- Email: [email protected]
Ovarian Tissue Cryopreservation Procedures; Who Is In?
e58
Poster Abstracts / J Pediatr Adolesc Gynecol 28 (2015) e41ee78
Comment: This case underscores the importance of a broad differential when considering etiologies of a cystic pelvic mass. Non-gynecological cysts such as enteric cysts (tailgut cysts and cystic rectal duplication), urachal and renal cysts, cystic sacrococcygeal teratomas, anterior sacral meningoceles, extra peritoneal adenomucinosis, cystic lymphangiomas, neurenteric cysts, mesenteric cysts, dermoid and epidermoid cysts should be considered, albeit many of these are rare occurrences. Complete surgical excision is indicated to establish the diagnosis and avoid complications. 39. Oophoropexy To Prevent Recurrent Ovarian Torsion: A Case Series Nicole Hubner MD, Lisa M. Allen MD, Sari Kives MD* The Hospital for Sick Children and the University of Toronto, Toronto, Ontario, Canada
Background: It has been suggested that particularly in children with torsion, pexy of the untwisted ovary, the contralateral ovary or both should be considered. Laparoscopic oophoropexy is overall a procedure with low morbidity and seems justifiable in order to avoid devastating consequences. If an oophoropexy is performed the ovary should be pexed to the pelvic sidewall, the back of the uterus or the ipsilateral utero-sacral ligament with a non-absorbable suture. Case: We present a case series of 11 patients less than 18 years old with surgically confirmed ovarian torsion that underwent unilateral or bilateral oophoropexy (OP) from January 1, 2004 to October 15, 2013. The rate of OP for surgically confirmed ovarian torsion was 11.3% (11/97). The mean age of patients in our series was 8.82 (+/-3.79) years. Half of the patients were pubertal (5/10), only 27% (3/11) were post-menarchal. A gynecologist was the primary surgeon for all cases. Eighty-two percent (9/11) of cases were completed by laparoscopy. The majority of patients had idiopathic adnexal torsion (82%). Forty-five and a half percent (5/11) had OP during their primary surgery, 45.5% (5/11) during a second surgery and 9% (1/11) during a third surgery. Indications for OP included: idiopathic adnexal torsion (8/11), recurrent torsion of ipsi-or contralateral ovary (5/11), bilateral torsion (2/11), torsion with concurrent or previous oophorectomy (2/11). Patients may have had more than one indication for OP. The ovary was pexed to the utero-sacral ligament in the majority of cases (82%), followed by pexy of the utero-ovarian ligament to utero-sacral ligament (9%), or shortening of the uteroovarian ligament (9%). Suture type used for OP was: non-absorbable (73%) and delayed absorbable (27%). There was one patient who experienced bleeding from the untwisted and pexed ovary intra-operatively as well as fever in the first 72 hours post operatively. One patient (9%) had a recurrent ovarian torsion after OP. This recurrence was the patient’s third surgery on the same ovary. At the previous surgery an OP had been performed after bivalving the ovary and a delayed absorbable suture was used to shorten the utero-ovarian ligament. At the third surgery the ovary was untwisted and a mature teratoma was identified and removed. Comments: In our institution oophoropexy was performed in 11% of ovarian torsions most commonly for the reasons of idiopathic ovarian torsion or recurrent torsion. The recurrence rate of ovarian torsion after oophoropexy in our series was nine percent, which is similar to the recurrence rate for torsions that occur in adnexa without leading pathology in the absence of oophoropexy. Future research should assess if there is a superior type of oophoropexy that has the lowest recurrence rate (i.e. pexy of the ovary to utero-sacral ligament vs. shortening of the utero-ovarian ligament). 40. Vulvar Abscesses in the Pediatric Population Maggie Dwiggins MD, Esther Fuchs MD, Thusitha Cotter MD University of Illinois College of Medicine-Peoria, Peoria, IL
Introduction: A variety of treatment approaches are available for vulvar abscess. Many specialties are involved in their management, but criteria for early recognition of high risk patients and prompt referral guidelines do not exist. Methods: A retrospective cohort of pediatric patients with vulvar abscess treated at a single, urban tertiary care center from 2011 to 2013 was identified. Patients equal to or under age 18 were included. Cases were
