Langerhans Cell Histiocytosis in Bilateral Basis Mandibularis: A Rare Case

Year 2023, , 154 - 157, 26.12.2023

Lokman Cin , Rabia Duman Tepe , Hülya Çakır Karabaş

https://doi.org/10.29228/erd.60

Abstract

Langerhans cell histiocytosis (LCH), an autoimmune disease, is a rare group of diseases characterized by abnormal proliferation of atypical histiocytes originating from the bone marrow. A 57-year-old male patient with no systemic disease applied to our clinic for consultation. The patient had mild pain in the bilateral mandible corpus and itching on the skin. CBCT examination revealed bilateral destruction areas on the mandible basis. No bone expansion and periosteal reaction were observed. The patient diagnosed with LCH was accepted as an eosinophilic granuloma variant since no extraskeletal extension was observed. This case report aims to present a rare case of LCH that destroyed the bilateral basis mandible. In addition, it is to present the radiographic data of this disease, in which dentists play an important role in early diagnosis since the first involvement area is usually the head and neck region.

Keywords

Langerhans Hücre Histiyositozu, eozinofilik granülom, cone beam computer tomography

Bilateral Basis Mandibulariste görülen Langerhans Hücreli Histiyositozis: Nadir Bir Olgu

Abstract

Otoimmün bir hastalık olan Langerhans hücreli histiyositoz (LHH), kemik iliği kaynaklı atipik histiositlerin anormal proliferasyonu ile karakterize nadir görülen bir hastalık grubudur. Sistemik hastalığı olmayan 57 yaşında erkek hasta kliniğimize konsültasyon amacı ile başvurdu. Hastada bilateral mandibula korpusta hafif düzeyde ağrı ve deride kaşıntı şikayeti mevcuttu. KIBT incelemesinde mandibula alt sınırında baziste, bilateral yıkım alanları izlendi. Kemikte ekspansiyon ve periost reaksiyonu izlenmedi. LHH tanısı alan hastada ekstraskeletal yayılım görülmediğinden eozinofilik granülom varyantı olarak kabul edildi. Bu olgu sunumunun amacı, bilateral basis mandibulada yıkıma sebep olmuş nadir yerleşimli LHH olgusunu sunmaktır. Ayrıca ilk tutulum bölgesi genellikle baş ve boyun bölgesi olması nedeniyle diş hekimlerinin erken tanıda önemli rol aldığı bu hastalığın radyografik verilerini sunmaktır.

Keywords

Langerhans Hücre Histiyositozu, Konik Işınlı Bilgisayarlı Tomografi, eozinofilik granülom

References

• Aruna D., Pushpalatha G., and Galgali S., Langerhans cell histiocytosis. Journal of Indian Society of Periodontology, 2011. 15(3): p. 276.
• Atalay F., E.A.Ö. Koç, and S. Yıldız, A rare presentation of langerhans cell histiocytosis tonsil infiltration: review of the literature: atypical presentation of langerhans cell histiocytosis. Indian Journal of Hematology and Blood Transfusion, 2014. 30(1): p. 437-439.
• Coleman M.A., et al., Bilateral temporal bone langerhans cell histiocytosis: radiologic pearls. The open neuroimaging journal, 2013. 7: p. 53.
• Fang J. and Jiang Z., Case Report Langerhans cell histiocytosis (LCH) occurring in the skull: report of 3 cases and review of the literature. Int J Clin Exp Pathol, 2017. 10(1): p. 695-701.
• Grois N., et al., Risk factors for diabetes insipidus in langerhans cell histiocytosis. Pediatric blood & cancer, 2006. 46(2): p. 228-233.
• Hwang D.-S., et al., Langerhans cell histiocytosis of the mandible: two case reports and literature review. Journal of the Korean Association of Oral and Maxillofacial Surgeons, 2019. 45(3): p. 167-172.
• Kapukaya A., et al., Langerhans-hücreli histiyositoz. TOTBID Dergisi, 2013. 12(6): p. 547-56.
• Koong, Bernard. Atlas of oral and maxillofacial radiology. John Wiley & Sons, United States 2017.
• Kumar Y.P., et al., Langerhans cell histiocytosis revisited: Case report with review. Contemporary Clinical Dentistry, 2015. 6(3): p. 432.
• Lajolo C., et al., Langerhans’s cell histiocytosis in old subjects: two rare case reports and review of the literature. Gerodontology, 2012. 29(2): p. e1207-e1214.
• Lieberman P.H., et al., Langerhans cell (eosinophilic) granulomatosis: a clinicopathologic study encompassing 50 years. The American journal of surgical pathology, 1996. 20(5): p. 519-552.
• Luz J., et al., Oral manifestation of Langerhans cell histiocytosis: a case report. BMC Oral Health, 2018. 18(1): p. 1-6.
• Madrigal-Martínez-Pereda C., et al., Langerhans cell histiocytosis: literature review and descriptive analysis of oral manifestations. Medicina oral, patologia oral y cirugia bucal, 2009. 14(5): p. E222-8.
• White S.C. and Pharoah M.J., Oral Radiology Principles and Interpretation. 6th Edition, St. Louis; Mosby 2009.
• Xie X., J. Wang and Y. Ding, Recurrent eosinophilic granuloma involving maxilla and mandible in an adult male: an unusual case report. Australian Dental Journal, 2021. 66: p. S88-S92.
• Yashoda-Devi, B., N. Rakesh, and M. Agarwal, Langerhans cell histiocytosis with oral manifestations: A rare and unusual case report. Journal of Clinical and Experimental Dentistry, 2012. 4(4): p. e252.