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      Prevalence of Testicular Microlithiasis in Males with Congenital Adrenal Hyperplasia and Its Association with Testicular Adrenal Rest Tumors

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          Abstract

          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). Patients and Methods: Scrotal ultrasound using a high-frequency linear transducer (12 MHz) was performed in 41 patients aged 12.1 ± 4.7 (range 3.5–23.3) years and 49 healthy similarly aged controls. TM was classified with respect to the number of microliths per ultrasound field as limited (LTM, <5 microliths) and classic (CTM, ≧ 5 microliths). CTM was graded as grade 1 (5–10 microliths), grade 2 (11–20 microliths), and grade 3 (>20 microliths). Results: TM was detected bilaterally in 9 (21.9%) patients and 2 (4.1%) control cases (1 bilateral, 1 unilateral). Four patients had LTM, one evaluated as grade 1, one as grade 2, and three as grade 3. There were 9 patients with TART. Four patients had TM and TART concomitantly. Conclusion: Because TM is frequently found in patients with CAH and may also exist concomitantly with TART, we recommend that these patients be followed annually by testicular ultrasound.

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          Growth references for Turkish children aged 6 to 18 years.

          Olcay Neyzi, Rüveyde Bundak, Feyza Darendeliler … (2006)
          To create up-to-date reference standards for Turkish children, and to compare these with growth standards for US children (CDC 2000 Growth Charts) and with previous local data. Height and weight measurements of 1100 boys and 1020 girls were obtained by biannual visits to six schools located in relatively well-off districts of Istanbul city. All children came from well-to-do families and all were healthy. All measurements were made by two trained technicians. The LMS method was used in the analyses. Heights of the boys and girls in all age groups were close to the updated 2000 USA growth references and showed an increase from data on Turkish children born 30 y earlier. Weight values were high compared to reference data on US children and to the older data on Turkish children. These results indicate that height growth in Turkish school-age children of high socio-economic level conforms to the updated growth data on US children. The data also show a secular upward trend in Turkey. Weight-for-age values indicate an increase in obesity. The results also point to the value of collecting and evaluating local growth data periodically.
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            Testicular tumors in patients with congenital adrenal hyperplasia due to 21-hydroxylase deficiency show functional features of adrenocortical tissue.

            Ad Hermus, Brian Ross, Eric J H Meuleman … (2007)
            In male patients with congenital adrenal hyperplasia (CAH), testicular adrenal rest tumors are frequently found that may interfere with gonadal function. Our objective was to determine steroid-producing features of testicular adrenal rest tumors. The study is descriptive and took place at a university medical center. Eight adult CAH patients with bilateral testicular adrenal rest tumors were treated with testis-sparing surgery. 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. Adrenal-specific steroids/enzymes were assessed. 21DF, 17OHP, and A levels were measurable in all spermatic vein samples. The ratio (mean +/- SD) between spermatic vein and simultaneously taken peripheral blood samples was 37.8 +/- 56.3 (21DF), 132.0 +/- 249 (17OHP), and 57.0 +/- 68.2 (A). 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. 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.
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              Prevalence of testicular adrenal rest tumours in male children with congenital adrenal hyperplasia due to 21-hydroxylase deficiency.

              Alice den Hertog, Ad Hermus, F Sweep … (2007)
              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. The aim of our study was to determine the presence of TART and their influence on gonadal function in childhood. Retrospective study. Scrotal ultrasound was performed in 34 children with CAH due to 21-hydroxylase deficiency who were between 2 and 18 years old. FSH, LH, testosterone and inhibin B concentrations were measured in serum of 27 patients. TART were detected by ultrasound in 8 out of 34 (24%) children. In two of them, bilateral tumours were found. All lesions were located in the rete testis. Seven patients had the salt-wasting type of CAH; one patient had the simple virilising type of CAH. Mean tumour size was 4.1 mm (range 2-8 mm). In none of the patients were the tumours palpable. Two children with TART were between 5 and 10 years old, the other six children were above 10 years old. In all children with TART, LH, FSH, testosterone and inhibin B levels were similar to the patients without TART. TART can be found in CAH children before the age of 10 years. The absence of gonadal dysfunction in our group of children suggests that gonadal dysfunction as frequently reported in adult CAH patients with TART develops after childhood.
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                Author and article information

                Journal
                Journal ID (publisher-id): HRP
                Journal ID (nlm-ta): Horm Res Paediatr
                Journal ID (doi): 10.1159/issn.1663-2818
                Title: Hormone Research in Paediatrics
                Publisher: S. Karger AG
                ISSN (Print): 1663-2818
                ISSN (Electronic): 1663-2826
                Publication date Subscription-year: 2010
                Publication date (Print): May 2010
                Publication date (Electronic): 20 April 2010
                Volume: 73
                Issue: 6
                Pages: 443-448
                Affiliations
                aPediatric Endocrinology Unit, Department of Pediatrics, and bDepartment of Radiology, Istanbul Faculty of Medicine, and cInstitute of Child Health, Istanbul University, Istanbul, Turkey
                Author notes
                *Sukran Poyrazoglu, Hamidiye sok. No: 9 Daire: 20, Gayrettepe, Beşiktaş, TR–34349 Istanbul (Turkey), Tel. +90 212 288 95 63, Fax +90 212 533 13 83, E-Mail sukranpoyrazoglu@yahoo.com
                Article
                Publisher ID: 313587 Other ID: Horm Res Paediatr 2010;73:443–448
                DOI: 10.1159/000313587
                PubMed ID: 20407230
                SO-VID: 51f9ec7d-a36b-446c-9a05-b387562ed74f
                Copyright © © 2010 S. Karger AG, Basel
                License:

                Copyright: All rights reserved. No part of this publication may be translated into other languages, reproduced or utilized in any form or by any means, electronic or mechanical, including photocopying, recording, microcopying, or by any information storage and retrieval system, without permission in writing from the publisher. Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug. Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.

                History
                Date received : 06 February 2009
                Date accepted : 12 August 2009
                Page count
                Figures: 1, Tables: 2, References: 37, Pages: 6
                Categories
                Subject: Original Paper

                ScienceOpen disciplines: Endocrinology & Diabetes,Neurology,Nutrition & Dietetics,Sexual medicine,Internal medicine,Pharmacology & Pharmaceutical medicine
                Keywords: Congenital adrenal hyperplasia,Microlithiasis, testicular,Testicular adrenal rest tumor
                Data availability:
                ScienceOpen disciplines: Endocrinology & Diabetes, Neurology, Nutrition & Dietetics, Sexual medicine, Internal medicine, Pharmacology & Pharmaceutical medicine
                Keywords: Congenital adrenal hyperplasia, Microlithiasis, testicular, Testicular adrenal rest tumor

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