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it is a chronic progressive disease involving single bone ( monostotic ) or many bones ( polyostotic ) of the body , characterized by rapid bone resorption and deposition , resulting in numerous reversal line formation which gives the mosaic pattern to the lamellar bone with profuse local vascularity and fibrous tissue in the marrow .
clinical symptoms include pain , deformity , and may lead to fracture of the affected bone , even though the initial course of the disease may be asymptomatic .
the enlarged and deformed bones may compress surrounding nerves and vessels causing neurological symptoms like hearing loss ; inexplicably , it is quite unusual in the facial bones .
radiograph shows characteristic cotton wool appearance and other radiographic finding include well - circumscribed radiolucency , loss of lamina dura , pulpal radio - opacity , root resorption , and hypercementosis.[46 ] the etiology of pd is still obscure , but genetic and environmental factors may play a role .
viruses such as paramyxovirus , canine distember virus , and respiratory syncytial virus are also reported to be the causative agents .
a recent clinical trial suggested that second and third generation bisphosphonate , such as pamidronate and alendronate , were more effective than calcitonin and editronate , the first generation bisphosphonate .
here , we are reporting a very rare case of pd with isolated mandibular involvement .
a 67-year - old female patient reported to a private dental clinic in coimbatore , complaining of swelling in the lower jaw for past few years .
history reveals a slow - progressive swelling with no other associated symptoms like pain , discharge , and non - responsive to any medication .
extra oral examination reveals broadening and widening of the lower facial view with thickened lower lip .
intra oral examination showed increased size and altered shape of the mandibular alveolar ridge , pseudo micrognathia of the maxillary arch [ figures 1 and 2 ] , and partially edentulous , thickened cortical plate .
orthopantomograph shows hypercementosis with displacement of involved teeth , cortical thickening of the body of the mandible , and mixed radiolucent and radio opaque areas [ figure 3 ] on both sides of the mandible .
whole - body x - rays and scans were taken to ascertain the involvement of other bones [ figure 4 ] , confirming that the lesion was restricted to mandible only .
extra oral view shows large mandible large size partially edentulous mandible opg shows hypercementosis of the teeth roots .
3d scan shows thickened mandibular cortical plate incisional biopsy was taken , histopathology of lesion showed increased osteoblastic activity and irregular cortical bones with presence of hematoxyphilic reversal line giving the characteristic mosaic pattern .
marrow spaces were filled with fibro vascular connective tissue , confirming osteitis deformans [ figure 5 ] .
biochemical analysis showed abnormal increase of alkaline phosphatase ( alp ) enzyme level ( 1368.1 u / l ) [ figure 6 ] while comparing with the normal values of 30 - 120 u / l ( above 17 years ) and 74 - 390 u / l ( below 17 years ) .
based on the clinical , radiographic , histopathological , and biochemical findings , a diagnosis of mandibular pd was rendered and was explained to the patient .
although pd is a relatively common disease in western countries like england , united states , new zealand , canada , south africa , and france , incidence in asian population that too among indians are very rare .
prevalence is markedly higher in new york than in atlanta , although the disease rare in africa .
pd is a geriatric disease reported above 5 to 6 decades of life , occurs both in men and women , with male predominance of approximately 3:2 ratio .
etiology of pd is unknown , but there is a hypothesis that vitamin d deficiency in children may express later as pd .
inclusion bodies resembling paramyxovirus nucleocapsid particles have been observed in pagetic osteoclasts by electron microscopy and canine distemper virus has been localized by in - situ hybridization in the bone cells of patients with disorder .
there is familial tendency of occurrence of pd and it is associated with polymorphisms in dna coding in centrosome structure .
but , it is evident from the decline in disease prevalence in britain that undifferentiated environmental factors is also a major contributor .
isolated involvement of one or both the jaws , the clinical and radiographic appearances may be interpreted as those of chronic sclerosing osteomyelitis in such condition , histological examination of bone or teeth may be suggestive if not conclusive performance of biochemical test and adequate follow - up of the patient .
if the disease affects isolated areas of bone such as maxilla or mandible , the biochemical values will be within the normal limits , whereas our case shows an increase of alp level to many folds ( 1368.1 u / l ) despite isolate involvement of mandible . as the stresses of mastication causes very active turnover of bone ,
healing infectious periostitis with excessive bone formation as a possible cause of numerous cement lines produces chaotic ( mosaic ) bone architecture .
but , when mosaics appear in conditions other than pd , their extent is limited and more regular , this fact coincide with our case , irregular mosaic pattern with numerous reversal line in our histopathological study . since the major defect in pd is exaggerated bone remodeling , treatment is based on powerful antiresorptive agent .
bisphosphonates form the main stay of medical management of pd . these agents suppress or reduce bone resorption by osteoclasts .
five bisphosphonates are approved by the us food and drug administration for the treatment of pd .
these include pamidronate , which is given intravenously , etidronate , tiludronate , alendronate , and risedronate , all of which are taken orally .
the case series from vellore and mumbai suggests that lower doses of alendronate might work well in indian patients .
we report a rare case of isolated mandibular pd in a 65-year - old indian female patient with abnormal increase of alp level ( 1368.1 u / l ) considered to be unique in our case .
paget 's disease ( pd ) is a chronic progressive disease of the bone characterized by abnormal bone resorption and deposition affecting either single bone ( monostotic ) or many bones ( polyostotic ) with uncertain etiology . 
 we report a case of isolated mandibular involvement in a 65-year - old female patient , clinically asymptomatic with abnormally increased alkaline phosphatase level ( 1 368.1 u / l ) . 
 although prevalence of pd is common in western countries , but rare in asian chapter , that too isolated mandibular involvement , considering this fact , we report this case of pd for documentation .