Gingival enlargement unveiling Crohn's disease - A report of a rarefied case.

This article highlights a peculiar case of Crohn's disease (CD) with the primary presenting symptom as localized gingival overgrowth in the anterior region of maxilla. The patient underwent surgical treatment with external bevel gingivectomy procedure. Follow-up after 6 months revealed no recurrence. A final diagnosis of CD was made, after following histopathological evaluation of the gingival biopsy and other special investigations. It is thus crucial to associate the localized granulomatous gingival overgrowth to systemic signs of CD. Intraoral occurrence preceding intestinal involvement is very rare in CD with an incidence of about 8%-9% of patients. Therefore, precise diagnosis, treatment planning, and timely management are vital to the overall health and welfare of patients.

INTRODUCTION

Gingival overgrowths are reasonably common which can be induced by an array of etiological factors and are aggravated by the localized accumulation of bacterial plaque. Gingival enlargements are classified into several categories based on the etiologic factors and pathologic changes.[1] Several systemic diseases may develop oral manifestations that can result in gingival enlargement, but it is very rare. Chronic granulomatous diseases such as tuberculosis[2] and sarcoidosis[3] can manifest as gingival enlargement in the oral mucosa.

This article highlights a rarefied case with localized gingival enlargement diagnosed to be related to Crohn's disease (CD) which is successfully managed by means of surgical periodontal therapy. This case is unique because intraoral occurrence is uncommon in CD with an incidence of about 8%–9%.[4] In most of the patients, intestinal involvement precedes the oral signs and symptoms.[5] However, in our case, gingival enlargement is seen without any intestinal symptoms. In 5%–10% of the affected patients, oral lesion may be the initial presenting sign before gastrointestinal symptoms.[6]

CASE REPORT

A female patient, 45-year-old, came to the department of periodontics with a complaint of pain and gingival overgrowth in the upper front tooth region for the past 2 years. No systemic problems were revealed in patient's medical history. The patient experienced fatigue, weakness, and loss of appetite for 6 months but no cough with expectoration. The patient did not give any history of other gastrointestinal disturbances. The patient had average built on general assessment; furthermore, the vital signs were within normal range. On clinical examination, the extraoral findings revealed competent lips, and there was no lymph node enlargement. Intraoral examination revealed diffuse gingival overgrowth in the upper anterior region covering more than two-thirds of the tooth surfaces with pseudo pockets of about 5–8 mm [Figure 1]. The color of the gingiva appeared pale pink. On palpation, it was firm and nontender. Slight bleeding on provocation was noted.

Figure 1

Intraoral photograph showing diffuse gingival enlargement

Phase I therapy comprising of oral hygiene maintenance instructions, scaling, and root debridement was performed. During re-evaluation, the growth was persistent, and hence, incisional biopsy was done. The tissue was submitted for histopathological examination, which was suggestive of chronic granulomatous diseases. The differential diagnosis considered is orofacial granulomatosis, angioedema, tuberculosis, sarcoidosis, and CD. Laboratory investigations were undertaken to rule out various granulomatous diseases. Mantoux test and sputum test were negative for tuberculosis and Kveim test proved negative, which ruled out sarcoidosis. Complete blood count reports revealed an increase in erythrocyte sedimentation rate (33 mm/h) and her hemoglobin was 9% and the red blood cell counts were 2.9 cells/Cumm. The serum angiotensin-converting enzyme levels were 39.2 U/L. The X-ray of chest revealed no abnormalities. Since the patient was not willing to undergo further investigations, surgical intervention by conventional gingivectomy was planned to remove the excessive gingival tissue.

