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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 15  |  Issue : 2  |  Page : 317-319

Monostotic fibrous dysplasia of mandible: A report of a case


1 Department of Oral and Maxillofacial Surgery, Sharad Pawar Dental College and Hospital, Datta Meghe Institute of Medical Sciences (Deemed to be University), Wardha, Maharashtra, India
2 Department of Oral and Maxillofacial Surgery, Sharad Pawar Dental College and Hospital Datta Meghe Institute of Medical Sciences (Deemed to be University), Wardha - 442 004, Maharashtra, India

Date of Submission16-Apr-2020
Date of Decision30-Apr-2020
Date of Acceptance15-May-2020
Date of Web Publication21-Dec-2020

Correspondence Address:
Dr. Hardik Girish Karia
Department of Oral and Maxillofacial Surgery, Sharad Pawar Dental College and Hospital Datta Meghe Institute of Medical Sciences (Deemed to be University), Wardha - 442 004, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdmimsu.jdmimsu_129_20

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  Abstract 


Fibrous Dysplasia (FD) is a benign, congenital, skeletal, and metabolic entity of immature bone-forming mesenchyme that is characterized as a defect of differentiation and of maturation of osteoblasts of unknown etiology, wherein pathogenesis is uncertain, and which shows varied histopathology. FD represents all bone tumors and all benign tumors of about 2.5% and over 7%, respectively. FD commonly involves long bones, pelvis, ribs, and craniofacial bones. Facial and cranial bones are affected in nearly 50% of polyostotic FD patients and in 10%–27% of monostotic FD (MFD). The mandible is less commonly involved than the maxilla in MFD jawbone lesions. Clinical examination revealed tender firm to hard buccal cortical expansion. Radiographic examination revealed mixed radiopaque and radiolucent picture giving ground-glass appearance. Bone biopsy was performed for confirmatory diagnosis of FD. The lesion was subjected to surgical curettage. The patient was kept on regular follow-ups once every 3 months up to 3 years and then once every year till the 5th year. At the conclusion of 5 years, the patient was disease free. The purpose of the current report is to enlighten the case of a 32-year female patient complaining of painful bony swelling over the left back region of the lower jaw successfully treated with surgical curettage.

Keywords: Curettage, fibrous dysplasia, mandible, monostotic, polyostotic


How to cite this article:
Karia HG, Jadhav A, Bhola N, Hande A. Monostotic fibrous dysplasia of mandible: A report of a case. J Datta Meghe Inst Med Sci Univ 2020;15:317-9

How to cite this URL:
Karia HG, Jadhav A, Bhola N, Hande A. Monostotic fibrous dysplasia of mandible: A report of a case. J Datta Meghe Inst Med Sci Univ [serial online] 2020 [cited 2021 Jan 24];15:317-9. Available from: http://www.journaldmims.com/text.asp?2020/15/2/317/304239




  Introduction Top


Lichtenstein[1] first described fibrous dysplasia (FD) as a progressive congenital disorder which is characterized by intermixed, thickened, and woven growth of immature and fibrous tissue with normal cancellous bone.[2]

FD represents all bone tumors and all benign bone tumors up to 2.5% and over 7%, respectively.[3] FD involves the facial and cranial bones in nearly 50% of polyostotic FD (PFD) patients and in 10%–27% of monostotic FD (MFD) patients. The maxilla is commonly involved than the mandible with male predilection in MFD jawbone lesions, which manifests usually before the third decade of life. Most FD starts slowly, and the initial maxillofacial signs and symptoms are not clear, so possibly, they are ignored by patients. Since it shows slow progression, symptoms such as loosening of teeth and occlusion disharmony can be seen. MFD of severe form results in deformity in the maxillofacial region, giving esthetic impact, and also, it brings a certain psychological burden to patients. There is obvious pain if the lesion is associated with infection. When there is invasion of nerve, then it may affect hearing and vision and may show varying degrees of facial paralysis. If there is invasion into the sinus cavity, it results in nasal congestion. Moreover, if the lesion is massive, it may lead to pathological fractures of the maxillofacial region, and also, symptoms may develop of craniocerebral origin.[4] The present case exhibited the rarity of MFD in the mandible in a female patient. The aim was to highlight curettage as a treatment modality for complete removal of tumor without jeopardizing the bony continuity.


