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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 26  |  Issue : 4  |  Page : 467-472

Pitfalls in diagnostic imaging: A case report


1 Department of Oral Medicine and Radiology, SGT Dental College, Gurgaon, India
2 Department of Oral Medicine and Radiology, Manav Rachna Dental College, Faridabad, Haryana, India

Date of Submission01-Aug-2014
Date of Acceptance09-Mar-2015
Date of Web Publication22-Apr-2015

Correspondence Address:
Ruchi Saharan
House No. 91/22, Mastana Bhawan, Sonepat Road, Rohtak - 124 001, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.155670

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   Abstract 

The new millennium has witnessed a sea change in diagnostic imaging sciences and the unfortunate 'tilt' toward 'over imaging' and 'over relying' on modern imaging needs to be relooked. This case report emphasizes the need to strengthen conventional diagnostic skills/acumen and the importance of case history taking in all its grandeur as a part of comprehensive patient work up. In this case report of a 7-year-old child, the entire prognosis and treatment outcome was fortuitous since the imaging findings and suspected diagnosis on magnetic resonance imaging were relegated to the findings of history and clinical examination.

Keywords: Ankylosis, chronic suppurative osteomyelitis, cone beam computed tomography (CBCT), magnetic resonance imaging (MRI)


How to cite this article:
Saharan R, Dang V, Chandar VV, Tanwar R. Pitfalls in diagnostic imaging: A case report. J Indian Acad Oral Med Radiol 2014;26:467-72

How to cite this URL:
Saharan R, Dang V, Chandar VV, Tanwar R. Pitfalls in diagnostic imaging: A case report. J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2020 Aug 5];26:467-72. Available from: http://www.jiaomr.in/text.asp?2014/26/4/467/155670


   Introduction Top


The term 'osteomyelitis' (originating from the Greek words 'osteon' for bone and 'muelinos' for marrow) refers to inflammation of bone and its marrow. Acute, subacute and chronic presentations of this condition have been described. [1],[2] The terminologies 'chronic suppurative osteomyelitis' and 'secondary chronic osteomyelitis' describing a relatively common suppurative variety have been interchangeably used in past medical literature. [3] Suppurative osteomyelitis can present as a triad of periosteal, cortical, and marrow involvement (i.e., all three components of bone). The clinical presentation of disease and radiological findings may span the spectrum of aggressive osteolytic putrefaction to a dry osteosclerotic phase depending on the host immunity and microbial virulence factors. [3]

Radiological examination is an important investigation in osteomyelitis in addition to other relevant investigations. Magnetic resonance imaging (MRI) may be used to complement radiography to depict soft tissue changes. A case report of chronic suppurative osteomyelitis of the left mandible accompanied by ankylosis of the left temporomandibular joint (TMJ) in a young female patient investigated radiologically and with MRI is presented. Accurate diagnosis is essential in the successful management of such cases which in turn impacts the treatment outcome.


   Case Report Top


A young girl aged 7 years reported to the department of Oral Medicine and Radiology of our dental institute with the chief complaint of a painless swelling on the lower part of the left side of the face of approximately 3-months duration [Figure 1]. The patient also complained of occasional pus discharge extraorally from in front of the left ear lobe. The child had experienced an episode of pain in the mandibular left deciduous molar region two years ago. This was followed by recurrent episodes of swelling on the left side of the face, pus discharge, and spontaneous remission. The father of the patient also informed that she had developed a reduced mouth opening for the past 3 years which was progressive for 1 and a half years and had remained thus so. No definitive history of trauma was reported by the family initially. However, on further enquiry the father informed that she had a fall while playing at home 4 years back. There was no history of birth injury, fever, cough, ear discharge, and any significant medical ailment. There was no history of consanguineous marriage between the parents. The patient was the youngest of six siblings and no history of any significant medical illness was reported in the others.
Figure 1: Extraoral swelling of approximately 70 × 50 mm involving the middle and lower half of the left side of face

