Home About us Editorial board Ahead of print Current issue Search Archives Submit article Instructions Subscribe Contact Us Login 
An Official Publication of the Indian Association of Oral and Maxillofacial Pathologists


 
  Table of Contents    
CASE REPORT - LESIONS INVOLVING BONE  
Year : 2020  |  Volume : 24  |  Issue : 4  |  Page : 51-54
 

Infantile anterior maxillary swelling: A diagnostician's dilemma


1 Department of Pedodontics and Preventive Dentistry, Guru Gobind Singh College of Dental Sciences and Research, Burhanpur, Madhya Pradesh, India
2 Department of Oral Pathology and Microbiology, Subharti Dental College, Swami Vivekanand Subharti University, Meerut, Uttar Pradesh, India
3 Denta Care Super Speciality Dental Clinic, Gulbarga, Karnataka, India

Date of Submission16-Oct-2019
Date of Acceptance23-Oct-2019
Date of Web Publication28-Feb-2020

Correspondence Address:
Vijay Wadhwan
Department of Oral Pathology and Microbiology, Subharti Dental College, Swami Vivekanand Subharti University, Meerut, Uttar Pradesh
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jomfp.JOMFP_300_19

Rights and Permissions

 

   Abstract 


Anterior maxillary swellings are commonly encountered in the adolescents and adults and they represent lesions ranging from cysts to tumors which can be both benign as well as malignant. However the anterior maxillary swellings are a rare phenomenon in the infants and toddlers and they generally are indicative of an aggressive lesion. We hereby present a case of a rapidly growing infantile swelling which was histopathologically diagnosed as Peripheral Giant Cell Granuloma.


Keywords: Giant cells, maxillary anterior, swelling


How to cite this article:
Patil DB, Wadhwan V, Patil SD. Infantile anterior maxillary swelling: A diagnostician's dilemma. J Oral Maxillofac Pathol 2020;24, Suppl S1:51-4

How to cite this URL:
Patil DB, Wadhwan V, Patil SD. Infantile anterior maxillary swelling: A diagnostician's dilemma. J Oral Maxillofac Pathol [serial online] 2020 [cited 2020 Apr 7];24, Suppl S1:51-4. Available from: http://www.jomfp.in/text.asp?2020/24/4/51/279765





   Introduction Top


The diagnosis of anterior maxillary swellings is a challenging task for a clinician just based on the clinical and radiological appearance of the lesion. The anterior maxillary swellings can be a result of some developmental disturbance like inclusion cysts, inflammatory in origin or can be a reactive or a neoplastic process. Although the swellings are seen throughout life, the anterior maxillary swellings in infants are usually fatal. The benign lesions do occur in infants, but the number of malignant cases exceeds than the benign ones.

These swellings can be best examined by inspection and palpation clinically, and radiographs may be an adjunct in ruling out pathologies such as abscesses and periapical inflammatory conditions. Routine panoramic radiography can help to discover bony masses arising from the maxilla. However, the final diagnosis is achieved after the histological examination.[1]

This case report is an attempt to describe an unusual presentation of peripheral giant cell granuloma (PGCG) in a 1½-year-old infant.


   Case Report Top


Parents of a 1½-year-old infant reported to the outpatient department with a chief complaint of swelling in the anterior maxillary region which was rapidly growing for 2 months. The patient was a known case of congenital acyanotic heart disease, and he was on medication for 2½ months. On examination of lesion, proper obvious facial asymmetry was noticed due to eversion of lip. Mouth opening was inadequate. Regional lymph nodes were palpable, tender and mobile. The swelling was roughly oval in shape, and measured about 1.5 cm × 2.5 cm approximately and had erythematous surface [Figure 1]. On palpation, it was firm, mobile, had smooth surface and was bleeding on provocation. The radiological changes were not marked and only showed superficial erosion of the underlying alveolus. A clinical diagnosis of malignant mesenchymal tumor was made. Incisional biopsy was carried out and sent for histopathological examination. The H and E stained section showed parakeratinized stratified squamous epithelium with underlying extremely cellular connective tissue stroma with numerous blood vessels and proliferation of multinucleated giant cells [Figure 2]. The giant cells were distributed throughout the stroma and had nuclei ranging from 8 to 10 in number [Figure 3]. Few areas showed the presence of hemosiderin pigment and also inflammatory cells such as lymphocytes and plasma cells [Figure 4]. Plump-, ovoid- and spindle-shaped mesenchymal cells were also seen. Based on these findings, a histopathological diagnosis of peripheral giant cell granuloma was made. The lesion was surgically excised, and the healing was uneventful.
Figure 1: Clinical photograph showing a reddish lesion on the labial aspect of incisors extending onto the palate

