Orofacial granulomatosis in children: Think about Crohn's disease

Lazzerini, Marzia; Martelossi, Stefano; Cont, Gabriele; Bersanini, Chiara; Ventura, Giovanna; Fontana, Massimo; Decorti, Giuliana; Ventura, Alessandro; Taddio, Andrea

doi:10.1016/j.dld.2014.12.012

Cited by 19 publications

(21 citation statements)

References 14 publications

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“…Eight studies [10,15,16,18,22,26,35,36] reported on thalidomide efficacy after failure of either infliximab or adalimumab. Of the 73 patients identified, 47 (64.4%) had a clinical response and 37 (50.7%) achieved clinical remission (Table 7).…”

Section: Resultsmentioning

confidence: 99%

Thalidomide for inflammatory bowel disease

et al. 2016

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Section: Resultsmentioning

confidence: 99%

Thalidomide for inflammatory bowel disease

et al. 2016

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“…There is strong evidence of the association between CG and CD, where 10% to 48% of CG cases were associated with CD. [7][8][9][10] It has even been postulated that CG, OFG, and MRS are oligosymptomatic manifestations of CD. 11 The estimated prevalence of CG is 0.08% in the general opulation, with no predilection for race, sex, or age.…”

Section: Discussionmentioning

confidence: 99%

Oral lymphangiectasia and gastrointestinal Crohn disease

et al. 2020

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Lip edema with non-caseating granulomas or lymphangiectasia pose a clinical and pathological challenge. These findings can be attributed to cheilitis granulomatosa (CG), Melkersson-Rosenthal syndrome (MRS), or Crohn disease (CD) depending on the appropriate clinical context. Lymphangiectasis, in particular, is a common pathological finding in CD due to lymphatic obstruction by granulomas and intralymphatic granulomas. Because oral symptoms can precede gastrointestinal symptoms of CD or be seen in patients with asymptomatic gastrointestinal disease, the identification of lymphangiectasia should raise the possibility of underlying CD. We present a case of a young woman with several years of lip swelling, with notable lymphangiectasia and subtle granulomas on pathological evaluation. The patient was diagnosed with MRS at an outside institution and treated with systemic steroids, without further systemic evaluation. We believe that early recognition of lymphangiectasia and consideration of CD early in the work-up are critical for early diagnosis and appropriate management. Neither clinical nor histopathological findings should be used in isolation to diagnose GC, MRS, or CD as there is significant debate as to the etiology and overlapping findings of these conditions.

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“… 18 Both physical examination and biopsy of oral lesions are can not distinguish OFG, MRS or CD. 2 , 4 , 11 , 19 …”

Section: Discussionmentioning

confidence: 99%

“… 1 - 3 There is no clear consensus on the definition of OFG, however, the main concern is to rule out CD. 4 …”

Section: Introductionmentioning

confidence: 99%

Orofacial granulomatosis and diet therapy: a review of the literature and two clinical cases

Espinoza

Navarrete

Benedetto

et al. 2018

An. Bras. Dermatol.

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Orofacial granulomatosis is a nonspecific term that contains a wide variety of granulomatous entities, which share a clinical and histopathological presentation. It manifests as persistent or recurrent orofacial swelling, amongst other findings. Idiopathic orofacial granulomatosis, characterized by an absence of systemic granulomatous disease, is a diagnosis of exclusion. The main differential diagnosis is Crohn's disease. Its pathogenesis is unknown, however, it seems to be immune-mediated. Patch-test sensitivity to multiple allergens is well documented. Currently, therapeutic options consider restrictive diets, topical, intralesional, and systemic agents. First-line therapy is currently a matter of debate. We present a review of the value of diet therapy in this syndrome, along with two illustrative cases.

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Orofacial granulomatosis in children: Think about Crohn's disease

Cited by 19 publications

References 14 publications

Thalidomide for inflammatory bowel disease

Thalidomide for inflammatory bowel disease

Oral lymphangiectasia and gastrointestinal Crohn disease

Orofacial granulomatosis and diet therapy: a review of the literature and two clinical cases

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