Long term outcomes of pituitary adenomas in Multiple Endocrine Neoplasia type 1 : a nationwide study
Valdés, Nuria (Hospital Universitario de Cruces (Barakaldo, País Basc))
Romero, Ana (Hospital Universitario Virgen del Rocío (Sevilla, Andalusia))
Diego, Estrella (Hospital Universitario de Cruces (Barakaldo, País Basc))
Calatayud, María (Hospital Universitario 12 de Octubre (Madrid))
Lamas, Cristina (Complejo Hospitalario Universitario de Albacete)
Araujo-Castro, Marta (Hospital Universitario Ramón y Cajal (Madrid))
Álvarez-Escolá, Cristina (Hospital Universitario La Paz (Madrid))
Díaz, José A. (Hospital Universitario Clínico San Carlos (Madrid))
Alcázar, Victoria (Hospital Universitario Severo Ochoa)
Sastre, Julia (Complejo Hospitalario de Toledo)
Martínez, Rosa (Hospital Universitario de Cruces (Barakaldo, País Basc))
Oriola, Josep (Hospital Clínic i Provincial de Barcelona)
Paja, Miguel (Hospital Universitario de Basurto (Bilbao, Biscaia))
Sánchez-Sobrino, Paula (Complejo Hospitalario Universitario de Pontevedra)
Salinas, Isabel (Institut Germans Trias i Pujol. Hospital Universitari Germans Trias i Pujol)
Recio-Córdova, José María (Hospital Universitario de Salamanca)
Navarro, Elena (Hospital Universitario Virgen del Rocío (Sevilla, Andalusia))
Chiara, María Dolores (Instituto de Investigación Sanitaria del Principado de Asturias)
Castaño, Luis (Hospital Universitario de Cruces (Barakaldo, País Basc))
Casterás, A. (Hospital Universitari Vall d'Hebron)

Data:	2024
Resum:	Introduction: Historically, Multiple Endocrine Neoplasia type 1 (MEN1)-related pituitary adenomas (PAs) were considered more aggressive and treatment-resistant than sporadic PAs. However, recent studies suggest similarities in their behavior. This study aimed to evaluate the long-term outcomes of MEN1 PAs and identify predictive factors. Methods: Nationwide multicenter retrospective cohort study of MEN1-related PAs with a minimum 1-year follow-up, collecting patient demographics, germline MEN1 pathogenic variants (PV), PA size, secretory profile, radiological characteristics, treatments, and outcomes. Results: We analyzed 84 PAs, 69%in females and 31% in males (P<0. 001), diagnosed at a mean age of 35. 2±14. 9 years, mostly through screening (60. 7%). Median follow-up was 9 years (IQR:4-16). Prolactin-secreting PAs (PRLomas) (53. 5%) and microadenomas (65. 5%) were most common. Dopamine agonist treatment was first line for 16 macroPRLomas and 25 microPRLomas, 60. 9% of them achieved PRL normalization. There was no significant association observed with tumor size, sex, treatment duration or MEN1 PV. The risk of progression from micro-PA to invasive macro-PA was 7. 2% (4/55), after 8 years (IQR:4-13), all of them were microPRLomas. Kaplan-Meier estimation curve showed significantly higher progression probability in microPRLomas than in other microadenomas subtypes (P=0. 017) or microNFPAs (P=0. 032). No differences were found between sex, age, or germline MEN1 PV. Conclusion: MEN1-related micro-PAs have a low risk of progressing to invasive macro-PAs, regardless of sex, age at diagnosis, or MEN1 germline PV. The risk is higher for microPRLomas over the long term. Therefore, long-term surveillance with reduced frequency, rather than intensive short-term monitoring, may be appropriate for patients with MEN1-related PAs.
Drets:	Aquest document està subjecte a una llicència d'ús Creative Commons. Es permet la reproducció total o parcial, la distribució, la comunicació pública de l'obra i la creació d'obres derivades, fins i tot amb finalitats comercials, sempre i quan es reconegui l'autoria de l'obra original.
Llengua:	Anglès
Document:	Article ; recerca ; Versió publicada
Matèria:	Pituitary adenomas ; Multiple Endocrine Neoplasia type 1 ; Non-functioning pituitary adenomas ; Prolactinomas ; Outcomes ; Microadenomas ; Macroadenomas ; Progression
Publicat a:	Frontiers in endocrinology, Vol. 15 (October 2024) , ISSN 1664-2392

DOI: 10.3389/fendo.2024.1427821
PMID: 39439563

9 p, 2.6 MB

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Documents de recerca > Documents dels grups de recerca de la UAB > Centres i grups de recerca (producció científica) > Ciències de la salut i biociències > Institut d'Investigació en Ciencies de la Salut Germans Trias i Pujol (IGTP)
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