identified by admission diagnosis of vulvar abscess in the electronic medical record and included both primary occurrences and recurrences within the given timeframe. Multivariate analysis and Fisher exact test was used to analyze data. IRB approval was obtained. Results: A total of 57 pediatric patients with a mean age of 5.33 (SD 6.24) years were identified. The prevalence of LOS 1 day was 57% (n ¼ 31). Characteristics that predicted LOS 1 day were 2 preoperative comorbidities (OR 2.07, 95% CI 1.59-3.37). All of patients were managed by surgical procedures by both Gynecologic (7%) and non-Gynecologic specialties (93%). Most surgeries were either one day after admission or scheduled as outpatient surgery, 40.4% and 36.8% respectively. Total recurrence rate was 40.4% after surgery. Upon admission, 11 patients (19%) met SIRS criteria. Of these patients, all were treated as inpatients with a recurrence rate of 45.5%, not significantly different from non-SIRS group (p ¼ 0.74). A total of 25 children were treated first with outpatient antibiotics before requiring surgery, and of that group, 13 (52%) had recurrence. This rate was again not significantly different than the group not receiving prior outpatient antibiotic therapy (p ¼ 0.17). Vulvar cultures were obtained in 57.9% of patients, 25 out of 33 were positive for MRSA infection. In this group, only 11 patients had recurrence (40%), all but one patient had been treated with antibiotics. Only 4 patients (7.01%) had gonorrhea or chlamydia testing. Pathology was sent on 19 patients (33.3%), six cases were HPV related condyloma acuminatum. Conclusion: This study identified a subset of high risk patients with vulvar abscess who would benefit from earlier treatment and more specialized services and interventions. A red flag that was noted during this study was that six children all under the age of eight were found to have HPV positive condyloma, and none of these patients had further work up for co-existent STIs or abuse. While many of these patients received treatment adequate to prevent recurrence, we believe some of these patients could have benefited from more comprehensive care.
41. Ovarian Tissue Cryopreservation Procedures; Who Is In? Janie Benoit MD 1, Laura Kruger MD 2, Karen Burns MD 1, Olivia Jaworek Frias RN 1, Lesley Breech MD 1, Leslie A. Appiah MD*3 1 Cincinnati Children’s Hospital Medical Center (CCHMC), Cincinnati, OH 2 Arkansas Children’s Hospital, Little Rock, AR 3 Kentucky Children’s Hospital, Lexington, KY
Background: There are now many conditions such as cancer and hematologic disorders, for which gonadotoxic therapy is used. The American Society for Reproductive Medicine (ASRM) and the American Society for Clinical Oncology (ASCO) recommend all patients be counseled about potential risks for infertility before starting any potentially gonadotoxic therapy. We present our experience in a single pediatric institution on a single fertility preservation technique: ovarian tissue cryopreservation (OTC). Although this is still considered experimental, it may be of significant benefit to pediatric patients. Methods: An Institutional Review Board approved retrospective chart review was performed of all OTC procedures conducted at CCHMC between November 2010 and October 2014. We reviewed the charts of 21 patients and obtained the following: age, pubertal status, marital status, previous gravidity and parity, primary diagnosis, therapy planned, premature ovarian insufficiency (POI) risk, possibility for delay of treatment, surgical technique and laterality of oophorectomy. Premature ovarian insufficiency risk category was defined as low: less than 20%, intermediate: 20 to 80% or high: more than 80%. Results: A total of 21 patients underwent OTC. The age range was three to 27 years and the average age was 12.5 years old. Thirteen (62%) patients were pre-menarchal. There were seven adult patients (18 years and older); one was married, and three were in relationships, one of which had a child. Of the patients who underwent OTC, there were five (24%) patients with solid tumors, four (19%) with neurologic tumors and 12 (57%) with hematologic disorders requiring bone marrow transplant (BMT). All the patients were characterized as high risk for POI from their planned therapy. The only patients who could delay treatment were those with hematologic conditions requiring BMT, of which only one was pubertal. All the patients diagnosed with a rapidly progressing tumor could not delay chemotherapy therefore their only option was OTC. The OTC procedure did not delay
