Dian Winkelhorst1, Marije M Kamphuis1, Liselotte C de Kloet2, Jaap Jan Zwaginga3,4, Dick Oepkes1, Enrico Lopriore2. 1. Department of Obstetrics, Leiden University Medical Center. 2. Division of Neonatology, Department of Pediatrics, Leiden University Medical Center. 3. Jon J. Van Rood Center for Clinical Transfusion Research, Sanquin Blood Supply Foundation. 4. Department of Immunohematology and Blood Transfusion, Leiden University Medical Center, Leiden, The Netherlands.
Abstract
BACKGROUND: The most feared bleeding complication in fetal and neonatal alloimmune thrombocytopenia (FNAIT) is an intracranial hemorrhage (ICH). However, FNAIT may also lead to other severe bleeding problems. The aim was to analyze this spectrum and evaluate the occurrence of severe hemorrhages other than ICH in fetuses or neonates with FNAIT. STUDY DESIGN AND METHODS: A retrospective chart analysis of cases of FNAIT presenting with severe bleeding complications other than ICH at our institution from 1990 to 2015 was conducted. Additionally, a review of the literature was performed to identify case reports and case series on FNAIT presenting with extracranial hemorrhage. RESULTS: Of 25 fetuses or neonates with severe bleeding due to FNAIT, three had isolated severe internal organ hemorrhage other than ICH, two pulmonary hemorrhages and one gastrointestinal hemorrhage. Two of these three neonates died due to this bleeding. Eighteen cases of extracranial bleeding complications as a first presentation of FNAIT were found in the literature, including ocular, gastrointestinal, spinal cord, pulmonary, renal, subgaleal, and genitourinary hemorrhages. CONCLUSION: Bleeding complications other than ICH may be more extensive, and the presentation of FNAIT may have a greater spectrum than previously described. A high index of suspicion on the possible diagnosis of FNAIT with any bleeding complication in a fetus or neonate may enable adequate diagnostics, adequate treatment, and appropriate follow-up in future pregnancies, as is especially relevant for FNAIT.
BACKGROUND: The most feared bleeding complication in fetal and neonatal alloimmune thrombocytopenia (FNAIT) is an intracranial hemorrhage (ICH). However, FNAIT may also lead to other severe bleeding problems. The aim was to analyze this spectrum and evaluate the occurrence of severe hemorrhages other than ICH in fetuses or neonates with FNAIT. STUDY DESIGN AND METHODS: A retrospective chart analysis of cases of FNAIT presenting with severe bleeding complications other than ICH at our institution from 1990 to 2015 was conducted. Additionally, a review of the literature was performed to identify case reports and case series on FNAIT presenting with extracranial hemorrhage. RESULTS: Of 25 fetuses or neonates with severe bleeding due to FNAIT, three had isolated severe internal organ hemorrhage other than ICH, two pulmonary hemorrhages and one gastrointestinal hemorrhage. Two of these three neonates died due to this bleeding. Eighteen cases of extracranial bleeding complications as a first presentation of FNAIT were found in the literature, including ocular, gastrointestinal, spinal cord, pulmonary, renal, subgaleal, and genitourinary hemorrhages. CONCLUSION: Bleeding complications other than ICH may be more extensive, and the presentation of FNAIT may have a greater spectrum than previously described. A high index of suspicion on the possible diagnosis of FNAIT with any bleeding complication in a fetus or neonate may enable adequate diagnostics, adequate treatment, and appropriate follow-up in future pregnancies, as is especially relevant for FNAIT.
Authors: Thijs W de Vos; Leendert Porcelijn; Suzanne Hofstede-van Egmond; Eva Pajkrt; Dick Oepkes; Enrico Lopriore; C Ellen van der Schoot; Dian Winkelhorst; Masja de Haas Journal: Br J Haematol Date: 2021-08-16 Impact factor: 8.615
Authors: C Ellen van der Schoot; Masja de Haas; Dian Winkelhorst; Thijs W de Vos; Marije M Kamphuis; Leendert Porcelijn; Enrico Lopriore; Dick Oepkes Journal: BMJ Open Date: 2020-07-20 Impact factor: 2.692