Autoimmune oophoritis and subsequent diminished ovarian reserve (DOR) or premature ovarian insufficiency (POI) is a rare cause of subfertility, representing approximately 4% of spontaneous POI. It often coexists with thyroid and/or adrenal autoimmunity, or can be a part of Polyglandular Autoimmune Syndrome (APS), Type I and Type II. The purpose of this study is to describe several cases of autoimmune DOR/POI in children/adolescents, as well as their ovarian tissue findings to help inform management of this rare condition.
Case
Patient A is a 16-year-old female with a history of Factor V Leiden heterozygous, who was diagnosed with POI during workup of sudden alopecia with labs notable for elevated FSH (129 mIU/mL) and undetectable estradiol. She was also noted to have anti-thyroid peroxidase antibodies, anti-thyroglobulin antibodies, and anti-adrenal antibodies, raising concern for polyglandular autoimmune failure. Menarche occurred at age 12 and she had regular menses, which became lighter but ongoing the time of her diagnosis. She underwent laparoscopic right oophorectomy and ovarian tissue cryopreservation. Ovarian histology demonstrated no ovarian follicles. Patient B is a 13-year-old who initially presented with weight loss, palpitations, and secondary amenorrhea. Menarche occurred at age 11 with subsequent regular menses until an abrupt stop at 13 years old. Labs demonstrated hyperthyroidism due to Grave's disease (TSH < 0.005 mIU/L, T4 15.8 mcg/dL, thyroid-stimulating immunoglobulin 413 IU/L) and hypergonadotropic hypogonadism consistent with POI (FSH 59.3 mIU/mL, LH 15.5 IU/L. estradiol 8 pg/mL, AMH 0.01 ng/m). She underwent laparoscopic right oophorectomy and ovarian tissue cryopreservation. Ovarian histology demonstrated no ovarian follicles.
Comments
This study demonstrated no follicles in either of the subjects with POI due to autoimmune oophoritis. It is possible that the absence of follicles represents generally worse outcomes of ovarian tissue cryopreservation for patients with autoimmune POI compared to other causes of POI. That said, little is known about the chronology of this condition, and earlier detection and recognition of autoimmune oophoritis might allow for improved fertility outcomes. Notably both of these patients were post-pubertal. It is unknown if fertility preservation outcomes for patients with autoimmune POI differ by pubertal status. More studies are needed to determine who are optimal candidates for ovarian tissue cryopreservation, especially for rare causes of POI like autoimmune oophoritis.
{"title":"44. Follicular findings in autoimmune oophoritis: 2 cases from the NIH ovarian tissue cryopreservation protocol","authors":"Carolyn Brookhart , Jacqueline Maher , Veronica Gomez-Lobo","doi":"10.1016/j.jpag.2025.01.077","DOIUrl":"10.1016/j.jpag.2025.01.077","url":null,"abstract":"<div><h3>Background</h3><div>Autoimmune oophoritis and subsequent diminished ovarian reserve (DOR) or premature ovarian insufficiency (POI) is a rare cause of subfertility, representing approximately 4% of spontaneous POI. It often coexists with thyroid and/or adrenal autoimmunity, or can be a part of Polyglandular Autoimmune Syndrome (APS), Type I and Type II. The purpose of this study is to describe several cases of autoimmune DOR/POI in children/adolescents, as well as their ovarian tissue findings to help inform management of this rare condition.</div></div><div><h3>Case</h3><div>Patient A is a 16-year-old female with a history of Factor V Leiden heterozygous, who was diagnosed with POI during workup of sudden alopecia with labs notable for elevated FSH (129 mIU/mL) and undetectable estradiol. She was also noted to have anti-thyroid peroxidase antibodies, anti-thyroglobulin antibodies, and anti-adrenal antibodies, raising concern for polyglandular autoimmune failure. Menarche occurred at age 12 and she had regular menses, which became lighter but ongoing the time of her diagnosis. She underwent laparoscopic right oophorectomy and ovarian tissue cryopreservation. Ovarian histology demonstrated no ovarian follicles. Patient B is a 13-year-old who initially presented with weight loss, palpitations, and secondary amenorrhea. Menarche occurred at age 11 with subsequent regular menses until an abrupt stop at 13 years old. Labs demonstrated hyperthyroidism due to Grave's disease (TSH < 0.005 mIU/L, T4 15.8 mcg/dL, thyroid-stimulating immunoglobulin 413 IU/L) and hypergonadotropic hypogonadism consistent with POI (FSH 59.3 mIU/mL, LH 15.5 IU/L. estradiol 8 pg/mL, AMH 0.01 ng/m). She underwent laparoscopic right oophorectomy and ovarian tissue cryopreservation. Ovarian histology demonstrated no ovarian follicles.</div></div><div><h3>Comments</h3><div>This study demonstrated no follicles in either of the subjects with POI due to autoimmune oophoritis. It is possible that the absence of follicles represents generally worse outcomes of ovarian tissue cryopreservation for patients with autoimmune POI compared to other causes of POI. That said, little is known about the chronology of this condition, and earlier detection and recognition of autoimmune oophoritis might allow for improved fertility outcomes. Notably both of these patients were post-pubertal. It is unknown if fertility preservation outcomes for patients with autoimmune POI differ by pubertal status. More studies are needed to determine who are optimal candidates for ovarian tissue cryopreservation, especially for rare causes of POI like autoimmune oophoritis.</div></div>","PeriodicalId":16708,"journal":{"name":"Journal of pediatric and adolescent gynecology","volume":"38 2","pages":"Pages 249-250"},"PeriodicalIF":1.7,"publicationDate":"2025-02-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143520213","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-02-28DOI: 10.1016/j.jpag.2025.01.080
Salman Ali Jan , Laura Rekedal , Greta Magerowski , Diana Robillard , Margaret Griffith , Eleanor Parker
Background
Mucinous cystadenomas are among the most common ovarian neoplasms in child and adolescent patients, but giant ovarian cysts measuring >15cm remain rare. To date, no cases have been reported among transmasculine patients on hormone replacement therapy with testosterone. This case aims to provide insight on the preoperative and intra-operative considerations in this patient population.