Click here to view
Figure 2: H&E stained section showing parakeratinized stratified squamous epithelium overlying an extremely cellular stroma (×4)

Click here to view
Figure 3: H&E stained section showing multinucleated giant cells in background cellular stroma (×40)

Click here to view
Figure 4: H&E stained section showing lots of vascular spaces and vascular channels (×10)

Click here to view



   Discussion Top


PGCG or so-called “Giant cell epulis” is a common giant cell lesion. It normally presents as a soft-tissue purplish-red nodule consisting of multinucleated giant cells in a background of mononuclear stromal cells and extravasated red blood cells. This lesion probably does not represent a true neoplasm but rather may be reactive in nature, believed to be stimulated by local irritation or trauma, but the exact etiology is not known. Although the PGCG occurs throughout life, the peak incidence is seen during the mixed dentition period and in adults during third to fourth decade of life. It is more common among females (60%). The mandible and maxilla is affected with equal frequency.

Several hypotheses had been proposed to explain the nature of multinucleated giant cells including the explanation that they were osteoclasts left from physiological resorption of teeth. Lim and Gibbins in 1995 stated that the multinucleated giant cells reacted strongly for a monoclonal antibody MB1 which reacts with lymphocytes and a proportion of T-cells and monocytes, thus laying stress on the lymphocytic–monocytic origin of these giant cells.[2]

Wülling et al. in 2001 revealed that the stromal cells secrete a variety of cytokines and differentiation factors, including monocyte chemoattractant protein-1, osteoclast differentiation factor and macrophage colony-stimulating factor. These molecules are monocyte chemoattractants and are essential for osteoclast differentiation, suggesting that the stromal cell stimulates blood monocyte immigration into tumor tissue and enhances their fusion into osteoclast-like, multinucleated giant cells.[3]

Another hypothesis suggests that giant cells may arise from the fusion of endothelial cells of proliferating capillaries.[4]

The differential diagnosis for the most common lesions of the maxillary anterior region includes reactive hyperplasias, related to a number of chronic irritant stimuli. However, there are a number of entities that have a predilection for the gingivae, which are much less common in other parts of the oral cavity.

  1. Pyogenic granuloma constitutes 85% of all reactive gingival swellings, representing a profuse mass of vascular granulation tissue. In oral cavity, it occurs most commonly in the maxillary buccal and interproximal gingiva. It is usually highly vascular, fast-growing, elevated, lobular, sessile or pedunculated and commonly ulcerated or covered by pseudomembrane. The color varies from red to pink with tendency to bleed on slight irritation. It occurs at any age and sex with a slight predilection for young females.[5] The size ranges from few millimeters to centimeters but neither cause alveolar bone loss nor do they disturb tooth positioning [4]
  2. Peripheral ossifying fibroma - It originates from the periodontal ligament or the periosteum. This lesion has a predilection for females and is most common in young patients between 1 and 19 years of age.[6] It occurs exclusively on the gingiva, especially in the anterior gingiva, with slight predilection to the maxilla and rare presentation in primary teeth.[7] It usually presents as a well-demarcated sessile nodules, often ulcerated. It is of the same color as normal mucosa or red if ulcerated, with firm to hard consistency, depending on the amount of ossification and calcifications [4]
  3. Sarcomas – rhabdomyosarcoma and fibrosarcoma


    • Rhabdomyosarcoma is a malignant neoplasm of skeletal muscle origin accounting for 4%–8% of all malignant diseases in children <15 years of age. It primarily occurs in the first decade of life with a peak incidence between 2 and 6 years with slightly more predilection in males. The tongue, palate and cheek are the most common locations in the mouth.[8] Gingiva is the additional site of growth.[4] The clinical appearance ranges from a small nodule to an extensive mucosal outgrowth. It may present as a painless, yet occasionally painful, facial swelling [8]
    • Infantile fibrosarcoma is a rare malignant neoplasm of fibroblast origin. They usually develop within the first 2 years of life with slightly more male predilection. Only 10% occur in the head and neck area, including the oral cavity,[9] with common site of occurrence in the buccal mucosa, palate, lips and periosteum of the mandible and maxilla. Fibrosarcomas present as fast-growing, fleshly, exophytic ulcerated lesions with tooth displacement and bone and tooth resorption [9]