Poster Abstracts / J Pediatr Adolesc Gynecol 28 (2015) e41ee78
treatment for any of the patients. The same surgical approach was utilized for all OTC: unilateral oophorectomy by laparoscopy. The laterality of the ovary removed for OTC was chosen by the gynecologist intra-operatively based on ovarian size, individual anatomy and pathologic process. Right oophorectomy was performed in 13 (62%) patients and left oophorectomy in eight (38%) patients. Only one patient had laterality determined preoperatively because she had a contralateral ovarian malignancy. Only one patient underwent more than one fertility preservation therapy: OTC and contralateral ovarian transposition. No complications were encountered from any of the procedures. Conclusion: Through our local experience, we found that OTC, despite being experimental, offers a viable fertility preservation option for many patients, who otherwise would not have any other options. None of our patients have used the cryopreserved ovarian tissue yet. Future fertility outcome with OTC initially performed in our center will provide us with a better knowledge about success rate. A combination of various fertility preservation options can be used and should be offered when possible. 42. Pregnancy Outcomes in Patients with Anorectal Malformations Serena H. Chan MD 1, Beth I. Schwartz MD 1, Morgan Alexander 1, Bruno Martinez-Leo MD 2, Andrea Bischoff MD 2, Belinda Dickie MD, PhD 2, Jason Frischer MD 2, Marc Levitt MD 3, Alberto Pena MD 2, Lesley L. Breech MD*1 1 Division of Pediatric and Adolescent Gynecology, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH 2 Colorectal Center, Division of Pediatric Surgery, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH 3 Center for Colorectal and Pelvic Reconstruction, Nationwide Children’s Hospital, Columbus, OH
Background: Anorectal malformations (ARM) in females encompass a variety of anomalies, and are often characterized by an imperforate anus with a rectal fistula that opens along the length of the perineum, vestibule, or vagina, with cloaca being the most complex malformations. Repair of ARM also vary in complexity, and, along with medical co-morbidities, can have implications for future reproduction. There is currently a paucity of data regarding pregnancy outcomes in patients with ARM. The aim of this study is to describe reproductive outcomes in female ARM patients. Methods: Under an IRB-approved protocol, we extracted data from a deidentified database of patients with ARM who received care from the participating authors. We examined data for female patients regarding type of ARM, reproductive anatomy, mode of conception and delivery, pregnancy complications, and gestational age (GA) of delivery. Results: We identified 38 pregnancies in 24 ARM patients, with five spontaneous abortions or fetal demise, one neonatal demise due to prematurity, and 32 live-born infants (one twin pregnancy). Eight patients had cloaca, one had a recto-perineal fistula, one had an H-type rectovaginal fistula, and the remaining 14 patients had recto-vestibular fistula. Seven patients were noted to have uterine didelphys, one had a unicornuate uterus, one had a bicornuate uterus, and five had a normal uterus (uterine anatomy not documented for the remaining 10 patients). Ten patients reported spontaneous conception, three underwent in vitro fertilization, and mode of conception data were not available for the remaining 11 patients. Four patients delivered prematurely in the 3rd trimester, one patient delivered prematurely at an unknown GA (neonatal demise), nine pregnancies were delivered at term, and GA data was not available for the remaining patients/pregnancies. Two patients with recto-vestibular fistula delivered via C-section prior to ARM repair. Another 18 pregnancies were delivered via C-section, either scheduled because of anatomy and prior repair, or for another indication, such as twin gestation or breech presentation. There were seven vaginal deliveries in three patients (two recto-vestibular fistulas, one recto-perineal fistula). Mode of delivery data was not available for five pregnancies, and one patient is currently still pregnant. Pregnancy and delivery complications included preterm labor and delivery, preterm premature rupture of membranes, and intra-op bladder injury. Conclusions: Our study suggests that many female ARM patients are able to conceive spontaneously. Also, the majority of our patients, and all
