Long-Term Outcome of Indomethacin Treatment in Pediatric Patients with Paroxysmal Hemicrania—A Case Series

Paroxysmal Hemicrania is a rare form of primary headache in children and adolescents, belonging to the group of trigeminal autonomic cephalalgias. Patients suffer from severe, short-lasting unilateral headaches accompanied by symptoms of the autonomic system on the same side of the head. The short d...

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Main Authors: Maximilian David Mauritz, Anna Enninger, Christine Wamsler, Julia Wager, Boris Zernikow
Format: Article
Language:English
Published: MDPI AG 2021-02-01
Series:Children
Subjects:
Online Access:https://www.mdpi.com/2227-9067/8/2/101
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spelling doaj-902a185d17eb4a9181c5db7723b0291b2021-04-02T19:20:51ZengMDPI AGChildren2227-90672021-02-01810110110.3390/children8020101Long-Term Outcome of Indomethacin Treatment in Pediatric Patients with Paroxysmal Hemicrania—A Case SeriesMaximilian David Mauritz0Anna Enninger1Christine Wamsler2Julia Wager3Boris Zernikow4German Paediatric Pain Centre, Children′s and Adolescents′ Hospital, Witten/Herdecke University, 45711 Datteln, GermanyDepartment of Children′s Pain Therapy and Paediatric Palliative Care, Faculty of Health, School of Medicine, Witten/Herdecke University, 58448 Witten, GermanyGerman Paediatric Pain Centre, Children′s and Adolescents′ Hospital, Witten/Herdecke University, 45711 Datteln, GermanyGerman Paediatric Pain Centre, Children′s and Adolescents′ Hospital, Witten/Herdecke University, 45711 Datteln, GermanyGerman Paediatric Pain Centre, Children′s and Adolescents′ Hospital, Witten/Herdecke University, 45711 Datteln, GermanyParoxysmal Hemicrania is a rare form of primary headache in children and adolescents, belonging to the group of trigeminal autonomic cephalalgias. Patients suffer from severe, short-lasting unilateral headaches accompanied by symptoms of the autonomic system on the same side of the head. The short duration of attacks distinguishes Paroxysmal Hemicrania from other trigeminal autonomic cephalalgias. Indomethacin is the treatment of choice, and its effectiveness provides a unique diagnostic criterion. However, the long-term outcomes in children are highly underreported. In this case-series, <i>n</i> = 8 patients diagnosed with Paroxysmal Hemicrania were contacted via telephone 3.1 to 10.7 years after initial presentation. A standardized interview was conducted. <i>n</i> = 6 patients were headache-free and no longer took indomethacin for 5.4 ± 3.4 years. The mean treatment period in these patients was 2.2 ± 1.9 years. Weaning attempts were undertaken after 1.7 ± 1.3 months; in <i>n</i> = 3 patients, more than one weaning attempt was necessary. <i>n</i> = 2 patients were still taking indomethacin (4.5 and 4.9 years, respectively). Both unsuccessfully tried to reduce the indomethacin treatment (two and six times, respectively). Adverse effects appeared in <i>n</i> = 6 (75%) patients and led to a discontinuation of therapy in <i>n</i> = 2 patients. Our long-term follow-up suggests that in a substantial proportion of pediatric patients, discontinuing indomethacin therapy is possible without the recurrence of Paroxysmal Hemicrania.https://www.mdpi.com/2227-9067/8/2/101headachepaintrigeminal autonomic cephalalgiasparoxysmal hemicraniachildrenindomethacin
collection DOAJ
language English
format Article
sources DOAJ
author Maximilian David Mauritz
Anna Enninger
Christine Wamsler
Julia Wager
Boris Zernikow
spellingShingle Maximilian David Mauritz
Anna Enninger
Christine Wamsler
Julia Wager
Boris Zernikow
Long-Term Outcome of Indomethacin Treatment in Pediatric Patients with Paroxysmal Hemicrania—A Case Series
Children
headache
pain
trigeminal autonomic cephalalgias
paroxysmal hemicrania
children
indomethacin
author_facet Maximilian David Mauritz
Anna Enninger
Christine Wamsler
Julia Wager
Boris Zernikow
author_sort Maximilian David Mauritz
title Long-Term Outcome of Indomethacin Treatment in Pediatric Patients with Paroxysmal Hemicrania—A Case Series
title_short Long-Term Outcome of Indomethacin Treatment in Pediatric Patients with Paroxysmal Hemicrania—A Case Series
title_full Long-Term Outcome of Indomethacin Treatment in Pediatric Patients with Paroxysmal Hemicrania—A Case Series
title_fullStr Long-Term Outcome of Indomethacin Treatment in Pediatric Patients with Paroxysmal Hemicrania—A Case Series
title_full_unstemmed Long-Term Outcome of Indomethacin Treatment in Pediatric Patients with Paroxysmal Hemicrania—A Case Series
title_sort long-term outcome of indomethacin treatment in pediatric patients with paroxysmal hemicrania—a case series
publisher MDPI AG
series Children
issn 2227-9067
publishDate 2021-02-01
description Paroxysmal Hemicrania is a rare form of primary headache in children and adolescents, belonging to the group of trigeminal autonomic cephalalgias. Patients suffer from severe, short-lasting unilateral headaches accompanied by symptoms of the autonomic system on the same side of the head. The short duration of attacks distinguishes Paroxysmal Hemicrania from other trigeminal autonomic cephalalgias. Indomethacin is the treatment of choice, and its effectiveness provides a unique diagnostic criterion. However, the long-term outcomes in children are highly underreported. In this case-series, <i>n</i> = 8 patients diagnosed with Paroxysmal Hemicrania were contacted via telephone 3.1 to 10.7 years after initial presentation. A standardized interview was conducted. <i>n</i> = 6 patients were headache-free and no longer took indomethacin for 5.4 ± 3.4 years. The mean treatment period in these patients was 2.2 ± 1.9 years. Weaning attempts were undertaken after 1.7 ± 1.3 months; in <i>n</i> = 3 patients, more than one weaning attempt was necessary. <i>n</i> = 2 patients were still taking indomethacin (4.5 and 4.9 years, respectively). Both unsuccessfully tried to reduce the indomethacin treatment (two and six times, respectively). Adverse effects appeared in <i>n</i> = 6 (75%) patients and led to a discontinuation of therapy in <i>n</i> = 2 patients. Our long-term follow-up suggests that in a substantial proportion of pediatric patients, discontinuing indomethacin therapy is possible without the recurrence of Paroxysmal Hemicrania.
topic headache
pain
trigeminal autonomic cephalalgias
paroxysmal hemicrania
children
indomethacin
url https://www.mdpi.com/2227-9067/8/2/101
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