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Ectopic Functioning Adrenocortical Oncocytic Adenoma (Oncocytoma) with Myelolipoma Causing Virilization  [PDF]
Lea F. Surrey,Ashesh A. Thaker,Paul J. Zhang,Giorgos Karakousis,Michael D. Feldman
Case Reports in Pathology , 2012, DOI: 10.1155/2012/326418
Abstract: Functioning adrenal adenomas are well-described entities that can rarely occur outside the adrenal gland in the ectopic adrenal tissue. Similarly, myelolipoma is an another benign lesion of the adrenal tissue which can rarely occur outside the adrenal gland. We report the first case of a testosterone producing an extra-adrenal adrenocortical oncocytoma accompanied by a myelolipoma. The patient presented with virilization and elevated androgen levels. Imaging revealed a retroperitoneal mass, which histologically consisted of oncocytes and intermingled myelolipoma. Postoperative androgen levels decreased to normal. The tumor cells were strongly positive for inhibin and Melan-A, supporting the adrenal origin. This case demonstrates a diagnostic challenge in which correlation with histology, immunohistochemistry, and serum endocrine studies led to the final diagnosis. 1. Introduction Adrenal cortical adenomas are common lesions of the adrenal cortex which may produce adrenal cortical hormones. These lesions are usually detected due to the clinical side effects of hormone production or found incidentally on imaging, especially in the case of nonfunctioning adenomas. Oncocytic adenomas in the adrenal gland are rare (approximately 132 reported cases) and have been described as adrenal oncocytomas [1]. Rarely adrenal cortical adenomas have been found outside the adrenal gland, in sites presumed to arise from ectopic adrenal tissue [2, 3]. However, adrenal oncocytoma has not been reported outside the adrenal gland. Myelolipoma is a benign proliferation of adipose tissue and trilineage hematopoietic elements typically found within the adrenal tissue. Myelolipomata have been associated with adrenal cortical adenomas both functioning and nonfunctioning but there are a few cases reported that occur outside the adrenal gland [4, 5]. We report a rare case of ectopic adrenocortical oncocytic adenoma (oncocytoma) accompanied by myelolipoma producing testosterone leading to virilization. To the best of our knowledge, there is no such case reported to date in the English literature. 2. Case Presentation A 55-year-old female presented with hirsutism which included thinning scalp hair and increased hair over her back and arms. She had no other complaints and denied changes in weight, dizziness, urinary symptoms, changes in bowel habits, and skin changes. Her past medical history was notable for hypertension controlled with hydrochlorothiazide and ramipril as well as hypothyroidism controlled with levothyroxine. Her surgical history included two cesarean sections, partial
Functioning adrenocortical oncocytoma: A case report and review of literature  [cached]
Geramizadeh Bita,Norouzzadeh Behdokht,Bolandparvaz Shahram,Sefidbakht Sepideh
Indian Journal of Pathology and Microbiology , 2008,
Abstract: Adrenocortical oncocytoma is very rare. Less than five functioning types of them are reported and most of the reported cases are incidentally found. We herein report a case of functioning adrenocortical oncocytoma of the left adrenal cortex in a young woman.
Aldosterone-secreting adrenocortical adenoma in children  [cached]
Gupta Vipul,Sharma Shyam
Journal of Indian Association of Pediatric Surgeons , 2007,
Abstract: Aldosterone-secreting adrenocortical adenoma is an unusual cause of secondary hypertension in childhood. We describe an eight year-old female child who presented clinically with weakness of lower limbs, headache and hypertension. Thorough clinical, laboratory and radiological evaluations confirmed the diagnosis of hyperaldosteronism due to a left adrenocortical adenoma. Left adrenalectomy after preoperative stabilization resulted in satisfactory recovery. Histopathological examination confirmed the diagnosis of an aldosteronoma. We review our experience with this rare entity and the pertinent literature.
Ectopic Functioning Adrenocortical Oncocytic Adenoma (Oncocytoma) with Myelolipoma Causing Virilization
Lea F. Surrey,Ashesh A. Thaker,Paul J. Zhang,Giorgos Karakousis,Michael D. Feldman
Case Reports in Pathology , 2012, DOI: 10.1155/2012/326418
Abstract: Functioning adrenal adenomas are well-described entities that can rarely occur outside the adrenal gland in the ectopic adrenal tissue. Similarly, myelolipoma is an another benign lesion of the adrenal tissue which can rarely occur outside the adrenal gland. We report the first case of a testosterone producing an extra-adrenal adrenocortical oncocytoma accompanied by a myelolipoma. The patient presented with virilization and elevated androgen levels. Imaging revealed a retroperitoneal mass, which histologically consisted of oncocytes and intermingled myelolipoma. Postoperative androgen levels decreased to normal. The tumor cells were strongly positive for inhibin and Melan-A, supporting the adrenal origin. This case demonstrates a diagnostic challenge in which correlation with histology, immunohistochemistry, and serum endocrine studies led to the final diagnosis.
