Radiofrequency therapy as an effective treatment for granulomatous cheilitis: A CARE case report.

INTRODUCTION: Granulomatous cheilitis is a rare disorder characterized by recurrent, idiopathic, and painless lip swelling. The diagnosis is proven by histopathological examination. The unknown aetiology and poorly understood underlying mechanism contribute to the difficulty in establishing an effective treatment. This case study proposes the effectiveness of radiofrequency therapy in the management of refractory granulomatous cheilitis. CASE SUMMARY: A 68-year-old patient presented with hypertrophy and swelling of the lower lip, and a biopsy revealed actinic cheilitis. The patient underwent lip shaving and an advancement mucosal flap, and definitive histologic examination confirmed the diagnosis of granulomatous cheilitis. No other signs of orofacial granulomatosis were observed, and the complementary aetiological study was negative for systemic disease. The lip swelling reappeared and persisted, which interfered with the quality of life. Hence, radiofrequency therapy was performed in the submucosal and subdermal layers of the lip, resulting in significant aesthetic and functional improvement and no further relapses after five years. DISCUSSION: The management of granulomatous cheilitis is challenging. The current mainstay treatment is corticotherapy or reduction cheiloplasty in severe cases. Radiofrequency has potential as a treatment option in debilitating macrocheilia, presenting worthy long-lasting functional and aesthetical results, with minimal morbidity.

Granulomatous Cheilitis Preceding Gastrointestinal Manifestations of Crohn's Disease.

ABSTRACT: We describe a 20-year-old male with no significant medical history who presented with a 1-month history of painless upper and lower lip edema initially treated with antibiotics for suspected cellulitis before arriving to the clinic. After a failed response to that treatment, a lip biopsy was eventually performed and consistent with a diagnosis of granulomatous cheilitis. In addition to oral and topical corticosteroids and tacrolimus, the patient adopted the cinnamon- and benzoate-free diet with some improvement of his lip swelling. Persistent mild tachycardia led to a cardiology referral for further evaluation and sarcoidosis workup. A gastroenterology consult was placed to correlate his presentation with Crohn's disease. The cardiology workup was noncontributory, and the patient was ultimately diagnosed with Crohn's disease after evaluation with laboratory studies and colonoscopy. This case highlights the need to evaluate for Crohn's disease in patients who present with granulomatous cheilitis, even in the absence of gastrointestinal symptoms, and the potential benefit of incorporating a cinnamon- and benzoate-free diet in treatment.

Granulomatous cheilitis of Miescher: a rare entity.

BACKGROUND: Granulomatous cheilitis (GC) is a rare entity of unknown etiology. It is a chronic inflammatory disorder with a predilection for young females. It is characterized by asymptomatic unrelenting swelling of lips. It is a monosymptomatic form or an incomplete variant of Melkersson-Rosenthal syndrome (a triad of recurrent orofacial swelling, relapsing facial paralysis, and fissuring of the tongue). CASE PRESENTATION: We herewith report a case of a 27-year-old female, presenting with persistent upper lip swelling, for 3 months. Biopsy from the lesion revealed features of granulomatous cheilitis. CONCLUSION: GC should be considered in the differential diagnosis of unrelenting swelling in the lip. Spontaneous remission is rare, and recurrences are common. Corticosteroids used for treatment provide temporary improvement.

Pathological and Immunohistochemical Characteristics of Granuloma and Lymphatics in Cheilitis Granulomatosa.

ABSTRACT: Cheilitis granulomatosa (CG) is an idiopathic, rare, and chronic granulomatous disorder involving the lips. We characterized the pathological and immunohistopathological findings of these granulomas and their relationship with the lymphatic vessels. Pathologically confirmed cases of primary CG from 2001 to 2016 were collected. Cases of inflammatory cheilitis without the presence of granuloma were included in the control group. Demographic data, clinical presentation, response to therapy, and pathological differences were compared. Periodic acid-Schiff and acid-fast stains excluded patients having infections. CD68, CD163, and D2-40 stains demonstrated features of granuloma, macrophage polarization, and the relationship between granuloma and lymphatic vessels. Thirteen patients diagnosed with CG were enrolled. Thirteen people were enrolled in the control group. The granulomas were either mononuclear or sarcoidal. They were predominantly positive for CD68 but negative for CD163. Perilymphatic granulomas were found in all patients. Intralymphatic histiocytosis and lymphatic dilatation were more commonly observed in patients diagnosed with CG than those in controls (54% vs. 15%, P = 0.03 and 92% vs. 23%, P < 0.01). TH1 immune response due to CD68+ M1 macrophages results in CG. Perilymphatic aggregation of macrophages and intralymphatic histiocytosis were important pathological clues for diagnosis.

An uncommon case of lip swelling: Granulomatous cheilitis associated with Crohn's disease.

Unbeknownst to most otolaryngologists, there is quite a range of oral manifestations which commonly manifest in the context of inflammatory bowel disease. As providers who will encounter such patients in consultation it is beneficial to be aware of that association. Lip swelling (granulomatous cheilitis) is just one such presentation, which is often otherwise mistaken for angioneurotic edema and can lead to unwarranted testing and misdirected treatment. We present such a case to highlight the educational value of this patient encounter.

Parálisis facial periférica recidivante como manifestación inicial del síndrome de Melkersson-Rosenthal / Recurrent peripheral facial palsy as an initial manifestation of Melkersson-Rosenthal syndrome

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The histopathological spectrum of Melkersson-Rosenthal syndrome: Analysis of 47 cases.

