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Case Report

Idiopathic Hypertrophic Pachymeningitis: Does Earlier Treatment Improve Outcome?

1
Faculty of Medicine and Surgery, University of Catania, Piazza dell’Università 2, 95124 Catania, Italy
2
Great Ormond Street Hospital for Children NHS Foundation Trust, London WC1N 3JH, UK
3
Paediatric Ophthalmology, Evelina London Children’s Hospital at Guy’s and St Thomas’ NHS Foundation Trust, London SE1 7EH, UK
4
Paediatric Rheumatology, Evelina London Children’s Hospital at Guy’s and St Thomas’ NHS Foundation Trust, London SE1 7EH, UK
5
Department of Neuroradiology, King’s College Hospital NHS Foundation Trust, London SE5 9RS, UK
6
Children’s Neurosciences, Evelina London Children’s Hospital at Guy’s and St Thomas’ NHS Foundation Trust, King’s Health Partners Academic Health Science Centre, London SE1 7RS, UK
7
Faculty of Life Sciences and Medicine, Kings College London, London SE1 7RS, UK
*
Author to whom correspondence should be addressed.
Children 2021, 8(1), 11; https://doi.org/10.3390/children8010011
Received: 23 November 2020 / Revised: 23 December 2020 / Accepted: 25 December 2020 / Published: 28 December 2020
(This article belongs to the Section Child Neurology)
Background/goal: Hypertrophic pachymeningitis is a rare chronic inflammatory disorder characterized by marked fibrous thickening of the cerebral and/or spinal dura mater. This condition has largely been reported in adults, but there are very few reports in children. Methods: We describe a 14-year-old boy with idiopathic hypertrophic pachymeningitis, who presented with deteriorating vision on a background of severe headache. We evaluated pediatric cases of hypertrophic pachymeningitis and compared treatments and their relation to outcomes. Results: There are only eleven pediatric cases of hypertrophic pachymeningitis reported in the literature. In the patients treated with steroids either at presentation or subsequent relapses, a good response was reported. In the cases with delayed initiation of steroid treatment, this was often related to an incomplete recovery. In our patient, this delay may have contributed to his poor visual outcome. Conclusions: Early initiation of steroid treatment in children with idiopathic hypertrophic pachymeningitis may improve outcomes. View Full-Text
Keywords: dural thickening; cranial neuropathy; immunotherapy; sarcoid; steroids; visual failure dural thickening; cranial neuropathy; immunotherapy; sarcoid; steroids; visual failure
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MDPI and ACS Style

Rizzo, E.; Ritchie, A.E.; Shivamurthy, V.; Siddiqui, A.; Lim, M. Idiopathic Hypertrophic Pachymeningitis: Does Earlier Treatment Improve Outcome? Children 2021, 8, 11. https://doi.org/10.3390/children8010011

AMA Style

Rizzo E, Ritchie AE, Shivamurthy V, Siddiqui A, Lim M. Idiopathic Hypertrophic Pachymeningitis: Does Earlier Treatment Improve Outcome? Children. 2021; 8(1):11. https://doi.org/10.3390/children8010011

Chicago/Turabian Style

Rizzo, Emilia, Ailsa E. Ritchie, Vinay Shivamurthy, Ata Siddiqui, and Ming Lim. 2021. "Idiopathic Hypertrophic Pachymeningitis: Does Earlier Treatment Improve Outcome?" Children 8, no. 1: 11. https://doi.org/10.3390/children8010011

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