A Case of Epulis Granulomatosa With Clinical and Radiological Findings Resembling Langerhans Cell Histiocytosis

Abstract

Aim Langerhans cell histiocytosis (LCH) is a rare disease characterized by the proliferation of Langerhans cells. The differential diagnosis of LCH from epulis granulomatosa may be more difficult, given that the major symptoms of LCH are swelling and a floating tooth appearance. The purpose of this study was to provide a case of epulis granulomatosa that resembled LCH in the jaw bones based on clinical and radiological symptoms, as well as to warn against the possibility of misdiagnosis. Case Report Smooth-surfaced, lobular, erythematous, sessile, and fibrotic tissue hyperplasias were seen in the maxilla anterior edentulous area and the mandible’s left posterior region. The patient’s radiographic findings revealed extensive alveolar bone destruction in the maxilla and mandible, as well as severe bone destruction in the posterior area of the left mandible, consistent with a floating tooth. Under local anesthetic, an excisional biopsy of the lesion was performed in the left posterior region of the mandible. Histopathological examination revealed that the patient had inflammatory fibrous tissue hyperplasia. Discussion The clinical symptoms of LCH patients vary depending on their location and degree of involvement. When completing a full mouth examination on a patient, it is critical to detect soft tissue abnormalities as well as provide an accurate diagnosis and treatment plan. Conclusion LCH, together with surrounding inflammatory alterations, should be considered in the differential diagnosis of osteolytic lesions of the jaw.

Keywords

• Differential diagnosis
• Epulis granulomatosa
• Histopathologic examination
• Langerhans cell histiocytosis
• Oral diagnosis

References

1. Harmon CM, Brown N. Langerhans cell histiocytosis a clinicopathologic review and molecular pathogenetic update. Archives of Pathology & Laboratory Medicine. 2015; 139(10): 1211–1214.
2. Altay MA, Sindel A, Özalp Ö, Kocabalkan B, Özbudak İH, Erdem R, Salim O, Baur DA. Langerhans Cell Histiocytosis: A Diagnostic Challenge in the Oral Cavity. Case Rep Pathol. 2017; 2017:1691403.
3. Lian C, Lu Y, Shen S. Langerhans cell histiocytosis in adults: A case report and review of the literatüre. Oncotarget. 2016; 7(14):18678–18683.
4. Stalemark H, Laurencikas E, Karis J , Gavhed D, Fadeel B, Henter JI. Incidence of Langerhans cell histiocytosis in children: A population-based study. Pediatric Blood & Cancer. 2008; 51(1):76–81.
5. Morcel M, Keribin P, Quenel L, Bertin H, Neel A, Lesclous P. Diagnosis, treatment and recurrence of a mandibular Langerhans cell histiocytosis: a three-year follow-up case report. J Oral Med Oral Surg. 2022; 28:46.
6. Aricò M, Girschikofsky M, Généreau T, Klersy C, McClain K, Grois N, et al. Langerhans cell histiocytosis in adults. Report from the International Registry of the Histiocyte Society. Eur J Cancer. 2003; 39:2341–2348.
7. AbdullGaffar B, Awadhi F. Oral manifestations of Langerhans cell histiocytosis with unusual histomorphologic features. Ann Diagn Pathol. 2020;47:151536.
8. Lau SK,Chu PG,Weiss LM.ImmunohistochemicalexpressionofLan- gerin in Langerhans cell histiocytosis and non-Langerhans cell histiocytic disorders. Am J Surg Pathol. 2008;32(4):615-619.

