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吲哚美辛反应性难治性头痛:2例拉斯姆森脑炎患儿半球切除术后病例报告

Indomethacin-responsive refractory headache: Two case reports in children after hemispherectomy for Rasmussen's encephalitis.

作者信息

Hauser Chatterjee Jessica, Monrad Priya A, Goldstein Hannah E, Shaw Dennis W, Ojemann Jeffrey G, Randle Stephanie Carapetian, Hauptman Jason S, Blume Heidi K

机构信息

Division of Pediatric Neurology, Department of Neurology, Seattle Children's Research Institute, Norcliffe Foundation Center for Integrative Brain Research, University of Washington School of Medicine, Seattle, Washington, USA.

Division of Pediatric Neurosurgery, Department of Neurological Surgery, Seattle Children's Hospital, University of Washington, Seattle, Washington, USA.

出版信息

Headache. 2025 Jun;65(6):1044-1048. doi: 10.1111/head.14928. Epub 2025 Mar 25.

DOI:10.1111/head.14928
PMID:40129243
Abstract

BACKGROUND

Indomethacin-responsive headaches occur in youth and include primary headache syndromes such as hemicrania continua and paroxysmal hemicrania. Both are trigeminal autonomic cephalalgias (TACs). In pediatrics TACs are rare. Moreover, they may differ in their phenotypes and response to indomethacin compared to adults. Secondary causes for side-locked headaches can have vascular, neoplastic, and inflammatory etiologies, emphasizing the importance of imaging in the evaluation of these headache types. Post-craniotomy indomethacin-responsive headaches have been described in adults, but not in children.

METHODS

Written consent was obtained from the patients' families and written assent from the two children for publication.

RESULTS

We report the course of two children, both with a history of Rasmussen's encephalitis treated with functional hemispherectomies, who subsequently developed debilitating, side-locked, medically refractory headaches several months after surgery. Headaches were on the same side of their encephalitis and surgery. In both instances, the headaches were exquisitely and rapidly responsive to indomethacin. Headache freedom was maintained on low doses of indomethacin (0.14-0.5 mg/kg/day).

CONCLUSION

In cases of refractory side-locked headaches following a craniotomy or neuroinflammatory condition in children, one may consider indomethacin as a treatment option after evaluation for other secondary etiologies.

摘要

背景

对吲哚美辛有反应的头痛在青少年中出现,包括原发性头痛综合征,如持续性偏侧头痛和发作性偏侧头痛。两者均为三叉自主神经性头痛(TACs)。在儿科中,TACs很罕见。此外,与成人相比,它们在表型和对吲哚美辛的反应方面可能有所不同。一侧固定性头痛的继发性病因可能有血管性、肿瘤性和炎症性病因,这强调了影像学在评估这些头痛类型中的重要性。成人中已描述了开颅术后对吲哚美辛有反应的头痛,但儿童中尚未见报道。

方法

获得了患者家属的书面同意以及两名儿童对发表的书面同意。

结果

我们报告了两名儿童的病程,他们都有接受功能性大脑半球切除术治疗的拉斯穆森脑炎病史,术后数月随后出现使人衰弱的、一侧固定性的、药物难治性头痛。头痛位于他们脑炎和手术的同一侧。在这两个病例中,头痛对吲哚美辛极为迅速地产生反应。低剂量吲哚美辛(0.14 - 0.5毫克/千克/天)可维持无头痛状态。

结论

对于儿童开颅术后或神经炎症性疾病后难治性一侧固定性头痛的病例,在评估其他继发性病因后,可考虑将吲哚美辛作为一种治疗选择。

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