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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 15  |  Issue : 3  |  Page : 302-305

Eosinophilic granuloma with metachronous actinomycosis – A case report with literature review


1 Department of Oral Pathology and Microbiology, Yenepoya Dental College, Yenepoya Deemed to be University, Mangalore, Karnataka, India
2 Centre for Forensic Odontology, Yenepoya Dental College, Yenepoya Deemed to be University, Mangalore, Karnataka, India

Date of Submission11-Mar-2022
Date of Acceptance28-Apr-2022
Date of Web Publication17-Sep-2022

Correspondence Address:
Dr. Sudeendra Prabhu
Centre for Forensic Odontology, Department of Oral Pathology, Yenepoya Dental College, Mangalore - 575 018, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/kleuhsj.kleuhsj_153_22

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  Abstract 


Eosinophilic granuloma (EG) is a type of histiocytosis X with uncertain etiology. The lesion manifests as single or multiple areas of bone destruction with mild-to-severe pain or may be asymptomatic in some cases. In this article, a case of EG with actinomycosis is reported which occurred in an 11-year-old male in the left buccal vestibule extending from 22 to 24. Histopathologically, it showed dense, cellular infiltrate in the connective tissue composed of abundant eosinophils intermixed with atypical cells. After 4 months, the same patient reported with a recurrence in the palatal region. Histopathologically, the section showed some colonies of actinomycosis along with the features of EG. EG with metachronous actinomycosis is very rare in the literature. This article reports a new case of EG with actinomycosis and reviews previously reported cases of actinomycosis associated with other lesions.

Keywords: Actinomycosis, eosinophilic granuloma, metachronus


How to cite this article:
Soundararajan K, Prabhu S, Sabarad P, Abdulla R. Eosinophilic granuloma with metachronous actinomycosis – A case report with literature review. Indian J Health Sci Biomed Res 2022;15:302-5

How to cite this URL:
Soundararajan K, Prabhu S, Sabarad P, Abdulla R. Eosinophilic granuloma with metachronous actinomycosis – A case report with literature review. Indian J Health Sci Biomed Res [serial online] 2022 [cited 2022 Dec 4];15:302-5. Available from: https://www.ijournalhs.org/text.asp?2022/15/3/302/356270




  Introduction Top


Lichtenstein and Jaffe in the year in 1940 first described the term eosinophilic granuloma (EG). Lichenstein considered EG, Hand–Schuller–Christian disease, and Letterer–Siwe disease under histiocytosis X because, in all these conditions, there is continuous proliferation of histiocytes.[1] The etiology is usually unknown, and it shows diverse manifestations but is most commonly caused by the abnormal proliferation of Langerhans cells. This proliferation of Langerhans cells may be due to viral infections such as Epstein–Barr virus, human herpesvirus-6, bacteria, and immune dysfunction trauma. Any bone can be involved; the more common sites include the skull, mandible, spine, ribs, and the long bones.[2] Clinically, very rarely, it may appear as a rapidly developing ulcer or indurated mass with a yellowish fibrinous base. EG appears in children and young adults but is common in all ages.[3] The same lesion occurring in infants and neonates is called as Riga–Fede disease.[4]

Microscopically, it shows numerous mononuclear histiocyte-like cells with irregular nuclear contours along with diffuse polymorphic cell infiltrate composed predominantly of eosinophils, necrosis, and inflammatory infiltrate involving both the superficial mucosa and submucosa.[5]

EG with metachronous actinomycosis is rare in the literature. Actinomyces is an anerobic, filamentous, Gram-positive bacterium which is found in the normal flora. It is classified according to the involvement of the area, in which craniofacial actinomycosis is the most common.[6] They have low pathogenicity but become pathologic when they enter into the subcutaneous tissue. Actinomycosis is generally afflicted to the soft tissues, but in fact, it has up to 15% of bone involvement.[7]

Here, we present a case of EG with metachronous actinomycosis in an 11-year-old male and to review the literature focusing on the etiopathogenesis and clinicopathologic features of this type of lesion in young patients.


  Case Report Top


An 11-year-old male patient reported to the outpatient department with pain and swelling on the left side of the face for the last 3 weeks. The swelling was seen a few days after a trivial toothbrush injury to the mucosa. Extraoral examination revealed facial asymmetry on the left side, and the overlying skin was warm and tender. Submandibular lymph node was tender and palpable. The patient visited a local dentist previously and was prescribed medication for pain and swelling.

