Abstract
Mowat-Wilson Syndrome is an autosomal dominant disorder caused by de novo heterozygous mutations of ZEB2 on 2q22. It is characterized by developmental delay, Hirschsprung's disease, seizures, and a wide variety of malformations affecting the neurologic, cardiac, and genitourinary systems. Reports describing the findings of Mowat-Wilson Syndrome at autopsy are sparse. Case reports of suprasellar spindle cell lipomas are even rarer, a circumstance that contributes to uncertainty regarding their etiology as true neoplasms rather than congenital malformations. Here we report the gross, histopathologic, and molecular findings of a 4-year-old female with Mowat-Wilson Syndrome presenting with sepsis in the setting of otitis media and incidentally found to have a rare suprasellar spindle cell lipoma demonstrating loss of RB1 by immunohistochemistry, suggestive of a neoplastic etiology.
| Original language | English |
|---|---|
| Pages (from-to) | 316-320 |
| Number of pages | 5 |
| Journal | Pediatric and Developmental Pathology |
| Volume | 28 |
| Issue number | 4 |
| DOIs | |
| State | Published - 2025 |
| Externally published | Yes |
Keywords
- Humans
- Female
- Child, Preschool
- Microcephaly/pathology
- Lipoma/pathology
- Hirschsprung Disease/pathology
- Autopsy
- Intellectual Disability/pathology
- Facies
- Retinoblastoma Binding Proteins/metabolism
- Ubiquitin-Protein Ligases/metabolism
- Brain Neoplasms/pathology
- Zinc Finger E-box Binding Homeobox 2/genetics
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