Outcome of autoimmune cytopenia after hematopoietic cell transplantation in primary immunodeficiency.
Identifieur interne : 000076 ( Main/Exploration ); précédent : 000075; suivant : 000077Outcome of autoimmune cytopenia after hematopoietic cell transplantation in primary immunodeficiency.
Auteurs : Su Han Lum [Pays-Bas] ; Sabeena Selvarajah [Royaume-Uni] ; Angela Deya-Martinez [Royaume-Uni] ; Peter Mcnaughton [Royaume-Uni] ; Ali Sobh [Royaume-Uni] ; Sheila Waugh [Royaume-Uni] ; Shirelle Burton-Fanning [Pays-Bas] ; Lisa Newton [Royaume-Uni] ; Julie Gandy [Royaume-Uni] ; Zohreh Nademi [Royaume-Uni] ; Stephen Owens [Royaume-Uni] ; Eleri Williams [Royaume-Uni] ; Marieke Emonts [Royaume-Uni] ; Terry Flood [Royaume-Uni] ; Andrew Cant [Royaume-Uni] ; Mario Abinun [Royaume-Uni] ; Sophie Hambleton [Royaume-Uni] ; Andrew R. Gennery [Royaume-Uni] ; Mary Slatter [Royaume-Uni]Source :
- The Journal of allergy and clinical immunology [ 1097-6825 ] ; 2020.
Abstract
BACKGROUND
Post hematopoietic cell transplantation (HCT) autoimmune cytopenia (AIC) is a potentially life-threatening complication, but studies focusing on large cohorts of patients transplanted for primary immunodeficiency are lacking.
OBJECTIVES
This study sought to determine the incidence, risk factors, and outcomes of post-HCT AIC and B-lymphocyte function following rituximab.
METHODS
We retrospectively studied 502 children with primary immunodeficiency who were transplanted at our center between 1987 and 2018.
RESULTS
Thirty-six patients (9%) developed post-HCT AIC, with a median onset of 6.5 months post-HCT. On univariate analysis, pre-HCT AIC, mismatched donor, alemtuzumab, anti-thymocyte antiglobulin, and acute and chronic graft versus host disease were significantly associated with post-HCT AIC. After multivariate analysis, alemtuzumab (subdistribution hazard ratio, 9.0; 95% CI, 1.50-54.0; P = .02) was independently associated with post-HCT AIC. Corticosteroid and high-dose intravenous immunoglobulin achieved remission in 50% (n = 18), additional rituximab led to remission in 25% (n = 9), and the remaining 25% were treated with a combination of various modalities including sirolimus (n = 5), bortezomib (n = 3), mycophenolate mofetil (n = 2), splenectomy (n = 2), and second HCT (n = 3). The mortality of post-HCT AIC reduced from 25% (4 of 16) prior to 2011 to 5% (1 of 20) after 2011. The median follow-up of 5.8 years (range, 0.4 to 29.1 years) showed that 26 of 30 survivors (87%) were in complete remission, and 4 were in remission with ongoing sirolimus and low-dose steroids. Of the 17 who received rituximab, 7 had B-lymphocyte recovery, 5 had persistent low B-lymphocyte count and remained on intravenous immunoglobulin replacement, 2 had second HCT, and 3 died.
CONCLUSIONS
The frequency of post HCT AIC in our cohort was 9%, and the most significant risk factors for its occurrence were the presence of graft versus host disease and the use of alemtuzumab.
DOI: 10.1016/j.jaci.2020.04.053
PubMed: 32442647
Affiliations:
Links toward previous steps (curation, corpus...)
