Calvarial hyperostosis syndrome is an uncommon and self-limiting disease affecting juvenile dogs. Only symptomatic treatment has been described, and diagnosis is based on clinical findings, imaging, and disease progression. This is the first reported case of calvarial hyperostosis syndrome in a Dalmatian dog. It is an uncommon osteoproliferative disease with diagnosis frequently based on clinical signs, imaging findings, and disease progression, with only symptomatic treatment described. Case describes a 5-month-old Dalmatian dog presented with a facial mass and difficulty eating. After imaging, mass was observed to be osteoproliferative, nonaggressive, and without affection of the temporomandibular joints and mandibles. Histology revealed an osseous-cartilaginous and proliferative lesion, together with scant amount of neutrophiles. Clinical improvement was observed after symptomatic treatment, and moderate lesion regression was observed in a CT reevaluation 6 months later.