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OCT2 expression in histiocytoses

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Abstract

Diagnosis of histiocytosis can be difficult and one of the biggest challenges is to distinguish between reactive and neoplastic histiocytes on histology alone. Recently, OCT2 nuclear expression was reported in Rosai-Dorfman disease (RDD). Our purpose was to expand the testing of OCT2 on a broader variety of sporadic or H syndrome-related histiocytoses. Cases of histiocytoses were retrieved from the files of Ambroise Paré Pathology Department. All slides and molecular analyses were reviewed, and staining was completed with immunohistochemistry for OCT2. A total of 156 samples from different localizations were tested. Among sporadic cases, 52 patients had RDD, and 10 patients had mixed histiocytosis combining RDD with Erdheim Chester disease (ECD, n = 8), Langerhans cell histiocytosis (LCH, n = 2) or juvenile xanthogranuloma (JXG, n = 1). All these patients were positive for OCT2 in RDD characteristic histiocytes. Twenty-three patients had ECD and all but two (91% − 21/23) were negative for OCT2. By contrast, OCT2 was positive in 11/27 (41%) LCH and 6/16 (38%) JXG. Among the 10 samples of H syndrome-associated histiocytosis, 3 had typical RDD histology, 6 had unclassified histiocytosis, and one had mixed RDD-LCH; all were positive for OCT2. On 16 samples of granulomatous lymphadenitis, OCT2 was negative in epithelioid histiocytes. Our study shows that OCT2 has a sensitivity of 100% for RDD cases and mixed histiocytoses with an RDD component. It is negative in 92% of ECD but expressed in at least 38% of LCH, JXG, and C group histiocytoses. Finally, OCT2 is positive in all H syndrome-related histiocytoses, independent of their histology.

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Funding

This article was supported by the Programme de Recherche Translationnelle sur le Cancer from the French National Cancer Institute [PRT-K19-143] and by unrestricted grants from the non-profit association AREP.

IAU received support from the European Society of Pathology for this work.

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Authors and Affiliations

  1. Pathology Department, Paris-Saclay University, Versailles SQY University (UVSQ), EA4340-BECCOH, Assistance Publique–Hôpitaux de Paris (APHP), Ambroise-Paré Hospital, 9 Avenue Charles de Gaulle, 92104, Boulogne-Billancourt, France

    Irena Antonia Ungureanu, Zofia Hélias-Rodzewicz & Jean-François Emile

  2. Internal Medicine Department 2, Sorbonne University, APHP, Pitié–Salpêtrière Hospital, French National Referral Center for Rare Systemic Diseases and Histiocytoses, Paris, France

    Fleur Cohen-Aubart & Julien Haroche

  3. Department of Pediatric Oncology and Hematology, Sorbonne University, APHP, Armand-Trousseau Hospital, Referral Center for Histiocytoses, Paris, France

    Sébastien Héritier

  4. Pathology Department, Necker-Enfants-Malades Hospital, Paris, France

    Sylvie Fraitag

  5. Pathology Department, Pierre Et Marie Curie University, APHP, Pitié-Salpétrière Hospital, Paris, France

    Frédéric Charlotte

  6. Department of Internal Medicine, Claude Bernard-Lyon1 University, Hôpital de La Croix-Rousse, Lyon, France

    Hippolyte Lequain

  7. Pediatric Oncology and Hematology, Armand-Trousseau Hospital, APHP, EA4340-BECCOH, Referral Center for Histiocytoses UVSQ, Paris, France

    Jean Donadieu

Authors
  1. Irena Antonia Ungureanu
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  2. Fleur Cohen-Aubart
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  3. Sébastien Héritier
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  4. Sylvie Fraitag
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  5. Frédéric Charlotte
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  6. Hippolyte Lequain
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  7. Zofia Hélias-Rodzewicz
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  8. Julien Haroche
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  9. Jean Donadieu
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  10. Jean-François Emile
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Contributions

IAU and JFE wrote de manuscript. JFE obtained funding. All co-authors contributed to collection and analysis of data, and correction of the manuscript. All co-authors accepted the final version of the manuscript.

Corresponding author

Correspondence to Jean-François Emile.

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Ethics approval and consent to participate

The article is an original work. The study (NCT04437381) has been approved by the ethical committee CESREES #2814848bis. Patients signed informed consent for translational research. This work was presented in part during the 38th annual meeting of the Histiocyte Society (Stockholm, September 2022).

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The authors declare no competing interests.

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Ungureanu, I.A., Cohen-Aubart, F., Héritier, S. et al. OCT2 expression in histiocytoses. Virchows Arch 483, 81–86 (2023). https://doi.org/10.1007/s00428-023-03508-7

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  • DOI: https://doi.org/10.1007/s00428-023-03508-7

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