Intraoral examination revealed an ulceroproliferative lesion with adherent pseudomembrane which extended from 22 to 25 regions. The lesion was not tender on palpation, and no pus discharge was noted. Cone-beam computed tomography (CBCT) revealed the decortication of the buccal plate from 23 to 24 regions. The clinical and radiological differential diagnoses were fungal infection and granulomatous lesion. An excisional biopsy was performed with adequate margins and sent to the department of oral pathology for histopathological diagnosis. On gross examination, the lesion measured 1.5 cm × 0.7 cm in diameter approximately. Hematoxylin- and eosin-stained sections revealed that the lesion is composed of abundant eosinophils intermixed with atypical cells and small round lymphocytes in the connective tissue. The atypical cells were mitotically active, large with irregular nuclear contours, multiple nucleoli, and abundant cytoplasm and stained positive for CD 1a [Figure 1]. Based on the clinical, histopathologic, and immunohistochemical findings, the final diagnosis of EG was made.
Figure 1: CD 1a positive lesional cells

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The same patient reported after 4 months with a swelling on the anterior palate extending from 23 to 24 regions. It was measuring about 3 cm × 3 cm. There were no associated symptoms and lymph node involvement. There were no radiographic findings. An excisional biopsy was done with adequate margins and sent to the department of oral pathology and microbiology for histopathological diagnosis. Grossly, the lesion was measuring about 3 cm × 2 cm approximately. Hematoxylin- and eosin-stained sections revealed stratified squamous epithelium with varying degrees of keratinization. The underlying connective tissue stroma showed extensive bundles of dense collagen fibers with inflammatory cells predominantly eosinophils, lymphocytes, and plasma cells [Figure 2]. The section showed colonies of actinomycosis along with dispersed actinomycete filaments which are found intervening in some areas [Figure 3] and [Figure 4]. Correlating with the clinical history and the histopathological features, the diagnosis was given as actinomycosis.
Figure 2: The H and E image showing the lesion with parakeratinized stratified squamous epithelium and granulomatous changes under × 4 magnification

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Figure 3: The H and E image of the lesion showing the colonies of actinomycosis under × 10 magnification

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Figure 4: The lesion showing numerous filaments of actinomycosis which stained positive for Periodic Acid Schiff under × 20 magnification

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  Discussion Top


EG is an obscure lesion with uncertain etiology and pathogenesis, but the current theory suggests that immunological factors may initiate the process. EG is a rare condition which accounts for 4 to 5 cases per million in a year. Most commonly, it occurs in young children below the age of 15 years with a mean age of 5.5 years. Males are being commonly affected than females.[1],[8]

Histopathologically, the eosinophilic granuloma is characterized by the presence of abundant histiocytes with prominent nuclear grooves admixed with a dense polymorphic infiltrate which is seen extending into the underlying mucosa. The presence of eosinophils is not completely understood.[9]

The suggested pathogenesis of the lesion is not clear, but it may occur as a manifestation of the immunological process.[2] One of the characteristic features is the abnormal proliferation of the Langerhans cells which may be a neoplastic or reactive process. This proliferation may be due to bacterial or viral infections and immunological dysfunction which leads to an increase in cytokines.[10]

Eosinophilic granuloma with metachronous actinomycosis is very unique because no such cases have been reported in the literature. In the present article, we are going to review the cases of actinomycosis correlating with the following conditions,

Zohreh et al. reported a case of actinomyces colonization in the keratocystic odontogenic tumors. In this study, the actinomycosis colonies were found to be in association with one out of two nonsyndromic multiple keratocystic odontogenic tumors. This study also showed only a limited number of colonies without a sufficiently great inflammatory cell infiltration around them, and the swelling may be related to diffuse inflammation of the cyst wall rather than the actinomyces colonies.[11]

Maiko et al. reported a case of actinomyces-associated calcifications in the dentigerous cyst of the mandible. In this case, there were multiple black actinomyces-associated calcifications in the mandible. They considered that actinomyces infected the cystic lesion around the crown of the right mandibular third molar from the pathologic periodontal pocket as a result of severe periodontitis of the right mandibular second molar. This corresponds to the second possible explanation of the relation between actinomyces and odontogenic cyst formation mentioned previously, namely, that after the cyst formation, actinomyces proliferated in hemorrhagic contents, ultimately filling the cystic cavity.[12]

Ahmed et al. reported a case of inflamed odontogenic cyst with actinomyces colonization in the mandible. In this report, the authors explained that it could be a reactive inflammatory odontogenic cyst which is stimulated by some coronal remnants of the prior deciduous tooth infected by actinomyces that gained access from the draining fistula. A retained root tip was seen by CBCT just apical to the lesion.[13]

Miller et al. reported a case of mandibular actinomyces osteomyelitis complicating florid cemento-osseous dysplasia. This case indicates an association between two chronic, destructive, and disfiguring conditions of the mandible, FCOD, and actinomyces osteomyelitis. “Ginger-root” pattern is the characteristic feature of FCOD. These typical findings of FCOD were seen in combination with the important features of actinomyces osteomyelitis which shows a filamentous Gram-positive organism, acute inflammation, and necrotic bone.[14]

[Table 1] discusses all the cases of actinomycosis with associated lesions.
Table 1: Actinomycosis co-existing with other oral lesions