e59
cloacas, delivered via C-section, whether due to prior repair, non-vertex presentation, or provider/patient preference. The majority of premature deliveries occurred after 32 weeks GA. Although there are a limited number of patients and pregnancies in this study, this is the largest cohort of ARM patients with pregnancies described to date. Moving forward, it will be important to gather information regarding changes in bowel and bladder function during pregnancy, antenatal and peripartum complications, and long-term outcomes after pregnancy and delivery. 43. Association of Uterovaginal Anomalies With Hydrocolpos and Common Channel Length in Patients With Cloacal Malformation Serena H. Chan MD 1, Beth I. Schwartz MD 1, Morgan Alexander 1, Md Monir Hossain PhD 3, Bruno Martinez-Leo MD 2, Andrea Bischoff MD 2, Belinda Dickie MD, PhD 2, Jason Frischer MD 2, Marc Levitt MD 4, Alberto Pena MD 2, Lesley L. Breech MD*1 1 Division of Pediatric and Adolescent Gynecology, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH 2 Colorectal Center, Division of Pediatric Surgery, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH 3 Division of Biostatistics and Epidemiology, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH 4 Center for Colorectal and Pelvic Reconstruction, Nationwide Children’s Hospital, Columbus, OH
Background: Cloaca is an anorectal malformation (ARM) that affects females and is associated with a high rate of uterovaginal (UV) anomalies. The length of the common channel (CC) can affect the surgical approach, and the potential need for complex reconstruction, which can have implications for future sexual and reproductive function. Hydrocolpos can be seen with cloaca and must be urgently addressed to prevent renal injury and damage to the reproductive tract. The purpose of this study is to evaluate the associations between the presence of hydrocolpos, UV anomalies, and CC length. Methods: Under an IRB-approved protocol, we extracted data for cloaca patients from a de-identified database of ARM patients who received care from the participating authors. We first determined the prevalence of UV duplication in all cloaca patients. We then assessed the association between hydrocolpos and UV anomalies, hydrocolpos and CC length, and UV duplication and CC length, using chi-square tests and regression models. Statistical analysis was performed using SAS. Prior literature has shown that a CC length >3 cm may require laparotomy and more complex repair, so we used three cm as a cut-off point. Results: We identified 645 cloaca patients in our database. Reproductive anatomy was recorded for 622 patients, with UV duplication in 363 (58.4%). Two-hundred and eighteen of 638 patients (34.2%) had hydrocolpos, with UV duplication in 155 of 211 patients (73.5%) and any UV anomaly in 171 of 211 patients (81.0%). The greater percentage of UV duplication in patients with hydrocolpos, compared to UV duplication in all cloacas, was statistically significant (p <0.05). Common channel length was available for 543 patients, with 244 (45.0%) with a length >3 cm. Of the 318 patients with UV duplication and recorded CC length, 184 (57.9%) had a CC length >3 cm and 134 (42.1%) had a CC length 3 cm (p <0.05). Using a regression model, the mean CC length in patients with and without UV duplication was 3.8 cm and 2.7 cm, respectively (p <0.05). Of the 196 patients with hydrocolpos present and CC length recorded, 136 (69.4%) had a CC length >3 cm, and 60 (30.6%) had a CC length 3 cm (p <0.05). Using a regression model, the mean CC length in patients with and without hydrocolpos was 4.1 cm and 2.9 cm, respectively (p <0.05). Conclusions: Our data suggest that cloaca patients with hydrocolpos are more likely to have UV duplication. Also, cloaca patients with UV duplication and hydrocolpos are more likely to have a longer CC. These findings are significant because adequate drainage of hydrocolpos is necessary to prevent damage to the reproductive and urinary tracts. With UV duplication, it is essential to ensure that both hemi-vaginas are drained. With longer CC length, need for laparotomy for repair increases, and with the strong association of UV duplication, it is critical to confirm patency of the entire Mullerian system. If laparotomy is not required, it is mandatory to
