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Indomethacin-responsive refractory headache: Two case reports in children after hemispherectomy for Rasmussen's
Jessica Hauser Chatterjee1, Priya A Monrad1, Hannah E Goldstein2
1Division 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.
Two children with headaches after surgery for Rasmussen's encephalitis experienced rapid relief with low-dose indomethacin. This suggests indomethacin may treat refractory side-locked headaches in pediatric neuroinflammatory conditions.
Area of Science:
- Pediatric Neurology
- Headache Medicine
- Neuroinflammation
Background:
- Indomethacin-responsive headaches, including trigeminal autonomic cephalalgias (TACs), are observed in youth.
- TACs are rare in pediatrics and may present differently than in adults.
- Secondary causes of side-locked headaches necessitate imaging; post-craniotomy headaches responsive to indomethacin are documented in adults but not children.
Purpose of the Study:
- To describe the occurrence and treatment of post-craniotomy headaches in children with a history of Rasmussen's encephalitis.
- To evaluate the efficacy of indomethacin in managing refractory side-locked headaches in this pediatric population.
Main Methods:
- Case report of two children with Rasmussen's encephalitis who underwent functional hemispherectomies.
- Observation of headache development and subsequent treatment with indomethacin.
Main Results:
- Both children developed debilitating, side-locked headaches ipsilateral to their encephalitis and surgery, months post-operation.
- Headaches were rapidly and completely responsive to low-dose indomethacin (0.14-0.5 mg/kg/day), with sustained headache freedom.
Conclusions:
- Refractory side-locked headaches in children following craniotomy or neuroinflammatory conditions warrant consideration of indomethacin.
- Indomethacin may be a valuable treatment option after excluding other secondary causes.
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