Andrew H. Chon

Fetal brain‐sparing after laser surgery for twin‐twin transfusion syndrome appears associated with two‐year neurodevelopmental outcomes

The cerebroplacental ratio (CPR) is a semi‐quantitative marker for fetal brain‐sparing. Our purpose was to measure the CPR at the time of treatment with selective laser photocoagulation of communicating vessels in gestations with twin‐twin transfusion syndrome (TTTS) to test its association with neurological outcomes at approximately 2 years.

Neonatal cerebral lesions predict 2-year neurodevelopmental impairment in children treated with laser surgery for twin–twin transfusion syndrome

Abstract Objective: The objective of this study is to assess whether postnatally detected cerebral abnormalities are predictive of neurodevelopmental impairment (NDI) in survivors of twin–twin transfusion syndrome (TTTS) that underwent laser surgery. Materials and methods: Ninety-nine children treated for TTTS had neurodevelopmental assessment at age 2-years (±6 weeks). ‘High-risk survivors’ had cerebral imaging in the neonatal period. ‘High-risk survivors’ were defined as (1) delivered at <32 weeks; or (2) cerebral imaging clinically indicated. NDI was a composite outcome of: Battelle Developmental Inventory 2nd edition (BDI-2) score <70, cerebral palsy, blindness, and/or deafness. Multilevel logistic regression with robust standard errors was used to evaluate associations between cerebral lesions and NDI. Results: Fifty-six children were ‘high-risk survivors’ and had neonatal cerebral imaging. Ten twins (18%) had at least one cerebral lesion, including grade 1–2 intraventricular hemorrhage (8), cystic periventricular leukomalacia (2), ventriculomegaly (1), and bilateral subependymal cyst (1). The risk of NDI in the ‘high-risk survivors’ was 7% (4/56) compared with 0% (0/43) in the remaining group. Among ‘high-risk survivors’, cerebral lesions were a significant risk factor for NDI (OR = 19.28, p < .001). Conclusions: Among ‘high-risk survivors’ of TTTS treated with laser surgery, cerebral lesions identified on neonatal imaging were associated with NDI at 2-years.

Amniopatch treatment of iatrogenic preterm premature rupture of membranes (iPPROM) after fetoscopic laser surgery for twin-twin transfusion syndrome.

Abstract Objectives: Amniopatch is a proposed treatment of iatrogenic preterm premature rupture of membranes (iPPROM). We studied characteristics associated with successful amniopatch treatment of iPPROM after fetoscopic laser surgery for twin–twin transfusion syndrome. Methods: Patients with iPPROM within 15 days of laser surgery treated with an amniopatch were studied. Factors associated with amniopatch success (i.e. cessation of leakage with normalization of amniotic fluid volume) were tested univariately and in multivariate logistic regression models. Continuous variables are expressed as median (range). Results: Of 1124 patients undergoing laser surgery, 19 (1.7%) had iPPROM and subsequent amniopatch. Twelve (63.2%) were successful. Latency in days from iPPROM to delivery was greater in the successful group (114.0 [87.0–141.0]) versus (44.0 [3.9–88.0], p = 0.0005), which translated into greater gestational age (GA) (weeks) at delivery (35.1 [30.9–39.4] versus 28.1 [22.0–31.0], p = 0.0005). The 30-day survival of the affected recipient twin was improved (100% versus 57.1%, p = 0.0361). After multivariate testing, GA < 20 weeks at the time of the amniopatch placement was the only variable that remained associated with successful sealing of the membranes. Conclusions: Treatment of iPPROM via amniopatch was successful in almost two-thirds of cases and was associated with higher GA at delivery and improved perinatal survival.

The impact of laser surgery on angiogenic and anti-angiogenic factors in twin–twin transfusion syndrome: a prospective study

Abstract Objective: To examine the effect of laser surgery on angiogenic and anti-angiogenic factors in patients with twin–twin transfusion syndrome (TTTS). Methods: Cases of TTTS and uncomplicated monochorionic diamniotic twin pregnancies between 16 and 26 weeks’ gestation were prospectively enrolled into the study. Maternal blood samples were obtained to measure angiogenic factors (vascular endothelial growth factor-A [VEGF], placental-derived growth factor [PlGF], and endothelin) and anti-angiogenic factors (soluble fms-like tyrosine kinase (sFlt-1), soluble endoglin (sEng), and sFlt-1/PlGF ratio). For cases, these factors were measured at visit 1 (pre-operatively), visit 2 (postoperative day one), and visit 3 (at least 3 weeks after surgery). In controls, the factors were measured at visit 1 (enrollment) and visit 2 (at least 3 weeks later). Levels of angiogenic and anti-angiogenic factors between cases and controls were compared. Results: At enrollment, the TTTS cases demonstrated an anti-angiogenic state with significantly higher sFlt-1, sEng, sFlt-1/PlGF ratio, and lower PlGF. Laser surgery, comparing visit 1–3, had a partial corrective effect on TTTS cases. sFlt-1 significantly decreased several weeks after surgery. The other factors (PlGF, endothelin, sFlt-1, sEng, and sFlt-1/PlGF ratio) were not statistically significantly different by visit 3. Conclusion: Laser surgery partially corrected the angiogenic profile in patients with TTTS.

