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Neuropathologic Findings in Mowat-Wilson Syndrome at Autopsy, Including a Suprasellar Spindle Cell Lipoma

  • Couger Jimenez Jaramillo
  • , Andrew Berman
  • , Jesse Fitzgerald
  • , Robert Brady
  • , Thomas Adams
  • , Nathan Clement

Research output: Contribution to journalArticlepeer-review

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 languageEnglish
Pages (from-to)316-320
Number of pages5
JournalPediatric and Developmental Pathology
Volume28
Issue number4
DOIs
StatePublished - 2025
Externally publishedYes

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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