Langerhans cell histiocytosis: unusual bone marrow infiltration-report of 2 cases in Ecuador
Langerhans cell histiocytosis (LCH) is a histiocytic neoplasm characterized by the abnormal proliferation of Langerhans cells. Bone marrow (BM) involvement is associated with high-risk disease and poor survival. Although BM involvement is particularly uncommon, no reported cases of LCH with BM infil...
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Published in | Frontiers in medicine Vol. 11; p. 1433463 |
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Language | English |
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Abstract | Langerhans cell histiocytosis (LCH) is a histiocytic neoplasm characterized by the abnormal proliferation of Langerhans cells. Bone marrow (BM) involvement is associated with high-risk disease and poor survival. Although BM involvement is particularly uncommon, no reported cases of LCH with BM infiltration have been documented in Latin America until now. The aim of this report is to highlight the clinical, hematological, and BM findings of two cases of LCH with BM infiltration, providing insights that may aid in detecting suspected patients. We present two cases of LCH with BM infiltration. One case involved a 23-month-old male patient, and the other a 16-month-old female patient. Common clinical findings in both cases included hepatosplenomegaly and fever. Hematological findings revealed anemia in both cases. The key diagnostic tool was the BM biopsy, which revealed histiocyte nests with characteristic morphology, CD1a-positive cells, increased eosinophils, and reactive paratrabecular lymphocytes. This report underscores the significance of clinical profiles in predicting BM infiltration in LCH. The presence of histiocyte nests displaying the characteristic morphology of Langerhans cells, accompanied by an elevation in eosinophils, indicates bone marrow involvement. Furthermore, the demonstration of CD1a-positive cells through immunohistochemistry serves as a crucial diagnostic tool. |
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AbstractList | Langerhans cell histiocytosis (LCH) is a histiocytic neoplasm characterized by the abnormal proliferation of Langerhans cells. Bone marrow (BM) involvement is associated with high-risk disease and poor survival. Although BM involvement is particularly uncommon, no reported cases of LCH with BM infiltration have been documented in Latin America until now. The aim of this report is to highlight the clinical, hematological, and BM findings of two cases of LCH with BM infiltration, providing insights that may aid in detecting suspected patients. We present two cases of LCH with BM infiltration. One case involved a 23-month-old male patient, and the other a 16-month-old female patient. Common clinical findings in both cases included hepatosplenomegaly and fever. Hematological findings revealed anemia in both cases. The key diagnostic tool was the BM biopsy, which revealed histiocyte nests with characteristic morphology, CD1a-positive cells, increased eosinophils, and reactive paratrabecular lymphocytes. This report underscores the significance of clinical profiles in predicting BM infiltration in LCH. The presence of histiocyte nests displaying the characteristic morphology of Langerhans cells, accompanied by an elevation in eosinophils, indicates bone marrow involvement. Furthermore, the demonstration of CD1a-positive cells through immunohistochemistry serves as a crucial diagnostic tool. Langerhans cell histiocytosis (LCH) is a histiocytic neoplasm characterized by the abnormal proliferation of Langerhans cells. Bone marrow (BM) involvement is associated with high-risk disease and poor survival. Although BM involvement is particularly uncommon, no reported cases of LCH with BM infiltration have been documented in Latin America until now. The aim of this report is to highlight the clinical, hematological, and BM findings of two cases of LCH with BM infiltration, providing insights that may aid in detecting suspected patients. We present two cases of LCH with BM infiltration. One case involved a 23-month-old male patient, and the other a 16-month-old female