Donor catch-up growth after laser surgery for twin–twin transfusion syndrome

OBJECTIVE To assess fetal growth after laser surgery for TTTS at the time of prenatal diagnosis, birth, and at 2years of age. DESIGN/METHODS Growth data were collected from surviving children treated between 2007 and 2010 as part of a study to assess neurodevelopment at 24months (±6weeks) corrected age. Fetal weights were obtained via ultrasound using Hadlocks formula at the time of preoperative assessment for laser surgery. Birth weights were recorded by the staff at the delivering institutions. Weights at 2years corrected age were recorded at the time of neurodevelopmental testing. Weights were converted into percentiles according to standard growth curves. Growth restriction was defined as <10th percentile for given age. Multilevel latent growth curve models in Mplus (twins nested in families) examined weight change over time as a function of donor status, and repeated measures ANOVA was utilized to assess in donor-recipient weight discordance over time for twin pairs. RESULTS 99 of 206 children (56 of 130 families) were studied. There were no differences between enrolled and non-enrolled patients in donor/recipient status and survival rates, fetal demise, intrauterine growth restriction, Quintero stage, and gestational age of surgery or delivery. 48.5% were donors. The median fetal, birth, and 2-year weights for all twins were 288g, 1.9kg, and 11.8kg, respectively, and the overall prevalence of growth restriction was 28%, 22%, and 3%, respectively. Growth restriction rates at prenatal diagnosis were 56% in donors vs. 2% in recipients (OR=64.3, p<0.001); at birth, 35% vs. 10% (OR=5.0, p<0.01); and at 2years, 6% vs. 0%. Donors showed significant gains in weight percentile (B=13.1, p<0.001) and a significant decrease in growth restriction rates over time (B=-1.6, p<0.001). Weight discordance between donor and recipient pairs also significantly decreased over time (linear F(1,42)=54.34, p<0.001). CONCLUSIONS After laser surgery for TTTS, donor twins exhibit significant catch-up growth by two years of age.

A Complication of Percutaneous Sclerotherapy for Congenital Pulmonary Airway Malformation: Intravascular Injection and Cardiac Necrosis

ABSTRACT Introduction: A congenital pulmonary airway malformation (CPAM) type III may become large enough to cause hydrops fetalis. In such circumstances, the fetus can be treated with open fetal resection, maternal betamethasone administration, or percutaneous sclerotherapy. Case report: A 24 week gestation fetus with a CPAM type III was treated by percutaneous sclerotherapy using ethanolamine oleate (EO). The EO inadvertently entered the left atrium and ventricle with subsequent fetal bradycardia and demise. Autopsy revealed myocardial necrosis. Conclusion: Percutaneous sclerotherapy has been previously described in the literature for the treatment of microcystic CPAMs with secondary hydrops. This is the first reported case of an adverse event after fetal sclerotherapy.

Incidental Septostomy after Laser Surgery for Twin-Twin Transfusion Syndrome: Perinatal Outcomes and Antenatal Management

Introduction: Incidental septostomy, or inadvertent perforation of the twins’ dividing membranes, can create a functional monoamniotic twin pregnancy. Our aim was to describe the perinatal outcomes and antenatal management of patients with incidental septostomy after laser surgery for twin-twin transfusion syndrome (TTTS). Materials and Methods: Monochorionic diamniotic multiple gestations with a gestational age (GA) between 16 and 26 weeks that underwent laser surgery from March 2006 to December 2015 were studied retrospectively. Perinatal outcomes were compared for patients with and without an incidental septostomy. A subgroup analysis was performed to evaluate antenatal management. Results: Of 532 TTTS patients who underwent laser surgery, incidental septostomy occurred in 34 (6.4%). Compared to all others, patients with a septostomy had an earlier median (range) GA at delivery (30.8 [18.6–37.9] vs. 34.0 [19.3–40.4] weeks, p < 0.0001) and lower 30-day survival of at least one twin (85.3 vs. 94.6%, p = 0.0452). We found no evidence that inpatient fetal surveillance was protective. Discussion: Incidental septostomy in TTTS patients treated via laser surgery appeared to be associated with lower GA at delivery and perinatal survival of at least one twin. Further study is required to determine the optimal antenatal surveillance strategy.

Types II and III congenital pulmonary airway malformation with hydrops treated in utero with percutaneous sclerotherapy

To present outcomes of fetuses with congenital pulmonary airway malformation (CPAM) treated with sclerotherapy.

The relationship between preoperative fetal head circumference and 2‐year cognitive performance after laser surgery for twin‐twin transfusion syndrome

To determine the relationship between preoperative fetal head circumference (HC) and cognitive performance among children treated with laser surgery for twin‐twin transfusion syndrome (TTTS).

Comparison of umbilical cord occlusion methods: Radiofrequency ablation versus laser photocoagulation

To compare outcomes between 2 umbilical cord occlusion (UCO) methods: radiofrequency ablation (RFA) versus fetoscopic‐guided laser photocoagulation.