Case Report: Enlarging symmetrical masses of the palate of idiopathic etiology

Introduction

Idiopathic fibrous hyperplasia is a rare benign condition, characterized by a slow and progressive increase in gingival volume^1,2. It manifests as a rosy swelling of hard consistency while, at histological examination, it is characterized by a proliferation of fibroblasts in a myxomatous stroma. We describe and discuss a case of idiopathic fibrous hyperplasia of the palate.

Case

A 33 year-old woman came to our attention with slowly enlarging exophytic masses of the palate, which had begun to grow 2 years before and caused her disturbances of phonation as well as in swallowing solids and liquids. The patient did not take any drugs; however she had been a frequent user of nonsteroidal anti-inflammatory drugs for the last 3 years because of chronic back pain. Remote personal and family histories were negative, except for recurrent gastric nuisance and back pain.

Clinical and rhinoscopy examination demonstrated bilateral and symmetrical exuberant hypertrophic tissue, of hard consistency and rosy color, at the posterior-later area of the palate, with a tendency to coalesce medially. This tissue was contiguous to the adjacent gingiva (Figure 1).

Figure 1. Clinical aspect: Enlarging symmetrical masses involving the palate.

Computerized tomography scan showed such lesions to be limited to the mucosal palate, with no underlying bone involvement (Figure 2).

Figure 2. Computerized tomography scan: Soft tissue masses, with absence of underlying bone involvement.

An incisional biopsy demonstrated a sub-epithelial fibrous proliferation with packed collagen bundles and increased fibroblasts number (Figure 3). We concluded for a diagnosis of localized idiopathic fibrous hyperplasia. We treated the patient with a personalized approach using surgical resection and insetting of a thermal-printed palate plaque (Figure 4). We obtained good functional results at 20 days post-op (Figure 5), and no sign of recurrence at the 12 months follow up (Figure 6).

Figure 3. Histologic aspect: Hematoxylin & eosin (magnification, x10).

Consistent with idiopathic fibrous hyperplasia.

Figure 4. Custom made, thermal printed plaque.

Figure 5. Clinical aspect, plaque in place after hyperplasia removal.

Figure 6. Clinical aspect, 12 months post-op: Satisfactory functional and cosmetic outcome and absence of recurrence.

Discussion

Two main subtypes of fibrous hyperplasia are known: generalized and localized. Generalized form has a genetic predisposition, appears at decimal or definitive dental eruption, and usually demonstrates a tendency to recur after surgery^1–4. Secondary forms associated to pregnancy, scurvy, leukemia and drugs are also known^1–3. Among the latter, various chemotherapy agents can elicit secondary forms⁵, including ipilimumab and vemurafenib⁶. However, no reports link gingival fibrous hyperplasia to the drugs administered in our patient. The localized form has its onset from the second decade, does not generally recur after surgery, and normally is not associated to genetic predisposition,^7,8 although investigations to exclude syndromes commonly associated to gingival fibromatosis should always be carried out in our experience; these include Laband, Rutherfurd, Cross and Ramon syndromes⁵. In both the localized and the generalized forms, local factors such as dental plaques, caries, and the action of chemical substances and their metabolites might contribute to the onset in susceptible patients⁶. The precise pathogenic mechanism of idiopathic forms is unknown, but it appears to confine to the gingival and mucosal fibroblasts with no involvement of the periodontal ligament or the palate underlying bones^1,2.

Regardless of the etiology, excess tissue removal is the treatment of choice in localized fibrous hyperplasia, either by scalpel or by CO₂ laser^7,8. Our personal approach includes the use of a thermal-printed palate plaque (Figure 4), to be left in place for 20 days after surgery (Figure 5), which in our experience yields excellent hemostasis by exerting compression and, at the same time, functions as a scaffold that promotes and guides second intention healing, preventing recurrence of exuberant tissue growth. The long term (12 months) results reported in our case testify to the efficacy of such an approach.

Consent

Written informed consent for publication of their clinical details and clinical images was obtained from the patient.