Le document en format XML
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<author><name sortKey="Lum, Su Han" sort="Lum, Su Han" uniqKey="Lum S" first="Su Han" last="Lum">Su Han Lum</name>
<affiliation wicri:level="3"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Department of Paediatrics, Leiden University Medical Centre, Leiden, The Netherlands. Electronic address: s.lum@nhs.net.</nlm:affiliation>
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<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Department of Paediatrics, Leiden University Medical Centre, Leiden</wicri:regionArea>
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<author><name sortKey="Selvarajah, Sabeena" sort="Selvarajah, Sabeena" uniqKey="Selvarajah S" first="Sabeena" last="Selvarajah">Sabeena Selvarajah</name>
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<author><name sortKey="Deya Martinez, Angela" sort="Deya Martinez, Angela" uniqKey="Deya Martinez A" first="Angela" last="Deya-Martinez">Angela Deya-Martinez</name>
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<author><name sortKey="Mcnaughton, Peter" sort="Mcnaughton, Peter" uniqKey="Mcnaughton P" first="Peter" last="Mcnaughton">Peter Mcnaughton</name>
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<author><name sortKey="Sobh, Ali" sort="Sobh, Ali" uniqKey="Sobh A" first="Ali" last="Sobh">Ali Sobh</name>
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<author><name sortKey="Waugh, Sheila" sort="Waugh, Sheila" uniqKey="Waugh S" first="Sheila" last="Waugh">Sheila Waugh</name>
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<author><name sortKey="Burton Fanning, Shirelle" sort="Burton Fanning, Shirelle" uniqKey="Burton Fanning S" first="Shirelle" last="Burton-Fanning">Shirelle Burton-Fanning</name>
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<author><name sortKey="Newton, Lisa" sort="Newton, Lisa" uniqKey="Newton L" first="Lisa" last="Newton">Lisa Newton</name>
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<author><name sortKey="Gandy, Julie" sort="Gandy, Julie" uniqKey="Gandy J" first="Julie" last="Gandy">Julie Gandy</name>
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<author><name sortKey="Nademi, Zohreh" sort="Nademi, Zohreh" uniqKey="Nademi Z" first="Zohreh" last="Nademi">Zohreh Nademi</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
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<author><name sortKey="Owens, Stephen" sort="Owens, Stephen" uniqKey="Owens S" first="Stephen" last="Owens">Stephen Owens</name>
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</affiliation>
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<author><name sortKey="Williams, Eleri" sort="Williams, Eleri" uniqKey="Williams E" first="Eleri" last="Williams">Eleri Williams</name>
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<author><name sortKey="Emonts, Marieke" sort="Emonts, Marieke" uniqKey="Emonts M" first="Marieke" last="Emonts">Marieke Emonts</name>
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<author><name sortKey="Flood, Terry" sort="Flood, Terry" uniqKey="Flood T" first="Terry" last="Flood">Terry Flood</name>
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</affiliation>
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<author><name sortKey="Cant, Andrew" sort="Cant, Andrew" uniqKey="Cant A" first="Andrew" last="Cant">Andrew Cant</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
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<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
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</affiliation>
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<author><name sortKey="Abinun, Mario" sort="Abinun, Mario" uniqKey="Abinun M" first="Mario" last="Abinun">Mario Abinun</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
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<author><name sortKey="Hambleton, Sophie" sort="Hambleton, Sophie" uniqKey="Hambleton S" first="Sophie" last="Hambleton">Sophie Hambleton</name>
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<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
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<author><name sortKey="Gennery, Andrew R" sort="Gennery, Andrew R" uniqKey="Gennery A" first="Andrew R" last="Gennery">Andrew R. Gennery</name>
