ArticleAACE clinical case reports
Xanthomatous Hypophysitis Presenting in an Adolescent Girl: A Long-Term Follow-Up of a Rare Case and Review of the Literature.
Article in AACE clinical case reports. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.
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Who cites it
4 citing papers in PubMed, 7 citations in OpenAlex.
- Autoimmune hypophysitis associated with rheumatoid arthritis: Histopathological insights from a rare biopsy-proven case.Surgical neurology international · 2025Article
- A novel approach to hypophysitis: outcomes using non-glucocorticoid immunosuppressive therapy.European journal of endocrinology · 2023Review
- Successful immunomodulatory treatment for recurrent xanthogranulomatous hypophysitis in an adolescent: illustrative case.Journal of neurosurgery. Case lessons · 2022Article
- Xanthomatous Hypophysitis Secondary to a Ruptured Rathke's Cleft Cyst: A Case Report.Brain tumor research and treatment · 2022Article
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Authors and funding
7 authors at 1 institution in 1 country.
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Abstract
objectivePrimary hypophysitis refers to the isolated inflammation of the pituitary gland not associated with other secondary causes. Among its histopathologic subtypes, xanthomatous is the rarest.
methodsWe describe a 22-year-old woman with xanthomatous hypophysitis (XH), its clinical progression over 8 years as well as the treatment effects of prednisolone and azathioprine. Our patient was first referred for severe short stature and delayed puberty at the age of 14 years.
resultsInvestigations revealed multiple pituitary deficiencies. Magnetic resonance imaging showed a pituitary mass whereby a partial resection was performed. A full resection was not feasible due to the location of the mass. The histopathologic analysis of the tissue was consistent with XH. The results of secondary workout for neoplasm, infection, autoimmune, and inflammatory disorders were negative. After surgery, a progressive enlargement of the mass was observed. Two courses of prednisolone were administered with a significant reduction in the mass size. Azathioprine was added due to the unsustained effects of prednisolone when tapered off and the concern of steroid toxicity with continued use. No further increase in the mass size was noted after 6 months on azathioprine.
conclusionGlucocorticoid and immunotherapy are treatment options for XH; however, more cases are needed to better understand its pathogenesis and clinical progression.
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