Nonfunctioning Juxtaglomerular Cell Tumor
Yoji Nagashima2013, Case Reports in Pathology
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The juxtaglomerular cell tumor (JGCT) is a rare renal tumor characterized by excessive renin secretion causing intractable hypertension and hypokalemia. However, asymptomatic nonfunctioning JGCT is extremely rare. Here, we report a case of nonfunctioning JGCT in a 31-year-old woman. The patient presented with a left renal tumor without hypertension or hypokalemia. Under a clinical diagnosis of renal cell carcinoma, radical nephrectomy was performed. The tumor was located in the middle portion adjacent to the renal pelvis, measuring 2 cm in size. Pathologically, the tumor was composed of cuboidal cells forming a solid arrangement, immunohistochemically positive for renin. Based on these findings, the tumor was diagnosed as JGCT. In cases with hyperreninism, preoperative diagnosis of JGCT is straightforward but difficult in nonfunctioning case. Generally, JGCT presents a benign biological behavior. Therefore, we should take nonfunctioning JGCT into the differential diagnoses for renal...
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Hindawi Publishing Corporation
Case Reports in Pathology
Volume 2013, Article ID 973865, 3 pages
http://dx.doi.org/10.1155/2013/973865
Case Report
Nonfunctioning Juxtaglomerular Cell Tumor
Ryoko Sakata,1 Hiroaki Shimoyamada,2,3 Masahiro Yanagisawa,1
Takayuki Murakami,1 Kazuhide Makiyama,1 Noboru Nakaigawa,1
Yoshiaki Inayama,4 Kenichi Ohashi,2,4 Yoji Nagashima,2
Masahiro Yao,1 and Yoshinobu Kubota1
1
Department of Urology, Yokohama City University Graduate School of Medicine, 3-9 Fukuura, Kanazawa-ku,
Yokohama 236-0004, Japan
2
Department of Pathology, Yokohama City University Graduate School of Medicine, 3-9 Fukuura, Kanazawa-ku,
Yokohama 236-0004, Japan
3
Department of Pathology, Kyorin University Graduate School of Medicine, 6-20-2, Shinkawa, Mitaka, Tokyo 181-8611, Japan
4
Division of Anatomical and Surgical Pathology, Yokohama City University Hospital, 3-9 Fukuura, Kanazawa-ku,
Yokohama 236-0004, Japan
Correspondence should be addressed to Ryoko Sakata; [email protected]
Received 31 January 2013; Accepted 27 February 2013
Academic Editors: S. Baeesa, T. Batinac, R. L. Haas, W. Khalbuss, P. Kornprat, M. Marino, and W. P. Wang
Copyright © 2013 Ryoko Sakata et al. This is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
The juxtaglomerular cell tumor (JGCT) is a rare renal tumor characterized by excessive renin secretion causing intractable
hypertension and hypokalemia. However, asymptomatic nonfunctioning JGCT is extremely rare. Here, we report a case of
nonfunctioning JGCT in a 31-year-old woman. The patient presented with a left renal tumor without hypertension or hypokalemia.
Under a clinical diagnosis of renal cell carcinoma, radical nephrectomy was performed. The tumor was located in the middle
portion adjacent to the renal pelvis, measuring 2 cm in size. Pathologically, the tumor was composed of cuboidal cells forming
a solid arrangement, immunohistochemically positive for renin. Based on these findings, the tumor was diagnosed as JGCT. In
cases with hyperreninism, preoperative diagnosis of JGCT is straightforward but difficult in nonfunctioning case. Generally, JGCT
presents a benign biological behavior. Therefore, we should take nonfunctioning JGCT into the differential diagnoses for renal
tumors, especially in younger patients to avoid excessive surgery.
1. Introduction
2. Case Report
Juxtaglomerular cell tumor (JGCT), a neoplasm derived from
the juxtaglomerular cell of the kidney, was first described
by Robertoson et al. and Kihara et al. [1, 2]. Since then,
approximately 100 cases have been reported in the literature.
Clinically, this tumor is characterized by hypertension due
to excessive renin secretion by tumor cells causing secondary hyperaldosteronism [1–3]. Generally, its preoperative
diagnosis is relatively easy, because of typical presence of
hypertension concomitant with hypokalemia. However, it is
quite difficult in cases of asymptomatic nonfunctioning JGCT
[3]. Here, we present a case of nonfunctioning JGCT, which
is the fourth case reported so far to the best of our knowledge
[4–6].