Surgical procedure

Informed consent was taken before the surgical procedure. The surgical site was anesthetized by local infiltration with 2% lignocaine containing 1:80,000 adrenaline. The depths of the pathological pockets were identified and at the level of the bottom of the pocket bleeding points were produced by a pocket marker. The primary incision (external bevel incision) was made with a No. 15 BP blade or a Kirkland knife No. 15/16 at a level apical to the bleeding points. The secondary incision through the interdental area was performed with the use of Orban knife No. 1 or 2. The incised tissues were carefully removed with curettes and tissue tags were removed and periodontal dressing given [Figure 2].

Figure 2

Intraoperative view showing external bevel gingivectomy

The excised tissue was sent to pathology department for histopathological analysis. Postoperative instructions, including antibiotics and analgesics, were given. Uneventful postoperative healing was observed. A 6-month follow-up showed no recurrence [Figure 3].

Figure 3

Postoperative picture showing well contour gingiva

Histopathological examination

Section stained with eosin and hematoxylin revealed stratified squamous epithelium with pseudoepitheliomatous hyperplasia and the underlying connective tissue shows diffuse chronic inflammation with focal aggregates of noncaseating epithelioid cell granulomas suggesting chronic granulomatous disease. Multinucleate giant cells were evident throughout the granuloma [Figure 4]. Since the histopathology once again revealed chronic granulomatous disease, the patient was referred back to the physician to rule out CD. The upper gastrointestinal tract endoscopy results were normal, but the colonoscopy report was suggestive of an early chronic granulomatous disease. On the basis of colonoscopy report, the case was finally diagnosed as CD. Its management is mainly based on the extent, degree of disease severity, and the individual patient situation. Since this patient had no gastrointestinal symptoms, she was kept under physician's follow-up with nutritional supplements alone.

Figure 4

Histopathological picture showing stratified squamous epithelium with pseudoepitheliomatous hyperplasia with diffuse multinucleated giant cells (×40)

DISCUSSION

CD also known as regional enteritis is a type of intestinal inflammatory disease which may involve any part of the gastrointestinal tract. The main gastrointestinal symptoms are pain in abdomen, vomiting, diarrhea, and weight loss. However, other than gastrointestinal complications, it can cause extraintestinal manifestations, namely skin rashes, arthritis, lesions of the oral cavity, tiredness, and lack of concentration.[7] Its incidence in the second and third decades of life is high. Colonoscopy along with ileoscopy and biopsy is worthy in the identification of CD.[8]. The distinguishing diagnostic colonoscopy findings comprise skip lesions, ulcerations, cobblestoning, and strictures. The additional diagnostic tests valuable in the finding of small bowel CD comprise capsule endoscopy, magnetic resonance enterography, and computed tomography enterography.[8]

Recent evidence showed a genetic link to CD. Mutations in the caspase-activating recruitment domain 15 gene also named as the nucleotide oligomerization domain 2 gene are linked with CD and to certain phenotype susceptibility of disease location as well as activity.[9] The intraoral findings related to CD were at first delineated by Dyer et al. in 1969.[10]

In the majority of CD patients, oral findings along with extraintestinal manifestations are more frequently seen though Plauth et al. did not support this in their outstanding review[11] In 8%–9% of patients, intraoral signs occur before intestinal involvement and oral signs may not reflect the intestinal disease activity and severity.[5] Amid the orofacial features have been mentioned in association with CD firm, nontender and persistent overgrowth of the labial or buccal mucosa is a common feature.[12] There was also a report of an unusual, granulomatous-appearing gingivitis, which was hyperplastic in the anterior labial or buccal mucosa with extension up to or above of the mucogingival margin.[13]

CONCLUSION

Gingival enlargement may occur due to various etiological factors. Diagnosis is often made with a careful history, clinical examination, and relevant investigations. Even though treatment of any gingival overgrowth is based on the etiology implicated, the importance of local factors such as plaque and calculus is considered as a modifying factor. Gingival hyperplasia may be the first sign in CD patients who are otherwise asymptomatic and may precede intestinal involvement. Hence, the dentist is in a unique position in the early diagnosis and treatment which is vital to the overall health as well as the welfare of patients.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.