The Adrenal Oncocytoma: About Two New Cases  

Mahdi  Ali Haddam1 , Djamila  Meskine1 , Farida  Chentli2 , Samia  Haddam3 , Rebiha  Baba  Ahmed3 , Soumeya  Fedala2
1. Department of endocrinology bologhine hospital, Algeria
2. Department of endocrinology Bab el oued hospital, Algeria
3. Department of pathology Bab el oued hospital, Algeria

Author    Correspondence author
International Journal of Clinical Case Reports, 2015, Vol. 5, No. 27   doi: 10.5376/ijccr.2015.05.0027
Received: 24 Apr., 2015    Accepted: 18 Jul., 2015    Published: 03 Jul., 2015
© 2015 BioPublisher Publishing Platform
This is an open access article published under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
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Mahdi Ali Haddam et al., 2015, The Adrenal Oncocytoma: About Two New Cases, International Journal of Clinical Case Reports, 5(27) 1-5 (doi: 10.5376/ijccr.2015.05.0027)

Abstract

The adrenal oncocytoma is an exceptional tumor. Only hundred fifty cases have been descripted to date. We report two new observations of a woman 47 years old and a man 58 years old hospitalized for exploration of adrenal mass discovered incidentally (in the woman, 10 cm) and during the exploration of pain in the right hypochondria (in the man, 7 cm). Both tumors were not secretant.

Due to the tumor size, adrenalectomy is performed for both. Histological and immunohistochemical study were in favor of oncocytoma.

Keywords
Oncocytic cells; Adrenal; Adrenal tumor; Histology
The adrenal oncocytoma is extremely rare. Since the first description in 1986 (Smirnova and Mikhailov, 1986), approximately 159 cases have been reported in the literature. Discovered incidentally in general, it is in most cases not functional and Benin. However, recent data indicate that about 20% of the adrenocortical oncocytomas demonstrate some elements of malignancy (Bisceglia et al., 2004) and 10-20% of them appear to affect hormone production in the adrenal gland particularly in children (Mearini et al., 2013).
We report two new cases of adrenal oncocytoma observations.
Observation 1
A man 58 years old was admitted to the department of endocrinology for exploration of an adrenal mass discovered during the exploration of pain in the right hypochondria to six months.
Physical examination had no abnormalities including no signs for an adrenergic or adrenocortical hypersecretion. Indeed, clinical examination did not reveal hypertension, headaches, palpitations or inappropriate perspiration and signs for a cushing syndrome. Adrenocortical hormones levels were normal (Table 1).


Table 1 Biological results

Radiologically, ultrasound and abdominal CT scan were objectified homogeneous adrenal tumor measuring 7 cm long axis. It was hypodense and was weakly enhanced after iodinated injection (Figure 1 A and B).


Figure 1 A and B abdominal CT scan objectified homogeneous a right adrenal tumor measuring 7 cm long axis. The mass has regular margins and a well circumscribed capsule, it was weakly enhanced after iodinated injection

This mass repressed the inferior vena cava (IVC) to retrohepatic towards front and right kidney down, but there is no evidence of infiltration of the neoplasm to periadrenal tissues with no lymphadenopathy or any other intra-abdominal neoplastic sites.
Due to the size of the tumor, right adrenalectomy by lumbar way is decided. No major bleeding or hemodynamic instability events were encountered perioperatively. After an uneventful postoperative recovery period of 10 days the patient was discharged in good medical condition.
Microscopically, the tumor was composed of large cells containing eosinophil granules without evidence of vascular or lymph node involvement (Figure 2).
In immunohistochemistry, the tumor cells expressing the anti-cytokeratin antibody and mitochondria; they were negative for synaptophysin, chromogranin, marker Melan-A, carcinoembryonic antigen (CEA), alpha-inhibin. Ki-67 had low labeling/proliferation index. The diagnosis was in favor of oncocytic adrenal tumor (Figure 2).


