These findings add a new mutant class to the list of ciliary mouse models of hyperphagia and obesity, which includes mutants of the BBS complex, of Alms1, of the IFT-B complex, and of the transition zone (Arsov et al., 2006; Collin et al., 2005; Davenport et al., 2007; Rahmouni et al., 2008; Stratigopoulos et al., 2014). Here, ALMS1 is linked to obesity due to melanocortin 4 receptor deficiency.