Case
This 19-year-old transgender patient presented for gender-affirming mastectomy but was referred preoperatively to the emergency department for evaluation of new, rapid-onset abdominal distention. Imaging showed a 17 × 26 × 36cm septated, cystic pelvic mass suspected to be of ovarian origin. Serum tumor markers were normal, and a benign pelvic mass was suspected. Gender affirming hysterectomy and bilateral salpingo-oophorectomy were discussed pre-operatively, and the patient ultimately decided to preserve future fertility yet was amenable to staging for oncologic benefit in the event a malignancy was identified. Exploratory laparotomy was initiated with a 4cm midline vertical incision and the cystic mass was encountered upon peritoneal entry. A controlled drainage was attempted via needle aspiration following the application of skin glue, wound protector and retention suture (Figure 1). Given concern for minimal spillage of cyst contents intra-operatively, as well as difficulty accessing the multiple loculations within the large mass, the incision was ultimately extended to 10cm and the mass was decompressed via suction to a volume of 7850mL. The cyst was externalized (Figure 2) and found to originate from the left ovary, while the right adnexa were normal in appearance. A unilateral salpingo-oophorectomy was performed, and pathology was suspected to be benign on frozen section. The final pathology confirmed benign mucinous cystadenoma with associated mural granulation tissue and an endometriotic cyst. Cytologic analysis of peritoneal washings was negative for malignancy.
Comments
Large cystic ovarian tumors with loculated morphology require special surgical considerations. While minimally invasive approaches are generally preferred, we advise a modest approach to drainage unless imaging is unequivocal. We recommend shared preoperative decision-making that addresses the risk of borderline and malignant pathology, as well as the transgender patient's goals for gender affirmation and future fertility. For patients undergoing surgery that will delay other gender-affirming care, it is imperative to provide clear communication about how surgical management may affect time to care.
{"title":"47. Giant Mucinous Cystadenoma in a Young Transgender Man: Case Report","authors":"Salman Ali Jan , Laura Rekedal , Greta Magerowski , Diana Robillard , Margaret Griffith , Eleanor Parker","doi":"10.1016/j.jpag.2025.01.080","DOIUrl":"10.1016/j.jpag.2025.01.080","url":null,"abstract":"<div><h3>Background</h3><div>Mucinous cystadenomas are among the most common ovarian neoplasms in child and adolescent patients, but giant ovarian cysts measuring >15cm remain rare. To date, no cases have been reported among transmasculine patients on hormone replacement therapy with testosterone. This case aims to provide insight on the preoperative and intra-operative considerations in this patient population.</div></div><div><h3>Case</h3><div>This 19-year-old transgender patient presented for gender-affirming mastectomy but was referred preoperatively to the emergency department for evaluation of new, rapid-onset abdominal distention. Imaging showed a 17 × 26 × 36cm septated, cystic pelvic mass suspected to be of ovarian origin. Serum tumor markers were normal, and a benign pelvic mass was suspected. Gender affirming hysterectomy and bilateral salpingo-oophorectomy were discussed pre-operatively, and the patient ultimately decided to preserve future fertility yet was amenable to staging for oncologic benefit in the event a malignancy was identified. Exploratory laparotomy was initiated with a 4cm midline vertical incision and the cystic mass was encountered upon peritoneal entry. A controlled drainage was attempted via needle aspiration following the application of skin glue, wound protector and retention suture (Figure 1). Given concern for minimal spillage of cyst contents intra-operatively, as well as difficulty accessing the multiple loculations within the large mass, the incision was ultimately extended to 10cm and the mass was decompressed via suction to a volume of 7850mL. The cyst was externalized (Figure 2) and found to originate from the left ovary, while the right adnexa were normal in appearance. A unilateral salpingo-oophorectomy was performed, and pathology was suspected to be benign on frozen section. The final pathology confirmed benign mucinous cystadenoma with associated mural granulation tissue and an endometriotic cyst. Cytologic analysis of peritoneal washings was negative for malignancy.</div></div><div><h3>Comments</h3><div>Large cystic ovarian tumors with loculated morphology require special surgical considerations. While minimally invasive approaches are generally preferred, we advise a modest approach to drainage unless imaging is unequivocal. We recommend shared preoperative decision-making that addresses the risk of borderline and malignant pathology, as well as the transgender patient's goals for gender affirmation and future fertility. For patients undergoing surgery that will delay other gender-affirming care, it is imperative to provide clear communication about how surgical management may affect time to care.</div></div>","PeriodicalId":16708,"journal":{"name":"Journal of pediatric and adolescent gynecology","volume":"38 2","pages":"Page 251"},"PeriodicalIF":1.7,"publicationDate":"2025-02-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143520216","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-02-28DOI: 10.1016/j.jpag.2025.01.094
Nikita Sood , Erin E Barlow
Background
The prevalence of endometriosis in adolescent populations is difficult to ascertain, with some studies estimating that almost 40% of post-menarchal adolescents with pelvic pain have endometriosis. Reports of endometriosis in pre-menarchal patients are even more limited and highlight a still-evolving understanding of the pathogenesis of the disease. No prior documented case has described labial endometriosis in a pre-menarchal female.
Case
An 11 year old pre-menarchal patient presented to the emergency room (ER) with a tender vulvar mass. The mass had been present for several weeks and was increasingly painful. She was afebrile, had no associated symptoms, and denied recent trauma to the area. On exam, a 6 × 6 cm mass was noted on the right labia minora with mild tenderness to palpation but no surrounding erythema or swelling. The patient was noted to have Tanner Stage 2 breast and pubic hair development. The presumed diagnosis was a hematoma and she was discharged by the ER team with planned follow up with Pediatric and Adolescent Gynecology for consultation. The patient presented again 2.5 weeks later with increased pain and size of the labial mass. She continued to be afebrile and without associated symptoms. Labial ultrasound demonstrated a 5 × 4.5 × 3 cm multi-septated structure with concentric solid and vascular components. Pelvic MRI also showed a 4.9 × 4.5 × 5 cm multiseptated cystic structure. Presumed diagnosis was a super-infected hair follicle and plan was made for incision and drainage in the operating room. Grossly, the mass was noted to be well-encapsulated, consistent with an encapsulated hematoma with a cystic wall structure and septations. Due to proximity to the clitoral hood, a small amount of capsule was left in situ and a Penrose drain was left in place. The mass was sent to pathology which reported hemosiderin-laden macrophages and CD10 positive stromal cells suggestive of endometriosis.
