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.