Poster Abstracts / J Pediatr Adolesc Gynecol 28 (2015) e41ee78
Comment: This case underscores the importance of a broad differential when considering etiologies of a cystic pelvic mass. Non-gynecological cysts such as enteric cysts (tailgut cysts and cystic rectal duplication), urachal and renal cysts, cystic sacrococcygeal teratomas, anterior sacral meningoceles, extra peritoneal adenomucinosis, cystic lymphangiomas, neurenteric cysts, mesenteric cysts, dermoid and epidermoid cysts should be considered, albeit many of these are rare occurrences. Complete surgical excision is indicated to establish the diagnosis and avoid complications. 39. Oophoropexy To Prevent Recurrent Ovarian Torsion: A Case Series Nicole Hubner MD, Lisa M. Allen MD, Sari Kives MD* The Hospital for Sick Children and the University of Toronto, Toronto, Ontario, Canada
Background: It has been suggested that particularly in children with torsion, pexy of the untwisted ovary, the contralateral ovary or both should be considered. Laparoscopic oophoropexy is overall a procedure with low morbidity and seems justifiable in order to avoid devastating consequences. If an oophoropexy is performed the ovary should be pexed to the pelvic sidewall, the back of the uterus or the ipsilateral utero-sacral ligament with a non-absorbable suture. Case: We present a case series of 11 patients less than 18 years old with surgically confirmed ovarian torsion that underwent unilateral or bilateral oophoropexy (OP) from January 1, 2004 to October 15, 2013. The rate of OP for surgically confirmed ovarian torsion was 11.3% (11/97). The mean age of patients in our series was 8.82 (+/-3.79) years. Half of the patients were pubertal (5/10), only 27% (3/11) were post-menarchal. A gynecologist was the primary surgeon for all cases. Eighty-two percent (9/11) of cases were completed by laparoscopy. The majority of patients had idiopathic adnexal torsion (82%). Forty-five and a half percent (5/11) had OP during their primary surgery, 45.5% (5/11) during a second surgery and 9% (1/11) during a third surgery. Indications for OP included: idiopathic adnexal torsion (8/11), recurrent torsion of ipsi-or contralateral ovary (5/11), bilateral torsion (2/11), torsion with concurrent or previous oophorectomy (2/11). Patients may have had more than one indication for OP. The ovary was pexed to the utero-sacral ligament in the majority of cases (82%), followed by pexy of the utero-ovarian ligament to utero-sacral ligament (9%), or shortening of the uteroovarian ligament (9%). Suture type used for OP was: non-absorbable (73%) and delayed absorbable (27%). There was one patient who experienced bleeding from the untwisted and pexed ovary intra-operatively as well as fever in the first 72 hours post operatively. One patient (9%) had a recurrent ovarian torsion after OP. This recurrence was the patient’s third surgery on the same ovary. At the previous surgery an OP had been performed after bivalving the ovary and a delayed absorbable suture was used to shorten the utero-ovarian ligament. At the third surgery the ovary was untwisted and a mature teratoma was identified and removed. Comments: In our institution oophoropexy was performed in 11% of ovarian torsions most commonly for the reasons of idiopathic ovarian torsion or recurrent torsion. The recurrence rate of ovarian torsion after oophoropexy in our series was nine percent, which is similar to the recurrence rate for torsions that occur in adnexa without leading pathology in the absence of oophoropexy. Future research should assess if there is a superior type of oophoropexy that has the lowest recurrence rate (i.e. pexy of the ovary to utero-sacral ligament vs. shortening of the utero-ovarian ligament). 40. Vulvar Abscesses in the Pediatric Population Maggie Dwiggins MD, Esther Fuchs MD, Thusitha Cotter MD University of Illinois College of Medicine-Peoria, Peoria, IL
Introduction: A variety of treatment approaches are available for vulvar abscess. Many specialties are involved in their management, but criteria for early recognition of high risk patients and prompt referral guidelines do not exist. Methods: A retrospective cohort of pediatric patients with vulvar abscess treated at a single, urban tertiary care center from 2011 to 2013 was identified. Patients equal to or under age 18 were included. Cases were
