Abstract
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, n = 8 patients diagnosed with Paroxysmal Hemicrania were contacted via telephone 3.1 to 10.7 years after initial presentation. A standardized interview was conducted. n = 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 n = 3 patients, more than one weaning attempt was necessary. n = 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 n = 6 (75%) patients and led to a discontinuation of therapy in n = 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.
Highlights
Paroxysmal Hemicrania is a rare form of primary headache in children and adolescents, belonging to the group of trigeminal autonomic cephalalgias
Between 2003 and 2017, n = 16 patients had a suspected or differential diagnosis of Paroxysmal Hemicrania (PH) at the German Paediatric Pain Centre, Children’s and Adolescents’ Hospital DattelnWitten/Herdecke University. n = 12 patients agreed to participate in the telephone interview. n = 4 patients were excluded because their suspected diagnosis of PH was not applicable, i.e., only patients with a confirmed PH diagnosis were included
The patients included in this study reported several side effects of indomethacin: The patientsproblems includedlike in this study reported several side of indomethacin: gasgastrointestinal abdominal pain, diarrhea, andeffects constipation (n = 4), edema trointestinal problems like abdominal pain, diarrhea, and constipation
Summary
Paroxysmal Hemicrania is a rare form of primary headache in children and adolescents, belonging to the group of trigeminal autonomic cephalalgias. The long-term outcomes in children are highly underreported In this case-series, n = 8 patients diagnosed with Paroxysmal Hemicrania were contacted via telephone 3.1 to 10.7 years after initial presentation. 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. 1. Introduction with regard to jurisdictional claims in Trigeminal autonomic cephalalgias (TAC) are a group of rare primary headache disorders. Introduction with regard to jurisdictional claims in Trigeminal autonomic cephalalgias (TAC) are a group of rare primary headache disorders They are characterized by sudden unilateral headaches in the trigeminal nerve region, accompanied by symptoms of the autonomic nervous system on the same side of the head. Distinguishes Cluster Headache, Paroxysmal Hemicrania, SUNCT syndrome (Short-lasting unilateral neuralgiform headache with conjunctival injection and tearing), SUNA syndrome (Short-lasting unilateral neuralgiform headache attacks with cranial autonomic symptoms), and Hemicrania Continua
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