A five year old  girl  presented  with  a  slowly  growing  gingival enlargement of both  arches which became  pronounced  during  the eruption of deciduous  teeth. A  severe and  generalized diffuse gingival hyperplasia involving the   marginal,  interdental    and     attached   gingiva    was    observed    covering   almost    all   the  surfaces, of  all  the  teeth  except   the   incisal   edges  of   the   incisors. The occlusal surfaces of the molars were partially covered.  The gingival hyperplasia was more severe  in the maxilla  than in the mandible.  Familial   history  was   negative.  The  case  was  diagnosed   as   idiopathic  gingival    hyperplasia    based    on    the   history,  clinical features and histological findings. The gingival hyperplastic tissue was excised  under general anesthesia. Recurrence necessitated repeated surgery.

Gingival  enlargements  are  quite  common  and may be either inflammatory, non inflammatory  or  a combination of both. Idiopathic gingival  hyperplasia is a  rare condition of undetermined  etiology described variously as fibromatosis  gingivae, gingivaematosis,1 hereditary gingival fibromatosis2 idiopathic fibromatosis,3 familial  elephantasis4 and diffuse fibroma.5 Diffuse gingival enlargement is also found to be associated  with  syndromes like Cross   syndrome,  Rutherford syndrome, Ramen syndrome,  Zimmerman Laband syndrome and Juvenile  hyaline syndrome. The purpose of this case report was to highlight the etiological factors and treatment.

Case Report

A  five year  old  girl  was  brought  to  the  department of pedodontics with the chief  complaint of a slow  growing,  nontender  gingival  enlargement  since  birth. The parent   indicated  that  the enlargement became  more  pronounced   at  the  time  of eruption of  the deciduous  teeth.  There was no history of epilepsy or long term medication for any ailments. Developmental  milestones and other systems of the child were  normal. This was an isolated case in their family according to the parents.  There was  a history  of  surgical exposure of maxillary deciduous anteriors, when the  child  was  3  year  old.  The  other associated problems were difficulty in  speech, mastication and swallowing.

Examination

Extra oral examination revealed a convex facial profile with a bimaxillary protrusion, incompetent   lips  and a broad  nasal bridge.  Intraorally, severe  diffuse gingival enlargement involving the marginal, interdental and attached gingiva of both arches was observed, covering almost all the surfaces of the teeth. The enlargement covered partially the occlusal  surfaces  except the incisal edges of the primary anteriors (Fig. 2).

The gingival hyperplasia was more severe in the maxilla than the mandible, especially  posteriorly. The gingival hyperplasia in the  maxillary right posterior segment  was  almost  projecting  into  the  lower  right  vestibule (Fig. 1).  The color of the gingiva appeared normal with  secondary inflammatory changes in the posterior segment, especially on the occlusal surfaces.  Melanin hyperpigmentation was observed in  most  of  the  areas.  On  isolation  and  drying, the gingival surface appeared pebbled with  increased  stippling. The hyperplastic tissue was firm in consistency and non tender on palpation.  Due  to  the massive gingival enlargement, normal lip  closure was prevented, but with force the child  was  able to approximate the lips closely.

The child experienced difficulty in mastication and had developed an altered tongue position with abnormal swallowing and open occlusal relationship. Drooling  of saliva  was also noticed. Based on the history and clinical features,  the  case was diagnosed as idiopathic gingival  fibromatosis.

Investigations

Full mouth periapical radiographs and orthopantomogram were taken to rule out bony changes, displacement and resorption  of the teeth. Routine hematological investigations were also done. Findings from these investigations were within normal limits.

Histopahological Findings

Sections showed a hyperparakeratinized   hyperplastic stratified squamous epithelium with the underlying fibrous connective tissue  showing  bundles of collagen fibers, aggregates of chronic  inflammatory cells, few blood vessels and extensive areas of hemorrhage.

