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1.
BMC Womens Health ; 24(1): 300, 2024 May 20.
Article in English | MEDLINE | ID: mdl-38769573

ABSTRACT

BACKGROUND: Concomitant invasive ovarian mucinous adenocarcinoma, unilateral renal agenesis and bicornuate uterus is a rare combination. Unilateral renal agenesis has been associated with genital anomalies, such as unicornuate and bicornuate uterus. Furthermore, a wealth of studies has reported the association between unicornuate uterus and ovarian anomalies, such as the absence of an ovary or ectopic ovaries, but rarely has there been a combination of the three to the best of our knowledge. The present case report is the first case presentation with a combination of the three syndromes: ovarian mucinous tumor, unilateral renal agenesis, and bicornuate uterus. CASE PRESENTATION: We report the case of a 17-year-old who presented with abdominal distension. On examination, a CT scan revealed a large multicystic abdominal mass on the right side, with an absence of the right kidney while the left kidney was normal in size, appearance, and position. Intraoperatively, massive blood-stained ascitic fluid was evacuated. Additionally, a large whitish polycystic intra-abdominal mass with mucus-like materials and solid areas was attached to the midpoint of the colon and the right ovary, with visible metastasis to the omentum. The uterus was bicornuate. The mass and omentum were taken for histopathology and a diagnosis of invasive ovarian mucinous cystadenocarcinoma with metastasis to the colon and omentum was made after a pathological report. CONCLUSIONS: The presence of these conditions in the same individual could potentially complicate medical management and fertility considerations. Thus, a need for a multidisciplinary medical team, including gynecologists, urologists, and oncologists, to address their unique needs and provide appropriate treatment and guidance. Further research and case studies are needed to better understand the possible association and implications of these rare co-occurring conditions.


Subject(s)
Adenocarcinoma, Mucinous , Bicornuate Uterus , Ovarian Neoplasms , Uterus , Adolescent , Female , Humans , Adenocarcinoma, Mucinous/complications , Adenocarcinoma, Mucinous/diagnosis , Adenocarcinoma, Mucinous/pathology , Bicornuate Uterus/complications , Congenital Abnormalities , Kidney/abnormalities , Kidney/pathology , Ovarian Neoplasms/complications , Ovarian Neoplasms/pathology , Solitary Kidney/complications , Tomography, X-Ray Computed , Uterus/abnormalities , Uterus/pathology
2.
Cureus ; 16(4): e58351, 2024 Apr.
Article in English | MEDLINE | ID: mdl-38756287

ABSTRACT

The presentation of a bicornuate uterus may include miscarriages and menstrual abnormalities. The diagnosis could be in an incident of caesarean delivery, miscarriage or hysteroscopy. The possibility of misdiagnosis to an ectopic pregnancy is real. There are sonographical similarities between a pregnant horn of a bicornuate uterus and an ectopic pregnancy. We present in this article a case of interstitial pregnancy in a woman with a bicornuate uterus simulating symptoms of miscarriage. Congenital abnormalities necessitate the availability of the best diagnostic tools at the disposal of the medical practitioners. Ultrasound scan is an important aid for practitioners to choose the best therapeutic approach.

3.
Article in English | MEDLINE | ID: mdl-38693698

ABSTRACT

INTRODUCTION: Uterus transplantation has revolutionized reproductive medicine for women with absolute uterine factor infertility, resulting in more than 40 reported successful live births worldwide to date. Small animal models are pivotal to refine this surgical and immunological challenging procedure aiming to enhance safety for both the mother and the child. MATERIAL AND METHODS: We established a syngeneic bicornuate uterus transplantation model in young female Lewis rats. All surgical procedures were conducted by an experienced and skilled microsurgeon who organized the learning process into multiple structured steps. Animals underwent meticulous preoperative preparation and postoperative care. Transplant success was monitored by sequential biopsies, monitoring graft viability and documenting histological changes long-term. RESULTS: Bicornuate uterus transplantation were successfully established achieving an over 70% graft survival rate with the passage of time. The bicornuate model demonstrated safety and feasibility, yielding outcomes comparable to the unicornuate model in terms of ischemia times and complications. Longitudinal biopsies were well-tolerated, enabling comprehensive monitoring throughout the study. CONCLUSIONS: Our novel bicornuate rat uterus transplantation model provides a distinctive opportunity for sequential biopsies at various intervals after transplantation and, therefore, comprehensive monitoring of graft health, viability, and identification of potential signs of rejection. Furthermore, this model allows for different interventions in each horn for comparative studies without interobserver differences contrary to the established unicornuate model. By closely replicating the clinical setting, this model stands as a valuable tool for ongoing research in the field of uterus transplantation, promoting further innovation and deeper insights into the intricacies of the uterus transplant procedure.