Comments
This report presents the first case of labial endometriosis in a pre-menarchal patient. Vulvar/perineal endometriosis is generally rare, with other cases in post-menarchal adolescents typically associated with recent vulvar trauma not seen in this case. In general, endometriosis is considered to be a progressive disease associated with menses, which highlights the atypical nature of this case. Though multiple theories exist as to the pathogenesis of the disease, the theory of retrograde menstruation is widely accepted. This case, however, suggests that alternate theories such as coelomic metaplasia or mullerian embryonic rests may also contribute.
{"title":"61. Labial Endometriosis in a Pre-Menarchal Patient","authors":"Nikita Sood , Erin E Barlow","doi":"10.1016/j.jpag.2025.01.094","DOIUrl":"10.1016/j.jpag.2025.01.094","url":null,"abstract":"<div><h3>Background</h3><div>The prevalence of endometriosis in adolescent populations is difficult to ascertain, with some studies estimating that almost 40% of post-menarchal adolescents with pelvic pain have endometriosis. Reports of endometriosis in pre-menarchal patients are even more limited and highlight a still-evolving understanding of the pathogenesis of the disease. No prior documented case has described labial endometriosis in a pre-menarchal female.</div></div><div><h3>Case</h3><div>An 11 year old pre-menarchal patient presented to the emergency room (ER) with a tender vulvar mass. The mass had been present for several weeks and was increasingly painful. She was afebrile, had no associated symptoms, and denied recent trauma to the area. On exam, a 6 × 6 cm mass was noted on the right labia minora with mild tenderness to palpation but no surrounding erythema or swelling. The patient was noted to have Tanner Stage 2 breast and pubic hair development. The presumed diagnosis was a hematoma and she was discharged by the ER team with planned follow up with Pediatric and Adolescent Gynecology for consultation. The patient presented again 2.5 weeks later with increased pain and size of the labial mass. She continued to be afebrile and without associated symptoms. Labial ultrasound demonstrated a 5 × 4.5 × 3 cm multi-septated structure with concentric solid and vascular components. Pelvic MRI also showed a 4.9 × 4.5 × 5 cm multiseptated cystic structure. Presumed diagnosis was a super-infected hair follicle and plan was made for incision and drainage in the operating room. Grossly, the mass was noted to be well-encapsulated, consistent with an encapsulated hematoma with a cystic wall structure and septations. Due to proximity to the clitoral hood, a small amount of capsule was left in situ and a Penrose drain was left in place. The mass was sent to pathology which reported hemosiderin-laden macrophages and CD10 positive stromal cells suggestive of endometriosis.</div></div><div><h3>Comments</h3><div>This report presents the first case of labial endometriosis in a pre-menarchal patient. Vulvar/perineal endometriosis is generally rare, with other cases in post-menarchal adolescents typically associated with recent vulvar trauma not seen in this case. In general, endometriosis is considered to be a progressive disease associated with menses, which highlights the atypical nature of this case. Though multiple theories exist as to the pathogenesis of the disease, the theory of retrograde menstruation is widely accepted. This case, however, suggests that alternate theories such as coelomic metaplasia or mullerian embryonic rests may also contribute.</div></div>","PeriodicalId":16708,"journal":{"name":"Journal of pediatric and adolescent gynecology","volume":"38 2","pages":"Page 258"},"PeriodicalIF":1.7,"publicationDate":"2025-02-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143520617","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
We present a case of intra-abdominal tuberculosis and ovarian yolk sac tumor in an adolescent. It highlights the challenges of overlapping genetic, infectious, and autoimmune factors in adolescents and the importance of multidisciplinary care in patients with rare diagnoses and balancing treatment with long-term quality of life.
Case
A 17-year-old female presented to the emergency room with a one-week history of abdominal distension; imaging demonstrated a 4 cm cystic pelvic mass with carcinomatosis and large volume ascites. Tumor markers showed a normal alpha-fetoprotein (AFP) of < 0.8, borderline lactate dehydrogenase (LDH) of 292, and an elevated CA-125 of 400. The histopathology from diagnostic laparoscopy of the omentum, peritoneum, and ovaries showed necrotizing granulomatous inflammation without malignancy. The patient received a rheumatologic, infectious, and genetic workup; positive Acid-Fast Bacilli cultures confirmed mycobacterial tuberculosis infection. She completed rifampin, isoniazid, pyrazinamide, ethambutol therapy 6 months later. Somatic genetic testing revealed a Nucleotide Binding Oligomerization Domain 2 (NOD2) mutation, which may have contributed to granulomatous disease, although the clinical significance was uncertain. One year later, she presented with abdominal pain; imaging revealed a 12 cm pelvic mass with retroperitoneal lymphadenopathy. Tumor markers were elevated with AFP > 1150 and LDH 801, suggesting a malignant process. She experienced acute worsening pain, and CT confirmed spontaneous rupture of the mass. She underwent exploratory laparotomy, unilateral salpingoophorectomy, and omentectomy with gynecology oncology. Pathology revealed a FIGO Stage IIB yolk sac tumor involving the left ovary with abundant necrosis. Omentum, peritoneum, and additional biopsies showed reactive changes without tumor involvement. She initiated chemotherapy with Bleomycin, Carboplatin, and Etoposide, and underwent three cycles without complications. At follow-up with pediatric gynecology, she showed no evidence of premature ovarian insufficiency and started on Slynd for menstrual management and contraception.
Comments
This case highlights the complexities adolescents face when undergoing treatment for rare disease processes and genetic mutations of uncertain clinical significance. NOD2 may play a role in carcinogenesis as a tumor suppressor and is linked to mycobacterial infections. This case highlights the importance of ongoing research of rare disease presentations and genetic variants as well as the importance of multidisciplinary care in adolescents with rare diagnoses at risk for decreased ovarian function.
