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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 30  |  Issue : 2  |  Page : 173-176

Osteonecrosis of jaw bones: A complication of severe dengue


Department of Oral Medicine and Radiology, Subharti Dental College, Meerut, Uttar Pradesh, India

Date of Submission12-Apr-2018
Date of Acceptance19-Jun-2018
Date of Web Publication16-Jul-2018

Correspondence Address:
Dr. S M Ravi Prakash
Department of Oral Medicine and Radiology, Subharti Dental College, Meerut, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_59_18

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   Abstract 


Dengue fever is an acute infection of viral origin caused by female Aedes aegypti mosquito. It has a flu-like illness that can be fatal if left untreated. Several systemic complications such as hypovolemia and circulatory shock with organ failure, oral complications such as post-extraction bleeding, and candidiasis have been reported. Rare complications such as lock jaw and osteonecrosis can also manifest in severe form of dengue fever. Very scanty literature exists to document this association. This case report is an evidence to document and emphasize that these complications develop in severe cases of dengue fever even without any preexisting odontogenic or periodontal etiology.

Keywords: Dengue fever, lock jaw, osteonecrosis


How to cite this article:
Ravi Prakash S M, Nasim A, Kamarthi N, Gupta S. Osteonecrosis of jaw bones: A complication of severe dengue. J Indian Acad Oral Med Radiol 2018;30:173-6

How to cite this URL:
Ravi Prakash S M, Nasim A, Kamarthi N, Gupta S. Osteonecrosis of jaw bones: A complication of severe dengue. J Indian Acad Oral Med Radiol [serial online] 2018 [cited 2019 Oct 17];30:173-6. Available from: http://www.jiaomr.in/text.asp?2018/30/2/173/236734




   Introduction Top


Dengue fever commonly named as “break bone fever” is a painful debilitating mosquito borne disease caused by a flavivirus transmitted by day biting female Aedes aegypti mosquitoes.[1] Due to its evolution in the past 50 years, it has become an important public health problem endangering an estimated 2.5 billion population across the globe.[2] It is endemic in many countries such as South East Asia, Africa, and Central and South America,[2] but most commonly involving Indians with approximately 33 million cases as reported in 2010.[3] The etiology, as hypothesized, could be either viral replication which occurs primarily in macrophages or direct infection of skin by virus. It can also be related to immunologic and chemically mediated mechanism induced by interaction of the virus with the host.[4] The fever is a benign disease characterized with acute onset of severe headache and myalgia, and it further leads to life-threatening dengue hemorrhagic fever (DHF). It mainly arises from damage to lymphatics, with plasma leakage, increased vascular permeability, thrombocytopenia, producing impaired hemostasis, epistaxis, and gingival hemorrhage. Plasma leakage results in pleural effusion and ascites leading to hypovolemia and circulatory shock with organ failure.[1] The World Health Organization mentions organ dysfunction as the indicator of the severity of dengue.[5] During second episodes of infection, hepatosplenomegaly increases with red blood cell destruction resulting in massive bleeding and dengue shock syndrome (DSS).[6]

Oral manifestations are more commonly associated with DHF presenting as gingival bleeding erythema, lip crusts, and vesicles on the lips and palate.[7],[8] Oral candidiasis [9] and post-extraction bleeding [10] were also reported. Few case reports documented the occurrence of osteonecrosis of jaws (ONJs) in association with dengue fever [1],[11] [Table 1]. This case report emphasizes that lock jaw and ONJ bones should be considered as oral manifestation occurring with severe form of dengue fever.
Table 1: Reported cases of osteonecrosis of jaw associated with dengue fever

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   Case Report Top


A 24 year-old male reported to the Department of Oral Medicine and Radiology with a chief complaint of multiple draining sinuses in the oral cavity for 2 months. The patient's medical history was significant for dengue fever which required hospitalization for 3 months. During the course of treatment, the patient noticed locking of jaws. Later on, on recovery from dengue, the patient developed diffuse swelling on the left side of the face and multiple draining sinuses from the left buccal vestibule. The patient had no history of deleterious oral habits, trauma, or any systemic disease before contacting dengue fever. There was no history of any drug allergies.

On extraoral examination, there was extensive diffuse erythematous swelling of the left side of the face [Figure 1]. Intraoral examination revealed good oral hygiene with no carious teeth. No evidence of gingival swelling, gingival recession, mobility, or attachment loss on periodontal probing was seen. There was slight obliteration of buccal vestibule extending from 38 till 43 region with multiple draining sinuses [Figure 2]. Teeth 38 to 43 were nonvital on electric pulp testing.
Figure 1: Extraoral photograph reveals diffuse erythematous swelling of left side of face

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Figure 2: Intraoral photograph depicting slight obliteration of buccal vestibule extending from 33 till 38region with multiple draining sinuses

