Minimally invasive therapy for the resolution of a subcapsular hepatic hematoma in a newborn. / Terapia mínimamente invasiva para el manejo de un hematoma subcapsular hepático en un recién nacido.

OBJECTIVE: To describe an innovative alternative to exploratory laparotomy in a newborn with a sub capsular hepatic hematoma secondary to umbilical vein catheterization. CLINICAL CASE: A preterm baby with a history of hyaline membrane disease, pulmonary hypertension, and large patent ductus arteriosus, requiring mechanical ventilation and the use of vasoactive drugs. Umbilical catheters were inserted and through an abdomen X-ray, we observed their proper position. The patient evolved with greater requirements of vasoactive drugs, abdominal wall pallor, and abdominal distention. Abdominal ultrasound showed a subcapsular hepatic hematoma, with no signs of active bleeding, so expectant management was decided. The patient required increased vasoactive drugs and presented a decrease in hematocrit. New ultrasound showed a larger subcapsular hematoma, abundant perihe patic fluid, and the intraparenchymal position of the umbilical catheter was confirmed. Endovascular embolization was performed through the umbilical catheter with Gelita®, achieving occlusion of the capsular path. Posterior ultrasound showed a reduction of the hematoma. CONCLUSIONS: The use of embolization through angiography is not commonly used in pediatric emergencies. It is a procedure with fewer comorbidities and complications than exploratory laparotomy, therefore it should be considered as first-line therapy in patients like the one presented above. The limitation for its routine performance is the lack of available angiography operating room and trained interventional radio logy team.

Neonatal Escherichia coli infection in twins: clinical spectrum and management dilemma.

The clinical manifestation of Escherichia coli could vary from asymptomatic bacteraemia to systemic bloodstream infection and meningitis. We describe an unusual course of E. coli infection in twins, emphasising commencement of appropriate antimicrobial therapy. A set of male dichorionic diamniotic twins were delivered at 34 weeks of gestation by caesarian section. Pregnancy was complicated by diabetes, pre-eclampsia and cholestasis. Antenatal ultrasounds noted a congenital pulmonary airway malformation in twin A. Following delivery, twin A developed respiratory distress, but twin B was asymptomatic. Partial septic work-up at admission in the neonatal intensve care unit was done. Twin A's blood culture grew E. coli, while twin B's blood culture was negative. Twin A was treated with 7 days of intravenous antibiotics. At 11 days of age, twin B acutely developed a scrotal swelling. On suspicion of testicular torsion, he was taken for urgent surgery, which revealed a scrotal abscess positive for E. coli The scrotum was irrigated and successfully treated with 4 weeks of antibiotics. Both twins were doing well at 3 months of follow-up.

Perinatal outcome of pregnancy complicated by twin anemia-polycythemia sequence: systematic review and meta-analysis.

