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epispadias patient
FERTILITY AND STERILITY Copyright
©
Vol. 60, No.4, October 1993
Printed on acid-free paper in U. S. A.
1993 The American Fertility Society
Fertility and the use of assisted reproductive techniques in the adult male exstrophy/epispadias patient
Martin D. Bastuba, M.D.* Michael M. Alper, M.D. t Robert D. Oates, M.D.*:j: Boston University Medical Center and Beth Israel Hospital, and Boston Fertility and Gynecology Associates/Boston IVF, Boston, Massachusetts
Exstrophy of the bladder with epispadias and separation of the pubes is an uncommon anomaly occurring in 1:30,000 live births. Surgical challenges have historically involved closure of the bladder and the achievement of urinary continence. The preservation and/or restoration of erectile and ejaculatory function are areas of more recent interest and refinement. With improvement in the lifestyle and general health of these patients, fertility issues have now assumed a level of increasing importance. Though fertility potential in the female exstrophy patient is reported to be adequate (1), spontaneous pregnancy has been reported in only 18% to 22% of couples (2, 3) in whom the male partner was born with classical bladder exstrophy. Etiologic factors implicated in the subfertility of this population include cryptorchidism, erectile dysfunction secondary to dissection of the corpora off the ischiopubic rami, retrograde ejaculation resulting from an incompetent bladder neck, recurrent epididymo-orchitis with subsequent epididymalocclusion and testicular atrophy, vasal injury at the time of herniorrhaphy, occlusive prostatic urethral stricture, dysfunctional urethral transport of semen secondary to deficient bulbospongiosus
Received December 21, 1992; revised and accepted June 14, 1993. * Department of Urology, Boston University Medical Center. t Department of Obstetrics and Gynecology, Beth Israel Hospital, and Boston Fertility and Gynecology Associates/Boston IVF. :j: Reprint requests: Robert D. Oates, M.D., DOB 606, 720 Harrison Avenue, Boston, Massachusetts 02118. Vol. 60, No.4, October 1993
muscle action, and iatrogenic injury to the seminal tract during reconstructive procedures. We report on three men, two born with classical exstrophy of the bladder and another with epispadias alone. Each couple presented after inability to initiate a pregnancy of a year or more duration. Each spouse's infertility evaluation was within normal limits. Innovative strategies, including assisted reproductive techniques in two, led to pregnancies in all cases. A review of relevant literature is presented with discussion. CASE REPORTS
Case 1 A 32-year-old male born with bladder exstrophy and bilateral inguinal hernias presented for evaluation of infertility. Shortly after birth, bladder closure, epispadias repair, and bilateral herniorrhaphy were performed without complication. Bladder neck reconstruction was carried out in early childhood. Because of continued incontinence, he underwent bilateral ureterosigmoidostomy at age 5, leaving the bladder and prostate in situ. Penile lengthening was accomplished at age 12. He married at age 30 and presented shortly thereafter. Erections were adequate for successful intravaginal intercourse. Although the patient sensed orgasm, no immediate antegrade flow of seminal fluid was present. Beginning approximately 0.5 hour after intercourse, the patient would note the slow emission of a scant amount of semen from the urethral meatus. The testicular exam was normal and the urethra was easily catheterizable. Bastuba et al.
Communications-in-brief
733
The patient was instructed to compress his urethra, from proximal to distal, to express more of the seminal fluid remaining stagnant in his proximal urethra after orgasm. In this fashion, an increased amount of semen was obtained, revealing a volume of 2.5 cc, a sperm density of 34 X 106 /cc, and a progressive motility of 85%. Seminal fluid was collected during the periovulatory period in the manner described above, with the couple performing cervical insemination. On the eighth attempt, without augmentative medications, a pregnancy was achieved with a healthy female infant delivered at term.
