Double lumbosacral lipomas of the dorsal and filar types associated with OEIS complex: case report
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So Tokunaga1, Takato Morioka, Kimiaki Hashiguchi
1Department of Neurosurgery, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan. tokunaga_so@yahoo.co.jp
Insights
This case study details a rare OEIS complex diagnosis in a newborn, highlighting the successful surgical correction of associated spinal lipomas after initial abdominogenital repairs.
Area of Science:
- Pediatric Surgery
- Neonatal Care
- Neuroscience
Background:
- The OEIS complex (Omphalocele, Exstrophy of the Cloaca, Imperforate Anus, Spinal Defects) is a rare congenital anomaly.
- Spinal lipomas are an uncommon association with OEIS complex, with terminal myelocystocele being more frequent.
Observation:
- A female infant presented with OEIS complex at birth.
- Magnetic resonance imaging revealed dual spinal lipomas: a dorsal-type and a filar-type.
- The filar-type lipoma was characterized by a thickened, fatty filum terminale.
Findings:
- The infant underwent abdominogenital repairs followed by neurosurgical intervention at 14 months.
- Surgical procedures included debulking of the dorsal lipoma and spinal cord untethering by sectioning the filar lipoma.
- Successful surgical outcomes were achieved for both the abdominal and spinal defects.
Implications:
- This case underscores the importance of comprehensive evaluation and management of OEIS complex, including associated spinal anomalies.
- Neurosurgical intervention for occult spinal dysraphism in OEIS patients is recommended after initial abdominogenital reconstructive surgeries.
- Timely surgical correction of spinal lipomas can prevent neurological complications in infants with OEIS complex.
Abstract:
A female baby was born at 37 weeks and 6 days gestation by vaginal delivery with omphalocele, exstrophy of the cloaca, and imperforate anus, indicating the presence of OEIS complex, a rare combination of defects consisting of omphalocele (O), exstrophy of the cloaca (E), imperforate anus (I), and spinal deformity (S), associated with lumbosacral lipoma. The most common associated spinal deformity is terminal myelocystocele, and spinal lipoma is rare. Constructive interference in steady-state magnetic resonance imaging clearly revealed double lipomas, a dorsal-type lipoma, located dorsal to the low-lying conus medullaris, and a filar-type lipoma, revealed by a thickened and fatty filum terminale. After recovery from abdominogenital repairs, debulking of the dorsal-type lipoma and untethering of the spinal cord by sectioning of the filar-type lipoma were performed at the age of 14 months. Neurosurgical treatment for occult spinal dysraphism should be undertaken after recovery from the initial series of major abdominogenital procedures.