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Asian Journal of Surgery

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Asian Journal of Surgery

Letter to Editor

Treatment of a case of adrenocortical sarcomatoid carcinoma

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Keywords: Adrenocortical carcinoma Sarcomatoid Recurrence Metastasis

To the editor,

Adrenal sarcomatoid carcinoma (ASC) is a very rare aggressive variant of adrenocortical carcinoma. It is a poorly differentiated car- cinoma with poor prognosis, and the diagnosis needs careful histo- logical and immunohistochemical investigation.1 To date, a total of 21 cases have been reported worldwide.2We recently admitted another patient with ASC and shared some of our experience in the treatment process.

A 68-year-old male was admitted to the hospital with “Right adrenal gland occupancy was found for 10+ days”.The whole abdominal CT: “A mass shadow about 43 mm*46 mm*53 mm in size was observed in the right adrenal region, with significant het- erogeneous enhancement at the edges of the mass and a large central unenhanced necrotic area.”(Fig. 1:A)Preoperative cardio- pulmonary function and adrenal hormone tests showed no signif- icant abnormalities.With a history of previous multiple abdominal surgeries, a robot-assisted laparoscopic right retroperitoneal occupied mass resection was performed in November 2021, and

a solid mass 5.5 cm*4.5 cm*3 cm in size in the right adrenal region was seen intraoperatively, with close adhesions to the adrenal gland, liver and surrounding vessels, and obvious edematous ad- hesions in the surrounding tissues(Fig. 1:B).Postoperative pathol- ogy revealed:KI-67 (70%); MDM2 (-); SMA (-); EMA (-), consistent with ASC(Fig. 1:C).Fluorescence in situ hybridization (FISH) did not show MDM2 (12q15) gene amplification.Postoper- atively, the patient refused adjuvant chemotherapy and was treated with hepatic tumor vascular embolization after a review 3 months after surgery revealed the possibility of liver recurrence of metastasis.He died 4 months after surgery due to severe anorexia, intestinal obstruction, and multiple organ failure.

ASC is highly aggressive and has a very poor prognosis, often combined with multiple metastases, where KI-67 > 10% in immu- nohistochemistry often indicates a high possibility of prognostic recurrence,1 and the KI-67 in this patient is 70%. Secondly, regarding the surgical approach, this report is the first robot- assisted surgical treatment.Although many guidelines recommend open surgery, laparoscopic operation can also be selected for pa- tients whose mass diameter is < 6 cm. However, this case has a his- tory of previous repeated abdominal surgery, which was found feasible after trying robotic laparoscopy, and it is also the first report that robotic surgery is completed.

Most patients with ASC have a postoperative survival of 3-12 months after surgical treatment, with the longest metastasis-free postoperative survival of 17 months.3 When there is no distant metastasis or local invasion, adjuvant chemotherapy is recommen- ded after the first operation.To date, no evidence-based consensus has been reported on the optimal oncologic treatment for ASC.

Fig. 1. A): A mass shadow about 43 mm*46 mm*53 mm in size was observed in the right adrenal region, with significant heterogeneous enhancement at the edges of the mass and a large central unenhanced necrotic area. B):Surgical specimen : a solid mass 5.5 cm*4.5 cm*3 cm in size. C):Postoperative pathological image.

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Fundation

This study is supported by Key research and development pro- jects of Sichuan Science and Technology Department ( 2022YFS0135 ) and “The General Item of Medical Engineering Cross ” (serial number ZYGX2021YGLH011) of Sichuan Provincial people’s Hospital, University of Electronic Science and Technology of China.

Authorship

Keyang Jia, Data analysis and/or interpretation, Drafting of manuscript and/or critical revision, Approval of final version of manuscript, Acquisition of data (laboratory or clinical): Yi Wei, Data analysis and/or interpretation, Drafting of manuscript and/or critical revision, Approval of final version of manuscript, Acquisi- tion of data (laboratory or clinical): Dong Wang, Data analysis and/or interpretation, Drafting of manuscript and/or critical revi- sion, Approval of final version of manuscript: Shangqing Ren, Data analysis and/or interpretation, Drafting of manuscript and/or critical revision, Approval of final version of manuscript, Concep- tion and design of study, Acquisition of data (laboratory or clinical)

Declaration of competing interest

The authors declared no competing interests.

Appendix A. Supplementary data

Supplementary data to this article can be found online at https://doi.org/10.1016/j.asjsur.2022.05.161.

References

1. Lam AK. Adrenocortical carcinoma: updates of clinical and pathological features af- ter renewed world health organisation classification and pathology staging. Feb 10 Biomedicines. 2021;9(2):175. https://doi.org/10.3390/biomedicines9020175. PMID:

33578929; PMCID: PMC7916702.

2. Sabrine D, Zakia B, Kaoutar Z. Adrenocortical sarcomatoid carcinoma: a case report and review of the literature. Jul 16;2020 J Surg Case Rep. 2020;(7). https://doi.org/10.1093/jscr/rjaa211. rjaa211, PMID: 32714505; PMCID: PMC7371253.

3. Papathomas TG, Duregon E, Korpershoek E, et al. Sarcomatoid adrenocortical carcinoma: a comprehensive pathological, immunohistochemical, and targeted next-generation sequencing analysis. Dec Hum Pathol. 2016;58:113-122. https://doi.org/10.1016/j.humpath.2016.08.006. Epub 2016 Aug 31. PMID: 27589897.

Keyang Jia1 School of Medicine, University of Electronic Science and Technology of China, Chengdu, 611731, China

Yi Wei1 Chengdu Medical College, Chengdu, 610500, China

Department of Robotic Minimally Invasive Surgery Center, Sichuan Academy of Medical Sciences & Sichuan Provincial People’s Hospital, Chengdu, 610072, China

Dong Wang

Department of Robotic Minimally Invasive Surgery Center, Sichuan Academy of Medical Sciences & Sichuan Provincial People’s Hospital, Chengdu, 610072, China

Chinese Academy of Sciences Sichuan Translational Medicine Research Hospital, Chengdu, 610072, China

Shangqing Ren Department of Robotic Minimally Invasive Surgery Center, Sichuan Academy of Medical Sciences & Sichuan Provincial People’s Hospital, Chengdu, 610072, China

Chinese Academy of Sciences Sichuan Translational Medicine Research Hospital, Chengdu, 610072, China

Corresponding author. E-mail address: rsq0516@163.com (S. Ren).

19 May 2022 Available online 16 June 2022

1 Contributed equally.