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Outcome and management of pregnancies in severe chronic neutropenia patients by the European Branch of the Severe Chronic Neutropenia International Registry

C. Zeidler, UA. Grote, A. Nickel, B. Brand, G. Carlsson, E. Cortesão, C. Dufour, C. Duhem, G. Notheis, HA. Papadaki, H. Tamary, GE. Tjønnfjord, F. Tucci, J. Van Droogenbroeck, C. Vermylen, J. Voglova, B. Xicoy, K. Welte,

. 2014 ; 99 (8) : 1395-402.

Jazyk angličtina Země Itálie

Typ dokumentu časopisecké články

Perzistentní odkaz   https://www.medvik.cz/link/bmc15023278

Long-term granulocyte-colony stimulating factor treatment has been shown to be safe and effective in severe chronic neutropenia patients. However, data on its use during pregnancy are limited. To address this issue, we analyzed all pregnancies reported to the European branch of the Severe Chronic Neutropenia International Registry since 1994. A total of 38 pregnancies in 21 women with chronic neutropenia (16 pregnancies in 10 women with congenital, 10 in 6 women with cyclic, 12 in 5 women with idiopathic neutropenia) were reported. Granulocyte-colony stimulating factor was administered throughout pregnancy in 16 women and for at least one trimester in a further 5 women. No major differences were seen between treated and untreated women with respect to pregnancy outcome, newborn complications and infections. In addition, we evaluated the genetic transmission of known or suspected genetic defects in 16 mothers having 22 newborns as well as in 8 men fathering 15 children. As a proof of inheritance, neutropenia was passed on to the newborn in 58% from female and in 62% from male patients with ELANE mutations, but also to some newborns from parents with unknown gene mutation. Based on our results, granulocyte-colony stimulating factor therapy has been shown to be safe for mothers throughout pregnancies and for newborns without any signs of teratogenicity. With an increasing number of adult patients, genetic counseling prior to conception and supportive care of mothers during pregnancy are crucial. The acceptance of having affected children may reflect the high quality of life obtained due to this treatment.

Citace poskytuje Crossref.org

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$a Long-term granulocyte-colony stimulating factor treatment has been shown to be safe and effective in severe chronic neutropenia patients. However, data on its use during pregnancy are limited. To address this issue, we analyzed all pregnancies reported to the European branch of the Severe Chronic Neutropenia International Registry since 1994. A total of 38 pregnancies in 21 women with chronic neutropenia (16 pregnancies in 10 women with congenital, 10 in 6 women with cyclic, 12 in 5 women with idiopathic neutropenia) were reported. Granulocyte-colony stimulating factor was administered throughout pregnancy in 16 women and for at least one trimester in a further 5 women. No major differences were seen between treated and untreated women with respect to pregnancy outcome, newborn complications and infections. In addition, we evaluated the genetic transmission of known or suspected genetic defects in 16 mothers having 22 newborns as well as in 8 men fathering 15 children. As a proof of inheritance, neutropenia was passed on to the newborn in 58% from female and in 62% from male patients with ELANE mutations, but also to some newborns from parents with unknown gene mutation. Based on our results, granulocyte-colony stimulating factor therapy has been shown to be safe for mothers throughout pregnancies and for newborns without any signs of teratogenicity. With an increasing number of adult patients, genetic counseling prior to conception and supportive care of mothers during pregnancy are crucial. The acceptance of having affected children may reflect the high quality of life obtained due to this treatment.
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$a Grote, Ulrike A H $u Molecular Hematopoiesis, Hannover Medical School, Germany.
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$a Nickel, Anna $u Molecular Hematopoiesis, Hannover Medical School, Germany.
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$a Brand, Beate $u Molecular Hematopoiesis, Hannover Medical School, Germany.
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$a Carlsson, Göran $u Childhood Cancer Research Unit, Department of Women's and Children's Health, Karolinska University Hospital, Karolinska Institutet, Stockholm, Sweden.
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$a Cortesão, Emília $u Department of Hematology, Hospitais da Universidade de Coimbra, Portugal.
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$a Dufour, Carlo $u Hematology Unit, G. Gaslini Children's Institute, Genova, Italy.
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$a Duhem, Caroline $u Department of Hematology-Oncology, Centre Hospitalier de Luxembourg, Luxembourg.
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$a Notheis, Gundula $u Department for Pediatric Hematology/Oncology and Infection/Immunity, Dr. von Haunersches Kinderspital, Ludwig-Maximilians-University, Munich, Germany.
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$a Papadaki, Helen A $u Department of Hematology, University Hospital of Heraklion, Greece.
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$a Tamary, Hannah $u Pediatric Hematology Unit, Schneider Children's Medical Center of Israel, Petah Tikva, Israel.
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$a Tjønnfjord, Geir E $u Department of Haematology, Oslo University Hospital and Institute of Clinical Medicine, University of Oslo, Norway.
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$a Tucci, Fabio $u Department of Pediatric Onco-Hematology, AOU Meyer, Florence, Italy.
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$a Van Droogenbroeck, Jan $u Department of Hematology, A.Z. Sint-Jan, Brugge, Belgium.
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$a Vermylen, Christiane $u Department of Pediatric Hematology, Université Catholique de Louvain, Cliniques Universitaires Saint Luc, Brussels, Belgium.
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$a Voglova, Jaroslava $u 4 Department of Internal Medicine - Haematology, University Hospital, Hradec Králové, Czech Republic.
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$a Xicoy, Blanca $u Department of Hematology, Germans Trias i Pujol Hospital, Badalona, Spain.
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$a Welte, Karl $u Molecular Hematopoiesis, Hannover Medical School, Germany.
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