OBJECTIVE: To report the perinatal outcome of monochorionic diamniotic (MCDA) twin pregnancies complicated by twin anemia-polycythemia sequence (TAPS), according to the type of TAPS (spontaneous or postlaser) and the management option adopted. METHODS: MEDLINE, EMBASE and The Cochrane Library databases were searched for studies reporting on the outcome of twin pregnancies complicated by TAPS. Inclusion criteria were non-anomalous MCDA twin pregnancies with a diagnosis of TAPS. The primary outcome was perinatal mortality; secondary outcomes were neonatal morbidity and preterm birth (PTB). The outcomes were stratified according to the type of TAPS (spontaneous or following laser treatment for twin-twin transfusion syndrome) and the management option adopted (expectant, laser surgery, intrauterine transfusion (IUT) or selective reduction (SR)). Random-effects meta-analysis of proportions was used to analyze the data. RESULTS: Perinatal outcome was assessed according to whether TAPS occurred spontaneously or after laser treatment in 506 pregnancies (38 studies). Intrauterine death (IUD) occurred in 5.2% (95% CI, 3.6-7.1%) of twins with spontaneous TAPS and in 10.2% (95% CI, 7.4-13.3%) of those with postlaser TAPS, while the corresponding rates of neonatal death were 4.0% (95% CI, 2.6-5.7%) and 9.2% (95% CI, 6.6-12.3%), respectively. Severe neonatal morbidity occurred in 29.3% (95% CI, 25.6-33.1%) of twins after spontaneous TAPS and in 33.3% (95% CI, 17.4-51.8%) after postlaser TAPS, while the corresponding rates of severe neurological morbidity were 4.0% (95% CI, 3.5-5.7%) and 11.1% (95% CI, 6.2-17.2%), respectively. PTB complicated 86.3% (95% CI, 77.2-93.3%) of pregnancies with spontaneous TAPS and all cases with postlaser TAPS (100% (95% CI, 84.3-100%)). Iatrogenic PTB was more frequent than spontaneous PTB in both groups. Perinatal outcome was assessed according to the management option adopted in 417 pregnancies (21 studies). IUD occurred in 9.8% (95% CI, 4.3-17.1%) of twins managed expectantly and in 13.1% (95% CI, 9.2-17.6%), 12.1% (95% CI, 7.7-17.3%) and 7.6% (95% CI, 1.3-18.5%) of those treated with laser surgery, IUT and SR, respectively. Severe neonatal morbidity affected 27.3% (95% CI, 13.6-43.6%) of twins in the expectant-management group, 28.7% (95% CI, 22.7-35.1%) of those in the laser-surgery group, 38.2% (95% CI, 18.3-60.5%) of those in the IUT group and 23.3% (95% CI, 10.5-39.2%) of those in the SR group. PTB complicated 80.4% (95% CI, 59.8-94.8%), 73.4% (95% CI, 48.1-92.3%), 100% (95% CI, 76.5-100%) and 100% (95% CI, 39.8-100%) of pregnancies after expectant management, laser surgery, IUT and SR, respectively. CONCLUSIONS: The present meta-analysis provides pooled estimates of the risks of perinatal mortality, neonatal morbidity and PTB in twin pregnancies complicated by TAPS, stratified by the type of TAPS and the management option adopted. Although a direct comparison could not be performed, the results from this systematic review suggest that spontaneous TAPS may have a better prognosis than postlaser TAPS. No differences in terms of mortality and morbidity were observed when comparing different management options for TAPS, although these findings should be interpreted with caution in view of the limitations of the included studies. Individualized prenatal management, taking into account the severity of TAPS and gestational age, is currently the recommended strategy. © 2021 International Society of Ultrasound in Obstetrics and Gynecology.

[Delayed delivery in multiple pregnancy: about a case and literature review]. / Accouchement différé d´une grossesse multiple: à propos d´un cas et revue de la littérature.

Delayed delivery is designed to allow the remaining fetus(es) to develop after premature expulsion of the first twin in the second trimester of pregnancy. This decision is aimed to allow the remaining fetus(es) to reach full fetal growth. We here report a clinical case of delayed delivery in a patient with triple pregnancy in whom the time between the expulsion of the first twin and the birth of the third twin was 10 weeks. The purpose of this study was to highlight the benefit and indications for delayed delivery.

COVID-19: Twin Research in Progress/Twin Research Reviews: Asthmatic and Nonasthmatic Twins; Stanford University Study of Anal Prints; Twin Mammals and COVID-19; Twin Delivery and the Three 'Rs'/Mixed Media: The Rainman Twins - The Film; Twins Irish Pub; Twins 'Covid and Corona'; Death of a Surgeon Who Separated Conjoined Twins; Twinco - Twin-Based Supply Company.

It is likely that genetic factors play a role in the susceptibility to, and recovery from, COVID-19. A survey of ongoing twin research could produce findings likely to help in the prevention and management of this pandemic. This survey is followed by a review of research comparing selected features of asthmatic and nonasthmatic twins, links between twin mammals and COVID-19, and relationships between twin delivery and the three 'Rs'. The final section of this article presents newsworthy twin-related items, some associated with COVID-19. They include a summary of the Rainman Twins film, a study of anal prints, the 'Twins' Irish pub, newborn twins 'Covid and Corona', the death of a surgeon who separated conjoined twins, and Twinco, a twin-based supply company.