Human tubal fluid
Hemitransection of Vas Deferens
Case 2
A 25-year-old male born with epispadias presented with a 2-year history of failure to initiate a pregnancy. He had his epispadias closure during infancy with bladder neck reconstruction at age 4. Although continent of urine at presentation, the patient experienced delayed and scant seminal emission. The antegrade milking technique reliably produced an increased volume of ejaculate, although still <1 cc. Because of the decreased seminal volume, it was believed that the addition of lUI would enhance their chances of conception. A pregnancy resulted after the second cycle of insemination coupled with ovulatory augmentation utilizing 50 mg of clomiphene citrate cycle days 5 to 9. Semen analysis at that time revealed a total count of 97 X 106 sperm with a progressive motility of 70%. A healthy female infant was delivered at term. Case 3
A 27-year-old male born with classic bladder exstrophy as well as bilateral cryptorchidism presented with infertility. The patient underwent early bladder closure and orchidopexy. A bilateral ureterosigmoidostomy was performed after multiple bladder neck reconstructions and urethroplasties failed to produce continence of urine. Penile lengthening was accomplished at age 16 and erectile function is presently adequate. The patient noted delayed and minimal ante grade emission (0.2 to 0.4 cc). Attempted urethral catheterization was unsuccessful, prompting retrograde urethrogram. The severe obstructing urethral stricture disease visualized was not amenable to surgical correction. Unreliable semen volume and delivery (0.2 to 0.6 cc), associated with widely fluctuating total sperm output (26 X 106 , 0, 3 X 106 , and 2 X 106 ) during unsuccessful lUI trials prompted microsurgical va734
Bastuba et al.
Communications-in-brief
Figure 1 After an anterior hemivasotomy, the exuding fluid is aspirated into prelubricated syringes with attached 22-gauge plastic tip. All fluid is expelled into appropriate media. Closure of the vas is accomplished with 10-0 mucosal and 9-0 seromuscular nylon sutures.
sal sperm aspiration coupled with GIFT. Direct aspiration assured retrieval of motile sperm before oocyte harvesting. Because of the intensive nature of the advanced reproductive techniques, we did not wish to risk that this patient's ejaculate would be inadequate for GIFT on that particular day. Knowing that he had the capability for adequate sperm production, a direct aspiration from the vas guaranteed sperm availability. A standard stimulation and oocyte-harvesting protocol was used. When the E2 was 2,141 pg/mL (conversion factor to SI unit, 3.671) and there were 10 follicles ~16 mm in diameter as visualized by transvaginal ultrasonography, 10,000 U hCG 1M was given. Thirty-two hours later, the sperm specimen was micro surgically aspirated from the straight portion of the scrotal vas deferens into 0.5 cc of medium (HEPES-buffered human tubal fluid; Irvine Scientific, Irvine, CA) (Fig. 1). Sperm processing was carried out using mini-Percoll (Pharmacia, Uppsala, Sweden) in conjunction with 3 mM pentoxiphylline and 3 mM 2 deoxyadenosine (Sigma Chemical Co., St. Louis, MO) as described in detail by Ord et al. (4). A total of36 X 106 sperm with a progressive motility of 40% were recovered. Eight oocytes were then laparoscopic ally retrieved, and four were transferred along with 200,000 motile sperm to the right fallopian tube. The remaining four oocytes were incubated in vitro with 200,000 motile sperm per oocyte. Two fertilized and are currently cryopreserved at the four-cell stage. A resultant twin pregnancy is ongoing. Fertility and Sterility
DISCUSSION
Advancements in the initial and secondary surgical treatment of exstrophy/epispadias have allowed fertility issues to take on increasing prominence. Spontaneous conception in the male exstrophy / epispadias population is still impaired, however. The restoration of normal erectile function is the first obstacle that must be overcome to enable the male to achieve satisfactory intravaginal intercourse. Even with improved operative technique, Mesrobian et al. (3) state that 39% oftheir postpubertal males still could not have successful intercourse. Therefore, spontaneous conception, at its maximum, could only occur in 61% of patients. Ejaculatory dysfunction is also a major cause of failure to conceive. In the exstrophy/epispadias