  Case Report Top


A countryside 32-year-old female visited at the Outpatient Department of Oral and Maxillofacial Surgery, Sharad Pawar Dental College, Sawangi, Wardha, with a complaint of painful slow-growing swelling over the lower left side of the jaw for 2 years. The medical history was not contributory. Root canal treatment was done with teeth 35 and 36 [Figure 1]a 3 years ago. The general physical examination was unremarkable. Extraorally, mild asymmetry was noticed over the left side of the jaw. Intraorally [Figure 1]b, the overlying mucosa was intact and normal and color same as that of adjacent mucosa. The affected area was tender and hard in consistency. The patient was further subjected to orthopantomograph (OPG) [Figure 1]c and three-dimensional computed tomography scan [Figure 1]d. OPG examination revealed mixed radiopaque and radiolucent picture giving ground-glass appearance. Considering clinical and radiographic examination, bone biopsy was demanded to confirm the diagnosis. Alkaline phosphatase levels were within normal limits (44–147 IU/L). H and E section [Figure 1]e of decalcified section revealed immature trabeculae in the arc-shaped pattern resembling a Chinese letter. The trabeculae were not lined by osteoblasts. The connective tissue was very sparse. The lesional tissue was blending with the surrounding normal bone.[5]
Figure 1: (a) Intraoral Periapical Radiograph (IOPA) showing root canal with 35 and 36. (b) Preoperative intraoral image. (c) Preoperative orthopantomograph showing mixed radiopaque and radiolucent lesion in the left mandibular body region. (d) Three-dimensional image showing buccal cortical expansion over the left mandibular body region. (e) Pictogram showing histopathological features of immature trabeculae in the arc-shaped Chinese letter pattern

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Since buccal cortical expansion was present and the patient was middle aged, surgery was undertaken where “surgical curettage” of the lesion was done, and to avoid pathological fracture, titanium plate and screw was adapted over the lower border of the mandible and closure was done in layers. Intentional root canal treatment was done with 37 postoperatively, and the patient was discharged and regularly followed up at 1 week, 1 month, every 3 months up to 3 years, and then once every year till the 5th year.


  Discussion Top


FD is majorly divided into three types: “monostotic” when single bone involved; “polyostotic,” multiple bones involved with multiple lesions; and “McCune–Albright syndrome,” type of PFD involving endocrine abnormalities.[6],[7]

MFD is most common, with a distinctive male predilection showing 70% of cases mostly in puberty and up to 50% female predilection in PFD, which involves craniofacial bones. The exhibited case presented with involvement of the mandibular bone in the middle-aged female patient, which is a rare finding in the MFD. It is usually observed in children and young adult patients, with 75% of them presenting before 30 years of age. The differential diagnosis of FD includes other fibro-osseous lesions such as cherubism, having various features which distinguish it from FD. Florid FD, Aneurysmal bone cyst, Giant cell granuloma, are often confused with FD. Ossifying fibroma which is a true bone tumour, contains variable mixture of bony trabeculae, cementum like spherules or both should be distinguished from other lesions. Cherubism, a benign giant cell lesion of jaw, which is, hereditary autosomal dominant pattern that shows painless bilateral swelling in childrens, usually between the age group of 2 to 5 years which shows progression till puberty and regresses spontaneously[8],[9] Aneurysmal bone cyst, giant cell granuloma and florid FD, other developmental disorders, often confused with FD. A true bone tumor, ossifying fibroma, contains a variable mixture of bony trabeculae, cementum-like spherules or both should be distinguished from these other lesions. Paget's disease where multiple lesions are bilateral and age over 40 years are affected more with pain as an important distinguishing feature with hypercementosis. Osteosarcoma has more aggressive radiographic feature changes including more bizarre internal structure.[10],[11] The gold standard for confirming the diagnosis of FD is a histologically proven fibro-osseous lesion radiographic finding with poorly defined margins. “FD is a bony developmental anomaly showing proliferation within the medullary bone as hamartomatous fibrous tissue, producing newly immature, weak calcified bone, with secondary bony metaplasia and without any maturation of osteoblast which on radiographs appear as radiolucent, described classically as ground-glass appearance.”[12]

Treatment protocols for FD include observation, medicinal treatment, and surgery. Clinical observation is suggested for FD lesions that have no risk of deformity or pathologic fracture. Medical treatment with bisphosphonates may have benefits including improvement of function, pain relief, and lower risk of fractures in properly selected FD patients. Indication for surgery is for confirmatory biopsy, deformity correction, prevention of pathologic fracture, and/or elimination of lesions which are symptomatic.[13] The patient reported with long-standing slow-growing lesion with bony hard swelling with significant amount of bony destruction and also patient was young so to eliminate the facial deformity, avoid pathological fracture surgery was planned and complete curettage was done by keeping safe margin of 1 cm around the remnant of healthy bone was insufficient to bear the occlusal load so titanium plate and screw was adapted over the body of the mandible and teeth where subjected to root canal and was differed from occlusal load by selective grinding and disoccluding. The patient was advised soft diet and avoid trauma to that site postoperatively. OPG [Figure 2] was repeated at 6-month follow-up which showed satisfactorily healing of surgical site and no evidence of any fracture or any recurrence.[14],[15]
Figure 2: (a) Postoperative intraoral image. (b) Orthopantomograph taken at 6-month follow-up showing a reduction in radiolucency and root canal obturation with 35, 36, and 37 and nonrigid titanium plate at roots of 33, 34, 35, 36, and 37