4.
Clin Case Rep ; 12(3): e8661, 2024 Mar.
Article in English | MEDLINE | ID: mdl-38487645

ABSTRACT

Key Clinical Message: OHVIRA syndrome can be misdiagnosed due to its rarity, resulting in the need for more invasive interventions than vaginoscopy. Also, delayed diagnosis of OHVIRA syndrome can affect patient's quality of life by leading to chronic gynecological diseases such as endometriosis and pelvic inflammatory disease. Abstract: Obstructive hemivagina and ipsilateral renal agenesis (OHVIRA) syndrome is one of the infrequent congenital Mullerian duct anomalies characterized by obstructed hemivagina and ipsilateral renal agenesis. This study presents a 20-year-old virgin female who was diagnosed with OHVIRA syndrome and treated by vaginoscopy using the hymen preservation technique. Also, she was misdiagnosed with non-communicating rudimentary uterine horn 4 years ago. Late or misdiagnosis of OHVIRA syndrome can affect fertility and pregnancy outcomes. Therefore, early diagnosis and management are crucial. OHVIRA syndrome's misdiagnosis is possible with other Mullerian duct anomalies, such as a rudimentary uterine horn. Also, patients with misdiagnosis undergo unnecessary interventions.

5.
Int Med Case Rep J ; 17: 181-185, 2024.
Article in English | MEDLINE | ID: mdl-38524802

ABSTRACT

Background: Primary rupture of an unscarred uterus is rare. Spontaneous rupture of an unscarred bicornuate uterus is a life-threatening obstetric emergency with high morbidity and mortality in the mother and fetus; however, it most commonly occurs in the first trimester of pregnancy. Case: A 20-year-old primigravid woman at 22 weeks of gestation, with no prior surgery, presented with severe abdominal pain, anemia, and hemodynamic instability. With a preoperative diagnosis of uterine rupture, she was transfused with three units of cross-matched whole blood and underwent emergency laparotomy. Intraoperative findings showed a ruptured bicornuate uterus and a dead fetus in the abdomen with huge hemoperitoneum. Postoperative recovery was smooth, and the patient was discharged after being counselled on family planning and subsequent pregnancy. Conclusion: A bicornuate uterus may be an independent risk factor for uterine rupture, which can occur in primigravid women at any stage of pregnancy. Each obstetrician should have a high index of suspicion for a rare condition like ruptured bicornuate uterus, especially for a pregnant woman presenting with acute abdominal pain and hemodynamic instability. Early ultrasonography plays a key role in the evaluation, follow-up, and management of these patients.