Figure 2 A: Necrosis; B: Immunohistochemistry: Expression synaptophysin synaptophysin: cytoplasmic expression of tumor cells; C: HE Aspect oncocytic, eosinophilic cytoplasm; D: Immunohistochemistry: bolus vascular

In the absence of major or minor criteria (mitotic activity, necrosis, invasion). Given the tumor size and necrosis (Two minor criteria) oncocytoma is classified indeterminate prognosis (borderline malignancy) when using Bisceglia-Weiss scoring system (Bisceglia et al., 2004). No supplementary treatment was indicated.
Clinical (Follow-up with periods of 3 months) and radiological controls (computed tomography with periods of 6 months) were favorable with current monitoring of 3 years.
Observation 2
A woman 48 years old was admitted to department of endocrinology to management of a left adrenal incidentalomas revealed scan performed for pelvic pain. Clinical examination did not found abnormalities including no signs for an adrenergic or adrenocortical hypersecretion like hypertension, headaches, palpitations or inappropriate perspiration and signs for a Cushing syndrome. Adrenocortical hormones levels were normal (Table 1).
Ultrasonography and abdominal computed tomography objective voluminous left adrenal tumor measuring 10 cm long axis. This tumor had hypodense at the CT, and took the contrast heterogeneously (Figure 3). The mass has regular margins and a well circumscribed capsule.


Figure 3 Left adrenal tumor measuring 10 cm long axis. This tumor was hypodense at the CT, and took the contrast heterogeneously. Total Adrenalectomy is performed

No major events were encountered perioperatively. After an uneventful postoperative recovery period of 12 days the patient was discharged in good medical condition.

