Original Article
Adrenal cortical carcinoma in Thailand: A review on the previous reported case
Joob B, Wiwanitkit V1
Sanitation 1 Medical Academic Center, 1Wiwinitkit House, Bangkhae, Bangkok Thailand
Correspondence to: Dr.Beuy Joob, E-mail: beuyjoob@hotmail.com
Abstract
BACKGROUND: Adrenal cortical carcinoma (ACC) is an uncommon cancer. MATERIALS AND METHODS: The author performed a literature review on the reports of ACC in Thailand in order to summarize the characteristics of this rare cancer among Thai patients. RESULTS: According to this study, there have been at least seven reports in the literature of nine individual cases of ACC, of which none was lethal directly to ACC. CONCLUSIONS: Here, the ACC is sporadically reported in Thailand. The diagnosis of ACC is usually by detection adrenal mass using imaging technique and the confirmation for the nature of cancer is usually by histopathology. Nevertheless, among the Thai population, the prognostic outcome is usually good.
Key Words: Adrenal cortical carcinoma, diagnosis, pathology
Introduction
Adrenal tumours, except for neuroblastoma, are relatively uncommon.[1] Most adrenal lesions are usually benign.[1] Since both benign and malignant tumours can have the ability to produce active hormone, it is very difficult to make a correct preoperative diagnosis.[1] Epidemiologically, adrenal cortical carcinoma (ACC) can be seen in estimated 0.02% of all cancers.[2] ACC is usually diagnosed at advanced stage at diagnosis.[2] Therefore, the overall 5 year survival is normally poor (20-45%).[2] However, the functional tumours in adult patients seems to have improved outcome due to the improvement of present medical management.[2]
Evaluation of a functional adrenal mass seems to be the basic requirement.[3] The starting point is usually overt symptomatic presentation or incidental detection of abnormal adrenal mass.[3] Differential diagnoses are needed for discrimination among different masses, which include pheochromocytoma, aldosteronoma, Cushing’s adenoma, and hormonally active ACC.[3] In Thailand, the first case report of ACC was published in 1983 by Chumaung.[4] Since the first case report, there have been sporadic case reports of ACCs in Thailand. Here, the author performed a literature review of the reports of ACC in Thailand in order to summarize the characteristics of this cancer among Thai patients.
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| DOI: 10.4103/0019-509X.123611 | |
Materials and Methods
This study was designed as a descriptive retrospective study. A literature review of the papers reporting ACC in Thailand was performed, using the referencing database of published works collected in IndexMedicus and Science Citation Index. The published works in all 256 local Thai journals which are not included in the international citation index was also reviewed using local medical database, ThaiIndexMedicus. The literature review focused on the years 1983-2012.
According to the preliminary review, seven reports were recruited for further study.[4-10] The important clinical presentation details of the included cases (such as clinical manifestation, diagnosis, treatment, and discharge status) in all included reports were extracted and summarised. The important demographic data including age, sex, and location were collected and summarized as well. Descriptive statistics, including range and percentage, were used in analysis with use of SPSS 7.0 SPSS inc., Chicago for windows computer programme.
Results
According to this study, there have been at least seven reports of overall nine cases of ACC in the Thai population, of which none was lethal due to ACC (but there were two death case due to other non-related disease) [Table 1]. In all cases, the surgical specimens were histologically confirmed to be ACC. Of all three cases with available data, there was no case with relapsing on the 10-year following-up (the other six cases have the following-up data less than 10 years until present and one case died due to other disease). Of the nine cases, the age ranged from 4 months to 67 years. All except one were males. Several different clinical characteristics among the nine cases are presented in Table 1.
Discussion
Adrenocortical carcinoma is considered rare. Its annual incidence is about 1.5 cases per million.[11] In this article, only nine cases were reported in Thailand, a country with about 60 million population. In our work, almost all cases are male, which is discordant with the findings in the Western countries (female more than male).[1]] Of interest, in this series, only six cases (about two-thirds) were hormone functional.[5,7-9] This is also concordant with the Western reports.[11] Focusing on the produced hormone, the cortisol and androgen are the two most common hormones in this series. This is similar to the finding in the previous Western reports (most common is cortisol following by androgen).[11]
For investigation, Computerised tomography scan and magnetic resonance imaging is the helpful investigation.[11,12] These imaging techniques help demonstrate necrotic lesions or areas of calcification in the mass which is the pathognomonic finding in this rare adrenal tumour.[11,12] These findings are also detected in cases in this series.[5,6] Complete surgical resection is presently the only one curative therapeutic option for ACC.[11] Additional chemotherapy with mitotane plus doxorubicin, etoposide and cisplatin is also suggested.[11] Although the high mortality of this cancer is reported in the previous case series, there is no death case in this series despite there is no administration of additional chemotherapy.[11,12] This observation is concordant with a recent report by Conzo et al., that “surgery represents the treatment of choice in case of primary adrenocortical cancer and is well indicated also for patients with advanced stage and recurrent loco-regional disease.”[13]
Based on this study, the ACC is sporadically reported in Thailand. The diagnosis of ACC is usually by
detection adrenal mass via imaging investigation and further histopathology study is usually used for confirming the nature of cancer. This might be general finding but it reflects the actual nature of diagnostic practice in Thailand. Indeed, in more developed countries, there might be more complex diagnostic approaches. The 2-[fluorine-18] fluoro-2-deoxy-D-glucose-positron emission tomography and metomidate is the good examples of new approaches in developed Western Countries.[14] The prognostic outcome is usually good. There was no death case due to ACC (but there were two deaths case due to nosocomial infection[7] and non-related peptic perforation induced respiratory failure[9]). The difference in the outcomes of disease from those reported in the Western population might reflect that there might be unknown ethnogenetic factors that might affect the outcome of ACC. Indeed, the relative good outcome of some endocrine tumours, which might imply a possible correlation to the ethnogenetic factors was recently reported by Yoshimoto et al.[15] However, due to the limitation due to few numbers of cases in this report, the finalized conclusion might not be made at this stage. Further researches to clarify this observation are needed.
