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Primary Intraosseous Xanthomas of the Jaws: A Series of Six Cases Including an Example with Formation of Apoptosis-Related Hyaline Globules, So-Called “Thanatosomes”

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Abstract

Primary intraosseous xanthomas of the jaws (PIXJ) are rare and predominantly affect the posterior mandible (86%) of normolipemic patients, with a mean age of 30 years and no gender predilection. Clinically, PIXJ exhibit indolent biologic behavior; curettage is considered treatment of choice. Only 36 PIXJ have been reported. Apoptosis-related hyaline globules (HGs), also known as “thanatosomes”, have not been previously reported in PIXJ. Cases diagnosed as xanthoma of bone were retrieved. Six cases fulfilling currently accepted criteria were identified and their clinicopathologic and immunohistochemical properties are presented herein. Mean age for PIXJ was 21.8 years (range = 12–33) and F:M ratio = 2:1. All cases presented as well-demarcated, unilocular or multilocular radiolucencies. Microscopically, PIXJ featured sheets of lipid-laden macrophages with eosinophilic or foamy cytoplasm. A secondary fibroblastic population lacking storiform pattern was evident in two cases. Adipocytes (3/6), peripheral neurovascular bundles (1/6), bone fragments (3/6) and dystrophic calcifications (3/6) were observed enclosed by the xanthoma cells. Notably, one case exhibited numerous, spherical, eosinophilic HGs containing apoptotic nuclei. PIXJ were consistently CD68(+) and negative for CD1α and S100. CD45 decorated lymphocytes and the membrane of foamy histiocytes. Xanthoma cells stained for lysozyme and plasma proteins including alpha-1 antitrypsin (AAT), IgG and IgA in one probed case. HGs were lysozyme(+), AAT(+), IgG(+), IgA(+), PAS(+) and diastase-resistant, and fuchsinophilic with Masson’s trichrome. PIXJ represent infrequent, solitary, mandibular lesions with a predilection for the second and third decade of life. Thanatosomes associated with cell injury and death can be present in PIXJ.

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Acknowledgements

The authors are indebted to Mr. Brian Dunnette (University of Minnesota, Minneapolis, MN) for sharing his expertise regarding the use of the Aperio ScanScope XT system. The authors would also like to thank Dr. Ioannis G. Koutlas, DDS, MS (University of Minnesota, Minneapolis, MN) for his guidance and support during the preparation of this manuscript.

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Authors and Affiliations

  1. Division of Oral and Maxillofacial Pathology, Department of Diagnostic and Biological Sciences, School of Dentistry, University of Minnesota, 515 Delaware Street SE 16-206B, Minneapolis, MN, 55455, USA

    Peter E. Wilkinson, Rajaram Gopalakrishnan & Prokopios P. Argyris

  2. School of Dentistry, National and Kapodistrian University of Athens, 11527, Athens, Greece

    Stavroula Merkourea

  3. Department of Biochemistry, Molecular Biology, and Biophysics, College of Biological Sciences, University of Minnesota, Minneapolis, MN, 55455, USA

    Prokopios P. Argyris

  4. Masonic Cancer Center, University of Minnesota, Minneapolis, MN, 55455, USA

    Prokopios P. Argyris

  5. Institute for Molecular Virology, University of Minnesota, Minneapolis, MN, 55455, USA

    Prokopios P. Argyris

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Wilkinson, P.E., Merkourea, S., Gopalakrishnan, R. et al. Primary Intraosseous Xanthomas of the Jaws: A Series of Six Cases Including an Example with Formation of Apoptosis-Related Hyaline Globules, So-Called “Thanatosomes”. Head and Neck Pathol 14, 859–868 (2020). https://doi.org/10.1007/s12105-020-01126-2

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