Fetal Diagnosis in a Unique Case of Vascular and Cardiac Interdependence in Omphaloischiopagus Conjoined Twins.
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ABSTRACT:
SUBMITTER: Gaitonde M
PROVIDER: S-EPMC8236376 | biostudies-literature | 2021 Jun
REPOSITORIES: biostudies-literature
Publications
Fetal Diagnosis in a Unique Case of Vascular and Cardiac Interdependence in Omphaloischiopagus Conjoined Twins.
Gaitonde Mansi M Kreeger Joseph J Border William W Roberts Jessica J Elshenawy Summer S Geary Franklyn F Michelfelder Erik E
CASE (Philadelphia, Pa.) 20210227 3
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Project description:Conjoined triplets are among the rarest of human malformations, as are asymmetric or parasitic conjoined twins. Based on a very modest corpus of recent literature, we applied the embryonic disk model of conjoined twinning to 10 previously reported cases involving asymmetric anatomical multiplications to determine whether they concerned conjoined twins or conjoined triplets. In spite of their phenotypic similarities, we diagnosed four of these cases as conjoined twins and three of them as conjoined triplets. In the remaining three cases, no definite diagnosis could be made, as essential information was lacking from the reports. We conclude that it is not necessarily the expected duplication or triplication of structures that points to the correct diagnosis in these cases, but the number and mutual position of the hearts they presented with. Considering their rarity we stress to thoroughly investigate and describe internal (dys)morphology in novel cases of (asymmetric) conjoined twins and triplets to further unravel their pathogenicity and come to the correct diagnoses.
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Project description:BackgroundConjoined twin is a rarely seen congenital anomaly associated with significant morbidity and mortality. The most common types of conjoined twins are thoracopagus. Conjoined twins are either symmetrical twins or asymmetrical or heteropagus. This report records the successful separation of 2 cases of asymmetrical twins and one symmetrical twin with fused livers and diaphragm and communicating peritoneal cavities.Patients and methodsThis study amended and strictly followed the ethical guidelines of the Helsinki declaration, our study included 4 case reports on conjoined twins (CT), 2 male and three female patients; 3 of them were parasitic and one was conjoined. We operated upon 2 parasitics and one conjoined. The 1st case died preoperatively.ResultsFour living children is our result.ConclusionAs connections between the bowel and bone of the parasite and the respective organs in the autosite are often absent, the parasite could be excised easily without any effect on the autosite. We recommend the separation of the parasite and the autosite as early as possible. In many cases, surgery results in the death of one or both of the conjoined twins, particularly if they are joined at the head or share a vital organ. Our separated twins are yet the youngest living separated twins.Trial registrationClinicalTrials.gov Identifier: NCT03388684.
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Project description:Medical imaging plays a crucial role in the management of conjoined twins. The role of imaging is to explore the anatomy, outline the shared organs and determine whether surgery is feasible. It also serves as a roadmap for successful separation. Additionally, imaging helps with counseling parents about prognosis. This review aims to illustrate recent advances in different imaging modalities and their role in the management of the various types of conjoined twins with an emphasis on relevant tips for optimal imaging.
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Project description:Introduction This report will discuss a case of minimally conjoined omphalopagus twins (MCOTs) with a body stalk anomaly (BSA). Case Report We experienced monochorionic diamniotic (MD) twins born at 31 weeks. One infant was suspicious of BSA before birth, and another infant was normal. But normal infant had anal atresia with small intestine which was inserted behind the umbilicus. Twins had very short common umbilicus and infant with BSA had intestinal conjunction, two appendixes at the site of the colon, and a blind-ending colon. We diagnosed MCOTs. Discussion On the basis of the Spencer hypothesis, the etiology of MCOTs was that MD twins shared a yolk sac. However, this could not explain the presence of a BSA. It is necessary to consider the possible reasons for a singleton BSA. In addition, intestinal fusion occurred unequally in this case, although two appendixes were found in the same place, which might have occurred because of the balanced fusion.