6.
Fertil Steril ; 121(5): 887-889, 2024 May.
Article in English | MEDLINE | ID: mdl-38316208

ABSTRACT

OBJECTIVE: To demonstrate the surgical techniques for improving safety in robotic-assisted abdominal cerclage in patients with bicornuate uteri complicated by recurrent pregnancy loss and cervical insufficiency. DESIGN: Stepwise demonstration with narrated video footage. SETTING: An academic tertiary care hospital. PATIENTS: Our patient is a 22-year-old G2P0020 with a history of recurrent pregnancy loss. During her first pregnancy, she was asymptomatic until 19 weeks and delivered because of a preterm premature rupture of membranes. A transvaginal cerclage was performed for her second pregnancy at 14 weeks, which ended at 16 weeks because of preterm premature rupture of membranes. The final magnetic resonance imaging report noted a "bicorporeal uterus with duplication of the uterine body, resulting in two markedly divergent uterine horns that are fused at the isthmus... unlike a typical didelphic uterus, a single, non septated cervix is noted, which shows normal appearances, measuring 3.8 cm in length." Given her history of a uterine anomaly and recurrent pregnancy loss in the absence of other biochemical factors, her maternal-fetal medicine specialist referred her to us as the patient strongly desired future viable pregnancies. The patient was counseled on multiple alternatives, including different methods of performing the cerclage, and ultimately decided on the robotic-assisted (Da Vinci Xi) prophylactic abdominal cerclage. INTERVENTIONS: The bicornuate uterus is a rare class IV mullerian duct anomaly caused by the impaired fusion of the mullerian ducts in the uterus, classically appearing in imaging studies as a heart-shaped uterus. This patient demographic reports a high incidence of obstetric complications. Pregnancy in such a uterus causes complications like first- and second-trimester pregnancy loss, preterm labor, low-birthweight infants, and malpresentation at delivery.1 Researchers have postulated that there is an abnormal ratio of muscle fibers to connective tissue in a congenitally abnormal cervix. During pregnancy, an inadequate uterine volume may lead to increased intrauterine pressure and stress on the lower uterine segment, which can lead to cervical incompetence.2 To address cervical incompetence, cervical cerclages are a commonly utilized procedure, as recent studies demonstrate that the incidence of term pregnancies in the group with documented cervical incompetence treated with cerclage placement increased from 26% to 63%.3 One observational study noted improved obstetrical outcomes occurred with interval placement, a cerclage placed in between pregnancies in the nongravid uterus, compared with cerclage placement between 9 and 10 weeks gestation, with the mean gestational age for delivery at 32.9 weeks and 34.5 weeks when a cerclage was placed in gravid and nongravid women, respectively.4 In addition, another retrospective study was done, which demonstrated a lower incidence of neonatal death with prophylactic cerclages.5 Operating on a nonpregnant uterus offers several benefits, including its reduced size, fewer and smaller blood vessels, and simplified handling. Moreover, there are clearly no concerns regarding the fetus. In the decision to use a robotic-assisted platform vs. laparoscopic, a systematic review showed the rates of third-trimester delivery and live birth (LB) using laparoscopy during pregnancy were found to be 70% and 70%-100%, respectively. The same review demonstrated slightly improved outcomes via the robotic route regarding gestational age at delivery (median, 37 weeks), rates of LB (90%), and third-trimester delivery (90%).6 Additional factors contributing to the preference for robotics in surgical procedures include incorporating advanced tools, which can enhance the robotic system's advantages compared with traditional laparoscopy. An invaluable tool in this context is the simultaneous utilization of Firefly mode, which employs a near-infrared camera system, achieved through injecting indocyanine green dye or integrating other light sources concurrently. The intravenous administration of indocyanine green is acknowledged widely for its safety and efficacy as a contrast agent in the evaluation of microvascular circulation and organ vascularization. This property equips surgeons with heightened precision when guiding the needle, proving especially advantageous when faced with challenges in visualizing vascular anatomy. In our specific case, we harnessed the capabilities of Firefly mode in conjunction with hysteroscopic light, enabling us to vividly illustrate the contours of a bicornuate uterus from both external and internal perspectives. We demonstrate a simplified technique of the abdominal cerclage, one cerclage around the internal cervical os of the uterus, using a robotic-assisted platform in a nongravid patient. The surgery began with the eversion of the umbilicus, and a 15-mm skin incision was made in the umbilicus. A Gelpoint mini advanced access site laparoscopy device was inserted into the incision, and CO2 was allowed to insufflate the abdominal cavity with careful attention given to intraabdominal pressure. Once the DaVinci was docked, the surgeon began the creation of a bladder flap. The bladder was carefully dissected from the lower uterine segment and both uteri using monopolar scissors. The anatomical differences of a bicornuate uterus prompted the surgeon to dissect a wider circumference for safety reasons, where a wider dissection offers a better view of the uterine vessels and ease of introducing the Mersilene tape later on. Bilateral uterine vessels were further skeletonized and exposed anteriorly using blunt dissection and monopolar scissors. After further dissection and lateralization, the final result creates a landmark medial to the right uterine vessels at the level of the internal cervical os with which the needle of the Mersilene tape will be able to pass through. The Mersilene tape was guided from anterior to posterior via a previously straightened needle. Similarly, a landmark was created on the left, and the Mersilene tape was directed from anterior to posterior. The Mersilene tape was placed circumferentially around the internal cervical os of the bicornuate uterus, medial to the uterine vessels. Both ends of the Mersilene tape were then gently pulled, ensuring that the tape was lying flat on the anterior of the internal cervical os with no bowels or uterine vessels within it. The tape was then tied posteriorly at the 6 o'clock position with appropriate tension. A 2-0 silk was then sutured to the tails of the tape using the purse-string technique to ensure that it would remain securely tied and in the correct position. Hemostasis was assured. Both a hysteroscopy and a cystoscopy were done after the completion of the cerclage to ensure that no tape or sutures were seen within the cervical canal or the uterine cavity. None were observed. MAIN OUTCOMES MEASURES: The success criteria for the surgery were identified as the patient's ability to attain a viable pregnancy after the cerclage placement, along with achieving LB. RESULTS: Subsequently, a spontaneous pregnancy was achieved. An infant weighing 3 pounds and 16 ounces was delivered by cesarean section at 36 weeks because of an oligohydramnios. The infant is currently healthy at 13 pounds. CONCLUSION: Robotic-assisted abdominal cerclage around the internal cervical os in a bicornuate uterus offers a possibly feasible and straightforward technique for surgeons seeking to reduce risks, although further research is needed.