complex, the urethra lacks circumferential bulbospongiosus and ischiocavernosus muscles, a situation not corrected by reconstructive surgery. Normally, rhythmic contraction of the periurethral musculature during ejaculation forcefully propels the seminal fluid in an antegrade direction. In its absence, the semen either remains in the proximal urethra or, occasionally, a small amount may dribble out the urethral meatus. Proper cervical deposition of semen during intercourse may not occur and, therefore, conception would be unlikely. As in case no. 3, multiple urethral surgical endeavors may lead to significant stricture disease, impeding further the antegrade flow of semen. In the diverted patient, severe strictures may not be relevant except in this context. Testicular function is believed to be intrinsic ally normal. Although many patients have "cryptorchidism," this may be more a result of inadequate scrotal development than a failure of descent due to an inherent testicular anomaly (5). At puberty, the scrotum will oftentimes enlarge while the gonads spontaneously "descend." It is unclear whether the high position of the testes during the prepubertal years will eventually have an impact on their spermatogenic capability. The reproductive ductal structures are embryologically and developmentally normal. However, epididymal obstruction may occur as a result of an episode of epididymitis whereas orchitis may lead to testicular atrophy. Review of the literature reveals only two studies that directly address semen quality and fertility. Lattimer et al. (2) report on nine patients (17 to 32 years old) and note that five were azoospermic, even in the retrograde specimen. Two patients were severely oligospermic (0.2 to 6 X 1Q6/CC ) and two were normospermic with excellent motility. These Vol. 60, No.4, October 1993
latter two patients, although with semen volumes of 0.5 to 0.6 cc, had both initiated pregnancies. It is unclear how many of this group were actively trying to conceive but it is highly unlikely that all were, given their reported ages. Therefore, the incidence of spontaneous pregnancy achievement is, at a minimum, 22%. Hanna and Williams (6) report on 16 exstrophy patients and 15 epispadias patients. In the exstrophy cohort, only 5 produced "normal" specimens and 11 produced specimens "incompatible with the probability of conception." Exact seminal parameters were not stated. It appeared that semen quality was better, overall, in those patients who did not have attempted bladder reconstruction but only excision of exposed bladder mucosa and urinary diversion. It is interesting to note that two patients who were detailed complained of slow, oozing ejaculation subsequent to orgasm. The epispadias cohort fared much better, with 14/15 ejaculating normally. Of 13 that produced semen specimens, 9 were considered "normal" and 4 were "substandard. " Woodhouse et al. (1) documented that of 29 patients who are married, 6 have initiated pregnancies for a minimal incidence of spontaneous conception of 21%. It is generally felt that the incidence of bladder exstrophy transmission is approximately 1 %, a 500-fold increase over the general population. Assisted reproductive techniques have improved the prognosis of infertility for a number of couples in whom female factors, male factors, or both have been preventing spontaneous conception. Cervical insemination is the least complicated, may be performed at home after appropriate instruction, and can overcome deficiencies in the normal deposition of an adequate volume of ejaculate to the cervical region. Intrauterine insemination is commonly used in a compensatory fashion when semen quality is poor and no remedial cause is apparent. Ovarian stimulation may be added, potentially increasing the probability of pregnancy. Our three cases illustrate that knowledge of both the pathophysiological mechanisms preventing spontaneous conception in the adult male exstrophy patient and the newer reproductive techniques, coupled with a little imagination, can lead to a long-awaited pregnancy. All three patients had normal emission/orgasm but noted only small amounts of dribbling seminal discharge either immediately after ejaculation or several hours subsequently. The first two had patent urethras but probable absence of normal periurethral musculature contraction and, therefore, no immediate antegrade Bastuba et al.
