Introduction: Pulmonary alveolar microlithiasis (PAM) is a rare hereditary disease caused by the mutation of the SLC34A2 gene, characterized by the deposition of calcium phosphate microliths throughout the alveolar spaces. Calcifications have also been reported in other organs such as the genitalia of male patients and can be associated with infertility cases. Our case report is the first Moroccan case reported in the literature of a male with PAM and infertility. Case Report: We present a case of a 47-year-old male patient (Moroccan, North African) presented with complaints of persistent grade I dyspnea for the last six years. He also claimed primary infertility of 20 years since marriage, PAM was diagnosed based on the typical appearance of the chest and pelvic high resolution computed tomography (HRCT). The infertility was due to distal obstruction of the seminal tract by small calcifications similar to pulmonary microliths. Low dose inhaled corticosteroids/long-acting beta2-agonists (ICS-LABA) was started as a treatment of symptoms with good control of symptoms. Conclusion: Pulmonary alveolar microlithiasis is a rare autosomal recessive disorder not easily described because of its rarity worldwide, its clinical course, and its evolution. Its association with infertility may be more common than is currently known, suggests the need to make a particularly close examination of the chest X-ray in some cases of male infertility.
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