Detailed Follow‐up Study of Pediatric Orofacial Granulomatosis Patients

Haaramo, Anu; Alapulli, Heikki; Aine, L; Saarnisto, Ulla; Tuokkola, Jetta; Ruuska, Tarja; Sipponen, Taina; Pitkäranta, Anne; Kolho, Kaija‐Leena

doi:10.1097/mpg.0000000000001554

Cited by 9 publications

(15 citation statements)

References 29 publications

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“…One-quarter of the patients reported suffering from angular cheilitis. In patients with OFG, angular cheilitis was found in half of the patients, and in patients with paediatric CD, it was the most frequently seen oral lesion, present in 15% of the patients [6,17]. However, angular cheilitis is not specific to CD or OFG, and it is also observed in otherwise healthy individuals.…”

Section: Discussionmentioning

confidence: 94%

“…In an earlier study, we investigated the connection between OFG and CD, and explored the factors leading to bowel disease in patients presenting with orofacial symptoms. We found that 72% of patients with paediatric-onset OFG developed a symptomatic bowel disease, namely CD, during a median of 3.1 years follow-up [6]. However, whether patients with MRS develop a symptomatic bowel disease in long-term follow-up has not been determined.…”

Section: Introductionmentioning

confidence: 86%

“…Primary symptoms are oedema of the lips and face, and ulceration in the mouth [5,6]. Patients with OFG may develop a symptomatic intestinal disease like Crohn's disease (CD), and patients with CD may develop oral lesions [7,8].…”

Section: Introductionmentioning

confidence: 99%

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A 30-year follow-up study of patients with Melkersson–Rosenthal syndrome shows an association to inflammatory bowel disease

et al. 2019

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Background: Melkersson-Rosenthal syndrome (MRS) is often classified under the term orofacial granulomatosis (OFG). A part of OFG patients eventually develop Crohn's disease (CD), but the relationship between MRS and CD is unknown. Goals: To evaluate the long-term outcomes of MRS patients, with specific interest in bowelrelated symptoms. Study: This follow-up study consisted of adult patients with MRSincluding the monosymptomatic form, cheilitis granulomatosa (CG)who had participated in our earlier MRS study (diagnosed after 1995 in Helsinki University Hospital). A phone interview was conducted with 27 patients (77.1% of the patients from the earlier study) and included questions about orofacial symptoms, facial palsy, intestinal symptoms, concomitant illnesses, medications, possible food avoidances and family history. Stool samples were collected to measure faecal calprotectin, a surrogate marker for intestinal inflammation. Results: The median follow-up time from symptom onset was 30 years. Three (11.1%) patients had developed inflammatory bowel disease (IBD); one CG patient developed CD, and two MRS patients with facial palsy developed ulcerative colitis. In addition, several other patients reported intestinal symptoms, but the examination of faecal calprotectin did not indicate intestinal inflammation. Conclusions: There was a connection between MRS and IBDnot only CD but ulcerative colitis as well. KEY MESSAGE Melkersson-Rosenthal syndrome (MRS) is a chronic condition often classified as a subtype of orofacial granulomatosis. Oral manifestations are common in MRS. We found a connection between MRS and inflammatory bowel disease, not only Crohn's disease but ulcerative colitis as well.

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Section: Discussionmentioning

confidence: 94%

Section: Introductionmentioning

confidence: 86%

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A 30-year follow-up study of patients with Melkersson–Rosenthal syndrome shows an association to inflammatory bowel disease

et al. 2019

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“…Both OFG and CD are intermittent with steady and acute phases. In OFG and oral CD, a predominant clinical finding is lip swelling followed by intra-oral features like hyperplastic gingivitis and cobblestoning, oral ulcers, and mucosal tags (Pittock et al 2001;Harty et al 2005;Al Johani et al 2010;Campbell et al 2011a;McCartan et al 2011;Haaramo et al 2017). However, it is reported that the overall discomfort, as well as the duration of symptoms and problems with appearance, are more severe in OFG than in oral CD patients (Gale et al 2015;Miest et al 2016).…”

Section: Discussionmentioning

confidence: 99%

“…Being a rare disease, no epidemiological data on OFG exist. Patient characteristics are mainly based on cohorts and case series (Mignogna et al 2003 ; Al Johani et al 2009 ; Saalman et al 2009 ; McCartan et al 2011 ; Gale et al 2015 ; Haaramo et al 2017 ). However, there seems to be an increase in the frequency of OFG in children and young adults in the Western world (Leao et al 2004 ; Saalman et al 2009 ; Campbell et al 2011a ; Lazzerini et al 2014 ) and a prevalence as high as 0.8% has been suggested in the Celtic population (McCartan et al 2011 ).…”

Section: Introductionmentioning

confidence: 99%

Orofacial granulomatosis: a questionnaire study among Norwegian dental clinicians

Skaare

Hovden²,

Herlofson

et al. 2020

Eur Arch Paediatr Dent

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Aims To evaluate awareness on orofacial granulomatosis (OFG) and oral Crohn disease (oral CD) among Norwegian dental clinicians. Methods A precoded questionnaire (QuestBack) was sent electronically to dentists and dental hygienists treating children and adolescents in the public dental service (PDS) in Norway. Data on the clinicians' knowledge of OFG and oral CD related to working experience were analysed by Chi square tests and bivariate logistic regression analyses. Results A total of 1097 clinicians were invited to participate, 778 dentists and 319 dental hygienists; 48.2% returned the completed form. Fifty-four percent of the participants had ≥ 10 year experience of clinical practice. Two-thirds (68.4%) of the dentists and all but one of the dental hygienists graduated in Norway. Approximately half of the respondents were aware of OFG (41.2%) and oral CD (57.8%). One-quarter (24.6%) reported that they most likely had seen a patient with OFG and 20.6% with oral CD. Recently graduated dentists (≤ 10 years ago) were more aware of OFG and oral CD than those who graduated > 10 years ago (p ≤ 0.001). Regarding dental hygienists, this difference was observed for OFG only (p < 0.05). Country of education did not affect the clinicians' reported knowledge. Approximately 90% would refer a patient suspected of having OFG or oral CD either to a dental specialist or to a physician. Conclusion The high prevalence of clinicians observing OFG and oral CD in this study may indicate that OFG and/or oral CD are under-reported and that OFG in particular is more common than hitherto believed. The high frequency of awareness was promising for the benefit of the patients.

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Oral Cavity

Betz¹,

Padilla²

2019

Practical Head and Neck Pathology

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Detailed Follow‐up Study of Pediatric Orofacial Granulomatosis Patients

Cited by 9 publications

References 29 publications

A 30-year follow-up study of patients with Melkersson–Rosenthal syndrome shows an association to inflammatory bowel disease

A 30-year follow-up study of patients with Melkersson–Rosenthal syndrome shows an association to inflammatory bowel disease

Orofacial granulomatosis: a questionnaire study among Norwegian dental clinicians

Oral Cavity

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