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1. Shin YM: Hepatic adrenal rest tumor mimicking hepatocellular carcinoma. Korean J Hepatol; 2010 Sep;16(3):338-41
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  • [Title] Hepatic adrenal rest tumor mimicking hepatocellular carcinoma.
  • [MeSH-major] Adrenal Rest Tumor / radiography. Liver Neoplasms / radiography

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  • (PMID = 20924220.001).
  • [ISSN] 1738-222X
  • [Journal-full-title] The Korean journal of hepatology
  • [ISO-abbreviation] Korean J Hepatol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Korea (South)
  • [Other-IDs] NLM/ PMC3304595
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2. Claahsen-van der Grinten HL, Duthoi K, Otten BJ, d'Ancona FC, Hulsbergen-vd Kaa CA, Hermus AR: An adrenal rest tumour in the perirenal region in a patient with congenital adrenal hyperplasia due to congenital 3beta-hydroxysteroid dehydrogenase deficiency. Eur J Endocrinol; 2008 Oct;159(4):489-91
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  • [Title] An adrenal rest tumour in the perirenal region in a patient with congenital adrenal hyperplasia due to congenital 3beta-hydroxysteroid dehydrogenase deficiency.
  • BACKGROUND: In contrast to the high incidence of testicular adrenal rest tumours (TART) in adult male patients with congenital adrenal hyperplasia (CAH), ovarian adrenal rest tumours in female CAH patients are very rare and other locations of adrenal rest tumours have never been reported.
  • Here, we report on an adult patient with CAH due to 3beta-hydroxysteroid dehydrogenase (HSD) deficiency with bilateral TART and additionally a large perirenal adrenal rest tumour.
  • Intensifying glucocorticoid medication did not result in decrease in POMC concentrations and shrinkage of size of the tumours.
  • Histological investigation after removal of the tumour showed sheets of large polygonal cells with abundant eosinophilic cytoplasm, separated by dense fibrous tissue strands.
  • The histological and immunohistochemical profile resulted in a diagnosis of an adrenal rest tumour.
  • CONCLUSION: In adult CAH patients, ectopic adrenal rest tumours can be present outside the testicular region.
  • Further investigations are necessary to determine whether regularly screening for these tumours is useful.


3. Claahsen-van der Grinten HL, Sweep FC, Blickman JG, Hermus AR, Otten BJ: Prevalence of testicular adrenal rest tumours in male children with congenital adrenal hyperplasia due to 21-hydroxylase deficiency. Eur J Endocrinol; 2007 Sep;157(3):339-44
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  • [Title] Prevalence of testicular adrenal rest tumours in male children with congenital adrenal hyperplasia due to 21-hydroxylase deficiency.
  • OBJECTIVE: Testicular adrenal rest tumours (TART) are a well-known complication in adult male patients with congenital adrenal hyperplasia (CAH), with a reported prevalence of up to 94%.
  • In adulthood, the tumours are associated with gonadal dysfunction most probably due to longstanding obstruction of the seminiferous tubules.
  • In two of them, bilateral tumours were found.
  • Mean tumour size was 4.1 mm (range 2-8 mm).
  • In none of the patients were the tumours palpable.


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4. Fernandes VO, Barros AI, Quidute AR, Montenegro AP, Fontenele EG, Sales AP, Montenegro RM, Ferreira FV, Montenegro RM Jr: [Bilateral testicular tumors caused by congenital adrenal rest hyperplasia]. Arq Bras Endocrinol Metabol; 2009 Nov;53(8):1052-8
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  • [Title] [Bilateral testicular tumors caused by congenital adrenal rest hyperplasia].
  • [Transliterated title] Tumores testiculares bilaterais por hiperplasia congênita de restos adrenais.
  • OBJECTIVES: Testicular tumors are a rare condition associated with congenital adrenal hyperplasia (CAH), originated from intratesticular adrenal rest tumors, and they are rarely associated with malignant tumors.
  • Their histological differentiation from Leydig-cell tumors is quite difficult, which would lead to inappropriate orchiectomies.
  • METHODS: Reported the case of 16-yr-old boy with previous diagnosis of CAH with bilateral testicular enlargement who was recommended to be submitted to a bilateral orchiectomy.
  • CONCLUSION: This case shows the importance of intratesticular adrenal rest tumors in the differential diagnosis of testicular tumors.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / pathology. Adrenal Rest Tumor / pathology. Leydig Cell Tumor / pathology. Testicular Neoplasms / pathology
  • [MeSH-minor] Adolescent. Antineoplastic Agents, Hormonal / therapeutic use. Diagnosis, Differential. Humans. Male. Prednisone / therapeutic use


5. Nomura K: [Adrenal rest tumor]. Nihon Rinsho; 2006 May 28;Suppl 1:717-9
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  • [Title] [Adrenal rest tumor].
  • [MeSH-major] Adrenal Rest Tumor
  • [MeSH-minor] 17-alpha-Hydroxyprogesterone / therapeutic use. Adrenal Hyperplasia, Congenital / complications. Adrenalectomy. Diagnosis, Differential. Diagnostic Imaging. Female. Humans. Kidney Neoplasms. Liver Neoplasms. Male. Ovarian Neoplasms. Prognosis. Testicular Neoplasms

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  • (PMID = 16776258.001).
  • [ISSN] 0047-1852
  • [Journal-full-title] Nihon rinsho. Japanese journal of clinical medicine
  • [ISO-abbreviation] Nippon Rinsho
  • [Language] jpn
  • [Publication-type] Journal Article; Review
  • [Publication-country] Japan
  • [Chemical-registry-number] 68-96-2 / 17-alpha-Hydroxyprogesterone
  • [Number-of-references] 15
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6. Stehr C, Velasco S, Velasco A, López M JM: [Is adrenal tumor size related to evolution time or does it represent a biological difference?]. Rev Med Chil; 2007 Dec;135(12):1526-9
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  • [Title] [Is adrenal tumor size related to evolution time or does it represent a biological difference?].
  • [Transliterated title] El tamaño de los tumores suprarrenales ¿está en relación al tiempo de evolución o expresa una diferencia biológica?
  • BACKGROUND: Adrenal tumor (AT) malignancy has been related to tumor size.
  • Since laparoscopic surgery is being used, smaller adrenal tumors are being excised.
  • AIM: To evaluate eventual clinical and histological differences between adrenal tumors smaller than 4 cm. and those larger than 6 cm.
  • PATIENTS AND METHODS: Retrospective review of pathological reports and clinical records of patients operated for adrenal tumors, dividing them in two groups.
  • Group 1 had 29 patients aged 52 +/- 13 years with AT < 4 cm operated during the period 2000-2005, and Group 2 was formed by 52 patients aged 46 +/-18 years with AT >6 cm operated between 1984-2005- Tumors between 4 and 6 cm were not included in the study to establish clear cut differences between groups.
  • RESULTS: Tumors were functional in 40 and 41% of cases in groups 1 and 2 respectively.
  • Fifty percent of functional tumors of group 1 were pheochromocytomas and the rest secreted aldosterone.
  • In group 2, 66% of tumors were pheochromocytomas and no aldosterone secreting tumors were found.
  • Fifty two and eight percent of tumors in Groups 1 and 2 were adenomas, respectively (p <0.001).
  • Nineteen tumors of group 2 were malignant, compared with one of group 1 (p <0.001).
  • CONCLUSIONS: The tumor size of adrenal cortical tumors may represent biological differences, suggesting two different tumor populations.
  • At time of diagnosis adrenal carcinomas are almost always larger than 6 cm.
  • [MeSH-major] Adrenal Gland Neoplasms / pathology. Adrenal Glands / pathology. Carcinoma / pathology
  • [MeSH-minor] Biomarkers, Tumor. Female. Humans. Hyperplasia. Incidental Findings. Male. Middle Aged. Neoplasm Invasiveness. Retrospective Studies. Time Factors

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  • (PMID = 18357352.001).
  • [ISSN] 0034-9887
  • [Journal-full-title] Revista médica de Chile
  • [ISO-abbreviation] Rev Med Chil
  • [Language] spa
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] Chile
  • [Chemical-registry-number] 0 / Biomarkers, Tumor
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7. Baba Y, Beppu T, Imai K, Masuda T, Iyama K, Sasano H, Baba H: A case of adrenal rest tumor of the liver: Radiological imaging and immunohistochemical study of steroidogenic enzymes. Hepatol Res; 2008;38(11):1154-8

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  • [Title] A case of adrenal rest tumor of the liver: Radiological imaging and immunohistochemical study of steroidogenic enzymes.
  • A case of adrenal rest tumor in the liver of a 67-year-old female was reported.
  • The tumor appeared on diagnostic imaging as a hypervascular and fat-containing mass at the periphery of right hepatic lobe and required differential diagnosis from hepatocellular carcinoma (HCC).
  • Pathological diagnosis of the intraoperative frozen section taken under laparoscopic liver biopsy could not exclude fat-containing HCC.
  • Histologically, the mass presented features similar to those of the adrenal cortex and was composed of pale cells.
  • Immunohistochemically, the mass expressed the adrenal 4-binding protein and several enzymes involved in the synthesis of adrenocortical steroids.
  • Based on these findings, the hepatic mass was finally diagnosed as an adrenal rest tumor of the liver.

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  • (PMID = 18564144.001).
  • [ISSN] 1386-6346
  • [Journal-full-title] Hepatology research : the official journal of the Japan Society of Hepatology
  • [ISO-abbreviation] Hepatol. Res.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Netherlands
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8. Claahsen-van der Grinten HL, Hermus AR, Otten BJ: Testicular adrenal rest tumours in congenital adrenal hyperplasia. Int J Pediatr Endocrinol; 2009;2009:624823

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  • [Title] Testicular adrenal rest tumours in congenital adrenal hyperplasia.
  • In adult patients with congenital adrenal hyperplasia (CAH), the presence of testicular adrenal rest tumours (TART) is an important complication leading to gonadal dysfunction and infertility.
  • These tumours can be already found in childhood and puberty.

