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dc.contributor.authorBuendía Martínez, Juan
dc.contributor.authorBarreda Sánchez, María
dc.contributor.authorRodríguez Peña, Lidya
dc.contributor.authorBallesta Martínez, María Juliana
dc.contributor.authorLópez González, Vanesa
dc.contributor.authorSánchez Soler, María José
dc.contributor.authorSerrano Antón, Ana Teresa
dc.contributor.authorPérez Tomás, María Elena
dc.contributor.authorGil Ferrer, Remedios
dc.contributor.authorAvilés Plaza, Francisco
dc.contributor.authorGlover López, Guillermo
dc.contributor.authorCarazo Díaz, Carmen
dc.contributor.authorGuillén Navarro, Encarna
dc.date.accessioned2026-02-10T13:29:24Z
dc.date.available2026-02-10T13:29:24Z
dc.date.issued2021
dc.identifier.citationBuendía-Martínez, J., Barreda-Sánchez, M., Rodríguez-Peña, L. et al. Health impact of acute intermittent porphyria in latent and non-recurrent attacks patients. Orphanet J Rare Dis 16, 106 (2021). https://doi.org/10.1186/s13023-021-01742-3es
dc.identifier.urihttp://hdl.handle.net/10952/10791
dc.descriptionTambién se accede al texto publicado del documento a través de la página web de la revista https://link.springer.com/article/10.1186/s13023-021-01742-3es
dc.description.abstractBackground: Acute intermittent porphyria (AIP) is a genetic disease characterized by acute neurovisceral attacks. Long-term clinical conditions, chronic symptoms and impaired health related quality of life (HRQoL) have been reported during non-attack periods but mainly in patients with recurrent attacks. Our aim was to investigate these aspects in sporadic AIP (SA-AIP) and latent AIP (L-AIP) patients. Fifty-five participants, 27 SA-AIP (< 4 attacks/year) and 28 L-AIP patients with a prevalent founder mutation from Spain were included. Medical records were reviewed, and individual interviews, physical examinations, biochemical analyses, and abdominal ultrasound scans were conducted. HRQoL was assessed through an EQ-5D-5L questionnaire. A comparative study was made between SA-AIP and L-AIP patients. Results: The earliest long-term clinical condition associated with SA-AIP was chronic kidney disease. Chronic symptoms were reported in 85.2 % of SA-AIP and 46.4 % of L-AIP patients. Unspecific abdominal pain, fatigue, muscle pain and insomnia were significantly more frequent in SA-AIP than in L-AIP patients. The EQ-5D-5L index was lower in SAAIP (0.809 vs. 0.926, p = 0.0497), and the impact of “pain”, “anxiety-depression” and “mobility” was more intense in the EQ-5D-5L domains in SA-AIP than in L-AIP subjects and the general Spanish population. Conclusions: AIP remains a chronically symptomatic disease that adversely affects health and quality of life, even in patients with low rate of acute attacks. We suggest a regular monitoring of patients with symptomatic AIP regardless of their attack rate or the time since their last attack, with proper pain management and careful attention to kidney function.es
dc.language.isoenes
dc.rightsAttribution-NonCommercial-NoDerivatives 4.0 Internacional*
dc.rights.urihttp://creativecommons.org/licenses/by-nc-nd/4.0/*
dc.subjectAcute intermittent porphyriaes
dc.subjectChronic symptomses
dc.subjectQuality of lifees
dc.subjectSporadic attackses
dc.subjectChronic kidney diseasees
dc.titleHealth impact of acute intermittent porphyria in latent and non‑recurrent attacks patientses
dc.typejournal articlees
dc.rights.accessRightsopen accesses
dc.journal.titleOrphanet Journal of Rare Diseaseses
dc.volume.number16es
dc.issue.number106es
dc.description.disciplineMedicinaes
dc.identifier.doi10.1186/s13023-021-01742-3es
dc.description.facultyMedicinaes


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Attribution-NonCommercial-NoDerivatives 4.0 Internacional
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