PMID- 37799353 OWN - NLM STAT- PubMed-not-MEDLINE LR - 20231026 IS - 2690-442X (Electronic) IS - 2690-442X (Linking) VI - 3 IP - 5 DP - 2023 Oct TI - Tolerance of sonidegib after intolerance of vismodegib-Experience in two patients with nevoid basal cell carcinoma syndrome (Gorlin syndrome). PG - e260 LID - 10.1002/ski2.260 [doi] LID - e260 AB - Nevoid basal cell carcinoma syndrome (NBCCS, Gorlin syndrome) is a rare genetic condition characterized by the early development of numerous cutaneous basal cell carcinomas (BCCs). Although most BCCs are surgically treated with total resection, some of the lesions may proceed to a locally advanced or metastatic stage. Systemic treatment with a hedgehog inhibitors (HHIs) such as Vismodegib or Sonidegib is indicated in this population. We report cases of two patients with confirmed diagnoses of NBCCS. Both patients had undergone multiple surgical excisions and had been treated with oral Vismodegib 150 mg/day for a locally advanced tumour. They both discontinued the therapy due to its specific adverse effects (AEs) and are now being treated with oral Sonidegib, which has had better tolerability and a complete response. The aims of this report was to demonstrate the efficacy of HHI treatment with Sonidegib in patients with NCBBS who had previously treated with Vismodegib but discontinued it because of its specific AEs. Our experience in two patients shows that Sonidegib can be considered in Gorlin patients intolerant but responding to Vismodegib. CI - (c) 2023 The Authors. Skin Health and Disease published by John Wiley & Sons Ltd on behalf of British Association of Dermatologists. FAU - Venturi, Federico AU - Venturi F AUID- ORCID: 0000-0001-5053-4172 AD - Section of Dermatology Department of Health Sciences University of Florence Florence Italy. AD - IRCCS Azienda Ospedaliero-Universitaria di Bologna Bologna Italy. AD - Dermatology Unit, Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum University of Bologna Bologna Italy. FAU - Trane, Luciana AU - Trane L AD - Cancer Research "Attilia Pofferi" Foundation Pistoia Italy. FAU - Silvestri, Flavia AU - Silvestri F AD - Section of Dermatology Department of Health Sciences University of Florence Florence Italy. FAU - Zuccaro, Biancamaria AU - Zuccaro B AD - Section of Dermatology Department of Health Sciences University of Florence Florence Italy. FAU - Magnaterra, Elisabetta AU - Magnaterra E AD - Section of Dermatology Department of Health Sciences University of Florence Florence Italy. FAU - De Giorgi, Vincenzo AU - De Giorgi V AUID- ORCID: 0000-0001-6531-7998 AD - Section of Dermatology Department of Health Sciences University of Florence Florence Italy. LA - eng PT - Case Reports DEP - 20230614 PL - England TA - Skin Health Dis JT - Skin health and disease JID - 9918227353706676 PMC - PMC10549827 COIS- The authors declared that they have no conflicts of interest to this work. EDAT- 2023/10/06 06:43 MHDA- 2023/10/06 06:44 PMCR- 2023/06/14 CRDT- 2023/10/06 03:48 PHST- 2023/04/30 00:00 [received] PHST- 2023/05/29 00:00 [revised] PHST- 2023/06/01 00:00 [accepted] PHST- 2023/10/06 06:44 [medline] PHST- 2023/10/06 06:43 [pubmed] PHST- 2023/10/06 03:48 [entrez] PHST- 2023/06/14 00:00 [pmc-release] AID - SKI2260 [pii] AID - 10.1002/ski2.260 [doi] PST - epublish SO - Skin Health Dis. 2023 Jun 14;3(5):e260. doi: 10.1002/ski2.260. eCollection 2023 Oct.