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A pregnant woman with thymoma-associated pure red cell aplasia

BACKGROUND: Pure red cell aplasia (PRCA) is a hematological disorder characterized by anemia with severe reticulocytopenia caused by a marked reduction in erythroid precursors in the bone marrow. PRCA is known to be associated with pregnancy, but thymoma-associated PRCA during pregnancy is very rare...

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Autores principales: Shibata, Megumi, Kaneko, Kayoko, Umehara, Nagayoshi, Matsui, Hitoshi, Kawai, Toshinao, Nakadate, Hisaya, Murashimia, Atsuko, Sago, Haruhiko
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9608914/
https://www.ncbi.nlm.nih.gov/pubmed/36303124
http://dx.doi.org/10.1186/s12884-022-05145-5
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author Shibata, Megumi
Kaneko, Kayoko
Umehara, Nagayoshi
Matsui, Hitoshi
Kawai, Toshinao
Nakadate, Hisaya
Murashimia, Atsuko
Sago, Haruhiko
author_facet Shibata, Megumi
Kaneko, Kayoko
Umehara, Nagayoshi
Matsui, Hitoshi
Kawai, Toshinao
Nakadate, Hisaya
Murashimia, Atsuko
Sago, Haruhiko
author_sort Shibata, Megumi
collection PubMed
description BACKGROUND: Pure red cell aplasia (PRCA) is a hematological disorder characterized by anemia with severe reticulocytopenia caused by a marked reduction in erythroid precursors in the bone marrow. PRCA is known to be associated with pregnancy, but thymoma-associated PRCA during pregnancy is very rare, and its successful management has not been reported. CASE PRESENTATION: A 37-year-old primiparous woman with severe anemia was referred to our center at 27 weeks’ gestation. She was diagnosed with PRCA based on bone aspiration findings at 33 weeks’ gestation. Magnetic resonance imaging (MRI) revealed an anterior mediastinal mass 4 cm in size suspected of being thymoma. She was therefore diagnosed with thymoma-associated PRCA during pregnancy. Surgery for thymoma was planned after delivery, since the imaging findings were suggestive of early-stage thymoma (Masaoka stage I or II). With transfusion of a total 3,360 ml of red blood cells (RBCs) during pregnancy, the patient gave birth to a baby girl weighing 2,548 g at 40 weeks’ gestation. The baby showed transient congenital cutaneous candidiasis. The placental pathology revealed subamniotic inflammation with a fungal structure. Treatment with topical anti-fungal cream immediately ameliorated the baby’s skin lesion. Maternal anemia did not improve after delivery; however, the thymoma did not increase in size. At five months after delivery, the mother underwent thymectomy with oral cyclosporine A. A pathological examination revealed Masaoka stage II-a thymoma. She completely had recovered from anemia at six months after surgery. Cyclosporine A treatment was discontinued three years after surgery. Remission has been sustained for four years since surgery. CONCLUSIONS: A very rare case of thymoma-associated PRCA during pregnancy was diagnosed without any subjective symptoms and was expectantly managed, resulting in a good prognosis. Although bone marrow aspiration during pregnancy is an invasive test, it is important to confirm the diagnosis. Conservative management with blood transfusion was possible for early-stage thymoma-associated PRCA during pregnancy. Active surveys, including MRI, for PRCA during pregnancy led to the detection of thymoma at an early stage and the achievement of a preferable pregnancy outcome. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s12884-022-05145-5.
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spelling pubmed-96089142022-10-28 A pregnant woman with thymoma-associated pure red cell aplasia Shibata, Megumi Kaneko, Kayoko Umehara, Nagayoshi Matsui, Hitoshi Kawai, Toshinao Nakadate, Hisaya Murashimia, Atsuko Sago, Haruhiko BMC Pregnancy Childbirth Case Report BACKGROUND: Pure red cell aplasia (PRCA) is a hematological disorder characterized by anemia with severe reticulocytopenia caused by a marked reduction in erythroid precursors in the bone marrow. PRCA is known to be associated with pregnancy, but thymoma-associated PRCA during pregnancy is very rare, and its successful management has not been reported. CASE PRESENTATION: A 37-year-old primiparous woman with severe anemia was referred to our center at 27 weeks’ gestation. She was diagnosed with PRCA based on bone aspiration findings at 33 weeks’ gestation. Magnetic resonance imaging (MRI) revealed an anterior mediastinal mass 4 cm in size suspected of being thymoma. She was therefore diagnosed with thymoma-associated PRCA during pregnancy. Surgery for thymoma was planned after delivery, since the imaging findings were suggestive of early-stage thymoma (Masaoka stage I or II). With transfusion of a total 3,360 ml of red blood cells (RBCs) during pregnancy, the patient gave birth to a baby girl weighing 2,548 g at 40 weeks’ gestation. The baby showed transient congenital cutaneous candidiasis. The placental pathology revealed subamniotic inflammation with a fungal structure. Treatment with topical anti-fungal cream immediately ameliorated the baby’s skin lesion. Maternal anemia did not improve after delivery; however, the thymoma did not increase in size. At five months after delivery, the mother underwent thymectomy with oral cyclosporine A. A pathological examination revealed Masaoka stage II-a thymoma. She completely had recovered from anemia at six months after surgery. Cyclosporine A treatment was discontinued three years after surgery. Remission has been sustained for four years since surgery. CONCLUSIONS: A very rare case of thymoma-associated PRCA during pregnancy was diagnosed without any subjective symptoms and was expectantly managed, resulting in a good prognosis. Although bone marrow aspiration during pregnancy is an invasive test, it is important to confirm the diagnosis. Conservative management with blood transfusion was possible for early-stage thymoma-associated PRCA during pregnancy. Active surveys, including MRI, for PRCA during pregnancy led to the detection of thymoma at an early stage and the achievement of a preferable pregnancy outcome. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s12884-022-05145-5. BioMed Central 2022-10-27 /pmc/articles/PMC9608914/ /pubmed/36303124 http://dx.doi.org/10.1186/s12884-022-05145-5 Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Shibata, Megumi
Kaneko, Kayoko
Umehara, Nagayoshi
Matsui, Hitoshi
Kawai, Toshinao
Nakadate, Hisaya
Murashimia, Atsuko
Sago, Haruhiko
A pregnant woman with thymoma-associated pure red cell aplasia
title A pregnant woman with thymoma-associated pure red cell aplasia
title_full A pregnant woman with thymoma-associated pure red cell aplasia
title_fullStr A pregnant woman with thymoma-associated pure red cell aplasia
title_full_unstemmed A pregnant woman with thymoma-associated pure red cell aplasia
title_short A pregnant woman with thymoma-associated pure red cell aplasia
title_sort pregnant woman with thymoma-associated pure red cell aplasia
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9608914/
https://www.ncbi.nlm.nih.gov/pubmed/36303124
http://dx.doi.org/10.1186/s12884-022-05145-5
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