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Histiocytosis Otomastoiditis X: About a Clinical Case

Received: 16 May 2022    Accepted: 6 September 2022    Published: 29 September 2022
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Abstract

Langerhans cell histiocytosis (LCH) or histiocytosis X refers to a group of diseases that are characterized by clonal proliferation of histiocyte with similar characteristics to Langerhans cells, which affect different organs of the human body, including the temporal bone. The present study describes the clinical case of a 42-year-old patient admitted to surgery for acute chronic otomastoiditis on the right temporal bone. Axial Computerized Tomography (CT) revealed petromastoid osteolytic lesions, dehiscence of the lateral sinus and external semicircular canal and a solution of continuity of the tympanic tegmen, but without infiltration of neurological structures. The diagnosis of temporal bone LCH was confirmed with histopathological and immunohistochemical exams. Depending on the type of lesion, there are different therapeutic modalities for LCH of the temporal bone that include surgery, chemotherapy, radiotherapy and even corticotherapy. In this case, the patient was submitted to mastoidectomy the right, followed by corticotherapy (dexamethasone) and chemotherapy (vinblastine). Up to date, he has not yet had any relapse, but is still on follow up.

Published in International Journal of Otorhinolaryngology (Volume 8, Issue 2)
DOI 10.11648/j.ijo.20220802.16
Page(s) 57-61
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2022. Published by Science Publishing Group

Keywords

Langerhans Cell Histiocytosis, Temporal Bone, Langerhans Cell

References
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[2] Brenda L. Nelson: Langerhans ‘cell histiocytosis of the temporal Bone. Head and Neck Pathol (2008); 2: 97-98. DOI 10.1007/s12105-008-0046-5.
[3] Nong Dong-xião, Nong Hui-tu, Tang Na-zhou, Chen Ruo-ze: Langerhans ‘Cell Histiocytosis (Histiocytosis X) of the Temporal Bone. Journal of Otology, 2007, Vol. 2, Nº1. https://doi.org/10.1016/S1672-2930(07)50007-3
[4] Asser A Elsharkawy, Mohamed M Sarhan: Langerhans ‘Cell Histiocytosis of Temporal Bone: A Study of 11 Egyptian Patients. Clinical Report. Journal International Advanced Otology, 2012; 8: (3) 463-469. www.advancedotology.org
[5] Lidiane Maria de Brito Macedo Ferreira, João Deodato Diógenes de Carvalho, Sérgio Tadeu Almeida Pereira, Marylane Galvão Tavares: Histiocytosis X in the temporal bone: Case report. Brazilian Journal of Otorhinolaryngology, 2006; 72 (4): 575. www.researchgate.net
[6] Viswanatha B. Granulomatous Diseases of the Middle Ear: Overview. [Internet] [updated 2018 Oct 29] Available from: https://emedicine.medscape.com/article/861035-overview
[7] Jeffrey M. Lipton, MD, Professor of Pediatrics and Molecular Medicine, Hofstra Northwell School of Medicine, Hempstead, NY; Professor, The Center for Autoimmune and Musculokletal Diease, Feinstein Institute for Medical Research; Chief, Hematology/Oncology and Stem Cell Transplantation, Cohen Chieldren̕s Medical Center of New York: Histiocitose das células de Langerhans. Manual MSD, 2019. www.msdmanuals.com
[8] Michael Girschikofsky, Maurizio Arico, Diego Castillo, Anthony Chu, et al: Management of adult patients with Langerhans cell histiocytosis: recommendations from an expert panel on behalf of Euro-Histio-Net. Orphanet Journal of Rare Diseases 2013, 8: 72. http://www.ojrd.com/content/8/1/72
[9] Margarida Dantas de Brito, Ângelo Martins, Joaquim Andrade, José Guimarães, José Mariz: Histiocitose de Langerhans no Adulto: Experiência de Dois Hospitais Portugueses. Acta Med Port 2014 Nov-Dec; 27 (6): 726-730. www.actamedicaportuguesa.com/revista/index.php/amp/article/download/5360/4149
[10] Robson Azevedo Dutra, Adriana Cartafina Perez-Boscollo, Luiz Maurício da Silva Júnior, Murilo Alexandre, Sebastião Dutra de Morais Júnior: Histiocitose de Células de Langerhans: Um Diagnóstico Diferencial dos Tumores do Mediastino Anterior em Crianças. Relato de Caso. http://www1.inca.gov.br/rbc/n_60/v04/pdf/07-relato-de-caso-histiocitose-de-celulas-de-langerhans-um-diagnostico-diferencial-dos-tumores-do-mediastino-anterior-em-criancas.pdf
[11] Fátima Satomi Nishimori, Gabriel Monteiro de Castro Chaves, Camila Nemoto de Mendonça, Priscila Mara Chaves e Silva, Calisqui Brandenburg Fernandes, Leonardo Vieira Neto, João Carlos Regazzi Avelleira: Histiocitose de células de Langerhans: Caso Clínico. Revista SPDV 72 (2) 2014. https://revista.spdv.com.pt/index.php/spdv/article/viewFile/260/235
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Cite This Article
  • APA Style