Comparison of central nervous system relapse outcomes following haploidentical vs identical-sibling transplant for acute lymphoblastic leukemia.

To explore the incidence, risk factors, and outcomes of central nervous system (CNS) relapse after allogeneic hematopoietic stem cell transplantation (allo-HSCT) for acute lymphoblastic leukemia (ALL) and to compare the differences in CNS relapse between haploidentical donor HSCT (HID-HSCT) and HLA-identical sibling donor HSCT (ISD-HSCT). We performed a retrospective nested case-control study on patients with CNS relapse after allo-HSCT. The cumulative incidence of CNS relapse was 4.06% after allo-HSCT in ALL, with a significantly poor prognosis. The incidence was 3.91% and 5.36% in HID-HSCT and ISD-HSCT, respectively (p = .227). Among the patients with CNS relapse, the overall survival (OS) at 3 years was 56.2 ± 6.8% in the HID-HSCT subgroup and 76.9 ± 10.2% in the ISD-HSCT subgroup (p = .176). The 3-year cumulative incidence of systemic relapse was also comparable between the two subgroups (HID-HSCT, 40.6 ± 7.4%; ISD-HSCT, 13.3 ± 8.7%, respectively, p = .085). Younger age (p = .045), T-ALL (p = .035), hyperleukocytosis at diagnosis (p < .001), advanced disease stage at transplant (p < .001), pre-HSCT CNS involvement (p < .001), and absence of chronic graft vs host disease (cGVHD) (p < .001) were independent risk factors for CNS relapse after allo-HSCT. In conclusion, CNS relapse was a significant complication after allo-HSCT in ALL and was associated with poor prognosis. The incidences and outcomes were comparable between HID-HSCT and ISD-HSCT.

Concomitant hypertriglyceridemia-induced pancreatitis in pregnant monozygotic twin siblings.

Hypertriglyceridemia-induced pancreatitis (HTIP) is the third most common cause of pancreatitis. Hypertriglyceridemia shows familial transition and pregnancy increases the risk of HTIP. The treatment of HTIP is initiated with supportive treatment and continues with specific treatments including plasmapheresis, insulin, heparin infusion, and hemofiltration. The current study reports monozygotic twins who are pregnant at the same time having concurrent HTIP attack.

US Evaluation of Twin Pregnancies: Importance of Chorionicity and Amnionicity.

The twin birth rate is increasing in the United States. Twin pregnancies can be dichorionic or monochorionic (MC). MC twins account for 20% of twin pregnancies but 30% of all-cause pregnancy-related complications. This article describes the imaging findings that establish chorionicity and amnionicity. Ideally, these are established in the first trimester when accuracy is high, but they can also be determined later in pregnancy. Complications unique to MC twin pregnancy include twin-twin transfusion syndrome, twin anemia polycythemia sequence, twin reversed arterial perfusion sequence, and selective fetal growth restriction. The US features, staging systems, and management of these complications are reviewed, and the consequences of MC twin demise are illustrated. Ongoing surveillance for these conditions starts at 16 weeks gestation. Monoamniotic (MA) twins are a small subset of MC twins. In addition to all of the MC complications, specific MA complications include cord entanglement and conjoined twinning. Radiologists must be able to determine chorionicity and amnionicity and should be aware of potential complications so that patients may be referred to appropriate regional specialized centers. A proposed algorithm for referral to specialized fetal treatment centers is outlined. Online supplemental material is available for this article. ©RSNA, 2019.

Positional treatment without mechanical ventilation in a very preterm infant with unilateral pulmonary interstitial emphysema: case report and review of the literature.

