Background: Infantile myofibromatosis is a rare mesenchymal disorder of infancy that can extensively involve the viscera including the gastrointestinal tract.
Case presentation: In this report, an exceptional case of infantile myofibromatosis is described in which rectal prolapse and sigmoid colo-colonic intussusception were the initial presentations of colorectal involvement in a 2-month-old premature female infant.
Conclusion: To the best of our knowledge, this is the first case reporting rectal prolapse and the second case documenting intussusception secondary to gastrointestinal involvement by infantile myofibromatosis.