• Jihan Ahmad, Adnan Ahmad, and Lama Hadid.
• Department of Endocrinology, Al-Assad University Hospital, Damascus University, Damascus, Syria.
• Medicine (Baltimore). 2023 Dec 8; 102 (49): e36061e36061.

RationaleCongenital adrenal hyperplasia (CAH) is considered one of the most common inherited disorders. In about more than 95% of all CAH cases, the deficient enzyme is 21-hydroxylase. Infertility is an important complication of this disease, and although this topic has been studied more frequently in females, cases, and literature reviews of the causes of infertility in male patients are constantly increasing.Patient ConcernsA 28 old male with congenital adrenal hyperplasia (we assume to be a nonclassical type) presented to our institution with infertility and suspected bilateral testicular masses after 4 years of stopping dexamethasone.DiagnosisTesticular adrenal rest tumors.InterventionsDexamethasone was reapplied in a supraphysiologic dose (1.5 mg before bedtime) with periodic monitoring of the patient.OutcomesTreatment with supraphysiologic dose of dexamethasone led to regression of these tumors and significant improvement in sperm count, resulting in being capable of having a child.LessonsThere are many suspected causes of reduced male fertility in male CAH patients and the presence of testicular adrenal rest tumors is the main cause of infertility in this population. These benign tumors are believed to arise from ectopic adrenal cells in the testes, that grow under adrenocorticotropic hormone stimulation in poorly controlled patients. Annual scrotal ultrasound is recommended in all males with CAH for detection and treatment of these tumors as early as possible before they cause permanent damage to the seminiferous tubules and irreversible infertility.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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