The pterygoideus proprius muscle can be found incidentally in the infratemporal fossa, where it is spatially associated with the muscles of mastication, the maxillary artery, and the trigeminal nerve and its branches. Anatomists have described the muscle in various ways over the past 160 years, chiefly as a musculotendinous structure that originates from the infratemporal crest of the sphenoid bone and inserts into the lateral pterygoid plate and the lateral pterygoid muscle. It is present in non‐human primates, albeit rarely, with similar anatomical findings. Embryologically, the pterygoideus proprius is thought to have developed from the first pharyngeal arch mesenchyme along with other muscles of mastication. Its close association with the maxillary artery and trigeminal nerve suggests possible clinical significance in trigeminal neuralgia and temporomandibular joint disorders. The literature was reviewed systematically to detail the historical background of research on the pterygoideus proprius muscle and explain its morphology, prevalence, embryology, and potential clinical significance. Despite its rarity, we propose that it is important to recognize its presence when the infratemporal fossa is approached.