Solitary primary intraosseous xanthoma of the mandible in a 15-year-old boy: a case report.
15-years-old boy
Case report
Mandible
Xanthoma
Journal
Journal of medical case reports
ISSN: 1752-1947
Titre abrégé: J Med Case Rep
Pays: England
ID NLM: 101293382
Informations de publication
Date de publication:
07 May 2024
07 May 2024
Historique:
received:
19
01
2024
accepted:
01
04
2024
medline:
7
5
2024
pubmed:
7
5
2024
entrez:
6
5
2024
Statut:
epublish
Résumé
A xanthoma is a rare bone condition consisting of a predominant collection of lipid-rich, foamy histiocytes. The central xanthoma of the jaws is a unique benign tumor. A 15-year-old Caucasian male has been presented to our department. He had radiological changes in the area of the left mandibular angle, with an area of diffuse osteolysis of 3.0 cm by 2.0 cm. Computed tomography reveals an area of diffuse osteolysis that starts from the distal root of the lower second molar and reaches the ascending process. A bone biopsy was performed, which revealed a benign proliferative process composed of histiocytic cells involving and infiltrating trabecular bone in a background of loose fibrous connective tissue devoid of any other significant inflammatory infiltrate. The size of the formation was 2.9 cm by 2.0 cm. Immunohistochemical staining for CD68 was strongly positive and negative for S-100 and CD1a. From routine blood tests, cholesterol, triglycerides, and blood sugar are within normal values, which excludes systemic metabolic disease. Subsequent to the surgical intervention, the patient underwent postoperative assessments at intervals of 14, 30, 60 days, and a year later, revealing the absence of any discernible complications during the aforementioned observation periods. The diagnosis of primary xanthoma of the mandible is rare and can often be confused with other histiocytic lesions. A differential diagnosis should be made with nonossifying fibroma and Langerhans cell histiocytosis, as in our case. In these cases, immunohistochemistry with CD 68, S-100, and CD1a, as well as blood parameters, are crucial for the diagnosis.
Sections du résumé
BACKGROUND
BACKGROUND
A xanthoma is a rare bone condition consisting of a predominant collection of lipid-rich, foamy histiocytes. The central xanthoma of the jaws is a unique benign tumor.
CASE REPORT
METHODS
A 15-year-old Caucasian male has been presented to our department. He had radiological changes in the area of the left mandibular angle, with an area of diffuse osteolysis of 3.0 cm by 2.0 cm. Computed tomography reveals an area of diffuse osteolysis that starts from the distal root of the lower second molar and reaches the ascending process. A bone biopsy was performed, which revealed a benign proliferative process composed of histiocytic cells involving and infiltrating trabecular bone in a background of loose fibrous connective tissue devoid of any other significant inflammatory infiltrate. The size of the formation was 2.9 cm by 2.0 cm. Immunohistochemical staining for CD68 was strongly positive and negative for S-100 and CD1a. From routine blood tests, cholesterol, triglycerides, and blood sugar are within normal values, which excludes systemic metabolic disease. Subsequent to the surgical intervention, the patient underwent postoperative assessments at intervals of 14, 30, 60 days, and a year later, revealing the absence of any discernible complications during the aforementioned observation periods.
CONCLUSION
CONCLUSIONS
The diagnosis of primary xanthoma of the mandible is rare and can often be confused with other histiocytic lesions. A differential diagnosis should be made with nonossifying fibroma and Langerhans cell histiocytosis, as in our case. In these cases, immunohistochemistry with CD 68, S-100, and CD1a, as well as blood parameters, are crucial for the diagnosis.
Identifiants
pubmed: 38711147
doi: 10.1186/s13256-024-04534-y
pii: 10.1186/s13256-024-04534-y
doi:
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
225Informations de copyright
© 2024. The Author(s).