Communications-in-brief
735
ejaculate. Absence of retrograde ejaculate was verified by catheterization and irrigation ofthe bladder. Each was taught to cleanse the urethra, to ejaculate, and then to milk the urethra from deep in the perineum to the tip of the penis to obtain an adequate semen specimen (0.4 cc, 120 X 106 ,65% and 2.5 cc, 84 X 106 ,85%, respectively). The first couple conceived after 8 months of relatively simple home cervical insemination whereas the other became pregnant after the second cycle of intrauterine insemination. Both mothers delivered healthy females without incident. Because there was no way in which to obtain a better semen specimen in the third patient because of his severe urethral stricture disease, a direct microsurgical vasal sperm aspiration was used to allow us to extract adequate numbers of high quality sperm for use in combination with GIFT. As can be seen by the fact that there is an ongoing twin pregnancy and that two additional embryos were generated, the fertilizing potential of this patient's sperm was excellent. SUMMARY
These three cases exemplify, in increasing order of complexity, how the commonsense application of already successful techniques might be applied to
736
Bastuba et al.
Communications-in-brief
augment the chance of pregnancy in this group of patients. We believe this to be the first report specifically outlining measures that may assist this group in their goal of pregnancy achievement. To our knowledge, vasal sperm aspiration has never been used for this indication. Key Words: Bladder exstrophy, epispadias, microsurgical sperm aspiration. REFERENCES 1. Woodhouse CRJ, Ransley PG, Williams Dr. The patient
with exstrophy in adult life. Br J Urol 1983;55:632-5. 2. Lattimer JK, Macfarlane MT, Puchner PJ. Male exstrophy patients: a preliminary report on the reproductive capability. Trans Am Assoc Genitourin Surg 1979;70:42-4. 3. Mesrobian HGJ, Kelalis PP, Kramer SA. Long-term followup of cosmetic appearance and genital function in boys with exstrophy: review of 53 patients. J UroI1986;136:256-8. 4. Ord T, Marello E, Patrizio P, Balmaceda JP, Silber SJ, Asch RH. The role of the laboratory in the handling of epididymal sperm for assisted reproductive technologies. Fertil Steril 1992;57:1103-6. 5. Lattimer JK, Puchner PJ, Hensle TW, Macfarlane MT. Delayed development of the scrotum in exstrophy. J Urol 1979;121:339-40. 6. Hanna MK, Williams Dr. Genital function in males with vesical exstrophy and epispadias. Br J UroI1972;44:169-74.
Note. Additional references and a complete literature review are available from the authors upon request.
Fertility and Sterility
©
Vol. 60, No.4, October 1993
Printed on acid-free paper in U. S. A.
1993 The American Fertility Society
Fertility and the use of assisted reproductive techniques in the adult male exstrophy/epispadias patient
Martin D. Bastuba, M.D.* Michael M. Alper, M.D. t Robert D. Oates, M.D.*:j: Boston University Medical Center and Beth Israel Hospital, and Boston Fertility and Gynecology Associates/Boston IVF, Boston, Massachusetts
Exstrophy of the bladder with epispadias and separation of the pubes is an uncommon anomaly occurring in 1:30,000 live births. Surgical challenges have historically involved closure of the bladder and the achievement of urinary continence. The preservation and/or restoration of erectile and ejaculatory function are areas of more recent interest and refinement. With improvement in the lifestyle and general health of these patients, fertility issues have now assumed a level of increasing importance. Though fertility potential in the female exstrophy patient is reported to be adequate (1), spontaneous pregnancy has been reported in only 18% to 22% of couples (2, 3) in whom the male partner was born with classical bladder exstrophy. Etiologic factors implicated in the subfertility of this population include cryptorchidism, erectile dysfunction secondary to dissection of the corpora off the ischiopubic rami, retrograde ejaculation resulting from an incompetent bladder neck, recurrent epididymo-orchitis with subsequent epididymalocclusion and testicular atrophy, vasal injury at the time of herniorrhaphy, occlusive prostatic urethral stricture, dysfunctional urethral transport of semen secondary to deficient bulbospongiosus
Received December 21, 1992; revised and accepted June 14, 1993. * Department of Urology, Boston University Medical Center. t Department of Obstetrics and Gynecology, Beth Israel Hospital, and Boston Fertility and Gynecology Associates/Boston IVF. :j: Reprint requests: Robert D. Oates, M.D., DOB 606, 720 Harrison Avenue, Boston, Massachusetts 02118. Vol. 60, No.4, October 1993
muscle action, and iatrogenic injury to the seminal tract during reconstructive procedures. We report on three men, two born with classical exstrophy of the bladder and another with epispadias alone. Each couple presented after inability to initiate a pregnancy of a year or more duration. Each spouse's infertility evaluation was within normal limits. Innovative strategies, including assisted reproductive techniques in two, led to pregnancies in all cases. A review of relevant literature is presented with discussion. CASE REPORTS
Case 1 A 32-year-old male born with bladder exstrophy and bilateral inguinal hernias presented for evaluation of infertility. Shortly after birth, bladder closure, epispadias repair, and bilateral herniorrhaphy were performed without complication. Bladder neck reconstruction was carried out in early childhood. Because of continued incontinence, he underwent bilateral ureterosigmoidostomy at age 5, leaving the bladder and prostate in situ. Penile lengthening was accomplished at age 12. He married at age 30 and presented shortly thereafter. Erections were adequate for successful intravaginal intercourse. Although the patient sensed orgasm, no immediate antegrade flow of seminal fluid was present. Beginning approximately 0.5 hour after intercourse, the patient would note the slow emission of a scant amount of semen from the urethral meatus. The testicular exam was normal and the urethra was easily catheterizable. Bastuba et al.
