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Introduction: Dissecting cellulitis of the scalp, or Hoffman disease, is described as an extremely rare condition. Clinically, it is represented by recurrent painful nodules, purulent drainage, interconnected sinus tracts and keloid formation, leading to scaring and cicatricial alopecia. Without a precise diagnosis and an adequate treatment, the repercussions consist of severe infectious complications along with psychological negative effects and serious aesthetic alterations. There is no standard treatment. In refractory cases, surgical management is reported. Patient concerns: We report a case of a 65-year-old Caucasian male patient, with a 5-year history of Hoffman disease, who presented with multiple abscesses and sinus tracts of the scalp and patches of alopecia. The lesions were non-responsive to medical treatment. Diagnosis: The diagnosis of DCS has been established on the basis of the clinical appearance and has been confirmed histopathologically. Interventions: The patient underwent wide excision of the scalp, followed by reconstruction using free latissimus dorsi flap and covered by meshed split-thickness skin graft. Outcomes: Eighteen-month follow-up revealed complete remission of symptoms and lesions along with satisfactory cosmetic result. Conclusion: The scope of this case report is to raise awareness of the following aspects: Hoffman disease has an extremely low occurrence rate, a difficult differential diagnosis and no standard therapeutical strategy. It also highlights the effectiveness of scalpectomy and free latissimus dorsi flap covered by meshed split-thickness skin graft in treating a very advanced stage of the disease together with providing a natural contouring of the scalp. Ultimately, it discusses the other treatment alternatives.