A 31-year-old woman was referred to our hospital with a
left renal tumor incidentally detected during examination for
left-side abdominal pain. She had no history of hypertension,
and her blood pressure at presentation was 116/62 mmHg. All
the laboratory data, including electrolyte levels, were within
normal ranges. Unfortunately, preoperative plasma renin
activity was not assayed. Unenhanced computed tomography (CT) revealed solitary well-circumscribed mass lesion
measuring 2 cm with fine calcifications in the middle portion
adjacent to the renal pelvis of the left kidney. Dynamicenhanced CT demonstrated that the tumor was not enhanced
in the corticomedullary (early) phase but enhanced in the
excretory (late) phase (Figure 1). Magnetic resonance image
2
Figure 1: Enhanced CT showing the tumor (excretory phase)
(arrow).
Figure 2: Gross findings of the resected kidney. The tumor located
in the middle portion and was well demarcated from the surrounding structures. The cut surface was white to tan in color.
(MRI) showed a well-defined, round mass in the middle
portion of the kidney that appears isointense on T1-weighted
image. The mass showed low intensity in fat saturation T2weighted image. With a clinical diagnosis of renal cell carcinoma, the patient underwent laparoscopic radical nephrectomy.
Grossly, the resected kidney contained an encapsulated
yellowish white mass in the middle portion (Figure 2).
Histologically, the tumor was composed of densely
packed cells surrounding tubular spaces with cuboidal lining.
The tumor cells were uniformly small in size, polygonal
in shape, possessed small round nuclei, and were clear to
eosinophilic cytoplasm. Their nuclei contained fine chromatin and inconspicuous nucleoli (Figure 3). Ultrastructurally, the tumor cells contained rhomboid-shaped granules in the cytoplasm, consistent with renin protogranules
(Figure 4). Immunohistochemically, the tumor cells showed
strong positivity for renin (Figure 5) but were negative
for cytokeratin and epithelial membrane antigen. Because
epithelial markers, but not renin, were positive in the spacelining cells, the tubular architectures seemed to be derived
from dilated tubules involved by the tumor.
Case Reports in Pathology
Figure 3: Histological findings of the tumor. The tumor was
composed of solid cell sheets containing tubular architectures.
Figure 4: Electron micrograph of a tumor cell. Rhomboidal crystalline bodies are observed in the cytoplasm of a tumor cell (arrow).
Based on these findings, the tumor was diagnosed as
nonfunctioning JGCT. Postoperatively, the patient is doing
well for more than 3 years of followup.
3. Discussion
JGCT is a rare benign renal neoplasm, which mostly affects
the younger females (second to third decades; mean age, 27
years old; male : female = 1 : 2). It is most frequently found
during the examination for intractable hypertension.
On plain CT, JGCT usually appears as a unilateral wellcircumscribed hypoattenuating cortical mass. At dynamic
CT, the tumors are not enhanced in the arterial phase despite
the profuse vascularity, possibly because of renin-induced
vasoconstriction. The tumors show moderate enhancement
during the delayed phase [7, 8]. At MRI, JGCT may appear
as a cortical-based renal mass of variable signal intensity that
shows delayed peripheral enhancement on dynamic contrastenhanced MRIs [9].
Grossly, the tumor is well circumscribed and firm in
consistency, and the cut surface is yellowish to tan in color.
Histologically, the tumor is composed of sheets, papillae,
and trabeculae of uniform polygonal cells. Their nuclei are
small with fine chromatin, and the cytoplasm is granular.
Case Reports in Pathology
Figure 5: Immunohistochemical micrograph of the tumor. The
majority of tumor cells were positive for renin, although the tubular
lining was negative.
3
not been precisely clarified due to the limited number of
cases. Enhanced pattern on CT of this case was not typical of
clear cell renal cell carcinoma. The possibility of other type of
RCC or low fat AML was considered to be discussed, because
the incidence of AML in a Japanese young female is relatively
high. Biopsy was an option of preoperative diagnosis. We
explained to the patient and her family that the tumor was
possibly not a renal cell carcinoma but a benign tumor such
as AML, and they chose an operation instead of a true cut
biopsy on FNA cytology.
In cases of renal tumors especially in young females,
even though no evidence of hyperreninism, JGCT should
be included in the candidates of differential diagnosis, and
excessive surgical treatment should be avoided to preserve the
renal function and not to lose the kidney.
