Goldenhar's syndrome.
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Biomedical subjects
Publications and source records attributed to S V Reddy.
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We report a 45-year-old man and a 60-year-old woman who presented with features of intermittent intestinal obstruction. Barium enema revealed narrowing at the pelvic-rectal junction in the man, and from the pelvic colon to the anal verge in the woman. Histology of the resected sections showed marked hypertrophy of the muscularis propria in both cases, with normal mucosa, submucosa and myenteric plexus. Both patients are asymptomatic at 4 years' and 2 years' follow up. This entity of primary hypertrophic colonopathy may be a variant of primary visceral myopathy.