Skip to main content
Log in

Rare Case of an Ancient Craniofacial Osteosarcoma with Probable Surgical Intervention

  • Original Article
  • Published:
Pathology & Oncology Research

Abstract

Osteosarcoma is the most common primary malignant bone tumor both today and in antiquity. Nevertheless, it is a comparatively rare tumor. This paper describes a case of a highly aggressive craniofacial lesion from the 11th–12th centuries AD, most likely representing osteosarcoma. During the paleopathological study, macroscopic, endoscopic, radiological, scanning-electron and light microscopic investigations were performed. The skull of the approximately 40–50 year-old female revealed several pathological findings. The most impressive macroscopic feature was an extensively spiculated periosteal reaction (“sunburst” pattern) in combination with a massive bone destruction most likely derived from a highly aggressive tumor originating in the ethmoidal area of the medial wall of the orbit. The central parts of the lesion showed excessive new and most probably neoplastic bone formation indicating an underlying high-grade osteosarcoma. The light microscopic examination revealed three different levels of bony structures representing different qualities of bone tissues. Besides the mass lesion, signs of a healed multiple incomplete trephination of the left parietal bone was observed. This case represents a unique example in which the concomitance of a tumor and an incomplete trephination could be observed from the skeletal remains of an ancient individual. The case opens new considerations as to whether surgical interventions, such as incomplete trephination, might have been used already in the Middle Ages as a therapeutic approach.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2
Fig. 3

Similar content being viewed by others

References

  1. David AR, Zimmerman MR (2010) Cancer: an old disease, a new disease or something in between? Nat Rev Cancer 10:728–733

    Article  CAS  PubMed  Google Scholar 

  2. Capasso LL (2005) Antiquity of cancer. Int J Cancer 113:2–13

    Article  CAS  PubMed  Google Scholar 

  3. Thillaud PL (2006) Paleopathology of cancers. Bull Cancer 93:767–773

    CAS  PubMed  Google Scholar 

  4. Schultz M, Parzinger H, Posdnjakov DV, Chikisheva TA, Schmidt-Schultz TH (2007) Oldest known case of metastasizing prostate carcinoma diagnosed in the skeleton of a 2,700-year-old Scythian king from Arzhan (Siberia, Russia). Int J Cancer 121:2591–2595

    Article  CAS  PubMed  Google Scholar 

  5. Ortner DJ (2011) What skeletons tell us. The story of human paleopathology. Virchows Arch 459(3):247–254. doi:10.1007/s00428-011-1122-x

    Article  PubMed  Google Scholar 

  6. Halperin EC (2004) Paleo-oncology: the role of ancient remains in the study of cancer. Perspect Biol Med 47:1–14

    Article  PubMed  Google Scholar 

  7. Rosenberg AE, Cleton-Jansen A-M, de Pinieux G, Deyrup AT, Hauben E, Squire J (2013) Conventional osteosarcoma. In: Fletcher CDM, Bridge JA, Hogendoorn PCW, Mertens F (eds) WHO classification of Tumours of Tumours of soft tissue and bone. IARC Press, Lyon, pp. 282–288

    Google Scholar 

  8. Gadwal SR, Gannon FH, Fanburg-Smith JC, Becoskie EM, Thompson LD (2001) Primary osteosarcoma of the head and neck in pediatric patients: a clinicopathologic study of 22 cases with a review of the literature. Cancer 91:598–605

    Article  CAS  PubMed  Google Scholar 

  9. Smith RB, Apostolakis LW, Karnell LH, Koch BB, Robinson RA, Zhen W, Menck HR, Hoffman HT (2003) National Cancer Data Base report on osteosarcoma of the head and neck. Cancer 98:1670–1680

    Article  PubMed  Google Scholar 

  10. Beutel A, Tänzer A (1963) Röntgendiagnostik der Orbitae, der Augen und der Tränenwege. In: Olsson O, Strnad F, Vieten H, Zuppinger A (eds) Handbuch der Medizinischen Radiologie, Bd VII/2. Röntgendiagnostik des Schädels II, 1st edn. Springer, Berlin-Göttingen-Heidelberg, pp 673–818

  11. Ortner DJ (2003) Tumors and tumor-like lesions of bone. In: Ortner DJ (ed) Identification of pathological conditions in human skeletal remains, 2nd edn. Academic Press, New York, pp. 503–544

    Chapter  Google Scholar 

  12. Farkas LG, Józsa L, Paja L, Molnár J (2007) Bone forming tumors on skeletons from a medieval Hungarian cemetery (Bátmonostor). Paleopathol Newsl 140:14–22

    Google Scholar 

  13. Józsa LG, Fóthi E (2003) Juxtacortical osteosarcoma on tibia and fibula from a medieval cemetery of Budapest. J Paleopath 15:23–31