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<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Slatter, Mary" sort="Slatter, Mary" uniqKey="Slatter M" first="Mary" last="Slatter">Mary Slatter</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
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</titleStmt>
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<date when="2020">2020</date>
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<idno type="pmid">32442647</idno>
<idno type="doi">10.1016/j.jaci.2020.04.053</idno>
<idno type="wicri:Area/Main/Corpus">000071</idno>
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<sourceDesc><biblStruct><analytic><title xml:lang="en">Outcome of autoimmune cytopenia after hematopoietic cell transplantation in primary immunodeficiency.</title>
<author><name sortKey="Lum, Su Han" sort="Lum, Su Han" uniqKey="Lum S" first="Su Han" last="Lum">Su Han Lum</name>
<affiliation wicri:level="3"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Department of Paediatrics, Leiden University Medical Centre, Leiden, The Netherlands. Electronic address: s.lum@nhs.net.</nlm:affiliation>
<country xml:lang="fr">Pays-Bas</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Department of Paediatrics, Leiden University Medical Centre, Leiden</wicri:regionArea>
<placeName><settlement type="city">Leyde</settlement>
<region nuts="2" type="province">Hollande-Méridionale</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Selvarajah, Sabeena" sort="Selvarajah, Sabeena" uniqKey="Selvarajah S" first="Sabeena" last="Selvarajah">Sabeena Selvarajah</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Deya Martinez, Angela" sort="Deya Martinez, Angela" uniqKey="Deya Martinez A" first="Angela" last="Deya-Martinez">Angela Deya-Martinez</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Mcnaughton, Peter" sort="Mcnaughton, Peter" uniqKey="Mcnaughton P" first="Peter" last="Mcnaughton">Peter Mcnaughton</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Sobh, Ali" sort="Sobh, Ali" uniqKey="Sobh A" first="Ali" last="Sobh">Ali Sobh</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Waugh, Sheila" sort="Waugh, Sheila" uniqKey="Waugh S" first="Sheila" last="Waugh">Sheila Waugh</name>
<affiliation wicri:level="1"><nlm:affiliation>Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Burton Fanning, Shirelle" sort="Burton Fanning, Shirelle" uniqKey="Burton Fanning S" first="Shirelle" last="Burton-Fanning">Shirelle Burton-Fanning</name>
<affiliation wicri:level="3"><nlm:affiliation>Department of Paediatrics, Leiden University Medical Centre, Leiden, The Netherlands.</nlm:affiliation>
<country xml:lang="fr">Pays-Bas</country>
<wicri:regionArea>Department of Paediatrics, Leiden University Medical Centre, Leiden</wicri:regionArea>
<placeName><settlement type="city">Leyde</settlement>
<region nuts="2" type="province">Hollande-Méridionale</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Newton, Lisa" sort="Newton, Lisa" uniqKey="Newton L" first="Lisa" last="Newton">Lisa Newton</name>
<affiliation wicri:level="1"><nlm:affiliation>Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Gandy, Julie" sort="Gandy, Julie" uniqKey="Gandy J" first="Julie" last="Gandy">Julie Gandy</name>
<affiliation wicri:level="1"><nlm:affiliation>Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Nademi, Zohreh" sort="Nademi, Zohreh" uniqKey="Nademi Z" first="Zohreh" last="Nademi">Zohreh Nademi</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Owens, Stephen" sort="Owens, Stephen" uniqKey="Owens S" first="Stephen" last="Owens">Stephen Owens</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Williams, Eleri" sort="Williams, Eleri" uniqKey="Williams E" first="Eleri" last="Williams">Eleri Williams</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Emonts, Marieke" sort="Emonts, Marieke" uniqKey="Emonts M" first="Marieke" last="Emonts">Marieke Emonts</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Flood, Terry" sort="Flood, Terry" uniqKey="Flood T" first="Terry" last="Flood">Terry Flood</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Cant, Andrew" sort="Cant, Andrew" uniqKey="Cant A" first="Andrew" last="Cant">Andrew Cant</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Abinun, Mario" sort="Abinun, Mario" uniqKey="Abinun M" first="Mario" last="Abinun">Mario Abinun</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Hambleton, Sophie" sort="Hambleton, Sophie" uniqKey="Hambleton S" first="Sophie" last="Hambleton">Sophie Hambleton</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Gennery, Andrew R" sort="Gennery, Andrew R" uniqKey="Gennery A" first="Andrew R" last="Gennery">Andrew R. Gennery</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Slatter, Mary" sort="Slatter, Mary" uniqKey="Slatter M" first="Mary" last="Slatter">Mary Slatter</name>