Adrenocortical Secreting Mass in a Patient with Gardner's Syndrome: A Case Report
Nabila Mejdoub Rekik,Sourour Ben Salah,Nozha Kallel,Mahdi Kamoun,Nadia Charfi,Mohamed Abid
Case Reports in Medicine , 2010, DOI: 10.1155/2010/682081
Abstract: Gardner's syndrome (GS) is a dysplasia characterized by neoformations of the intestine, soft tissue, and osseous tissue. Endocrine neoplasms have occasionally been reported in association with GS. Adrenal masses in GS are rare, and few have displayed clinical manifestations. In the current paper, The authors report a 37-year-old male patient with GS including familial adenomatous polyposis (FAP) and mandible osteoma who presented with an incidental adrenal mass. Computerized tomography adrenal scan identified bilateral masses. Functional analyses showed a hormonal secretion pattern consistent with pre-Cushing's syndrome. Other extraintestinal manifestations were hypertrophy of the pigmented layer of the retina and histiocytofibroma in the right leg. This paper describes a rare association of adrenocortical secreting mass in an old male patient with Gardner syndrome.
The comparison of free androgen index and serum free testosterone levels in women with hirsutism or polycystic ovary syndrome  [PDF]
Oya Güng?r,G?nül Erden,Ceylan Bal,Nihal U?uz1
Journal of Clinical and Experimental Investigations , 2011,
Abstract: In many laboratories free testosterone can not be measured, so that free androgen index is suggested instead. The aim of this study was to compare free androgen index and serum free testosterone levels measured by radioimmunoassay in women with hirsutism or polycystic ovary syndrome.Materials and methods: Totally 94 women referred to the polyclinics of Ankara Numune Hospital were retrospectively included. Three patient groups were composed; 55 of hirsutism, 20 of polycystic ovary syndrome and 19 of both hirsutism and polycystic ovary syndrome. Total testosterone and sex hormone binding globuline levels were measured by chemiluminescence method and free testosterone levels were measured by radioimmunoassay. Free androgen index was calculated from total testosterone and sex hormone binding globuline.Results: There was a significant positive correlation between free testosterone and free androgen index in patients with hirsutism, in patients with polycystic ovary syndrome, in patients with hirsutism and polycystic ovary syndrome, and in total patient group [r(hirsutism)=0,597, r(PCOS)=0,617, r(hirsutism and PCOS)=0,779, r(total patient group)=0,649, P<0,01].Receiver operating characteristics curves were drawn to assess the diagnostic power of parameters for all patient groups [For hirsutism (n=55) auROC (FT)=0,431 auROC (FAI)=0,485] [For PCOS (n=20) auROC (FT)=0,431 auROC (FAI)=0,359] [For hirsutism and PCOS (n=19) auROC (FT)=0,676 auROC (FAI)=0,669]. In our study, free testosterone and free androgen index were found useful to diagnose ‘hirsutism and polycystic ovary syndrome’ but not others.Conclusion: Free androgen index can be used instead of free testosterone in hirsutism and polycystic ovary syndrome for diagnosis. J Clin Exp Invest 2011;2(2):152-6
Pure Androgen-Secreting Adrenal Adenoma Associated with Resistant Hypertension  [PDF]
René Rodríguez-Gutiérrez,Mario Arturo Bautista-Medina,Ana Eugenia Teniente-Sanchez,Maria Azucena Zapata-Rivera,Juan Montes-Villarreal
Case Reports in Endocrinology , 2013, DOI: 10.1155/2013/356086
Abstract: Pure androgen-secreting adrenal adenoma is very rare, and its diagnosis remains a clinical challenge. Its association with resistant hypertension is uncommon and not well understood. We present an 18-year-old female with a 10-year history of hirsutism that was accidentally diagnosed with an adrenal mass during the evaluation of a hypertensive crisis. She had a long-standing history of hirsutism, clitorimegaly, deepening of the voice, and primary amenorrhea. She was phenotypically and socially a male. FSH, LH, prolactin, estradiol, 17-hydroxyprogesterone, and progesterone were normal. Total testosterone and DHEA-S were elevated. Cushing syndrome, primary aldosteronism, pheochromocytoma, and nonclassic congenital adrenal hyperplasia were ruled out. She underwent adrenalectomy and pathology reported an adenoma. At 2-month followup, hirsutism and virilizing symptoms clearly improved and blood pressure normalized without antihypertensive medications, current literature of this unusual illness and it association with hypertension is presented and discussed. 