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  • (PMID = 19956703.001).
  • [ISSN] 1687-9856
  • [Journal-full-title] International journal of pediatric endocrinology
  • [ISO-abbreviation] Int J Pediatr Endocrinol
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Egypt
  • [Other-IDs] NLM/ PMC2777016
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9. Reisch N, Scherr M, Flade L, Bidlingmaier M, Schwarz HP, Müller-Lisse U, Reincke M, Quinkler M, Beuschlein F: Total adrenal volume but not testicular adrenal rest tumor volume is associated with hormonal control in patients with 21-hydroxylase deficiency. J Clin Endocrinol Metab; 2010 May;95(5):2065-72
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  • [Title] Total adrenal volume but not testicular adrenal rest tumor volume is associated with hormonal control in patients with 21-hydroxylase deficiency.
  • CONTEXT: Patients with 21-hydroxylase deficiency (21-OHD) have been shown to develop adrenal adenomas and, in males, testicular adrenal rest tumors (TARTs) at a high percentage.
  • OBJECTIVE: The aim of this study was to evaluate the interrelation of adrenal masses and TARTs as well as factors stimulating tumor growth of orthotopic and ectopic adrenal tissue in 21-OHD.
  • Adrenal size and morphology was compared to 26 age-matched controls.
  • RESULTS: Combined adrenal volume of 21-OHD patients was significantly higher (median, 9.3 ml; range, 3.2-124.5 ml) in comparison to controls (median, 7.4 ml; range, 5.5-10.8 ml; P = 0.005).
  • None of the controls, but 42% of 21-OHD patients had an overall adrenal volume higher than 11 ml.
  • Total adrenal volume and tumor size but not TART volume correlated positively with current parameters of hormonal control (androstenedione, morning 17-OHP in serum, pregnanetriol in 24-h urine; P < 0.001 for each).
  • Baseline ACTH was independent of adrenal and TART volume.
  • There was no correlation of total adrenal or adrenal tumor size with TART volume.
  • CONCLUSION: These data provide indirect evidence that different factors regulate the growth of orthotopic adrenal tissue and ectopic adrenal remnants in TARTs.
  • [MeSH-major] Adrenal Glands / anatomy & histology. Adrenal Hyperplasia, Congenital / genetics. Adrenal Rest Tumor / genetics. Adrenal Rest Tumor / pathology. Steroid 21-Hydroxylase / genetics. Testicular Neoplasms / genetics

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  • MedlinePlus Health Information. consumer health - Testicular Cancer.
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  • (PMID = 20190160.001).
  • [ISSN] 1945-7197
  • [Journal-full-title] The Journal of clinical endocrinology and metabolism
  • [ISO-abbreviation] J. Clin. Endocrinol. Metab.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 7S5I7G3JQL / Dexamethasone; EC 1.14.99.10 / Steroid 21-Hydroxylase; WI4X0X7BPJ / Hydrocortisone
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10. Tiosano D, Vlodavsky E, Filmar S, Weiner Z, Goldsher D, Bar-Shalom R: Ovarian adrenal rest tumor in a congenital adrenal hyperplasia patient with adrenocorticotropin hypersecretion following adrenalectomy. Horm Res Paediatr; 2010;74(3):223-8
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  • [Title] Ovarian adrenal rest tumor in a congenital adrenal hyperplasia patient with adrenocorticotropin hypersecretion following adrenalectomy.
  • OBJECTIVE: Ovarian adrenal rest tumors (OARTs) are rare in contrast to testicular adrenal rest tumors.
  • We report a case of OART in a patient with congenital adrenal hyperplasia who developed Nelson's syndrome after bilateral adrenalectomy.
  • RESULTS: An 18-year-old female with congenital adrenal hyperplasia, who had undergone bilateral adrenalectomy at the age of 10 years, presented with severe hyperpigmentation and hirsutism.
  • Laparoscopic removal of the ovarian mass confirmed the diagnosis of OART.
  • CONCLUSIONS: Common to all documented cases of OART are sustained high ACTH levels that activate the adrenal anlagen tissue in the ovaries.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Rest Tumor / complications. Nelson Syndrome. Ovarian Neoplasms / complications


11. Bayhan GI, Cetinkaya S, Cinar HG, Aycan Z: Testicular adrenal rest tumor in a patient with 11beta-hydroxylase deficient congenital adrenal hyperplasia. J Pediatr Endocrinol Metab; 2010 Jul;23(7):729-32
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  • [Title] Testicular adrenal rest tumor in a patient with 11beta-hydroxylase deficient congenital adrenal hyperplasia.
  • In patients with congenital adrenal hyperplasia, testicular adrenal hyperplasia and tumors can develop.
  • 11beta-hydroxylase deficient congenital adrenal hyperplasia was diagnosed and hydrocortisone treatment was started.
  • Almost two years later the tumor completely disappeared with high dose steroid treatment.
  • In conclusion, the monitoring of congenital adrenal hyperplasia with ultrasonography is recommended, especially in puberty, because it is important that testicular adrenal rest hyperplasia should be determined before testicular adrenal rest tumors develop.
  • In this case we observed that small testicular adrenal rest tumors disappeared completely with high dose steroid treatment in nearly two years.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Rest Tumor / etiology. Testicular Neoplasms / etiology


12. Tiryaki T, Aycan Z, Hücümenoğlu S, Atayurt H: Testis sparing surgery for steroid unresponsive testicular tumors of the congenital adrenal hyperplasia. Pediatr Surg Int; 2005 Oct;21(10):853-5
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  • [Title] Testis sparing surgery for steroid unresponsive testicular tumors of the congenital adrenal hyperplasia.
  • The association between testicular tumors/nodules and congenital adrenal hyperplasia (CAH) has been recognized for many years.
  • Tumors are considered to be an aberrant adrenal tissue that has descended with the testes and has become hyperplastic due to ACTH stimulation.
  • We diagnosed steroid unresponsive testicular tumors of the CAH in two patients who were treated by testicular sparing tumor enucleation.
  • We believe that testis sparing surgery is the procedure of choice for all patients with testicular adrenal rest tumor, since it maximizes future fertility potential.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Rest Tumor / surgery. Testicular Neoplasms / surgery
  • [MeSH-minor] Adolescent. Child. Humans. Leydig Cell Tumor / diagnosis. Male


13. Basaran C, Karcaaltincaba M, Akata D, Karabulut N, Akinci D, Ozmen M, Akhan O: Fat-containing lesions of the liver: cross-sectional imaging findings with emphasis on MRI. AJR Am J Roentgenol; 2005 Apr;184(4):1103-10
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  • CONCLUSION: These lesions can contain macroscopic fat (i.e., angiomyolipoma, lipoma, liposarcoma, hydatid cyst, lipopeliosis, adrenal rest tumor, pseudolipoma, hepatic teratoma, pericaval fat, extramedullary hematopoiesis, and metastases) or intracellular lipid (i.e., focal steatosis, adenoma, focal nodular hyperplasia, regenerative nodules, and hepatocellular carcinoma).
  • CT, MRI, and sonographic findings of these lesions can help in characterization by allowing specific diagnosis or narrowing the differential diagnosis of liver lesions.
  • [MeSH-major] Fatty Liver / diagnosis. Liver Neoplasms / diagnosis. Magnetic Resonance Imaging. Neoplasms, Adipose Tissue / diagnosis
  • [MeSH-minor] Diagnosis, Differential. Humans

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  • (PMID = 15788580.001).
  • [ISSN] 0361-803X
  • [Journal-full-title] AJR. American journal of roentgenology
  • [ISO-abbreviation] AJR Am J Roentgenol
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 17
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14. Vajda Z, Borbély G, Jakab Z, Riesz P, Rácz K: [Bilateral testicular tumor in a young man with congenital 11β-hydroxylase deficiency]. Orv Hetil; 2010 Nov 7;151(45):1863-8
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  • [Title] [Bilateral testicular tumor in a young man with congenital 11β-hydroxylase deficiency].
  • Adrenal rest tumor presenting as palpable testicular mass has been well described in boys and adult males with congenital adrenal hyperplasia.
  • It develops most commonly in patients with 21- hydroxylase deficiency, but the entity may also occur in rare forms of congenital adrenal hyperplasia, including 11β-hydroxylase deficiency.
  • Because the management of testicular adrenal rest tumors is substantially different from that applied in benign and malignant testicular tumors, an accurate differentiation between these entities is particularly important.
  • Authors present the history of a young adult male with 11β-hydroxylase deficiency who developed adrenal rest tumors presenting as palpable bilateral testicular masses during treatment with glucocorticoids, then testicular masses showed a rapid regression after an adequate glucocorticoid treatment.
  • Considering lessons obtained from this case, authors review the pathomechanism, symptoms, as well as current diagnostic and treatment modalities of testicular adrenal rest tumors.
  • [MeSH-major] Adrenal Rest Tumor. Glucocorticoids / therapeutic use. Steroid 11-beta-Hydroxylase / metabolism. Testicular Neoplasms

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  • (PMID = 20980225.001).
  • [ISSN] 0030-6002
  • [Journal-full-title] Orvosi hetilap
  • [ISO-abbreviation] Orv Hetil
  • [Language] hun
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Hungary
  • [Chemical-registry-number] 0 / Glucocorticoids; EC 1.14.15.4 / Steroid 11-beta-Hydroxylase
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15. Prasad SR, Wang H, Rosas H, Menias CO, Narra VR, Middleton WD, Heiken JP: Fat-containing lesions of the liver: radiologic-pathologic correlation. Radiographics; 2005 Mar-Apr;25(2):321-31
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • Fat-containing tumors of the liver are a heterogeneous group of tumors with characteristic histologic features, variable biologic profiles, and variable imaging findings.
  • Benign liver lesions that contain fat include focal or geographic fatty change (steatosis), pseudolesions due to postoperative packing material (omentum), adenoma, focal nodular hyperplasia, lipoma, angiomyolipoma, cystic teratoma, hepatic adrenal rest tumor, pseudolipoma of the Glisson capsule, and xanthomatous lesions in Langerhans cell histiocytosis.
  • The imaging characteristics of a lesion coupled with the pattern of intratumoral fatty change are helpful in narrowing the differential diagnosis.
  • [MeSH-major] Fatty Liver / diagnosis. Liver Neoplasms / diagnosis

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  • [Copyright] (c) RSNA, 2005.
  • (PMID = 15798052.001).
  • [ISSN] 1527-1323
  • [Journal-full-title] Radiographics : a review publication of the Radiological Society of North America, Inc
  • [ISO-abbreviation] Radiographics
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 31
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16. Claahsen-van der Grinten HL, Otten BJ, Sweep FC, Span PN, Ross HA, Meuleman EJ, Hermus AR: Testicular tumors in patients with congenital adrenal hyperplasia due to 21-hydroxylase deficiency show functional features of adrenocortical tissue. J Clin Endocrinol Metab; 2007 Sep;92(9):3674-80
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Testicular tumors in patients with congenital adrenal hyperplasia due to 21-hydroxylase deficiency show functional features of adrenocortical tissue.
  • CONTEXT: In male patients with congenital adrenal hyperplasia (CAH), testicular adrenal rest tumors are frequently found that may interfere with gonadal function.
  • OBJECTIVE: Our objective was to determine steroid-producing features of testicular adrenal rest tumors.
  • PATIENTS: Eight adult CAH patients with bilateral testicular adrenal rest tumors were treated with testis-sparing surgery.
  • INTERVENTIONS: In all but one patient, spermatic veins were cannulated during surgery and blood samples collected to measure the adrenal-specific steroid 21-deoxycortisol (21DF) and 17-hydroxyprogesterone (17OHP) and androstenedione (A).
  • The same parameters were measured in simultaneously taken peripheral blood. mRNA concentrations of adrenal-specific enzymes CYP11B1 and CYP11B2 and ACTH and angiotensin II (AII) receptors were measured in tumor tissue.
  • MAIN OUTCOME MEASURES: Adrenal-specific steroids/enzymes were assessed.
  • CYP11B1, CYP11B2, and ACTH and AII receptor mRNAs were detected in all tumors with a strong correlation between ACTH receptor mRNA in tumors and 21DF (r = 0.85; P = 0.015), 17OHP (r = 1; P = 0.01) and A (r = 0.89; P = 0.007) concentrations in peripheral blood.
  • CONCLUSION: Testicular adrenal rest tumors produce adrenal-specific steroids and express adrenal-specific enzymes and ACTH and AII receptors, confirming the strong resemblance with adrenal tissue.
  • Because AII receptors are present in tumor tissue, it can be hypothesized that AII may be an additional factor responsible for testicular adrenal rest tumor growth.
  • [MeSH-major] Adrenal Cortex / physiopathology. Adrenal Hyperplasia, Congenital / complications. Adrenal Hyperplasia, Congenital / physiopathology. Adrenal Rest Tumor / etiology. Testicular Neoplasms / etiology