<affiliation wicri:level="1"><nlm:affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne</wicri:regionArea>
<wicri:noRegion>Newcastle upon Tyne</wicri:noRegion>
</affiliation>
</author>
</analytic>
<series><title level="j">The Journal of allergy and clinical immunology</title>
<idno type="eISSN">1097-6825</idno>
<imprint><date when="2020" type="published">2020</date>
</imprint>
</series>
</biblStruct>
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<front><div type="abstract" xml:lang="en"><p><b>BACKGROUND</b>
</p>
<p>Post hematopoietic cell transplantation (HCT) autoimmune cytopenia (AIC) is a potentially life-threatening complication, but studies focusing on large cohorts of patients transplanted for primary immunodeficiency are lacking.</p>
</div>
<div type="abstract" xml:lang="en"><p><b>OBJECTIVES</b>
</p>
<p>This study sought to determine the incidence, risk factors, and outcomes of post-HCT AIC and B-lymphocyte function following rituximab.</p>
</div>
<div type="abstract" xml:lang="en"><p><b>METHODS</b>
</p>
<p>We retrospectively studied 502 children with primary immunodeficiency who were transplanted at our center between 1987 and 2018.</p>
</div>
<div type="abstract" xml:lang="en"><p><b>RESULTS</b>
</p>
<p>Thirty-six patients (9%) developed post-HCT AIC, with a median onset of 6.5 months post-HCT. On univariate analysis, pre-HCT AIC, mismatched donor, alemtuzumab, anti-thymocyte antiglobulin, and acute and chronic graft versus host disease were significantly associated with post-HCT AIC. After multivariate analysis, alemtuzumab (subdistribution hazard ratio, 9.0; 95% CI, 1.50-54.0; P = .02) was independently associated with post-HCT AIC. Corticosteroid and high-dose intravenous immunoglobulin achieved remission in 50% (n = 18), additional rituximab led to remission in 25% (n = 9), and the remaining 25% were treated with a combination of various modalities including sirolimus (n = 5), bortezomib (n = 3), mycophenolate mofetil (n = 2), splenectomy (n = 2), and second HCT (n = 3). The mortality of post-HCT AIC reduced from 25% (4 of 16) prior to 2011 to 5% (1 of 20) after 2011. The median follow-up of 5.8 years (range, 0.4 to 29.1 years) showed that 26 of 30 survivors (87%) were in complete remission, and 4 were in remission with ongoing sirolimus and low-dose steroids. Of the 17 who received rituximab, 7 had B-lymphocyte recovery, 5 had persistent low B-lymphocyte count and remained on intravenous immunoglobulin replacement, 2 had second HCT, and 3 died.</p>
</div>
<div type="abstract" xml:lang="en"><p><b>CONCLUSIONS</b>
</p>
<p>The frequency of post HCT AIC in our cohort was 9%, and the most significant risk factors for its occurrence were the presence of graft versus host disease and the use of alemtuzumab.</p>
</div>
</front>
</TEI>
<pubmed><MedlineCitation Status="In-Data-Review" Owner="NLM"><PMID Version="1">32442647</PMID>
<DateRevised><Year>2020</Year>
<Month>08</Month>
<Day>10</Day>
</DateRevised>
<Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1097-6825</ISSN>
<JournalIssue CitedMedium="Internet"><Volume>146</Volume>
<Issue>2</Issue>
<PubDate><Year>2020</Year>
<Month>Aug</Month>
</PubDate>
</JournalIssue>
<Title>The Journal of allergy and clinical immunology</Title>
<ISOAbbreviation>J Allergy Clin Immunol</ISOAbbreviation>
</Journal>
<ArticleTitle>Outcome of autoimmune cytopenia after hematopoietic cell transplantation in primary immunodeficiency.</ArticleTitle>
<Pagination><MedlinePgn>406-416</MedlinePgn>
</Pagination>
<ELocationID EIdType="pii" ValidYN="Y">S0091-6749(20)30682-5</ELocationID>
<ELocationID EIdType="doi" ValidYN="Y">10.1016/j.jaci.2020.04.053</ELocationID>
<Abstract><AbstractText Label="BACKGROUND" NlmCategory="BACKGROUND">Post hematopoietic cell transplantation (HCT) autoimmune cytopenia (AIC) is a potentially life-threatening complication, but studies focusing on large cohorts of patients transplanted for primary immunodeficiency are lacking.</AbstractText>
<AbstractText Label="OBJECTIVES" NlmCategory="OBJECTIVE">This study sought to determine the incidence, risk factors, and outcomes of post-HCT AIC and B-lymphocyte function following rituximab.</AbstractText>
