Abstract Introduction Acalculous cholecystitis has been shown to occur in the context of malarial infection with Plasmodium vivax and Plasmodium falciparum and requires prompt diagnosis and treatment to prevent complications. To the best of our knowledge this is the first reported case of the disease occurring in a patient infected with Plasmodium malariae. Case presentation We report the first case of acalculous cholecystitis associated with Plasmodium malariae in a 59-year-old male Nepalese ex-Gurkha soldier. He presented with fever and vomiting and later developed right upper quadrant pain. Abdominal ultrasound and computed tomography scans confirmed acalculous cholecystitis for which he was treated medically with chloroquine, gentamicin and metronidazole. He made a full recovery. Conclusions Physicians should be aware that in addition to Plasmodium vivax and Plasmodium falciparum infections, acalculous cholecystitis can occur in the context of Plasmodium malariae infection. Mechanisms for this are discussed but further studies are needed to establish the etiology of this association.