Since this case series comply few cases, the interpretation of the results for further generalization might be limited. Here, the authors try to describe the features of ACC in Thailand via retrospective review of the previously published literatures. Nevertheless, the size of the sample is too small and might not representative of the population of Thailand (60 millions). The conclusion might not be made. Ones might make a query on the way to increase the sample size for this report and suggest for some alternative approaches. Indeed, reviewing the records of national hospitals in order to spot the cases of all ACC that was
| Table 1: Clinical characteristics of the 9 Thai cases of ACC | |||||
|---|---|---|---|---|---|
| Authors | Age (year) | Sex | Chief complaint | Diagnosis | Treatment |
| Chumaung[4] | 49 | Male | Large left upper quadrant mass | Intravenous pyelography | Surgical removal |
| Sriphatphiriyakun[5] | 46 | Male | Cushingoid features and virilisation | Utrasonography, CT scan | Surgical removal |
| Churesigaew[6] | 5 | Male | Low grade fever with low back pain | CT scan | Surgical removal |
| Jaruratanasirikul[7] | 4 | Male | Hyperandrogenism | CT scan | Surgical removal |
| Jaruratanasirikul et al.[7] | 6 | Male | Hyperandrogenism | CT scan | Surgical removal |
| Jaruratanasirikul et al.[7] | 5 | Female | Huge abdominal mass, heterosexual pseudorecocity | CT scan | Surgical removal |
| Pusantisampan et al.[8] | 4/12 | Male | Cushingoid features | CT scan | Surgical removal |
| Siriwong et al.[9] | 23 | Male | Cushingoid features and neck mass and superior | CT scan | Surgical removal |
| Muttarak et al.[10] | 67 | Male | Vena cava syndrome Chronic dyspepsia | CT scan | Surgical removal |
ACC=Adrenal cortical carcinoma; CT=Computerised tomography
Joob and Wiwanitkit: Adrenal cortical carcinoma in Thailand
surgically treated or prove to be inoperative and received palliative therapy might be helpful method to get more evidence. However, there is no good cancer registry report in Thailand and the record cannot be available, hence, it is not possible to perform such study. Since the focus of this work is on Thailand, which has several differences especially for ethnic background, to other Southeast Asian (SEA) countries, the extension to other published case reports in SEA is not done. Also, when the searching is done on the publications from the most ethnically similar countries in SEA to Thailand (Laos, Cambodia and Myanmar), there is also no report from those countries.
References
1. Rescorla FJ. Malignant adrenal tumors. Semin Pediatr Surg 2006; 15:48-56.
2. Roman S. Adrenocortical carcinoma. Curr Opin Oncol 2006; 18:36-42. Stifelman MD, Fenig DM. Work-up of the functional adrenal mass. Curr Urol Rep 2005;6:63-71.
3. Chumaung C. Adrenal cortical carcinoma: A case report. Thai J Urol 1983;5:27-9.
4. Sriphatphiriyakun P. Adrenal cortical carcinoma: A case report. Med J 2002; 16: 103-5.
5. Churesigaew S. Adrenocortical carcinoma in a 5-year-old child. Bull Dept Med Serv 1990; 12: 173-5.
6. Jaruratanasirikul S, Patarapinyokul S, Mitranun W. Androgen-producing
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7. Pusantisampan T, Sangkhathat S, Kayasut K, Kanngurn S, Jaruratanasirikul S, Chotsampancharoen T, et al. Cushing’s syndrome in an infant secondary to malignant adrenocortical tumors with somatic mutation of beta-catenin. Pediatr Dev Pathol 2010; 13:238-42.
8. Siriwong S, Shuangshoti S, Saritsiri S, Pak-art P, Khovidhunkit W, Snabboon T. Functioning adrenocortical carcinoma with superior vena cava and upper airway obstructions. J Med Assoc Thai 2006;89: 1511-5.
9. Muttarak M, Chotirosniramit A, Unsrisong K, Na Chiangmai W. Adrenal carcinoma. Biomed Imaging Interv J 2006;2:e9.
10. Tupikowski W, Bednarek-Tupikowska G, Florczak A. Adrenocortical carcinoma and its treatment. Postepy Hig Med Dosw (Online) 2004;58:27-36.
11. Iihara M, Obara T. Diagnosis and treatment for adrenocortical carcinoma. Gan To Kagaku Ryoho 2004;31:342-5.
12. Conzo G, Grillo M, Campione M, Amore A, Di Marzo M, Santini L. The role of surgery in the treatment of adrenocortical carcinoma. Ann Ital Chir 2002;73:619-22.
13. Bourdeau I, Mackenzie-Feder J, Lacroix A. Recent advances in adrenocortical carcinoma in adults. Curr Opin Endocrinol Diabetes Obes 2013;20: 192-7.
14. 4. Yoshimoto K, Iwahana H, Itakura M. Relatively good prognosis of multiple endocrine neoplasia type 2B in Japanese: Review of cases in Japan and analysis of genetic changes in tumors. Endocr J 1993;40:649-57.
How to site this article: Joob B, Wiwanitkit V. Adrenal cortical carcinoma in Thailand: A review on the previous reported case. Indian J Cancer 2013;50:327-9.
Source of Support: Nil, Conflict of Interest: None declared.
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