Lipomyelocele is an uncommon type of lipoma that occurs with spina bifida. We present the clinical course and therapeutic process of a female who underwent resection of a lipomyelocele and developed a massive lumbosacral subcutaneous exudate postoperatively. The therapeutic process is described in detail, and a review of the relevant literature on lipomyelocele is presented.A 23-year-old woman presented to our institution complaining of a large lumbosacral subcutaneous mass. She underwent surgical resection of the mass and untethering of the spinal cord under intraoperative neurophysiologic monitoring. A massive lumbosacral subcutaneous exudate developed postoperatively. After excluding cerebrospinal fluid (CSF) leakage, we placed a suction drain. Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the editor of this journal. Because of this, there is no need to conduct special ethic review and the ethical approval is not necessary.Postoperative pathologic examination confirmed the diagnosis of lipomyelocele. Continuation of the negative-pressure drain for 1 week yielded >1000 mL of fluid. The patient recovered well and developed no further subcutaneous exudate.In a patient with massive lumbosacral subcutaneous exudate after surgical treatment of a large lipomyelocele, continuous negative-pressure drainage can be an effective treatment method after excluding CSF leakage.