Treatment

After routine clinical and hematological examination, the maxillary and mandibular deciduous central and lateral incisors were extracted. The  child  was  reviewed  after 4 months  to see  if   the  hyperplastic  tissue  had  regressed.  Since  the  enlargement persisted, a  single sitting  full mouth gingivectomy procedure was done under general  anesthesia.  After  a  4 year  follow up, eruption  of 11,12, 21,22,31and 41 had occurred and recurrence of the gingival hyperplasia in the posterior segment of both  arches was observed (Fig. 3), necessitating a second stage surgery which was done. The  child is currently undergoing orthodontic  treatment for the anterior open bite and a habit breaking appliance has been placed for the correction of tongue thrust. The child is under follow up observation.

Idiopathic gingival fibromatosis may be congenital or hereditary. Though the genetic mechanism is not well  understood,  the  majority   of the authors of reported cases attributed the  condition to hereditary factors.  The mode of  transmission is mainly autosomal dominant.6,7 The first polymorphic marker for hereditary gingival  fibromatosis (HGF) phenotype in chromosome  2p21. Many cases are sporadic with no familial background.

Gingival  hyperplasia can occur after therapy with drugs like phenytoin, cyclosporine, nifidepine and nitrendipine. Long  term use of  these  drugs  has  to  be  ruled out. The incidence  of gingival  enlargement caused by phenytoin,8 an  anticonvulsant used in the treatment of epilepsy varies from 3 to 84.5%. whereas, cyclosporine a fairly potent immunosupressive agent used to prevent organ transplant rejection and to treat several disease  of  autoimmune  origin  induced  gingival enlargement in 30% of the cases.9 Nifidepine, which  is  a  calcium  channel  blocker  used  in  the treatment of acute and chronic coronary insufficiency, including  angina pectoris and refractory hypertension  and nitrendipine an analogue of nifedipine have also  been  reported to induced gingival enlargement.10,11

The condition may be associated with   physical developmental retardation and hypertricosis.12 Although gingival tissue may  appear normal at birth, hyperplastic gingival fibromatosis may become evident with the eruption of primary or permanent dentition,  suggesting a trauma - induced tissue reaction  during the eruption.13

Sometimes  gingival  enlargement  does  not occur until the eruption of the permanent dentition.  Further enlargement does not occur once the growth of jaw is completed.14 It  has  been  suggested that gingival enlargement may be  due  to nutritional and hormonal factors, but these  have not been completely substantiated. The constant increase in the tissue mass can result  in delayed eruption  and displacement of teeth, arch deformity, spacing and migration of teeth.15  The  condition  is  not  painful  until the tissue enlarges to partially cover the occlusal surface of the molars and become traumatized during mastication, which was observed in the present case.  Due to massive gingival enlargement, an affected child usually develops abnormal swallowing pattern and experiences difficulty in speech and mastication. Along with these features, there may  be some interference with the oral hygiene measures and normal mastication.  All  these  will  favor accumulation of materia alba and plaque, which further complicates the existing  hyperplastic tissue.  Maintenance of  good  oral  hygiene is very important. It is not known if  plaque control measures are effective in this condition, but it is good practice to maintain the plaque control following gingivectomy   procedure.

Histologically, the gingival hyperplasia is mainly due to an increase and thickening of mature collagen bundles in the connective tissue stroma.16 The nodular appearance can be attributed to the thickened para hyperkeratinized epithelium.17 Various modalities of treatment  had  been proposed including radical treatment with extraction of the involved teeth, which was reported not to favor a recurrence of the growth.16 The only treatment of choice in this  condition  was  gingivectomy to satisfy the  patient's  esthetics. Though  the  tissue  appeared  to  be  pale  and firm, the surgical procedure  was  complicated  with excessive  hemorrhage. Some have reported a case where apically positioned flap surgery and CO2 laser evaporation were used  to  reduce  the gingival tissue. Since recurrence could be  expected  within  a few  months after surgery and may return to the original condition  within  few  years, the patient  may have to undergo   repeated gingivectomy procedures. This often causes  further increase in the patients and parents'  psychological and emotional stress. Hence psychological counselling is a must for patients and parents.