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Rationale: Kerion Celsi, a severe form of tinea capitis, is generally caused by zoophilic and geophilic fungi. This is the first report of an unusual case of kerion Celsi caused by Microsporum gypseum in a 6-year-old boy.Patient concerns: A 6-year-old boy presented to the dermatology clinic with the complaint of multiple pustules, edematous plaques over the scalp with hair loss for 1 month.Diagnosis: Clinical and laboratory investigations, including reverse transcriptase-quantitative polymerase chain reaction, confirmed M gypseum causing kerion Celsi.Interventions: Upon combination therapy using oral itraconazole and oral prednisolone along with the topical terbinafine, kerion Celsi remitted in the patient.Outcome: New hair growth was noted during the 4-month follow-up.Lesson: We presented the first case of kerion Celsi infection secondary to M gypseum that was probably transmitted from a guinea pig.

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