31789919

Adrenocortical Scintigraphy With SPECT/CT for the Diagnosis of Hepatic Adrenal Rest

Masako Kawasaki, MD,* Mana Yoshimura, MD,* Katsutoshi Sugimoto, MD,1- Hirohito Takeuchi, MD,¡ and Kazuhiro Saito, MD*

Abstract: Hepatic adrenal rest include ectopic adrenal tissue in the liver and adrenohepatic fusion. Long-lasting hyperstimulation of that tissue induces hyperplasia sometimes simulating true tumors. True autonomic tumors orig- inating from ectopic adrenal tissue with or without hyperfunction are rare. Hepatic adrenal rest sometimes has similar imaging findings to lipid-rich hepatocellular carcinoma or angiomyolipoma, containing obvious lipid com- ponent. But in our cases, tumors showed hypervascular patterns similar to he- patocellular adenoma or carcinoma. Therefore, adrenocortical scintigraphy with SPECT/CT should be taken into consideration to diagnose tumoral lesions in the posterior segment of the right hepatic lobe with or without an obvious lipid component.

Key Words: adrenocortical scintigraphy, hepatic adrenal rest, hepatocellular carcinoma, SPECT/CT

(Clin Nucl Med 2019;00: 00-00)

Received for publication July 26, 2019; revision accepted September 8, 2019.

From the Departments of *Radiology and ¡Gastroenterology, Tokyo Medical University, Tokyo, Japan.

Conflicts of interest and sources of funding: none declared.

Correspondence to: Masako Kawasaki, MD, Department of Radiology, Tokyo Medical University, 6-7-1 Nishishinjuku, Shinjuku-ku, Tokyo 160-0023, Japan. E-mail: 1107bluemoon@gmail.com.

Copyright @ 2019 Wolters Kluwer Health, Inc. All rights reserved. ISSN: 0363-9762/19/0000-0000

DOI: 10.1097/RLU.0000000000002852

REFERENCES

1. Sugiyama T, Tajiri T, Hiraiwa S, et al. Hepatic adrenal rest tumor: diagnostic pitfall and proposed algorithms to prevent misdiagnosis as lipid-rich hepato- cellular carcinoma. Pathol Int. 2015;65:95-99.

2. Enjoji M, Sanada K, Seki R, et al. Adrenal rest tumor of the liver preopera- tively diagnosed as hepatocellular carcinoma. Case Rep Surg Volume. 2017; 2017:8231943.

3. Anderson JR, Ross AH. Ectopic adrenal tissue in adults. Postgrad Med J. 1980;56:806-808.

4. Jayarajah U, Herath KB, Fernando MH, et al. Testicular adrenal rest tumour in an adult patient with congenital adrenal hyperplasia: a case report and review of literature. J Surg Case Rep. 2018;10:rjy255.

5. Saeger W. Ectopia of the adrenal. Pathologe. 2018;39:409-414.

6. Honma K. Adreno-hepatic fusion. An autopsy study. Zentralbl Pathol. 1991; 137:117-122.

7. Woo HS, Lee KH, Park SY, et al. Adrenal cortical adenoma in adrenohepatic fusion tissue: a mimic of malignant hepatic tumor at CT. Am J Roentgenol. 2007;188:W246-W248.

8. Matsukuma S, Kono T, Takeo H, et al. Intra-adrenal bile ductules associated with adreno-hepatic fusion: a possible origin for adrenal epithelial cysts. Histopathology. 2013;62:799-804.

FIGURE 1. The patient was a 64-year-old woman who was being treated with prednisolone for autoimmune hepatitis. A well-defined mass of about 10 mm was demonstrated in S7 of the liver. The lesion was enhanced in the arterial phase (A-1) and washed out in the equilibrium phase (A-2) of the enhanced CT. Dynamic contrast-enhanced MRI with Gd-EOB-DTPA (gadolinium ethoxybenzyl diethylenetriaminepentaacetic acid) showed high intensity in the arterial phase (B-1) and low in the hepatobiliary phase (B-2). The signal was weaker in the opposed phase (B-4) than in the in-phase (B-3) on chemical shift imaging, which confirmed the presence of a fat component in the lesion.1,2 As the lesion was located close to the right adrenal gland,3-5 and the level of a-fetoprotein was normal, adrenocortical scintigraphy with NCL-6-1311 (1311-6-B-iodomethyl-19- norcholesterol) SPECT/CT was performed. Obvious uptake was displayed in the lesion; on the other hand, there was faint uptake in the bilateral adrenal glands (C-1 and C-2). Liver biopsy (D-1: hematoxylin-eosin staining, D-2: immunostaining) confirmed the presence of many clear cytoplasm cells forming a cluster, which was histologically similar to the zona fasciculata of the adrenal cortex (the red square in D-1) with normal hepatic tissue adjacent to the lesion. The border between the lesion and normal tissue was indistinct, and hyperplasia of the fibrous stroma was also observed (arrow in D-1), which suggested adrenohepatic union6-8 (D-1 and D-2). There was no tumor component in the lesion associated with adrenal rest or hepatic cells. Normal adrenocortical activity might be suppressed because of the treatment with prednisolone.

A-1

A-2

C-1

C-2

B-1

B-2

D-1

2

B-3

B-4

D-2

FIGURE 2. The patient was a 39-year-old woman with an increased CA-19-9 level. Dynamic CT displayed a mass lesion of about 16 mm in S7, which was well enhanced in the arterial phase (A-1) and was washed out in the equilibrium phase (A-2). Dynamic contrast-enhanced MRI displayed hypervascularity in the arterial phase (B-1) and low intensity in the hepatobiliary phase (B-2). The signal was weaker in the opposed phase (B-4) than in the in-phase (B-3) on chemical shift imaging, similarly to the previous case. On adrenocortical scintigraphy, uptake was detected in the mass lesion together with comparable uptake in the bilateral adrenal glands (C-1 and C-2). The lesion (arrow in C-2) was located on the dorsal side of the normal adrenal tissue (arrowhead in C-2). As there was no evidence of autonomic secretion of cortisol, follow-up observation was performed.

A-1

A-2

C-1

C-2

B-1

B-2

B-3

B-4