Case report
An otherwise healthy 18-year-old female was referred for gingival
overgrowth and desquamative gingivitis to the Department of Oral and
Maxillofacial Diseases Head and Neck Center, Helsinki University
Hospital, Helsinki, Finland.
The patient did not have any systemic diseases, medications, or
gastrointestinal symptoms; neither did she smoke or use alcohol
regularly. She had an allergy to cats, dogs, and horses. There was no
family history of IBD or other autoimmune diseases.
Histopathological examination of a biopsy from gingival overgrowth in
the right incisal-premolar region of the maxilla revealed normal surface
epithelium with a slight decrease in cell cohesion. Under the
epithelium, among the inflammatory cells, granuloma structures with
histiocytic and multinucleated giant cells were seen (August 2017)
(Figure 1). Having found this granulomatous inflammation, the
pathologist raised the possibility of Crohn’s disease. Consequently, the
patient was referred for gastro- and colonoscopy with biopsies, which
all turned out normal. Liver and kidney laboratory tests, including
celiac antibodies and CRP, were normal. Microcytic anemia was considered
to be associated with menstruation and was treated with iron
supplementation. Sarcoidosis was ruled out by an ear, nose, and throat
specialist.
The first appointment at the Department of Oral and Maxillofacial
Diseases Head and Neck Center, Helsinki University Hospital was one year
after initial examinations (August 2018). The patient still had no
gastrointestinal symptoms at that time. According to the patient, the
only symptom was bleeding gums when brushing her teeth. The patient
brushed her teeth twice a day with fluorine toothpaste, but interdental
cleaning was not performed regularly. Non-foaming, sodium lauryl sulfate
free toothpaste and interdental cleaning with a silicone brush were
recommended. Eating and drinking habits were recorded in more detail.
The patient consumed cola drinks three times per week; accordingly, she
was asked to stop drinking cola drinks with benzoate compounds.
Extra- and intraoral clinical examination with panoramic tomography was
performed. Extraoral findings were normal; facial skin and lips were
healthy. Intraorally, the interproximal gums were swollen, with some
periodontal pseudopockets 4 mm deep. The bleeding on probing (BOP) index
was 18%, measured from six sites per tooth. There were no infection
foci, no alveolar bone loss, no caries, and no periapical lesions in
panoramic tomography. Professional anti-infective treatment was
performed, and oral self-care instructions were given. After five
months, oral self-care was improved and the BOP index dropped under
10%; however, swollen gums in interproximal areas were still seen in
the upper jaw. A biopsy was taken in March 2019 from the marginal
gingiva in the maxillary canine region, revealing chronic inflammation.
Clinical examination with new laboratory tests were carried out after
six months (October 2019). Clinically, the lower lip was slightly
swollen, angular cheilitis with cracks in the corners of the mouth was
seen (Figure 2A), and gingiva in right upper maxilla was swollen (Figure
2B). Other oral mucosal lesions (OMLs) included small tissue tags in the
sulcus in the lower jaw (Figure 2C) and in the base of the mouth. In
laboratory tests, complete blood count, antinuclear antibody (ANCA), and
angiotensin-converting enzyme (ACE) turned out to be normal, but fecal
calprotectin, which reflects inflammation in the colon and is a useful
marker for IBD, was slightly elevated – 115 μg/g (reference <
100 μg/g). The patient had a gastroenterology consultation, but since no
intestinal symptoms were found, and calprotectin was only slightly
elevated, no further examinations were made at that point. Oral mucosal
monitoring was recommended in our department.
A new biopsy was taken in November 2019 from the buccal sulcular fold
and revealed chronic inflammation. Since the gums still bled easily,
some extra laboratory tests (S-Ferrit, P-TfR, S-B12-TC2, fS-Folaat, P-D
25) were made. Ferritin (S-Ferrit) 11 μg/l (15–125 μg/l), and vitamin D
23 nmol/l (> 50 nmol/l) were under the reference values.
Ferritin and vitamin D supplementation were recommended. Still, no other
symptoms except bleeding on brushing were noticed by the patient.
Laboratory values were controlled, and both ferritin and vitamin D
values were corrected.
The patient was closely followed up in our clinic, and supportive
periodontal therapy was arranged on regular basis.
Almost four years after oral granulomatous inflammation was first
diagnosed, the patient complained of abdominal pain for the first time
(February 2021). Beginning autumn 2020, she suffered from diarrhea twice
a week. The gums were swollen and the mouth was sore, she had cracked
lips and angular cheilitis. Intraoral examination revealed swollen
marginal hyperplastic strawberry-like gingivitis and slightly folded
mucosa in the sulcus areas of the maxilla (Figure 2D-E). The gingiva
bled easily on probing. A biopsy was taken from the swollen, inflamed
area of the left premolar area of the maxilla. Histopathologically,
granulomatous inflammation was diagnosed. Fecal calprotectin was
elevated – 186 µg/l. fS-ACE, P-Ferrit, S-D-25, and ANCA were within the
reference limits. See Table 1 for details for main oral and systemic
symptoms, manifestations, and pathology, laboratory and endoscopy
findings.
A colonoscopy performed in April 2021 showed segmental mild inflammation
in the colon and rectum. Mucosal biopsies confirmed a diagnosis of
Crohn’s disease. Gastroscopy was normal. Magnetic resonance imaging
(MRI) revealed an uncomplicated and asymptomatic perianal fistula. The
patient was started on standard intravenous infliximab. A repeated
colonoscopy in March 2022 showed moderate pancolitis, and the patient
was shown to be a non-responder to infliximab therapy. In addition, oral
lesions (sore mouth, lips and corners of the mouth cracking easily,
swollen gingiva/folded mucosa) persisted (Figure 2F-I). She was switched
to subcutaneous ustekinumab in May 2022.
At the time of the diagnosis of Crohn’s disease, alkaline phosphatase
(ALP) was elevated – 425 U/L (reference 35–105 U/L) – and alanine
aminotransferase (ALT) was 214 U/L (reference < 35 U/L). A
liver biopsy showed findings consistent with primary sclerosing
cholangitis (PSC). An endoscopic retrograde cholangiography (ERC) in
November 2021 showed slight biliary narrowing of the common hepatic duct
and choledochus, in line with mild PSC.