Developmental outcomes for survivors of placental laser photocoagulation for the management of twin-to-twin transfusion syndrome.

BACKGROUND: Fetoscopic laser coagulation of placental anastomoses reverses the pathological process in twin-to-twin transfusion syndrome, thereby increasing survival, but there are a paucity of studies addressing long-term neurodevelopmental outcome of survivors. This study aimed to ascertain the presence of neurodevelopmental disabilities in child survivors of monochorionic pregnancies managed by placental laser photocoagulation in the Australian state of Victoria. METHODS: All pregnancies undergoing placental laser photocoagulation with the Victorian Fetal Therapy Service between 2006-2017 were included. Information on each surviving child, including demographics, perinatal course, and developmental progress was collected from parents, and consent was sought to complete the Child Behaviour Checklist. Interviewers evaluated whether this information was consistent with a diagnosis of any of 14 neurodevelopmental conditions. A three-tiered outcome measure was allocated for each child: (1) unimpaired or developmentally normal, (2) mild or moderate neurological impairment, or (3) severe neurological impairment. Clinical predictors for adverse outcome were identified. RESULTS: Of 116 pregnancies (113 twin, 3 triplet), 96 (83%) resulted in 1 + surviving fetuses. 57/113 (50%) twin pregnancies resulted in 2 survivors, 36 (32%) in 1 survivor, and 20 (18%) in no survivors. Of the 235 fetuses, 154 (65.5%) survived to follow-up. Survival increased from 59% in 2006-2008 to 73% in 2015-2017. 90/154 (58%) survivors were followed up at a mean age of 7.5 [SD 3.0] years. Based on parental interview and Child Behaviour Checklist data, 28/90 (31%) participants were assessed as having neurodevelopmental impairment, 27 of mild-moderate severity and 1 severe. Speech/language disorders, attention deficit (hyperactivity) disorders, and fine motor impairment were most common. Neonatal length of stay conferred the highest risk of impairment. CONCLUSION: Substantial variation exists between fetal therapy services in the type and length of neonatal follow-up following fetoscopic laser coagulation, contributing to a lack of data on long-term outcomes. The findings from this study support increasingly urgent calls to undertake systematic and sustained follow-up of fetoscopic laser coagulation survivors until school age. Information from this study may assist parents in their decision-making when offered fetal surgery. Importantly, it highlights a group for targeted surveillance and early intervention.

Twin-to-twin transfusion syndrome neurodevelopmental follow-up study (neurodevelopmental outcomes for children whose twin-to-twin transfusion syndrome was treated with placental laser photocoagulation).

BACKGROUND: Twin-to-twin transfusion syndrome (TTTS) is a serious complication of 10-15% of twin or triplet pregnancies in which multiple fetuses share a single placenta. Communicating placental vessels allow one fetus (the donor) to pump blood to the other (the recipient). Mortality rates without intervention are high, approaching 100% in some series, with fetal deaths usually due to cardiac failure. Surgical correction using laser photocoagulation of communicating placental vessels was developed in the 1980s and refined in the 1990s. Since it was introduced in Victoria in 2006, laser surgery has been performed in approximately 120 pregnancies. Survival of one or more fetuses following laser surgery is currently > 90%, however the neurodevelopmental outcomes for survivors remain incompletely understood. Prior to laser therapy, at least one in five survivors of TTTS had serious adverse neurodevelopmental outcomes (usually cerebral palsy). Current estimates of neurological impairment among survivors following laser surgery vary from 4 to 31% and long-term follow-up data are limited. METHODS: This paper describes the methodology for a retrospective cohort study in which children aged 24 months and over (corrected for prematurity), who were treated with laser placental photocoagulation for TTTS at Monash Health in Victoria, Australia, will undergo comprehensive neurodevelopmental assessment by a multidisciplinary team. Evaluation will include parental completion of pre-assessment questionnaires of social and behavioural development, a standardised medical assessment by a developmental paediatrician or paediatric neurologist, and age-appropriate cognitive and academic, speech and fine and gross motor assessments by psychologists, speech and occupational therapists or physiotherapists. Assessments will be undertaken at the Murdoch Children's Research Institute/Royal Children's Hospital, at Monash Health or at another mutually agreed location. Results will be recorded in a secure online database which will facilitate future related research. DISCUSSION: This will be the first study to report and evaluate neurodevelopmental outcomes following laser surgery for twin-to-twin transfusion syndrome in Victoria, and will inform clinical practice regarding follow-up of children at risk of adverse outcomes.

Multiple birth families with children with special needs: a qualitative investigation of mothers' experiences.

Multiple birth remains prevalent, with prematurity and subsequent disability being common complications. However, little is known of the experiences of mothers living with the combined circumstances of multiple birth, prematurity, and special needs. This paper reports an exploratory study using the qualitative technique of thematic analysis, to describe and interpret the experiences of 10 mothers of prematurely born multiple birth children with diverse special needs. Mothers were shocked to learn they were carrying multiple fetuses, including those who underwent in vitro fertilization with dual embryo transfer. Most experienced protracted concern over one or more babies' survival during pregnancy, and prescribed bed rest was frequently associated with increased anxiety and other adverse psychological effects. Some experienced disenfranchised grief, such as those grieving the opportunity to bear a single child. The contrast with experiences of mothers of healthy, term singletons caused considerable distress. Feelings of detachment and unreality were common in the immediate postpartum period, possibly due to transient depersonalization. Having more than one newborn created practical and psychological problems during the neonatal period, particularly when infants were separated due to differences in medical status. The extent to which hospitals accommodated the multiple birth relationship varied and significantly affected mothers' postnatal experiences. Mothers often felt guilty, particularly regarding inequality of care and attention they were able to provide to each child. This was especially problematic for multiples discordant for special needs status. The presence of one normally developing child complicated adjustment to the other's difficulties, and several experienced chronic sorrow. Serious maternal mental health difficulties were common but not universal. Available formal supports were generally perceived as inadequate, addressing some, but not all, of the mothers' needs. Further work is needed to advance understanding of the relationships between mothers and their multiples, and to explore the implications of special needs within multiple birth families.