<AbstractText Label="METHODS" NlmCategory="METHODS">We retrospectively studied 502 children with primary immunodeficiency who were transplanted at our center between 1987 and 2018.</AbstractText>
<AbstractText Label="RESULTS" NlmCategory="RESULTS">Thirty-six patients (9%) developed post-HCT AIC, with a median onset of 6.5 months post-HCT. On univariate analysis, pre-HCT AIC, mismatched donor, alemtuzumab, anti-thymocyte antiglobulin, and acute and chronic graft versus host disease were significantly associated with post-HCT AIC. After multivariate analysis, alemtuzumab (subdistribution hazard ratio, 9.0; 95% CI, 1.50-54.0; P = .02) was independently associated with post-HCT AIC. Corticosteroid and high-dose intravenous immunoglobulin achieved remission in 50% (n = 18), additional rituximab led to remission in 25% (n = 9), and the remaining 25% were treated with a combination of various modalities including sirolimus (n = 5), bortezomib (n = 3), mycophenolate mofetil (n = 2), splenectomy (n = 2), and second HCT (n = 3). The mortality of post-HCT AIC reduced from 25% (4 of 16) prior to 2011 to 5% (1 of 20) after 2011. The median follow-up of 5.8 years (range, 0.4 to 29.1 years) showed that 26 of 30 survivors (87%) were in complete remission, and 4 were in remission with ongoing sirolimus and low-dose steroids. Of the 17 who received rituximab, 7 had B-lymphocyte recovery, 5 had persistent low B-lymphocyte count and remained on intravenous immunoglobulin replacement, 2 had second HCT, and 3 died.</AbstractText>
<AbstractText Label="CONCLUSIONS" NlmCategory="CONCLUSIONS">The frequency of post HCT AIC in our cohort was 9%, and the most significant risk factors for its occurrence were the presence of graft versus host disease and the use of alemtuzumab.</AbstractText>
<CopyrightInformation>Crown Copyright © 2020. Published by Elsevier Inc. All rights reserved.</CopyrightInformation>
</Abstract>
<AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Lum</LastName>
<ForeName>Su Han</ForeName>
<Initials>SH</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Department of Paediatrics, Leiden University Medical Centre, Leiden, The Netherlands. Electronic address: s.lum@nhs.net.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Selvarajah</LastName>
<ForeName>Sabeena</ForeName>
<Initials>S</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Deya-Martinez</LastName>
<ForeName>Angela</ForeName>
<Initials>A</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>McNaughton</LastName>
<ForeName>Peter</ForeName>
<Initials>P</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Sobh</LastName>
<ForeName>Ali</ForeName>
<Initials>A</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Waugh</LastName>
<ForeName>Sheila</ForeName>
<Initials>S</Initials>
<AffiliationInfo><Affiliation>Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Burton-Fanning</LastName>
<ForeName>Shirelle</ForeName>
<Initials>S</Initials>
<AffiliationInfo><Affiliation>Department of Paediatrics, Leiden University Medical Centre, Leiden, The Netherlands.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Newton</LastName>
<ForeName>Lisa</ForeName>
<Initials>L</Initials>
<AffiliationInfo><Affiliation>Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Gandy</LastName>
<ForeName>Julie</ForeName>
<Initials>J</Initials>
<AffiliationInfo><Affiliation>Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Nademi</LastName>
<ForeName>Zohreh</ForeName>
<Initials>Z</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Owens</LastName>
<ForeName>Stephen</ForeName>
<Initials>S</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Williams</LastName>
<ForeName>Eleri</ForeName>
<Initials>E</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Emonts</LastName>
<ForeName>Marieke</ForeName>
<Initials>M</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Flood</LastName>
<ForeName>Terry</ForeName>
<Initials>T</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Cant</LastName>
<ForeName>Andrew</ForeName>
<Initials>A</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Abinun</LastName>
<ForeName>Mario</ForeName>
<Initials>M</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Microbiology and Virology, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Hambleton</LastName>
<ForeName>Sophie</ForeName>
<Initials>S</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Gennery</LastName>
