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BMC Pregnancy and Childbirth
Published in: BMC Pregnancy and Childbirth 1/2010

Open Access 01-12-2010 | Study protocol

Long-Term follow up after intra-Uterine transfusionS; the LOTUS study

Authors: Esther P Verduin, Irene TM Lindenburg, Vivianne EHJ Smits-Wintjens, Jeanine MM van Klink, Henk Schonewille, Inge L van Kamp, Dick Oepkes, Frans J Walther, Humphrey HH Kanhai, Ilias IN Doxiadis, Enrico Lopriore, Anneke Brand

Published in: BMC Pregnancy and Childbirth | Issue 1/2010

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Abstract

Background

The Leiden University Medical Center (LUMC) is the Dutch national referral centre for pregnancies complicated by haemolytic disease of the fetus and newborn (HDFN) caused by maternal alloimmunization. Yearly, 20-25 affected fetuses with severe anaemia are transfused with intra-uterine blood transfusions (IUT). Mothers of whom their fetus has undergone IUT for HDFN are considered high responders with regard to red blood cell (RBC) antibody formation. Most study groups report high perinatal survival, resulting in a shift in attention towards short- and long-term outcome in surviving children.

Methods/Design

We set up a large long-term observational follow-up study (LOTUS study), in cooperation with the Sanquin Blood Supply Foundation and the LUMC departments of Obstetrics, Neonatology and ImmunoHematology & Bloodtransfusion.
The first part of this study addresses several putative mechanisms associated with blood group alloimmunization in these mothers. The second part of this study determines the incidence of long-term neurodevelopment impairment (NDI) and associated risk factors in children treated with IUT. All women and their life offspring who have been treated with IUT for HDFN in the LUMC from 1987-2008 are invited to participate and after consent, blood or saliva samples are taken. RBC and HLA antigen profile and antibodies are determined by serologic or molecular techniques. Microchimerism populations are tested by real time polymerase chain reaction (RT PCR).
All children are tested for their neurological, cognitive and psychosocial development using standardised tests and questionnaires. The primary outcome is neurodevelopmental impairment (NDI), a composite outcome defined as any of the following: cerebral palsy, cognitive or psychomotor development < 2 standard deviation, bilateral blindness and/or bilateral deafness.

Discussion

The LOTUS study includes the largest cohort of IUT patients ever studied and is the first to investigate post-IUT long-term effects in both mother and child. The results may lead to a change in transfusion policy, in particular future avoidance of certain incompatibilities. Additionally the LOTUS study will provide clinicians and parents better insights in the long-term neurodevelopmental outcome in children with HDFN treated with IUTs, and may improve the quality of antenatal counselling and long-term guidance.
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Metadata
Title
Long-Term follow up after intra-Uterine transfusionS; the LOTUS study
Authors
Esther P Verduin
Irene TM Lindenburg
Vivianne EHJ Smits-Wintjens
Jeanine MM van Klink
Henk Schonewille
Inge L van Kamp
Dick Oepkes
Frans J Walther
Humphrey HH Kanhai
Ilias IN Doxiadis
Enrico Lopriore
Anneke Brand
Publication date
01-12-2010
Publisher
BioMed Central
Published in
BMC Pregnancy and Childbirth / Issue 1/2010
Electronic ISSN: 1471-2393
DOI
https://doi.org/10.1186/1471-2393-10-77

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