Subject(s)
Bicornuate Uterus , Cerclage, Cervical , Robotic Surgical Procedures , Uterine Cervical Incompetence , Female , Humans , Pregnancy , Young Adult , Abortion, Habitual/surgery , Abortion, Habitual/etiology , Abortion, Habitual/prevention & control , Bicornuate Uterus/complications , Bicornuate Uterus/diagnostic imaging , Bicornuate Uterus/surgery , Cerclage, Cervical/methods , Robotic Surgical Procedures/methods , Treatment Outcome , Urogenital Abnormalities/surgery , Urogenital Abnormalities/diagnostic imaging , Urogenital Abnormalities/complications , Uterine Cervical Incompetence/surgery , Uterine Cervical Incompetence/diagnostic imaging , Uterus/abnormalities , Uterus/surgery , Uterus/diagnostic imaging
8.
Cureus ; 15(11): e48841, 2023 Nov.
Article in English | MEDLINE | ID: mdl-38106706

ABSTRACT

Choriocarcinoma, an aggressive gestational trophoblastic disease, infrequently manifests with spontaneous uterine perforation. We report the case of a 22-year-old female with five months of amenorrhea presenting with acute abdominal pain. Ultrasound and MRI assessment highlighted a uterine perforation with choriocarcinoma. Subsequent total abdominal hysterectomy revealed choriocarcinoma in the bicornuate uterus with uterine perforation. Histopathological analysis confirmed the diagnosis of choriocarcinoma in the cornu of the uterus. Timely diagnosis is vital to reduce mortality. Notably, choriocarcinoma in a bicornuate uterus is exceptionally rare. Radiological evaluations are critical for diagnosis, staging, and follow-up.

9.
Case Rep Womens Health ; 39: e00524, 2023 Sep.
Article in English | MEDLINE | ID: mdl-37954229

ABSTRACT

Bicornuate uterus (BU) is a rare congenital anomaly that may present with various obstetric complications, and very rarely may be a risk factor for uterine rupture, even of an unscarred uterus. A 21-year-old primigravida woman, at 19 weeks and 5 days of gestation, presented with severe abdominal pain and features of hypovolemic shock. Urgent laparotomy showed a large hemoperitoneum, a ruptured left horn of a BU and a dead fetus in the abdominal cavity. Excision of the ruptured left horn was performed and the uterine defect was sutured. Postoperative recovery was uneventful. Bicornuate uterus is a rare uterine anomaly and is associated with various obstetric complications at different gestational ages. Ruptured uterus should be considered in the differential diagnoses of acute abdominal pain and a picture of hypovolemia in women with mid-trimester pregnancies.