    Google Scholar 

  14. Bona A, Papai Z, Maasz G, Gabor A, Toth GA, Jambor E, Schmidt J, Cs T, Cs F, Mark L (2014) Mass spectrometric identification of ancient proteins as potential molecular biomarkers for a 2000-year-old osteogenic sarcoma. PLoS One 9(7):e103862. doi:10.1371/journal.pone.0103862

    Article  Google Scholar 

  15. Russeva V (2012) Religion, Magic or Medicine? New finds of trepanned skulls from Southeastern Bulgaria, 11th–13th c. Archaeologia Bulgarica XVI:77–95

  16. Mariani-Costantini R, Catalano P, di Gennaro F, di Tota G, Angeletti LR (2000) New light on cranial surgery in ancient Rome. Lancet 355:5–7

    Article  Google Scholar 

  17. Jordanov J, Br D, Sp N (1988) Symbolic trephination of skulls from the middle ages (IXth-Xth century) in Bulgaria. Acta Neurochir 92:15–18

    Article  CAS  PubMed  Google Scholar 

  18. Zs B, Évinger S, Fóthi E (2006) New symbolic trephination cases from Hungary. Annls hist-nat Mus nat hung 98:177–183

    Google Scholar 

  19. Bereczki Z, Molnár E, Marcsik A, Pálfi G (2015) Rare types of trephination from Hungary shed new light on possible cross-cultural connections in the Carpathian Basin. Int J Osteoarchaeol 25(3):322–333

    Article  Google Scholar 

  20. Allodiatoris I (1937) Adatok az Árpádkori alföldi magyarság anthropológiájához (data to the anthropology of the lowland Hungarians from the Arpadian-age). University of Pázmány Péter, Dissertation

    Google Scholar 

  21. Zádori P, Bajzik G, Biró G, Lelovics Zs, Balassa T, Bernert Zs, Évinger S, Hajdu T, Marcsik A, Molnár E, Ősz B, Pálfi Gy, Wolff K, Repa I (2016) Koponyacsont-laesiók komputertomográfiás vizsgálata és paleoradiológiai aspektusai (Computed tomographic examination of cranial lesions, a paleoradiological approach) Ideggyogy Sz 69: (in press)

  22. Schultz M (2001) Paleohistopathology of bone: a new approach to the study of ancient diseases. Yearb Phys Anthropol 44:106–147

    Article  Google Scholar 

  23. Schultz M (2003) Light microscopic analysis in skeletal paleopathology. In: Ortner DJ (ed) Identification of pathological conditions in human skeletal remains, 2nd edn. Academic Press, New York, pp. 73–108

    Chapter  Google Scholar 

  24. Schultz M (2011) Light microscopic analysis of macerated pathologically changed bone. In: Crowder C, Stout S (eds) Bone histology. An anthropological perspective. CRC Press, Taylor & Francis Group, Boca Raton, pp. 253–296

    Chapter  Google Scholar 

  25. Molnár M, Janovics R, Major I, Molnár M, Janovics R, Major I, Orsovszki J, Gönczi R, Veres M, Leonard AG, Castle SM, Lange TE, Wacker L, Hajdas I, Jull AJT (2013) Status report of the new AMS 14C sample preparation lab of the Hertelendi Laboratory of environmental studies, Debrecen, Hungary. In: Jull AJT, Hatté C (eds). Proceedings of the 21st International Radiocarbon Conference. Radiocarbon 55:665–76

  26. Weidenreich F (1930) Das Knochengewebe. In: v. Möllendorff W (ed) Handbuch der Mikroskopischen Anatomie des Menschen, Bd. II, Die Gewebe, Zweiter Teil, Stützgewebe, Knochengewebe, Skeletsystem. Verlag von Julius Springer, Berlin, pp. 391–520

Download references

Acknowledgements

This research was supported by grants from the Hungarian National Scientific Research Foundation (OTKA) project number NN 78696, TÁMOP 4.2.4. A/1-11-1-2012-0001 „National Excellence Program – Elaborating and operating an inland student and researcher personal support system” and ELTE Talent Management Council.

We would like to acknowledge our debt of gratitude to Kendra Sirak for language editing of the manuscript.

The authors thank Michael Brandt for the production of the thin-ground samples and Ingrid Hettwer-Steeger for the preparation of the samples for scanning-electron microscopy, both Department of Anatomy, University Medical School Göttingen, Germany.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Erika Molnár.

Additional information

Michael Schultz is a shared first author of the manuscript.

Electronic supplementary material

ESM 1

(PDF 921 kb)

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Molnár, E., Schultz, M., Schmidt-Schultz, T.H. et al. Rare Case of an Ancient Craniofacial Osteosarcoma with Probable Surgical Intervention. Pathol. Oncol. Res. 23, 583–587 (2017). https://doi.org/10.1007/s12253-016-0153-7

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12253-016-0153-7

Keywords

Navigation