identified by admission diagnosis of vulvar abscess in the electronic medical record and included both primary occurrences and recurrences within the given timeframe. Multivariate analysis and Fisher exact test was used to analyze data. IRB approval was obtained. Results: A total of 57 pediatric patients with a mean age of 5.33 (SD 6.24) years were identified. The prevalence of LOS 1 day was 57% (n ¼ 31). Characteristics that predicted LOS 1 day were 2 preoperative comorbidities (OR 2.07, 95% CI 1.59-3.37). All of patients were managed by surgical procedures by both Gynecologic (7%) and non-Gynecologic specialties (93%). Most surgeries were either one day after admission or scheduled as outpatient surgery, 40.4% and 36.8% respectively. Total recurrence rate was 40.4% after surgery. Upon admission, 11 patients (19%) met SIRS criteria. Of these patients, all were treated as inpatients with a recurrence rate of 45.5%, not significantly different from non-SIRS group (p ¼ 0.74). A total of 25 children were treated first with outpatient antibiotics before requiring surgery, and of that group, 13 (52%) had recurrence. This rate was again not significantly different than the group not receiving prior outpatient antibiotic therapy (p ¼ 0.17). Vulvar cultures were obtained in 57.9% of patients, 25 out of 33 were positive for MRSA infection. In this group, only 11 patients had recurrence (40%), all but one patient had been treated with antibiotics. Only 4 patients (7.01%) had gonorrhea or chlamydia testing. Pathology was sent on 19 patients (33.3%), six cases were HPV related condyloma acuminatum. Conclusion: This study identified a subset of high risk patients with vulvar abscess who would benefit from earlier treatment and more specialized services and interventions. A red flag that was noted during this study was that six children all under the age of eight were found to have HPV positive condyloma, and none of these patients had further work up for co-existent STIs or abuse. While many of these patients received treatment adequate to prevent recurrence, we believe some of these patients could have benefited from more comprehensive care.
41. Ovarian Tissue Cryopreservation Procedures; Who Is In? Janie Benoit MD 1, Laura Kruger MD 2, Karen Burns MD 1, Olivia Jaworek Frias RN 1, Lesley Breech MD 1, Leslie A. Appiah MD*3 1 Cincinnati Children’s Hospital Medical Center (CCHMC), Cincinnati, OH 2 Arkansas Children’s Hospital, Little Rock, AR 3 Kentucky Children’s Hospital, Lexington, KY
Background: There are now many conditions such as cancer and hematologic disorders, for which gonadotoxic therapy is used. The American Society for Reproductive Medicine (ASRM) and the American Society for Clinical Oncology (ASCO) recommend all patients be counseled about potential risks for infertility before starting any potentially gonadotoxic therapy. We present our experience in a single pediatric institution on a single fertility preservation technique: ovarian tissue cryopreservation (OTC). Although this is still considered experimental, it may be of significant benefit to pediatric patients. Methods: An Institutional Review Board approved retrospective chart review was performed of all OTC procedures conducted at CCHMC between November 2010 and October 2014. We reviewed the charts of 21 patients and obtained the following: age, pubertal status, marital status, previous gravidity and parity, primary diagnosis, therapy planned, premature ovarian insufficiency (POI) risk, possibility for delay of treatment, surgical technique and laterality of oophorectomy. Premature ovarian insufficiency risk category was defined as low: less than 20%, intermediate: 20 to 80% or high: more than 80%. Results: A total of 21 patients underwent OTC. The age range was three to 27 years and the average age was 12.5 years old. Thirteen (62%) patients were pre-menarchal. There were seven adult patients (18 years and older); one was married, and three were in relationships, one of which had a child. Of the patients who underwent OTC, there were five (24%) patients with solid tumors, four (19%) with neurologic tumors and 12 (57%) with hematologic disorders requiring bone marrow transplant (BMT). All the patients were characterized as high risk for POI from their planned therapy. The only patients who could delay treatment were those with hematologic conditions requiring BMT, of which only one was pubertal. All the patients diagnosed with a rapidly progressing tumor could not delay chemotherapy therefore their only option was OTC. The OTC procedure did not delay