17. Fenichel P, Bstandig B, Roger C, Chevallier D, Michels JF, Sadoul JL, Hieronimus S, Brucker-Davis F: Unilateral testicular tumour associated to congenital adrenal hyperplasia: Failure of specific tumoral molecular markers to discriminate between adrenal rest and leydigioma. Ann Endocrinol (Paris); 2008 Nov;69(5):453-8
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Unilateral testicular tumour associated to congenital adrenal hyperplasia: Failure of specific tumoral molecular markers to discriminate between adrenal rest and leydigioma.
  • Testicular adrenal rest tumours are frequently associated with congenital adrenal hyperplasia (CAH).
  • These ACTH-dependent tumours cannot be easily distinguished histologically from Leydig-cell tumours.
  • We report the case of a 30-year-old man who was explored for infertility, azoospermia and unilateral testicular tumour.
  • High levels of 17-OH progesterone and ACTH, low cortisol and undetectable gonadotropins levels, associated to bilateral adrenal hyperplasia, led to the diagnosis of CAH by 21-OH deficiency with a composite heterozygoty.
  • The testicular tumour was first considered as adrenal rest.
  • However, histological analysis of this unilateral painful tumour showed a steroid-hormone-secreting cell proliferation with atypical and frequent mitosis.
  • To discriminate between a benign adrenal rest tumour and a possible malignant leydigioma, tumoral expression of specific gene products was analyzed by RT-PCR.
  • No 11-beta-hydroxylase nor ACTH receptor mRNAs could be found in the tumour, which did not behave like usual adrenal rest cells.
  • For this unilateral testicular tumour, the lack of adrenal-specific markers associated with a high rate of mitosis and pleiomorphism supported a leydigian origin with malignant potential.
  • However, lack of tumoral LH-R mRNA expression and a tumour-free 3-year follow-up led us to retain the diagnosis of adrenal rest tumour with loss of adrenal gene expression and progressive autonomous behaviour.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Hyperplasia, Congenital / diagnosis. Adrenal Rest Tumor / diagnosis. Leydig Cell Tumor / diagnosis. Testicular Neoplasms / complications. Testicular Neoplasms / diagnosis
  • [MeSH-minor] Adrenal Cortex Hormones / blood. Adrenal Cortex Hormones / genetics. Adult. Anti-Inflammatory Agents / therapeutic use. Azoospermia / etiology. Biomarkers, Tumor. Dexamethasone / therapeutic use. Diagnosis, Differential. Gonadal Steroid Hormones / blood. Gonadal Steroid Hormones / genetics. Gonadotropins / blood. Humans. Infertility, Male / etiology. Male. RNA, Messenger / biosynthesis. RNA, Messenger / genetics. Reverse Transcriptase Polymerase Chain Reaction. Testis / pathology


18. Claahsen-van der Grinten HL, Otten BJ, Stikkelbroeck MM, Sweep FC, Hermus AR: Testicular adrenal rest tumours in congenital adrenal hyperplasia. Best Pract Res Clin Endocrinol Metab; 2009 Apr;23(2):209-20
MedlinePlus Health Information. consumer health - Testicular Cancer.

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Testicular adrenal rest tumours in congenital adrenal hyperplasia.
  • In adult patients with congenital adrenal hyperplasia (CAH) the presence of testicular adrenal rest tumours (TART) is an important cause of gonadal dysfunction and infertility.
  • In the last decade several papers have focused on the origin and pathogenesis of these tumours.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Rest Tumor / etiology. Testicular Neoplasms / etiology


19. Mouritsen A, Jørgensen N, Main KM, Schwartz M, Juul A: Testicular adrenal rest tumours in boys, adolescents and adult men with congenital adrenal hyperplasia may be associated with the CYP21A2 mutation. Int J Androl; 2010 Jun 1;33(3):521-7
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Testicular adrenal rest tumours in boys, adolescents and adult men with congenital adrenal hyperplasia may be associated with the CYP21A2 mutation.
  • Congenital adrenal hyperplasia (CAH) is an autosomal recessive disorder with impaired function of the adrenal cortex caused by mutations in the CYP21A2 gene.
  • Testicular adrenal rest tumours (TART) may be prevalent in up to 95% of CAH adults and may already appear during childhood.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / genetics. Adrenal Hyperplasia, Congenital / physiopathology. Adrenal Rest Tumor / physiopathology. Mutation / physiology. Steroid 21-Hydroxylase / genetics

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  • (PMID = 19531083.001).
  • [ISSN] 1365-2605
  • [Journal-full-title] International journal of andrology
  • [ISO-abbreviation] Int. J. Androl.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] England
  • [Chemical-registry-number] EC 1.14.99.10 / CYP21A2 protein, human; EC 1.14.99.10 / Steroid 21-Hydroxylase
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20. Claahsen-van der Grinten HL, Otten BJ, Hermus AR, Sweep FC, Hulsbergen-van de Kaa CA: Testicular adrenal rest tumors in patients with congenital adrenal hyperplasia can cause severe testicular damage. Fertil Steril; 2008 Mar;89(3):597-601
MedlinePlus Health Information. consumer health - Testicular Cancer.

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Testicular adrenal rest tumors in patients with congenital adrenal hyperplasia can cause severe testicular damage.
  • OBJECTIVE: To evaluate the histological features of testicular tumors and residual testicular parenchyma in male patients with congenital adrenal hyperplasia (CAH) and longstanding bilateral testicular adrenal rest tumors (TART).
  • RESULT(S): All tumors had a similar histological appearance, with sheets of polygonal cells separated by dense fibrous tissue with focal lymphocytic infiltrates and without Reinke crystals.
  • CONCLUSION(S): Testicular adrenal rest tumors can lead to end-stage damage of testicular parenchyma, most probably as a result of longstanding obstruction of the seminiferous tubules.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Rest Tumor / etiology. Infertility, Male / etiology. Testicular Neoplasms / etiology. Testis / pathology


21. Knape P, Reisch N, Dörr HG, Reincke M, Lenk S, Quinkler M: [Testicular adrenal rest tumors (TART) in adult men with classic congenital adrenal hyperplasia (CAH)]. Urologe A; 2008 Dec;47(12):1596-7, 1599-602
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [Testicular adrenal rest tumors (TART) in adult men with classic congenital adrenal hyperplasia (CAH)].
  • BACKGROUND: Information about treatment of adult male patients with congenital adrenal hyperplasia (CAH) and testicular adrenal rest tumors (TART) is scarce.
  • RESULTS: The aims of treatment in adult male patients with CAH are prevention of adrenal crisis and TART, improvement of general well-being, good quality of life and sexual well-being, fertility, and prevention of side effects of gluco- and mineralocorticoid therapy.
  • [MeSH-major] Adrenal Rest Tumor / therapy. Testicular Neoplasms / therapy
  • [MeSH-minor] Adrenal Hyperplasia, Congenital / diagnosis. Adrenal Hyperplasia, Congenital / pathology. Adrenal Hyperplasia, Congenital / therapy. Glucocorticoids / administration & dosage. Humans. Infertility, Male / etiology. Male. Mineralocorticoids / administration & dosage. Orchiectomy. Prognosis. Testis / pathology

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  • (PMID = 18762902.001).
  • [ISSN] 0340-2592
  • [Journal-full-title] Der Urologe. Ausg. A
  • [ISO-abbreviation] Urologe A
  • [Language] ger
  • [Publication-type] English Abstract; Journal Article; Review
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Glucocorticoids; 0 / Mineralocorticoids
  • [Number-of-references] 46
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22. Poyrazoglu S, Saka N, Agayev A, Yekeler E: Prevalence of testicular microlithiasis in males with congenital adrenal hyperplasia and its association with testicular adrenal rest tumors. Horm Res Paediatr; 2010;73(6):443-8
MedlinePlus Health Information. consumer health - Testicular Disorders.

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Prevalence of testicular microlithiasis in males with congenital adrenal hyperplasia and its association with testicular adrenal rest tumors.
  • BACKGROUND: Testicular microlithiasis (TM) is characterized by calcium deposits within the seminiferous tubules and is associated with benign and malign conditions.
  • AIM: To determine TM prevalence in patients with congenital adrenal hyperplasia (CAH) and its association with testicular adrenal rest tumors (TART).
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Rest Tumor / epidemiology. Lithiasis / epidemiology. Testicular Diseases / epidemiology. Testicular Neoplasms / epidemiology. Testis / pathology


23. Mouritsen A, Juul A, Jørgensen N: Improvement of semen quality in an infertile man with 21-hydroxylase deficiency, suppressed serum gonadotropins and testicular adrenal rest tumours. Int J Androl; 2010 Jun 1;33(3):518-20
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Improvement of semen quality in an infertile man with 21-hydroxylase deficiency, suppressed serum gonadotropins and testicular adrenal rest tumours.
  • Here, we report improvement of semen quality in a 30-year-old man with congenital adrenal hyperplasia (CAH) because of 21-hydroxylase deficiency, bilateral testicular adrenal rest tumours (TART) and a 1.5-year infertility history.
  • His adrenal substitution therapy was changed from hydrocortisone 10 mg once daily to 10 mg three times daily in combination with dexamethasone, 0.1 mg once daily.
  • Upon this change, the testicular tumours showed regression and his sperm concentration increased from 0.1 to 98 mio/mL and total sperm count from 0.64 to 392 mio. sp., total number of motile spermatozoa increased from 0.23 to 258.72 mio.
  • [MeSH-major] Adrenal Rest Tumor / diagnosis. Adrenal Rest Tumor / etiology. Testicular Neoplasms / diagnosis. Testicular Neoplasms / etiology
  • [MeSH-minor] Adrenal Hyperplasia, Congenital / complications. Adrenal Hyperplasia, Congenital / diagnosis. Adrenal Hyperplasia, Congenital / pathology. Adult. Dexamethasone / administration & dosage. Fertility. Follow-Up Studies. Gonadotropins. Hormones. Humans. Hydrocortisone. Male. Metabolism, Inborn Errors / complications. Metabolism, Inborn Errors / pathology. Retrospective Studies. Semen Analysis. Sperm Count. Steroid 21-Hydroxylase / genetics. Testis / pathology. Time Factors. Treatment Outcome