10.
Cureus ; 15(10): e47618, 2023 Oct.
Article in English | MEDLINE | ID: mdl-38022269

ABSTRACT

Placenta accreta is defined as an abnormal trophoblast invasion of part or all of the placenta into the myometrium of the uterine wall. It is a well-known cause of maternal morbidity and mortality. Here, we present a unique case of focal placenta accreta due to a bicornuate uterus and a history of septum resection. We also discuss its management and outcome. The patient underwent a classical cesarean section and reinforcement of the anterior and posterior uterine wall. The patient had a history of surgery for correction of uterine malformation, which may have resulted in an abnormal adherence of the placenta.

11.
Arch Gynecol Obstet ; 2023 Sep 30.
Article in English | MEDLINE | ID: mdl-37777621

ABSTRACT

OBJECTIVE: This study aimed to evaluate whether a trial of labor after cesarean delivery (TOLAC) in women with a bicornuate uterus is associated with increased maternal and neonatal morbidity compared to women with a non-malformed uterus. METHODS: A multicenter retrospective cohort study was conducted at two university-affiliated centers between 2005 and 2021. Parturients with a bicornuate uterus who attempted TOLAC following a single low-segment transverse cesarean delivery (CD) were included and compared to those with a non-malformed uterus. Failed TOLAC rates and the rate of adverse maternal and neonatal outcomes were compared using both univariate and multivariate analyses. RESULTS: Among 20,844 eligible births following CD, 125 (0.6%) were identified as having a bicornuate uterus. The overall successful vaginal delivery rate following CD in the bicornuate uterus group was 77.4%. Failed TOLAC rates were significantly higher in the bicornuate group (22.4% vs. 10.5%, p < 0.01). Uterine rupture rates did not differ between the groups, but rates of placental abruption and retained placenta were significantly higher among parturients with a bicornuate uterus (9.8% vs. 4.4%, p < 0.01, and 9.8% vs. 4.4%, p < 0.01, respectively). Neonatal outcomes following TOLAC were less favorable in the bicornuate group, particularly in terms of neonatal intensive care unit admission and neonatal sepsis. Multivariate analysis revealed an independent association between the bicornuate uterus and failed TOLAC. CONCLUSIONS: This study found that parturients with a bicornuate uterus who attempted TOLAC have a relatively high overall rate of vaginal birth after cesarean (VBAC). However, their chances of achieving VBAC are significantly lower compared to those with a non-malformed uterus. Obstetricians should be aware of these findings when providing consultation to patients.

12.
BMC Pregnancy Childbirth ; 23(1): 559, 2023 Aug 02.
Article in English | MEDLINE | ID: mdl-37533012

ABSTRACT

Pregnancies in the bicornuate uterus are usually considered high-risk because of their association with poor reproductive outcomes such as recurrent pregnancy loss, cervical insufficiency, low birthweight, preterm birth, malpresentation, cesarean delivery, and uterine rupture. The objective of the report was to show that patients with a scared bicornuate uterus at post-term could have successful pregnancy outcomes. We are presenting a 28-year-old gravida III para II lady with a bicornuate scared uterus at post-term. She has a history of early third-trimester pregnancy loss, and her second pregnancy was delivered via cesarean section. In her first pregnancy, the patient's uterus was not identified as bicornuate. However, an ultrasound during her second pregnancy revealed that she had a bicornuate uterus, which led to the diagnosis. At term, the lady had a successful cesarean section. Lastly, for the current post-term pregnancy she had no prenatal check-up. Even though this, she was coming at the latent first stage of labor and an emergency cesarean section was done.Conclusion Successful outcomes could be achieved in patients with the bicornuate uterus at post-term gestation.


