Abstract Purpose To demonstrate the capabilities of 4D ultrasound calf muscle imaging in patients with genetic types of distal myopathies (DMs). Methods Three patients with DM were studied: a 58-year-old man with Vocal Cord and Pharyngeal Weakness Distal Myopathy (VCPDM), a 36-year-old woman with Tibial Muscular Dystrophy (TMD) and a 27-year-old woman with Hereditary Inclusion Body Myopathy Type 2 (HIBM2). Their calf muscles were evaluated in rest and during maximal plantar flexion using 3D/4D ultrasound imaging. The results were compared to myosonograms of healthy controls. Results All patients had myopathic syndrome due to advanced muscular dystrophy. In comparison to controls abnormal calf muscle architectonics, reduced muscle contractility and a combination of spot-like hypo- and hyperechoic areas were established on 4D ultrasound imaging. The changes were associated with the degree of muscle atrophy, fat and fibrous tissue infiltration. Conclusions Four-dimensional myosonology gives additional information for muscle architectonics in patients with genetic types of DM. Further studies are needed to evaluate if the described findings are typical for specific genetic types of myopathy.