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Sertoli-Leydig cell tumours of the ovary (SLCT) are rare tumours predominantly caused by mutations in the  DICER1  gene. We present a patient with a unilateral SLCT who had an underlying germline  DICER1  gene mutation. We discuss the underlying pathology, risks, and screening opportunities available to those with a mutation in this gene as SLCT is only one of a multitude of other tumours encompassing  DICER1  syndrome. The condition is inherited in an autosomal dominant fashion. As such, genetic counselling is a key component of the management of women with SLCT.

Sertoli-Leydig Cell Tumour and DICER1 Mutation: A Case Report and Review of the Literature