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On clinical examination a marked facial asymmetry with fullness of cheek on the left side of the face associated with a retruded chin was obvious. A diffuse swelling measuring approximately 7 cm (across) × 5 cm (vertically) involving the middle and lower one-third of the left face and the presence of a discharging sinus 2-cm anterior to angle of mandible with enlarged and tender left submandibular and submental lymph nodes [Figure 2] were noted. Multiple scars were observed 1-cm anterior to left tragus of the ear and at the angle of mandible. Palpation revealed a soft ovoid and non-tender swelling approximately 7.5 × 6 cm extending over the middle and lower one-third of the face without any local rise in temperature. The swelling extended from the lower border of mandible inferiorly to the malar process superiorly and from the ala of nose to 1 cm in front of the external auditory meatus in the antero-posterior direction. No condylar movements were palpable in the left TMJ region and there was deviation of chin towards the left side on mouth opening. However, reduced condylar movements were palpable in the right TMJ region. An interincisal opening of 1 cm with anterior open bite was noted. The mandibular midline was deviated to the left. Intra-orally, a bony hard brownish mass approximately 5 × 3 cm adherent to the buccal vestibule and extending posteriorly from the mandibular left deciduous canine was also seen. The buccal mucosa per se was unremarkable.
Figure 2: Extraoral photograph showing sinus on the left angle of mandible and multiple scars of healed sinuses

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A computed tomography (CT) scan had been performed 1 month ago on the advice of a local practitioner but the diagnosis was inconclusive. The axial sections available confirmed the location and size of the lesion revealing it to be radio opaque in nature. Additionally, the outlines of the coronoid process and condyle were not discernable on the left side with complete osseous union between the coronoid-condylar complex and the base of the skull. The left TMJ space was not discernible [Figure 3]. [Figure 4] shows the hard brownish mass with a coarse 'moth-eaten appearance' after it was pried out from the left buccal vestibule under local anesthesia.
Figure 3: Three-dimensional view of CT showing complete osseous union of the coronoid-condylar complex with the base of skull and obliteration of joint space on the left side

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Figure 4: Sequestrum extract measuring 50 × 30 mm

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Based on the above findings a provisional diagnosis of chronic suppurative osteomyelitis (of odontogenic origin) with ankylosis of the left TMJ (of traumatic origin) was made. A differential diagnosis of tuberculous osteomyelitis and juvenile chronic arthritis was also considered. A routine hemogram, smear from the sinus for acid-fast bacilli (AFB) and culture/sensitivity, Mantoux test, X-ray chest- AP view, digital panoramic radiograph (OPG) and Cone Beam CT (CBCT) scan were advised. To assess the involvement of the soft tissues an MRI scan was also carried out.

The OPG revealed an indiscernible outline of the left condylar head and obliteration of the joint space. It showed increase in the width of ramus of the mandible on the left side. A radiolucency extending to the mid-coronal region was suggestive of caries in 75. Near total root resorption was seen in 75 and other deciduous teeth conforming to the normal exfoliative sequence [Figure 5]. An ill-defined peri-radicular radiolucency suggestive of periapical abscess was also noted in 46.
Figure 5: Preoperative OPG showing indiscernible outline of the left condylar head and obliteration of joint space

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The hard mass removed from the buccal sulcus was examined radiologically using a standard adult periapical (IOPA) film before submitting for histopathological examination. The histopathologic findings of marrow spaces and an eosinophilic trabecular pattern containing empty lacunae were consistent with a bony sequestrum [Figure 6]. Few mixed inflammatory cells were also noted at some foci. The radiograph also revealed a radiodensity compatible with bone [Figure 7]. The routine blood examination findings were within normal limits and the chest X-ray did not reveal any abnormality. Sputum was negative for AFB and the culture showed the presence of Streptococcus species sensitive to most antibiotics.
Figure 6: Histopathological view of the sequestrum revealing marrow spaces around which eosinophilic trabecular pattern was appreciated containing empty lacunae

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Figure 7: IOPA of sequestrum showing radiodensity compatible with bone

