FERTILITY AND STERILITY
Vol. 28, No. 10, October 1977 Printed in U.S.A.
Copyright' 1977 The American Fertility Society
INFERTILITY AS A CONSEQUENCE OF BILATERAL HERNIORRHAPHIES
JOHN A, SWANSON, M,D, FREDERICK K. CHAPLER, M,D,* Division of Reproductive Endocrinology, Department of Obstetrics and Gynecology, University of Iowa School of Medicine, Iowa City, Iowa 52242
The involvement of internal genitalia in inguinal hernias occurring in female infants and children has been reported, We present here an interesting infertility problem as a consequence of accidental "tubal ligation" secondary to bilateral inguinal herniorrhaphies during childhood, Several postulated etiologic factors in the development of such hernias are presented, and the potential occurrence of similar problems in other individuals is emphasized. We urge very meticulous technique when such hernias are repaired, and we reiterate the importance of a thorough medical history during infertility investigation. The potential frequency of this uncommon but devastating problem presenting in an infertility investigation may be underestimated. Additionally, it is conceivable that, with the increasing survival rates of prematurely born infants who are at increased risk for such hernias, this problem may become more frequent.
Tubal problems have been estimated to account for 30% to 35% of infertility problems. 1 Everyadequate medical history contains information regarding previous abdominal and pelvic surgical procedures, and the importance of previous acute and chronic pelvic infections resulting in compromised tubal function is obvious. How frequently, however, do we place relative unimportance on seemingly minor surgical procedures performed in infancy and early childhood? We describe just such a situation and discuss what may be a more important clinical situation than we had previously realized. CASE REPORT
Mrs. C. was a 24-year-old white female who first came to the University of Iowa Hospitals in October 1975 with the complaint of3 years of primary infertility. Her medical history revealed menarche at age 12 years with subsequent regular cycles every 28 to 30 days. Her only episode of menstrual irregularity had occurred in early 1974, Received April 20, 1977; revised May 16, 1977; accepted May 17, 1977. *To whom reprint requests should be addressed.
at which time she had skipped two menses, followed by an episode of vaginal bleeding. Dilatation and curettage performed at that time revealed secretory endometrium. The remainder of the medical history and review of systems were unremarkable, with the exception of inguinal herniorrhaphies performed at ages 12 and 18 months on opposite inguinal sides. Unfortunately, the operative summaries of each of these procedures were not available for review. Mr. C. had fathered a child during a previous marriage, and a recent semen analysis demonstrated normal fertility potential. The remainder of the infertility investigation was unremarkable, with the exception of a hysterosalpingogram demonstrating normal uterine contour but permitting visualization of the proximal fallopian tubes only, with no evidence of spillage of contrast material into the peritoneal cavity (see Fig. 1). Mrs. C. denied any history compatible with previous pelvic or intra-abdominal infections. The general physical examination was unremarkable, and a pelvic examination revealed a normal-sized, mid-positioned, freely mobile uterus with no palpable adnexal masses or tenderness. In early January 1976, laparoscopy was performed. The pelvic contents were easily vis1118
Vol. 28, No. 10
1119
COMMUNICATIONS-IN-BRIEF
FIG. 1. Preoperative hysterosalpingogram showing proximal portion of fallopian tubes only, with no patency (obstruction marked by arrows).
ualized, demonstrating a freely mobile, midpositioned uterus, and normal-appearing ovaries bilaterally. Each fallopian tube was adherent laterally to the pelvic sidewall near the internal inguinal ring. Each fimbriated end appeared normal, but there was marked atrophy of the fallopian tube and mesosalpinx for a distance of about 2.5 cm in its proximal ampullary portion bilaterally. Chromotubation with diluted indigo carmine failed to demonstrate patency of either tube. Laparotomy was performed, and, upon closer inspection, each tube was found to be adherent to the pelvic sidewall at the internal inguinal ring, in the same position as the previously described tubal atrophy. The oviduct at this site appeared very much like that in a previous tubal ligation. The areas of atrophy were excised, and a bilateral tubal reanastomosis was performed by the technique of Clyman. 2 At the termination of the procedure, both tubes were patent to chromotubation and there was excellent tubal mobility. Each tube apparently had been ligated during the previous inguinal herniorrhaphies. Pathologic examination of the excised
specimen revealed fibromuscular tissue only, with no discernible tubal lumen. Postoperatively the patient did very well, and hydrotubation performed during the early postoperative period, and later in the outpatient clinic, suggested tubal patency. Conception occurred in June 1976. The pregnancy progressed uneventfully and terminated at 40 weeks with the spontaneous delivery of an 8 lb, 2 oz, healthy female infant. DISCUSSION
Inguinal hernias in female infants occur with about one-tenth the frequency of their occurrence in male infants. Twenty per cent to thirty per cent of female inguinal hernias are sliding hernias of the broad ligament containing the internal genitalia. 3 • 4 In 1963 Richardson 3 wrote, " ... the discovery of a sliding hernia involving internal genitalia in a female infant evokes both surprise and confusion in a general surgeon of average experience." Therefore, it is not surprising ·that early articles reported the sacrifice of internal
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genitalia during inguinal hernia repair with a frequency as high as 6%.5 There seems to be a general agreement that sliding hernias of the ovaries and fallopian tubes are the most frequent findings, accounting for about 50% of these hernias. 6 Occurring less frequently are hernias containing the ovary only, the fallopian tube only, and the uterus with or without the adnexa. We emphasize that the fallopian tube is found, with or without other internal genitalia, in the vast majority of these sliqing hernias in female infants and children. Ultimately, 15% to 25% will be bilatera1. 4 It should be remembered that an inguinal hernia may be the first clinical indication of an intersex problem, and a nuclear chromatin smear as well as a biopsy of the gonad if present in the hernia should be obtained. There is imperfect closure of the processus vaginalis in an estimated 20% of full-term newborn females. 3 This finding, coupled with visceral clouding of pelvic structures, marked peritoneal mobility, and adnexal proximity to the internal inguinal ring in the newborn female infant, are considered important etiologic factors. The rigorous activities and intermittent episodes of increased intra-abdominal pressures associated with crying and straining may then lead to the development of an inguinal hernia containing the internal genitalia.
