed during a fertility workup. In one case there was a history of recurrent attacks of abdominal pain during the patient's teenage years, and the second patient had an unremarkable medical history. The importance of diagnosing such a condition during childhood and adolescence is emphasized, and the consequences regarding future fertility are discussed. CASE REPORTS CASE 1
A 23-year-old woman (gravida 3, para 0, abortion 2) was admitted for laparoscopy because of
Received February 28, 1984; revised and accepted June 26, 1984. *Supported by a donation from Dr. E. Gruenewald, Poole, England. tReprint requests: Yoram Beyth, M.D., Department of Obstetrics and Gynecology, Hadassah University Hospital, P.O.B. 12 000, il-91 120 Jerusalem, Israel. . :j:Present address: Department of Obstetrics and Gynecology, Shaare-Zedek Medical Center, Jerusalem, Israel.
932
Beytb and Bar-On Comrffiinications-.in-brief
extrauterine pregnancy. The only pertinent detail in her medical history was a laparotomy at the age of 16 because of suspected acute appendicitis. A normal, uninflamed appendix was removed. At that time, the right ovary was described as being slightly enlarged, having a smooth surface, while the left ovary and both tubes were normal. Ten days before admission the patient suffered from vaginal bleeding. Clinically, the diagnosis of incomplete abortion was made, and the patient underwent curettage. Products of conception were not recovered during this procedure; yet repeat serum ~-subunit human chorionic gonadotropin levels were 400 mIU/ml. In view of these results and because of lower abdominal pain with continuous vaginal bleeding, the diagnosis of ectopic pregnancy was highly suspected. Physical examination revealed tenderness in the left lower abdomen, moderate vaginal bleeding, a normalsized uterus, and a very tender mass in the pouch of Douglas, measuring 4 x 5 cm. At laparoscopy, blood clots were seen in the peritoneal cavity, and therefore laparotomy was performed. Upon opening the abdomen, a small uterus was revealed, with adhesions of bowel loops to the sacrouterine ligaments. Surprisingly, on the right side only, the proximal part of the tubal isthmus was identified, being covered by peritoneal adhesions. The tube seemed to have been amputated. Neither the rest of the tube nor the ovary was seen or palpated. On the left side an ovarian dermoid cyst 6 em in diameter was found and enucleated. The ampulla of the left fallopian tube was congested and swollen, and a few blood clots were removed by a "milking" maneuver. Periadnexal adhesions Fertility and Sterility
pected following a rectal examination, but this diagnosis was not confirmed. Hysterosalpingogram, which was performed later as part of the patient's fertility workup, revealed mild intrauterine adhesions, no filling of the left tube, and suspected distal occlusion of the right tube. At laparoscopy, a normal uterus was noted. The left ovary was not seen, and only a short stump of the left tubal isthmus could be identified. The right ovary and tube were covered by adhesions. Dye, which was injected into the uterus, passed through the right tube, but remained pooled in between the peritubal adhesions. After this procedure the patient underwent reconstructive surgery. The absence of the left adnexa was confirmed. All periadnexal adhesions surrounding the right adnexa were removed. A normal-looking tube including fimbria and ovary were then revealed (Fig. 1). DISCUSSION
Figure 1 Pelvic appearance following left tuboovarian autoamputation. (A), Notice the adhesion between uterus and bowel and the right periadnexal adhesions (right Teflon rod demonstrates right isthmus). (B), Following resection of adhesions. Notice the proximal part of the amputated left tube (indicated by the Teflon rod) and the normal-looking right ovary and tube, including fimbriae.
