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1. 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).
  • INTERVENTION(S): Changing glucocorticoid medication from 30 mg of hydrocortisone to 0.75 mg of dexamethasone (DXM) daily.
  • In an attempt to decrease tumor size and improve testicular function, his glucocorticoid medication was changed from hydrocortisone to an equivalent dosage of DXM.
  • Due to his progressive weight gain and striae, his medication was changed back to hydrocortisone, and he again developed again azoospermia.
  • 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


2. 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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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [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
  • [MeSH-minor] Adolescent. Adult. Body Mass Index. Child. Child, Preschool. Dexamethasone. Drug Therapy, Combination. Follow-Up Studies. Humans. Hydrocortisone / therapeutic use. Infant. Male. Middle Aged. Patient Selection. Young Adult

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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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3. 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.
  • The change from paediatric to adult medicine should be handled in a transition outpatient clinic organized by paediatric and adult endocrinologists.
  • The steroid therapy is usually orientated on an individual basis; but, general guidelines are lacking.
  • 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


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4. 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


5. 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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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [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.


6. Stikkelbroeck NM, Otten BJ, Pasic A, Jager GJ, Sweep CG, Noordam K, Hermus AR: High prevalence of testicular adrenal rest tumors, impaired spermatogenesis, and Leydig cell failure in adolescent and adult males with congenital adrenal hyperplasia. J Clin Endocrinol Metab; 2001 Dec;86(12):5721-8
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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 testicular adrenal rest tumors, impaired spermatogenesis, and Leydig cell failure in adolescent and adult males with congenital adrenal hyperplasia.
  • In male patients with congenital adrenal hyperplasia, testicular tumors, or so-called adrenal rest tumors, have been described, but their presence in well controlled patients is thought to be rare.
  • In this study, the prevalence of testicular tumors in 17 adolescent and adult male patients with congenital adrenal hyperplasia (age, 16-40 yr) was investigated.
  • In 16 of 17 patients, one or more testicular tumors, ranging in maximal length from 0.2-4.0 cm, were found on ultrasonography.
  • In 6 patients, the testicular tumors were palpable.
  • Nevertheless, 11 of these 12 patients showed testicular tumors on ultrasonography.
  • Neither the presence of undertreatment at the time of investigation nor characteristics of the therapeutic regimen (daily dose of hydrocortisone equivalents per body surface, the use of glucocorticoid medication either two or three times a day, or the time of taking the highest glucocorticoid dose either in the morning or the evening) could predict tumor size (maximal diameter of largest tumor).
  • In patients who were heterozygous or homozygous for the deletion or conversion of the CYP21 gene, tumor size was significantly larger than in patients who did not have this genotype.
  • We conclude that, when carefully sought for, testicular adrenal rest tumors are frequently present in adolescent and adult males with congenital adrenal hyperplasia and are often accompanied by impaired spermatogenesis and Leydig cell failure.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Hyperplasia, Congenital / physiopathology. Adrenal Rest Tumor / epidemiology. Adrenal Rest Tumor / etiology. Testicular Neoplasms / epidemiology. Testicular Neoplasms / etiology


7. Stikkelbroeck NM, Hermus AR, Suliman HM, Jager GJ, Otten BJ: Asymptomatic testicular adrenal rest tumours in adolescent and adult males with congenital adrenal hyperplasia: basal and follow-up investigation after 2.6 years. J Pediatr Endocrinol Metab; 2004 Apr;17(4):645-53
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Asymptomatic testicular adrenal rest tumours in adolescent and adult males with congenital adrenal hyperplasia: basal and follow-up investigation after 2.6 years.
  • AIM: To study the course of asymptomatic testicular adrenal rest tumours in patients with congenital adrenal hyperplasia (CAH) and the association between tumour changes and glucocorticoid therapy adjustments.
  • PATIENTS AND METHODS: Fifteen male patients with CAH (21-hydroxylase deficiency), in whom asymptomatic testicular adrenal rest tumours had been found at a baseline investigation, underwent scrotal ultrasonography and venous blood sampling (for LH, FSH and testosterone) on average 2.6 years later.
  • RESULTS: Tumour decrease, defined as > or =30% decrease in the sum of the longest diameter(s) of the lesion(s), was found in six patients; tumour increase, defined as > or =20% increase, in six and stable tumours in three patients.
  • All three patients with overtreatment showed tumour decrease and of the six patients with undertreatment only one showed tumour decrease.
  • Tumour increase was not only observed in undertreated patients but also in patients with adequate treatment.
  • Changing the night dose of hydrocortisone into dexamethasone, to obtain prolonged ACTH suppression, had resulted in better adrenal suppression in only one patient.
  • CONCLUSIONS: Tumour decrease could be achieved by aiming at adrenal oversuppression, but the required high glucocorticoid doses may induce side effects.
  • In asymptomatic tumours in young male patients with CAH, a practical guideline could be to optimise adrenal suppression to a maximal tolerable glucocorticoid dose and to offer analysis and cryopreservation of semen as soon as the patient can be motivated.
  • [MeSH-major] Adrenal Hyperplasia, Congenital / complications. Adrenal Hyperplasia, Congenital / drug therapy. Adrenal Rest Tumor / etiology. Dexamethasone / administration & dosage. Glucocorticoids / administration & dosage. Hydrocortisone / administration & dosage. Testicular Neoplasms / etiology
  • [MeSH-minor] Adolescent. Adult. Androstenedione / antagonists & inhibitors. Dose-Response Relationship, Drug. Follicle Stimulating Hormone / blood. Follow-Up Studies. Humans. Luteinizing Hormone / blood. Male. Saliva / metabolism. Testosterone / blood


8. Hughes I: Testicular adrenal rest tumours. J Pediatr Endocrinol Metab; 2004 Apr;17(4):589-90
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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.
  • [MeSH-major] Adrenal Rest Tumor. Testicular Neoplasms
  • [MeSH-minor] Adrenal Hyperplasia, Congenital / complications. Dose-Response Relationship, Drug. Glucocorticoids / administration & dosage. Glucocorticoids / adverse effects. Glucocorticoids / therapeutic use. Humans. Male

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  • [CommentOn] J Pediatr Endocrinol Metab. 2004 Apr;17(4):645-53 [15198296.001]
  • (PMID = 15198289.001).
  • [ISSN] 0334-018X
  • [Journal-full-title] Journal of pediatric endocrinology & metabolism : JPEM
  • [ISO-abbreviation] J. Pediatr. Endocrinol. Metab.
  • [Language] eng
  • [Publication-type] Comment; Editorial
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Glucocorticoids
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