Communications-in-brief
733
The patient was instructed to compress his urethra, from proximal to distal, to express more of the seminal fluid remaining stagnant in his proximal urethra after orgasm. In this fashion, an increased amount of semen was obtained, revealing a volume of 2.5 cc, a sperm density of 34 X 106 /cc, and a progressive motility of 85%. Seminal fluid was collected during the periovulatory period in the manner described above, with the couple performing cervical insemination. On the eighth attempt, without augmentative medications, a pregnancy was achieved with a healthy female infant delivered at term.
Human tubal fluid
Hemitransection of Vas Deferens
Case 2
A 25-year-old male born with epispadias presented with a 2-year history of failure to initiate a pregnancy. He had his epispadias closure during infancy with bladder neck reconstruction at age 4. Although continent of urine at presentation, the patient experienced delayed and scant seminal emission. The antegrade milking technique reliably produced an increased volume of ejaculate, although still <1 cc. Because of the decreased seminal volume, it was believed that the addition of lUI would enhance their chances of conception. A pregnancy resulted after the second cycle of insemination coupled with ovulatory augmentation utilizing 50 mg of clomiphene citrate cycle days 5 to 9. Semen analysis at that time revealed a total count of 97 X 106 sperm with a progressive motility of 70%. A healthy female infant was delivered at term. Case 3
A 27-year-old male born with classic bladder exstrophy as well as bilateral cryptorchidism presented with infertility. The patient underwent early bladder closure and orchidopexy. A bilateral ureterosigmoidostomy was performed after multiple bladder neck reconstructions and urethroplasties failed to produce continence of urine. Penile lengthening was accomplished at age 16 and erectile function is presently adequate. The patient noted delayed and minimal ante grade emission (0.2 to 0.4 cc). Attempted urethral catheterization was unsuccessful, prompting retrograde urethrogram. The severe obstructing urethral stricture disease visualized was not amenable to surgical correction. Unreliable semen volume and delivery (0.2 to 0.6 cc), associated with widely fluctuating total sperm output (26 X 106 , 0, 3 X 106 , and 2 X 106 ) during unsuccessful lUI trials prompted microsurgical va734
Bastuba et al.
Communications-in-brief
Figure 1 After an anterior hemivasotomy, the exuding fluid is aspirated into prelubricated syringes with attached 22-gauge plastic tip. All fluid is expelled into appropriate media. Closure of the vas is accomplished with 10-0 mucosal and 9-0 seromuscular nylon sutures.