were removed. In the pouch of Douglas 40 ml of blood clots were found, among them the products of conception. CASE 2
A 27-year-old woman with a 3-year history of primary infertility was admitted for diagnostic laparoscopy. Her pertinent medical history revealed several episodes of abdominal pain. At the age of 12 years, she had recurrent attacks of colicky abdominal pains. She was admitted to an emergency room and to the pediatric department three times during 1 year; however, the cause of her pain was not found despite extensive physical and laboratory investigations. Barium enema and intravenous pyelogram revealed a normal lower gastrointestinal tract and a normal urinary tract. At one time, a left ovarian cyst was susVol. 42, No.6, December 1984
Congenital anomalies of Mullerian ducts result in abnormalities of the uterus, cervix, and upper vagina; whereas the ovaries and fallopian tubes usually develop in a normal manner. A total absence of adnexa (tube and ovary) in the presence of a normal uterus, cervix, and vagina is usually considered to be acquired, because these two organs originate from two different embryonal elements. 3 Tuboovarian autoamputation may be totally asymptomatic, as described in our first patient. Some of the patients in whom the absence of adnexa was incidentally diagnosed did not have any medical history of an abdominal catastrophe. 4 In these patients neither their parents nor they themselves could recall any episodes of even mild lower abdominal pains. However, there are patients in whom a history of intermittent attacks of lower abdominal pain during childhood or later life can be obtained. These are the patients, similar to our second case, in whom the adnexa could eventually have been saved. It is of great importance that surgeons and pediatricians be aware of a possible torsion of adnexa in young girls and teenagers who present themselves with acute attacks oflower abdominal pain. This diagnosis may be difficult to establish in young girls in whom vaginal examinations are not feasible, and one has to rely on rectal examination. It is especially difficult in cases in which the torsion is of normal-sized adnexa and not secondary to an ovarian cyst. During the last decade, Beyth and Bar-On Communications-in-brief
933
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ultrasound has become a routine diagnostic tool which was added to the armamentarium of almost all departments. We would like to suggest that a pelvic screening ultrasound be used as a routine test by physicians who are presented with a similar diagnostic dilemma, in young girls in whom there exists an uncertainty as to the presence of an adnexal mass. It is interesting to mention that both our patients came to us with fertility problems. The absence of adnexa was discovered in one patient during laparoscopy, which was part of her infertility workup. Multiple pelvic peritoneal, periovarian, and peritubal adhesions surrounding the remaining adnexa were noted. In the second patient, laparotomy for tubal pregnancy was performed, at which time the absence of the contralateral adnexa was revealed. It may well be speculated that the inflammatory process which developed secondary to the amputation of the adnexa in this patient caused the tubal pregnancy, either through direct damage to the tubal mucosa or by impairing the tubal muscle function, because of the peritubal adhesions. Moreover, it is almost certain that the pelvic peritoneal adhesions and the periadnexal adhesions surrounding the single remaining ovary in both patients were
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the result of the pelvic inflammatory process which developed at the time of the adnexal torsion and amputation. These adhesions are an additional contribution to infertility. SUMMARY
Asymptomatic or undiagnosed tuboovarian autoamputation in teenagers may have its impact on fertility later in life. Two cases are presented with infertility following unilateral adnexal autoamputation. The importance of awareness for possible subtorsion or torsion of adnexa in childhood and adolescence and its impact on fertility are discussed. REFERENCES 1. Sebastian JA, Baher RL, Cordray D: Asymptomatic in-
farction and separation of ovary and distal uterine tube. Obstet Gynecol 41:531, 1973 2. Georgy FM, Viechnicki MB: Absence of an ovary and uterine tube. Obstet Gynecol 44:441, 1974 3. Nissen ED, Kent DR, Nissen SE, Feldman BM: Unilateral tuboovarian autoamputation. J Reprod Med 19:151, 1977 4. Bates GW, Abide JK: Bilateral autoamputation of the fallopian tubes. Fertil Steril 38:253, 1982
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934
Beytb and Bar-On Communications-in-brief
Fertility and Sterility
....
i
FERTILITY AND STERILITY Copyright 0 1984 The American Fertility Society
Vol. 42, No.6, December 1984 Printed in U.SA.
Tuboovarian auto amputation and infertility*
Yoram Beyth, M,D, t Elchanan Bar-On, M,D,:j: Department of Obstetrics and Gynecology, Hadassah University Hospital, Jerusalem, Israel
rl
I,
I,
I
I.'j . ~!