9. Allen CE, Li L, Peters TL, et al. Cell-specific gene expressions in Langerhans cell histiocytosis lesions reveals a distinct profile compared with epidermal Langerhans cells. J lmmunol. 2010;184(8):4557-4567.
10. Pekiner FN, Borahan MO , Özbayrak S, Alatlı C, Kızılyel G. Oral manifestations of chronic disseminated langerhans cell histiocytosis: A case report. Clinical and Experimental Health Sciences. 2014; 2(3): 138-142.
11. Zheng JW. The terminology and treatment of epulis. Chin J Oral Maxillofac Surg 2010;8(3):287–8.
12. Zheng JW, Zhou Q, Yang XJ, He Y, Wang YA, Ye WM, et al. In- tralesional injection of Pingyangmycin may be an effective treatment for epulis. Med Hypotheses. 2009;72:453
13. Jafarzadeh H, Sanatkhani M, Mohtasham N. Oral pyogenic granu- loma: a review. J Oral Sci. 2006;48:167-75.
14. Sümer P, Bodur FZ. Langerhans hücresi histiositozisi: Olgu sunumu. Uluslararası Diş Hekimliği Bilimleri Dergisi. 2019; 5(3): 62-65.
15. Günaldı Ö, Tuğcu B, Tanrıverdi O, Gümüş E, Güler AK, Eseoğlu M. Parietal kemik yerleşimli langerhanc hücreli histiositoz: Olgu sunumu J Neurol Sci. 2008;25:200-204.
16. Islinger RB, Kuklo TR, Owens BD, Horan PJ, ChomaTJ, Murphey MD, Temple HT. Langerhans’ cell histiocytosis in patients older than 21 years. Clin Orthop Relat Res.2000;379:231-23.
17. White SC, Pharoach MJ. Oral Radiology Principles and Interpretation. 4th ed. Philadelphia: Mosby Inc; 2000.
18. Yashoda Devi BK, Rakesh N, Agarwal M. Langerhans cell histiocytosis with oral manifestations: a rare and unusual case report. J Clin Exp Dent. 2012;4:e252-255.
19. Donadieu J, Chalard F, Jeziorski E. Medical management of Langerhans cell histiocytosis from diagnosis to treatment. Expert Opin Pharmacother. 2012; 13:1309-1322.
20. Gonçalves CF, Morai MO, de Cassia Gonçalves Alencar R, Batista AC, Mendonça EF. Solitary langerhans cell histiocytosis in an adult: case report and literature review. BMC Res Notes. 2016;9:19
21. Muramatsu T, Hall GL, Hashimoto S, Miyauchi J, Shimono M. Clinico-pathologic conference: case 4. Langerhans cell histiocytosis (LCH) Head Neck Pathol. 2010;4:343-346.
22. Kapukaya A, Işık R, Alemdar C, Yıldırım A. Langerhans hücreli histiyositoz TOTBİD Dergisi. 2013;12:547-556.
23. Madrigal-Martínez-Pereda C, Guerrero-Rodríguez V, Guisado-Moya B, Meniz-García C. Langerhans cell histiocytosis: literature review and descriptive analysis of oral manifestations. Med Oral Patol Oral Cir Bucal. 2009;14:E222–228.
24. Atarbashi Moghadam S, Lotfi A, Piroozhashemi B, Mokhtari S. A retrospective analysis of oral langerhans cell histiocytosis in an Iranian population: a 20-year evaluation. J Dent (Shiraz) 2015;16 (3 Suppl.):274–277.
25. Faustino ISP, Fernandes PM, Pontes HAR, Mosqueda-Taylor A, Santos-Silva AR, Vargas PA, et al. Langerhans cell histiocytosis in the oral and maxillofacial region: an update. J Oral Pathol Med.2021;50:565–571.
26. Giovannetti F, Aboh IV, Chisci G, Gennaro P, Gabriele G, Cascino F, et al. Langerhans cell histiocytosis: treatment strategies. J Craniofac Surg. 2014; 25:1134–1136.
27. Gadner H, Grois N, Arico M, Broadbent V, Ceci A, Jakobson A, et al. A randomized trial of treatment for multisystem Langerhans’ cell histiocytosis. J Pediatr. 2001; 138:728–734.
28. Kim JE, Yi WJ, Heo MS, Lee SS, Choi SC, Huh KH. Langerhans cell histiocytosis of the jaw, a mimicker of osteomyelitis on CT and MR images: A retrospective analysis. Medicine (Baltimore). 2019;98(27):e16331.
29. Azariah ED, Chandrasekaran D, Chinnaswami R, Balasubramaniam S, Jagdish E. Histiocytosis X- a rare case report. J Clin Diagn Res. 2016; 10:ZD19-22.
30. Kumar YP, Agrawal J, Mohanlakshmi J, Kumar PS. Langerhans cell histiocytosis revisited: Case report with review. Contemp Clin Dent. 2015;6(3):432-6.