patient. Common clinical findings in both cases included hepatosplenomegaly and fever. Hematological findings revealed anemia in both cases. The key diagnostic tool was the BM biopsy, which revealed histiocyte nests with characteristic morphology, CD1a-positive cells, increased eosinophils, and reactive paratrabecular lymphocytes. This report underscores the significance of clinical profiles in predicting BM infiltration in LCH. The presence of histiocyte nests displaying the characteristic morphology of Langerhans cells, accompanied by an elevation in eosinophils, indicates bone marrow involvement. Furthermore, the demonstration of CD1a-positive cells through immunohistochemistry serves as a crucial diagnostic tool.Langerhans cell histiocytosis (LCH) is a histiocytic neoplasm characterized by the abnormal proliferation of Langerhans cells. Bone marrow (BM) involvement is associated with high-risk disease and poor survival. Although BM involvement is particularly uncommon, no reported cases of LCH with BM infiltration have been documented in Latin America until now. The aim of this report is to highlight the clinical, hematological, and BM findings of two cases of LCH with BM infiltration, providing insights that may aid in detecting suspected patients. We present two cases of LCH with BM infiltration. One case involved a 23-month-old male patient, and the other a 16-month-old female patient. Common clinical findings in both cases included hepatosplenomegaly and fever. Hematological findings revealed anemia in both cases. The key diagnostic tool was the BM biopsy, which revealed histiocyte nests with characteristic morphology, CD1a-positive cells, increased eosinophils, and reactive paratrabecular lymphocytes. This report underscores the significance of clinical profiles in predicting BM infiltration in LCH. The presence of histiocyte nests displaying the characteristic morphology of Langerhans cells, accompanied by an elevation in eosinophils, indicates bone marrow involvement. Furthermore, the demonstration of CD1a-positive cells through immunohistochemistry serves as a crucial diagnostic tool. |
Author | Santana, Paulina Izquierdo-Condoy, Juan S Arias-Intriago, Marlon |
AuthorAffiliation | 1 Department of Pathology, Medical Science Faculty, Universidad Central del Ecuador , Quito , Ecuador 2 Department of Anatomic Pathology, Hospital de Especialidades Carlos Andrade Marín , Quito , Ecuador 3 One Health Research Group, Faculty of Medicine, Universidad de las Américas , Quito , Ecuador |
AuthorAffiliation_xml | – name: 2 Department of Anatomic Pathology, Hospital de Especialidades Carlos Andrade Marín , Quito , Ecuador – name: 1 Department of Pathology, Medical Science Faculty, Universidad Central del Ecuador , Quito , Ecuador – name: 3 One Health Research Group, Faculty of Medicine, Universidad de las Américas , Quito , Ecuador |
Author_xml | – sequence: 1 givenname: Paulina surname: Santana fullname: Santana, Paulina organization: Department of Anatomic Pathology, Hospital de Especialidades Carlos Andrade Marín, Quito, Ecuador – sequence: 2 givenname: Marlon surname: Arias-Intriago fullname: Arias-Intriago, Marlon organization: One Health Research Group, Faculty of Medicine, Universidad de las Américas, Quito, Ecuador – sequence: 3 givenname: Juan S surname: Izquierdo-Condoy fullname: Izquierdo-Condoy, Juan S organization: One Health Research Group, Faculty of Medicine, Universidad de las Américas, Quito, Ecuador |
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Cites_doi | 10.2350/09-05-0651-OA.1 10.1002/pbc.21227 10.1182/blood.2020005209 10.1016/j.annepidem.2018.04.005 10.1111/apa.16014 10.1177/107327481402100409 10.1111/petr.12244 10.1002/mpo.2950110307 10.1182/blood.2021014343 10.1177/03008916211039555 10.1177/0300060519864807 10.1056/NEJMra1607548 |
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Copyright | Copyright © 2024 Santana, Arias-Intriago and Izquierdo-Condoy. Copyright © 2024 Santana, Arias-Intriago and Izquierdo-Condoy. 2024 Santana, Arias-Intriago and Izquierdo-Condoy |
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Keywords | Langerhans cells Ecuador histiocytes bone marrow Latin America |