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Project description:BackgroundThe surgical separation of two Conjoined Twins is a particularly complex operation. Surgical times are particularly long and post-operative complications are very frequent in this type of procedure. We report a clinical case of surgical separation of two thoraco-omphalopagus conjoined twins in which, thanks to the use of (3D) three dimensional technologies, we were able to significantly reduce operative times and improve clinical outcomes.MethodsWe performed a 3D reconstruction of the anatomical parts involved in congenital fusion using Computer Tomography (CT) images.We obtained virtual anatomical models of the patients which allowed us to estimate essential details as the residual post-operative thoracic volume as well as the exact position of resection planes for both the general separation and for the hepatic splitting procedure. Subsequently, we printed 3D anatomical models of the thoracic cage and sternum and of the liver with the plane of resection. Finally, we printed an additional 3D anatomical model of the two patients representing different organs with multiple colors and materials.ResultsThe use of 3D printing reduced the duration of surgery by 30% with a favorable patient outcome. Two years after the operation, the patients do not present any type of deficit and have a normal life without any significant complication.ConclusionVirtual anatomical 3D models and 3D printing represent a valid technological tool to support complex surgical operations, especially in pre-surgical planning. 3D models are important tools to better understand complex anatomy and to discuss clinical cases among members of the surgical team.
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Project description:Objective Describe a case of cloaca prenatally diagnosed in one of a set of monoamniotic twins. Study Design Retrospective review of a case. Results Cloaca is one of the most complex and severe degrees of anorectal malformations in girls. We present a discordant cloaca in monoamniotic twins. Fetal ultrasound showed a female fetus with a pelvic midline cystic mass, a phallus-like structure, a probable anorectal atresia with absence of anal dimple and a flat perineum, and renal anomalies. The diagnosis was confirmed by fetal magnetic resonance imaging postnatally. Conclusions The rarity of the malformation in a monoamniotic pregnancy, the difficulties of prenatal diagnosis, the pathogenic assumptions, and the consequences of adequate amniotic fluid for fetal lung development are discussed.
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Project description:BACKGROUND: A large number of monozygotic and dizygotic twin pairs with inflammatory bowel disease have been reported. To date no twin pair has developed phenotypically discordant inflammatory bowel disease. This case report is the first documented occurrence of discordant inflammatory bowel disease occurring in monozygotic twins. CASE REPORT: Twenty two year old identical male twins presented within three months of each other with inflammatory bowel disease that proved to be discordant in overall disease type, disease distribution, clinical course, and histopathological findings. Twin 1 developed a severe pancolitis necessitating total colectomy while twin 2 developed a predominantly distal patchy colitis with frequent granulomas, controlled by aminosalicylates. Twin 1 was antineutrophil cytoplasmic antibody (ANCA) negative at the time of testing while twin 2 (Crohn's disease) was ANCA positive. Significantly, the twins possessed the HLA type DR3-DR52-DQ2 previously associated with extensive colitis. CONCLUSION: This case report confirms the important role played by genetic factors in the development of inflammatory bowel disease. It also highlights the crucial role of undetermined environmental agents in dictating disease expression and phenotype.
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Project description:BackgroundDiagnosis of constrictive pericarditis requires demonstration of interventricular interdependence which can prove difficult even with invasive haemodynamics. Its treatment requires invasive surgical procedures prior to which diagnostic certainty is necessary. Cardiac magnetic resonance imaging (MRI) is an underutilized tool for identification of this pathology.Case summaryWe present two cases of heart failure due to interventricular interdependence with inconclusive invasive haemodynamic. Prior to recommending invasive surgical treatment, confirmation of the diagnosis was required. This was achieved using cardiac MRI leading to pericardiectomy followed by clinical improvement.DiscussionThese cases demonstrate the clinical utility, sensitivity, and specificity of cardiac MRI for ventricular interdependence.
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Project description:BackgroundDetermining the cause of intrauterine fetal death is essential for patients to manage their next pregnancy. However, in the majority of cases of fetal death, the cause remains unexplained despite comprehensive evaluation, especially in the cases of twins. Among twin pregnancies, conditions of monochorionic twinning, commonly regarded as monozygotic, are more complicated than dichorionic ones.Case summaryWe systematically evaluated the cause of fetal death for a Han Chinese woman with monochorionic twinning following in vitro fertilization/embryo transfer. Discrepant karyotypes were unexpectedly discovered between the twins. One fetus had an aneuploid male karyotype (46, XY), dup (9) (p24.3-q13), and the other had a normal female karyotype (46, XX). We considered that the male died of aberration of chromosome 9 and the female died of subsequent acute exsanguination through vascular anastomosis.ConclusionThis study demonstrated the importance of recognizing the presence of monochorionic dizygotic twinning and the challenges of clinical management for twins following in vitro fertilization/double embryo transfer.
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