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Detailed Follow-up Study of Pediatric Orofacial Granulomatosis Patients
Anu Haaramo1, Heikki Alapulli, Liisa Aine
1*Department of Otorhinolaryngology, Head and Neck Surgery †Children's Hospital ‡Department of Oral and Maxillofacial Diseases, Helsinki University Hospital and Helsinki University, Helsinki §Department of Dentistry, Tampere University Hospital and Tampere University, Tampere ||Department of Gastroenterology, Tampere University Hospital and Tampere University, Tampere ¶Department of Gastroenterology, Helsinki University Hospital and Helsinki University, Helsinki, Finland.
Insights
Pediatric orofacial granulomatosis (OFG) patients show good long-term prognosis with no significant otorhinolaryngological issues. Elevated anti-Saccharomyces cerevisiae antibody A may indicate underlying Crohn disease (CD) in OFG patients.
Area of Science:
- Oral and Maxillofacial Medicine
- Pediatric Inflammatory Diseases
- Gastroenterology
Background:
- Orofacial granulomatosis (OFG) is a chronic inflammatory condition of the orofacial region.
- The association between OFG and Crohn disease (CD) remains debated.
- Understanding pediatric OFG and its potential links to CD is crucial for diagnosis and management.
Purpose of the Study:
- To characterize a cohort of pediatric patients with OFG.
- To assess the long-term course and prognosis of pediatric OFG.
- To identify predictors of concurrent Crohn disease in OFG patients.
Main Methods:
- A cohort of 29 pediatric OFG patients from two Finnish university hospitals underwent dental and otorhinolaryngological examinations.
- Orofacial findings were documented using digital photography and a structured schema.
- Patients completed questionnaires on general health and diet, and nutrition was assessed via food records. Comparisons were made between OFG-only and OFG with CD groups.
Main Results:
- Patients with CD exhibited more orofacial findings (median score 11) than those with OFG only (median score 7.5).
- The upper lip was more frequently affected in the CD group.
- Elevated anti-Saccharomyces cerevisiae antibody A levels were exclusively found in CD patients and correlated with increased orofacial findings.
Conclusions:
- Pediatric-onset OFG generally has a favorable long-term prognosis with minimal otorhinolaryngological comorbidity.
- Anti-Saccharomyces cerevisiae antibody A may serve as a potential biomarker for underlying CD in OFG patients.
- Further research is warranted to validate the predictive value of anti-Saccharomyces cerevisiae antibody A for CD in OFG.
Objectives:
Orofacial granulomatosis (OFG) is a chronic inflammatory condition affecting the orofacial area. Its connection to Crohn disease (CD) is debated. Our aim was to describe a cohort of pediatric patients with OFG in detail, study the long-term behavior of OFG, and evaluate factors predicting CD in patients with OFG.
Methods:
We invited patients diagnosed with OFG at 2 university hospitals, Finland for a follow-up appointment. Patients (n = 29) were examined by a dentist and an otorhinolaryngologist using a structural schema. Orofacial findings were also recorded using digital photographing. Patients filled in questionnaires about general health and special diets. Patients' nutrition was evaluated from food records. The findings were compared between patients with OFG only and OFG with CD.
Results:
Patients with CD had more findings in the orofacial area (total score for orofacial findings median 11) compared to patients with OFG only (total score median 7.5). There was no statistically significant difference in the type of lesions between these groups, except the upper lip was more often affected in patients with CD (n = 11) than in patients with OFG only (n = 0). Most of the patients had normal otorhinolaryngological findings. All patients with elevated anti-Saccharomyces cerevisiae antibody A levels had CD (n = 6) and they presented with more orofacial findings (total score) than patients with normal levels of anti-S cerevisiae antibody A (P = 0.0311).
Conclusions:
Long-term follow-up of pediatric-onset patients with OFG shows good prognosis. Patients with OFG do not seem to have otorhinolaryngological comorbidity. Anti-S cerevisiae antibody A may serve as a factor to indicate the possible presence of underlying CD in patients with OFG, but further studies are requested.