{"title":"2. A Dual Diagnosis: The Rare Connection Between Pelvic Tuberculosis and Yolk Sac Tumor","authors":"Kendall Bielak , Ellen Myers , Vasiliki Anemikos , Vrunda Patel","doi":"10.1016/j.jpag.2025.01.035","DOIUrl":"10.1016/j.jpag.2025.01.035","url":null,"abstract":"<div><h3>Background</h3><div>We present a case of intra-abdominal tuberculosis and ovarian yolk sac tumor in an adolescent. It highlights the challenges of overlapping genetic, infectious, and autoimmune factors in adolescents and the importance of multidisciplinary care in patients with rare diagnoses and balancing treatment with long-term quality of life.</div></div><div><h3>Case</h3><div>A 17-year-old female presented to the emergency room with a one-week history of abdominal distension; imaging demonstrated a 4 cm cystic pelvic mass with carcinomatosis and large volume ascites. Tumor markers showed a normal alpha-fetoprotein (AFP) of < 0.8, borderline lactate dehydrogenase (LDH) of 292, and an elevated CA-125 of 400. The histopathology from diagnostic laparoscopy of the omentum, peritoneum, and ovaries showed necrotizing granulomatous inflammation without malignancy. The patient received a rheumatologic, infectious, and genetic workup; positive Acid-Fast Bacilli cultures confirmed mycobacterial tuberculosis infection. She completed rifampin, isoniazid, pyrazinamide, ethambutol therapy 6 months later. Somatic genetic testing revealed a Nucleotide Binding Oligomerization Domain 2 (NOD2) mutation, which may have contributed to granulomatous disease, although the clinical significance was uncertain. One year later, she presented with abdominal pain; imaging revealed a 12 cm pelvic mass with retroperitoneal lymphadenopathy. Tumor markers were elevated with AFP > 1150 and LDH 801, suggesting a malignant process. She experienced acute worsening pain, and CT confirmed spontaneous rupture of the mass. She underwent exploratory laparotomy, unilateral salpingoophorectomy, and omentectomy with gynecology oncology. Pathology revealed a FIGO Stage IIB yolk sac tumor involving the left ovary with abundant necrosis. Omentum, peritoneum, and additional biopsies showed reactive changes without tumor involvement. She initiated chemotherapy with Bleomycin, Carboplatin, and Etoposide, and underwent three cycles without complications. At follow-up with pediatric gynecology, she showed no evidence of premature ovarian insufficiency and started on Slynd for menstrual management and contraception.</div></div><div><h3>Comments</h3><div>This case highlights the complexities adolescents face when undergoing treatment for rare disease processes and genetic mutations of uncertain clinical significance. NOD2 may play a role in carcinogenesis as a tumor suppressor and is linked to mycobacterial infections. This case highlights the importance of ongoing research of rare disease presentations and genetic variants as well as the importance of multidisciplinary care in adolescents with rare diagnoses at risk for decreased ovarian function.</div></div>","PeriodicalId":16708,"journal":{"name":"Journal of pediatric and adolescent gynecology","volume":"38 2","pages":"Page 230"},"PeriodicalIF":1.7,"publicationDate":"2025-02-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143519877","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-02-28DOI: 10.1016/j.jpag.2025.01.068
Leandra Cole , Elizabeth Boskey , Ava Scatoni , Jessica Shim
Background
Longitudinal vaginal septum (LVS) is a congenital anomaly that may be excised in adolescents with symptoms such as tampon difficulty. Several techniques for surgical excision exist, including excision with cautery or a bipolar electrosurgical instrument. Our study objective was to evaluate if any difference in blood loss or postoperative complications existed between excision with Bovie® cautery versus with the LigaSure™ vessel sealing device. We hypothesized that blood loss and complications were low with both surgical techniques.
Methods
This was a retrospective study of all patients who underwent LVS excision at a single pediatric tertiary care institution under the care of the Pediatric and Adolescent Gynecology division from 2012 to 2024. Patients either underwent excision using cautery or a vessel sealing device; the vaginal mucosa was oversewn after either technique. Demographics, clinical characteristics, estimated blood loss, and postoperative complications were abstracted from the medical chart. This study was approved by the Institutional Review Board.
Results
44 patients underwent excision of LVS during the study time period, mean age 17 years. The most common presenting symptom was tampon difficulty (52%) followed by dysmenorrhea (39%), and dyspareunia (9%). Eleven percent had no symptoms reported. The majority of patients (84%) had 2 cervices; 7 (16%) had 1 cervix. Thirty-two patients (73%) underwent cautery, 11 (25%) underwent vessel sealing, and 1 (2%) both. Mean blood loss was not significantly different with both techniques (13 vs. 4 ml, p=.10). Postoperative complications were rare. 7 (16%) patients presented to the emergency department (ED), average 12 days post-surgery. Of these 7 patients, 3 presented for vaginal bleeding, 1 for vaginal discharge, 3 for symptoms deemed unrelated to surgery by the ED. One patient underwent emergent revision due to an estimated 400 ml blood loss prior in the ED, with 100 ml blood evacuated in the OR and 10 ml active blood loss with the revision. Of the 3 patients who presented to the ED for bleeding concerns, 2 underwent cautery, and 1 underwent vessel sealing device.
Conclusions
The overall blood loss at time of surgery for LVS was low and did not significantly differ by surgical technique, although there was a trend towards less blood loss with vessel sealing that might become significant with a larger sample. Postoperative complications were also rare with either method, though 2 patients (4%) experienced unanticipated problems – one significant interoperative tissue friability and the other bacterial vaginosis. Surgeons may consider using several techniques to safely perform LVS excision.