Subject(s)
Bicornuate Uterus , Pregnancy Complications , Pregnancy, Prolonged , Premature Birth , Pregnancy , Humans , Infant, Newborn , Female , Adult , Cesarean Section/adverse effects , Pregnancy Complications/etiology , Premature Birth/etiology , Pregnancy Outcome
13.
Cureus ; 15(6): e40938, 2023 Jun.
Article in English | MEDLINE | ID: mdl-37496525

ABSTRACT

Intrauterine devices (IUDs) have become one of the most frequently used forms of long-acting reversible contraception (LARC) in women of childbearing age. While complications are generally considered to be minimal, they can occur during the insertion, during use, or upon removal. Uterine anomalies, such as a bicornuate uterus, can increase the risk of complications during all stages. Here, we describe a case of a patient with a bicornuate uterus who had a levonorgestrel IUD in place for five years before she experienced a dislodging of the IUD, fragmentation upon attempted removal, and ultimately required a hysteroscopy to remove an embedded fragment from the endocervical canal. Due to the limited reporting on fragmented IUDs, further studies will be required to assess the optimal management. While symptomatic patients should have the fragment removed, asymptomatic patients should have their individual history and desire for future pregnancy weighed against the risk and benefits of treatment.

14.
Clin Case Rep ; 11(6): e7440, 2023 Jun.
Article in English | MEDLINE | ID: mdl-37305878

ABSTRACT

Key Clinical Message: Twin pregnancies in uterine didelphys and uterus bicornuate bicollis represent dicavitary twin pregnancies that can be managed using similar principles. Consideration must be given to delivery planning including mode of delivery and uterine incision. Abstract: Dicavitary twin pregnancies present unique challenges for obstetric management. This case demonstrates an approach to management of a bicornuate bicollis twin pregnancy and provides a contemporary review of the literature on dicavitary twin pregnancies.

15.
Cureus ; 15(4): e38248, 2023 Apr.
Article in English | MEDLINE | ID: mdl-37252564

ABSTRACT

Congenital Mullerian anomalies are rare developmental defects that result in malformation of the fallopian tubes, uterus, cervix, and vagina. The bicornuate uterus is one of the many variants of Mullerian anomalies, defined as having an external fundal indentation of greater than one centimeter. Pelvic ultrasound has a sensitivity of 99% for identifying bicornuate uteruses and is the predominant imaging device used for diagnosis. The cervical and uterine cavity anatomy in patients with a bicornuate uterus varies. The effect of maternal uterine structure on offspring development has been poorly documented. This report details a rare case of dichorionic-diamniotic twin pregnancy in a bicornuate uterus with one fetus affected by Ebstein's anomaly. Twin A was diagnosed by first-trimester ultrasound with right renal agenesis and Ebstein's anomaly. Twin B did not have any anatomical defects identified on ultrasound. Both twins were delivered via emergency repeat cesarean section at 34 weeks and four days due to nonreassuring fetal heart tracings and twin A in the breech presentation. Twin A and twin B were found to be in separate horns within the uterus during low transverse cesarean section. Twin A required endotracheal intubation in the delivery room due to respiratory distress. Both twins required neonatal intensive care treatment. Twin A was found to have a right pelvic kidney, rather than right renal agenesis, while in the neonatal intensive care unit. Females with germline mutations in the Mullerian duct and urogenital sinus development have resulted in concomitant malformations in the uterus and kidneys. This is a rare case of an infant with a cardiac anomaly born to a mother with a germline mutation. The relationship between congenital heart defects and uterine anomalies has not been identified. As seen in this case, maternal malformations impacting fetal cardiac development can be sporadic or result from germline mutations in mesoderm that have not been reported yet.

