Year : 2021 Month : May Volume : 10 Issue : 22 Page : 1741-1745

A Non-Sinus Forming Mandibular Actinomycotic Osteomyelitis with a Submandibular Gland Swelling - A Case Mimicking a Periapical Infection

Suma Gundareddy Nagendra Reddy1, Ravina2, Dayashankara Rao Jingade Krishnojirao3 Debdip Mandal4

1, 2, 4 Department of Oral Medicine & Radiology, SGT University, Gurugram, Haryana, India. 3Department of Maxillofacial Surgery & Diagnostic Sciences. College of Dentistry, Qassim University. Saudi Arabia.

CORRESPONDING AUTHOR

Dr. Suma Gundareddy Nagendra Reddy, E 101, RV 2, Plot 16, Sector 23, Dwarka - 110077, Delhi, India.
Email : sumagndaya@gmail.com

INTRODUCTION

Involvement of the submandibular gland in actinomycotic osteomyelitis in the absence of sinus is rare. Cervicofacial form is characterized by contiguous spread, suppurative osteomyelitic & granulomatous inflammation of the mandible and formation of multiple abscesses and hallmark discharging sinuses draining sero- sanguinous fluid containing sulphur granules.

As the imaging finding of this entity is rarely described, in this case report, a rare case of mandibular actinomycotic osteomyelitis, with a submandibular swelling, but without draining sinus is reported. The imaging findings of cone beam computed tomography (CBCT) & ultrasonography (US) of the case are discussed with a review.

In subacute stage, mandibular actinomycosis may show no hallmark sinuses. Sclerotic margins around the lesion may be found on radiograph. Imaging is useful in ruling out clinical diagnostic challenge when it includes involvement of mandible & submandibular region.

Cervicofacial actinomycosis commonly occurs as perimandibular infection, rarely as osteomyelitis of the mandible.1 It may present as two distinct morphological patterns; first, "lumpy jaw," and second, simulating an acute pyogenic infection affecting the submandibular area, discharging sinus being a hallmark finding.1 Other variant reported include chronic osteitis, osteolytic lesion, hard nodule on the tongue, lockjaw, periapical, or paradental abscess. Diagnosis of cases presenting in multiple areas in the absence of multiple discharging sinuses is a challenge.2 Usefulness the imaging findings are rarely enumerated.

This report is of a twin presentation of a non - sinus forming actinomycotic mandibular osteomyelitis and a submandibular swelling, along with a review of various diagnostic imaging features.

PRESENTATION OF CASE

In the third decade, a male patient presented with six - month - old painful swelling below the angle of the lower jaw. History revealed, fracture of the right mandible in an accident a year back, treated with inter-maxillary fixation. A few months later, the patient returned with a painful right mandibular tooth mimicking a periapical infection. The attempted endodontic treatment failed to relieve the symptoms. The tooth was extracted due to a persistent infection, in spite of endodontic treatment. Three months later, a soft and painless swelling appeared in the right submandibular region, which gradually enlarged to become firm and painful. There was no history of draining sinus, fever, cough, weight loss. Medical, family, and psychosocial history was unremarkable. Despite the repeated medication of antibiotics and analgesics, partial remissions and exacerbations of the swelling continued.

Extraoral examination revealed a diffuse mild swelling over the right angle of the mandible, and an oval, 2.5 cm x 3.5 cm, swelling in the ipsilateral submandibular region 1 cm anterior to the sternocleidomastoid muscle, 1 cm below the lower border of the mandible, with well - defined borders, smooth surface, & bluish - purple coloured overlying skin. (Figure1). Swelling in the submandibular gland region was firm, tender, non – fluctuant. The skin over the swelling was indurated, but no sinus/pus discharge was present. Right submandibular tender lymphadenopathy was present.

On intraoral examination, there was no source of dental sepsis in the molar region. On palpation, the vestibule showed mild obliteration of the vestibule and tenderness. The panoramic image revealed ill-defined radiolucency, 3.05 x 1.68 cm in size, in the mandible extending from the second molar till the anterior ramus, resorbing the external oblique ridge. The lesion showed a moth-eaten appearance, with irregular

sclerotic borders. (Fig. 2). CBCT showed an osteolytic lesion,

 

mild expansion of the buccolingual cortical plate predominantly in tooth no. 48 region, effacement, and disruption of the lingual cortex, with irregular areas of dense trabecular pattern (Fig. 3). Radiographic impression was chronic osteomyelitis of the mandible.