{"title":"35. Estimated blood loss and safety by technique for longitudinal vaginal septum excision","authors":"Leandra Cole , Elizabeth Boskey , Ava Scatoni , Jessica Shim","doi":"10.1016/j.jpag.2025.01.068","DOIUrl":"10.1016/j.jpag.2025.01.068","url":null,"abstract":"<div><h3>Background</h3><div>Longitudinal vaginal septum (LVS) is a congenital anomaly that may be excised in adolescents with symptoms such as tampon difficulty. Several techniques for surgical excision exist, including excision with cautery or a bipolar electrosurgical instrument. Our study objective was to evaluate if any difference in blood loss or postoperative complications existed between excision with Bovie® cautery versus with the LigaSure™ vessel sealing device. We hypothesized that blood loss and complications were low with both surgical techniques.</div></div><div><h3>Methods</h3><div>This was a retrospective study of all patients who underwent LVS excision at a single pediatric tertiary care institution under the care of the Pediatric and Adolescent Gynecology division from 2012 to 2024. Patients either underwent excision using cautery or a vessel sealing device; the vaginal mucosa was oversewn after either technique. Demographics, clinical characteristics, estimated blood loss, and postoperative complications were abstracted from the medical chart. This study was approved by the Institutional Review Board.</div></div><div><h3>Results</h3><div>44 patients underwent excision of LVS during the study time period, mean age 17 years. The most common presenting symptom was tampon difficulty (52%) followed by dysmenorrhea (39%), and dyspareunia (9%). Eleven percent had no symptoms reported. The majority of patients (84%) had 2 cervices; 7 (16%) had 1 cervix. Thirty-two patients (73%) underwent cautery, 11 (25%) underwent vessel sealing, and 1 (2%) both. Mean blood loss was not significantly different with both techniques (13 vs. 4 ml, p=.10). Postoperative complications were rare. 7 (16%) patients presented to the emergency department (ED), average 12 days post-surgery. Of these 7 patients, 3 presented for vaginal bleeding, 1 for vaginal discharge, 3 for symptoms deemed unrelated to surgery by the ED. One patient underwent emergent revision due to an estimated 400 ml blood loss prior in the ED, with 100 ml blood evacuated in the OR and 10 ml active blood loss with the revision. Of the 3 patients who presented to the ED for bleeding concerns, 2 underwent cautery, and 1 underwent vessel sealing device.</div></div><div><h3>Conclusions</h3><div>The overall blood loss at time of surgery for LVS was low and did not significantly differ by surgical technique, although there was a trend towards less blood loss with vessel sealing that might become significant with a larger sample. Postoperative complications were also rare with either method, though 2 patients (4%) experienced unanticipated problems – one significant interoperative tissue friability and the other bacterial vaginosis. Surgeons may consider using several techniques to safely perform LVS excision.</div></div>","PeriodicalId":16708,"journal":{"name":"Journal of pediatric and adolescent gynecology","volume":"38 2","pages":"Pages 246-247"},"PeriodicalIF":1.7,"publicationDate":"2025-02-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143519882","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-02-28DOI: 10.1016/j.jpag.2025.01.042
Samantha Lowe , Sara Vincent , Megan Sumida , Jennifer Dietrich
Background
Mayer-Rokitansky-Küster-Hauser Syndrome (MRKH) is a congenital anomaly in which the Mullerian duct is underdeveloped, leading to absence of the uterus and vagina. Limited research exists describing abdominopelvic pain (AP) in women with MRKH, with most studies focusing solely on pain related to sexual function. Our objectives were to: 1) estimate the prevalence of AP in women with MRKH, 2) compare the prevalence of AP in women with anatomic variations and extragenital anomalies in MRKH, and 3) estimate the prevalence of coexisting pain disorders (CPD) in women with MRKH.
Methods
We conducted an IRB-approved retrospective cohort study of patients with MRKH seen at an academic center between 8/20/2014 – 8/20/2024. Patients were identified using ICD-10 codes for agenesis of uterus or agenesis of vagina. Inclusion criteria were as follows: 1) patients with a confirmed diagnosis of MRKH, as assessed by post-pubertal MRI or surgical findings. Patients with abnormal karyotypes were excluded. Data included demographics, MRKH type and associated extragenital malformations, presence of uterine remnants (UR), pelvic pathology, presence and quality of AP, and CPD. Persistent AP was defined as pain lasting more than six months or occurring at intervals which spanned over a period longer than six months. Cyclic AP was defined as pain occurring at monthly intervals for any time period. Persistent and cyclic AP were not mutually exclusive. Chi-square and t-test statistics were performed.
Results
117 patients were identified and then subcategorized into those with UR (N=63) and those without UR (N=54) and separately evaluated by MRKH type (Type 1 N=65, Type 2 N=52). Overall, 43 patients (36.8%) reported persistent AP and 24 patients (20.5%) reported cyclic AP. Presence of UR was associated with both the presence of persistent AP (p = 0.008) and cyclic AP (p = 0.001) (Table 1). There was no association between the type of MRKH and the presence of persistent AP (p = 0.732) or cyclic AP (p = 0.282) (Table 2). 28 (23.9%) patients were diagnosed with or treated for one or more CPD, with migraine headache (21/117) and IBS (8/117) being the most common. Secondarily, MRKH patients had a wide range of pelvic pathologies, including ovarian cysts (14/117), paratubal cysts (15/117), and endometriosis (3/117). Endometriosis may be underestimated due to need for surgical diagnosis.
Conclusions
In this cohort of women with MRKH, over a third of patients reported persistent AP. Persistent AP and cyclic AP were associated with the presence of UR and MRKH types with AP were similar. The limited number of MRKH patients with CPD may reflect overlapping diagnoses.
{"title":"9. Abdominopelvic Pain and Coexisting Pain Disorders in Patients with Mayer-Rokitansky-Küster-Hauser Syndrome","authors":"Samantha Lowe , Sara Vincent , Megan Sumida , Jennifer Dietrich","doi":"10.1016/j.jpag.2025.01.042","DOIUrl":"10.1016/j.jpag.2025.01.042","url":null,"abstract":"<div><h3>Background</h3><div>Mayer-Rokitansky-Küster-Hauser Syndrome (MRKH) is a congenital anomaly in which the Mullerian duct is underdeveloped, leading to absence of the uterus and vagina. Limited research exists describing abdominopelvic pain (AP) in women with MRKH, with most studies focusing solely on pain related to sexual function. Our objectives were to: 1) estimate the prevalence of AP in women with MRKH, 2) compare the prevalence of AP in women with anatomic variations and extragenital anomalies in MRKH, and 3) estimate the prevalence of coexisting pain disorders (CPD) in women with MRKH.</div></div><div><h3>Methods</h3><div>We conducted an IRB-approved retrospective cohort study of patients with MRKH seen at an academic center between 8/20/2014 – 8/20/2024. Patients were identified using ICD-10 codes for agenesis of uterus or agenesis of vagina. Inclusion criteria were as follows: 1) patients with a confirmed diagnosis of MRKH, as assessed by post-pubertal MRI or surgical findings. Patients with abnormal karyotypes were excluded. Data included demographics, MRKH type and associated extragenital malformations, presence of uterine remnants (UR), pelvic pathology, presence and quality of AP, and CPD. Persistent AP was defined as pain lasting more than six months or occurring at intervals which spanned over a period longer than six months. Cyclic AP was defined as pain occurring at monthly intervals for any time period. Persistent and cyclic AP were not mutually exclusive. Chi-square and t-test statistics were performed.</div></div><div><h3>Results</h3><div>117 patients were identified and then subcategorized into those with UR (N=63) and those without UR (N=54) and separately evaluated by MRKH type (Type 1 N=65, Type 2 N=52). Overall, 43 patients (36.8%) reported persistent AP and 24 patients (20.5%) reported cyclic AP. Presence of UR was associated with both the presence of persistent AP (p = 0.008) and cyclic AP (p = 0.001) (Table 1). There was no association between the type of MRKH and the presence of persistent AP (p = 0.732) or cyclic AP (p = 0.282) (Table 2). 28 (23.9%) patients were diagnosed with or treated for one or more CPD, with migraine headache (21/117) and IBS (8/117) being the most common. Secondarily, MRKH patients had a wide range of pelvic pathologies, including ovarian cysts (14/117), paratubal cysts (15/117), and endometriosis (3/117). Endometriosis may be underestimated due to need for surgical diagnosis.</div></div><div><h3>Conclusions</h3><div>In this cohort of women with MRKH, over a third of patients reported persistent AP. Persistent AP and cyclic AP were associated with the presence of UR and MRKH types with AP were similar. The limited number of MRKH patients with CPD may reflect overlapping diagnoses.</div></div>","PeriodicalId":16708,"journal":{"name":"Journal of pediatric and adolescent gynecology","volume":"38 2","pages":"Pages 233-234"},"PeriodicalIF":1.7,"publicationDate":"2025-02-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143519933","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-02-28DOI: 10.1016/j.jpag.2025.01.028
