Woodhouse–Sakati syndrome (WSS) is a rare autosomal recessive disease, known to have distinctive neuroendocrine manifestations.WSS covers diverse phenotypes (1). Neurological involvement is common, with dystonia being the most common neurological manifestation. Management of WSS necessitates a multidisciplinary team targeting symptomatic therapy. Like other types of dystonia, the first-line treatment includes oral medications and botulinum toxin injection followed by a surgical intervention such as Deep Brain Stimulation (DBS) of the Globus Pallidus Internus (GPI)(2). No clear guidelines are available for the treatment of dystonia in WSS. A thorough literature review showed one patient with WSS underwent bilateral GPI DBS, which improved his cervical and truncal dystonia (3)