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A difficult case
Xanthomatous hypophysitis relapsing and remitting over two decades
  1. Kabir Khan Nazeer1,
  2. Salhin Alatrash2,
  3. Sawsan Elsheikh3,
  4. Saiju Jacob4,5
  1. 1Neurology, Royal Stoke University Hospital, Stoke-on-Trent, UK
  2. 2University Hospitals Birmingham NHS Foundation Trust, Birmingham, England, UK
  3. 3Ophthalmology, University Hospitals Birmingham NHS Foundation Trust, Birmingham, Birmingham, UK
  4. 4Neurology, University of Birmingham Institute of Immunology and Immunotherapy, Birmingham, UK
  5. 5Department of Neurology, University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK
  1. Correspondence to Dr Kabir Khan Nazeer; Kabir-Khan.Nazeer{at}nhs.net

Abstract

Inflammation of the pituitary gland can be primary (without another underlying cause) or secondary (associated with a systemic inflammatory condition). Primary hypophysitis is very rare, among which xanthomatous hypophysitis as a histological type is extremely unusual. A woman in her late 50s presented with recurrent pituitary lesions over 20 years. Her general practitioner had diagnosed panhypopituitarism in her 30s; a decade later, she had presented to ophthalmology with visual loss and restricted visual fields, and a pituitary lesion was found. This recurred several times requiring multiple resections. Histopathology showed atypical inflammation in keeping with xanthomatous hypophysitis; this responded well to corticosteroid therapy. Xanthomatous hypophysitis is a rare form of steroid-responsive primary pituitary inflammation, to consider in the differential diagnosis of recurring pituitary lesions.

  • NEUROIMMUNOLOGY
  • NEUROOPHTHALMOLOGY
  • NEUROPATHOLOGY
  • VISION
  • NEUROENDOCRINOLOGY

Data availability statement

Data sharing not applicable as no datasets generated and/or analysed for this study. Not applicable.All references are included within the article."No publicly available datasets were used in the preparation of this case report’.

https://doi.org/10.1136/pn-2024-004430

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    Data availability statement

    Data sharing not applicable as no datasets generated and/or analysed for this study. Not applicable.All references are included within the article."No publicly available datasets were used in the preparation of this case report’.

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    Footnotes

    • Contributors KKN contributed to writing the first draft of the manuscript, gathered images from radiology, histopathology for inclusion in the manuscript and was the corresponding author. SE also contributed to writing the first draft of the manuscript and collating ophthalmology records. SA consented the patient for case report, reviewed and edited the manuscript and images. SJ was the senior author who guided the drafts and revisions. KKN is the guarantor.

    • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

    • Competing interests None declared.

    • Provenance and peer review Not commissioned; externally peer reviewed by Claire Rice, Bristol, UK.