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  Conclusion Top


The present case report demonstrates the importance of the practice of differential diagnosis with a multidisciplinary approach for accurate diagnosis and treatment planning. Furthermore, the detailed systemic and dental history of the patient is helpful in the treatment. The present case report is very rare in the literature which demonstrates the importance of differential diagnosis with a multidisciplinary approach for accurate diagnosis and treatment. Therefore, detailed history of the patients should be taken which will be helpful in the management of the disease.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The guardian understands that names and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Jha SK, De Jesus O. Eosinophilic Granuloma. Florida, USA: StatPearls; 2021.  Back to cited text no. 1
    
2.
Kaul R, Gupta N, Gupta S, Gupta M. Eosinophilic granuloma of skull bone. J Cytol Indian Acad Cytologists 2009;26:156.  Back to cited text no. 2
    
3.
Sherwani RK, Akhtar K, Qadri S, Ray PS. Eosinophilic granuloma of the mandible: A diagnostic dilemma. BMJ Case Rep 2014;2014:10:1-4.  Back to cited text no. 3
    
4.
Park SH, Park J, Hwang JH, Hwang SK, Hamm IS, Park YM. Eosinophilic granuloma of the skull: A retrospective analysis. Pediatr Neurosurg 2007;43:97-101.  Back to cited text no. 4
    
5.
Dhull AK, Aggarwal S, Kaushal V, Singh S. Into the wild world of eosinophilic granuloma. Case Reports. 2013 Nov 19;2013:bcr2013200522  Back to cited text no. 5
    
6.
Valour F, Sénéchal A, Dupieux C, Karsenty J, Lustig S, Breton P, et al. Actinomycosis: Etiology, clinical features, diagnosis, treatment, and management. Infect Drug Resist 2014;7:183-97.  Back to cited text no. 6
    
7.
D'Amore F, Franchini R, Moneghini L, Lombardi N, Lodi G, Sardella A, et al. Actinomycosis of the tongue: A case report and review of literature. Antibiotics (Basel) 2020;9:E124.  Back to cited text no. 7
    
8.
Allen CE, Ladisch S, McClain KL. How I treat langerhans cell histiocytosis. Blood 2015;126:26-35.  Back to cited text no. 8
    
9.
Haouimi AS, Al-Hawsawi ZM, Jameel AN. Unusual location of eosinophilic granuloma. Saudi Med J 2004;25:1489-91.  Back to cited text no. 9
    
10.
Cambazard F, Dezutter-Dambuyant C, Staquet MJ, Schmitt D, Thivolet J. Eosinophilic granuloma of bone and biochemical demonstration of 49-kDa CD1a molecule expression by langerhans-cell histiocytosis. Clin Exp Dermatol 1991;16:377-82.  Back to cited text no. 10
    
11.
Jafari-Ashkavandi Z, Kamali F. Actinomyces colonization in keratocystic odontogenic tumor: A case report. J Dent (Shiraz) 2015;16:134-7.  Back to cited text no. 11
    
12.
Shibasaki M, Iwai T, Chikumaru H, Mitsudo K, Inayama Y, Tohnai I. Actinomyces-associated calcifications in a dentigerous cyst of the mandible. J Craniofac Surg 2013;24:e311-4.  Back to cited text no. 12
    
13.
Soolari A, Soolari A, Fielding C. Inflamed odontogenic cyst with Actinomyces colonization: Management of an atypical case in a 16-year-old patient. Int J Periodontics Restorative Dent 2020;40:e35-41.  Back to cited text no. 13
    
14.
Smith MH, Harms PW, Newton DW, Lebar B, Edwards SP, Aronoff DM. Mandibular Actinomyces osteomyelitis complicating florid cemento-osseous dysplasia: Case report. BMC Oral Health 2011;11:21.  Back to cited text no. 14
    
15.
Sun Y, Kuyama K, Fifita SF, Nogami T, Fukui K, Kondoh T, et al. Actinomycosis in radicular cyst: A case report and literature survey. Oral Med Pathol 2010;14:113-6.  Back to cited text no. 15
    
16.
Finley AM, Beeson MS. Actinomycosis osteomylelitis of the mandible. Am J Emerg Med 2010;28:4.e1-4.  Back to cited text no. 16
    
17.
Gannepalli A, Ayinampudi BK, Baghirath PV, Reddy GV. Actinomycotic osteomyelitis of maxilla presenting as oroantral fistula: A rare case report. Case Rep Dent 2015;2015:689240.  Back to cited text no. 17
    
18.
Baldawa P, Shirol P, Kulkarni D, Koshti S, Mishra N. Actinomycotic osteomyelitis of palate masquerading periapical pathology: A rare case report. Indian J Dent Res 2018;29:247-51.  Back to cited text no. 18
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19.
Kaldas M, Barghorn A, Schmidlin PR. Actinomycosis as a rare local manifestation of severe periodontitis. Case Rep Dent 2020;2020:5961452.  Back to cited text no. 19
    


    Figures

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