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Asymptomatic paraganglioma of urinary bladder
Tai-Li Chen1, Yuan-Hong Jiang2, Yung-Hsiang Hsu3
1 Department of Medical Education, Hualien Tzu Chi Hospital, Buddhist Tzu Chi Medical Foundation, Hualien, Taiwan
2 Department of Urology, Hualien Tzu Chi Hospital, Buddhist Tzu Chi Medical Foundation and Tzu Chi University, Hualien, Taiwan
3 Department of Pathology, Hualien Tzu Chi Hospital, Buddhist Tzu Chi Medical Foundation and Tzu Chi University, Hualien, Taiwan
| Date of Submission | 09-Dec-2020 |
| Date of Decision | 07-Jan-2021 |
| Date of Acceptance | 18-Jan-2021 |
| Date of Web Publication | 05-Apr-2021 |
Correspondence Address:
Yung-Hsiang Hsu,
Department of Pathology, Hualien Tzu Chi Hospital, Buddhist Tzu Chi Medical Foundation, 707, Section 3, Chung-Yang Road, Hualien
Taiwan
 Source of Support: None, Conflict of Interest: None DOI: 10.4103/tcmj.tcmj_292_20
A 58-year-old previously healthy woman was referred to a urologist for an incidental finding of bladder tumor on computed tomography (CT) [Figure 1]a. There was no urinary frequency, urgency, hematuria, or dysuria. No fever, weight loss, or micturition syncope was complained. She denied any family history of malignancy or hereditary disease. | Figure 1: (a) A 2-cm hyperdensity lesion at the right urinary bladder was indicated in the abdominal computed tomography. (b) The image of cystoscope revealed a 2-cm broad-base tumor at the right bladder base. (c) Pathology indicated trabeculae of polygonal cells, with pepper-salt-like nuclei and abundant granular cytoplasm (H and E, ×100). (d) S-100 stain demonstrated the rare sustentacular cells, which were highlighted by the arrows (S-100, ×100)
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Physical examination showed a vitally stable female with normal blood pressure. No discomfort was indicated during the lower abdominal examination. She was admitted for further workup. Laboratory studies revealed no significant finding. A cystoscope was conducted and showed a 2-cm broad-base tumor at the right bladder base [Figure 1]b. Transurethral resection of bladder tumor (TUR-BT) with tumor biopsy was performed. The results of histopathology revealed nests (zellballen) or trabeculae of polygonal cells, with pepper-salt-like nuclei and abundant granular cytoplasm [Figure 1]c. The pathological tumor margin was free indicating that the tumor was completely resected. Immunohistochemical stain revealed positive for cluster of differentiation 56 (CD56), chromogranin, synaptophysin, and S-100 and negative for cytokeratin 7 (CK7). Sustentacular cells were highlighted by S-100 staining [Figure 1]d. The diagnosis of paraganglioma of the urinary bladder was made.
The patient was discharged 1 day after the surgery. Postoperative urine examination for vanillylmandelic acid was followed, and the results were within normal range. Her condition was good when she returned to the urology outpatient department for a follow-up 1 month after the operation.
Pheochromocytomas, which are rare tumors of neuroendocrine origin, arise from neural crest-derived chromaffin cells [1]. Extra-adrenal pheochromocytomas are often refer to as paragangliomas [1]. In the genitourinary tract, urinary bladder is the most prevalent site for paragangliomas [2]. Case reports have described the presentations of paraganglioma of the bladder [3],[4]. In this case report, we presented a case of asymptomatic paraganglioma of the urinary bladder, with typical pathological characteristics. Symptoms such as paroxysmal headache, palpitations, and sweating may not always be encountered in such patients.
Based on the activity of catecholamine that arises from the tumor, paraganglioma can be classified as functional or nonfunctional [2]. Functional bladder paragangliomas may be detected throughout the catecholamine-secreting symptoms including micturition syncope, hypertension, and hematuria [3]. Nevertheless, patients presenting without catecholamine-secreting symptoms are often underdiagnosis [4]. Careful physical examination for palpable mass and appropriate image study may be beneficial for such patients. In our case, the patient was incidentally found to have a bladder tumor on CT image.
General neuroendocrine markers such as CD56, chromogranin, synaptophysin, and S-100 were applied in our pathologic analysis. To distinguish between paraganglioma and carcinoid tumor, we utilized CK7 stain. Carcinoid tumors may stain positive for CK7 in the parenchyma, while paragangliomas are negative for CK7. In our histopathologic examination, CK7 showed negative, which was favored for paraganglioma.
Management of paraganglioma of the urinary bladder required subsequent surgical resection [5]. However, the extent of tumor invasion needs to be taken into consideration. Besides TUR-BT, partial cystectomy or chemotherapy may be beneficial for patients with vesicular muscle invasion or distant metastases.
In conclusion, we reported a relatively uncommon patient of asymptomatic bladder paraganglioma, which is difficult to diagnose preoperatively. Despite the rarity of paraganglioma of urinary bladder, patients with asymptomatic presentations required more attention.
Acknowledgments
The author thank the Department of Medical Research, Hualien Tzu Chi Hospital, Buddhist Tzu Chi Medical Foundation, for the invaluable contribution to the methodological aspects of the present case report.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initials will not be published and due efforts will be made to conceal the identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
Dr. Yuan-Hong Jiang and Yung-Hsiang Hsu, the editorial board members at Tzu Chi Medical Journal, had no roles in the peer review process of or decision to publish this article. The other author declared no conflict of interest in writing this paper.
| References | |  |
| 1. | Elder EE, Elder G, Larsson C. Pheochromocytoma and functional paraganglioma syndrome: No longer the 10% tumor. J Surg Oncol 2005;89:193-201.  |
| 2. | Purnell S, Sidana A, Maruf M, Grant C, Agarwal PK. Genitourinary paraganglioma: Demographic, pathologic, and clinical characteristics in the surveillance, epidemiology, and end results database (2000-“2012). Urol Oncol 2017;35:457.e9-14. |
| 3. | Beilan JA, Lawton A, Hajdenberg J, Rosser CJ. Pheochromocytoma of the urinary bladder: A systematic review of the contemporary literature. BMC Urol 2013;13:22. |
| 4. | Johnson JT, Cherian KE, Kapoor N, Paul TV. Micturition syncope secondary to urinary bladder paraganglioma. BMJ Case Rep 2020;13:e233556. |
| 5. | Andrade MO, Cunha VS, Oliveira DC, Moraes OL, Lofrano-Porto A. What determines mortality in malignant pheochromocytoma? Report of a case with eighteen-year survival and review of the literature. Arch Endocrinol Metab 2018;62:264-9. |
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