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Panoramic radiograph revealed multiple small irregular osteolytic areas involving the left body of mandible from 38 to 43 regions extending to left ramus, coronoid, and condylar regions giving moth-eaten appearance. Interdental vertical and horizontal pattern of bone loss was evident from 38 to 43 regions [Figure 3]. Advanced imaging such as contrast-enhanced computed tomography and cone-beam computed tomography was carried out which showed evidence of diffuse osteolysis with intermingled area of sclerosis. Multifocal areas of cortical break in left coronoid, condylar process, and ramus extended up to symphysis menti and involved the right half of the body of mandible [Figure 4]a,[Figure 4]b,[Figure 4]c,[Figure 4]d.
Figure 3: Orthopantomograph shows multiple small irregular osteolytic areas involving left ramus, coronoid, condylar and body of mandible extending from 38 to 43 region giving moth-eaten appearance. Interdental Vertical and horizontal pattern of bone loss was evident from 38 – 43 regions

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Figure 4: (a and b) CECT in 3D imaging and sagittal section reveal evidence of diffuse osteolysis involving left condyle, coronoid , ramus upto symphysis region with interdental vertical and horizontal bone loss. (c and d) CBCT in 3D and axial section unveiled diffuse osteolysis with intermingled area of sclerosis with multi focal area of cortical break in left coronoid, condylar process, ramus upto symphysis extending to the right half of the body of mandible

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Erythrocyte sedimentation rate was raised to 94 mm/1st h. Other test results such as complete blood count, hemoglobin, and blood sugar levels (fasting and PP) were within normal limits. The patient was nonreactive to HIV and hepatitis. Aspirated material did not show any evidence of acid-fast bacilli with Gram staining and was negative to fungal growth. Histopathological examination of necrosed hard tissue sample at 40× magnification revealed cancellous bone with empty lacunae. Lacunae indicating acellular marrow space infiltrated with chronic inflammatory cell with superadded secondary infection. [Figure 5].
Figure 5: H&E stained section at ×40 magnification of the received specimen depicting necrotic bone with empty lacunae and relatively acellular marrow space with superimposed secondary infection

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Based on history, clinical findings, and radiological examination, diagnosis of ONJ secondary to dengue fever was made. Osteomyelitis was ruled out as there was neither odontogenic infection nor primary periodontal infection. The only suitable diagnosis that could be made was ONJs.

The patient was on conservative management with oral amoxicillin with combination of potassium clavulanate (625 mg) and analgesic thrice in a day for 15 days. For maintenance of oral hygiene, the patient was advised with 0.2% chlorhexidine oral rinse twice daily for 3 months along with desensitizing toothpaste. The patient showed signs of improvement in 3 months and is on regular follow-up. [Figure 6].
Figure 6: (a) Patient presented with signs of improvement with increase in mouth opening.after conservative management was done. (b) OPG revealed a marked reduction in the radiolucent areas with increase in bone density

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   Discussion Top


Dengue fever is a viral disease associated with flu-like illness that affects infants, children, and adolescents.[12] They are of three classical forms including classic dengue, DHF and DSS.[5] The febrile period is accompanied with a rash which appears as diffuse maculopapular or petechiae formation.[12] Oral manifestations associated are reddish swelling of gingiva and lips and bleeding of gums, raised hemorrhagic plaques on buccal mucosa and tongue, and post-extraction hemorrhage.[13] Purpura and ecchymosis reflect platelet levels.[4]

As the life cycle of dengue virus begins, it has the tendency to enter blood stream and penetrate macrophages and monocytes which enable reproduction in the circulation.[14] The virus also activates CD4+ T cells. The monocytes release tumor necrosis factor-α which creates cascade of cytokine, complement activation, and monocyte chemotactic protein-1 activation. The cytokine and chemokine cause leukopenia, raised vascular permeability, thrombocytopenia, and trigger coagulation and fibrinolysis.[11] Fibrinolysis and thrombocytopenia are factors that lead to hemorrhage and widespread capillary permeability giving rise to edema and disseminated intravascular coagulopathy and contribute to osteonecrosis. Complex cytokine interaction also causes bone marrow suppression and osteonecrosis.[15]

Osteonecrosis could be due to marrow suppression and intraosseous hemorrhage leading to bone infarction and periodontal infection.[16],[17] The present case with clinical and radiological features is compared with osteomyelitis, osteoradionecrosis, bisphosphonate-related osteonecrosis, osteonecrosis associated with herpes virus, and alveolar osteitis.[16] No history of drug therapy and radiotherapy ruled out the possibility of osteoradionecrosis and bisphosphonate ONJ. Good oral hygiene, no history of periodontal problems, no evidence of carious teeth, and clinical periodontal disease like mobility, recession, and periodontal pockets exclude osteomyelitis secondary to primary odontogenic or periodontal cause. Negative acid-fast bacilli and fungal culture exclude the possibilities of tubercular and fungal osteomyelitis. History of recent episodes of severe dengue fever supports the diagnosis of osteonecrosis as one of the oral manifestations caused by it.

One of the most characteristic clinical features that delineate osteomyelitis secondary to odontogenic and periodontal infection with ONJs secondary to dengue fever is the absence of recession, mobility, attachment loss, and carious teeth as demonstrated in the present case. The proximity of the jaws to oral microorganisms could be one factor that predisposes jaws more than any other bone to develop osteonecrosis with dengue fever. Another important finding that was seen in this case was lock jaw that was absent in the other two reported cases of osteonecrosis associated with dengue. We emphasize that lock jaw should be considered as one of the significant findings associated with dengue.