Références
Alden KJ, McCarthy EF, Weber KL. Xanthoma of bone: a report of three cases and review of the literature. Iowa Orthop J. 2008;28:58–64. https://doi.org/10.1016/j.jdcr.2020.06.010 .
doi: 10.1016/j.jdcr.2020.06.010
pubmed: 19223950
pmcid: 2603351
Rudy HN, Scheingold SS. Solitary xanthogranuloma of the mandible; report of a case. Oral Surg Oral Med Oral Pathol. 1964;18:262–71.
doi: 10.1016/0030-4220(64)90436-0
pubmed: 14176614
Bonhomme GR, Loevner LA, Yen DM, Deems DA, Bigelow DC, Mirza N. Extensive intracranial xanthoma associated with type II hyperlipidemia. Am J Neuroradiol. 2000;21:353–5. https://doi.org/10.3174/ajnr.A1780 .
doi: 10.3174/ajnr.A1780
pubmed: 10696023
pmcid: 7975347
Harsanyi BB, Larsson A. Xanthomatous lesions of the mandible: osseous expression of non-X histiocytosis and benign fibrous histiocytoma. Oral Surg Oral Med Oral Pathol. 1988;65(5):551–66.
doi: 10.1016/0030-4220(88)90138-7
pubmed: 2836774
Bell A, Shreenath AP. Xanthoma. In: StatPearls. StatPearls Publishing; 2023.
Edelman A, Wetzel M, Owen C, Schadt CR, Callen JP. Xanthomas heralding pediatric coronary artery disease. JAAD Case Rep. 2020;6(8):753–4. https://doi.org/10.1016/j.jdcr.2020.06.010 .
doi: 10.1016/j.jdcr.2020.06.010
pubmed: 32715071
pmcid: 7369508
Dhanawat A, Padhan P. Tuberous xanthomas mimicking tophaceous gout. Indian J Med Res. 2020;152(Suppl 1):S29. https://doi.org/10.4103/ijmr.IJMR_1426_19 .
doi: 10.4103/ijmr.IJMR_1426_19
pubmed: 35345101
pmcid: 8257178
Morel D, Kelsch RD, Nolan PJ. Primary xanthoma of the mandible: report of a rare case. Head Neck Pathol. 2016;10(2):245–51. https://doi.org/10.1007/s12105-015-0643-z .
doi: 10.1007/s12105-015-0643-z
pubmed: 26254177
Hicks J, Flaitz CM. Langerhans cell histiocytosis: current insights in a molecular age with emphasis on clinical oral and maxillofacial pathology practice. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2005;100(2 Suppl):S42-66. https://doi.org/10.1016/j.tripleo.2005.06.016 .
doi: 10.1016/j.tripleo.2005.06.016
pubmed: 16037792
Kademani D, Patel SG, Prasad ML, Huvos AG, Shah JP. Intraoral presentation of Rosai-Dorfman disease: a case report and a case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2002;93(6):699–704. https://doi.org/10.1067/moe.2002.123495 .
doi: 10.1067/moe.2002.123495
pubmed: 12142877
Marques Mateo M, Puche Torres M, Miragall Alba L, Iglesias Gimilio ME, Pascual Gil JV. Primary mandibular bone xanthoma. A case report. Int J Oral Maxillofac Surg. 2004;33(8):806–7. https://doi.org/10.1016/j.ijom.2003.12.006 .
doi: 10.1016/j.ijom.2003.12.006
pubmed: 15556333
de Moraes Ramos-Perez FM, de Padua JM, Silva-Sousa YT, de Almeida OP, da Cruz Perez DE. Primary xanthoma of the mandible. Dentomaxillofac Radiol. 2011;40(6):393–6. https://doi.org/10.1259/dmfr/51850495 .
doi: 10.1259/dmfr/51850495
pubmed: 21831981
pmcid: 3520337
Daley T, Dunn G, Darling MR. Central xanthoma of the jaws: a clinicopathologic entity? Oral Surg Oral Med Oral Pathol Oral Radiol. 2015;119(1):92–100. https://doi.org/10.1016/j.oooo.2014.09.018 .
