Bilateral internuclear ophthalmoplegia (INO) has been linked with various pathological conditions of the central nervous system such as multiple sclerosis, stroke, tumours and brainstem inflammatory processes. This unusual case report describes a case of a 45-year-old female patient presenting with diplopia due to bilateral INO, but with no evidence of brainstem lesion on brain magnetic resonance imaging (MRI). She was diagnosed with Sjogrens’ syndrome, which is a systemic autoimmune disease that usually affects salivary and lacrimal glands. The authors report this case as the first known reported case of bilateral INO as a presenting symptom of primary Sjogrens’ syndrome. A variety of neuro-ophthalmological manifestations have been reported previously in the setting of primary Sjogrens’ syndrome, including one case report of unilateral INO.