Background <p>Testicular malakoplakia (MP) is an extremely rare chronic granulomatous inflammatory lesion. It lacks specific clinical or imaging features and is highly prone to misdiagnosis as a testicular tumor. To our knowledge, no reports have described the contrast-enhanced ultrasound (CEUS) manifestations of this condition. Herein, we present a case with the aim of improving the understanding of testicular MP and reducing unnecessary misdiagnosis and oversight.</p> Patient presentation <p>A 41-year-old Han Chinese male presented with unexplained right scrotal swelling for more than 20&#xa0;days, accompanied by intermittent fever, chills, and dysuria, along with mild distending pain in the enlarged testicle. CEUS revealed that the right affected testicle had rapid high enhancement and slow washout. Computed tomography (CT) and magnetic resonance imaging (MRI) showed marked enlargement of the right testicle and altered parenchymal density, suggestive of a neoplastic lesion. Urine culture confirmed a bacterial infection with <i>Escherichia coli</i> (<i>E. coli</i>). Treatment with ceftriaxone and other antimicrobial agents yielded no significant improvement. Subsequently, a right orchiectomy was performed, and pathological examination confirmed the diagnosis of testicular MP. The patient’s urinary symptoms gradually resolved postoperatively. During the 3-month follow-up, no recurrence was observed, and inflammatory markers remained within the normal range.</p> Conclusions <p>The clinical and imaging presentations of testicular MP are nonspecific, posing a significant challenge for preoperative diagnosis. Definitive diagnosis still relies on pathological examination. Therefore, clinicians should increase their awareness of this rare disease to avoid misdiagnosis. To our knowledge, this report represents the first description and analysis of CEUS manifestations in a case of testicular MP.</p>