1.1
i l~
Torsion of uterine adnexa may be the cause of acute abdomen in children as well as in adult females. However, silent torsion of ovary and tube followed by autoamputation of these organs is a distinct, although rare, gynecologic problem. The first case report of an asymptomatic separation of an adnexa was described by Sebastian et al. 1 in 1973, and since then there have been three additional reports 2- 4 in the literature. We would like to describe two women in whom the absence of ovary and tube were incidentally diagnosed during a fertility workup. In one case there was a history of recurrent attacks of abdominal pain during the patient's teenage years, and the second patient had an unremarkable medical history. The importance of diagnosing such a condition during childhood and adolescence is emphasized, and the consequences regarding future fertility are discussed. CASE REPORTS CASE 1
A 23-year-old woman (gravida 3, para 0, abortion 2) was admitted for laparoscopy because of
Received February 28, 1984; revised and accepted June 26, 1984. *Supported by a donation from Dr. E. Gruenewald, Poole, England. tReprint requests: Yoram Beyth, M.D., Department of Obstetrics and Gynecology, Hadassah University Hospital, P.O.B. 12 000, il-91 120 Jerusalem, Israel. . :j:Present address: Department of Obstetrics and Gynecology, Shaare-Zedek Medical Center, Jerusalem, Israel.
932
Beytb and Bar-On Comrffiinications-.in-brief
extrauterine pregnancy. The only pertinent detail in her medical history was a laparotomy at the age of 16 because of suspected acute appendicitis. A normal, uninflamed appendix was removed. At that time, the right ovary was described as being slightly enlarged, having a smooth surface, while the left ovary and both tubes were normal. Ten days before admission the patient suffered from vaginal bleeding. Clinically, the diagnosis of incomplete abortion was made, and the patient underwent curettage. Products of conception were not recovered during this procedure; yet repeat serum ~-subunit human chorionic gonadotropin levels were 400 mIU/ml. In view of these results and because of lower abdominal pain with continuous vaginal bleeding, the diagnosis of ectopic pregnancy was highly suspected. Physical examination revealed tenderness in the left lower abdomen, moderate vaginal bleeding, a normalsized uterus, and a very tender mass in the pouch of Douglas, measuring 4 x 5 cm. At laparoscopy, blood clots were seen in the peritoneal cavity, and therefore laparotomy was performed. Upon opening the abdomen, a small uterus was revealed, with adhesions of bowel loops to the sacrouterine ligaments. Surprisingly, on the right side only, the proximal part of the tubal isthmus was identified, being covered by peritoneal adhesions. The tube seemed to have been amputated. Neither the rest of the tube nor the ovary was seen or palpated. On the left side an ovarian dermoid cyst 6 em in diameter was found and enucleated. The ampulla of the left fallopian tube was congested and swollen, and a few blood clots were removed by a "milking" maneuver. Periadnexal adhesions Fertility and Sterility
pected following a rectal examination, but this diagnosis was not confirmed. Hysterosalpingogram, which was performed later as part of the patient's fertility workup, revealed mild intrauterine adhesions, no filling of the left tube, and suspected distal occlusion of the right tube. At laparoscopy, a normal uterus was noted. The left ovary was not seen, and only a short stump of the left tubal isthmus could be identified. The right ovary and tube were covered by adhesions. Dye, which was injected into the uterus, passed through the right tube, but remained pooled in between the peritubal adhesions. After this procedure the patient underwent reconstructive surgery. The absence of the left adnexa was confirmed. All periadnexal adhesions surrounding the right adnexa were removed. A normal-looking tube including fimbria and ovary were then revealed (Fig. 1). DISCUSSION
Figure 1 Pelvic appearance following left tuboovarian autoamputation. (A), Notice the adhesion between uterus and bowel and the right periadnexal adhesions (right Teflon rod demonstrates right isthmus). (B), Following resection of adhesions. Notice the proximal part of the amputated left tube (indicated by the Teflon rod) and the normal-looking right ovary and tube, including fimbriae.