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  • (PMID = 19281490.001).
  • [ISSN] 1365-2605
  • [Journal-full-title] International journal of andrology
  • [ISO-abbreviation] Int. J. Androl.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Gonadotropins; 0 / Hormones; 7S5I7G3JQL / Dexamethasone; EC 1.14.99.10 / Steroid 21-Hydroxylase; WI4X0X7BPJ / Hydrocortisone
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24. Sha YW, Song YQ, Zheng LK, Ma XM, Yang D, Huang P, Han B: [Congenital adrenal hyperplasia complicated by testicular adrenal rest tumors: one-case clinical analysis]. Zhonghua Nan Ke Xue; 2010 Sep;16(9):816-21
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [Congenital adrenal hyperplasia complicated by testicular adrenal rest tumors: one-case clinical analysis].
  • OBJECTIVE: To investigate the early diagnosis and treatment of congenital adrenal hyperplasia (CAH) complicated by testicular adrenal rest tumors (TART).
  • Imaging examinations revealed soft tissue density in the bilateral adrenal glands, especially on the right, and irregularly increased volume of the bilateral testes, particularly on the left, with heterogeneous signals and septas and surrounded by the fluid signals.
  • Immunohistochemical results were negative for testicular interstitial cell tumor.
  • The clinical signs of the patient were improved after 3 months of dexamethasone treatment, the hyperplastic nodules in the left testis decreased obviously and those in the right testis disappeared after 6 months, and the hyperplastic nodules in the adrenal glands vanished after 9 months.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Rest Tumor / complications

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  • (PMID = 21171267.001).
  • [ISSN] 1009-3591
  • [Journal-full-title] Zhonghua nan ke xue = National journal of andrology
  • [ISO-abbreviation] Zhonghua Nan Ke Xue
  • [Language] chi
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] China
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25. Martinez-Aguayo A, Rocha A, Rojas N, García C, Parra R, Lagos M, Valdivia L, Poggi H, Cattani A, Chilean Collaborative Testicular Adrenal Rest Tumor Study Group: Testicular adrenal rest tumors and Leydig and Sertoli cell function in boys with classical congenital adrenal hyperplasia. J Clin Endocrinol Metab; 2007 Dec;92(12):4583-9
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Testicular adrenal rest tumors and Leydig and Sertoli cell function in boys with classical congenital adrenal hyperplasia.
  • CONTEXT: Infertility observed in adult males with congenital adrenal hyperplasia (CAH) has been associated with testicular adrenal rest tumors (TART) that may originate during childhood.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / physiopathology. Adrenal Rest Tumor / physiopathology. Leydig Cells / physiology. Sertoli Cells / physiology. Testicular Neoplasms / physiopathology


26. Claahsen-van der Grinten HL, Otten BJ, Sweep FC, Hermus AR: Repeated successful induction of fertility after replacing hydrocortisone with dexamethasone in a patient with congenital adrenal hyperplasia and testicular adrenal rest tumors. Fertil Steril; 2007 Sep;88(3):705.e5-8
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Repeated successful induction of fertility after replacing hydrocortisone with dexamethasone in a patient with congenital adrenal hyperplasia and testicular adrenal rest tumors.
  • OBJECTIVE: To report repeated successful induction of fertility in an adult male patient with congenital adrenal hyperplasia (CAH) and testicular adrenal rest tumors (TART).
  • In an attempt to decrease tumor size and improve testicular function, his glucocorticoid medication was changed from hydrocortisone to an equivalent dosage of DXM.
  • CONCLUSION(S): In male CAH patients with testicular adrenal rest tumors, infertility may be reversible by replacing hydrocortisone with short courses of an equivalent dosage of DXM.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Dexamethasone / therapeutic use. Fertility / physiology. Hydrocortisone / therapeutic use. Testicular Neoplasms / complications


27. Claahsen-van der Grinten HL, Otten BJ, Takahashi S, Meuleman EJ, Hulsbergen-van de Kaa C, Sweep FC, Hermus AR: Testicular adrenal rest tumors in adult males with congenital adrenal hyperplasia: evaluation of pituitary-gonadal function before and after successful testis-sparing surgery in eight patients. J Clin Endocrinol Metab; 2007 Feb;92(2):612-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Testicular adrenal rest tumors in adult males with congenital adrenal hyperplasia: evaluation of pituitary-gonadal function before and after successful testis-sparing surgery in eight patients.
  • CONTEXT: In male patients with congenital adrenal hyperplasia (CAH), testicular adrenal rest tumors (TART) are frequently present.
  • These tumors can interfere with testicular function.
  • Intensifying glucocorticoid therapy does not always lead to tumor regression and improvement of testicular function.
  • OBJECTIVE: The aim of this study was to evaluate tumor volume, symptoms, and pituitary-gonadal function in male patients with CAH caused by 21-hydroxylase deficiency and bilateral TART before and after testis-sparing surgery.
  • MAIN OUTCOME MEASURES: The main outcome measures were absence of residual tumor and improvement of symptoms and pituitary-gonadal function.
  • RESULTS: Residual tumors were not found on any of the patients' magnetic resonance imaging after surgery.
  • CONCLUSION: Testis-sparing surgery did not improve pituitary-gonadal function despite successful removal of the tumors.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / surgery. Adrenal Rest Tumor / surgery. Pituitary Gland / physiology. Testicular Neoplasms / surgery. Testis / physiology. Testis / surgery

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  • (PMID = 17090637.001).
  • [ISSN] 0021-972X
  • [Journal-full-title] The Journal of clinical endocrinology and metabolism
  • [ISO-abbreviation] J. Clin. Endocrinol. Metab.
  • [Language] eng
  • [Publication-type] Clinical Trial; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / inhibin B; 2DI9HA706A / Estrone; 3XMK78S47O / Testosterone; 409J2J96VR / Androstenedione; 57285-09-3 / Inhibins; 68-96-2 / 17-alpha-Hydroxyprogesterone; 9002-60-2 / Adrenocorticotropic Hormone; 9002-67-9 / Luteinizing Hormone; 9002-68-0 / Follicle Stimulating Hormone
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28. Bercovici JP, Fiet J, Gibault L, Volant A, Abalain JH, Floch HH, Sonnet E, Fournier G: Testicular adrenal rest tumours in salt wasting congenital adrenal hyperplasia (in vivo and in vitro studies). J Steroid Biochem Mol Biol; 2005 Jan;93(1):67-72
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Testicular adrenal rest tumours in salt wasting congenital adrenal hyperplasia (in vivo and in vitro studies).
  • We describe the case of a 20-year-old patient with salt-wasting congenital adrenal hyperplasia (CAH) related to 21-hydroxylase deficiency.
  • Bilateral craggy testicular tumours were found, requiring histological evaluation.
  • High levels of 11beta-OH steroids measured in the gonadal vein, compared with peripheral blood samples suggested the presence of adrenal rests.
  • Incubation of the tumours (which could not be differentiated histologically, from Leydig tissue), with radioactive steroid precursors was carried out.
  • The results revealed the testicular tumours were of adrenal tissue origin, associated with 21-hydroxylase deficiency.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Rest Tumor / etiology. Steroid 21-Hydroxylase / metabolism. Testicular Neoplasms / etiology
  • [MeSH-minor] Adrenocorticotropic Hormone / pharmacology. Adult. Dehydroepiandrosterone Sulfate / blood. Dexamethasone / therapeutic use. Diagnosis, Differential. Fludrocortisone / therapeutic use. Follicle Stimulating Hormone / blood. Follow-Up Studies. Glucocorticoids / therapeutic use. Gonadotropin-Releasing Hormone / metabolism. Humans. Hydrocortisone / deficiency. Hypogonadism / etiology. Inhibins / blood. Leydig Cell Tumor / diagnosis. Luteinizing Hormone / blood. Male. Prolactin / blood. Renin / blood. Treatment Refusal

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  • (PMID = 15748834.001).
  • [ISSN] 0960-0760
  • [Journal-full-title] The Journal of steroid biochemistry and molecular biology
  • [ISO-abbreviation] J. Steroid Biochem. Mol. Biol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Glucocorticoids; 0 / inhibin B; 33515-09-2 / Gonadotropin-Releasing Hormone; 57285-09-3 / Inhibins; 57B09Q7FJR / Dehydroepiandrosterone Sulfate; 7S5I7G3JQL / Dexamethasone; 9002-60-2 / Adrenocorticotropic Hormone; 9002-62-4 / Prolactin; 9002-67-9 / Luteinizing Hormone; 9002-68-0 / Follicle Stimulating Hormone; EC 1.14.99.10 / Steroid 21-Hydroxylase; EC 3.4.23.15 / Renin; U0476M545B / Fludrocortisone; WI4X0X7BPJ / Hydrocortisone
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29. Ors F, Lev-Toaff A, O'Kane P, Qazi N, Bergin D: Paraovarian adrenal rest with MRI features characteristic of an adrenal adenoma. Br J Radiol; 2007 Sep;80(957):e205-8

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Paraovarian adrenal rest with MRI features characteristic of an adrenal adenoma.
  • We report MR and sonographic imaging features of an incidentally detected paraovarian adrenal rest in a 44-year-old woman who was being evaluated for menorrhagia.
  • This is the first report of chemical shift imaging identifying the presence of lipid within an adrenal rest as well as rapid washout of contrast.
  • Both of these MR characteristics are typically seen with an adrenal adenoma.
  • [MeSH-major] Adrenal Rest Tumor / diagnosis. Fallopian Tube Neoplasms / diagnosis
  • [MeSH-minor] Adenoma / diagnosis. Adenoma / ultrasonography. Adrenal Gland Neoplasms / diagnosis. Adrenal Gland Neoplasms / ultrasonography. Adult. Diagnosis, Differential. Female. Humans. Magnetic Resonance Imaging

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  • (PMID = 17928489.001).
  • [ISSN] 1748-880X
  • [Journal-full-title] The British journal of radiology
  • [ISO-abbreviation] Br J Radiol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] England
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30. Claahsen-van der Grinten HL, Hulsbergen-van de Kaa CA, Otten BJ: Ovarian adrenal rest tissue in congenital adrenal hyperplasia--a patient report. J Pediatr Endocrinol Metab; 2006 Feb;19(2):177-82
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  • [Title] Ovarian adrenal rest tissue in congenital adrenal hyperplasia--a patient report.
  • We report a young girl who died in an Addisonian crisis due to previously undiagnosed congenital adrenal hyperplasia (CAH), in whom ovarian adrenal rest tissue was detected at postmortem histopathological examination.
  • [MeSH-major] Addison Disease / complications. Adrenal Hyperplasia, Congenital / complications. Adrenal Rest Tumor / complications. Ovarian Neoplasms / complications


31. Erdogan S, Ergin M, Cevlik F, Yuksel B, Tuncer R, Tunali N, Polat S: Testicular adrenal rest hyperplasia due to 21-hydroxylase deficiency: a case report. Endocr Pathol; 2006;17(1):83-7
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  • [Title] Testicular adrenal rest hyperplasia due to 21-hydroxylase deficiency: a case report.
  • Bilateral testicular tumors are a rare complication of congenital adrenal hyperplasia.
  • It can be extremely difficult to distinguish histologically between Leydig cell tumors and adrenocortical rest hyperplasia, which may lead in some cases to unnecessary orchidectomy.
  • Hormonal studies established the diagnosis of congenital adrenal hyperplasia due to 21-hydroxylase deficiency.
  • Here we report a case of testicular adrenal rest hyperplasia in congenital adrenal hyperplasia and discuss the pathological and clinical features and origin of this rare lesion by using immunohistochemical evaluation.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / enzymology. Adrenal Rest Tumor / enzymology. Puberty, Precocious / etiology. Steroid 21-Hydroxylase / metabolism. Testicular Neoplasms / enzymology. Testis / pathology

