Scintigraphic progress of the liver in a patient with Alagille syndrome (arteriohepatic dysplasia)

Springer Science and Business Media LLC - Tập 17 - Trang 693-697 - 2003
Megumi Jinguji1, Shinsaku Tsuchimochi1, Masayuki Nakajo1, Hiroyuki Hamada1, Takuro Kamiyama1, Tomokazu Umanodan1, Atsushi Tani1, Yoshiaki Nakabeppu1, Tatsuru Kaji2, Hideo Takamatsu2, Hironori Haga3
1Department of Radiology, Faculty of Medicine, Kagoshima University, Kagoshima, Japan
2Department ofPediatric Surgery, Faculty of Medicine, Kagoshima University, Japan
3Department ofOrgan Transplant Unit and Laboratory of Anatomic Pathology, Kyoto University Hospital, Japan

Tóm tắt

We encountered a 9-year-old Japanese girl with Alagille syndrome. Her scintigraphic examinations of the liver were performed at the ages of 16 months and 9 years.99mTc-PMT, a hepatobiliary imaging agent, was distributed homogeneously in the liver at the younger age, but unevenly produced an area of focally increased uptake in the medial segment of the liver surrounded by peripheral atrophy at the older age.99mTc-GSA, a hepatoreceptor binding agent, was highly accumulated in the area, corresponding to the focally increased uptake of99mTc-PMT. These imaging findings suggest that the pathophysiological and morphological changes of the liver occurred in our patient during the clinical course.

Tài liệu tham khảo

Alagille D, Odievre M, Gautier M, Dommergues JP. Hepatic ductular hypoplasia associated with characteristic facies, vertebral malformations, retarded physical, mental, and sexual development, and cardiac murmur.J Pediatr 1975; 86:63–71. Alagille D, Estrada A, Hadchouel M, Gautier M, Odievre M, Dommergues JP. Syndromic paucity of intralobular bile ducts (Alagille syndrome or arteriohepatic dysplasia): Review of 80 cases.J Pediatr 1987; 110:195–200. Mueller RF. The Alagille syndrome (arteriohepatic dysplasia).J Med Genet 1987; 24:621–626. Balich S, Jarboe GL, Stevens SJ. Alagille’s syndrome, new manifestation.Clin Nucl Med 1996; 21:98–101. Torizuka T, Tamaki N, Fujita T, Yonekura Y, Uemoto S, Tanaka K, et al. Focal liver hyperplasia in Alagille syndrome: Assessment with hepatoreceptor and hepatobiliary imaging.J Nucl Med 1996; 37:1365–1367. Tajima T, Honda H, Yanaga K, Kuroiwa T, Yoshimitsu K, Irie H, et al. Hepatic nodular hyperplasia in a boy with Alagille syndrome: CT and MR appearances.Pediatr Radiol 2001; 31:584–588. Aburano T, Yokoyama K, Takayama T, Tonami N, Hisada K. Distinct hepatic retention of Tc-99m IDA in arteriohepatic dysplasia (Alagille syndrome).Clin Nucl Med 1989; 14:874–876. DuCret RP, Cefalu JB, Alford BA, Drake G, Boudreau RJ. Hepatocarcinoma in association with the Alagille syndrome.Clin Nucl Med 1988; 13:920–921. Halvorsen RA Jr, Garrity S, Kuni C, duCret RP, Letourneau JG, Bloomer J. Arteriohepatic dysplasia (Alagille’s syndrome): unusual hepatic architecture and function.Abdom Imaging 1995; 20:191–196. International working party. Terminology of nodular hepa-tocellular lesions.Hepatology 1995; 22:983–993. Saito K, Koizumi K, Abe K, Goto Y, Seki T. Potential for qualitative diagnosis of tumors and tumorous lesions in the liver with Tc-99m-GSA SPECT—Correlation with pathological evaluation and MRI findings—.Ann Nucl Med 1998; 12:275–280. Hasegawa Y, Nakano S, Ibuka K, Hashizume T, Sasaki Y, Imaoka S, et al. The importance of delayed imaging in the study of hepatoma with a new hepatobiliary agent.J Nucl Med 1984; 25:1122–1126.