 

 

 

An ultrasound examination suggested an inflammatory lesion in the submandibular gland. It showed a roughly circular hypoechogenic area in the right submandibular region measuring 2.1 x 1.4 cm, with multiple echogenic foci, the largest measuring 2.5 mm, oedematous changes in the overlying skin, subcutaneous tissue. (Fig. 3) Fine needle aspiration cytology (FNAC) from the submandibular swelling confirmed the final diagnosis as actinomycosis. (Fig. 3)

 

 

 

CLINICAL DIAGNOSIS

Inflammatory swelling of the right submandibular space, secondary to a residual infectious source in the tooth 48 region.

DIFFERENTIAL DIAGNOSIS

Residual abscess, chronic suppurative osteomyelitis, antibioma, and TB lymphadenitis

PATHOLOGICAL DISCUSSION

Actinomyces, earlier classified as a fungal lesion, are now classified as bacteria.3 (Fig. 4). The spores follow a cycle of haploid & diploid generations by the process of reduction division and conjugation. Spores gain access into the body in the presence of characteristic risk factors.4 (Fig. 5). Cervical actinomycotic infection without the hallmark draining sinus is rare. Characteristic series of diagnostic manifestations are chronicity, extensive propagation across tissue planes, and firm to hard mass like features often mimicking malignancy, multiple draining sinuses, and frequent remissions & relapses.5 The most com­monly involved site is the angle of the jaw or the submandibular region, followed by cheek, submental space, masticator space, and temporomandibular joint.6 As in the present case, dual presentation of mandibular osteomyelitis and a submandibular swelling, in the absence of discharging sinus, may be mistaken as dental abscess involving the submandibular space. However, 40 % of cases report a discharging sinus. It is not clear if this finding is absent in patients with good host resistance, low virulence of the organism, or subclinical disease.

Cervicofacial actinomycosis infection of the mandible is often a peri mandibular, granulomatous infection. However, the mandibular osteomyelitis with a submandibular gland swelling is rare, as reported here. Bone infection (periostitis and osteomyelitis) may develop in 11.7 % of cases.7 Reports show sclerosis within the lesion, but not a sclerotic periphery, around a minimal expansile, moth-eaten type of radiolucency as seen in the present case. It is not clear if this characteristic is pathognomonic of the disease or is a feature in subclinical forms.

Not many studies reported CBCT findings of cervicofacial actinomycosis. In the present case, CBCT was useful in showing the buccolingual expansion, irregular areas of dense trabecular pattern, disruption of the lingual cortex, & status of the mandibular canal. Various imaging findings of actinomycotic mandibular osteomyelitis, described in the literature reported between 1970 to 2017, include lytic and sclerotic lesion, minimal expansion, destruction of cortices on computerised tomography (CT), heterogeneous lesion on magnetic resonance imaging (MRI), homogenous radiotracer uptake on bone scan. The lesion may extend into the adjacent skin, fat tissues, muscle, masticator spaces, and the salivary gland.1,2,5-16 (Table 1).

 

 

Ultrasound examination of cervical swelling is useful to rule out other causes like submandibular gland involvement, tubercular lymphadenitis, and other tumours. Reports on US findings are rare. In the present case, the US showed hypoechoic areas with multiple echogenic areas. On US examination, an actinomycotic lesion showed a mass with relatively homogeneous enhance­ment that has small necrotic areas or cystic por­tions. The abundant granulation tissue, fibrosis & necrosis may result in such enhancement on ultrasound imaging.6 In the subacute stage, mandibular actinomycosis may present as a slowly progressing osteomyelitis with no draining sinuses that may easily mimic a periapical infection. Sclerotic margins around the lesion may be found on the radiograph. Involvement of mandible & submandibular region challenges clinical diagnostic skills, & CBCT, US are useful in such cases. Prolonged treatment is required to control the disease, as in the present case.

DISCUSSION OF MANAGEMENT

Pharmacotherapy included-Amoxycillin and Clavulanic acid 625mg TID and Diclofenac Sodium BID & Serratiopeptidase. six months of follow up gave complete remission of clinical signs and symptoms. (Fig. 1B), The post-therapy panoramic radiograph showed partial resolution of the lesion. (Fig. 2B.) (Time line of the case is given in the table). The patient reported no adverse event. He reported improved quality of life in the physical, psychological, and social domains.

