Discussion
Oral manifestations may precede diagnosis of IBD; thus, the role of the dental practitioner is important in early detection, and collaboration between an oral specialist and gastroenterologist is important. We reported a case of an otherwise healthy young female with desquamative gingivitis and gingival overgrowth, which revealed granulomatous inflammation in the biopsy specimen (Figure 1). Granulomatous inflammation is rare in the oral cavity but can be seen in CD, sarcoidosis, foreign body reaction, perioral dermatitis, infectious disease, mycobacterial infections (tuberculosis), granulomatosis with polyangiitis, and orofacial granulomatosis6. Orofacial granulomatosis (OFG) is an uncommon granulomatous disorder in which systemic granulomatous diseases have been excluded. Cinnamon or bentzoate compounds, among others, have been suggested as playing an important role as hypersensitivities in OFG patients6.
The presence of oral manifestations that precede or follow intestinal symptoms of IBD must be taken into serious consideration by both dentists and gastroenterologists in order to allow early diagnosis. The prevalence of oral manifestations in IBD has been reported to range from 0.7% to 37% in adults and 7% to 23% in children.3,7 A healthy oral mucosa is dependent on several vitamins and minerals. The integrity of the oral mucosa may be compromised in IBD due to intestinal malabsorption, leading to a deficit of iron, zinc, or vitamin B12.
Oral manifestations of IBD were first reported in the 1950s and initially focused on aphthous ulcerations, which are the most common type of oral lesions in Crohn’s disease (CD), with a prevalence of 0.7% to 50% in adults.5,8 Aphthous ulcerations are strongly associated (p = 0.001) with the active phase of CD and they resemble the ulcers present in the gastrointestinal (GI) tract.9 Other oral manifestations characteristic of CD include swelling of the lips, cobblestoning or edema of the buccal mucosa, deep linear ulcerations, mucosal tags, and mucogingivitis.3,4,10,11 Pyostomatitis vegetans, a rare disorder characterized by friable pustules on the gingiva and mucosa, has been described in connection with IBD and PSC and liver disease.12 Pyostomatitis vegetans is more often associated with UC than CD.3 The most common non-specific manifestations, such as aphthous stomatitis and angular cheilitis, occur in both diseases. Non-specific lesions in the oral cavity can also be the result of malnutrition and drugs. These oral manifestations may be either asymptomatic or symptomatic with pain and impairment of oral function.4,13
Caries and periodontitis are also common oral diseases in CD.3 Over 50 systemic diseases or conditions are associated with periodontal diseases, including inflammatory bowel diseases.14 In a meta-analysis by Nijakowski et al, the risk of periodontal disease in IBD patients was almost two and a half times more than that of controls.15 Dry mouth, dysphagia, taste alterations, and halitosis have also been reported.3 As said, oral manifestations may precede diagnosis of IBD, and they can significantly impact the quality of life (QoL) of IBD patients, sometimes more so than the intestinal disease itself.5 In a study by Rikardsson conducted in Sweden, CD patients themselves also perceived their oral health to be worse and have a greater need for dental treatment compared to a control group. That study comprised 1943 patients with CD recruited from the Swedish National Patients Organization of IBD and 1000 randomly selected controls. Patients with CD reported significantly more mouth-related problems than controls (OR 3.2), such as significantly more caries and more gingival bleeding.16
IBD should be considered a systemic disease, since extraintestinal manifestations (EIMs) present in 5% to 50% of all IBD cases. Some EIMs correlate with intestinal disease activity, other manifestations are activity independent, such as primary sclerosing cholangitis (PSC).54,17,18 Musculoskeletal manifestations such as spondyloarthopatia, cutaneous manifestations such as oral aphthous lesions, ocular manifestations such as uveitis, pulmonary, renal, and urological manifestations, neurological manifestations, anemia, osteopenia, osteoporosis and hepatobiliary manifestations such as primary sclerosing cholangitis (PSC), autoimmune and granulomatous hepatitis, and fatty liver disease should be kept in mind.5 PSC is the most common hepatobiliary manifestation of IBD, since 75% of PSC patients are diagnosed with IBD. Intestinal diseases might occur years before the liver disease is diagnosed. UC is far more common, since 90% of PSC patients with IBD have UC and only 10% have CD. Still, only about 3% to 8% of colitis patients may have PSC, which results in inflammation and fibrosis of the intra- and extrahepatic biliary tract.17 PSC is also associated with a high risk of cholangiocarcinoma.
The frequency for at least one EIM varies between 6% and 47 %. Just one EIM seems to increase the risk for developing further manifestations. Autoimmune and genetic factors (HLA) may have an important role for EIMs.18
In the present case, a healthy young female with no abdominal or other systemic symptoms presented desquamative gingivitis and gingival overgrowth with granulomatous inflammation in a biopsy. However, initial gastro- and colonoscopy with biopsies and laboratory tests turned out normal. Oral mucosal lesions (OMLs) were followed up on a regular basis. In addition to the existing mucogingivitis, linear ulceration and tissue tags with pain were noted later on. After four years of initial symptoms, she developed diarrhea and fecal calprotectin was elevated (186 ug/l). She was referred to a gastroenterologist for further investigation, and IBD, namely Crohn´s disease, was diagnosed together with primary sclerosing cholangitis (PSC). Early recognition and follow-up are, thus, crucial, and a dental practitioner may be the first clinician to suspect IBD.