关键词: 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

来  源:   DOI:10.3171/CASE22191   PDF(Pubmed)

Abstract:
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.
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.
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.
摘要:
背景:垂体黄色瘤病变与Rathke裂囊肿破裂或出血性有关。据报道,大多数病例在根治性切除术后消退。当复发发生时,没有既定的治疗方案。据报道,在一些已发表的病例中,大剂量糖皮质激素是有益的;然而,一旦停止治疗,它们的效果通常不会持续。
方法:作者报告了一例青少年男性,尽管进行了两次手术干预,但还是出现了与Rathke囊肿相关的复发性黄色肉芽肿性垂体炎。他接受了短期地塞米松治疗,然后接受了塞来昔布和霉酚酸酯的维持治疗。该方案被证明是安全和耐受性良好的,它成功地阻止了他的黄色肉芽肿性垂体炎的另一次复发。
结论:本病例证明了一种治疗复发性黄色肉芽肿性垂体炎的新的非手术方法。这表明保留皮质类固醇的免疫抑制和抗炎联合方案在其他难治性黄色肉芽肿性垂体炎病例中的潜在应用。
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