Dupuytren's disease was first described by Baron Guillaume Dupuytren in 1831. He outlined the pathology of palmar fascia thickening and contracture resulting in flexion of one or more digits. Dupuytren's disease usually affects the little or ring fingers, with the thumb rarely affected. Furthermore, the thumb in isolation is only affected in 0.5% of cases, with contracture at the metacarpophalangeal joint. We present a unique and interesting case of Dupuytren's disease predominantly of the thumbs, with isolated contractures bilaterally at the interphalangeal joints, in a 59 year old lady with epilepsy who was otherwise fit and well. To our knowledge this disease pattern has never been reported in the literature. Furthermore a relevant literature review of Dupuytren's disease affecting the thumb, in particular the interphalangeal joint, is presented. Our interesting and unique case reinforces the notion that the disease pathogenesis is diverse and multifactorial, and provides a further interesting example for the literature.