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  • (PMID = 16760584.001).
  • [ISSN] 1046-3976
  • [Journal-full-title] Endocrine pathology
  • [ISO-abbreviation] Endocr. Pathol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] EC 1.14.99.10 / Steroid 21-Hydroxylase
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32. Reisch N, Flade L, Scherr M, Rottenkolber M, Pedrosa Gil F, Bidlingmaier M, Wolff H, Schwarz HP, Quinkler M, Beuschlein F, Reincke M: High prevalence of reduced fecundity in men with congenital adrenal hyperplasia. J Clin Endocrinol Metab; 2009 May;94(5):1665-70
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] High prevalence of reduced fecundity in men with congenital adrenal hyperplasia.
  • CONTEXT: Testicular adrenal rest tumors (TARTs) are regarded to contribute to the high prevalence of subfertility in males with congenital adrenal hyperplasia (CAH).
  • Of the other 17 well-controlled patients with normal or suppressed adrenal androgens and 17-hydroxyprogesterone levels, five presented with TARTs.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Hyperplasia, Congenital / epidemiology. Fertility / physiology. Infertility, Male / epidemiology. Infertility, Male / etiology
  • [MeSH-minor] Adrenal Rest Tumor / blood. Adrenal Rest Tumor / ultrasonography. Adult. Cohort Studies. Follicle Stimulating Hormone / blood. Hormones / blood. Hormones / metabolism. Humans. Inhibins / blood. Leydig Cells / physiology. Luteinizing Hormone / blood. Male. Middle Aged. Saliva / chemistry. Semen / chemistry. Semen / cytology. Sperm Count. Spermatogenesis. Testis / ultrasonography. Testosterone / blood. Treatment Outcome. Young Adult


33. Knape P, Reisch N, Dörr HG, Reincke M, Quinkler M: [Treatment of adult men with congenital adrenal hyperplasia syndrome due to 21-hydroxylase deficiency]. Dtsch Med Wochenschr; 2008 May;133(19):1025-9
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [Treatment of adult men with congenital adrenal hyperplasia syndrome due to 21-hydroxylase deficiency].
  • Information about the treatment of males with congenital adrenal hyperplasia (CAH) is scarce and there are no therapeutical guidelines.
  • The aim in the treatment of adult male CAH patients is preservation of fertility, prevention of an addisonian crisis, blood pressure management, prevention of testicular adrenal rest tumors (TART), maintaining well-being and good quality of life, satisfactory sexual function and prevention of long-term side effects of gluco- and mineralocorticoid therapy.
  • Some of these tumors are responsive to altered glucocorticoid therapy.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / therapy. Adrenogenital Syndrome / therapy. Glucocorticoids / therapeutic use. Mineralocorticoids / therapeutic use. Steroid 21-Hydroxylase / metabolism
  • [MeSH-minor] Addison Disease / etiology. Addison Disease / prevention & control. Adrenal Medulla / physiopathology. Adrenal Rest Tumor / etiology. Adrenal Rest Tumor / prevention & control. Adult. Continuity of Patient Care. Humans. Hypertension / etiology. Hypertension / prevention & control. Infertility, Male / etiology. Infertility, Male / prevention & control. Male. Testicular Neoplasms / etiology. Testicular Neoplasms / prevention & control


34. El Otmani H, Jodoin N, Yaici S, Welter ML, Bonnet AM, Navarro S, Pidoux B, Dormont D, Cornu P, Agid Y: [Can subthalamic nucleus stimulation reveal parkinsonian rest tremor?]. Rev Neurol (Paris); 2009 Jan;165(1):81-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [Can subthalamic nucleus stimulation reveal parkinsonian rest tremor?].
  • [Transliterated title] La stimulation du noyau subthalamique peut-elle révéler un tremblement de repos parkinsonien ?
  • INTRODUCTION: Rest tremor, one of the main symptoms in Parkinson's disease (PD), is dramatically improved following subthalamic nucleus stimulation (STN).
  • The occurrence of rest tremor after neurosurgery in patients without preoperative tremor is uncommon.
  • AIM: The aim of this work was to investigate the role of subthalamic nucleus stimulation in the appearance of parkinsonian rest tremor.
  • PATIENTS-RESULTS: Thirty PD patients (14%) out of 215 undergoing STN deep brain stimulation had an akinetorigid form of the disease, without preoperative tremor 11 years after onset of the disease.
  • This de novo parkinsonian tremor was improved by l-dopa treatment and disappeared when the stimulator was turned on.
  • CONCLUSION: This finding suggests that infraclinical parkinsonian tremor is probably present in all PD patients.
  • [MeSH-major] Adrenal Rest Tumor / diagnosis. Deep Brain Stimulation. Parkinson Disease / diagnosis. Subthalamic Nucleus / physiology

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  • (PMID = 18808774.001).
  • [ISSN] 0035-3787
  • [Journal-full-title] Revue neurologique
  • [ISO-abbreviation] Rev. Neurol. (Paris)
  • [Language] fre
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] France
  • [Chemical-registry-number] 0 / Antiparkinson Agents; 46627O600J / Levodopa
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35. Barwick TD, Malhotra A, Webb JA, Savage MO, Reznek RH: Embryology of the adrenal glands and its relevance to diagnostic imaging. Clin Radiol; 2005 Sep;60(9):953-9

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Embryology of the adrenal glands and its relevance to diagnostic imaging.
  • An understanding of the embryology of the adrenal glands is necessary to appreciate the location of adrenal ectopic, or rest, tissue which can occur anywhere along the course of gonadal descent.
  • This tissue usually has no clinical significance, but may become hyperplastic in patients with primary or secondary adrenal pathology.
  • In congenital adrenal hyperplasia, hyperplastic rest tissue may present as a soft-tissue mass, particularly in the gonads and retroperitoneum, and may be mistaken for tumour.
  • The adrenal in the neonate is proportionately much larger than in the adult; in renal ectopy or agenesis the ipsilateral adrenal is normally sited and may be mistaken for a kidney because of its size.
  • This review article illustrates the embryology of the adrenal with particular emphasis on the relevance of embryology to pathology.
  • [MeSH-major] Adrenal Glands / embryology. Adrenal Glands / radiography. Adrenal Hyperplasia, Congenital / radiography
  • [MeSH-minor] Adrenal Gland Neoplasms / radiography. Adrenal Rest Tumor / radiography. Female. Humans. Infant, Newborn. Kidney / abnormalities. Kidney / radiography. Male. Testicular Neoplasms / radiography. Testis / radiography. Tomography, X-Ray Computed

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  • (PMID = 16124976.001).
  • [ISSN] 0009-9260
  • [Journal-full-title] Clinical radiology
  • [ISO-abbreviation] Clin Radiol
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] England
  • [Number-of-references] 33
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36. De Padua M, Rajagopal V: Myxoid adrenal adenoma with focal pseudoglandular pattern. Indian J Med Sci; 2008 May;62(5):199-203

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Myxoid adrenal adenoma with focal pseudoglandular pattern.
  • Adrenal cortical tumors with myxoid change are rare tumors.
  • A pseudoglandular pattern has been described in 9 of these tumors.
  • We report a case of a myxoid adenoma of the left adrenal gland in a 67-year-old woman, with a focal pseudoglandular pattern involving about 20% of the studied tumor.
  • Rest of the tumor was composed of anastomosing cords of tumor cells.
  • Immunophenotype was consistent with an adrenal tumor, i.e., positive for vimentin, inhibin, and melan A.
  • [MeSH-major] Adrenal Cortex Neoplasms / pathology. Adrenocortical Adenoma / pathology

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  • (PMID = 18579979.001).
  • [ISSN] 0019-5359
  • [Journal-full-title] Indian journal of medical sciences
  • [ISO-abbreviation] Indian J Med Sci
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] India
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37. Gazymov MM, Filipov DS: [Adrenal tumors: topical diagnosis and surgical outcomes]. Urologiia; 2005 Sep-Oct;(5):9-12
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [Adrenal tumors: topical diagnosis and surgical outcomes].
  • A total of 128 adrenalectomies were made for catechol-producing tumor (n = 69), mineralocorticism (n = 27), primary and metastatic adrenal cancer (n = 20), other tumors (n = 12).
  • The rest patients improved, i.e. their malignant hypertension converted to a benign one.
  • If cancer involves the upper segment of the kidney, the tumor often invades the adrenal.
  • Therefore, pre- and intraoperative search for adrenal tumor must be made.
  • [MeSH-major] Adrenal Gland Neoplasms / diagnosis. Adrenal Gland Neoplasms / surgery. Adrenalectomy. Hypertension / surgery

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  • (PMID = 16281831.001).
  • [ISSN] 1728-2985
  • [Journal-full-title] Urologii︠a︡ (Moscow, Russia : 1999)
  • [ISO-abbreviation] Urologiia
  • [Language] rus
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] Russia (Federation)
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38. Fitoz S, Atasoy C, Adiyaman P, Berberoglu M, Erden I, Ocal G: Testicular adrenal rests in a patient with congenital adrenal hyperplasia: US and MRI features. Comput Med Imaging Graph; 2006 Dec;30(8):465-8
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Testicular adrenal rests in a patient with congenital adrenal hyperplasia: US and MRI features.
  • We present ultrasonographic and magnetic resonance imaging findings of intratesticular adrenal rests in a 16-year-old patient with congenital adrenal hyperplasia.
  • This case suggests that radiological evaluation of testes, even in the presence of normal physical examination findings, should be included in periodical follow-up of patients with congenital adrenal hyperplasia.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / pathology. Adrenal Hyperplasia, Congenital / ultrasonography. Adrenal Rest Tumor / pathology. Adrenal Rest Tumor / ultrasonography. Testicular Neoplasms / pathology. Testicular Neoplasms / ultrasonography