sal sperm aspiration coupled with GIFT. Direct aspiration assured retrieval of motile sperm before oocyte harvesting. Because of the intensive nature of the advanced reproductive techniques, we did not wish to risk that this patient's ejaculate would be inadequate for GIFT on that particular day. Knowing that he had the capability for adequate sperm production, a direct aspiration from the vas guaranteed sperm availability. A standard stimulation and oocyte-harvesting protocol was used. When the E2 was 2,141 pg/mL (conversion factor to SI unit, 3.671) and there were 10 follicles ~16 mm in diameter as visualized by transvaginal ultrasonography, 10,000 U hCG 1M was given. Thirty-two hours later, the sperm specimen was micro surgically aspirated from the straight portion of the scrotal vas deferens into 0.5 cc of medium (HEPES-buffered human tubal fluid; Irvine Scientific, Irvine, CA) (Fig. 1). Sperm processing was carried out using mini-Percoll (Pharmacia, Uppsala, Sweden) in conjunction with 3 mM pentoxiphylline and 3 mM 2 deoxyadenosine (Sigma Chemical Co., St. Louis, MO) as described in detail by Ord et al. (4). A total of36 X 106 sperm with a progressive motility of 40% were recovered. Eight oocytes were then laparoscopic ally retrieved, and four were transferred along with 200,000 motile sperm to the right fallopian tube. The remaining four oocytes were incubated in vitro with 200,000 motile sperm per oocyte. Two fertilized and are currently cryopreserved at the four-cell stage. A resultant twin pregnancy is ongoing. Fertility and Sterility
DISCUSSION
Advancements in the initial and secondary surgical treatment of exstrophy/epispadias have allowed fertility issues to take on increasing prominence. Spontaneous conception in the male exstrophy / epispadias population is still impaired, however. The restoration of normal erectile function is the first obstacle that must be overcome to enable the male to achieve satisfactory intravaginal intercourse. Even with improved operative technique, Mesrobian et al. (3) state that 39% oftheir postpubertal males still could not have successful intercourse. Therefore, spontaneous conception, at its maximum, could only occur in 61% of patients. Ejaculatory dysfunction is also a major cause of failure to conceive. In the exstrophy/epispadias complex, the urethra lacks circumferential bulbospongiosus and ischiocavernosus muscles, a situation not corrected by reconstructive surgery. Normally, rhythmic contraction of the periurethral musculature during ejaculation forcefully propels the seminal fluid in an antegrade direction. In its absence, the semen either remains in the proximal urethra or, occasionally, a small amount may dribble out the urethral meatus. Proper cervical deposition of semen during intercourse may not occur and, therefore, conception would be unlikely. As in case no. 3, multiple urethral surgical endeavors may lead to significant stricture disease, impeding further the antegrade flow of semen. In the diverted patient, severe strictures may not be relevant except in this context. Testicular function is believed to be intrinsic ally normal. Although many patients have "cryptorchidism," this may be more a result of inadequate scrotal development than a failure of descent due to an inherent testicular anomaly (5). At puberty, the scrotum will oftentimes enlarge while the gonads spontaneously "descend." It is unclear whether the high position of the testes during the prepubertal years will eventually have an impact on their spermatogenic capability. The reproductive ductal structures are embryologically and developmentally normal. However, epididymal obstruction may occur as a result of an episode of epididymitis whereas orchitis may lead to testicular atrophy. Review of the literature reveals only two studies that directly address semen quality and fertility. Lattimer et al. (2) report on nine patients (17 to 32 years old) and note that five were azoospermic, even in the retrograde specimen. Two patients were severely oligospermic (0.2 to 6 X 1Q6/CC ) and two were normospermic with excellent motility. These Vol. 60, No.4, October 1993