1. Introduction Benign and malignant tumors of the adrenal gland might be functional or silent. The majority of these tumors are benign, nonfunctioning adenomas that are incidentally discovered on abdominal image studies. Others are functional adenomas able to secrete cortisol, aldosterone, or less commonly androgens or estrogens [1]. Pure androgen-secreting adrenal tumors are very unusual, and their diagnosis represents a clinical challenge. Hirsutism and virilization syndrome, characterized by clitorimegaly, male pattern baldness, and deepening of the voice along with menstrual irregularities are the most common findings [2–4]. Resistant hypertension is also a clinical feature in these cases, and it is defined as arterial hypertension above goals in spite of the concurrent use of three different antihypertensive drugs of different classes, including a diuretic [5]. The association of pure androgen-secreting adrenal tumors with hypertension has exceptionally been reported. Most of the cases have been carcinomas and mixed hormone-secreting tumors [6–8]. Even though it is well known that testosterone can increase blood pressure and epidemiological studies have demonstrated a higher blood pressure in males than females, most studies agree that androgens are only an aggravating factor rather than the exclusive cause of resistant hypertension [9, 10]. Herein, we present the case of an 18-year-old female with a 10-year history of hirsutism, virilization, and primary amenorrhea associated to an incidental
Giant adrenal myelolipoma associated with 21-hydroxylase deficiency: unusual association mimicking an androgen-secreting adrenocortical carcinoma
Mermejo, Lívia Mara;Elias Junior, Jorge;Saggioro, Fabiano Pinto;Tucci Junior, Silvio;Castro, Margaret de;Moreira, Ayrton Custódio;Elias, Paula C. Lamparelli;
Arquivos Brasileiros de Endocrinologia & Metabologia , 2010, DOI: 10.1590/S0004-27302010000400012
Abstract: the objective of this study was to describe a case of giant myelolipoma associated with undiagnosed congenital adrenal hyperplasia (cah) due to 21-hydroxylase (21oh) deficiency. five seven year-old male patient referred with abdominal ultrasound revealing a left adrenal mass. biochemical investigation revealed hyperandrogenism and imaging exams characterized a large heterogeneous left adrenal mass with interweaving free fat tissue, compatible with the diagnosis of myelolipoma, and a 1.5 cm nodule in the right adrenal gland. biochemical correlation has brought concerns about differential diagnosis with adrenocortical carcinoma, and surgical excision of the left adrenal mass was indicated. anatomopathologic findings revealed a myelolipoma and multinodular hyperplasic adrenocortex. further investigation resulted in the diagnosis of cah due to 21oh deficiency. concluded that cah has been shown to be associated with adrenocortical tumors. although rare, myelolipoma associated with cah should be included in the differential diagnosis of adrenal gland masses. moreover, cah should always be ruled out in incidentally detected adrenal masses to avoid unnecessary surgical procedures.
Oncocytoma of the caruncle  [cached]
Mohan Ravindra,Biswas Jyotirmay,Krishnakumar S
Indian Journal of Ophthalmology , 2002,
Abstract: A rare case of oncocytoma of the caruncle in a 75-year-old female is reported. Clinical differential diagnosis and histopathologic features are discussed
A rare case of an aldosterone secreting metastatic adrenocortical carcinoma and papillary thyroid carcinoma in a 31-year-old male  [cached]
Stephen M. Wanta,Marina Basina,Steven D. Chang,Daniel T. Chang
Rare Tumors , 2011, DOI: 10.4081/rt.2011.e45
Abstract: We report a rare synchronous presentation of adrenocortical carcinoma (ACC) and papillary thyroid carcinoma (PTC). A 31-year-old male first presented with a large left adrenal mass that was identified during the workup for refractory hypertension due to hyperaldosteronism. The mass was removed surgically with pathology showing ACC. The patient was then treated with adjuvant radiation therapy and mitotane chemotherapy. Four months post ACC resection, metastatic ACC to the right upper lung and PTC in the left lobe of the thyroid were found in surveillance imaging. He subsequently developed pulmonary, contralateral adrenal and brain metastases from his ACC. Li Fraumeni syndrome and Multiple Endocrine Neoplasia Type I (MEN I) were considered, but testing of both P53 and menin genes showed no mutation. We also performed a review of the literature and found three similar cases, however gene mutation analysis was not performed.
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