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  Conclusion Top


Isolated FD cases in maxillofacial regions are minimal and not easy to distinguish from other disorders of benign and malignant bone origin. Imaging studies and histological and laboratory tests are mandatory to be carried out for gaining definite diagnosis, treatment planning, and for management of FD.[14]

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.
Lichtenstein L. Polyostotic fibrous dysplasia. Arch Surg 1938;36:874-98.  Back to cited text no. 1
    
2.
Riminucci M, Fisher LW, Shenker A, Spiegel AM, Bianco P, Gehron Robey P. Fibrous dysplasia of bone in the McCune-Albright syndrome: Abnormalities in bone formation. Am J Pathol 1997;151:1587-600.  Back to cited text no. 2
    
3.
Boltin D, Gingold-Belfer R, Lichtenstein L, Levi Z, Niv Y. Long-term treatment outcome of patients with gastric vascular ectasia treated with argon plasma coagulation. Eur J Gastroenterol Hepatol 2014;26:588-93.  Back to cited text no. 3
    
4.
Ni Y, Yang Z, Xu H, Lu P, Wang P, Liu F. Assessment of preoperative and postoperative quality of life of 24 patients with fibrous dysplasia of the mandible-Y. Br J Oral Maxillofac Surg 2019;57:913-917. doi: 10.1016/j.bjoms.2019.08.001. Epub 2019 Aug 26.  Back to cited text no. 4
    
5.
Gujjar P, Sahni P, Jayadeva HM, Shylaja MD. Monostotic fibrous dysplasia of mandible: Report of a case in a young female.International Journal of Applied Dental Sciences 2015; 1(5): 10-12.  Back to cited text no. 5
    
6.
Ameli NO, Rahmat H, Abbassioun K. Monostotic fibrous dysplasia of the cranial bones: Report of fourteen cases. Neurosurg Rev 1981;4:71-7.  Back to cited text no. 6
    
7.
Zenn MR, Zuniga J. Treatment of fibrous dysplasia of the mandible with radical excision and immediate reconstruction: Case report. J Craniofac Surg 2001;12:259-63.  Back to cited text no. 7
    
8.
Zachariades N, Papanicolaou S, Xypolyta A, Constantinidis I. Cherubism. Int J Oral Surg 1985;14:138-45.  Back to cited text no. 8
    
9.
Zohar Y, Grausbord R, Shabtai F, Talmi Y. Fibrous dysplasiaand cherubism as an hereditary familial disease. J Craniomaxillofac Surg 1989;17:340-4.  Back to cited text no. 9
    
10.
Slootweg P, Muller H. Differential diagnosis of tibro-osseousjaw lesions. J Craniomaxillofac Surg 1990;18:210-4.  Back to cited text no. 10
    
11.
Reed RJ, Hagy DM. Benign nonodontogenic fibro-osseous lesions of the skull; report of two cases. Oral Surg Oral Med Oral Pathol 1965;19:214-27.  Back to cited text no. 11
    
12.
Ben hadj Hamida F, Jlaiel R, Ben Rayana N. Craniofacial fibrous dysplasia: A case report. J Fr Ophtalmol 2005;28:e6.  Back to cited text no. 12
    
13.
DiCaprio MR, Enneking WF. Fibrous dysplasia. Pathophysiology, evaluation, and treatment. J Bone Joint Surg Am 2005;87:1848-64.  Back to cited text no. 13
    
14.
Cholakova RP, Kanasirska N, Kanasirski IV, ChenchevAD. Fibrous dysplasia in the maxillomandibular Region –case report. JIMAB. 2010; 16(4):10-13.Ricalde P, Magliocca KR, Lee JS:Craniofacial fibrous dysplasia. Oral Maxillofac Surg Clin North Am 24:427,2012.  Back to cited text no. 14
    
15.
Hotwani K, Sharma K, Jaiswal A. Evaluation of Tongue/Mandible Volume Ratio in Children with Obstructive Sleep Apnea. Dental Press J Orthod 2018;23:72-8. Available from: https://doi.org/10.1590/2177-6709.23.4.072-078.oar. [Last accessed on 2020 Jan 05].  Back to cited text no. 15
    


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