ASIAN

CALI

FOUNDED 1976

Contents lists available at ScienceDirect

Asian Journal of Surgery

journal homepage: www.e-asianjournalsurgery.com

Asian Journal of Surgery

Letter to Editor

Adrenocortical oncocytic carcinoma misdiagnosed as adrenal cortical adenoma

Check for updates

Dear editor,

Adrenal cortical carcinoma, originating from adrenal cortical cells, is a rarely malignant tumor in clinic, with an annual incidence rate of only 0.5-2 per million.1 Adrenocortical oncocytic carcinoma (AOC) is one of the exceptional subtypes. Compared to adrenal cortical adenomas, which usually only secrete one type of steroid hormone, adrenal cortical carcinomas can secrete multiple steroid hormones, and most commonly co-secrete cortisol and androgens, with less than a third of adrenal cortical carcinomas being non- functional.2,3 However, as we reported in this case, about two- thirds of oncocytic adrenocortical neoplasms are nonfunctional.4

A 35-years-old female presented to our department for a right adrenal gland tumor found during a physical examination. We completed the hormonal and radiological assessments. Preopera- tive plasma cortisol, plasma aldosterone and the 24-h urinary cat- echolamines and their metabolites test was normal. Adrenal computed tomography (CT) showed the right adrenal region showed irregular shape mass with heterogeneous low-density, size about 70 mm*66 mm, and the enhanced scan was unevenly enhanced, considering pheochromocytoma (Fig. 1 a and b). because the tumor was too large and had the possibility of malignancy, we performed a preoperative needle biopsy on the patient according to the Chinese urology guidelines. The preoperative needle biopsy

Fig. 1. a: Patient's preoperative CT. b: The patient's preoperative contrast CT. A. Melan-A (+). B. SF-1 (+). C. Calretinin (+). D. Inhibin-a (+). E. VIM (+). F. Syn (+). G. CgA (+). H. KI- 67(15 %).

a

b

6.90 cm

105 cm

6.50 cm

A

B

C

D

E

F

G

H

was adrenocortical adenoma.

After preoperative preparation, the patient underwent laparo- scopic right adrenalectomy. Two hours later, we had the tumor removed completely. Post-operative histopathological examina- tion: Right adrenal tumor, complete grey-white capsule, one grey-brown tenacious mass, size 8.5*6.5*5 cm, cut on multiple sides, grey-yellow, grey-brown, slightly tenacious, local hemor- rhage. Pathological diagnosis: adrenocortical oncocytic carcinoma, see mitotic rate)5/50HFP, atypical mitotic, small foci necrosis, suspi- cious capsular invasion, according to Lin-Weiss-Bisceglia criteria,3 in line with adrenocortical oncocytic carcinoma. Immunohisto- chemistry (Fig. 1 A to H): Melan-A (+), SF-1 (+), Calretinin (+), Inhibin-a (+), VIM (+), Syn (+), CgA (+), KI-67(15 %). The first three months follow up, we conducted CT scan and hormone assessment, there was no tumor metastasis and hormone secretion abnormality.

The case report concern: Adrenocortical oncocytic carcinoma is an extremely rare tumour that is mostly nonfunctional. According to few reports, adrenocortical oncocytic carcinoma seem to have a better prognosis than other types of adrenal cortical carcinoma. Expert knowledge of adrenocortical oncocytic carcinoma as an extremely rare tumor is limited, and we hope that more case re- ports will be reported in the future to emphasize awareness of the disease; Based on this case, the pathological results of preoper- ative biopsy are not completely credible, especially when you thought malignment tumor. We recommend that the diagnosis of malignment adrenal tumor suggestion is combination with multi- ple evidence, such as patient signs, biochemical tests, medical im- aging (CT, MRI, and PET-CT), etc.

Conflicts of interest

There are no potential financial and non-financial conflicts of interest.

References

1. Fassnacht M, Assie G, Baudin E, et al. Adrenocortical carcinomas and malignant phaeochromocytomas: ESMO-EURACAN clinical practice guidelines for diag- nosis, treatment and follow-up. Ann Oncol. 2020;31:1476-1490.

2. Libè R, Fratticci A, Bertherat J. Adrenocortical cancer: pathophysiology and clin- ical management. Endocr Relat Cancer. 2007;14:13-28.

3. Allolio B, Fassnacht M. Adrenocortical carcinoma: clinical update. J Clin Endocri- nol Metabol. 2006;91:2027-2037.

4. Wong DD, Spagnolo DV, Bisceglia M, et al. Oncocytic adrenocortical neoplasms-a clinicopathologic study of 13 new cases emphasizing the importance of their recognition. Hum Pathol. 2011;42:489-499.

5. Bisceglia M, Ludovico O, Di Mattia A, et al. Adrenocortical oncocytic tumors: report of 10 cases and review of the literature. Int J Surg Pathol. 2004;12: 231-243.

Yang Li, Pei Li Department of Urology, The Second Affiliated Hospital of Kunming Medical University, No.374 Dian-Mian Avenue, Kunming, 650101, China E-mail addresses: 2742890767@qq.com (Y. Li), peilinli@live.cn (P. Li).

Fu-kai Tao Chuxiong People’s Hospital, China E-mail address: 974965457@qq.com.

Jiong-ming Li*

Department of Urology, The Second Affiliated Hospital of Kunming Medical University, No.374 Dian-Mian Avenue, Kunming, 650101, China

* Corresponding author. Department of Urology, The Second Affiliated Hospital of Kunming Medical University, No.374 Dian- Mian Avenue, Kunming, 650101, China. E-mail address: jiongmingli@aliyun.com (J .- m. Li).

13 December 2023 Available online 1 February 2024