Nicole Smith , Justin Lim , Carolyn Ziegler , Lisa Allen , Andrea Simpson , Sari Kives
<div><h3>Background</h3><div>Ovarian dermoid cysts are benign germ-cell tumors commonly found in pediatric and adolescent patients. Laparoscopic ovarian cystectomy has become the preferred surgical approach over laparotomy due to its benefits of shorter hospital stay, reduced post-operative pain, and quicker recovery. However, there remains concern for higher rates of cyst rupture (and possible peritonitis) and future dermoid cyst recurrence, when compared to laparotomy. This systematic review and meta-analysis evaluates the existing evidence on these surgical methods to assess whether the shift to laparoscopy has led to these unintended outcomes.</div></div><div><h3>Methods</h3><div>A comprehensive literature search in five academic medical databases was conducted with an information specialist, using keywords and MeSH terms for ovarian dermoid cysts, laparotomy, laparoscopy, or minimally invasive surgery in children, from database inception to August 29, 2024. This study was exempt from Research Ethics Board (REB) approval. Randomized, quasi-randomized trials, and prospective/retrospective cohort studies were eligible. Two independent authors reviewed titles/abstracts, full texts, and extracted data, resolving conflicts with a third author as required. Risk of bias was assessed with appropriate tools. A meta-analysis using risk ratios with 95% confidence intervals was performed with a Mantel-Haenszel fixed-effect model. Heterogeneity among studies was assessed by I², with I2 > 50% interpreted as high degree of heterogeneity requiring random-effects modelling.</div></div><div><h3>Results</h3><div>1021 title/abstracts were reviewed, with six studies eligible for inclusion (2000 – 2022; Canada, United States, Poland). In total, 492 (58%) patients underwent laparoscopy and 353 (42%) underwent laparotomy. For meta-analysis, five studies were included for cyst rupture and four studies were included for dermoid cyst recurrence. Random-effect meta-analysis (I2=58%) showed a significantly higher risk of cyst rupture with laparoscopy (RR = 2.47, 95% CI 1.40-4.37). Four studies reported on peritonitis, with no cases observed. Fixed-effect meta-analysis (I2 =5%) found no significant difference in dermoid cyst recurrence between laparoscopy and laparotomy (RR = 0.92, 95% CI 0.41-2.08).</div></div><div><h3>Conclusions</h3><div>Laparoscopy remains the preferred approach for the surgical management of ovarian dermoid cysts in children. Despite a higher risk of intraoperative cyst rupture, our systematic review highlights that</div><div>subsequent post-operative peritonitis was not clinically observed. Further, dermoid cyst recurrence rates remained similar between laparoscopy and laparotomy, further encouraging the use of laparoscopy when possible. Limitations of our systematic review include the limited number of included studies, all of which were observational studies with small sample sizes, and the presence of unaccounted confounders such as cyst size,
{"title":"15. Surgical management of ovarian dermoid cysts in children: a systematic review and meta-analysis","authors":"Nicole Smith , Justin Lim , Carolyn Ziegler , Lisa Allen , Andrea Simpson , Sari Kives","doi":"10.1016/j.jpag.2025.01.028","DOIUrl":"10.1016/j.jpag.2025.01.028","url":null,"abstract":"<div><h3>Background</h3><div>Ovarian dermoid cysts are benign germ-cell tumors commonly found in pediatric and adolescent patients. Laparoscopic ovarian cystectomy has become the preferred surgical approach over laparotomy due to its benefits of shorter hospital stay, reduced post-operative pain, and quicker recovery. However, there remains concern for higher rates of cyst rupture (and possible peritonitis) and future dermoid cyst recurrence, when compared to laparotomy. This systematic review and meta-analysis evaluates the existing evidence on these surgical methods to assess whether the shift to laparoscopy has led to these unintended outcomes.</div></div><div><h3>Methods</h3><div>A comprehensive literature search in five academic medical databases was conducted with an information specialist, using keywords and MeSH terms for ovarian dermoid cysts, laparotomy, laparoscopy, or minimally invasive surgery in children, from database inception to August 29, 2024. This study was exempt from Research Ethics Board (REB) approval. Randomized, quasi-randomized trials, and prospective/retrospective cohort studies were eligible. Two independent authors reviewed titles/abstracts, full texts, and extracted data, resolving conflicts with a third author as required. Risk of bias was assessed with appropriate tools. A meta-analysis using risk ratios with 95% confidence intervals was performed with a Mantel-Haenszel fixed-effect model. Heterogeneity among studies was assessed by I², with I2 > 50% interpreted as high degree of heterogeneity requiring random-effects modelling.</div></div><div><h3>Results</h3><div>1021 title/abstracts were reviewed, with six studies eligible for inclusion (2000 – 2022; Canada, United States, Poland). In total, 492 (58%) patients underwent laparoscopy and 353 (42%) underwent laparotomy. For meta-analysis, five studies were included for cyst rupture and four studies were included for dermoid cyst recurrence. Random-effect meta-analysis (I2=58%) showed a significantly higher risk of cyst rupture with laparoscopy (RR = 2.47, 95% CI 1.40-4.37). Four studies reported on peritonitis, with no cases observed. Fixed-effect meta-analysis (I2 =5%) found no significant difference in dermoid cyst recurrence between laparoscopy and laparotomy (RR = 0.92, 95% CI 0.41-2.08).</div></div><div><h3>Conclusions</h3><div>Laparoscopy remains the preferred approach for the surgical management of ovarian dermoid cysts in children. Despite a higher risk of intraoperative cyst rupture, our systematic review highlights that</div><div>subsequent post-operative peritonitis was not clinically observed. Further, dermoid cyst recurrence rates remained similar between laparoscopy and laparotomy, further encouraging the use of laparoscopy when possible. Limitations of our systematic review include the limited number of included studies, all of which were observational studies with small sample sizes, and the presence of unaccounted confounders such as cyst size,","PeriodicalId":16708,"journal":{"name":"Journal of pediatric and adolescent gynecology","volume":"38 2","pages":"Pages 228-229"},"PeriodicalIF":1.7,"publicationDate":"2025-02-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143520011","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2025-02-28DOI: 10.1016/j.jpag.2025.01.047
Rabab Isa , Noor Zwayne
Background
Hematometra and hematocolpos are uncommon conditions involving fluid accumulation in the uterus and vagina, often due to primary or secondary outflow obstruction. Their occurrence in young patients, particularly those with hematologic disorders, poses diagnostic challenges and can lead to significant morbidity. This case report aims to enhance awareness of acquired hematometrocolpos resulting from graft versus host disease (GvHD) in a post-hematopoietic stem cell transplant patient.