BACKGROUND: Pulmonary interstitial emphysema (PIE) in very low birth weight infants is a rare but severe complication. Although most of these air leaks develop in mechanically ventilated infants, they have also been reported in infants exposed only to nasal continuous positive airway pressure (CPAP). The optimal treatment for PIE is still under discussion and includes different approaches such as unilateral intubation, high frequency oscillation ventilation and even surgical lobectomy. However, as yet, there has been no report on complete resolution of unilateral PIE by positioning therapy without mechanical ventilation. CASE PRESENTATION: We report the case of a 28+1gestational week twin, 990 g birth weight, Apgar 9-10-10. After stabilization with nasal CPAP the baby received surfactant by less invasive surfactant application (LISA) technique in the delivery room after 35 min of life, and continued respiratory support with nasal CPAP. At day 5 X-ray presented unilateral PIE, while pCO2 increased from 40 mmHg to 55 mmHg and FiO2 from 0.21 to 0.28 to achieve SpO2 in the target range of 89-94%. The baby was treated by strict positioning on the affected hemithorax in a special splint while spontaneously breathing on High Flow Nasal Cannula (HFNC). Complete resolution of the unilateral PIE was observed after 96 h. No chronic lung disease developed. CONCLUSION: For unilateral PIE in very preterm infants, positioning on the affected hemithorax without mechanical ventilation is a therapeutic option.

Discordant immune complete heart block and growth restriction in dichorionic twin pregnancy with permanent pacemaker implantation of an 1140 g neonate.

Fetal atrioventricular block is a rare pathology, mostly due to placental transmission of maternal SSA/Ro and SSB/La antibodies, and can lead to severe fetal or neonatal outcomes. We report a case of dichorionic, diamniotic twin pregnancy, with maternal SSA/Ro antibodies. Isolated complete atrioventricular block was diagnosed at 23 weeks in one fetus (Twin A), while the second fetus (Twin B) remained in normal sinus rhythm. Severe asymmetric intrauterine growth restriction occurred in Twin A. Delivery was by caesarean section at 32 + 2 weeks. Neonatal permanent pacemaker was inserted on the first day after birth in 1140 g neonate. Discordant heart block in twin pregnancy has already been reported in a few dichorionic pregnancies, but the pathway of discordant disease expression remains unclear. Extraction decision is a dilemma between cardiac failure prevention and prematurity associated twin morbidity. This case shows a successful pacing in a very low birth weight neonate.

Study protocol: developing, disseminating, and implementing a core outcome set for selective fetal growth restriction in monochorionic twin pregnancies.

BACKGROUND: Selective fetal growth restriction in monochorionic twin pregnancies is associated with an increased risk of perinatal mortality and morbidity and represents a clinical dilemma. Interventions include expectant management with early preterm delivery if there are signs of fetal compromise, selective termination of the compromised twin, fetoscopic laser coagulation of the communicating placental vessels or termination of the whole pregnancy. Previous studies evaluating interventions have reported many different outcomes and outcome measures. Such variation makes comparing, contrasting, and combining results challenging, limiting ongoing research on this uncommon condition to inform clinical practice. We aim to produce, disseminate, and implement a core outcome set for selective fetal growth restriction research in monochorionic twin pregnancies. METHODS: An international steering group, including professionals, researchers, and lay experts, has been established to oversee the development of this core outcome set. The methods have been guided by the Core Outcome Measures in Effectiveness Trials Initiative Handbook. Potential core outcomes will be developed by undertaking a systematic review of studies evaluating interventions for selective fetal growth restriction in monochorionic twin pregnancies. Potential core outcomes will be entered into a three-round Delphi survey and key stakeholders including clinical professionals, researchers, and lay experts will be invited to participate. Repeated reflection and rescoring of individual outcomes should encourage group and individual stakeholder convergence towards consensus outcomes which will be entered into a modified Nominal Group Technique to finalize the core outcome set. Once core outcomes have been agreed, we will establish standardized definitions and recommend high-quality measurement instruments for each outcome. DISCUSSION: The development, dissemination, and implementation of a core outcome set for selective fetal growth restriction should ensure that future research protocols select, collect, and report outcomes and outcome measures in a standardized manner. Data synthesis will be possible on a broad level and rigorous implementation should advance the quality of research studies and their effective use in order to guide clinical practice, improve patient care, maternal, short-term perinatal outcomes, and long-term neurodevelopmental outcomes. TRIAL REGISTRATION: Core Outcome Measures in Effectiveness Trials (COMET) registration number: 998. International Prospective Register of Systematic Reviews (PROSPERO) registration number: CRD42018092697 . 18th April 2018.