 

FINAL DIAGNOSIS

A non-sinus forming mandibular subacute actinomycotic osteomyelitis with submandibular gland involvement.

REFERENCES

1

Moniruddin ABM, Begum H, Nahar K. Actinomycosis: an update. Medicine Today 2010;22(1):43-7.                          

Google Scholar |
2

Kura MM, Rane VK. Cervicofacial actinomycosis mimicking lymphangioma circumscriptum. Indian J Dermatol 2011;56(3):321-3.            

CrossRef | Google Scholar | PubMed
3

Lee YC, Lim LR, Lee KH, et al. Actinomycotic osteomyelitis of the mandible: a case report. J Oral Med Pain 2019;44(3):140-4.           

Google Scholar |
4

Klieneberger-Nobel E. The life cycle of sporing actinomyces as revealed by a study of their structure and septation. J Gen Microbiol 1947;1(1):22-32.         

CrossRef | Google Scholar | PubMed
5

Sezer B, Akdeniz BG, Günbay S, et al. Actinomycosis osteomyelitis of the jaws: report of four cases and a review of the literature. J Dent Sci 2013;12(3):301-7.                            

CrossRef | Google Scholar | PubMed
6

Heo SH, Shin SS, Kim JW, et al. Imaging of actinomycosis in various organs: a comprehensive review. Radiographics 2014;34(1):19-33.            

CrossRef | Google Scholar | PubMed
7

Wong VK, Turmezei TD, Weston VC. Actinomycosis. BMJ 2011;343:d6099.                       

CrossRef | Google Scholar | PubMed
8

Yuki K, Kazuyuki A, Tomomi H, et al. CT and US findings of actinomycosis of the jaw. J Global 2001;41(4):231-9.

9

Figueiredo LMG, Trindade SC, Sarmento VA, et al. Actinomycotic osteomyelitis of the mandible: an unusual case. Oral Maxillofac Surg 2013;17(4):299-302.                                          

CrossRef | Google Scholar | PubMed
10

Smith MH, Harms PW, Newton DW, et al. Mandibular Actinomyces osteomyelitis complicating florid cemento- osseous dysplasia: case report. BMC Oral Health 2011;11(1):21.               

CrossRef | Google Scholar | PubMed
11

Abbaszadeh H, Sheibani MS. Actinomycotic osteomyelitis of mandible. J Craniofac Surg 2016;27(5):e452-4.                               

CrossRef | Google Scholar | PubMed
12

Kirsch WM, Stears JC. Actinomycotic osteomyelitis of the skull and epidural space: case report. J Neurosurg 1970;33(3):347-51.         

CrossRef | Google Scholar | PubMed
13

Volante M, Contucci AM, Fantoni M, et al. Cervicofacial actinomycosis: still a difficult differential diagnosis. Acta Otorhinolaryngol Ital 2005;25(2):116-9.                      

CrossRef | Google Scholar | PubMed
14

Sasaki Y, Kaneda T, Uyeda JW, et al. Actinomycosis in the mandible: CT and MR findings. AJNR Am J Neuroradiol 2014;35(2):390-4.            

CrossRef | Google Scholar | PubMed
15

Valour F, Sénéchal A, Dupieux C, et al. Actinomycosis: etiology, clinical features, diagnosis, treatment and management. Infect Drug Resist 2014;7:183-97.

CrossRef | Google Scholar | PubMed
16

Sasaki Y, Ono J, Akashiba T, et al. Diagnostic value of computed tomography, magnetic resonance imaging, and scintigraphy in diagnosing actinomycosis of the mandible. Oral Radiology 2017;33(3):241-5.        

Google Scholar |
17

Belmont MJ, Behar PM, Wax MK. Atypical presentations of actinomycosis. Head Neck 1999;21(3):264-8.                        

CrossRef | Google Scholar | PubMed

DISCLOSURE AND FUNDING

Disclosure forms provided by the authors are available with the full text of this article at jemds.com

ICMJE Forms

Financial or other competing interests: None.

Disclosure forms provided by the authors are available with the full text of this article at jemds.com.

How to cite this article

Reddy SGN, Ravina, Krishnojirao DRJ, et al. A non-sinus forming mandibular actinomycotic osteomyelitis with a submandibular gland swelling - a case mimicking a periapical infection. J Evolution Med Dent Sci 2021;10(22):1741-1745, DOI: 10.14260/jemds/2021/360

Videos :

watch?v