H. Iijima et al., Spontaneous regression of hepatic adenoma in a patient with glycogen storage disease type I after hemodialysis: Ultrasonographic and CT findings, INTERN MED, 40(9), 2001, pp. 891-895
A 23-year-old woman was admitted to our hospital with recurrent gouty arthr
itis. Laboratory findings showed hypoglycemia, lactic acidosis, hyperlipide
mia, and hyperuricemia, with normal values of serum alfa-fetoprotein (AFP)
and protein induced by vitamin K absence (PIVKA-II). A diagnosis of glycoge
n storage disease type I (GSD-type I) was made on the basis of the laborato
ry data, liver biopsy findings, and partially deficient thrombocyte glucose
-6-phosphatase (G-6-Pase) activity. Ultrasonography and computed tomography
revealed multiple focal hepatic masses. Biopsied specimens of the lesion d
emonstrated a hepatic adenoma, which changed in appearance in the relativel
y short period between echography and computed tomography. This interesting
phenomenon may highlight the importance for careful follow-up of hepatic a
denomas, because of the potential of rapture, hemorrhage, or malignant tran
sformation. During follow-up, the present patient received hemodialysis due
to renal failure, and the adenoma regressed spontaneously after 8 years. I
ncluded are diagnostic images, demonstrating the association of hepatic ade
noma. and GSD-type I.