<ForeName>Andrew R</ForeName>
<Initials>AR</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
<Author ValidYN="Y"><LastName>Slatter</LastName>
<ForeName>Mary</ForeName>
<Initials>M</Initials>
<AffiliationInfo><Affiliation>Children's Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom; Translational and Clinical Research Institute, Newcastle upon Tyne Hospital National Health System Foundation Trust, Newcastle upon Tyne, United Kingdom.</Affiliation>
</AffiliationInfo>
</Author>
</AuthorList>
<Language>eng</Language>
<PublicationTypeList><PublicationType UI="D016428">Journal Article</PublicationType>
</PublicationTypeList>
<ArticleDate DateType="Electronic"><Year>2020</Year>
<Month>05</Month>
<Day>19</Day>
</ArticleDate>
</Article>
<MedlineJournalInfo><Country>United States</Country>
<MedlineTA>J Allergy Clin Immunol</MedlineTA>
<NlmUniqueID>1275002</NlmUniqueID>
<ISSNLinking>0091-6749</ISSNLinking>
</MedlineJournalInfo>
<CitationSubset>AIM</CitationSubset>
<CitationSubset>IM</CitationSubset>
<KeywordList Owner="NOTNLM"><Keyword MajorTopicYN="N">pediatrics</Keyword>
<Keyword MajorTopicYN="N">posttransplant autoimmune cytopenia</Keyword>
<Keyword MajorTopicYN="N">primary immunodeficiency</Keyword>
</KeywordList>
</MedlineCitation>
<PubmedData><History><PubMedPubDate PubStatus="received"><Year>2019</Year>
<Month>10</Month>
<Day>06</Day>
</PubMedPubDate>
<PubMedPubDate PubStatus="revised"><Year>2020</Year>
<Month>04</Month>
<Day>02</Day>
</PubMedPubDate>
<PubMedPubDate PubStatus="accepted"><Year>2020</Year>
<Month>04</Month>
<Day>09</Day>
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<affiliations><list><country><li>Pays-Bas</li>
<li>Royaume-Uni</li>
</country>
<region><li>Hollande-Méridionale</li>
</region>
<settlement><li>Leyde</li>
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</list>
<tree><country name="Pays-Bas"><region name="Hollande-Méridionale"><name sortKey="Lum, Su Han" sort="Lum, Su Han" uniqKey="Lum S" first="Su Han" last="Lum">Su Han Lum</name>
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<name sortKey="Burton Fanning, Shirelle" sort="Burton Fanning, Shirelle" uniqKey="Burton Fanning S" first="Shirelle" last="Burton-Fanning">Shirelle Burton-Fanning</name>
</country>
<country name="Royaume-Uni"><noRegion><name sortKey="Selvarajah, Sabeena" sort="Selvarajah, Sabeena" uniqKey="Selvarajah S" first="Sabeena" last="Selvarajah">Sabeena Selvarajah</name>
</noRegion>
<name sortKey="Abinun, Mario" sort="Abinun, Mario" uniqKey="Abinun M" first="Mario" last="Abinun">Mario Abinun</name>
<name sortKey="Cant, Andrew" sort="Cant, Andrew" uniqKey="Cant A" first="Andrew" last="Cant">Andrew Cant</name>
<name sortKey="Deya Martinez, Angela" sort="Deya Martinez, Angela" uniqKey="Deya Martinez A" first="Angela" last="Deya-Martinez">Angela Deya-Martinez</name>
<name sortKey="Emonts, Marieke" sort="Emonts, Marieke" uniqKey="Emonts M" first="Marieke" last="Emonts">Marieke Emonts</name>
<name sortKey="Flood, Terry" sort="Flood, Terry" uniqKey="Flood T" first="Terry" last="Flood">Terry Flood</name>
<name sortKey="Gandy, Julie" sort="Gandy, Julie" uniqKey="Gandy J" first="Julie" last="Gandy">Julie Gandy</name>
<name sortKey="Gennery, Andrew R" sort="Gennery, Andrew R" uniqKey="Gennery A" first="Andrew R" last="Gennery">Andrew R. Gennery</name>
<name sortKey="Hambleton, Sophie" sort="Hambleton, Sophie" uniqKey="Hambleton S" first="Sophie" last="Hambleton">Sophie Hambleton</name>
<name sortKey="Mcnaughton, Peter" sort="Mcnaughton, Peter" uniqKey="Mcnaughton P" first="Peter" last="Mcnaughton">Peter Mcnaughton</name>
<name sortKey="Nademi, Zohreh" sort="Nademi, Zohreh" uniqKey="Nademi Z" first="Zohreh" last="Nademi">Zohreh Nademi</name>
<name sortKey="Newton, Lisa" sort="Newton, Lisa" uniqKey="Newton L" first="Lisa" last="Newton">Lisa Newton</name>
<name sortKey="Owens, Stephen" sort="Owens, Stephen" uniqKey="Owens S" first="Stephen" last="Owens">Stephen Owens</name>
<name sortKey="Slatter, Mary" sort="Slatter, Mary" uniqKey="Slatter M" first="Mary" last="Slatter">Mary Slatter</name>
<name sortKey="Sobh, Ali" sort="Sobh, Ali" uniqKey="Sobh A" first="Ali" last="Sobh">Ali Sobh</name>
<name sortKey="Waugh, Sheila" sort="Waugh, Sheila" uniqKey="Waugh S" first="Sheila" last="Waugh">Sheila Waugh</name>
<name sortKey="Williams, Eleri" sort="Williams, Eleri" uniqKey="Williams E" first="Eleri" last="Williams">Eleri Williams</name>
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