Usually a hernia, if present, is found in the first few months of life. 4 , 6 It seems to be generally accepted that prematurity increases the incidence of this type of hernia, and Richardson 3 reported that about 25% of his series of female infants and children with inguinal hernias of the internal genitalia were born prematurely. It is possible that with increasing sophistication in dealing with the prematurely born infant we may be seeing more female infants requiring herniorrhaphies. Meticulous surgical technique is mandatory to avoid more infertility patients presenting as our patient. REFERENCES 1. Behrman SJ, Kistner RW: A rational approach to the
2.
3. 4. 5. 6.
evaluation of infertility. In Progress in Infertility, Second Edition, Edited by SJ Behrman, RW Kistner. Boston, Little, Brown and Co, 1975, p 1 Clyman MJ: Tubal reconstructive surgery for infertility. In Pathways to Conception, Edited by AI Sherman. Springfield, Ill, Charles C Thomas, 1971, Chap 8, p 110 Richardson WR: Inguinal hernia of the internal genitalia in female infants and children. Am Surg 29:446, 1963 Goldstein IR, Potts WJ: Inguinal hernia in female infants and children. Ann Surg 148:819, 1958 Donovan EJ, Stanley-Brown EG: Inguinal hernia in female infants and children. Surg Gynecol Obstet 107:663, 1958 Arnheim EE, Linder JM: Inguinal hernia of the pelvic viscera in female infants. Am J Surg 92:436, 1956
Vol. 28, No. 10, October 1977 Printed in U.S.A.
Copyright' 1977 The American Fertility Society
INFERTILITY AS A CONSEQUENCE OF BILATERAL HERNIORRHAPHIES
JOHN A, SWANSON, M,D, FREDERICK K. CHAPLER, M,D,* Division of Reproductive Endocrinology, Department of Obstetrics and Gynecology, University of Iowa School of Medicine, Iowa City, Iowa 52242
The involvement of internal genitalia in inguinal hernias occurring in female infants and children has been reported, We present here an interesting infertility problem as a consequence of accidental "tubal ligation" secondary to bilateral inguinal herniorrhaphies during childhood, Several postulated etiologic factors in the development of such hernias are presented, and the potential occurrence of similar problems in other individuals is emphasized. We urge very meticulous technique when such hernias are repaired, and we reiterate the importance of a thorough medical history during infertility investigation. The potential frequency of this uncommon but devastating problem presenting in an infertility investigation may be underestimated. Additionally, it is conceivable that, with the increasing survival rates of prematurely born infants who are at increased risk for such hernias, this problem may become more frequent.
Tubal problems have been estimated to account for 30% to 35% of infertility problems. 1 Everyadequate medical history contains information regarding previous abdominal and pelvic surgical procedures, and the importance of previous acute and chronic pelvic infections resulting in compromised tubal function is obvious. How frequently, however, do we place relative unimportance on seemingly minor surgical procedures performed in infancy and early childhood? We describe just such a situation and discuss what may be a more important clinical situation than we had previously realized. CASE REPORT
Mrs. C. was a 24-year-old white female who first came to the University of Iowa Hospitals in October 1975 with the complaint of3 years of primary infertility. Her medical history revealed menarche at age 12 years with subsequent regular cycles every 28 to 30 days. Her only episode of menstrual irregularity had occurred in early 1974, Received April 20, 1977; revised May 16, 1977; accepted May 17, 1977. *To whom reprint requests should be addressed.