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License | Copyright © 2024 Santana, Arias-Intriago and Izquierdo-Condoy. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms. |
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References | Özyörük (ref14) 2021; 107 Xiao (ref12) 2020; 136 Cueva (ref9) 2019 (ref7) 2023 Galluzzo (ref6) 2010; 13 Kumar (ref1) 2015; 9 Goyal (ref13) 2022; 139 Peckham-Gregory (ref8) 2018; 28 Rodriguez-Galindo (ref11) 2021; 110 Allen (ref5) 2018; 379 Minkov (ref3) 2007; 49 Pan (ref15) 2019; 47 Grana (ref2) 2014; 21 Jun (ref10) 2014; 18 McClain (ref4) 1983; 11 |
References_xml | – volume: 13 start-page: 101 year: 2010 ident: ref6 article-title: Bone marrow findings at diagnosis in patients with multisystem langerhans cell histiocytosis publication-title: Pediatr Dev Pathol doi: 10.2350/09-05-0651-OA.1 contributor: fullname: Galluzzo – volume: 9 start-page: 193 year: 2015 ident: ref1 article-title: Bone marrow infiltration in Langerhan's cell histiocytosis—an unusual but important determinant for staging and treatment publication-title: Int J Hematol Oncol Stem Cell Res contributor: fullname: Kumar – volume: 49 start-page: 694 year: 2007 ident: ref3 article-title: Bone marrow assessment in Langerhans cell histiocytosis publication-title: Pediatr Blood Cancer doi: 10.1002/pbc.21227 contributor: fullname: Minkov – volume: 136 start-page: 2188 year: 2020 ident: ref12 article-title: Bone marrow–derived myeloid progenitors as driver mutation carriers in high- and low-risk Langerhans cell histiocytosis publication-title: Blood doi: 10.1182/blood.2020005209 contributor: fullname: Xiao – year: 2023 ident: ref7 – volume-title: Epidemiología del Cáncer en Quito 2011–2015 year: 2019 ident: ref9 contributor: fullname: Cueva – volume: 28 start-page: 521 year: 2018 ident: ref8 article-title: The role of parental and perinatal characteristics on Langerhans cell Histiocytosis: characterizing increased risk among Hispanics publication-title: Ann Epidemiol doi: 10.1016/j.annepidem.2018.04.005 contributor: fullname: Peckham-Gregory – volume: 110 start-page: 2892 year: 2021 ident: ref11 article-title: Clinical features and treatment of Langerhans cell histiocytosis publication-title: Acta Paediatr doi: 10.1111/apa.16014 contributor: fullname: Rodriguez-Galindo – volume: 21 start-page: 328 year: 2014 ident: ref2 article-title: Langerhans cell Histiocytosis publication-title: Cancer Control doi: 10.1177/107327481402100409 contributor: fullname: Grana – volume: 18 start-page: E124 year: 2014 ident: ref10 article-title: Haploidentical parental hematopoietic stem cell transplantation in pediatric refractory Langerhans cell histiocytosis publication-title: Pediatr Transplant junio de doi: 10.1111/petr.12244 contributor: fullname: Jun – volume: 11 start-page: 167 year: 1983 ident: ref4 article-title: Bone marrow involvement in histiocytosis X publication-title: Med Pediatr Oncol doi: 10.1002/mpo.2950110307 contributor: fullname: McClain – volume: 139 start-page: 2601 year: 2022 ident: ref13 article-title: International expert consensus recommendations for the diagnosis and treatment of Langerhans cell histiocytosis in adults publication-title: Blood doi: 10.1182/blood.2021014343 contributor: fullname: Goyal – volume: 107 start-page: NP120 year: 2021 ident: ref14 article-title: Successful treatment of multiple relapsed–refractory Langerhans cell histiocytosis with cyclosporine publication-title: Tumori doi: 10.1177/03008916211039555 contributor: fullname: Özyörük – volume: 47 start-page: 4522 year: 2019 ident: ref15 article-title: Autologous hematopoietic stem cell transplantation for efficient treatment of multisystem, high-risk, BRAF V600E-negative Langerhans cell histiocytosis publication-title: J Int Med Res doi: 10.1177/0300060519864807 contributor: fullname: Pan – volume: 379 start-page: 856 year: 2018 ident: ref5 article-title: Langerhans-cell Histiocytosis publication-title: N Engl J Med doi: 10.1056/NEJMra1607548 contributor: fullname: Allen |
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Title | Langerhans cell histiocytosis: unusual bone marrow infiltration-report of 2 cases in Ecuador |
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