TY - JOUR
T1 - Updated diagnostic criteria and nomenclature for neurofibromatosis type 2 and schwannomatosis
T2 - An international consensus recommendation
AU - International Consensus Group on Neurofibromatosis Diagnostic Criteria (I-NF-DC)
AU - Plotkin, Scott R.
AU - Messiaen, Ludwine
AU - Legius, Eric
AU - Pancza, Patrice
AU - Avery, Robert A.
AU - Blakeley, Jaishri O.
AU - Babovic-Vuksanovic, Dusica
AU - Ferner, Rosalie
AU - Fisher, Michael J.
AU - Friedman, Jan M.
AU - Giovannini, Marco
AU - Gutmann, David H.
AU - Hanemann, Clemens Oliver
AU - Kalamarides, Michel
AU - Kehrer-Sawatzki, Hildegard
AU - Korf, Bruce R.
AU - Mautner, Victor Felix
AU - MacCollin, Mia
AU - Papi, Laura
AU - Rauen, Katherine A.
AU - Riccardi, Vincent
AU - Schorry, Elizabeth
AU - Smith, Miriam J.
AU - Stemmer-Rachamimov, Anat
AU - Stevenson, David A.
AU - Ullrich, Nicole J.
AU - Viskochil, David
AU - Wimmer, Katharina
AU - Yohay, Kaleb
AU - Anten, Monique
AU - Aylsworth, Arthur
AU - Baralle, Diana
AU - Barbarot, Sebastien
AU - Barker, Fred
AU - Ben-Shachar, Shay
AU - Bergner, Amanda
AU - Bessis, Didier
AU - Blanco, Ignacio
AU - Cassiman, Catherine
AU - Ciavarelli, Patricia
AU - Clementi, Maurizio
AU - Frébourg, Thierry
AU - Gomes, Alicia
AU - Halliday, Dorothy
AU - Helen Hanson Arvid Heiberg, Chris Hammond
AU - Joly, Pascal
AU - Jordan, Justin T.
AU - Karajannis, Matthias
AU - Kroshinsky, Daniela
AU - Larralde, Margarita
N1 - Publisher Copyright:
© 2022 The Authors
PY - 2022/9
Y1 - 2022/9
N2 - Purpose: Neurofibromatosis type 2 (NF2) and schwannomatosis (SWN) are genetically distinct tumor predisposition syndromes with overlapping phenotypes. We sought to update the diagnostic criteria for NF2 and SWN by incorporating recent advances in genetics, ophthalmology, neuropathology, and neuroimaging. Methods: We used a multistep process, beginning with a Delphi method involving global disease experts and subsequently involving non-neurofibromatosis clinical experts, patients, and foundations/patient advocacy groups. Results: We reached consensus on the minimal clinical and genetic criteria for diagnosing NF2 and SWN. These criteria incorporate mosaic forms of these conditions. In addition, we recommend updated nomenclature for these disorders to emphasize their phenotypic overlap and to minimize misdiagnosis with neurofibromatosis type 1. Conclusion: The updated criteria for NF2 and SWN incorporate clinical features and genetic testing, with a focus on using molecular data to differentiate the 2 conditions. It is likely that continued refinement of these new criteria will be necessary as investigators study the diagnostic properties of the revised criteria and identify new genes associated with SWN. In the revised nomenclature, the term “neurofibromatosis 2” has been retired to improve diagnostic specificity.
AB - Purpose: Neurofibromatosis type 2 (NF2) and schwannomatosis (SWN) are genetically distinct tumor predisposition syndromes with overlapping phenotypes. We sought to update the diagnostic criteria for NF2 and SWN by incorporating recent advances in genetics, ophthalmology, neuropathology, and neuroimaging. Methods: We used a multistep process, beginning with a Delphi method involving global disease experts and subsequently involving non-neurofibromatosis clinical experts, patients, and foundations/patient advocacy groups. Results: We reached consensus on the minimal clinical and genetic criteria for diagnosing NF2 and SWN. These criteria incorporate mosaic forms of these conditions. In addition, we recommend updated nomenclature for these disorders to emphasize their phenotypic overlap and to minimize misdiagnosis with neurofibromatosis type 1. Conclusion: The updated criteria for NF2 and SWN incorporate clinical features and genetic testing, with a focus on using molecular data to differentiate the 2 conditions. It is likely that continued refinement of these new criteria will be necessary as investigators study the diagnostic properties of the revised criteria and identify new genes associated with SWN. In the revised nomenclature, the term “neurofibromatosis 2” has been retired to improve diagnostic specificity.
KW - NF2
KW - Neurofibromatosis
KW - SMARCB1
KW - Schwannomatosis
KW - lztr1
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U2 - 10.1016/j.gim.2022.05.007
DO - 10.1016/j.gim.2022.05.007
M3 - Article
C2 - 35674741
AN - SCOPUS:85132738125
SN - 1098-3600
VL - 24
SP - 1967
EP - 1977
JO - Genetics in Medicine
JF - Genetics in Medicine
IS - 9
ER -