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The CBCT findings were suggestive of a left condylar neck fracture with an anteromedially displaced condyle and an extra-articular ankylosis in the left TMJ region (between the lateral aspect of the hypertrophied left condylar neck, left zygomatic arch and root of the left coronoid process) [Figure 8]a-c. There was a depressed fracture of the left zygomatic arch with hypertrophy of the arch accompanied by an irregular extra-articular ankylosis of the left TMJ due to new bone formation from the lateral aspect of the glenoid fossa and temporal process of the zygomatic arch. New bone formation also extended from the inferomedial aspect of the left zygomatic arch to the root of the coronoid process close to the lateral cortex of the ramus. Additionally surface erosion and sub-articular sclerosis was noted. The glenoid fossa and the articular eminence were flattened and deformed. The inferior border of the mandible was everted laterally corresponding to 36 and 37 regions. The basal trabecular pattern of the left posterior body showed mild to moderate sclerosis.
Figure 8: CBCT (a) axial section showing the anteromedial displacement of the left condyle (b) sagittal section showing hypertrophy of the remaining condylar neck and reduced height with increased width of the ramus, and (c) 3-D reconstructed image showing left condylar neck

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The patient was also subjected to an MRI for the delineation and assessment of the marked soft tissue involvement. The MRI was performed on a 1.5 Tesla and revealed fatty infiltration of the left parotid gland, masseter, medial pterygoid, and temporalis muscle [Figure 9].
Figure 9: T1-weighted magnetic resonance image showing findings suggestive of fatty infiltration involving the masseter, lateral pterygoid and temporalis muscle of the left side

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fatty infiltration of the left parotid gland, masseter, medial pterygoid, and temporalis muscle [Figure 9]. The left lateral pterygoid muscle showed reduced bulk without fatty infiltration. The areas of fatty infiltration showed hyperintense signal on T1-weighted image and completely suppressed on short T1 inversion recovery (STIR) images [Figure 10]. The submandibular glands, oral and base of the tongue appeared normal in signal intensity and morphology. The radiologist reported these findings to be compatible with features of congenital lipomatosis of the face.
Figure 10: T1-weighted fat suppressed image

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Despite the contradictory radiological findings, surgical intervention in the form of a gap arthroplasty and local debridement was performed under general anesthesia relying more on the clinical and radiologic findings rather than the MRI. A soft tissue biopsy from the region of the swelling and curettage of the sinus tract was performed. The histopathology revealed non-specific inflammation. The patient had an uneventful recovery without any recurrence of the disease over a regular follow-up of 2 months. The post-operative panoramic radiograph taken after 2 weeks of surgery is shown in [Figure 11]. A mouth opening of 3.2 cm was achieved after surgery [Figure 12] and cosmetic surgery was deferred till puberty.
Figure 11: Postoperative orthopanoramic view

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Figure 12: Interincisal opening after 2 weeks following surgery

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   Discussion Top


Osteomyelitis of the jaw bones has been frequently reported in literature with a propensity toward mandibular involvement. This lower jaw predilection may be explained on the basis of the hard cortical encasement of the mandible and limited collateral vascularity. [4] Historically, the disease incidence graph dipped considerably with the advent of antibiotics. However, growing microbial resistance to antibiotics has seen re-emergence of disease presenting diagnostic and therapeutic challenges. Osteomyelitis remains a significant cause of patient morbidity often necessitating multiple surgeries. Prolonged treatment time, hard tissue (teeth and or bone) loss, and associated soft tissue changes add to the challenge in successful management. [5]

Odontogenic infection, trauma (especially compound fractures), surgery with subsequent ingress of pathogenic micro-organisms, infections of the oral cavity leading to periostitis, infections from furuncles or lacerations in addition to the less common hematogenous deposition of microbes have all been listed as causative factors. [6] The posterior body of the mandible is the most common site for odontogenic infection to cause osteomyelitis in both acute and chronic forms. [7]

An abrupt onset of symptoms and signs during the initial stage of infection are the hallmark of acute osteomyelitis. Sub-acute or chronic disease might present as sequelae of the acute infection or may result from inadequate or improper treatment. [6] Use of drugs such as steroids, chemotherapeutic agents and bisphosphonates have also been linked to the development of osteomyelitis as also the presence of underlying systemic disease such as diabetes, malignancies, malnutrition, acquired immunodeficiency syndrome, and autoimmune status. [8],[9] In the present case none of these factors were identified and the cause of infection could be attributed to the fact that the patient had a severely carious mandibular left second deciduous molar which may have facilitated the entry of bacteria. An undisclosed trauma to the left side of the face in the past may be a further confounding factor. The present case posed a diagnostic dilemma as to the exact origin of the infection and the finding of bony ankylosis of the left TMJ - which is a rare association with osteomyelitis.