    Luzbel Von-Haff, Miguel Rito, João Figueira, Anabela Palma, Cristóvão Ribeiro, et al. (2022). Histiocytosis Otomastoiditis X: About a Clinical Case. International Journal of Otorhinolaryngology, 8(2), 57-61. https://doi.org/10.11648/j.ijo.20220802.16

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    ACS Style

    Luzbel Von-Haff; Miguel Rito; João Figueira; Anabela Palma; Cristóvão Ribeiro, et al. Histiocytosis Otomastoiditis X: About a Clinical Case. Int. J. Otorhinolaryngol. 2022, 8(2), 57-61. doi: 10.11648/j.ijo.20220802.16

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    AMA Style

    Luzbel Von-Haff, Miguel Rito, João Figueira, Anabela Palma, Cristóvão Ribeiro, et al. Histiocytosis Otomastoiditis X: About a Clinical Case. Int J Otorhinolaryngol. 2022;8(2):57-61. doi: 10.11648/j.ijo.20220802.16

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  • @article{10.11648/j.ijo.20220802.16,
      author = {Luzbel Von-Haff and Miguel Rito and João Figueira and Anabela Palma and Cristóvão Ribeiro and Lígia Ferreira and Pedro Montalvão and João Roque},
      title = {Histiocytosis Otomastoiditis X: About a Clinical Case},
      journal = {International Journal of Otorhinolaryngology},
      volume = {8},
      number = {2},
      pages = {57-61},
      doi = {10.11648/j.ijo.20220802.16},
      url = {https://doi.org/10.11648/j.ijo.20220802.16},
      eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.ijo.20220802.16},
      abstract = {Langerhans cell histiocytosis (LCH) or histiocytosis X refers to a group of diseases that are characterized by clonal proliferation of histiocyte with similar characteristics to Langerhans cells, which affect different organs of the human body, including the temporal bone. The present study describes the clinical case of a 42-year-old patient admitted to surgery for acute chronic otomastoiditis on the right temporal bone. Axial Computerized Tomography (CT) revealed petromastoid osteolytic lesions, dehiscence of the lateral sinus and external semicircular canal and a solution of continuity of the tympanic tegmen, but without infiltration of neurological structures. The diagnosis of temporal bone LCH was confirmed with histopathological and immunohistochemical exams. Depending on the type of lesion, there are different therapeutic modalities for LCH of the temporal bone that include surgery, chemotherapy, radiotherapy and even corticotherapy. In this case, the patient was submitted to mastoidectomy the right, followed by corticotherapy (dexamethasone) and chemotherapy (vinblastine). Up to date, he has not yet had any relapse, but is still on follow up.},
     year = {2022}
    }
    

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  • TY  - JOUR
    T1  - Histiocytosis Otomastoiditis X: About a Clinical Case
    AU  - Luzbel Von-Haff
    AU  - Miguel Rito
    AU  - João Figueira
    AU  - Anabela Palma
    AU  - Cristóvão Ribeiro
    AU  - Lígia Ferreira
    AU  - Pedro Montalvão
    AU  - João Roque
    Y1  - 2022/09/29
    PY  - 2022
    N1  - https://doi.org/10.11648/j.ijo.20220802.16
    DO  - 10.11648/j.ijo.20220802.16
    T2  - International Journal of Otorhinolaryngology
    JF  - International Journal of Otorhinolaryngology
    JO  - International Journal of Otorhinolaryngology
    SP  - 57
    EP  - 61
    PB  - Science Publishing Group
    SN  - 2472-2413
    UR  - https://doi.org/10.11648/j.ijo.20220802.16
    AB  - Langerhans cell histiocytosis (LCH) or histiocytosis X refers to a group of diseases that are characterized by clonal proliferation of histiocyte with similar characteristics to Langerhans cells, which affect different organs of the human body, including the temporal bone. The present study describes the clinical case of a 42-year-old patient admitted to surgery for acute chronic otomastoiditis on the right temporal bone. Axial Computerized Tomography (CT) revealed petromastoid osteolytic lesions, dehiscence of the lateral sinus and external semicircular canal and a solution of continuity of the tympanic tegmen, but without infiltration of neurological structures. The diagnosis of temporal bone LCH was confirmed with histopathological and immunohistochemical exams. Depending on the type of lesion, there are different therapeutic modalities for LCH of the temporal bone that include surgery, chemotherapy, radiotherapy and even corticotherapy. In this case, the patient was submitted to mastoidectomy the right, followed by corticotherapy (dexamethasone) and chemotherapy (vinblastine). Up to date, he has not yet had any relapse, but is still on follow up.
    VL  - 8
    IS  - 2
    ER  - 

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Author Information
  • Department of Otolaryngology - Head and Neck Sugery, Hospital Center Setúbal, Setúbal, Portugal

  • Department of Pathological Anatomy, Portuguese Institute, Oncology Lisbon Francisco Gentil, Lisbon, Portugal

  • Department of Otolaryngology - Head and Neck Sugery, Hospital Center Setúbal, Setúbal, Portugal

  • Department of Otolaryngology - Head and Neck Sugery, Portuguese Institute, Oncology of Lisbon Francisco Gentil, Lisbon, Portugal

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