16.
Radiol Case Rep ; 18(6): 2107-2111, 2023 Jun.
Article in English | MEDLINE | ID: mdl-37089975

ABSTRACT

Bicornuate uterus is a rare congenital uterine malformation and structural abnormality that affects less than 05 % of women in the United States. Resulting pregnancies are rarely carried to term and commonly result in fetal abnormalities and complications such as decreased fertility and risks of complicated pregnancies such as spontaneous abortion and miscarriages, IUGR, preterm delivery, cervical incompetence, and fetal malpresentations. This report highlights a case in which a 32-year-old patient with a bicornis bicollis uterus who had a viable pregnancy in one of the uterine horns later delivered a live male neonate at term without complications via spontaneous vaginal delivery (SVD). The study underscores the importance of using the imaging modalities of ultrasonography, magnetic resonance imaging, and hysterosalpingograms to aid in the diagnosis and monitoring throughout the duration of the pregnancy.

17.
Ann Med Surg (Lond) ; 85(4): 1166-1171, 2023 Apr.
Article in English | MEDLINE | ID: mdl-37113970

ABSTRACT

A solid pseudopapillary neoplasm (SPN) of the pancreas is a rare tumor of the pancreas. Concomitant SPN with urogenital anomalies is a very rare presentation. Case Presentation: A 16-year-old female presented with a chief complaint of abdominal pain 30 days back. Solid pseudopapillary neoplasm (SPN) of the pancreas was diagnosed with the aid of ultrasonography and contrast-enhanced computed tomography of the abdomen and pelvis. Incidentally, concomitant left unilateral renal agenesis and bicornuate uterus were also detected in radiological findings. The patient underwent spleen-preserving distal pancreatectomy, and SPN was confirmed with the histopathological report. Discussion: Symptomatic SPN patients present with an abdominal mass and pain or very rarely jaundice. Most of the SPNs are benign. Complete surgical excision results in more than 95% cure. SPN with concomitant urogenital anomalies is extremely rare, and their concurrent occurrence can be better attributed to Wnt signaling pathway owing to their similar pathogenic mechanism. Conclusion: The solid pseudopapillary tumor has an excellent prognosis if timely resected. Proper evaluation of the patient with imaging is necessary to suspect and diagnose SPN who has urogenital anomalies and vice versa.

18.
CEN Case Rep ; 12(1): 122-129, 2023 02.
Article in English | MEDLINE | ID: mdl-36056295

ABSTRACT

Congenital anomalies of the kidney and urinary tract (CAKUT) are frequently associated with Mullerian anomalies. This can be explained by the fact that Mullerian duct elongation depends on the preformed Wolffian duct during embryogenesis. While CAKUT such as unilateral renal agenesis and multicystic dysplastic kidney are commonly identified prenatally by routine ultrasound, the diagnosis of Mullerian anomalies is often delayed, increasing the risk of complications such as endometriosis or pelvic inflammatory disease. Herein, we report a case of a premenarchal girl who had initially been diagnosed with right multicystic dysplastic kidney. She presented with continuous urinary incontinence at 4 years old and further evaluation by contrast-enhanced computed tomography, cystoscopy, colposcopy, ureterography, and hysterosalpingography led to the final diagnosis of right hypodysplastic kidney and ectopic ureter associated with bicornuate uterus. A strong family history of uterine malformations prompted the examination of the uterus. Genetic testing was suggested but the family declined. She is planned to be referred to a gynecologist at puberty for further assessment. The recognition and screening rate of concurrent Mullerian anomalies in CAKUT patients varies between institutions. Screening for Mullerian anomalies in prediagnosed CAKUT girls may enable to provide timely counseling and to prevent gynecological complications.


Subject(s)
Bicornuate Uterus , Multicystic Dysplastic Kidney , Ureter , Ureteral Obstruction , Female , Humans , Child, Preschool , Ureter/abnormalities , Vagina/abnormalities , Kidney/diagnostic imaging , Kidney/abnormalities
19.
Ginecol. obstet. Méx ; 91(2): 119-125, ene. 2023. graf
Article in Spanish | LILACS-Express | LILACS | ID: biblio-1448322

