DeCou Sarah, Recinos Pablo F, Prayson Richard A, Karakasis Christopher, Haider Anzar, Patel Neha
1Cleveland Clinic Lerner College of Medicine of Case Western Reserve University, Cleveland, Ohio.
3Rose Ella Burkhardt Brain Tumor & Neuro-Oncology Center, Neurological Institute, Cleveland Clinic, Cleveland, Ohio.
J Neurosurg Case Lessons. 2022 Aug 29;4(9):CASE22191. doi: 10.3171/CASE22191.
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.
垂体黄色瘤样病变与破裂或出血性拉克氏囊肿有关。据报道，大多数病例在根治性切除后可缓解。当复发确实发生时，尚无既定的治疗方案。在几例已发表的病例中，高剂量糖皮质激素已被报道有益；然而，一旦停药，其效果往往难以持续。
作者报告了一例青少年男性病例，尽管进行了两次手术干预，但仍发生了与拉克氏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，随后接受塞来昔布和霉酚酸酯维持治疗。该方案被证明是安全且耐受性良好的，并成功预防了他的黄色肉芽肿性垂体炎再次复发。
该病例展示了一种治疗复发性黄色肉芽肿性垂体炎的新型非手术方法。它提示了联合使用减少糖皮质激素用量的免疫抑制和抗炎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。
