Several hallmark features of ALMS such as cardiomyopathy, widespread fibrosis, and severe obesity with metabolic syndrome are known to be related to deregulation of TGF-β/BMP signaling (Zamani and Brown, 2011; Grgurevic et al., 2016; Heger et al., 2016; Walton et al., 2017); however, the possible connection between this pathway and the ALMS1 protein has not been extensively studied. Here, ALMS1 is linked to obesity due to melanocortin 4 receptor deficiency.