Cheilitis granulomatosa associated with lupus erythematosus discoid and treated with methotrexate: report of a case

Nazzaro, Gianluca; Muratori, Simona; Carrera, Carlo Giovanni; Coggi, Antonella; Gianotti, Raffaele

doi:10.1590/abd1806-4841.20153762

Cited by 4 publications

(3 citation statements)

References 9 publications

(9 reference statements)

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“…Autoimmunity has also been suggested as an aetiological factor, as there is an association of this syndrome with several autoimmune diseases such as Crohn’s disease and sarcoidosis 26 27. Melkersson-Rosenthal syndrome has also been reported in association with mixed connective tissue disease, discoid lupus erythematosus and Hashimoto’s thyroiditis 2 28–31…”

Section: Discussionmentioning

confidence: 99%

Melkersson-Rosenthal syndrome with coeliac and allergic diseases

Martins¹,

Azenha

Almeida

et al. 2019

BMJ Case Rep

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A 45-year-old man presented with a 10-year history of relapsing oedema of the lips. Moreover, he exhibited recurrent facial nerve palsy since the age of 10 years, coeliac disease since the age of 12 years, atopic eczema, allergic rhinitis and asthma. Physical examination revealed lip swelling and lingua plicata. Thus, he presented the classic triad of Melkersson-Rosenthal syndrome which includes recurrent orofacial oedema, facial nerve palsy and fissured tongue. A lip biopsy confirmed our clinical diagnosis.This case is particularly rare, as the classic triad is seen only in a minority of the cases. Moreover, allergic and coeliac diseases were observed concomitantly. This paper illustrates a potential pathophysiological interconnection between these pathologies in which interferon gamma could play a key role. To our knowledge, this is the first case report in which Melkersson-Rosenthal syndrome has been observed concurrently with coeliac disease.

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Section: Discussionmentioning

confidence: 99%

Melkersson-Rosenthal syndrome with coeliac and allergic diseases

Martins¹,

Azenha

Almeida

et al. 2019

BMJ Case Rep

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“…Miescher’s cheilitis is a chronic disfiguring condition clinically characterized by persistent swelling of the lips, consisting in a granulomatous cheilitis [1] . The etiology of CG is still unknown, but a genetic predisposition may be possible [2] .…”

Section: Introductionmentioning

confidence: 99%

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

Innocenti

Taverna

et al. 2017

International Journal of Surgery Case Reports

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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.

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“…Alfred Guido Miescher was a professor of Dermatology at the University Hospital of Zurich and was passionate about dermatopathology . His landmark work, Histologie de 100 cas de naevi pigmentaires d'après les methods de Masson , published in 1935, included 100 hemispherical melanocytic nevi found mostly on women's faces and nowadays known as “Miescher's nevi.” Ten years later, he described cheilitis granulomatosa, subsequently also called Miescher cheilitis, one of the components of Melkersson‐Rosenthal syndrome.…”

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confidence: 99%