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• W2004372259 abstract "Studies of three related patients with familial non-hemolytic jaundice with conjugated bilirubin in the serum are described. An unidentified pigment was present in the liver cells in two patients (Cases I and II) and a diagnosis of Dubin-Johnson syndrome was made. A liver specimen was obtained from the third patient (Case III) on four separate occasions and detailed histologic examination failed to reveal any abnormal intrahepatic pigmentation. The survival of erythrocytes labelled with Cr51 was normal in the two patients in whom it was determined. Urinary and fecal urobilinogen excretion, menthol tolerance test and in vitro assay of hepatic glucuronyl transferase activity were within normal limits in all three cases. Determination of the distribution of bile pigments in the plasma, the relative amounts of dipyrrol azo pigments A and B after the addition of diazotized sulfanilic acid to serum, the percentage of dipyrrol azo pigment B which remained after alkaline and enzymatic hydrolysis, and the relative amounts of free and conjugated bromsulphalein in the serum forty-five and 100 minutes after the intravenous injection of 5 mg. bromsulphalein per kg. body weight were not significantly different in the three cases. The hepatic Tm for bromsulphalein was markedly reduced in all three cases. The relative hepatic storage of bromsulphalein was normal. Histochemical studies revealed acid phosphatase-positive pericanalicular pigment granules in liver tissue obtained in Cases I and II. Four specimens of liver obtained in Case III did not reveal abnormal hepatic pigmentation. Histochemical study revealed increased pericanalicular acid phosphatase activity. On electron microscopic examination, abundant amounts of the abnormal pigment were seen in pericanalicular lysosomes in Case I. The possibility is discussed that cases of chronic familial non-hemolytic jaundice with conjugated bilirubin in the serum with and without abnormal hepatic pigmentation may represent varied expressivity of a common functional defect in the poorly understood mechanisms by which bilirubin and other metabolites are transported from the liver cells into the bile." @default.
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• W2004372259 title "Studies of chronic familial non-hemolytic jaundice with conjugated bilirubin in the serum with and without an unidentified pigment in the liver cells" @default.
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• W2004372259 doi "https://doi.org/10.1016/0002-9343(61)90135-8" @default.
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