Gorlin syndrome (GS) is an oncogenetic autosomal dominant syndrome causing multiple basal cell carcinomas (BCCs), odontogenic keratocysts and bone disorders.BCCs are challenging to diagnose, multiple and frequently occur on the face, notably periorificial areas, leading to difficult surgery.New non-invasive imaging systems combined with artificial intelligence (AI) may improve early detection and management of challenging BCC in GS patients.We performed a clinical retrospective study on GS patients evaluating the impact of their monitoring with an innovative workflow based on multimodal imaging using VECTRA 3D total body photography (TBP) and line-field confocal optical coherence tomography (LC-OCT) enhanced by AI for early BCC detection in GS patients.Methods: We included in this clinical retrospective study 3 GS patients.They were diagnosed and followed up for GS in our department since 2015.Since 2022 a unique multimodal screening approach was based on a standardized workflow.First VECTRA 3D was used for longitudinal follow-up and early detection of suspicious lesions for BCC, then AI-assisted videodermoscopy (DEXI) provided malignancy scores of these lesions, and when the score was high, followed by LC-OCT screening, which offered in vivo cellular resolution and AI-derived confirmation (ConvNeXt).BCC's diagnosis could be non-invasively confirmed and surgical safety margins established based on LC OCT before the removal surgery enabling a one-step surgical management.Results: We included 3 GS patients, 37, 40, 73 years old, 2 females and 1 male.These patients presented multiple pigmented and non-pigmented lesions less than 5 mm without any specific dermoscopy criteria.We included 29 difficult-to-diagnose lesions, analysed in our specific multimodal workflow approach since 2022.The median size was small (<2 mm) and lacking specific dermoscopic criteria.All lesions suspected to be BCC after our analysis were surgically excised: 26 BCCs (18 nodular/infiltrative BCCs, 8 superficial BCCs confirmed histologically) and 3 actinic keratosis.The BCCs were localized on the face, scalp, nose, medial canthus, alar fossa, and trunk.The multimodal approach achieved 100% sensitivity and 90% specificity, reducing unnecessary biopsies.One-step surgical management was facilitated in 24 BCCs 93%, and 2 BCC needed a 2 steps surgery because of margins.AI-assisted TBP by VECTRA 3D, videodermoscopy and LC-OCT, contributed to reducing unnecessary surgery.This workflow also supported diagnostic training for less experienced clinicians.Conclusions:
Schuh et al. (2026) studied this question.