Morning glory syndrome is a congenital anomaly of the optic disc in which the disc is enlarged and excavated, with white glial tissue in the center. A case is presented of morning glory syndrome associated with sphenoid encephalocele, median cleft lip, and agenesis of the corpus callosum. A 22-day-old boy was referred to the Wakayama Medical College Hospital for management of dyspnea due to a soft-tissue mass in the oral cavity. Magnetic resonance imaging revealed a mass extending through a bone defect in the sphenoid region and into the oral cavity. Surgical repair was attempted through a bifrontal craniotomy. A bone defect was identified in the sphenoid plate, through which the arachnoid membrane was connected to the oral cavity. Both optic nerves were elongated and adhered to the encephalocele. The wall of the meningocele was compressed digitally through the oral cavity and sutured to the dura mater of the bone defect. The operative findings suggest that a basal encephalocele protruding from a bone defect in the sphenoid plate may disturb the normal development of the optic nerve.
Paneth cells secrete bactericidal substances in response to bacterial proliferation on the mucosal surface without directly contacting bacteria. However, the induction mechanism of this transient secretion has not been clarified, although nervous system and/or immunocompetent cells in the lamina propria (LP) might be involved. In this study, we ultrastructurally and immunohistochemically investigated which LP cells are localized beneath Paneth cells and examined the relationship between the Paneth cell-derived cellular processes which extended into the LP and the LP cells. The results showed that various cells-including blood capillary, subepithelial stromal cell, and nerve fiber-were present in the LP beneath Paneth cells. Endothelial cells of blood capillary were the cells most frequently found in this location; they were situated within 1 μm of the Paneth cells and possessed fenestration on the surfaces adjacent to Paneth cells. The Paneth cells rarely extended the cellular processes toward the LP across the basal lamina. Most of the cellular processes of Paneth cells contacted the subepithelial stromal cells. Immunohistochemistry revealed that the CD34 CD31 αSMA stromal cells preferentially localized in the LP beneath the intestinal crypt base, while PDGFRα αSMA stromal cells mainly localized around the lateral portions of the intestinal crypt and PDGFRα αSMA stromal cells localized in the intestinal villus. From these findings, the existence of blood capillaries beneath Paneth cells might reflect the active exocrine function of Paneth cells. Furthermore, subepithelial stromal cells, probably with a CD34 CD31 αSMA PDGFRα phenotype, beneath the crypt base might affect Paneth cell activity by interacting with their cellular processes. Anat Rec, 301:1074-1085, 2018. © 2018 Wiley Periodicals, Inc.
Twenty three cases of primary intracranial germinomas including five cases of germinomas with syncytiotrophoblastic giant cells are studied and analyzed, with special reference to the recurrence under radiotherapy. The follow-up period for all cases was 7 months to 12 years (average: 5.8 years) with that for pure germinomas ranging from 8 months to 12 years (average: 5.7 years) and that for germinomas with syncytiotrophoblastic giant cells ranging from 7 months to 11 years (average: 6.3 years). Late recurrence was observed in three cases (3/23, 13%), developing outside of the initial irradiation field. With regard to recurrence, significant correlation to radiation fields was evident, while it was not to radiation doses. Furthermore, germinoma with syncytiotrophoblastic giant cells showed a more significant tendency to recur than pure germinoma. The radiotherapy of germinomas is discussed and the clinical features of germinoma with syncytiotrophoblastic giant cells are presented.
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