Orofacial granulomatosis in children—A challenge for diagnosis and treatment

Kolho, Kaija‐Leena; Heiskanen, Kaarina; Verkasalo, M; Pitkäranta, Anne

doi:10.1016/j.ijporl.2011.03.003

Cited by 16 publications

(11 citation statements)

References 21 publications

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“…Systemic agents (including steroids, dapsone, hydroxyquinolone and sulfasalazine) failed to ameliorate the oral disease, but infliximab resolved most oral features, save for mild residual labial swelling (Peitsch et al , 2007). Although many cases of OFG with CD (or previous intestinal disease) may be responsive to infliximab, use of infliximab in one child with concomitant intestinal CD produced no benefit (Kolho et al , 2011). Subsequent to this report, the same authors trialled infliximab in two other children with CD and oral disease, and while in one case its use improved intestinal inflammation, there was little benefit seen in the oral disease (K‐L Kolho, personal communication).…”

Section: Orofacial CD and Ofgmentioning

confidence: 99%

Biologics in oral medicine: oral Crohn’s disease and orofacial granulomatosis

O’Neill¹,

Scully²

2012

Oral Diseases

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Antitumour necrosis factor (TNF-α) therapy has a potential to benefit patients with oral lesions of Crohn's disease (CD) and patients with orofacial granulomatosis (OFG). The most appropriate use would appear to be in patients with severe or multisystem features, where other available agents have failed or have been associated with adverse effects. TNF-α antagonists (infliximab in particular) have a role in the management of orofacial CD and OFG, but potential adverse effects of TNF-α antagonists include acute infusion reactions, infection and increased risk of malignancy. Thus, a full risk-benefit analysis is indicated, with patient selection, use and subsequent monitoring coordinated with gastroenterologists with appropriate training and experience in biological therapies.

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Section: Orofacial CD and Ofgmentioning

confidence: 99%

Biologics in oral medicine: oral Crohn’s disease and orofacial granulomatosis

O’Neill¹,

Scully²

2012

Oral Diseases

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“…About 50% of children with OFG would develop Chron’s disease in the future (10), although intestinal lesions may take up to 10 years to develop (11). These cases of CD progressing from OFG, initially included lip swelling and are frequently related to atopia, allergic rhinitis or asthma (12), which were not found in this clinical case.…”

Section: Discussionmentioning

confidence: 65%

Unifocal orofacial granulomatosis in retromolar mucosa: surgical treatment with Er,Cr:YSGG laser

et al. 2014

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Orofacial granulomatosis is defined by permanent or recurrent swelling of orofacial tissues with different multiform and multifocal clinical patterns. An 11-year old boy presented with a 2-month history of mucosa enlargement. Intraoral examination revealed an erythematous, polylobulated, exophytic lesion with a smooth surface located in retromolar mucosa, non-tender and non-infiltratated to palpation. The diagnosis was inflammatory lesion compatible with pyogenic granuloma and laser excision was decided. Haematological parameters were within normal range, as well as chest Xrays. These findings lead to a diagnosis of non-symptomatic orofacial granulomatosis, whose early diagnosis can minimize the impact of systemic-related disorders, like Chron’s disease. Key words:Laser, orofacial granulomatosis, childhood, oral lesions, diagnosis.

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“…Oral disease may predate intestinal CD involvement, especially in young patients. 9 Currently, OCD and OFG are separated on clinical grounds, where OFG tends to present with more labial pathology like isolated cheilitis and less buccal ulceration or cobblestoning. 8 Several drugs have been used for the management of granulomatous cheilitis, with very variable results, and there is no consensus regarding optimal treatment.…”

Section: Discussionmentioning

confidence: 99%

“…In Table 1 we summarize the characteristics of patients with granulomatous cheilitis, with or without coexistent CD, treated with biologics. 4,9,10,[13][14][15][16][17][18][19][20][21][22][23][24][25][26][27][28] There was a clear predominance of cheilitis in children and young adults (mean age 26 years, SD 15), and 55% of the patients were female. Dupuy et al have reported a greater male predominance and a young age of onset in CD patients who present with oral manifestations.…”

Section: Discussionmentioning

confidence: 99%

Recurrent granulomatous cheilitis associated with Crohn’s disease successfully treated with ustekinumab: case report and literature review

Taxonera

Alba

Colmenares

et al. 2020

Therap Adv Gastroenterol

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Granulomatous cheilitis, characterized by persistent inflammation of the lips and a granulomatous histology, is sometimes associated with Crohn’s disease and is a therapeutic challenge. Reported evidence indicates treatment with an anti-TNF agent (mainly infliximab) is the most recommended therapeutic option after failure of conventional treatments. The clinical case reported the effectiveness of ustekinumab, a monoclonal antibody against interleukins 12/23, to induce the remission of severe and recurrent granulomatous cheilitis in a patient with Crohn’s disease.

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Orofacial granulomatosis in children—A challenge for diagnosis and treatment

Cited by 16 publications

References 21 publications

Biologics in oral medicine: oral Crohn’s disease and orofacial granulomatosis

Biologics in oral medicine: oral Crohn’s disease and orofacial granulomatosis

Unifocal orofacial granulomatosis in retromolar mucosa: surgical treatment with Er,Cr:YSGG laser

Recurrent granulomatous cheilitis associated with Crohn’s disease successfully treated with ustekinumab: case report and literature review

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