Poster Abstracts / J Pediatr Adolesc Gynecol 28 (2015) e41ee78
treatment for any of the patients. The same surgical approach was utilized for all OTC: unilateral oophorectomy by laparoscopy. The laterality of the ovary removed for OTC was chosen by the gynecologist intra-operatively based on ovarian size, individual anatomy and pathologic process. Right oophorectomy was performed in 13 (62%) patients and left oophorectomy in eight (38%) patients. Only one patient had laterality determined preoperatively because she had a contralateral ovarian malignancy. Only one patient underwent more than one fertility preservation therapy: OTC and contralateral ovarian transposition. No complications were encountered from any of the procedures. Conclusion: Through our local experience, we found that OTC, despite being experimental, offers a viable fertility preservation option for many patients, who otherwise would not have any other options. None of our patients have used the cryopreserved ovarian tissue yet. Future fertility outcome with OTC initially performed in our center will provide us with a better knowledge about success rate. A combination of various fertility preservation options can be used and should be offered when possible. 42. Pregnancy Outcomes in Patients with Anorectal Malformations Serena H. Chan MD 1, Beth I. Schwartz MD 1, Morgan Alexander 1, Bruno Martinez-Leo MD 2, Andrea Bischoff MD 2, Belinda Dickie MD, PhD 2, Jason Frischer MD 2, Marc Levitt MD 3, Alberto Pena MD 2, Lesley L. Breech MD*1 1 Division of Pediatric and Adolescent Gynecology, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH 2 Colorectal Center, Division of Pediatric Surgery, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH 3 Center for Colorectal and Pelvic Reconstruction, Nationwide Children’s Hospital, Columbus, OH
Background: Anorectal malformations (ARM) in females encompass a variety of anomalies, and are often characterized by an imperforate anus with a rectal fistula that opens along the length of the perineum, vestibule, or vagina, with cloaca being the most complex malformations. Repair of ARM also vary in complexity, and, along with medical co-morbidities, can have implications for future reproduction. There is currently a paucity of data regarding pregnancy outcomes in patients with ARM. The aim of this study is to describe reproductive outcomes in female ARM patients. Methods: Under an IRB-approved protocol, we extracted data from a deidentified database of patients with ARM who received care from the participating authors. We examined data for female patients regarding type of ARM, reproductive anatomy, mode of conception and delivery, pregnancy complications, and gestational age (GA) of delivery. Results: We identified 38 pregnancies in 24 ARM patients, with five spontaneous abortions or fetal demise, one neonatal demise due to prematurity, and 32 live-born infants (one twin pregnancy). Eight patients had cloaca, one had a recto-perineal fistula, one had an H-type rectovaginal fistula, and the remaining 14 patients had recto-vestibular fistula. Seven patients were noted to have uterine didelphys, one had a unicornuate uterus, one had a bicornuate uterus, and five had a normal uterus (uterine anatomy not documented for the remaining 10 patients). Ten patients reported spontaneous conception, three underwent in vitro fertilization, and mode of conception data were not available for the remaining 11 patients. Four patients delivered prematurely in the 3rd trimester, one patient delivered prematurely at an unknown GA (neonatal demise), nine pregnancies were delivered at term, and GA data was not available for the remaining patients/pregnancies. Two patients with recto-vestibular fistula delivered via C-section prior to ARM repair. Another 18 pregnancies were delivered via C-section, either scheduled because of anatomy and prior repair, or for another indication, such as twin gestation or breech presentation. There were seven vaginal deliveries in three patients (two recto-vestibular fistulas, one recto-perineal fistula). Mode of delivery data was not available for five pregnancies, and one patient is currently still pregnant. Pregnancy and delivery complications included preterm labor and delivery, preterm premature rupture of membranes, and intra-op bladder injury. Conclusions: Our study suggests that many female ARM patients are able to conceive spontaneously. Also, the majority of our patients, and all