Macroscopic examination of the surgical pieces showed the presence of a homogeneous encapsulated mass of yellowish color.
Microscopically, the tumor was composed of polygonal oncocytes with abundant granular, eosinophilic cytoplasm. Cells were arranged in a diffuse pattern. No mitotic figures, neither lymphovascular invasion or capsular was seen. The tumor was immune-reactive for cytokeratin and mitochondrial antibodies were negative for chromogranin, synaptophysin, carcinoembryonic antigen (CEA), and alpha-inhibin. Ki-67 had low labeling/proliferation index. The diagnosis was in favor of oncocytic tumor of the adrenal.
Given the tumor size and necrosis (Two minor criteria) oncocytoma is classified indeterminate prognosis (borderline malignancy) when using Bisceglia-Weiss scoring system (Bisceglia et al., 2004).
Clinical (Follow-up with periods of 3 months) and radiological controls (CT with periods of 6 months were favorable with current monitoring of 2 years.
Discussion
The definition of oncocytoma was given by Jaffe in 1932 to designate tumors composed entirely or almost oncocytic cells (Jaffe, 1932). These tumors are observed in thyroid, parathyroid, kidney and salivary glands. The adrenal localization is rare (Tallini, 1998). This definition excludes adrenal tumors containing some oncocytic cells within and compact spongiocytaires cells. Since 1986, approximately 159 cases have been described in the literature (Bisceglia et al., 2004; Song et al., 2004).
The average age of onset is 47.6 years but their occurrence in all ages has been described. It occurs more frequently in females (Corsi et al., 2002).
The adrenal oncocytoma is often a non-functional tumor; among the reported cases, 8 cases were functional; it was virilization, Cushing's syndrome, bilateral gynecomastia (in two cases) and pseudo-puberty (Poretti et al., 2003; Xiao et al., 2005). Therefore, a careful biochemical and hormonal evaluation should be performed to eliminate the presence of functioning tumor (Mearini et al., 2013). Any finding of hormonal hypersecretion is an indication for surgical therapy.
The tumor frequently locates at the right side (2/1). Its average size is 8.4 cm in major cases with extremes from 3 to 15 cm (Corsi et al., 2002; Kitching and Harach, 1999).
They consist exclusively or almost of oncocytes cells which are large with many eosinophilic granules corresponding to mitochondria by electron microscopy. Immunohistochemistry shows typically a diffuse and intense immunostaining for anti-mitochondrial antibodies and frequently for vimentin and keratin (El-Naggar et al., 1991).
Histologically, it is a diffuse proliferation and more rarely clusters cell. The tumor cells are large and often include an eosinophilic granular cytoplasm. The kernel is often atypical pleomorphic and monstrous. Mitoses are rare and not atypical. The stroma is reduced and often hyalinized with myxoid and hemorrhagic areas. Tumor is surrounded by a thick fibrous capsule. There was no necrosis (El-Naggar et al., 1991; Hoang et al., 2002). Its confirmation by immunohistochemistry (using the mES-13 or SDHA/SDHB stainings) or electron microscopy is of paramount importante. In the last decade, it was recognized by gross and micro- scopic examination that.
In the majority of cases, the oncocytoma is Benin and non-functional. However, malignant cases were reported in literature. The first case was described in 1991. It has local invasion and distant metastases. Nine cases of adrenal oncocytic carcinoma oncocytic were described few years after (Doublet, 2007; Juliano et al., 2008).
The biologic behaviour of adrenal oncocytomas could not be correctly predicted by the widely accepted Weiss and Weiss modified systems. Indeed, of the nine criteria used to classify the tumour as benign or malignant, three are almost always present in oncocytic neoplasms (high nuclear grade, clear cell composition ≤25 % and diffuse architectural pattern), which would categorize the vast majority of AOs as malignant lesions (Weiss score ≥3 criteria) (Ferlicot et al.,2005).
Bisceglia et al. (2004) proposed a review of the common Weiss criteria. The morphologic parameters of the Weiss system were reviewed in the context of their possible application to the oncocytic tumor variant. Proposed major (high mitotic rate, atypical mitoses and venous invasion) and minor (large size and huge weight, necrosis, capsular invasion and sinusoidal invasion) criteria in distinguishing malignant tumors have been analyzed by the authors. Defining criteria (predominantly cells with eosinophilic and granular cytoplasm, a high nuclear grade and a diffuse architectural pattern) in common with all types of oncocytic tumors have been outlined. The presence of 1 major criterion indicating malignancy, 1-4 minor criteria indicating uncertain malignant potential (borderline) and the absence of all major and minor criteria indicative of benign mass (Ferlicot et al., 2005).
Hoang et al. (2002) suggested that p53 tumor suppressor gene may play a role in malignant transformation of some oncocytic adrenal cortical tumors. However, the role of either Ki-67 or p53 as biomarkers likely remains uncertain due to the availability of so few of these tumors (Hoang et al., 2002).
There are no specific criteria on both CT scan and MRI with chemical shift subtraction for adrenal oncocytic neoplasm and its malignant variant (Friedrich-Rust et al., 2011).
Treatmentof adrenal oncocytoma is surgical, with the most complete resection. Adrenalectomy is the standard, since adrenal oncocytic neoplasm usually presented as large adrenal mass (more than 6 cm). Recent advances in endoscopic techniques have allowed the performance of an increasing number of laparoscopic adrenalectomies. This approach should be considered when the preoperative CT and MRI findings reveal a well-encapsulated tumor, with no evidence of invasion into surrounding tissue and no regional adenopathy (Pazooki and Vaziri, 2008; Tian et al., 2008; Eldahshan et al., 2008).
Because the likehood of adrenal cancer has been revealed to increase in adrenal lesions with a diameter greater than 6 cm, a recent National Institutes at Health State-of-the-Science Statement recommended that nonsecretory adrenal incidentalomas larger than 6 cm or with suspicious features of malignancy on imaging procedures should be treated using open adrenalectomy because of the increased prevalence of malignancy (Tabarin et al., 1997).
Furthermore, when laparoscopic surgery is performed for adrenal tumors larger than 6 cm or for adrenal tumors of certain malignant potential, it should be carried out by only a highly experienced laparoscopic surgeon because there is a high risk of surgical complications and incomplete resections. Also, laparoscopic surgery increases the risk of tumor recurrence.
After surgery, adrenal oncocytic neoplasm can be assessed conservatively in the absence of mitotic activity, necrosis, or invasion.
Actually, there are no recommendations for the follow-up of benign adrenal oncocytic neoplasms.
When the diagnosis of adrenal oncocytoma with uncertain malignant potential is raised because of histological criteria (as patient 2), and it should be closely monitored. Follow-up of metastasis and recurrences can be CT or MRI with periods of 3-6 months. MRI has not an advantage on CT. Preventive treatment with mitotane was considered by some authors.
The oncocytic variant of adrenocortical carcinoma has a poor prognosis, with a postoperative recurrence rate of 75%, and a tumour-related mortality of 40% in a small group of 24 patients with a median follow-up of 21 months (Wong et al., 2011). An adjuvant or palliative chemotherapy with mitotane can be administered in patients with adrenocortical carcinomas with beneficial effects, but there is no evidence of efficacy when applied to oncocytic variant.
The role of adjuvant therapy remains to be determined and will be discussed in the presence of malignancy. The literature reports a favorable response to adjuvant radiotherapy. As for adrenal carcinoma, an adjuvant or palliative chemotherapy with mitotane can be administered but there is no evidence of efficacy when applied to oncocytic variant (Tallini, 1998; Icard et al., 2001; Gargah et al., 2008).
Conclusion
The adrenal oncocytoma is extremely rare tumor and preoperative diagnosis is difficult. They only the histogical and immunohistochemical examination allows to pose the diagnosis. It needs careful evaluation and surgical treatment, usually presents as an incidental, large adrenal mass; it is mostly benign tumor and the prognostic is usually favorable. Long-term follow-up is necessary.
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