Background: Congenital amusia (CA) is a rare disorder characterized by deficits in pitch perception, and many structural and functional magnetic resonance imaging studies have been conducted to better understand its neural bases. However, a structural magnetic resonance imaging analysis using a surface-based morphology method to identify regions with cortical features abnormalities at the vertex-based level has not yet been performed. Methods: Fifteen participants with CA and 13 healthy controls underwent structural magnetic resonance imaging. A surface-based morphology method was used to identify anatomical abnormalities. Then, the surface parameters' mean value of the identified clusters with statistically significant between-group differences were extracted and compared. Finally, Pearson's correlation analysis was used to assess the correlation between the Montreal Battery of Evaluation of Amusia (MBEA) scores and surface parameters. Results: The CA group had significantly lower MBEA scores than the healthy controls ( p = 0.000). The CA group exhibited a significant higher fractal dimension in the right caudal middle frontal gyrus and a lower sulcal depth in the right pars triangularis gyrus ( p < 0.05; false discovery rate-corrected at the cluster level) compared to healthy controls. There were negative correlations between the mean fractal dimension values in the right caudal middle frontal gyrus and MBEA score, including the mean MBEA score ( r = -0.5398, p = 0.0030), scale score ( r = -0.5712, p = 0.0015), contour score ( r = -0.4662, p = 0.0124), interval score ( r = -0.4564, p = 0.0146), rhythmic score ( r = -0.5133, p = 0.0052), meter score ( r = -0.3937, p = 0.0382), and memory score ( r = -0.3879, p = 0.0414). There was a significant positive correlation between the mean sulcal depth in the right pars triangularis gyrus and the MBEA score, including the mean score ( r = 0.5130, p = 0.0052), scale score ( r = 0.5328, p = 0.0035), interval score ( r = 0.4059, p = 0.0321), rhythmic score ( r = 0.5733, p = 0.0014), meter score ( r = 0.5061, p = 0.0060), and memory score ( r = 0.4001, p = 0.0349). Conclusion: Individuals with CA exhibit cortical morphological changes in the right hemisphere. These findings may indicate that the neural basis of speech perception and memory impairments in individuals with CA is associated with abnormalities in the right pars triangularis gyrus and middle frontal gyrus, and that these cortical abnormalities may be a neural marker of CA., Competing Interests: The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest., (Copyright © 2022 Liao, Sun, Jin, Wu and Liu.)