e59
cloacas, delivered via C-section, whether due to prior repair, non-vertex presentation, or provider/patient preference. The majority of premature deliveries occurred after 32 weeks GA. Although there are a limited number of patients and pregnancies in this study, this is the largest cohort of ARM patients with pregnancies described to date. Moving forward, it will be important to gather information regarding changes in bowel and bladder function during pregnancy, antenatal and peripartum complications, and long-term outcomes after pregnancy and delivery. 43. Association of Uterovaginal Anomalies With Hydrocolpos and Common Channel Length in Patients With Cloacal Malformation Serena H. Chan MD 1, Beth I. Schwartz MD 1, Morgan Alexander 1, Md Monir Hossain PhD 3, Bruno Martinez-Leo MD 2, Andrea Bischoff MD 2, Belinda Dickie MD, PhD 2, Jason Frischer MD 2, Marc Levitt MD 4, Alberto Pena MD 2, Lesley L. Breech MD*1 1 Division of Pediatric and Adolescent Gynecology, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH 2 Colorectal Center, Division of Pediatric Surgery, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH 3 Division of Biostatistics and Epidemiology, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH 4 Center for Colorectal and Pelvic Reconstruction, Nationwide Children’s Hospital, Columbus, OH
Background: Cloaca is an anorectal malformation (ARM) that affects females and is associated with a high rate of uterovaginal (UV) anomalies. The length of the common channel (CC) can affect the surgical approach, and the potential need for complex reconstruction, which can have implications for future sexual and reproductive function. Hydrocolpos can be seen with cloaca and must be urgently addressed to prevent renal injury and damage to the reproductive tract. The purpose of this study is to evaluate the associations between the presence of hydrocolpos, UV anomalies, and CC length. Methods: Under an IRB-approved protocol, we extracted data for cloaca patients from a de-identified database of ARM patients who received care from the participating authors. We first determined the prevalence of UV duplication in all cloaca patients. We then assessed the association between hydrocolpos and UV anomalies, hydrocolpos and CC length, and UV duplication and CC length, using chi-square tests and regression models. Statistical analysis was performed using SAS. Prior literature has shown that a CC length >3 cm may require laparotomy and more complex repair, so we used three cm as a cut-off point. Results: We identified 645 cloaca patients in our database. Reproductive anatomy was recorded for 622 patients, with UV duplication in 363 (58.4%). Two-hundred and eighteen of 638 patients (34.2%) had hydrocolpos, with UV duplication in 155 of 211 patients (73.5%) and any UV anomaly in 171 of 211 patients (81.0%). The greater percentage of UV duplication in patients with hydrocolpos, compared to UV duplication in all cloacas, was statistically significant (p <0.05). Common channel length was available for 543 patients, with 244 (45.0%) with a length >3 cm. Of the 318 patients with UV duplication and recorded CC length, 184 (57.9%) had a CC length >3 cm and 134 (42.1%) had a CC length 3 cm (p <0.05). Using a regression model, the mean CC length in patients with and without UV duplication was 3.8 cm and 2.7 cm, respectively (p <0.05). Of the 196 patients with hydrocolpos present and CC length recorded, 136 (69.4%) had a CC length >3 cm, and 60 (30.6%) had a CC length 3 cm (p <0.05). Using a regression model, the mean CC length in patients with and without hydrocolpos was 4.1 cm and 2.9 cm, respectively (p <0.05). Conclusions: Our data suggest that cloaca patients with hydrocolpos are more likely to have UV duplication. Also, cloaca patients with UV duplication and hydrocolpos are more likely to have a longer CC. These findings are significant because adequate drainage of hydrocolpos is necessary to prevent damage to the reproductive and urinary tracts. With UV duplication, it is essential to ensure that both hemi-vaginas are drained. With longer CC length, need for laparotomy for repair increases, and with the strong association of UV duplication, it is critical to confirm patency of the entire Mullerian system. If laparotomy is not required, it is mandatory to