39. Rich T, Innominato PF, Boerner J, Mormont MC, Iacobelli S, Baron B, Jasmin C, Lévi F: Elevated serum cytokines correlated with altered behavior, serum cortisol rhythm, and dampened 24-hour rest-activity patterns in patients with metastatic colorectal cancer. Clin Cancer Res; 2005 Mar 1;11(5):1757-64
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  • [Title] Elevated serum cytokines correlated with altered behavior, serum cortisol rhythm, and dampened 24-hour rest-activity patterns in patients with metastatic colorectal cancer.
  • EXPERIMENTAL DESIGN: Eighty patients with metastatic colorectal cancer with either normal (group I, n = 40) or dampened (group II, n = 40) 24-hour rest/activity patterns measured by actigraphy were identified.
  • Actigraphy patterns were correlated with QOL indices, serum cortisol obtained at 8:00 a.m. and 4:00 p.m. and with serum levels of transforming growth factor-alpha, tumor necrosis factor-alpha, and interleukin 6 (IL-6) obtained at 8:00 a.m. and analyzed in duplicate by ELISA.
  • CONCLUSIONS: Significant correlations were found between serum levels of TGF-alpha and IL-6, circadian patterns in wrist activity and serum cortisol and tumor-related symptoms in patients with metastatic colorectal cancer.
  • These data support the hypothesis that some cancer patient's symptoms of fatigue, poor QOL, and treatment outcome are related to tumor or host generated cytokines and could reflect cytokine effects on the circadian timing system.
  • This interplay between cytokine signaling pathways, the hypothalamic-pituitary-adrenal axis, the autonomic nervous system, and efferent pathways of the suprachiasmatic nucleus that control circadian physiology, opens the way to new rational interventions for symptom management in cancer patients.
  • [MeSH-major] Circadian Rhythm. Colorectal Neoplasms / immunology. Colorectal Neoplasms / psychology. Interleukin-6 / blood. Neoplasm Metastasis. Transforming Growth Factor alpha / blood. Tumor Necrosis Factor-alpha / analysis

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  • (PMID = 15755997.001).
  • [ISSN] 1078-0432
  • [Journal-full-title] Clinical cancer research : an official journal of the American Association for Cancer Research
  • [ISO-abbreviation] Clin. Cancer Res.
  • [Language] eng
  • [Grant] United States / NCRR NIH HHS / RR / M01 RR 00847
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S.
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Interleukin-6; 0 / Transforming Growth Factor alpha; 0 / Tumor Necrosis Factor-alpha; WI4X0X7BPJ / Hydrocortisone
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40. Singh V, Sinha RJ, Sankhwar SN, Kumar S, Mehrotra B, Puri M, Sengottayan VK: Primary hepatocellular carcinoma in ectopic liver masquerading as left adrenal carcinoma: a rare occurrence. Rare Tumors; 2010;2(2):e35
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Primary hepatocellular carcinoma in ectopic liver masquerading as left adrenal carcinoma: a rare occurrence.
  • We report a unique case of primary hepatocellular carcinoma in an ectopic liver rest in the left renal hilum masquerading as a left adrenal tumor.
  • Adrenal tumors have been reported within adrenal rests inside the liver but hepatocellular carcinoma in ectopic liver rests in the adrenal area is an extremely rare entity.
  • He had chronic obstructive pulmonary disease and was hepatitis B surface antigen positive.
  • The provisional diagnosis of an adrenal tumor was made and open transperitoneal excision of the tumor along with two enlarged lymph nodes was done.
  • We concluded that chronic hepatitis B infection and chronic and excessive alcohol consumption and/or chronic smoking may have been the predisposing factors for the occurrence of primary hepatocellular carcin oma in the liver rest in our patient.

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  • (PMID = 21139837.001).
  • [ISSN] 2036-3613
  • [Journal-full-title] Rare tumors
  • [ISO-abbreviation] Rare Tumors
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Italy
  • [Other-IDs] NLM/ PMC2994502
  • [Keywords] NOTNLM ; carcinoma / hepatocellular / liver neoplasms
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41. Mirzaei MR, Rezvanian H, Siavash M, Parham M, Mahzouni P: A patient with refractory testicular adrenal rest tumour in the setting of cyp11b1 deficiency congenital adrenal hyperplasia. BMJ Case Rep; 2009;2009

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] A patient with refractory testicular adrenal rest tumour in the setting of cyp11b1 deficiency congenital adrenal hyperplasia.
  • Testicular adrenal rest tumour (TART) due to CYP11B1 deficiency is a very rare clinical finding.
  • The most important differential diagnosis of his latter presentation is Leydig cell tumour (LCT).
  • It was found that positive family history of congenital adrenal hyperplasia (CAH), hypertension, bilaterality, hypokalaemia and multiple hypoechoic masses on ultrasonography of the testes are in favour of a diagnosis of TART.
  • Conversely, high titres of tumour markers and presence of Reinke crystalloids are supportive of a diagnosis of LCT.

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  • [Cites] AJR Am J Roentgenol. 1999 May;172(5):1235-8 [10227495.001]
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  • (PMID = 21686875.001).
  • [ISSN] 1757-790X
  • [Journal-full-title] BMJ case reports
  • [ISO-abbreviation] BMJ Case Rep
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] England
  • [Other-IDs] NLM/ PMC3028312
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42. Jain SH, Sadow PM, Nosé V, Dluhy RG: A patient with ectopic cortisol production derived from malignant testicular masses. Nat Clin Pract Endocrinol Metab; 2008 Dec;4(12):695-700
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • DIAGNOSIS: Cushing syndrome was diagnosed on the basis of a markedly elevated 24-hour urine free cortisol level and classic cushingoid features.
  • The etiology of Cushing syndrome was determined to be an adrenocortical carcinoma arising from testicular adrenal rest cells.
  • Nevertheless, the possibility of a malignant Leydig cell tumor with ectopic cortisol production could not be excluded.
  • Despite initial success with this regimen, the patient died as a result of tumor progression and complications of poorly controlled hypercortisolism.
  • [MeSH-major] Adrenal Rest Tumor / complications. Cushing Syndrome / diagnosis. Cushing Syndrome / drug therapy. Hydrocortisone / blood. Testicular Neoplasms / complications
  • [MeSH-minor] Adrenocortical Carcinoma / blood. Adrenocortical Carcinoma / diagnosis. Adrenocortical Carcinoma / pathology. Adrenocorticotropic Hormone / blood. Aged. Humans. Male. Metyrapone / therapeutic use. Mitotane / therapeutic use

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  • (PMID = 18941436.001).
  • [ISSN] 1745-8374
  • [Journal-full-title] Nature clinical practice. Endocrinology & metabolism
  • [ISO-abbreviation] Nat Clin Pract Endocrinol Metab
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 78E4J5IB5J / Mitotane; 9002-60-2 / Adrenocorticotropic Hormone; WI4X0X7BPJ / Hydrocortisone; ZS9KD92H6V / Metyrapone
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43. Mönig H, Sippell W: Congenital adrenal hyperplasia in adulthood: do men need to continue treatment? Horm Res; 2005;64 Suppl 2:71-3
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Congenital adrenal hyperplasia in adulthood: do men need to continue treatment?
  • Due to early diagnosis and appropriate paediatric treatment, internists and endocrinologists are now caring for an increasing number of adult patients with congenital adrenal hyperplasia (CAH).
  • Problems that may be encountered in adult males with CAH are the continuing risk of developing an adrenal crisis and impaired gonadal function due to suppression of the hypothalamic-gonadal axis.
  • Fertility may be further compromised by testicular adrenal rest tumours.
  • Similar to female CAH patients, male patients may develop adrenal tumours due to inadequate adrenocorticotropic hormone suppression.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / drug therapy
  • [MeSH-minor] Adrenal Gland Neoplasms / etiology. Adrenal Insufficiency / etiology. Adult. Child. Fludrocortisone / therapeutic use. Humans. Hydrocortisone / therapeutic use. Hypertrophy / etiology. Infant. Infertility, Male / etiology. Male. Testis / pathology


44. Marazuela M, Domínguez-Gadea L, Larrañaga E, Rodríguez-Ramos R, López-Gallardo G, Rodríguez-Eyre JL, Gómez-Pan A: [The use of suprarenal scintigraphy in the differential diagnosis of suprarenal incidentaloma]. Rev Clin Esp; 2005 Jul;205(7):316-21
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [The use of suprarenal scintigraphy in the differential diagnosis of suprarenal incidentaloma].
  • [Transliterated title] Utilidad de la gammagrafía suprarrenal en el diagnóstico diferencial del incidentaloma suprarrenal.
  • OBJECTIVES: To assess the usefulness of adrenal scintigraphy for clinical evaluation of adrenal incidentalomas, and its relation with pathological diagnosis and follow-up.
  • PATIENTS AND METHODS: We have studied 46 patients with unilateral adrenal incidentaloma of size between 10 and 100 mm (average 30.5 +/- 19 mm).
  • The lesions were discovered with abdominal computerized tomography in the study of a primary tumor (22%) or in the evaluation of benign pathology (78%).
  • Adrenal scintigraphy assessed uptake in adrenal incidentaloma.
  • Hormonal study included urinary catecholamines, plasma cortisol after dexamethasone, adrenal androgens, and renin and aldosterone in hypertensive patients.
  • Five patients were operated, FNAB was carried out in three patients, and in the rest average follow-up was 29 +/- 21 months.
  • Adrenal incidentaloma was considered nonfunctional if the lesion did not modified its size nor showed analytical alterations along a follow-up higher than 8 months.
  • RESULTS: Of 46 adrenal lesions, seven didn't show uptake (three metastases, one cyst, one adrenal carcinoma, one pheochromocytoma, and one angiomyolipoma), 34 showed excessive uptake (29 nonfunctional adrenal nodules and 5 hyperfunctional adrenal nodules), and five had normal uptake (nonfunctional adrenal nodules).
  • Adrenal scintigraphy was compatible in all cases with cytological study or the response to chemotherapy.
  • CONCLUSIONS: Detection of a lesion with no uptake in adrenal scintigraphy forces to carry out complementary explorations in order to rule out malignant pathology.
  • A lesion with excessive uptake is indicative of a benign process and should be assessed with hormonal determinations.
  • [MeSH-major] Adrenal Gland Neoplasms / radionuclide imaging

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  • (PMID = 16029757.001).
  • [ISSN] 0014-2565
  • [Journal-full-title] Revista clínica española
  • [ISO-abbreviation] Rev Clin Esp
  • [Language] spa
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] Spain
  • [Chemical-registry-number] WI4X0X7BPJ / Hydrocortisone
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45. Charmandari E, Chrousos GP, Merke DP: Adrenocorticotropin hypersecretion and pituitary microadenoma following bilateral adrenalectomy in a patient with classic 21-hydroxylase deficiency. J Pediatr Endocrinol Metab; 2005 Jan;18(1):97-101
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • Objections to surgical adrenalectomy have been based on surgical risk, possible loss of protective adrenal function, and the risk of ACTH-induced activation of adrenal rest tissue.
  • We report a young female with salt-wasting CAH, who underwent bilateral adrenalectomy and developed severe hyperpigmentation, progressively marked corticotropin hypersecretion to concentrations seen in Nelson's syndrome (5,000-7,000 pg/ml), a pituitary microadenoma 5 years postoperatively, and probable ectopic adrenal rest tissue.
  • Patients with CAH have hyperactivity of the hypothalamic-pituitary-adrenal axis and are at risk for pituitary tumor formation.
  • [MeSH-major] Adenoma / etiology. Adrenal Glands. Adrenal Hyperplasia, Congenital / surgery. Adrenalectomy / adverse effects. Adrenocorticotropic Hormone / secretion. Choristoma. Pituitary Neoplasms / etiology