latter two patients, although with semen volumes of 0.5 to 0.6 cc, had both initiated pregnancies. It is unclear how many of this group were actively trying to conceive but it is highly unlikely that all were, given their reported ages. Therefore, the incidence of spontaneous pregnancy achievement is, at a minimum, 22%. Hanna and Williams (6) report on 16 exstrophy patients and 15 epispadias patients. In the exstrophy cohort, only 5 produced "normal" specimens and 11 produced specimens "incompatible with the probability of conception." Exact seminal parameters were not stated. It appeared that semen quality was better, overall, in those patients who did not have attempted bladder reconstruction but only excision of exposed bladder mucosa and urinary diversion. It is interesting to note that two patients who were detailed complained of slow, oozing ejaculation subsequent to orgasm. The epispadias cohort fared much better, with 14/15 ejaculating normally. Of 13 that produced semen specimens, 9 were considered "normal" and 4 were "substandard. " Woodhouse et al. (1) documented that of 29 patients who are married, 6 have initiated pregnancies for a minimal incidence of spontaneous conception of 21%. It is generally felt that the incidence of bladder exstrophy transmission is approximately 1 %, a 500-fold increase over the general population. Assisted reproductive techniques have improved the prognosis of infertility for a number of couples in whom female factors, male factors, or both have been preventing spontaneous conception. Cervical insemination is the least complicated, may be performed at home after appropriate instruction, and can overcome deficiencies in the normal deposition of an adequate volume of ejaculate to the cervical region. Intrauterine insemination is commonly used in a compensatory fashion when semen quality is poor and no remedial cause is apparent. Ovarian stimulation may be added, potentially increasing the probability of pregnancy. Our three cases illustrate that knowledge of both the pathophysiological mechanisms preventing spontaneous conception in the adult male exstrophy patient and the newer reproductive techniques, coupled with a little imagination, can lead to a long-awaited pregnancy. All three patients had normal emission/orgasm but noted only small amounts of dribbling seminal discharge either immediately after ejaculation or several hours subsequently. The first two had patent urethras but probable absence of normal periurethral musculature contraction and, therefore, no immediate antegrade Bastuba et al.
Communications-in-brief
735
ejaculate. Absence of retrograde ejaculate was verified by catheterization and irrigation ofthe bladder. Each was taught to cleanse the urethra, to ejaculate, and then to milk the urethra from deep in the perineum to the tip of the penis to obtain an adequate semen specimen (0.4 cc, 120 X 106 ,65% and 2.5 cc, 84 X 106 ,85%, respectively). The first couple conceived after 8 months of relatively simple home cervical insemination whereas the other became pregnant after the second cycle of intrauterine insemination. Both mothers delivered healthy females without incident. Because there was no way in which to obtain a better semen specimen in the third patient because of his severe urethral stricture disease, a direct microsurgical vasal sperm aspiration was used to allow us to extract adequate numbers of high quality sperm for use in combination with GIFT. As can be seen by the fact that there is an ongoing twin pregnancy and that two additional embryos were generated, the fertilizing potential of this patient's sperm was excellent. SUMMARY
These three cases exemplify, in increasing order of complexity, how the commonsense application of already successful techniques might be applied to
736
Bastuba et al.
Communications-in-brief
augment the chance of pregnancy in this group of patients. We believe this to be the first report specifically outlining measures that may assist this group in their goal of pregnancy achievement. To our knowledge, vasal sperm aspiration has never been used for this indication. Key Words: Bladder exstrophy, epispadias, microsurgical sperm aspiration. REFERENCES 1. Woodhouse CRJ, Ransley PG, Williams Dr. The patient
with exstrophy in adult life. Br J Urol 1983;55:632-5. 2. Lattimer JK, Macfarlane MT, Puchner PJ. Male exstrophy patients: a preliminary report on the reproductive capability. Trans Am Assoc Genitourin Surg 1979;70:42-4. 3. Mesrobian HGJ, Kelalis PP, Kramer SA. Long-term followup of cosmetic appearance and genital function in boys with exstrophy: review of 53 patients. J UroI1986;136:256-8. 4. Ord T, Marello E, Patrizio P, Balmaceda JP, Silber SJ, Asch RH. The role of the laboratory in the handling of epididymal sperm for assisted reproductive technologies. Fertil Steril 1992;57:1103-6. 5. Lattimer JK, Puchner PJ, Hensle TW, Macfarlane MT. Delayed development of the scrotum in exstrophy. J Urol 1979;121:339-40. 6. Hanna MK, Williams Dr. Genital function in males with vesical exstrophy and epispadias. Br J UroI1972;44:169-74.
Note. Additional references and a complete literature review are available from the authors upon request.
Fertility and Sterility