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Oral verruciform xanthoma associated with stem cell transplantation and graft versus host disease in a paediatric patient

Bone Marrow Transplantation volume 55, pages 1832–1833 (2020)Cite this article

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This case outlines the presentation of verruciform xanthoma in a 11-year-old boy who presented with oral lesions with a history of stem cell transplant and subsequent graft versus host disease affecting the skin. This report aims to explore the risk factors associated with this unusual pathology as well as reviewing current literature explaining its pathogenesis.

A 11-year-old boy was referred to the Oral and Maxillofacial Surgery department with regard to two asymptomatic oral lesions. Both were well-defined, pink, raised lesions situated on the hard palate and the mucosa at the right commissure. The patient had undergone prior treatment for relapsed T-cell acute lymphoblastic leukaemia, including chemotherapy followed by 6/8 cord blood (fludarabine, cyclophosphamide and total body irradiation) stem cell transplantation (SCT) 29 months previously. Post SCT, he developed skin and oral mucosal graft versus host disease (GVHD), which responded well to medical management including oral steroid mouthwash. Systemic immunosuppression was discontinued 15 months after SCT. Intermittent oral budesonide was required up to 27 months post transplant. He had a normal CD4 count from 12 months post transplant and was therefore not significantly immunosuppressed at the time of presentation.

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Fig. 1: Histological image of left palate lesion at ×40 magnification and right oral commissure lesion at ×200 magnification taken on Olympus BX-51.

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Acknowledgements

The authors wish to thank Dr Liz Hook (honorary consultant paediatric histopathologist, Addenbrookes Hospital, Cambridge) for the provision of the histological images.

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Authors and Affiliations

  1. Oral and Maxillofacial Surgery Department, Addenbrookes Hospital, Cambridge, Hills Road, Cambridge, CB2 0QQ, UK

    Ian Jenkyn & Christopher Fowell

  2. The University of Cambridge School of Clinical Medicine, Addenbrooke’s Hospital, Hills Road, Cambridge, CB2 0SP, UK

    George Woodward

  3. Children’s and Adolescent Services, Addenbrookes Hospital, Cambridge, Hills Road, Cambridge, CB2 0QQ, UK

    Anne Kelly

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  1. Ian Jenkyn
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  2. George Woodward
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  3. Anne Kelly
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Correspondence to Ian Jenkyn.

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Jenkyn, I., Woodward, G., Kelly, A. et al. Oral verruciform xanthoma associated with stem cell transplantation and graft versus host disease in a paediatric patient. Bone Marrow Transplant 55, 1832–1833 (2020). https://doi.org/10.1038/s41409-019-0772-1

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