التفاصيل البيبلوغرافية
| العنوان: |
Undervirilized male infant with in utero exposure to maternal use of high dose antifungal therapy |
| المؤلفون: |
Gujral, Jasmine, Costin, Gertrude, Khurana, Divya, Yau, Mabel, Wallach, Elizabeth, Romero, Christopher J., Wilkes, Meredith, Sethuram, Swathi, Rapaport, Robert |
| المصدر: |
International Journal of Pediatric Endocrinology ; volume 2020, issue 1 ; ISSN 1687-9856 |
| بيانات النشر: |
Springer Science and Business Media LLC |
| سنة النشر: |
2020 |
| الوصف: |
Background Antifungals act on fungal sterols structurally similar to human cholesterol. Ketoconazole reversibly suppresses steroidogenesis by inhibiting cytochrome P450 enzymes and interferes with dihydrotestosterone (DHT) activity by binding to the androgen receptor. Hypospadias was reported in infants exposed to nystatin in utero. Case presentation A male infant exposed to antepartum nystatin presented with severe under-undervirilization and transient adrenal corticosteroid abnormalities. He was born in USA at 31 weeks gestation to a mother treated with vaginal Polygynax capsules (nystatin-100,000 international units, neomycin sulphate-35,000 international units and polymyxin B-35,000 international units) for vaginal discharge in the Ivory Coast. She used approximately 60 capsules between the first trimester until delivery. The infant was born with micropenis, chordee, perineo-scrotal hypospadias and bifid scrotum with bilaterally palpable gonads. The karyotype was 46,XY. No Mullerian structures were seen on ultrasound. Serum 17-hydroxyprogesterone (17 OHP) on newborn screening was high (304 ng/ml, normal < 35). Cortisol response to cosyntropin on the 3rd day of life (DOL) was 10 mcg/ml; the subnormal cortisol response may have resulted from prematurity and the predelivery treatment with betamethasone. The elevation of several adrenal corticosteroids was not consistent with any specific enzymatic defect. Hydrocortisone and fludrocortisone were initiated at another hospital for suspected mild glucocorticoid and mineralocorticoid deficiencies. Genetic screening for adrenal and gonadal developmental defects performed when transferred to our care were normal. All medications were gradually discontinued over 5–8 months. Adrenal and testicular responses to cosyntropin and human chorionic gonadotropin (hCG) were normal at 8 months. Conclusions We report severe undervirilization in a 46,XY infant born to a mother treated with prolonged and high dose nystatin during pregnancy. This presentation suggests ... |
| نوع الوثيقة: |
article in journal/newspaper |
| اللغة: |
English |
| DOI: |
10.1186/s13633-020-00087-1 |
| DOI: |
10.1186/s13633-020-00087-1.pdf |
| DOI: |
10.1186/s13633-020-00087-1/fulltext.html |
| الإتاحة: |
https://doi.org/10.1186/s13633-020-00087-1Test |
| حقوق: |
https://creativecommons.org/licenses/by/4.0Test ; https://creativecommons.org/licenses/by/4.0Test |
| رقم الانضمام: |
edsbas.CF53885F |
| قاعدة البيانات: |
BASE |