doi: 10.1016/j.oooo.2014.09.018
pubmed: 25446505
de Araujo MR, Scariot R, Uetanabaro L, Luvison Gomes da Silva L, Giovanini AF. Primary mandibular xanthoma: case report. Oral Surg Oral Med Oral Pathol Oral Radiol. 2015;120(4):177–82. https://doi.org/10.1016/j.oooo.2015.01.015 .
doi: 10.1016/j.oooo.2015.01.015
Rawal YB, Chandra SR, Hall JM. Central xanthoma of the jaw bones: a benign tumor. Head and Neck Pathol. 2017;11:192–202. https://doi.org/10.1007/s12105-016-0785-3 .
doi: 10.1007/s12105-016-0785-3
Shoor H, Pai KM, Shergill AK, Kamath AT. Benign fibrous histiocytoma: a rare case involving jaw bone. Contemp Clin Dent. 2015;6(Suppl 1):S266–8. https://doi.org/10.4103/0976-237X.166828 .
doi: 10.4103/0976-237X.166828
pubmed: 26604585
pmcid: 4632234
Bowers LM, Cohen DM, Bhattacharyya I, Pettigrew JC Jr, Stavropoulos MF. The non-ossifying fibroma: a case report and review of the literature. Head Neck Pathol. 2013;7(2):203–10. https://doi.org/10.1007/s12105-012-0399-7 .
doi: 10.1007/s12105-012-0399-7
pubmed: 23008139
Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and maxillofacial pathology. 3rd ed. St. Louis: Saunders; 2009.
Czerniak B. Bone tumors. 2nd ed. Philadelphia: Elsevier; 2016.
Heo MS, Cho HJ, Kwon KJ, Lee SS, Choi SC. Benign fibrous histiocytoma in the mandible. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2004;97(2):276–80. https://doi.org/10.1016/j.tripleo.2003.07.002 .
doi: 10.1016/j.tripleo.2003.07.002
pubmed: 14970788
Fink IJ, Lee MA, Gregg RE. Radiographic and CT appearance of intraosseous xanthoma mimicking a malignant lesion. Br J Radiol. 1985;58:262–4.
doi: 10.1259/0007-1285-58-687-262
pubmed: 4063668
Kyriakos M. Benign fibrous histiocytoma of bone. In: Fletcher CDM, Unni KK, Mertens F, editors. World health organization classification of tumours. Pathology and genetics of tumours of soft tissue and bone. Lyon: IARC Press; 2002. p. 292–3.
Muthusamy KA, Azmi K, Narayanan P, et al. Bilateral temporal bone xanthoma: case report. J Neurosurg. 2008;108:361–4. https://doi.org/10.3171/JNS/2008/108/2/0361 .
doi: 10.3171/JNS/2008/108/2/0361
pubmed: 18240935
Huang CF, Cheng SN, Hung CH, et al. Xanthoma of bone in the normolipidic child: report of one case. Acta Paediatr Taiwan. 2000;41:158–60.
pubmed: 10920551
Mosby EL, Albright JE, Messer EJ, Nealis MF, Werning JT. Clincopathologic conferences. Case 44, part I. J Oral Maxillofac Surg. 1983;41(3):180–1.
doi: 10.1016/0278-2391(83)90077-0
pubmed: 6572229
Mosby EL, Albright JE, Messer EJ, Nealis MF, Werning JT. Case 44, part II: xanthoma of the mandible. J Oral Maxillofac Surg. 1983;41(4):268–70.
doi: 10.1016/0278-2391(83)90274-4
pubmed: 6572700
Slootweg PJ, Swart JG, van Kaam N. Xanthomatous lesion of the mandible. Report of a case. Int J Oral Maxillofac Surg. 1993;22(4):236–7.
doi: 10.1016/S0901-5027(05)80644-7
pubmed: 8409567