Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jul 16, 2021; 9(20): 5675-5682
Published online Jul 16, 2021. doi: 10.12998/wjcc.v9.i20.5675
Oncocytic adrenocortical tumor with uncertain malignant potential in pediatric population: A case report and review of literature
Xiao-Chun Chen, Yun-Man Tang, Yu Mao, Dao-Rui Qin
Xiao-Chun Chen, Yun-Man Tang, Yu Mao, Dao-Rui Qin, Department of Pediatric Surgery, Sichuan Academy of Medical Sciences Sichuan Provincial People's Hospital, Chengdu 610072, Sichuan Province, China
Author contributions: Chen XC and Qin DR designed the research study; Chen XC, Tang YM, Mao Y and Qin DR performed the research; Chen XC analyzed the data and wrote the manuscript; All authors have read and approve the final manuscript.
Informed consent statement: Written informed consent was obtained from the patients for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors have nothing to disclose.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Dao-Rui Qin, PhD, Surgeon, Department of Pediatric Surgery, Sichuan Academy of Medical Sciences Sichuan Provincial People's Hospital, No. 32 West Two Section, First Ring Road, Qingyang District, Chengdu 610072, Sichuan Province, China. daorui1982@163.com
Received: March 3, 2021
Peer-review started: March 3, 2021
First decision: April 14, 2021
Revised: April 25, 2021
Accepted: May 8, 2021
Article in press: May 8, 2021
Published online: July 16, 2021
Processing time: 125 Days and 19.5 Hours
Abstract
BACKGROUND

Oncocytic adrenocortical tumor (OACT) is rare, with few cases reported in the literature. No more than 20 cases in children have been reported. The clinical characteristics, diagnosis, treatment and prognosis of children with OACT are summarized based on a literature review, in order to improve the understanding of OACT in children.

CASE SUMMARY

We report a case of a 17-mo-old patient who was admitted to our hospital due to symptoms of odynuria and fever, which are clinical features consistent with a functional adrenocortical tumor. The patient was diagnosed with OACT of uncertain malignant potential. Computed tomography indicated a soft tissue giant tumor in the right adrenal region, approximately 4.3 cm × 5.5 cm in size. Multiple nodular and speckled calcifications were observed in the lesion. The patient received robot-assisted laparoscopic right adrenal tumor resection. Postoperative pathological results were consistent with OACT, and immunohistochemical results showed cytokeratin+/-, chromogranin A+, synaptophysin-, neuron-specific enolase-, S100-, Ki67 about 10%, CD34- and D2-40-. After surgery, urinary tract ultrasonography was reviewed monthly, catecholamine hormone and sex hormone levels were examined every 2 mo and computed tomography was performed every 6 mo. To date, no tumor metastasis or recurrence has been identified in this patient. The levels of sex hormones and catecholamine hormones decreased to normal 1 mo after surgery.

CONCLUSION

OACT is rare in the pediatric population, with few cases reported in the literature. Although most pediatric OACTs are benign, malignant cases have been reported. Surgical resection is the preferred option in most patients.

Keywords: Oncocytic adrenocortical tumor; Adrenocortical tumor; Robotic surgery; Children; Case report

Core Tip: Oncocytic adrenocortical tumor (OACT) is a very rare tumor, with few cases reported in the literature. Surgical resection is the preferred treatment option in most cases. We report the first case of robot-assisted laparoscopic resection of an OACT in a pediatric patient. The findings from this case report and the literature review show that robot-assisted laparoscopic resection of pediatric OACT with a complete tumor capsule, clear boundary between the tumor and surrounding tissues and no regional lymph node invasion is safe. Radiation and chemotherapy do not seem to be necessary in pediatric OACT without metastasis.