We present a series of three infants who underwent keystone design perforator island flap (KDPIF) closure for myelomeningocele in a paediatric neurosurgical centre in Australia. This is the first recorded utilization of this flap for primary closure of myelomeningocele (MMC). METHODS: The prospective data from the Monash Neurosurgical Database were used to select all cases of MMC between December 2008 and September 2010. Retrospective analysis of these cases revealed three patients who underwent KDPIF at Monash Medical Centre for MMC repair at birth. RESULTS: Wound healing was prompt and satisfactory in all three cases. No minor or major complications were noted. In particular, there was no associated skin flap separation, skin flap dehiscence, skin flap necrosis, infection, cerebrospinal fluid leak, or need for return to theatre for further intervention to the wound. This keystone design perforator island flap is based on random perforating musculo/fasciocutaneous perforators. In our experience, this robust flap provides better tissue bulk, more reliable vascularity and a wider geometrical versatility than traditional random 1:1 cutaneous flaps. CONCLUSION: Whilst primary closure remains an option for myelomeningocele closure, primary repair of larger defects can lead to closure site tension, stretching of inelastic scar tissue and inadequate soft tissue cover. In this small series, we have demonstrated the use of keystone design perforator island flap closure as an alternative for larger and more complex lesions.