were removed. In the pouch of Douglas 40 ml of blood clots were found, among them the products of conception. CASE 2
A 27-year-old woman with a 3-year history of primary infertility was admitted for diagnostic laparoscopy. Her pertinent medical history revealed several episodes of abdominal pain. At the age of 12 years, she had recurrent attacks of colicky abdominal pains. She was admitted to an emergency room and to the pediatric department three times during 1 year; however, the cause of her pain was not found despite extensive physical and laboratory investigations. Barium enema and intravenous pyelogram revealed a normal lower gastrointestinal tract and a normal urinary tract. At one time, a left ovarian cyst was susVol. 42, No.6, December 1984
Congenital anomalies of Mullerian ducts result in abnormalities of the uterus, cervix, and upper vagina; whereas the ovaries and fallopian tubes usually develop in a normal manner. A total absence of adnexa (tube and ovary) in the presence of a normal uterus, cervix, and vagina is usually considered to be acquired, because these two organs originate from two different embryonal elements. 3 Tuboovarian autoamputation may be totally asymptomatic, as described in our first patient. Some of the patients in whom the absence of adnexa was incidentally diagnosed did not have any medical history of an abdominal catastrophe. 4 In these patients neither their parents nor they themselves could recall any episodes of even mild lower abdominal pains. However, there are patients in whom a history of intermittent attacks of lower abdominal pain during childhood or later life can be obtained. These are the patients, similar to our second case, in whom the adnexa could eventually have been saved. It is of great importance that surgeons and pediatricians be aware of a possible torsion of adnexa in young girls and teenagers who present themselves with acute attacks oflower abdominal pain. This diagnosis may be difficult to establish in young girls in whom vaginal examinations are not feasible, and one has to rely on rectal examination. It is especially difficult in cases in which the torsion is of normal-sized adnexa and not secondary to an ovarian cyst. During the last decade, Beyth and Bar-On Communications-in-brief
933
.----------------~-~--~--------
i:
:1
-
---- ---------
ultrasound has become a routine diagnostic tool which was added to the armamentarium of almost all departments. We would like to suggest that a pelvic screening ultrasound be used as a routine test by physicians who are presented with a similar diagnostic dilemma, in young girls in whom there exists an uncertainty as to the presence of an adnexal mass. It is interesting to mention that both our patients came to us with fertility problems. The absence of adnexa was discovered in one patient during laparoscopy, which was part of her infertility workup. Multiple pelvic peritoneal, periovarian, and peritubal adhesions surrounding the remaining adnexa were noted. In the second patient, laparotomy for tubal pregnancy was performed, at which time the absence of the contralateral adnexa was revealed. It may well be speculated that the inflammatory process which developed secondary to the amputation of the adnexa in this patient caused the tubal pregnancy, either through direct damage to the tubal mucosa or by impairing the tubal muscle function, because of the peritubal adhesions. Moreover, it is almost certain that the pelvic peritoneal adhesions and the periadnexal adhesions surrounding the single remaining ovary in both patients were
-----
the result of the pelvic inflammatory process which developed at the time of the adnexal torsion and amputation. These adhesions are an additional contribution to infertility. SUMMARY
Asymptomatic or undiagnosed tuboovarian autoamputation in teenagers may have its impact on fertility later in life. Two cases are presented with infertility following unilateral adnexal autoamputation. The importance of awareness for possible subtorsion or torsion of adnexa in childhood and adolescence and its impact on fertility are discussed. REFERENCES 1. Sebastian JA, Baher RL, Cordray D: Asymptomatic in-
farction and separation of ovary and distal uterine tube. Obstet Gynecol 41:531, 1973 2. Georgy FM, Viechnicki MB: Absence of an ovary and uterine tube. Obstet Gynecol 44:441, 1974 3. Nissen ED, Kent DR, Nissen SE, Feldman BM: Unilateral tuboovarian autoamputation. J Reprod Med 19:151, 1977 4. Bates GW, Abide JK: Bilateral autoamputation of the fallopian tubes. Fertil Steril 38:253, 1982
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934
Beytb and Bar-On Communications-in-brief
Fertility and Sterility
....