ABSTRACT

Resumen ANTECEDENTES: Las anomalías uterinas congénitas son consecuencia de la formación anormal, fusión o reabsorción de los conductos de Müller durante el desarrollo embriológico. El útero bicorne, unicorne y didelfo (U1, U2, U3 ESHRE-ESGE) son defectos en la unificación con una baja prevalencia en la población general y en la que tiene antecedente de aborto e infertilidad; se estima en alrededor de 0.4 a 1.1%, respectivamente. La presentación del caso puede aportar información útil relacionada con la evolución del embarazo y los desenlaces perinatales cuando aparece esta anomalía. CASO CLÍNICO: Paciente de 35 años, con amenorrea secundaria y prueba inmunológica de embarazo positiva, con antecedentes de dismenorrea primaria severa y anomalía uterina congénita compatible con útero bicorpóreo (U3 completo ESHRE-ESGE), bicorne completo (4a-AFS) y didelfo. El embarazo trascurrió sin complicaciones hasta la semana 37, cuando finalizó mediante cesárea, con hipotonía uterina que no ameritó transfusión de hemoderivados. CONCLUSIÓN: Los embarazos gemelares con útero bicorne son excepcionales y casi todos se logran espontáneamente; los que llegan a término finalizan mediante cesárea electiva.


Abstract BACKGROUND: Congenital uterine anomalies are the result of abnormal formation, fusion, or resorption of the Müllerian ducts during embryological development. The bicornuate, unicornuate and didelphic uterus (U1, U2, U3 ESHRE/ESGE), are unification defects with a low prevalence both in the general population and in the population with a history of abortion and infertility, is estimated at around 0.4 to 1.1%, respectively. The presentation of the case can provide useful information related to with the evolution of the pregnancy and the perinatal outcomes when this anomaly appears. CLINICAL CASE: A 35-year-old patient with secondary amenorrhea and a positive immunological pregnancy test with a history of severe primary dysmenorrhea and previous magnetic resonance imaging with a congenital uterine anomaly compatible with a bicorporeal uterus (U3 complete ESHRE-ESGE), complete bicornuate (4a /AFS) , uterus didelphys (ASRM). The pregnancy was uncomplicated until 37 weeks, when it was terminated by cesarean section, with uterine hypotonia that did not require transfusion of blood products. CONCLUSION: Twin pregnancy with bicornuate uterus is a rare entity, most of them have been achieved spontaneously and those that reach term have been resolved by elective cesarean section.

20.
J Med Case Rep ; 16(1): 130, 2022 Apr 02.
Article in English | MEDLINE | ID: mdl-35365244

ABSTRACT

BACKGROUND: Uterine anomalies occur because of Müllerian duct maldevelopment. Few of them are associated with adverse obstetric outcome (Reyes-Muñoz et al. in Diagnostics. 2019;9:4. https://doi.org/10.3390/diagnostics9040149 ). Genital outflow tract obstructive uterine anomalies invariably present in the adolescent age group. CASE DETAILS: We report a case series of uterine anomalies. Ten such cases presented like a "cluster" within a short span of just one month. Eight of these ten cases were diagnosed intraoperatively during cesarean section. One case was diagnosed during laparoscopic sterilization, and the other case was diagnosed before doing manual vacuum aspiration. There were four cases of bicornuate uterus, two cases each of unicornuate uterus and uterine didelphys, and one case each of septate uterus and arcuate uterus. All eight babies were healthy and without any obvious congenital anomalies. To the best of the authors' knowledge, literature regarding these anomalies has been mentioned mostly as case reports (Bruand et al. in Cureus. 2020;12:3. https://doi.org/10.7759/cureus.7191 ) and a few case series (Ross et al. in BMJ Case Rep. 2018. https://doi.org/10.1136/bcr-2017-221815 ). All women were of Kannadiga ethnicity and in the age range of 19-35 years. They were from places nearby to our institute within a range of approximately 250 km. CONCLUSION: We describe herein almost all types of uterine anomalies. These rare uterine anomalies presented in a short span of just four weeks like a "cluster". This incidental finding is unusual. We need to design studies to understand the reasons for clustering of such cases in our clinical practice.


Subject(s)
Cesarean Section , Urogenital Abnormalities , Adolescent , Adult , Female , Humans , Mullerian Ducts/abnormalities , Pregnancy , Urogenital Abnormalities/surgery , Uterus/abnormalities , Uterus/surgery , Young Adult
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