Successful treatment of fulminant neonatal enteroviral myocarditis in monochorionic diamniotic twins with cardiopulmonary support, intravenous immunoglobulin and pocapavir.

Neonatal cardiogenic shock most commonly occurs due to critical congenital heart disease, sepsis, metabolic disorder or arrhythmias. In particular, enterovirus infections are common in the neonatal period, and patients can present with fulminant myocarditis. Early recognition is imperative due to its high morbidity and mortality without prompt and aggressive treatment. We present the successful treatment of fulminant neonatal enteroviral myocarditis in a pair of monochorionic diamniotic twins with cardiopulmonary support, intravenous immunoglobulin and pocapavir, an enteroviral capsid inhibitor. The twins took an almost exact parallel hospital course, including day of extracorporeal membrane oxygenation (ECMO) cannulation, day of ECMO decannulation, improvement of cardiac function, discharge and status at follow-up. While it was difficult to assess the relative contribution of each intervention, our case shows promise in the use of pocapavir for treatment of severe enteroviral infections. Remarkably, both twins demonstrated remarkable recovery within 2 weeks, underscoring that early aggressive cardiopulmonary support, and potentially pocapavir, contributed to their recovery.

Successful treatment of intrauterine cytomegalovirus infection with an intraventricular cyst in a dichorionic diamniotic twin gestation using cytomegalovirus immunoglobulin.

Congenital cytomegalovirus (CMV) infection is the leading cause of severe congenital abnormalities. CMV immunoglobulin (CMVIG) may lower risk for symptomatic disease in congenital CMV infection. In a twin pregnancy, only one fetus shows CMV infection, raising a dilemma about intervention since the uninfected fetus would be exposed to treatment unnecessarily. CMVIG (2 × 200 U/kg) was given due to high viral load and development of an intraventricular cyst. The cyst growth plateaued, no other brain damage developed, and at 8 months, the infant was symptom-free. CMVIG appears appropriate to treat intrauterine CMV infection in this setting.

Different evolution in the treatment of a severe persistent asthma in 2 twins: Case report and review of the literature.

RATIONALE: Asthma is a multifactorial disease with complex genetic inheritance. In children under the age of 5 years, the diagnosis of asthma is a challenge. PATIENT CONCERN: We present the case of twin sisters under the same treatment for persistent asthma, but with different evolution over the time. DIAGNOSES: One of the sister is diagnosed with severe persistent bronchial asthma associated with bronchiectasis and dyslipidemia and the other one only with mild persistent asthma. INTERVENTIONS: At each admission the treatment for the exacerbations and the underlying respiratory infections was represented by antibiotics, short-acting ß2 agonists, and, sometimes, oxygen and systemic corticosteroids. As chronic treatment, they received in the last period inhaled corticosteroids associated with long-acting ß2 agonist. OUTCOMES: Until the age of 6.5 years, they had similar diagnoses and treatment; from this point one was asymptomatic, with normal pulmonary function tests. The other one had a more complicated evolution which led to a severe crisis by the age of 10 years old. LESSONS: Although asthma is a multifactorial disease with complex genetic inheritance, the genetics has its limits. Our twins had a similar onset with the same genetic inheritance, with the same risk factors, with the same comorbidities and with the same treatment. In this context, different evolutions of severe persistent asthma require more extensive genetic investigations. PATIENT CONCERN: We present the case of twin sisters under the same treatment for persistent asthma, but with different evolution over the time.