References
Ultrastructurally, rhomboid renin protogranules are noted in
the cytoplasm. Immunohistochemically, the tumor cells are
positive for renin, but negative for epithelial markers. The
candidates of differential diagnosis are oncocytoma, epithelioid angiomyolipoma, renal cell carcinoma, and mesenchymal tumors such as glomus tumor, solitary fibrous tumor,
and hemangiopericytoma. Pathological diagnosis is usually
straightforward, because of characteristic clinical presentation [4]. In cases of nonfunctioning JGCT, immunohistochemistry is helpful for differential diagnosis. Because of negative reaction with epithelial markers (cytokeratin, epithelial
membrane antigen), RCC and oncocytoma are excluded.
Unlike epithelioid angiomyolipoma, JGCT is not reactive
with HMB45 antibody raised against melanosome-associated
antibody. Although hemangiopericytomatous proliferating
pattern is common with hemangiopericytoma and solitary
fibrous tumor, differential diagnosis can be performed by
detection of intracytoplasmic rennin with immunohistochemistry and/or electron microscopy [3].
Because JGCT is mostly benign with an exceptional case
causing metastasis [10], nephron-sparing surgery, such as
partial nephrectomy, should be considered.
Although the classical triad of JGCT is poorly controlled
hypertension, elevated renin level, and renal mass, some
cases had been reported which lacked hypertension and/or
hypokalemia [4–6]. According to presence or absence of
the symptoms, JGCT is subdivided into three groups [6].
First, the typical variant, accounting for the majority, presents
elevated plasma renin activity and secondary hyperaldosteronism causing hypokalemia and hypertension. Second is the
atypical variant showing hypertension but normal potassium
level, which is common next to the typical variant. The third
is the nonfunctioning variant, characterized by normal blood
pressure and normal potassium level. Nonfunctioning variant
is the rarest and is thought to produce inactive rennin [5]. Our
case is classified into nonfunctioning variant. Due to lack of
evidence of excessive renin secretion, we failed to consider
the possibility of JGCT preoperatively. Preoperative imaging
analysis on predictable histological types was limited because
characteristics of renal tumors except typical clear cell renal
cell carcinoma as seen in different imaging techniques have
[1] P. W. Robertoson, A. Klidijan, K. Harding, G. Walters, M. R.
Lee, and A. H. T. Robb-Smith, “Hypertension due to a renninsecreting renal tumor,” The American Journal of Medicine, vol.
43, no. 6, pp. 963–976, 1967.
[2] I. Kihara, S. Kitamura, T. Hoshino, H. Seida, and T. Watanabe,
“A hitherto unreported vascular tumor of the kidney: a proposal
of ‘juxtaglomerular cell tumor’,” Acta Pathologica Japonica, vol.
18, no. 2, pp. 197–206, 1968.
[3] N. Kuroda, H. Gotoda, C. Ohe et al., “Review of juxtaglomerular
cell tumor with focus on pathobiological aspect,” Diagnostic
Pathology, vol. 6, article 80, 2011.
[4] D. Dong, H. Li, W. Yan, and W. Xu, “Juxtaglomedular cell tumor
of the kidney: a new classification schme,” Urologic Oncology,
vol. 28, no. 1, pp. 34–38, 2010.
[5] Y. Endoh, T. Motoyama, S. Hayami, and I. Kihara, “Juxtaglomerular cell tumor of the kidney: report of a nonfunctioning variant,” Pathology International, vol. 47, no. 6, pp.
393–396, 1997.
[6] H. Takahashi, T. Ohsawa, and A. Nakamura, “A case of juxtaglomerular cell tumor,” Nippon Hinyokika Gakkai Zasshi, vol.
85, article 531, 1985.
[7] S. R. Prasad, N. C. Dalrymple, and V. R. Surabhi, “Crosssectional imaging evaluation of renal masses,” Radiologic Clinics
of North America, vol. 46, no. 1, pp. 95–111, 2008.
[8] A. Tanabe, M. Naruse, T. Ogawa et al., “Dynamic computer
tomography is useful in the differential diagnosis of juxtaglomerular cell tumor and renal cell carcinoma,” Hypertension
Research, vol. 24, no. 4, pp. 331–336, 2001.
[9] R. Agrawal, S. Z. H. Jafri, D. P. Gibson, K. G. Bis, and A. AliReza, “Juxtaglomerular cell tumor: MR findings,” Journal of
Computer Assisted Tomography, vol. 19, no. 1, pp. 140–142, 1995.
[10] X. Duan, P. Bruneval, R. Hammadeh et al., “Metastatic juxtaglomerular cell tumor in a 52-year-old man,” The American
Journal of Surgical Pathology, vol. 28, no. 8, pp. 1098–1102, 2004.
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