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  • (PMID = 15679075.001).
  • [ISSN] 0334-018X
  • [Journal-full-title] Journal of pediatric endocrinology & metabolism : JPEM
  • [ISO-abbreviation] J. Pediatr. Endocrinol. Metab.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Anti-Inflammatory Agents; 9002-60-2 / Adrenocorticotropic Hormone; WI4X0X7BPJ / Hydrocortisone
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46. Eisenhofer G, Sharabi Y, Pacak K: Unexplained symptomatic paroxysmal hypertension in pseudopheochromocytoma: a stress response disorder? Ann N Y Acad Sci; 2008 Dec;1148:469-78
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • Among overall numbers of patients tested for pheochromocytoma, less than 2% harbor the tumor.
  • Among the rest, there is often no satisfactory explanation for the signs and symptoms leading to suspicion of pheochromocytoma.
  • Similar to patients with the real tumor, patients with pseudopheochromocytoma can be misdiagnosed with panic disorder.
  • Because the clinical manifestations of pseudopheochromocytoma are similar, if not identical, to those due to excess circulating catecholamines in patients with the tumor, the most attractive explanation for the disorder is that it involves altered function of the autonomic nervous system.
  • In line with this hypothesis, recent findings suggest that enhanced adrenal release of epinephrine and exaggerated cardiovascular responsiveness to catecholamines both contribute to the paroxysmal hypertension and symptoms of catecholamine excess in pseudopheochromocytoma.
  • From this pattern, one would predict that therapeutic interventions that inhibit adrenal secretion of epinephrine or block adrenoceptor-mediated responses to catecholamines might provide a logical approach to therapy.
  • [MeSH-minor] Adrenal Gland Neoplasms / physiopathology. Adrenal Gland Neoplasms / therapy. Diagnosis, Differential. Epinephrine / metabolism. Humans. Panic Disorder / physiopathology


47. Claahsen-van der Grinten HL, Stikkelbroeck NM, Sweep CG, Hermus AR, Otten BJ: Fertility in patients with congenital adrenal hyperplasia. J Pediatr Endocrinol Metab; 2006 May;19(5):677-85
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Fertility in patients with congenital adrenal hyperplasia.
  • Congenital adrenal hyperplasia (CAH) is generally regarded as a paediatric endocrine disease, but nowadays nearly all patients reach adulthood as a result of improved diagnosis and treatment.
  • In males with CAH the fertility rate is reduced compared with the normal population, the most frequent cause being testicular adrenal rest tumours.
  • Development and growth of these tumours is assumed to be ACTH dependent and undertreatment may play an important role.
  • If intensifying glucocorticoid treatment does not lead to tumour decrease, surgical intervention may be considered, but the effect on fertility is not yet known.
  • Age of menarche and regularity of the menstrual cycle depends on the degree of adrenal suppression.
  • Not only adrenal androgens have to be normalised but also the levels of adrenal progestins (progesterone and 17-OH-progesterone) that interfere with normal ovulatory cycles.
  • Other factors that contribute to impaired fertility in females with CAH are ovarian hyperandrogenism (polycystic ovary syndrome), ovarian adrenal rest tumours, genital surgery and psychological factors.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Fertility / physiology

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  • (PMID = 16789634.001).
  • [ISSN] 0334-018X
  • [Journal-full-title] Journal of pediatric endocrinology & metabolism : JPEM
  • [ISO-abbreviation] J. Pediatr. Endocrinol. Metab.
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] England
  • [Number-of-references] 75
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48. Bhatt S, Rubens DJ, Dogra VS: Sonography of benign intrascrotal lesions. Ultrasound Q; 2006 Jun;22(2):121-36
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Sonography of benign intrascrotal lesions.
  • Ultrasound plays an important role and adds essential information in diagnosing benign intrascrotal lesions.
  • Characterization of benign intrascrotal lesions with sonography, in combination with clinical assessment, can lead to nonsurgical management or testicular sparing surgery.
  • We present important sonographic features of benign intrascrotal lesions, including extratesticular lesions: adenomatoid tumors, papillary cystadenomas, spermatoceles, hydroceles, varicoceles, hernias; and intratesticular lesions: tunica albuginea cysts, testicular simple cysts, epidermoid cysts, tubular ectasia of the rete testis, intratesticular varicoceles, adrenal rest tumors, and splenogonadal fusion.
  • The goal of this review is to provide the radiologist with a better understanding of benign lesions that occur in the scrotum.
  • [MeSH-minor] Diagnosis, Differential. Humans. Male

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  • (PMID = 16783242.001).
  • [ISSN] 0894-8771
  • [Journal-full-title] Ultrasound quarterly
  • [ISO-abbreviation] Ultrasound Q
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 73
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49. Szyfter W, Kopeć T, Kawczyński M: [Glomus caroticum, jugulare and vagale--problems in diagnosis and treatment]. Otolaryngol Pol; 2006;60(3):305-12

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [Glomus caroticum, jugulare and vagale--problems in diagnosis and treatment].
  • The rest (20%) are with close association with autonomic nerves and some cranial nerves.
  • Paraganglioma, being tumors arising from the paraganglion cells of the parasympathetic system, were first reported in man in 1935.
  • Although histologically these tumors have been shown to contain neurosecretory granules, they are rarely functional.
  • The first three of them are clinically recognized as tumor of the neck.
  • The tumor was removed totally with partial excision of communis, external and internal carotid artery.
  • The internal carotid artery was closed by the tumor.
  • [MeSH-major] Carotid Body Tumor. Glomus Jugulare Tumor. Glomus Tympanicum Tumor. Paraganglioma, Extra-Adrenal
  • [MeSH-minor] Adult. Diagnosis, Differential. Female. Humans. Male. Middle Aged. Treatment Outcome

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  • (PMID = 16989440.001).
  • [ISSN] 0030-6657
  • [Journal-full-title] Otolaryngologia polska = The Polish otolaryngology
  • [ISO-abbreviation] Otolaryngol Pol
  • [Language] pol
  • [Publication-type] Case Reports; English Abstract; Journal Article; Review
  • [Publication-country] Poland
  • [Number-of-references] 34
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50. Liu GH, Li HZ, Li YQ, Ji ZG, Jin W, Li HJ: [Treatment of male adrenogenital syndrome (report of 17 cases)]. Zhonghua Nan Ke Xue; 2006 Jul;12(7):633-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • METHODS: The clinical data of 17 patients with male adrenogenital syndrome, including 15 cases of congenital adrenal hyperplasia (CAH) and 2 cases of adrenocortical tumors, were analyzed retrospectively.
  • The patients with 21-hydroxylase deficiency (21-OHD) and 11beta-hydroxylase deficiency (11beta-OHD) were treated with adrenocortical hormone, those with 17-hydroxylase deficiency (17-OHD) received sexual glands excision and estrogen besides adrenocortical hormone, and those with adrenocortical tumors underwent surgical removal.
  • The testicular adrenal rest (TAR) tumor was reduced dramatically in 1 case of 21-OHD after treatment.
  • No relapse was found after resection of the adrenocortical tumors.
  • CONCLUSION: Adrenocortical hormone therapy helps improve the height growth and testicular function of CAH patients, and surgical removal is necessary for adrenocortical tumors.
  • [MeSH-minor] Adolescent. Adrenal Cortex Neoplasms / surgery. Adrenal Hyperplasia, Congenital / drug therapy. Adult. Child. Child, Preschool. Glucocorticoids / therapeutic use. Humans. Infant. Infant, Newborn. Male. Retrospective Studies

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  • (PMID = 16894944.001).
  • [ISSN] 1009-3591
  • [Journal-full-title] Zhonghua nan ke xue = National journal of andrology
  • [ISO-abbreviation] Zhonghua Nan Ke Xue
  • [Language] chi
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] China
  • [Chemical-registry-number] 0 / Glucocorticoids
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51. Somuncu S, Cakmak M, Caglayan F, Akman H, Ulusoy S, Kaya M: Idiopathic benign bilateral testicular enlargement in a pubertal boy: a case report and review of literature. J Pediatr Surg; 2005 Nov;40(11):e23-5

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Idiopathic benign bilateral testicular enlargement in a pubertal boy: a case report and review of literature.
  • We present a case of 12-year-old boy with idiopathic benign bilateral testicular enlargement.
  • We eliminated precocious puberty, juvenile hypothyroidism, adrenal rest tumors, X-linked mental retardation, and bilateral testicular neoplasms.
  • The clinical and laboratory features and differential diagnosis of benign bilateral testicular enlargement are discussed.
  • [MeSH-minor] Biomarkers / analysis. Child. Diagnosis, Differential. Functional Laterality. Humans. Hypertrophy. Male. Puberty

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  • (PMID = 16291135.001).
  • [ISSN] 1531-5037
  • [Journal-full-title] Journal of pediatric surgery
  • [ISO-abbreviation] J. Pediatr. Surg.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Biomarkers
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52. Otten BJ, Stikkelbroeck MM, Claahsen-van der Grinten HL, Hermus AR: Puberty and fertility in congenital adrenal hyperplasia. Endocr Dev; 2005;8:54-66
MedlinePlus Health Information. consumer health - Puberty.

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Puberty and fertility in congenital adrenal hyperplasia.
  • Congenital adrenal hyperplasia (CAH) is a disorder of adrenal steroid synthesis.
  • Adrenal progestagens and androgens are the main cause of disturbed ovarian activity.
  • In males, the main cause of subfertiltiy is the presence of testicular adrenal rest tumors, which are thought to originate from aberrant adrenal tissue and respond to treatment with glucocorticoids.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / physiopathology. Fertility / physiology. Puberty / physiology


53. Skrivan J, Zvĕrina E, Kluh J, Chovanec M, Pádr R: Our experience with surgical treatment of tympanojugular pragangliomas. Prague Med Rep; 2010;111(1):25-34

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • Tympanojugular paragangliomas are benign and slow growing lesions of the lateral skull base.
  • Treatment modalities include preoperative embolization of feeding vessels and tumour itself, surgical removal and irradiation.
  • Surgical radicality has been achieved in 62.5%, in the rest of patients the tumour remnants were either irradiated with a Leksell gamma knife, or left without treatment in a wait-end-rescan approach.
  • According to the benign character of the tumour, radicality is not always the highest goal, since a radical removal may be compromised by a high postoperative morbidity.
  • [MeSH-major] Ear Neoplasms / surgery. Glomus Jugulare Tumor / surgery. Paraganglioma, Extra-Adrenal / surgery

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  • (PMID = 20359435.001).
  • [ISSN] 1214-6994
  • [Journal-full-title] Prague medical report
  • [ISO-abbreviation] Prague Med Rep
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Czech Republic
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54. Nzegwu MA, Aghaji A: Neuroblastoma occurring in a 38-year old Nigerian man: a rare finding. Rare Tumors; 2009;1(1):e15

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Neuroblastoma occurring in a 38-year old Nigerian man: a rare finding.
  • In adults, abdomen/retroperitoneum are the primary sites and in children the adrenal gland.
  • We report a 38-year old civil servant who presented at our urology clinic on the 21(st) of December 2007 with a six month history of right flank dull pain which was worse on walking and relieved by rest, hypertension and a large right retroperitoneal mass.
  • Tumor resection revealed a grade III NB.