Prenatal management of fetal intrapericardial teratoma: a systematic review.

OBJECTIVES: The purpose of this systematic review is to provide a comprehensive overview on the clinical course, perinatal outcome, and effectiveness of prenatal management options for pericardial teratoma. METHODS: A comprehensive search including Ovid MEDLINE, Ovid EMBASE, and Scopus was conducted from inception to September 2016. All studies that reported the prenatal course of pericardial teratoma in singleton or twin gestations were considered eligible. Standardized forms were used for data abstraction by two independent reviewers. RESULTS: Out of 217 screened abstracts, 59 studies reporting 67 fetuses with pericardial teratoma were included. Twenty-three singleton fetuses and 3 fetuses in twin gestations underwent prenatal treatment, and 20 (76.9%) of them were hydropic at the time of intervention. Of those, 15/20 (75%) had a favorable outcome. In the non-intervention group (n = 41), 26 (63.4%) developed hydrops, and out of those, 8 (30.8%) had a favorable outcome. CONCLUSION: Prenatal fluid drainage and other prenatal techniques have been utilized in the treatment of intrapericardial teratoma. While most fetuses tolerated pericardiocentesis, the neonatal benefit of this procedure is still uncertain, and outcomes of other interventions had variable success. Prenatal intervention for pericardial teratoma may be an option in specialized units but, given the maternal and fetal risks, needs careful consideration. © 2017 John Wiley & Sons, Ltd.

Prenatal diagnosis and management of vasa previa in twin pregnancies: a case series and systematic review.

BACKGROUND: Twin pregnancies are at higher risks of velamentous cord insertion and vasa previa. In vitro fertilization is an additional risk factor of abnormal cord insertion and thus the incidence of vasa previa is likely to increase over the next decades. OBJECTIVE: We sought to evaluate the role of ultrasound imaging in optimizing the management of twins diagnosed with vasa previa antenatally. STUDY DESIGN: We searched our database for twin pregnancies diagnosed with vasa previa and managed antenatally using measurements of cervical length and performed a systematic review of articles that correlated prenatal diagnosis of vasa previa in twins and pregnancy outcome. PubMed and MEDLINE were searched for studies published from 1987 through October 20, 2016, using specific medical subject heading terms, key words, and their combination. The primary eligibility criteria were articles that correlated prenatal ultrasound imaging of vasa previa and pregnancy outcome in twins. The secondary eligibility criteria was the use of cervical length in the management of twin pregnancies diagnosed antenatally with vasa previa. Two authors independently assessed inclusion criteria, data extraction, and analysis. The final selection included 3 case report series, 9 retrospective cohort studies, and 1 retrospective case-control study of vasa previa diagnosed prenatally and confirmed at birth in twin pregnancies. RESULTS: The search of our databases identified 6 cases of dichorionic-diamniotic twins and 1 case of monochorionic-diamniotic twins diagnosed prenatally with vasa previa between 22-29 weeks and managed using cervical length. Two cases were delivered by emergency because of rapid changes in cervical length in one and bleeding on placenta previa in the other at 33 and 30 weeks, respectively. The systematic review identified data on 56 cases. The incidence of twin pregnancies diagnosed antenatally with vasa previa in the cohort and case-control studies was 11.0%. Data on chorionicity were available in only 34 cases and cervical length measurements were used by only the authors of 2 case reports and 4 cohort studies. Velamentous cord insertion was the most common additional ultrasound findings in twins presenting with vasa previa in both our series and the systematic review. CONCLUSION: Vasa previa is associated with specific prenatal and obstetric complications with different outcomes in singletons compared to twins. Data on the diagnosis and management of vasa previa in twin pregnancies are limited but there is enough evidence to warrant guidelines for targeted screening. To enable the development of efficient management protocols tailored to the need of individual cases, future studies of the screening, diagnosis, and management of vasa previa should be prospective and multicentric with detailed data on twins including chorionicity and use of cervical length.