Case
A 17-year-old female with a history of acute myeloid leukemia presented with severe pelvic pain after treatment for cancer, GvHD, and premature ovarian insufficiency. Evaluation revealed significant hematocolpos and a distended uterus to 16 cm. Diagnostic laparoscopy and hysteroscopy identified atrophic vaginal and cervical tissues with substantial adhesions. Approximately 300 cc of dark blood was drained, and a levonorgestrel-releasing intrauterine device was placed. Despite initial improvement, the patient experienced recurrent hematocolpos requiring repeat surgical decompression. She is currently doing well with vaginal estrogen and dilators to maintain patency.
Comments
This case highlights consideration of hematometrocolpos in adolescents with complex medical histories, particularly those undergoing treatment for hematologic malignancies. Early recognition and intervention can prevent long-term complications and improve patient outcomes. Continued education on the implications of GvHD on the female genital tract is essential for optimizing care in this vulnerable population.
{"title":"14. An Unusual Case of Hematometrocolpos in a Young Patient with Acute Myeloid Leukemia","authors":"Rabab Isa , Noor Zwayne","doi":"10.1016/j.jpag.2025.01.047","DOIUrl":"10.1016/j.jpag.2025.01.047","url":null,"abstract":"<div><h3>Background</h3><div>Hematometra and hematocolpos are uncommon conditions involving fluid accumulation in the uterus and vagina, often due to primary or secondary outflow obstruction. Their occurrence in young patients, particularly those with hematologic disorders, poses diagnostic challenges and can lead to significant morbidity. This case report aims to enhance awareness of acquired hematometrocolpos resulting from graft versus host disease (GvHD) in a post-hematopoietic stem cell transplant patient.</div></div><div><h3>Case</h3><div>A 17-year-old female with a history of acute myeloid leukemia presented with severe pelvic pain after treatment for cancer, GvHD, and premature ovarian insufficiency. Evaluation revealed significant hematocolpos and a distended uterus to 16 cm. Diagnostic laparoscopy and hysteroscopy identified atrophic vaginal and cervical tissues with substantial adhesions. Approximately 300 cc of dark blood was drained, and a levonorgestrel-releasing intrauterine device was placed. Despite initial improvement, the patient experienced recurrent hematocolpos requiring repeat surgical decompression. She is currently doing well with vaginal estrogen and dilators to maintain patency.</div></div><div><h3>Comments</h3><div>This case highlights consideration of hematometrocolpos in adolescents with complex medical histories, particularly those undergoing treatment for hematologic malignancies. Early recognition and intervention can prevent long-term complications and improve patient outcomes. Continued education on the implications of GvHD on the female genital tract is essential for optimizing care in this vulnerable population.</div></div>","PeriodicalId":16708,"journal":{"name":"Journal of pediatric and adolescent gynecology","volume":"38 2","pages":"Pages 235-236"},"PeriodicalIF":1.7,"publicationDate":"2025-02-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143520016","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Adolescent and young adult cancer survivors are at risk of infertility due to the gonadotoxic effects of treatment. Although ovarian function testing (OFT) is an essential aspect of fertility care, there is currently no standard of care for monitoring reserve after gonadotoxic therapy. At our institution, we measure AMH and FSH at 12-, 18-, and 24-months post-therapy for early detection of DOR (diminished ovarian reserve) and POI (premature ovarian insufficiency) to avoid delays in referral for assisted reproductive technologies (ART). The primary objective of this study is to assess whether a novel ovarian reserve monitoring algorithm enables early detection of DOR and POI after gonadotoxic therapy to improve reproductive outcomes.
Methods
In this IRB-approved observational cohort study at University of Colorado Hospital, we evaluated the algorithm to (1) identify survivors at risk of DOR and POI due to gonadotoxic therapy and (2) minimize time from treatment to ART. Group A (n=106) consisted of a retrospective cohort of survivors who completed therapy between October 1, 2016, and September 31, 2019, with AMH and FSH evaluated at 12, 18, and 24 months post-treatment completion (n=27). Group B (n=418) included survivors who completed cancer therapy after September 31, 2019, assessed prospectively at similar time points (n=64). AMH and FSH values, along with time since treatment, were analyzed to determine if the algorithm enables early detection of DOR before POI. Ovarian status was categorized as normal, diminished, impending POI, or POI. Time from treatment to diagnosis of DOR and POI was compared between the prospective group and historical controls using the log-rank test.
Results
Demographics between groups A and B were similar. OFT within 36 months after completing cancer treatment was performed in only 17.4% (91/524) of patients, and AMH was rarely tested before 2019. At the 24-month follow-up, the prospective group had a higher prevalence of normal ovarian reserve (44.3% [27/61] vs. 17% [4/23], p=0.05) compared to the retrospective group. Time from treatment to diagnosis of DOR or POI was shorter in Group B, although not significantly due to the small sample size (8.2 months in group A [n=19] vs. 6.1 months in group B [n=24], p=0.7).