BACKGROUND: Melkersson-Rosenthal syndrome (MRS) is a rare disease characterized by the triad of recurrent orofacial edema, relapsing facial paralysis and plicated tongue. Histopathological features of MRS have not been extensively analyzed. METHODS: This study investigated the histopathological aspects of oral lesions from 47 MRS patients. RESULTS: The most common biopsy site was the upper lip, followed by the lower lip, gingiva and palate. The most important findings were ill-defined and well-formed granulomas. Lymphoplasmacytic inflammatory infiltrate was seen in early and late stages of MRS. Edema, fibrosis, vasodilatation and congestion were the most common finding in the lamina propria. Gingival and palate exams also demonstrated granulomatous infiltrates. Regarding the evolution time of the disease, we demonstrated that, in initial phases, there is a lymphoplasmacytic inflammatory infiltrates, followed by a granulomatous infiltrate and, subsequently, fibrosis. CONCLUSION: Histopathological examination of oral lesions is helpful for the diagnosis of MRS; the absence of granulomatous inflammation does not exclude the diagnosis of syndrome. Clinical and histopathological analysis of the rare gingival and palate lesions is important, since all histopathological findings of the disease were detected in these sites.

Aspectos clínico-patológicos de queilite granulomatosa: relato de caso / Clinical and pathological aspects of Granulomatosis Queilite: Case Report

Introdução:A queilite granulomatosa (QG) é uma lesão oral rara, apresentando clinicamente inchaço de aspecto granulomatoso no lábio.Objetivo:O presente estudo, tem como objetivo apresentar um relato de caso de QG, tendo em vista, a dificuldade do diagnóstico e tratamento de condições inflamatórias com etiologias desconhecidas que acometem a cavidade oral.Relato de caso:Paciente do sexo feminino, apresentou aumento de volume no lábio superior exibindo sintomatologia dolorosa, porém, sem relatar associações com alterações sistêmicas. Durante o exame clínico notou-se que o lábio superior apresentava fissuras profundas, observando tambémedema que se estendia da comissura labial, atravessando a linha média. Levantou-se então, a hipótese diagnóstica de QG. Foi realizada uma biópsia incisional para confirmação do diagnóstico. A análise histopatológica evidenciou, presença de granulomas não caseosos e infiltrado inflamatório linfoplasmocitário perivascular. De acordo com os dados clínicos e análise histopatológica foi dado o diagnóstico inicial de QG. Após o diagnóstico inicial de QG, a paciente foi encaminhada para uma avaliação médica, a fim de avaliar possíveis alterações intestinais que pudessem caracterizar a síndrome de Melkersson-Rosenthal ou Cronh, como também possível reação de hipersensibilidade tardia. Conclusões:O cirurgião-dentista deve estar preparado para diagnosticar aumentos de volume significativos nos lábios, a fim de manejar corretamente o tratamento do paciente, estabelecendo desta forma, prognósticos favoráveis para essas condições (AU).

Introduction:Granulomatous cheilitis (HQ) is a rare oral lesion, presenting clinically granulomatous swelling on the lip. Objective: This study aims to present a case report of HG, considering the difficulty of diagnosis and treatment of inflammatory conditions with unknown etiologies that affect the oral cavity.Objective:This study aims to present a case report of HG, considering the difficulty of diagnosis and treatment of inflammatory conditions with unknown etiologies that affect the oral cavity.Case report:A female patient presented increased volume in the upper lip showing painful symptoms, but without reporting associations with systemic alterations. During the clinical examination it was observed that the upper lip presented deep fissures, also observing edema that extended from the labial commissure, crossing the middle line. The diagnostic hypothesis of GQ was then raised. An incisional biopsy was performed to confirm the diagnosis. Histopathological analysis revealed the presence of non-caseous granulomas and perivascular lymphoplasmacytic inflammatory infiltrate. According to the clinical data and histopathological analysis the initial diagnosis of GH was given. After initial diagnosis of HG, the patient was referred for medical evaluation to evaluate possible intestinal changes that could characterize Melkersson-Rosenthal syndrome or Cronh, as well as possible late hypersensitivity reactions.Conclusions:The dentist should be prepared to diagnose significant volume increases in the lips in order to correctly manage the patient's treatment, thus establishing favorable prognoses for these conditions (AU).

Clinical and genetic heterogeneity in Melkersson-Rosenthal Syndrome.

Melkersson Rosenthal syndromes (MRS) is a rare autosomal dominantly inherited neurocutaneous syndrome characterised by a triad of facial (seventh cranial) nerve palsy, recurrent orofacial swelling and fissuring of the tongue. A recent report implicated a heterozygous missense variant in SLC27A1 (FATP1) as the cause of this condition in members of an affected Chinese family. We undertook Sanger sequencing of this gene in 14 affected unrelated individuals affected by MRS. We did not detect any putative pathogenic variants. Our data indicates that there is both clinical and genetic heterogeneity in this condition and that the causative gene remains to be identified for the majority of cases.

[Melkersson-Rosenthal syndrome in a young man].

A 24-year-old man presented with orofacial swelling, peripheral facial palsy and fissured tongue. Apart from a previous episode of peripheral palsy, he had no history of illness. Biopsy of the lip, blood tests, magnetic resonance imaging of the head and lumbar puncture showed no pathology. He received systemic prednisone for ten days, and follow-up showed sparse improvement in facial movements. The patient could have been misdiagnosed with Bell's palsy, but the symptoms rather indicated a case of Melkersson-Rosenthal syndrome. This rare syndrome is presenting in oligo- or monosymptomatic forms and is often mistaken for Bell's palsy. There is no existing gold standard of treatment, and different modalities have been considered with no consistent response.