Isolated juvenile xanthogranuloma of the subglottis: Case report

Authors

  • Mahilravi S. Thevasagayam FRCS,

    1. Department of Otolaryngology, Sheffield Children's Hospital, The Royal Hallamshire Hospital, Glossop Road, Sheffield S10 2JF, United Kingdom
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  • Sudip Ghosh FRCS,

    1. Department of Otolaryngology, Sheffield Children's Hospital, The Royal Hallamshire Hospital, Glossop Road, Sheffield S10 2JF, United Kingdom
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  • D. O'Neill MRCP, MRCPath,

    1. Department of Histopathology, Sheffield Children's Hospital, University of Sheffield, Sheffield, United Kingdom
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  • A. Panarese FRCS,

    1. Department of Otolaryngology, Sheffield Children's Hospital, The Royal Hallamshire Hospital, Glossop Road, Sheffield S10 2JF, United Kingdom
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  • Peter D. Bull FRCS

    Corresponding author
    1. Department of Otolaryngology, Sheffield Children's Hospital, The Royal Hallamshire Hospital, Glossop Road, Sheffield S10 2JF, United Kingdom
    • Department of Otolaryngology, Sheffield Children's Hospital, The Royal Hallamshire Hospital, Glossop Road, Sheffield S10 2JF, United Kingdom
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Abstract

Background

Juvenile xanthogranulomatosis (JXG) is a relatively rare macrophage proliferative disorder. It usually presents as a localized cutaneous lesion but may affect other organs. Until now it has never been described in the subglottic region of the larynx.

Methods

We report the first case of juvenile xanthogranulomatosis (JXG) in the subglottis in a 3 year old child.

Results

The localization in the subglottis caused airway obstruction requiring tracheostomy to secure the airway. On the basis that most cutaneous lesions regress spontaneously the lesion was managed expectantly and regressed over a period of 28 months allowing decannulation of the child.

Conclusion

JXG should be considered in the differential diagnosis of subglottic lesions. Once the airway has been secured, JXG of the subglottis can be managed conservatively. Long-term follow-up is required because of the possibility of relapse at other sites. © 2001 John Wiley & Sons, Inc. Head Neck 23: 426–429, 2001.

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