Orofacial granulomatosis: an unsuccessful response to weekly azithromycin pulse therapy

Atkin, Philip A.; Simms, Melanie

doi:10.1016/j.oooo.2018.01.002

Cited by 5 publications

(5 citation statements)

References 13 publications

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“…Effective use of low‐level laser was described in a case of OFG with good clinical response 35 . Azithromycin was also used without clinical improvement 31 . Surgical cheiloplasty remains the only effective solution for unresponsive cases, 11 and also in our patient cohort, it gave good results in two patients.…”

Section: Discussionmentioning

confidence: 67%

“…Many medical therapeutic options have been described such as GCs, immunosuppressants (azathioprine, methotrexate, thalidomide, tacrolimus), anti-TB agents (ethambutol, isoniazid), antimalarials (chloroquine), anti-leprosy drugs (clofazimine and dapsone), metronidazole, anti-TNFα monoclonal antibodies (infliximab, adalimumab), sulfasalazine, antibiotics, and non-steroidal antiinflammatory drugs (NSAIDs). 1,2,7,12,25,26,[28][29][30][31][32][33][34][35][36][37][38] Among these therapeutic options, GCs are the most used.…”

Section: Trigeminal Neuralgiamentioning

confidence: 99%

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

Galdiero

Maio

Arcoleo

et al. 2021

Allergy

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Background Orofacial granulomatosis (OFG) is characterized by granulomatous inflammation of the soft tissues of maxillofacial region. We explored OFG patients from 10 different Italian centers and summarized the most recent literature data. Methods A review of patients with OFG was carried out. An extensive online literature search was performed to identify studies reporting diagnosis and management of OFG. Results Thirty‐nine patients were recruited between January 2018 and February 2020. Most of them (97.4%) displayed involvement of the lips, and 28.2% suffered from Melkersson‐Rosenthal syndrome. Two patients received diagnosis of CD and one patient of sarcoidosis, suggesting secondary OFG. Oral aphthosis and cervical lymphadenopathy were also described. The mean diagnostic delay was 3.4 years. Histological evaluation was performed in 34/39 patients (87.2%); non‐caseating granulomas were found in 73.5% of them. Neurological symptoms (28.2%), gastrointestinal symptoms in absence of overt inflammatory bowel disease (IBD) (20.5%), and atopy (35.9%) were also identified. Therapeutic approaches varied among the centers. Steroids (51.3%) were used with good or partial results. Anti‐TNF‐α and anti‐IgE monoclonal antibodies were used in 6 (15.4%) and 1 (2.6%) patients, respectively, with variable results. Surgery was the choice for 2 patients with good response. Conclusions OFG is a rare and neglected disease showing multiple clinical phenotypes. While early diagnosis is crucial, management is difficult and highly dependent on the expertise of clinicians due to the lack of international guidelines. There is a need to establish registry databases and address challenges of long‐term management.

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Section: Discussionmentioning

confidence: 67%

Section: Trigeminal Neuralgiamentioning

confidence: 99%

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

Galdiero

Maio

Arcoleo

et al. 2021

Allergy

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“…This is in line with another report showing short-term improvement in only one of five patients treated with azithromycin. 4 We achieved the best response with methotrexate, but only one patient had a durable remission (Fig. 1).…”

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confidence: 82%

Low response of granulomatous cheilitis to currently established treatments

Kreuter

Koushk‐Jalali

Oellig

et al. 2021

Acad Dermatol Venereol

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“…Orofacial granulomatosis (OFG) is a chronic inflammatory disease that is clinically characterized by lymphedema of the mouth and/or the face [1][2][3][4]. Lip edema is the most common finding (over 90%), although it is rarely the sole clinical feature.…”

Section: Introductionmentioning

confidence: 99%

“…The etiology of OFG is currently poorly understood. Causes are expected to be largely multifactorial [3,13,[15][16][17] with the potential participation of delayed hypersensitivity to food substances and food preservatives [5,18,19], microbial infections [20,21], inflammatory/immunologic dysregulation, genetics, and hereditary factors [1,7,22]. In addition, dental infections [23] and metals used for dental restorative materials (e.g., mercury [24][25][26], gold [25,27], cobalt and indium [28] constitutive of amalgam fillings and dental crowns) might also contribute to the initiation or worsening of OFG.…”

Section: Introductionmentioning

confidence: 99%

Gingival Orofacial Granulomatosis Clinical and 2D/3D Microscopy Features after Orthodontic Therapy: A Pediatric Case Report

et al. 2023

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Orofacial granulomatosis (OFG) represents a heterogeneous group of rare orofacial diseases. When affecting gingiva, it appears as a chronic soft tissue inflammation, sometimes combined with the enlargement and swelling of other intraoral sites, including the lips. Gingival biopsy highlights noncaseating granulomatous inflammation, similar to that observed in Crohn’s disease and sarcoidosis. At present, the etiology of OFG remains uncertain, although the involvement of the genetic background and environmental triggers, such as oral conditions or therapies (including orthodontic treatment), has been suggested. The present study reports the results of a detailed clinical and 2D/3D microscopy investigation of a case of gingival orofacial granulomatosis in an 8-year-old male patient after orthodontic therapy. Intraoral examination showed an erythematous hyperplasia of the whole gingiva with a granular appearance occurring a few weeks after the installation of a quad-helix. Peri-oral inspection revealed upper labial swelling and angular cheilitis. General investigations did not report ongoing extra-oral disturbances with the exception of a weakly positive anti-Saccharomyces cerevicae IgG auto-antibody. Two- and three-dimensional microscopic investigations confirmed the presence of gingival orofacial granulomatosis. Daily corticoid mouthwashes over a period of 3 months resulted in a slight improvement in clinical signs, despite an intermittent inflammation recurrence. This study brings new insights into the microscopic features of gingival orofacial granulomatosis, thus providing key elements to oral practitioners to ensure accurate and timely OFG diagnosis. The accurate diagnosis of OFG allows targeted management of symptoms and patient monitoring over time, along with early detection and treatment of extra-oral manifestations, such as Crohn’s disease.

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Orofacial granulomatosis: an unsuccessful response to weekly azithromycin pulse therapy

Cited by 5 publications

References 13 publications

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

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

Low response of granulomatous cheilitis to currently established treatments

Gingival Orofacial Granulomatosis Clinical and 2D/3D Microscopy Features after Orthodontic Therapy: A Pediatric Case Report

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