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  • (PMID = 21139886.001).
  • [ISSN] 2036-3613
  • [Journal-full-title] Rare tumors
  • [ISO-abbreviation] Rare Tumors
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Italy
  • [Other-IDs] NLM/ PMC2994447
  • [Keywords] NOTNLM ; adult. / neuroblastoma
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55. Bachelot A, Plu-Bureau G, Thibaud E, Laborde K, Pinto G, Samara D, Nihoul-Fékété C, Kuttenn F, Polak M, Touraine P: Long-term outcome of patients with congenital adrenal hyperplasia due to 21-hydroxylase deficiency. Horm Res; 2007;67(6):268-76
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Long-term outcome of patients with congenital adrenal hyperplasia due to 21-hydroxylase deficiency.
  • AIMS: Conflicting results exist regarding bone mineral density (BMD), metabolism and reproductive function of adult patients with congenital adrenal hyperplasia (CAH).
  • Three men presented with testicular adrenal rest tumors.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / drug therapy. Glucocorticoids / therapeutic use


56. Ishido K, Koizumi W, Tanabe S, Higuchi K, Sasaki T, Katada C, Azuma M, Saigenji K, Futawatari N, Saegusa M: A patient with stage IV type 4 advanced gastric cancer who had a complete pathological response to short-term treatment with S-1 alone. Anticancer Drugs; 2008 Oct;19(9):921-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • The left adrenal gland and the paragastric, mediastinal, and abdominal para-aortic lymph nodes were enlarged.
  • The patient started to receive S-1 (100 mg/day), given orally for 4 weeks followed by 2 weeks of rest.
  • The tumor had shrunk and was severely deformed.
  • Abdominal computed tomography revealed that ascites and enlargement of the left adrenal gland and paragastric lymph nodes had resolved.
  • The primary gastric tumor, resected lymph nodes, and a peritoneal-lavage specimen were all negative for tumor.
  • Histologically, the tumor had a complete pathological response to S-1.

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  • (PMID = 18766007.001).
  • [ISSN] 0959-4973
  • [Journal-full-title] Anti-cancer drugs
  • [ISO-abbreviation] Anticancer Drugs
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Drug Combinations; 150863-82-4 / S 1 (combination); 1548R74NSZ / Tegafur; 5VT6420TIG / Oxonic Acid
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57. Aktas S, Celebiler AC, Zadeoğlulari Z, Diniz G, Kargi A, Olgun N: Expression and methylation pattern of p16 in neuroblastoma tumorigenesis. Pathol Oncol Res; 2010 Mar;16(1):1-6
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • P16 is a tumour suppressor gene contributing in cell cycle arrest as cyclin dependent kinase inhibitor.
  • Methylation is an important mechanism for silencing tumor suppressor genes.
  • P16 was studied in 5 samples of normal adrenal medullar tissue, 5 samples of adrenal tissue including blastic rests, 5 samples of neuroblastoma in situ tissue and in 8 samples of neuroblastoma tissues primary and after chemotherapy in each group.
  • The adrenal gland tissues were obtained from paediatric autopsy cases.
  • P16 expression was positive in normal adrenal tissues, in one of 5 samples of adrenal blastic rest tissue and in all of samples of after chemotherapy; while no expression was observed in neuroblastoma and neuroblastoma in situ tissues.

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  • (PMID = 19466588.001).
  • [ISSN] 1532-2807
  • [Journal-full-title] Pathology oncology research : POR
  • [ISO-abbreviation] Pathol. Oncol. Res.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Netherlands
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58. Karasavvidou F, Potamianos SP, Barbanis S, Stathakis E, Psychos A, Kapsoritakis AN, Koukoulis G: Malakoplakia of the colon associated with colonic adenocarcinoma diagnosed in colonic biopsies. World J Gastroenterol; 2007 Dec 7;13(45):6109-11
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • It has also been found in many other sites such as the gastrointestinal tract, pancreas, liver, lymph nodes, skin, respiratory tract, adrenal gland, vagina and brain.
  • We present a case of a 64-year-old man referred to our hospital with cachexia and radiologic evidence of metastatic tumor of the liver.
  • Colonoscopy revealed a large malignant - appearing polypoid mass of the ascending colon and multiple distinct polyps throughout the rest of the colon.
  • Biopsies of the ascending colon mass confirmed the diagnosis of adenocarcinoma.

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  • (PMID = 18023111.001).
  • [ISSN] 1007-9327
  • [Journal-full-title] World journal of gastroenterology
  • [ISO-abbreviation] World J. Gastroenterol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] China
  • [Other-IDs] NLM/ PMC4250902
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59. Zhou Y, Chen BA, Zhao G: [Novel progress on treatment of acute graft-versus-host disease]. Zhongguo Shi Yan Xue Ye Xue Za Zhi; 2010 Feb;18(1):238-41
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [Novel progress on treatment of acute graft-versus-host disease].
  • Acute graft-versus-host disease (aGVHD) is a common complication after allogeneic hematopoietic stem-cell transplantation.
  • Over the past decade, monoclonal antibodies, biologic engineering products, and chemotherapeutics with immunomodulatory effects are being used as novel therapies in this disease.
  • Many of these agents, such as mycophenolate mofetil, anti-tumor necrosis factor antibodies, and anti-interleukin-2Ralpha-chain antibodies, have demonstrated promising activity in steroid-refractory aGVHD.
  • The key to improving aGVHD outcomes may, in fact, rest upon successful initial therapy, and timely taper corticosteroids to promote immune reconstitution.

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  • (PMID = 20137155.001).
  • [ISSN] 1009-2137
  • [Journal-full-title] Zhongguo shi yan xue ye xue za zhi
  • [ISO-abbreviation] Zhongguo Shi Yan Xue Ye Xue Za Zhi
  • [Language] CHI
  • [Publication-type] English Abstract; Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] China
  • [Chemical-registry-number] 0 / Adrenal Cortex Hormones; 0 / Antibodies, Monoclonal
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60. Ho VT, Cutler C: Current and novel therapies in acute GVHD. Best Pract Res Clin Haematol; 2008 Jun;21(2):223-37
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • Despite improvements in our understanding of transplant immunology and clinical and supportive care, acute graft-versus-host disease (GVHD) remains a clinical challenge and a major cause of morbidity and mortality for patients after allogeneic hematopoietic stem-cell transplantation.
  • New generations of monoclonal antibodies, biologics, and chemotherapeutics with immunomodulatory effects have been developed over the past decade, and are being tested as novel therapies in this disease.
  • Many of these agents - including, among others, mycophenolate mofetil, anti-tumor necrosis factor-alpha antibodies, denileukin diftitox, and anti-interleukin-2Ralpha-chain antibodies - have demonstrated promising activity in steroid-refractory acute GVHD.
  • The key to improving acute GVHD outcomes may, in fact, rest upon successful initial therapy, and timely taper of corticosteroids to promote healthier immune reconstitution.
  • [MeSH-major] Adrenal Cortex Hormones / therapeutic use. Antibodies, Monoclonal / therapeutic use. Graft vs Host Disease / drug therapy

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  • (PMID = 18503988.001).
  • [ISSN] 1521-6926
  • [Journal-full-title] Best practice & research. Clinical haematology
  • [ISO-abbreviation] Best Pract Res Clin Haematol
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Adrenal Cortex Hormones; 0 / Antibodies, Monoclonal; 0 / Immunologic Factors
  • [Number-of-references] 104
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61. Zöllner EW, Pitcher R: Adrenal rest tumours in congenital adrenal hyperplasia. S Afr Med J; 2007 Dec;97(12):1254
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Adrenal rest tumours in congenital adrenal hyperplasia.
  • [MeSH-major] 17-alpha-Hydroxyprogesterone / blood. Adrenal Hyperplasia, Congenital / complications. Adrenal Rest Tumor / etiology. Testicular Neoplasms / etiology
  • [MeSH-minor] Diagnosis, Differential. Follow-Up Studies. Humans. Infant, Newborn. Male. Time Factors


62. Maizlin ZV, Strauss S: Testicular adrenal rest tumors. Isr Med Assoc J; 2005 Mar;7(3):206-7
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Testicular adrenal rest tumors.
  • [MeSH-major] Adrenal Rest Tumor / diagnosis. Testicular Neoplasms / diagnosis
  • [MeSH-minor] Adolescent. Adrenal Hyperplasia, Congenital / complications. Humans. Male

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  • (PMID = 15792275.001).
  • [ISSN] 1565-1088
  • [Journal-full-title] The Israel Medical Association journal : IMAJ
  • [ISO-abbreviation] Isr. Med. Assoc. J.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Israel
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63. Collet TH, Pralong FP: Reversal of primary male infertility and testicular adrenal rest tumors in salt-wasting congenital adrenal hyperplasia. J Clin Endocrinol Metab; 2010 May;95(5):2013-4
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Reversal of primary male infertility and testicular adrenal rest tumors in salt-wasting congenital adrenal hyperplasia.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / drug therapy. Fludrocortisone / therapeutic use. Pregnenediones / therapeutic use
  • [MeSH-minor] Addison Disease. Adrenal Rest Tumor / drug therapy. Adrenal Rest Tumor / pathology. Adult. Dexamethasone / therapeutic use. Exons. Humans. Infertility, Male / etiology. Male. Mutation, Missense. Patient Compliance. Steroid 21-Hydroxylase / genetics


64. Nagamine WH, Mehta SV, Vade A: Testicular adrenal rest tumors in a patient with congenital adrenal hyperplasia: sonographic and magnetic resonance imaging findings. J Ultrasound Med; 2005 Dec;24(12):1717-20
MedlinePlus Health Information. consumer health - Testicular Cancer.

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Testicular adrenal rest tumors in a patient with congenital adrenal hyperplasia: sonographic and magnetic resonance imaging findings.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / pathology. Adrenal Hyperplasia, Congenital / ultrasonography. Adrenal Rest Tumor / pathology. Adrenal Rest Tumor / ultrasonography. Testicular Neoplasms / pathology. Testicular Neoplasms / ultrasonography


65. Lee D, Rodgers SK: Testicular adrenal rests. Ultrasound Q; 2008 Jun;24(2):105-7
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Testicular adrenal rests.
  • [MeSH-major] Adrenal Rest Tumor / ultrasonography. Testicular Neoplasms / ultrasonography
  • [MeSH-minor] Adrenal Hyperplasia, Congenital / ultrasonography. Child. Diagnosis, Differential. Humans. Male

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  • (PMID = 18528248.001).
  • [ISSN] 0894-8771
  • [Journal-full-title] Ultrasound quarterly
  • [ISO-abbreviation] Ultrasound Q
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
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66. Kim HK, Crotty E: Bilateral testicular adrenal rests in a boy with congenital adrenal hyperplasia. Pediatr Radiol; 2010 Dec;40 Suppl 1:S25
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Bilateral testicular adrenal rests in a boy with congenital adrenal hyperplasia.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Hyperplasia, Congenital / ultrasonography. Adrenal Rest Tumor / complications. Adrenal Rest Tumor / ultrasonography. Testicular Neoplasms / complications. Testicular Neoplasms / ultrasonography






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