The peculiar reparative bone stimulating response accompanying the infective/destructive process (including the findings of a large sequestrum) was quite unique in our case. A negative AFB smear and normal chest X-ray indicated the likely absence of tuberculosis. Oral tuberculous in the form of chronic ulcers, tuberculous gingivitis, erythematous patches, indurated lesions with a granular surface, nodules, and fissures, and tuberculous osteomyelitis have also been reported occurring in approximately 3% of cases involving long standing pulmonary and/or systemic infection. [10],[11]

A panoramic radiograph is a valuable aid for initial evaluation of widespread jaw bone pathoses, which is widely available. Radiographic changes associated with osteolytic or osteogenic bone disease, potential sources of infection, fractures and underlying bone changes can be visualized. [5] Computed tomography scans provide three-dimensional imaging not available in an OPG. Early cortical erosion of bone in osteomyelitis can be visualized in a CT scan, along with the extent of the lesion, bony sequestra and pathologic fractures. However, like conventional films, CT requires 30-50% demineralization of bone for changes to be seen, which may cause a delay in early diagnosis. [12] Multi-detector computed tomography (MDCT) and CBCT with better resolution have been recently added to the imaging armamentarium. CBCT allows accurate and objective assessment of an abnormality in addition to 3D volumetric rendering at relatively small radiation doses. [13] The comparatively lower radiation dose in a CBCT scan (as compared to conventional CT), large diagnostic yield and a range of diagnostic applications in the maxillofacial region have found favor with most clinicians. These include evaluating the presence of osseous defects in the jaws, cysts, lesions, calcifications, teeth and bone trauma and fractures. [14] Evaluation of soft tissue lesions can be better performed with the help of MRI. Since there is a loss of the marrow signal, it can assist in identifying earlier stages of osteomyelitis, before cortical erosion or sequestrum of the bone appears. [15] MRI may complement radiological examination by accurately depicting the anatomic extent of lipomatous tumors. [16]

In the present case our provisional diagnosis of chronic osteomyelitis with ankylosis of the left TMJ were contradicted by the MRI findings suggestive of congenital lipomatosis of the face. This rare congenital disorder is characterized by non-encapsulated infiltrating collections of mature lipocytes into the tissues which may in turn lead to craniofacial deformity. On occasion the onset of this condition may be delayed to infancy. The adipose cell infiltration is often accompanied by rapid growth, osseous deformity and a high risk of recurrence after surgery. [16],[17] Infiltrating lipomatosis of the face has been found on the cheek, buccal sulcus, tongue, lip, floor of the mouth, mental area, and parotid gland. [16],[17],[18],[19] The etiology of this unusual condition remains unknown though trauma, chronic irradiation, muscular metaplasia, degeneration with subsequent fatty change, hormonal stimulation of multipotent cells of embryogenic origin, cytomegalovirus infection and alteration in chromosome 12 have been proposed as possible etiologic factors. [18],[19],[20] In the present case diagnostic difficulties posed a dilemma in management since osseous deformity may also accompany changes of congenital lipomatosis (as evidenced on the radiograph). The recurrent nature of the swelling with discharge and spontaneous remission including the presence of non-specific inflammatory reaction in the histopathology did not favor the diagnosis of lipomatosis. Further no evidence of lipomatosis was seen on any other parts of the body which helped to rule out the diagnosis of congenital lipomatosis.


   Conclusion Top


The diagnosis of chronic osteomyelitis of the left mandible accompanied by ankylosis of the left TMJ in the present case formed the basis of surgical intervention despite MRI findings to the contrary highlighting the importance of case history, clinical examination and radiological investigations. In conclusion, we strongly support the idea of a 'holistic' approach in diagnosis rather than over relying on any one imaging modality.

