AbstractLipomyelomeningocele is a form of spina bifida occulta with a distinct pathogenesis that differentiates it from open neural tube defects. Familial forms are rare and the condition may be polygenic. We report on 2 affected siblings with similar lesions and raise the possibility of an autosomal recessive pattern of inheritance with implications for genetic counselling. © 2004 Wiley‐Liss, Inc.