A standard of care for individuals with PIK3CA-related disorders: An international expert consensus statement

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TitreA standard of care for individuals with PIK3CA-related disorders: An international expert consensus statement
Type de publicationJournal Article
Year of Publication2022
AuteursDouzgou S, Rawson M, Baselga E, Danielpour M, Faivre L, Kashanian A, Keppler-Noreuil KM, Kuentz P, Mancini GMS, Maniere M-C, Martinez-Glez V, Parker VE, Semple RK, Srivastava S, Vabres P, De Wit M-CY, , Clayton-Smith J, Mirzaa GM, Biesecker LG
JournalCLINICAL GENETICS
Volume101
Pagination32-47
Date PublishedJAN
Type of ArticleReview
ISSN0009-9163
Mots-clésclinical management, expert consensus, Mosaic, PIK3CA-related overgrowth spectrum
Résumé

Growth promoting variants in PIK3CA cause a spectrum of developmental disorders, depending on the developmental timing of the mutation and tissues involved. These phenotypically heterogeneous entities have been grouped as PIK3CA-Related Overgrowth Spectrum disorders (PROS). Deep sequencing technologies have facilitated detection of low-level mosaic, often necessitating testing of tissues other than blood. Since clinical management practices vary considerably among healthcare professionals and services across different countries, a consensus on management guidelines is needed. Clinical heterogeneity within this spectrum leads to challenges in establishing management recommendations, which must be based on patient-specific considerations. Moreover, as most of these conditions are rare, affected families may lack access to the medical expertise that is needed to help address the multi-system and often complex medical issues seen with PROS. In March 2019, macrocephaly-capillary malformation (M-CM) patient organizations hosted an expert meeting in Manchester, United Kingdom, to help address these challenges with regards to M-CM syndrome. We have expanded the scope of this project to cover PROS and developed this consensus statement on the preferred approach for managing affected individuals based on our current knowledge.

DOI10.1111/cge.14027