 
   References Top

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2.
Cierny G 3 rd , Mader JT, Penninck JJ. A clinical staging system for adult osteomyelitis. Clin Orthop Relat Res 2003;414:7-24.  Back to cited text no. 2
    
3.
Baltensperger MM, Eyrich GK. Osteomyelitis of the Jaws: Definition and classification. In: Baltensperger MM, Eyrich GK, editors. Osteomyelitis of the Jaws. Berlin: Springer-Verlag; 2009. p. 5-56.  Back to cited text no. 3
    
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Theologie-Lygidakis N, Schoinohoriti O, Iatrou I. Surgical management of primary chronic osteomyelitis of the jaws in children: A prospective analysis of five cases and review of the literature. Oral Maxillofac Surg 2011;15:41-50.  Back to cited text no. 4
    
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Khushner MG, Alpert B. Osteomyelitis and osteoradionecrosis. In: Miloro M, Ghali GE, Larsen PE, Waite PD. Peterson's principles of Oral and Maxillofacial Surgery. 2 nd ed. Hamilton, Ontario: BC Decker Inc.; 2004. p. 313-25.  Back to cited text no. 5
    
6.
Prasad KC, Prasad SC, Mouli N, Agarwal S. Osteomyelitis in the head and neck. Acta Otolaryngol 2007;127:194-205.  Back to cited text no. 6
    
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Shafer WG. Diseases of the pulp and periapical tissues. In: Shafer WG, Hine MK, Levy BM, editors. A Textbook of Oral Pathology. 4 th ed. Philadelphia, PA: WB Saunders Co.; 1993. p. 479-525.  Back to cited text no. 7
    
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Marx RE. Chronic osteomyelitis of the jaws. Oral Maxillofac Surg Clin North Am 1991;3:367-81.  Back to cited text no. 8
    
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Marx RE. Pamidronate (Aredia) and zoledronate (Zometa) induced avascular necrosis of the jaws: A growing epidemic. J Oral Maxillofac Surg 2003;61:1115-7.  Back to cited text no. 9
    
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Eng HL, Lu SY, Yang CH, Chen WJ. Oral tuberculosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1996;81:415-20.  Back to cited text no. 10
    
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Bhatt AP, Jayakrishnan A. Tuberculous osteomyelitis of the mandible: A case report. Int J Paediatr Dent 2001;11:304-8.  Back to cited text no. 11
    
12.
de Bont LG, van der Kuijl B, Stegenga B, Vencken LM, Boering G. Computed tomography in differential diagnosis of temporomandibular joint disorders. Int J Oral Maxillofac Surg 1993;22:200-9.  Back to cited text no. 12
    
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Cohnen M, Kemper J, Möbes O, Pawelzik J, Mödder U. Radiation dose in dental radiology. Eur Radiol 2002;12:634-7.  Back to cited text no. 13
    
14.
Scarfe WC, Farman AG, Sukovic P. Clinical applications of cone-beam computed tomography in dental practice. J Can Dent Assoc 2006;72:75-80.  Back to cited text no. 14
    
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Schuknecht BF, Carls FR, Valavanis A, Sailer HF. Mandibular osteomyelitis: Evaluation and staging in 18 patients, using magnetic resonance imaging, computed tomography and conventional radiographs. J Craniomaxillofac Surg 1997;25:24-33.  Back to cited text no. 15
    
16.
Chen CM, Lo LJ, Wong HF. Congenital infiltrating lipomatosis of the face: Case report and literature review. Chang Gung Med J 2002;25:194-200.  Back to cited text no. 16
    
17.
De Rosa G, Cozzolino A, Guarino M, Giardino C. Congenital infiltrating lipomatosis of the face: Report of cases and review of the literature. J Oral Maxillofac Surg 1987;45:879-83.  Back to cited text no. 17
    
18.
Slavin SA, Baker DC, McCarthy JG, Mufarrij A. Congenital infiltrating lipomatosis of the face: Clinicopathologic evaluation and treatment. Plast Reconstr Surg 1983;72:158-64.  Back to cited text no. 18
    
19.
Scherl MP, Som PM, Biller HF, Shah K. Recurrent infiltrating lipoma of the head and neck. Case report and literature review. Arch Otolaryngol Head Neck Surg 1986;112:1210-2.  Back to cited text no. 19
    
20.
Enzinger FM, Weiss SW. Benign lipomatous tumors. In: Weiss SW, Enzinger FM, editors. Soft Tissue Tumors. 3 rd ed. St. Louis, Missouri: Mosby; 1995. p. 381-430.  Back to cited text no. 20
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12]



 

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