High risk of long‐term neurodevelopmental impairment in donor twins with spontaneous twin anemia–polycythemia sequence

Abstract

CONTRIBUTIONWhat are the novel findings of this work? Donor twins of pregnancies complicated by spontaneous twin anemia-polycythemia sequence (TAPS) have four-fold higher odds of neurodevelopmental impairment compared with their recipient cotwins, are at increased risk of cognitive delay and have a high rate of deafness. What are the clinical implications of this work?Based on our findings, we strongly advise that routine long-term follow-up, including testing for hearing loss, should be an essential part of care of twins diagnosed with spontaneous TAPS. ABSTRACTObjectives To evaluate the long-term neurodevelopmental and behavioral outcomes in surviving infants of pregnancies with spontaneous twin anemia-polycythemia sequence (TAPS), to compare outcome between donors and recipients, and to investigate potential risk factors for neurodevelopmental impairment (NDI).Methods This was a retrospective study of a consecutive cohort of spontaneous-TAPS survivors delivered between 2005 and 2017 at the . Neurological, motor, cognitive and behavioral development were assessed at a median age of 4 years. The primary outcome was NDI, which was a composite outcome of cerebral palsy, deafness, blindness and motor and/or cognitive delay. NDI was subdivided into two grades of severity: mild-to-moderate and severe Correspondence to: Ms L. NDI. Outcome was compared between surviving donor and recipient twins. Logistic regression analysis was used to assess risk factors for NDI.Results Forty-nine twin pregnancies complicated by spontaneous TAPS were eligible for inclusion. The perinatal survival rate was 83% (81/98) of twins. Neurodevelopmental assessment was performed in 91% (74/81) of surviving twins. NDI occurred in 30% (22/74) of TAPS survivors, and was found more often in donors (44%; 15/34) than in recipients (18%; 7/40) (odds ratio (OR), 4.1; 95% CI, 1.8-9.1; P = 0.001). Severe NDI was detected in 9% (7/74) of survivors and was higher in donors compared with recipients (18% (6/34) vs 3% (1/40)), although the difference did not reach statistical significance; P = 0.056). Donors demonstrated lower cognitive scores compared with recipients (P = 0.011). Bilateral deafness was identified in 15% (5/34) of donors compared with 0% (0/40) of recipients (P = 0.056). Parental concern regarding development was reported more often for donor than for recipient twins (P = 0.001). On multivariate analysis, independent risk factors for NDI were gestational age at delivery (OR, 0.7; 95% CI, 0.5-0.9; P = 0.003) and severe anemia (OR, 6.4; 95% CI,; P < 0.001).Conclusion Surviving donor twins of pregnancies complicated by spontaneous TAPS have four-fold higher odds of NDI compared with recipient cotwins, are at increased risk of cognitive delay and have a high rate of deafness.

Treatment and outcome of 370 cases with spontaneous or post‐laser twin anemia–polycythemia sequence managed in 17 fetal therapy centers

“…However, laser surgery was associated with a high diagnosis-to-birth interval, especially in comparison to treatment with IUT (± PET). As prematurity has a profound impact on short-and long-term health in TAPS twins, prolongation of pregnancy is of the utmost importance to improve outcome 6,7,10,11 . Notably, a comparable prolongation of pregnancy was achieved with selective feticide and expectant management.…”

“…Moreover, long-term outcome was not investigated in this study. Previous studies have shown that the detrimental effects of TAPS are not limited to the perinatal period, but also manifest later in life 6,7 . Therefore, the true effect of management for TAPS can only be properly investigated when TAPS cases are randomized between treatment groups, when stratification for risk factors is applied, and when long-term consequences are taken into account.…”

“…Options include expectant management, preterm delivery, intrauterine transfusion (IUT) in the donor with or without partial exchange transfusion (PET) in the recipient, fetoscopic laser surgery of the placental vascular anastomoses and selective feticide. Since TAPS is associated with high rates of adverse short-and long-term outcomes, it is crucial to investigate which management strategy offers TAPS twins the best outcome [6][7][8] . Unfortunately, due to the low incidence of the condition, studies are limited to small sample sizes, thus hampering generalizability of the results and necessitating caution when comparing the outcomes.…”

Performance of Antenatal Diagnostic Criteria of Twin-Anemia-Polycythemia Sequence

“…It has a reported incidence of spontaneous onset in 3-5% of MCDA twins, and 2-16% in cases after laser photocoagulation for TTTS [2][3][4][5][6]. It is known to be associated with increased perinatal morbidity and mortality [1], and neurodevelopmental impairment [7,8]. The optimal management of TAPS remains uncertain, but strategies varying from fetoscopic laser therapy, intrauterine transfusion with or without partial exchange transfusion, selective reduction, early delivery, to expectant management have been described [7,8].…”

Screening, Management and Delivery in Twin Pregnancy

“…▪ MC-Zwillinge mit TAPS haben ein erhöhtes Risiko für eine neurologische Entwicklungsverzögerung. ▪ Es wird eine zerebrale Bildgebung im dritten Trimenon sowie eine entwicklungsneurologische Untersuchung im Alter von 2 Jahren empfohlen[52,53] (▶ Tab. 2).…”