Anophthalmia refers to the most severe ocular congenital malformation that results from inadequate development of the primitive forebrain and is always associated with blindness. The exact etiology of anophthalmia is not well understood; however, heritable and environmental factors may be involved. A 2-month-old Asian Shepherd puppy was presented with the absence of one eye. The puppy was alert, responsive, and had a good body score. The eyelids with eyelashes and palpebral conjunctiva were bilaterally present. However, in the right eye, the palpebral fissure was narrow, and the orbit was shallow without a distinct globe. Further inspection revealed no ocular structures, although the left eye was ophthalmoscopically quite normal. Also, there were no cytologic characteristics of conjunctivitis in either eye. Using the transpalpebral ultrasonography technique, a semi-oval anechoic area without any obvious ocular chambers or structures was observed in the right eye, and finally, unilateral clinical anophthalmia was diagnosed. General physical examination revealed no other malformation. The long-term prognosis is favorable for this pup. However, surgical strategies (e.g., implants and expanders) can be used to improve cosmetic appearance.