Conclusions
Results suggests that patients followed under the fertility preservation program since 2019 have received more frequent monitoring and earlier OFT with increased use of AMH testing to detect changes before significant decline. The ovarian reserve monitoring algorithm allows early detection of DOR and POI after gonadotoxic therapy, potentially improving reproductive outcomes.
{"title":"1. A Novel Ovarian Reserve Monitoring Algorithm for Patients at Risk of Ovarian Injury from Gonadotoxic Therapy","authors":"Guluzar Turan , Victoria Kennerley , Marie-Teresa Colbert , Marcy Hoath , Kamilah Abdur-Rashid , Mikayla Ecker , Mary Sammel , Leslie Appiah","doi":"10.1016/j.jpag.2025.01.013","DOIUrl":"10.1016/j.jpag.2025.01.013","url":null,"abstract":"<div><h3>Background</h3><div>Adolescent and young adult cancer survivors are at risk of infertility due to the gonadotoxic effects of treatment. Although ovarian function testing (OFT) is an essential aspect of fertility care, there is currently no standard of care for monitoring reserve after gonadotoxic therapy. At our institution, we measure AMH and FSH at 12-, 18-, and 24-months post-therapy for early detection of DOR (diminished ovarian reserve) and POI (premature ovarian insufficiency) to avoid delays in referral for assisted reproductive technologies (ART). The primary objective of this study is to assess whether a novel ovarian reserve monitoring algorithm enables early detection of DOR and POI after gonadotoxic therapy to improve reproductive outcomes.</div></div><div><h3>Methods</h3><div>In this IRB-approved observational cohort study at University of Colorado Hospital, we evaluated the algorithm to (1) identify survivors at risk of DOR and POI due to gonadotoxic therapy and (2) minimize time from treatment to ART. Group A (n=106) consisted of a retrospective cohort of survivors who completed therapy between October 1, 2016, and September 31, 2019, with AMH and FSH evaluated at 12, 18, and 24 months post-treatment completion (n=27). Group B (n=418) included survivors who completed cancer therapy after September 31, 2019, assessed prospectively at similar time points (n=64). AMH and FSH values, along with time since treatment, were analyzed to determine if the algorithm enables early detection of DOR before POI. Ovarian status was categorized as normal, diminished, impending POI, or POI. Time from treatment to diagnosis of DOR and POI was compared between the prospective group and historical controls using the log-rank test.</div></div><div><h3>Results</h3><div>Demographics between groups A and B were similar. OFT within 36 months after completing cancer treatment was performed in only 17.4% (91/524) of patients, and AMH was rarely tested before 2019. At the 24-month follow-up, the prospective group had a higher prevalence of normal ovarian reserve (44.3% [27/61] vs. 17% [4/23], p=0.05) compared to the retrospective group. Time from treatment to diagnosis of DOR or POI was shorter in Group B, although not significantly due to the small sample size (8.2 months in group A [n=19] vs. 6.1 months in group B [n=24], p=0.7).</div></div><div><h3>Conclusions</h3><div>Results suggests that patients followed under the fertility preservation program since 2019 have received more frequent monitoring and earlier OFT with increased use of AMH testing to detect changes before significant decline. The ovarian reserve monitoring algorithm allows early detection of DOR and POI after gonadotoxic therapy, potentially improving reproductive outcomes.</div></div>","PeriodicalId":16708,"journal":{"name":"Journal of pediatric and adolescent gynecology","volume":"38 2","pages":"Page 219"},"PeriodicalIF":1.7,"publicationDate":"2025-02-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143520095","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Background: Non-gestational ovarian choriocarcinoma (NGOC) is extremely rare, particularly in the pediatric population. Few case reports have discussed NGOC with precocious puberty and mentioned both tumor markers (β-hCG, AFP, CA125) and the dynamics of gonadotropic and gonadal hormones.
Case: A 7-year-old girl presented with early puberty and an abdominal mass. Laboratory tests revealed elevated estradiol, β-hCG, and CA125 levels, with suppressed LH and FSH. She was diagnosed with ovarian choriocarcinoma and precocious puberty. Following tumor resection, her hormone levels normalized and symptoms resolved.
Conclusion: Pediatric ovarian choriocarcinomas are rare and may present with precocious puberty. Comprehensive endocrine evaluation, including serum hormone assays, is essential for diagnosis. Hormonal profiles in pediatric patients may differ from those in adults, with estradiol levels often being markedly elevated compared to age-specific reference ranges, and progesterone levels being relatively low.
{"title":"Nongestational Ovarian Choriocarcinoma with Precocious Puberty in a 7-Year-Old Girl: A rare case report and literature review.","authors":"Kako Ono, Reina Hoshi, Takayuki Hirano, Yosuke Watanabe, Shumpei Goto, Takashi Hosokawa, Shuichiro Uehara","doi":"10.1016/j.jpag.2025.02.007","DOIUrl":"https://doi.org/10.1016/j.jpag.2025.02.007","url":null,"abstract":"<p><strong>Background: </strong>Non-gestational ovarian choriocarcinoma (NGOC) is extremely rare, particularly in the pediatric population. Few case reports have discussed NGOC with precocious puberty and mentioned both tumor markers (β-hCG, AFP, CA125) and the dynamics of gonadotropic and gonadal hormones.</p><p><strong>Case: </strong>A 7-year-old girl presented with early puberty and an abdominal mass. Laboratory tests revealed elevated estradiol, β-hCG, and CA125 levels, with suppressed LH and FSH. She was diagnosed with ovarian choriocarcinoma and precocious puberty. Following tumor resection, her hormone levels normalized and symptoms resolved.</p><p><strong>Conclusion: </strong>Pediatric ovarian choriocarcinomas are rare and may present with precocious puberty. Comprehensive endocrine evaluation, including serum hormone assays, is essential for diagnosis. Hormonal profiles in pediatric patients may differ from those in adults, with estradiol levels often being markedly elevated compared to age-specific reference ranges, and progesterone levels being relatively low.</p>","PeriodicalId":16708,"journal":{"name":"Journal of pediatric and adolescent gynecology","volume":" ","pages":""},"PeriodicalIF":1.7,"publicationDate":"2025-02-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143531542","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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