Oral ATP treatment in alternating hemiplegia of childhood: a case report and review
Alternating hemiplegia of childhood (AHC) is a rare neurological disorder that usually manifests before 18 months of age and is characterized by recurrent, alternating episodes of hemiparesis with variable frequency and can last from a few minutes to several days. We present a case of AHC in a littl...
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Frontiers Media S.A.
2025-01-01
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| Series: | Frontiers in Medicine |
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| Online Access: | https://www.frontiersin.org/articles/10.3389/fmed.2024.1433217/full |
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| author | Marco Carrozzi Maria Elisa Morelli Mario Cirino Alessandra Maestro Gilda Paternuosto Giulia Benericetti Giada Bennati Maura Bin Anna Flamigni Federico Pigato Natalia Maximova Egidio Barbi Egidio Barbi Davide Zanon |
| author_facet | Marco Carrozzi Maria Elisa Morelli Mario Cirino Alessandra Maestro Gilda Paternuosto Giulia Benericetti Giada Bennati Maura Bin Anna Flamigni Federico Pigato Natalia Maximova Egidio Barbi Egidio Barbi Davide Zanon |
| author_sort | Marco Carrozzi |
| collection | DOAJ |
| description | Alternating hemiplegia of childhood (AHC) is a rare neurological disorder that usually manifests before 18 months of age and is characterized by recurrent, alternating episodes of hemiparesis with variable frequency and can last from a few minutes to several days. We present a case of AHC in a little girl carrying a sporadic mutation in the ATP1A3 gene (p.Glu815Lys) refractory to flunarizine and non-compliant to topiramate due to adverse effects treated with oral compound of adenosine-5′-triphosphate (ATP) capsules. Outcome was evaluated through the follow-up and side effects and safety were monitored regularly. Compounded drug showed effectiveness and safety. Indeed, during the four-year follow-up, with the dose of adenosine-5′-triphosphate gradually increasing up to 21 mg/kg, the patient showed a substantial benefit in controlling the frequency and duration of hemiplegic episodes and an improvement in neurological deterioration. |
| format | Article |
| id | doaj-art-363dfc03b7754a4ea79cd4e7f311de8c |
| institution | Kabale University |
| issn | 2296-858X |
| language | English |
| publishDate | 2025-01-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Medicine |
| spelling | doaj-art-363dfc03b7754a4ea79cd4e7f311de8c2025-01-07T06:51:41ZengFrontiers Media S.A.Frontiers in Medicine2296-858X2025-01-011110.3389/fmed.2024.14332171433217Oral ATP treatment in alternating hemiplegia of childhood: a case report and reviewMarco Carrozzi0Maria Elisa Morelli1Mario Cirino2Alessandra Maestro3Gilda Paternuosto4Giulia Benericetti5Giada Bennati6Maura Bin7Anna Flamigni8Federico Pigato9Natalia Maximova10Egidio Barbi11Egidio Barbi12Davide Zanon13Institute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyInstitute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyInstitute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyInstitute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyInstitute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyInstitute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyInstitute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyInstitute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyInstitute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyInstitute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyInstitute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyInstitute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyDepartment of Medicine, Surgery and Health Sciences, University of Trieste, Trieste, ItalyInstitute for Maternal and Child Health Burlo Garofolo (IRCCS), Trieste, ItalyAlternating hemiplegia of childhood (AHC) is a rare neurological disorder that usually manifests before 18 months of age and is characterized by recurrent, alternating episodes of hemiparesis with variable frequency and can last from a few minutes to several days. We present a case of AHC in a little girl carrying a sporadic mutation in the ATP1A3 gene (p.Glu815Lys) refractory to flunarizine and non-compliant to topiramate due to adverse effects treated with oral compound of adenosine-5′-triphosphate (ATP) capsules. Outcome was evaluated through the follow-up and side effects and safety were monitored regularly. Compounded drug showed effectiveness and safety. Indeed, during the four-year follow-up, with the dose of adenosine-5′-triphosphate gradually increasing up to 21 mg/kg, the patient showed a substantial benefit in controlling the frequency and duration of hemiplegic episodes and an improvement in neurological deterioration.https://www.frontiersin.org/articles/10.3389/fmed.2024.1433217/fulladenosine-5′-triphosphateATPalternating hemiplegia of childhoodrare diseasecompoundclinical galenic |
| spellingShingle | Marco Carrozzi Maria Elisa Morelli Mario Cirino Alessandra Maestro Gilda Paternuosto Giulia Benericetti Giada Bennati Maura Bin Anna Flamigni Federico Pigato Natalia Maximova Egidio Barbi Egidio Barbi Davide Zanon Oral ATP treatment in alternating hemiplegia of childhood: a case report and review Frontiers in Medicine adenosine-5′-triphosphate ATP alternating hemiplegia of childhood rare disease compound clinical galenic |
| title | Oral ATP treatment in alternating hemiplegia of childhood: a case report and review |
| title_full | Oral ATP treatment in alternating hemiplegia of childhood: a case report and review |
| title_fullStr | Oral ATP treatment in alternating hemiplegia of childhood: a case report and review |
| title_full_unstemmed | Oral ATP treatment in alternating hemiplegia of childhood: a case report and review |
| title_short | Oral ATP treatment in alternating hemiplegia of childhood: a case report and review |
| title_sort | oral atp treatment in alternating hemiplegia of childhood a case report and review |
| topic | adenosine-5′-triphosphate ATP alternating hemiplegia of childhood rare disease compound clinical galenic |
| url | https://www.frontiersin.org/articles/10.3389/fmed.2024.1433217/full |
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