madman
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Combined surgical and medical treatment in an adolescent with severe gynecomastia due to excessive estradiol secretion: a case report
Abstract
Background: Gynecomastia develops due to the reversed estradiol-to-Testosterone ratio in adolescence, and symptoms typically improve within 2 years. The causes vary widely, including estrogen excess and tumors, and surgical treatment is usually given in late adolescence because postoperative symptoms may recur in adolescents. This study reports a case of a pediatric patient with severe gynecomastia due to excessive estradiol secretion who showed a positive outcome after receiving surgical treatment combined with aromatase inhibitor administration.
Case presentation: A 9-year old boy visited to the Department of Pediatric Endocrinology for breast budding. At that time, the patient showed breasts at Tanner stage II and no abnormality on hormone tests. During a follow-up, both gynecomastia had progressed to Tanner stage III–IV at age 13. Tamoxifen 10 mg bid was administered; however, the condition rapidly progressed to Tanner stage V at 13.5 years. The evaluation of pathologic gynecomastia showed an increase of estradiol to 296 pg/mL with normal range 10 ~ 36 pg/mL and microlithiasis in both testes. As the condition worsened, total mastectomy was performed at the age of 13.5 years. Based on the assessment that elevated aromatase activity had induced breast budding, we changed the medication to anastrozole (Arimidex) 1 mg once a day, after which the estradiol level improved to 38.5 pg/mL and was maintained well in the two-year postoperative follow-up.
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Conclusions: This case report shows a combined plastic surgery and appropriate medical management bring a positive outcome in severe gynecomastia patient.
Background
Gynecomastia, enlargement of the breast tissue in men, is often found in infancy, adolescence, and late adulthood, with 50–60% of incidences occurring in adolescence [1–3]. Gynecomastia is caused by ductal epithelial hyperplasia and increased growth of stromal and periductal connective tissue due to elevated estradiol activity and lowered androgen activity [1, 4]. The etiology has not been clearly identified, but it is predicted to be from abnormal testosterone-to estrogen ratio and it is believed to be related to increased peripheral conversion of testosterone to estradiol as testosterone levels rise during puberty [5]. Although gynecomastia is a physiologic finding that spontaneously resolves in most cases [4, 6], it is important to rule out pathologic gynecomastia in severe and progressive cases. Causes of gynecomastia vary and include hypogonadism, thyroid disease, hyperprolactinemia, estrogen excess, and tumors [7]. Estrogen biosynthesis involves a conversion to steroid precursor androgens and the conversion of androgens to estrogens by enzymatic aromatase activity [8]. The increase in excess estradiol also results from elevated aromatase activity, and its causes include the elevated secretion of enzymes in Sertoli and Leydig cell tumors and aromatase gene mutation [9, 10]. Pathologic gynecomastia is uncommon, and it is crucial to perform appropriate tests to diagnose and provide proper treatment. For idiopathic gynecomastia patients in adolescent period, the first line choice is Tamoxifen, and in aromatase excess patients, aromatase inhibitor is used and if there is a tumor, then the tumor is removed by principle. However, the decision of the best therapeutic approach should be made considering the psychological distress in patients with gynecomastia [11].
Conclusions
We report a case of severe gynecomastia in which identification of aromatase excess syndrome leading to pathologic gynecomastia and a combination of endocrine therapy and proactive surgical treatment resulted in positive outcomes in terms of both symptoms and prognosis in a pediatric patient. This case report shows a combined plastic surgery and appropriate medical management bring a positive outcome in severe gynecomastia patient, and it suggests a need for endocrine evaluation in pediatric patients with severe and progressive gynecomastia.
Abstract
Background: Gynecomastia develops due to the reversed estradiol-to-Testosterone ratio in adolescence, and symptoms typically improve within 2 years. The causes vary widely, including estrogen excess and tumors, and surgical treatment is usually given in late adolescence because postoperative symptoms may recur in adolescents. This study reports a case of a pediatric patient with severe gynecomastia due to excessive estradiol secretion who showed a positive outcome after receiving surgical treatment combined with aromatase inhibitor administration.
Case presentation: A 9-year old boy visited to the Department of Pediatric Endocrinology for breast budding. At that time, the patient showed breasts at Tanner stage II and no abnormality on hormone tests. During a follow-up, both gynecomastia had progressed to Tanner stage III–IV at age 13. Tamoxifen 10 mg bid was administered; however, the condition rapidly progressed to Tanner stage V at 13.5 years. The evaluation of pathologic gynecomastia showed an increase of estradiol to 296 pg/mL with normal range 10 ~ 36 pg/mL and microlithiasis in both testes. As the condition worsened, total mastectomy was performed at the age of 13.5 years. Based on the assessment that elevated aromatase activity had induced breast budding, we changed the medication to anastrozole (Arimidex) 1 mg once a day, after which the estradiol level improved to 38.5 pg/mL and was maintained well in the two-year postoperative follow-up.
'
Conclusions: This case report shows a combined plastic surgery and appropriate medical management bring a positive outcome in severe gynecomastia patient.
Background
Gynecomastia, enlargement of the breast tissue in men, is often found in infancy, adolescence, and late adulthood, with 50–60% of incidences occurring in adolescence [1–3]. Gynecomastia is caused by ductal epithelial hyperplasia and increased growth of stromal and periductal connective tissue due to elevated estradiol activity and lowered androgen activity [1, 4]. The etiology has not been clearly identified, but it is predicted to be from abnormal testosterone-to estrogen ratio and it is believed to be related to increased peripheral conversion of testosterone to estradiol as testosterone levels rise during puberty [5]. Although gynecomastia is a physiologic finding that spontaneously resolves in most cases [4, 6], it is important to rule out pathologic gynecomastia in severe and progressive cases. Causes of gynecomastia vary and include hypogonadism, thyroid disease, hyperprolactinemia, estrogen excess, and tumors [7]. Estrogen biosynthesis involves a conversion to steroid precursor androgens and the conversion of androgens to estrogens by enzymatic aromatase activity [8]. The increase in excess estradiol also results from elevated aromatase activity, and its causes include the elevated secretion of enzymes in Sertoli and Leydig cell tumors and aromatase gene mutation [9, 10]. Pathologic gynecomastia is uncommon, and it is crucial to perform appropriate tests to diagnose and provide proper treatment. For idiopathic gynecomastia patients in adolescent period, the first line choice is Tamoxifen, and in aromatase excess patients, aromatase inhibitor is used and if there is a tumor, then the tumor is removed by principle. However, the decision of the best therapeutic approach should be made considering the psychological distress in patients with gynecomastia [11].
Conclusions
We report a case of severe gynecomastia in which identification of aromatase excess syndrome leading to pathologic gynecomastia and a combination of endocrine therapy and proactive surgical treatment resulted in positive outcomes in terms of both symptoms and prognosis in a pediatric patient. This case report shows a combined plastic surgery and appropriate medical management bring a positive outcome in severe gynecomastia patient, and it suggests a need for endocrine evaluation in pediatric patients with severe and progressive gynecomastia.
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