  4. Other less aggressive mesenchymal (infantile myofibromatosis and aggressive fibromatosis) lesions can also be considered when a lesion recurs in such a short period of time and especially in a child. They present as locally aggressive, firm, painless and poorly demarcated masses, that may be either rapidly growing or slowly growing [4]
  5. Lymphoma and leukemia have a nonspecific clinical presentation but often present with swelling and reddening of the gingival tissues. Advanced cases are likely to be accompanied by bone loss and tooth mobility. More localized swelling may be mistaken for pyogenic granuloma or giant cell epulis.[10]



   Conclusion Top


Although the present case in the anterior maxillary region was growing at an alarming pace and was causing discomfort to the patient, the diagnosis for the same turned out to be a reactive lesion. Wide local excision was carried out, and the healing was uneventful. Hence, a careful histopathological examination is mandatory for all oral lesions for proper treatment planning.

Acknowledgment

The authors wish to thank Dr Suhasini GP(Assitant Professor), Subharti Dental College, Meerut for helping in preparation of this manuscript.

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.



 
   References Top

1.
Patil S, Khandelwal S, Doni B, Rahman F. A review of 9 palatal swellings. JPDA 2013;22:134-9.  Back to cited text no. 1
    
2.
Lim L, Gibbins JR. Immunohistochemical and ultrastructural evidence of a modified microvasculature in the giant cell granuloma of the jaws. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1995;79:190-8.  Back to cited text no. 2
    
3.
Wülling M, Engels C, Jesse N, Werner M, Delling G, Kaiser E. The nature of giant cell tumor of bone. J Cancer Res Clin Oncol 2001;127:467-74.  Back to cited text no. 3
    
4.
Sivapathasundaram B, Satish Muthukumar R, Rajendran R. Non epithelial tumors of oral cavity. Shafer's Textbook of Oral Pathology 8th ed. India: Elsevier; 2016. p. 188-9.  Back to cited text no. 4
    
5.
Fantasia JE, Damm DD. Red nodular lesion of tongue. Pyogenic granuloma. Gen Dent 2003;51:190, 194.  Back to cited text no. 5
    
6.
Hanemann JA, Pereira AA, Ribeiro Júnior NV, Oliveira DT. Peripheral ossifying fibroma in a child: Report of case. J Clin Pediatr Dent 2003;27:283-5.  Back to cited text no. 6
    
7.
Cuisia ZE, Brannon RB. Peripheral ossifying fibroma – A clinical evaluation of 134 pediatric cases. Pediatr Dent 2001;23:245-8.  Back to cited text no. 7
    
8.
Chigurupati R, Alfatooni A, Myall RW, Hawkins D, Oda D. Orofacial rhabdomyosarcoma in neonates and young children: A review of literature and management of four cases. Oral Oncol 2002;38:508-15.  Back to cited text no. 8
    
9.
Grohn ML, Borzi P, Mackay A, Suppiah R. Management of extensive congenital fibrosarcoma with preoperative chemotherapy. ANZ J Surg 2004;74:919-21.  Back to cited text no. 9
    
10.
Brierley DJ, Crane H, Hunter KD. Lumps and bumps of the gingiva: A pathological miscellany. Head Neck Pathol 2019;13:103-13.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

Top
Print this article  Email this article
            

    

 
   Search
 
  
    Similar in PUBMED
    Search Pubmed for
    Search in Google Scholar for
  Related articles
    Article in PDF (1,928 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
   Introduction
   Case Report
   Discussion
   Conclusion
    References
    Article Figures

 Article Access Statistics
    Viewed39    
    Printed0    
    Emailed0    
    PDF Downloaded20    
    Comments [Add]    

Recommend this journal

Journal of Oral and Maxillofacial Pathology | Published by Wolters Kluwer - Medknow
Online since 15th Aug, 2007