at which time she had skipped two menses, followed by an episode of vaginal bleeding. Dilatation and curettage performed at that time revealed secretory endometrium. The remainder of the medical history and review of systems were unremarkable, with the exception of inguinal herniorrhaphies performed at ages 12 and 18 months on opposite inguinal sides. Unfortunately, the operative summaries of each of these procedures were not available for review. Mr. C. had fathered a child during a previous marriage, and a recent semen analysis demonstrated normal fertility potential. The remainder of the infertility investigation was unremarkable, with the exception of a hysterosalpingogram demonstrating normal uterine contour but permitting visualization of the proximal fallopian tubes only, with no evidence of spillage of contrast material into the peritoneal cavity (see Fig. 1). Mrs. C. denied any history compatible with previous pelvic or intra-abdominal infections. The general physical examination was unremarkable, and a pelvic examination revealed a normal-sized, mid-positioned, freely mobile uterus with no palpable adnexal masses or tenderness. In early January 1976, laparoscopy was performed. The pelvic contents were easily vis1118
Vol. 28, No. 10
1119
COMMUNICATIONS-IN-BRIEF
FIG. 1. Preoperative hysterosalpingogram showing proximal portion of fallopian tubes only, with no patency (obstruction marked by arrows).
ualized, demonstrating a freely mobile, midpositioned uterus, and normal-appearing ovaries bilaterally. Each fallopian tube was adherent laterally to the pelvic sidewall near the internal inguinal ring. Each fimbriated end appeared normal, but there was marked atrophy of the fallopian tube and mesosalpinx for a distance of about 2.5 cm in its proximal ampullary portion bilaterally. Chromotubation with diluted indigo carmine failed to demonstrate patency of either tube. Laparotomy was performed, and, upon closer inspection, each tube was found to be adherent to the pelvic sidewall at the internal inguinal ring, in the same position as the previously described tubal atrophy. The oviduct at this site appeared very much like that in a previous tubal ligation. The areas of atrophy were excised, and a bilateral tubal reanastomosis was performed by the technique of Clyman. 2 At the termination of the procedure, both tubes were patent to chromotubation and there was excellent tubal mobility. Each tube apparently had been ligated during the previous inguinal herniorrhaphies. Pathologic examination of the excised
specimen revealed fibromuscular tissue only, with no discernible tubal lumen. Postoperatively the patient did very well, and hydrotubation performed during the early postoperative period, and later in the outpatient clinic, suggested tubal patency. Conception occurred in June 1976. The pregnancy progressed uneventfully and terminated at 40 weeks with the spontaneous delivery of an 8 lb, 2 oz, healthy female infant. DISCUSSION
Inguinal hernias in female infants occur with about one-tenth the frequency of their occurrence in male infants. Twenty per cent to thirty per cent of female inguinal hernias are sliding hernias of the broad ligament containing the internal genitalia. 3 • 4 In 1963 Richardson 3 wrote, " ... the discovery of a sliding hernia involving internal genitalia in a female infant evokes both surprise and confusion in a general surgeon of average experience." Therefore, it is not surprising ·that early articles reported the sacrifice of internal
1120
October 1977
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genitalia during inguinal hernia repair with a frequency as high as 6%.5 There seems to be a general agreement that sliding hernias of the ovaries and fallopian tubes are the most frequent findings, accounting for about 50% of these hernias. 6 Occurring less frequently are hernias containing the ovary only, the fallopian tube only, and the uterus with or without the adnexa. We emphasize that the fallopian tube is found, with or without other internal genitalia, in the vast majority of these sliqing hernias in female infants and children. Ultimately, 15% to 25% will be bilatera1. 4 It should be remembered that an inguinal hernia may be the first clinical indication of an intersex problem, and a nuclear chromatin smear as well as a biopsy of the gonad if present in the hernia should be obtained. There is imperfect closure of the processus vaginalis in an estimated 20% of full-term newborn females. 3 This finding, coupled with visceral clouding of pelvic structures, marked peritoneal mobility, and adnexal proximity to the internal inguinal ring in the newborn female infant, are considered important etiologic factors. The rigorous activities and intermittent episodes of increased intra-abdominal pressures associated with crying and straining may then lead to the development of an inguinal hernia containing the internal genitalia.
Usually a hernia, if present, is found in the first few months of life. 4 , 6 It seems to be generally accepted that prematurity increases the incidence of this type of hernia, and Richardson 3 reported that about 25% of his series of female infants and children with inguinal hernias of the internal genitalia were born prematurely. It is possible that with increasing sophistication in dealing with the prematurely born infant we may be seeing more female infants requiring herniorrhaphies. Meticulous surgical technique is mandatory to avoid more infertility patients presenting as our patient. REFERENCES 1. Behrman SJ, Kistner RW: A rational approach to the
2.
3. 4. 5. 6.
evaluation of infertility. In Progress in Infertility, Second Edition, Edited by SJ Behrman, RW Kistner. Boston, Little, Brown and Co, 1975, p 1 Clyman MJ: Tubal reconstructive surgery for infertility. In Pathways to Conception, Edited by AI Sherman. Springfield, Ill, Charles C Thomas, 1971, Chap 8, p 110 Richardson WR: Inguinal hernia of the internal genitalia in female infants and children. Am Surg 29:446, 1963 Goldstein IR, Potts WJ: Inguinal hernia in female infants and children. Ann Surg 148:819, 1958 Donovan EJ, Stanley-Brown EG: Inguinal hernia in female infants and children. Surg Gynecol Obstet 107:663, 1958 Arnheim EE, Linder JM: Inguinal hernia of the pelvic viscera in female infants. Am J Surg 92:436, 1956
