Miescher’s cheilitis: Surgical management and long term outcome of an extremely severe case

Abstract

IntroductionMiescher’s cheilitis is clinically characterized by persistent swelling of the lip(s). Its pathogenesis is still unknown. Histopathologically is characterized by sub-epithelial edema, increased number of dilated lymphatic vessels and an inflammatory infiltrate and/or non-caseating/non- necrotic granulomas. Even if the disorder must be controlled by medical therapy, surgery may be required to treat most severe cases.Presentation of the caseWe report a 30-year-old man who presented a persistent swelling of both lips since 8 years, previously treated with intralesional steroid and immunosuppressive therapy. Clinical examination did not show facial nerve palsy or other associated conditions. On the base of clinical and histopathological findings, a diagnosis of Miescher’s syndrome was made. Patient underwent Conway’s reduction cheilopasty repaired with local flaps. At one-year follow-up, the patient does not show local recurrence of the deformity; both oral continence and lip sensation are preserved.DiscussionBecause of its extreme rarity and unknown etiopathogenesis, Miescher’s cheilitis receives poor attention and may often remain misdiagnosed. Several medical therapies are proposed, in particular steroids and immunosuppression. Even if medical therapy remains the main treatment, surgery may be required.ConclusionSatisfactory results have been obtained combining medical therapy and surgical approach.

Orofacial granulomatosis: Clinical and therapeutic features in an Italian cohort and review of the literature

“…In one patient, based on high circulating levels of total IgE, the anti‐IgE monoclonal antibody omalizumab (300 mg s.c. monthly for 6 months) was used, with good clinical response but relapse upon drug discontinuation 10 . Lip reduction surgery was the choice for two patients with good and persistent results 11 . No response to anti‐H1 drugs was observed.…”

Miescher’s cheilitis: A case report with literature review

“…Various other treatment options for MC have been reported including antibiotics like tetracycline, roxithromycin and clofazimine 16 , metronidazole 13 , hydroxychloroquine 17 , oral and intralesional steroids 13,18 , adalimumab (TNF alpha inhibitor) 13 and surgical resection. 19…”

<p>Management Strategies of Melkersson-Rosenthal Syndrome: A Review</p>

“…In one such case, based on Conway's technique, en-block removal of mucosa dorsal to vermillion was done. 38 Part of orbicularis muscle and grossly edematous tissue were also removed. 38 Helium laser ablation is an alternative procedure for cheiloplasty.…”

“…38 Part of orbicularis muscle and grossly edematous tissue were also removed. 38 Helium laser ablation is an alternative procedure for cheiloplasty. Both these procedures may cause loss of sensation in the affected lip and do not affect disease recurrence.…”