Uncommon Oral Manifestations and Growth Failure in A Pediatric Multisystem Langerhans Cell Histiocytosis: A Case Report
DOI:
https://doi.org/10.52783/jns.v14.1623Keywords:
Gingival hypertrophy, failure to thrive, Langerhans cell histiocytosisAbstract
Langerhans cell histiocytosis (LCH) is a rare condition characterized by the clonal proliferation of Langerhans cells, leading to varying degrees of organ dysfunction. This report presents a challenging case of multisystem LCH in a 1-year-8-month-old female, exhibiting symptoms like gingival hypertrophy, cutaneous lesions, and failure to thrive. Diagnosis was confirmed by histopathology and immunohistochemistry, which revealed CD1a-positive Langerhans cells. The patient underwent chemotherapy, achieving significant clinical improvement. This report emphasizes the need for vigilance when evaluating pediatric patients with nonspecific symptoms and the importance of a multidisciplinary approach in diagnosing and managing this rare disorder.
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Altay MA, Sindel A, Özalp Ö, et al. Langerhans cell histiocytosis: a diagnostic challenge in the oral cavity. Case Rep Pathol.2017;2017:1-¬ 6.
Emile JF, Abla O, Fraitag S, Horne A, Haroche J, Donadieu J, Requena-Caballero L, Jordan MB, Abdel-Wahab O, Allen CE, Charlotte F, Diamond EL, Egeler RM, Fischer A, Herrera JG, Henter JI, Janku F, Merad M, Picarsic J, Rodriguez-Galindo C, Rollins BJ, Tazi A, Vassallo R, Weiss LM; Histiocyte Society. Revised classification of histiocytoses and neoplasms of the macrophage-dendritic cell lineages. Blood. 2016 Jun 2;127(22):2672-81. doi: 10.1182/blood-2016-01-690636. Epub 2016 Mar 10. PMID: 26966089; PMCID: PMC5161007.
Brenner M. Current status of gene transfer into hamatopoietic progenitor cells: application to langerhans cell histiocytosis. Br J Cancer suppl.1994;23:S56-7
Yashoda Devi B, Rakesh N, Agarwal M. Langerhans cell histiocytosis with oral manifestations: A rare and unusual case report. J Clin Exp Dent 2012;4:e252 5.
Rao DG, Trivedi MV, Havale R, Shrutha SP. A rare and unusual case report of Langerhans cell histiocytosis. J Oral MaxillofacPathol. 2017;21(1):140-¬ 144. doi:10.4103/jomfp.JOMFP
Tesluk EW, Szutkowski Z, Kawecki A. Langerhans cell histiocytosis of bone – A case report and review of literature. J Onco 2003;53:161 4.
Greenberger JS, Crocker AC, Vawter G, Jaffe N, Cassady JR. Results of treatment of 127 patients with systemic histiocytosis. Medicine (Baltimore) 1981;60:311 38
Kumar YP, Agrawal J, Mohanlakshmi J, Kumar PS. Langerhans cell histiocytosis revisited: Case report wit review. Contemp Clin Dent 2015;6:432-6.
Badalian-Very G, Vergilio JA, Degar BA, MacConaill LE, Brandner B, Calicchio ML, Kuo FC, Ligon AH, Stevenson KE, Kehoe SM, Garraway LA, Hahn WC, Meyerson M, Fleming MD, Rollins BJ. Recurrent BRAF mutations in Langerhans cell histiocytosis. Blood 2010; 116: 1919-1923.
Berres ML, Merad M, Allen CE. Progress in understanding the pathogenesis of Langerhans cell histiocytosis: back to Histiocytosis X? Br J Haematol 2015; 169: 3-13.
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