Gorham's disease is a rare disorder involving the proliferation of endothelial channels resulting in resorption and disappearance of bone. An unusual case of polyostotic Gorham's disease affecting the scapula, humerus, radius, and ulna in a 39-year-old woman is described. The patient had extensive disease spreading across both the glenohumeral and humeroulnar joints. This is the first report of Gorham's disease spreading across multiple joints in the upper extremity.
Intramucosal ganglion cells are incompletely understood. Reviewed were 100 normal specimens from colorectal biopsies of nonconstipated adults to firmly establish the existence of intramucosal ganglion cells in normal adult colorectal mucosa, determine whether intramucosal giant ganglia exist, and compare the prevalence of colorectal intramucosal ganglion cells between men and women. Fifty specimens from each gender were examined. Twenty specimens contained intramucosal ganglion cells, including 15 from women (75%) and only 5 from men (25%). Intramucosal ganglion cells occurred singly or in clusters. One woman had large disciform and globular clusters with 7 ganglion cells, resembling giant ganglia. Awareness of ganglion cells and large ganglia in normal mucosa is necessary to avoid potential confusion with ectopic ganglion cells and submucosal giant ganglia seen in adult intestinal neuronal dysplasia. Intramucosal ganglion cells in normal colorectal mucosa are particularly common in women. This disparity might contribute to colorectal neurophysiologic differences between genders.
It generally is believed that perikarya of ganglion cells in the human colorectum are confined to plexuses that lie deep to the mucosa, and that intramucosal perikarya are rare. We retrospectively reviewed 100 specimens from biopsies of normal and abnormal mucosa to further characterize intramucosal perikarya. The presence of intramucosal perikarya, their number, location, and grouping were recorded. Twenty-one specimens (21.0%) contained intramucosal perikarya. Intramucosal perikarya occurred throughout the colorectum in the muscularis mucosae or lamina propria and in normal mucosa, acute self-limited colitis, inflammatory bowel disease, cytomegalovirus-associated colitis, hyperplastic polyps, tubular adenomas, and high-grade intraepithelial neoplasia. In some specimens, intramucosal perikarya morphologically resembled microgranulomas or cytomegalovirus-infected cells. We demonstrated that intramucosal perikarya of ganglion cells are surprisingly common in normal and abnormal mucosa. Awareness of intramucosal perikarya is necessary to avoid confusion with microgranulomas or cytomegalovirus-infected cells.
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