| dc.contributor.author | Buendía Martínez, Juan | |
| dc.contributor.author | Barreda Sánchez, María | |
| dc.contributor.author | Rodríguez Peña, Lidya | |
| dc.contributor.author | Ballesta Martínez, María Juliana | |
| dc.contributor.author | López González, Vanesa | |
| dc.contributor.author | Sánchez Soler, María José | |
| dc.contributor.author | Serrano Antón, Ana Teresa | |
| dc.contributor.author | Pérez Tomás, María Elena | |
| dc.contributor.author | Gil Ferrer, Remedios | |
| dc.contributor.author | Avilés Plaza, Francisco | |
| dc.contributor.author | Glover López, Guillermo | |
| dc.contributor.author | Carazo Díaz, Carmen | |
| dc.contributor.author | Guillén Navarro, Encarna | |
| dc.date.accessioned | 2026-02-10T13:29:24Z | |
| dc.date.available | 2026-02-10T13:29:24Z | |
| dc.date.issued | 2021 | |
| dc.identifier.citation | Buendía-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/s13023-021-01742-3 | es |
| dc.identifier.uri | http://hdl.handle.net/10952/10791 | |
| dc.description | También se accede al texto publicado del documento a través de la página web de la revista https://link.springer.com/article/10.1186/s13023-021-01742-3 | es |
| dc.description.abstract | Background: 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.iso | en | es |
| dc.rights | Attribution-NonCommercial-NoDerivatives 4.0 Internacional | * |
| dc.rights.uri | http://creativecommons.org/licenses/by-nc-nd/4.0/ | * |
| dc.subject | Acute intermittent porphyria | es |
| dc.subject | Chronic symptoms | es |
| dc.subject | Quality of life | es |
| dc.subject | Sporadic attacks | es |
| dc.subject | Chronic kidney disease | es |
| dc.title | Health impact of acute intermittent porphyria in latent and non‑recurrent attacks patients | es |
| dc.type | journal article | es |
| dc.rights.accessRights | open access | es |
| dc.journal.title | Orphanet Journal of Rare Diseases | es |
| dc.volume.number | 16 | es |
| dc.issue.number | 106 | es |
| dc.description.discipline | Medicina | es |
| dc.identifier.doi | 10.1186/s13023-021-01742-3 | es |
| dc.description.faculty | Medicina | es |