ONJs are generally managed conservatively. Conservative therapy includes maintaining optimal oral hygiene, elimination of active dental and periodontal disease, topical antibiotic mouth rinses, and systemic antibiotic therapy as indicated. Conservative therapy is the mainstay of care, and although it may not necessarily lead to complete resolution of lesions, it symptomatically may provide long-term relief. For nonresponsive ONJ lesions, surgery is an option and includes ostectomy of the affected area with resection margins that extend into adjacent normal appearing bone.


   Conclusion Top


A case of extensive ONJs as one of the severe and unusual complications associated with severe dengue fever has been documented. However, underlying possibilities for the described pathology cannot be completely ruled out as there have been limited studies reporting the association between ONJ and dengue fever. Therefore, more such cases need to be documented to determine this unique clinical presentation of dengue fever so that the possibility of its occurrence and remedy can be sorted out at the earliest for patients' betterment.

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.
Al-Namnam NM, Nambiar P, Shanmuhasuntharam P, Harris M. Dengue relatedosteonecrosis of the maxillary dento-alveolar bone. Aust Dent J 2017;62:228-32.  Back to cited text no. 1
    
2.
World Health Organization. Dengue and severe dengue. Factsheet No. 117. Updated March 2014. Available from: http://www.who.int/mediacentre/factsheets/fs117/en. [Last accessed on 2014 Sep 20 ].  Back to cited text no. 2
    
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Chakravarti A, Arora R, Luxemburger C. Fifty years of dengue in India. Trans R Soc Trop Med Hyg 2012;106:273-82.  Back to cited text no. 3
    
4.
Mithra R, Baskaran P, Sathyakumar M. Oral presentation in dengue hemorrhagic fever: A rare entity. J Nat Sci Biol Med 2013;4:264-7.  Back to cited text no. 4
    
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World Health Organization (WHO). Dengue Guidelines for Diagnosis, Treatment, Prevention and Control. New ed. Geneva, Switzerland: World Health Organization; 2009. Available from: http://www.who.int/tdr/publications/documents/dengue-diagnosis. [Last accessed on Mar 2018].  Back to cited text no. 5
    
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Halstead SB. Pathogenesis of dengue: Challenges to molecular biology. Science 1988;239:476-81.  Back to cited text no. 6
    
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Ahmed S, Mohammad WW, Hamid F, Akhter A, Afzal RK, Mahmood A. The 2011 dengue haemorrhagic fever outbreak in Lahore An account of clinical parameters and pattern of haemorrhagic complications. J Coll Physicians Surg Pak 2013;23:463-7.  Back to cited text no. 7
    
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Azfar NA, Malik LM, Jamil A, Jahangir M, Tirmizi N, Majid A,et al. Cutaneous manifestations in patients of dengue fever. J Pak Assoc Dermatol 2012;22:320-4.  Back to cited text no. 8
    
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Indurkar MS, Sethi R. An unusual case of osteonecrosis of jaw associated with dengue fever and periodontitis. Aust Dent J 2016;61:113-9.  Back to cited text no. 9
    
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Sheikh KR, Shehzad A, Mufti S, Mirza UA, Shamsuddin A. Skin involvement in patients of dengue fever during the 2011 epidemic in Lahore, Pakistan.J Pak Assoc Dermatol 2012;22:325-30  Back to cited text no. 10
    
11.
Dubey P, Kumar S, Bansal V, Kumar KV, Mowar A, Khare GPostextraction bleeding following a fever: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol 2013;115:27-31.  Back to cited text no. 11
    
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Thomas EA, John M, Kanish B. Mucocutaneous manifestations of dengue fever. Indian J Dermatol 2010;55:79-85.  Back to cited text no. 12
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13.
Pontes FS, Frances LT, Carvalho Mde V, Fonseca FP, Neto NC, do Nascimento LS,et al. Severe oral manifestation of dengue viral infection: A rare clinical description. Quintessence Int 2014;45:151-6.  Back to cited text no. 13
    
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Chaturvedi UC, Dhawan R, Khanna M, Mathur A. Breakdown of the blood-brain barrier during dengue virus infection of mice. J Gen Virol 1991;72:859-66.  Back to cited text no. 14
    
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Chuansumrit A, Chaiyaratana W. Hemostatic derangement in dengue hemorrhagic fever. Thromb Res 2014;133:10-6.  Back to cited text no. 15
    
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Drozdzowska B. Osteonecrosis of the jaw. Pol J Endocrinol 2011;62:88-92.  Back to cited text no. 16
    
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Khan AA, Morrison A, Hanley DA, Felsenberg D, McCauley LK, O'Ryan F, et al. Diagnosis and management of osteonecrosis of the jaw: A systematic review and international consensus. J Bone Miner Res 2015;30:3-23.  Back to cited text no. 17
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]
 
 
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