Successful immunomodulatory treatment for recurrent xanthogranulomatous hypophysitis in an adolescent: illustrative case.
CSF = cerebrospinal fluid
MMF = mycophenolate mofetil
MRI = magnetic resonance imaging
RCC = Rathke’s cleft cyst
Rathke’s cleft cyst
XG = xanthogranuloma
XGH = xanthogranulomatous hypophysitis
XH = xanthomatous hypophysitis
hypophysitis
immunosuppressive therapy
suprasellar
xanthogranulomatous
xanthomatous
Journal
Journal of neurosurgery. Case lessons
ISSN: 2694-1902
Titre abrégé: J Neurosurg Case Lessons
Pays: United States
ID NLM: 9918227275606676
Informations de publication
Date de publication:
29 Aug 2022
29 Aug 2022
Historique:
received:
25
04
2022
accepted:
09
06
2022
entrez:
2
9
2022
pubmed:
3
9
2022
medline:
3
9
2022
Statut:
epublish
Résumé
Xanthomatous lesions of the pituitary have been linked to ruptured or hemorrhagic Rathke's cleft cysts. Most cases are reported to resolve following radical resection. When recurrence does occur, there is no established treatment regimen. High-dose glucocorticoids have been reported to be beneficial in several published cases; however, their effects are often not sustained once therapy is discontinued. The authors report the case of an adolescent male who developed recurrent xanthogranulomatous hypophysitis associated with a Rathke's cleft cyst despite two surgical interventions. He was treated with a short course of dexamethasone followed by a maintenance course of celecoxib and mycophenolate mofetil. This regimen proved to be safe and well-tolerated, and it successfully prevented another recurrence of his xanthogranulomatous hypophysitis. This case demonstrates a novel nonsurgical approach to the management of recurrent xanthogranulomatous hypophysitis. It suggests a potential application of a combined corticosteroid-sparing immunosuppressive and anti-inflammatory regimen in other cases of refractory xanthogranulomatous hypophysitis.
Sections du résumé
BACKGROUND
BACKGROUND
Xanthomatous lesions of the pituitary have been linked to ruptured or hemorrhagic Rathke's cleft cysts. Most cases are reported to resolve following radical resection. When recurrence does occur, there is no established treatment regimen. High-dose glucocorticoids have been reported to be beneficial in several published cases; however, their effects are often not sustained once therapy is discontinued.
OBSERVATIONS
METHODS
The authors report the case of an adolescent male who developed recurrent xanthogranulomatous hypophysitis associated with a Rathke's cleft cyst despite two surgical interventions. He was treated with a short course of dexamethasone followed by a maintenance course of celecoxib and mycophenolate mofetil. This regimen proved to be safe and well-tolerated, and it successfully prevented another recurrence of his xanthogranulomatous hypophysitis.
LESSONS
CONCLUSIONS
This case demonstrates a novel nonsurgical approach to the management of recurrent xanthogranulomatous hypophysitis. It suggests a potential application of a combined corticosteroid-sparing immunosuppressive and anti-inflammatory regimen in other cases of refractory xanthogranulomatous hypophysitis.
Identifiants
pubmed: 36051774
doi: 10.3171/CASE22191
pii: CASE22191
pmc: PMC9426350
doi:
pii:
Types de publication
Case Reports
Langues
eng
Pagination
CASE22191Informations de copyright
© 2022 The authors.
Déclaration de conflit d'intérêts
Disclosures Dr. Recinos reported consulting fees from Stryker outside the submitted work, and ownership interest in Acera Surgical. No other disclosures were reported.
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