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Estrogen-Only–Producing Adrenal Mass As an Overlooked Etiology of Isosexual Precocious Puberty in Girls: A Case Report and Literature Review Publisher



Haghshenas Z1 ; Mollaian M2 ; Alizadeh H3 ; Alehossein M3 ; Harsini S1 ; Zoham MH4
Authors
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Authors Affiliations
  1. 1. Department of Pediatric Endocrinology, Bahrami Children's Hospital, Tehran University of Medical Sciences, Tehran, Iran
  2. 2. Department of Pediatric Surgery, Bahrami Children's Hospital, Tehran University of Medical Sciences, Tehran, Iran
  3. 3. Department of Pediatric Radiology, Bahrami Children's Hospital, Tehran University of Medical Sciences, Tehran, Iran
  4. 4. Pediatric Intensive Care Unit, Bahrami Children's Hospital, Tehran University of Medical Sciences, Tehran, Iran

Source: AACE Clinical Case Reports Published:2017


Abstract

Objective: An estrogen-only–producing adrenal tumor is a rare etiology of isosexual precocious puberty (PP) in girls. Methods: We describe a 2.5-year-old girl who presented with signs and symptoms of isosexual PP. In primary laboratory and imaging investigations, serum estradiol level was found to be increased, while follicle-stimulating hormone, luteinizing hormone, adrenocorticotropic hormone, cortisol, testosterone, and 17-hydroxyprogesterone levels were shown to be within normal ranges. Abdominopelvic ultrasound and abdominal computed tomography revealed a right-sided adrenal mass, which was initially assumed to be an incidentaloma. Diagnosis of an adrenal cortical tumor was confirmed by tumor resection. Histologic examination revealed the tumor to be a benign adenoma with scattered areas of necrosis. Results: Tumor resection resulted in normalized serum estradiol and diminished clinical signs after 3 weeks. The child received no additional treatment and remains symptom free after 30 months of close observation. Conclusion: With the intent to spread the awareness of adrenocortical tumors as a potentially malignant cause of PP in children, and due to the rarity of an estrogen-producing adrenal mass, we discuss the clinical and biochemical features of our patient and a brief review of the literature. Abbreviations: CT computed tomography;PP precocious puberty © 2017 Elsevier Inc.