Palatal manifestations of Langerhan cell histicytosis in pediatric age: a case report with review of literature

Authors

  • Ishwar Singh Department of ENT and Head Neck and Surgery, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi, India
  • Sakshi Negi Department of ENT and Head Neck and Surgery, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi, India
  • Shruti Dhingra Department of ENT and Head Neck and Surgery, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi, India
  • Raghav Singh Department of Pathology, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi, India
  • Varuna Mallya

DOI:

https://doi.org/10.18203/issn.2454-5929.ijohns20231469

Keywords:

Langerhan cell histiocytosis, Pediatric LCH, Palatal ulcer

Abstract

Langerhans cell histiocytosis (LCH) is characterised by multisystem disorder with various entities grouped together under same name. Oral cavity manifestations of this disease is yet to be explored. In oral cavity it can present with ulceration, swelling or osteolysis of underlying bone. Oral cavity involvement due to LCH might confuse the examining doctor resulting in misdiagnosis of such dreadful disease. So here we are reporting a case of LCH in a 4 years old female presented to us with ulceration over hard palate mucosa. Biopsy was taken from the ulcer site which was positive for LCH. On further examination and evaluation of the patient multisystem involvement was found with involvement of skull bone.

References

Langerhans P. Oeber die Nerven der menshlichen Haut. Arch Pathol Anat. 1868;44:325-37.

Howarth DM, Gilchrist GS, Mullan BP, Wiseman GA, Edmonson JH, Schomberg PJ. Langerhans cell histiocytosis. Cancer. 1999;15:2278-90.

Galindo CR, Allen CE. Langerhans cell histiocytosis. Blood. 2020;135:1319-31.

Zhang Q, Wu X, Wang X, Pan E, Ying L. Molecular and oral manifestations of Langerhans cell histiocytosis preceding acute myeloid leukaemia. BMC Oral Health. 2022;22:1-6.

Gadner H. Minkov M, Grois N, Pötschger U, Theim E, Aricò M, et al. Histiocyte Society. Therapy prolongation improves outcome in multisystem Langerhans cell histiocytosis. Blood. 2013;121:5006-14.

Milián MA, Bagán JV, Jiménez Y, Pérez A, Scully C, Antoniades d et al. Langerhans’ cell histiocytosis restricted to the oral mucosa. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2001;91:76-9.

Merglova V, Hrusak D, Boudova L, Mukensnabl P, Valentova E, Hosticka L. Langerhans cell histiocytosis in childhood-Review, symptoms in the oral cavity, differential diagnosis and report of two cases. J Cranio Maxillofacial Surg. 2014;42:93-100.

Henter JJ, Tondini C, Pritchard J. Histiocyte disorders. Crit Rev Oncol Hematol. 2004;50:157-74.

Kilborn TN, The J, Goodman TR. Paediatric manifestations of Langerhans cell histiocytosis: a review of the clinical and radiological findings. Clin Radiol. 2003;269-78:58.

Roper M, Kelly D, PArmley RT, Kheir SM, Hicks JN. Eosinophilic granuloma of the palatal mucosa in a nine week old infant. Med Pediatr Oncol. 1981;9:153-6.

Loh HS, Quah TC. X Histiocytosis (Langerhans-cell histiocytosis) of the palate. Case Report Aus Dent J. 1990;35:117-20.

Davis SE, Rice DH. Langerhans cell histiocytosis: Current trends and the role of the head and neck surgeon. Ear Nose Throat J. 2004:83;340-4.

Alajbeg I, Boras VV, Femenic R, Cekic-Arambasin A, Anicic M, Kelecic J, et al. Unrecognized oral manifestations of Langerhans cell histiocytosis which progressed to systemic disease. Oral Oncol EXTRA. 2006:42;10-3.

Sasaoka K, Mogi K, Kanou A, Kanno K, Negishi A. Clinical study of five cases of Langerhan cell histiocytosis occurred in jaws. Kitakanto Med J. 2008;58:317-24.

Murray M, Dean J, Slater L. Multifocal oral Langerhans cell histiocytosis. J Oral Maxillofac Surg. 2011;69:2585-91.

Halperson E, Weintraub M. Oral Langerhans cell histiocytosis in an infant. J Dent Child. 2018;85:75-8.

Neves-Silva R, Fernandes DT, Fonseca FP, Pontes HAR, Brasilerio BF, Santos-Silva AR, et al. Oral menifestations of LAngerhan cell histiocytosis: a case series. Spec Care Dent. 2018;38:426-33.

Hammouri EH, Sweidan HA, Ashokaibi O, Omari LA. Langerhans cell histiocytosis: A case report with oral manifestations and the role of pediatric dentist in the diagnosis. Clin Case Rep. 2020;8:545-9.

Togawa T, Sugauchi A, Oya K, Yokota Y, Isomura ET. Langerhans cell histiocytosis of the hard palate in an infant. J Oral Maxillofacial Surg Med Pathol. 2023;35:56-9.

Downloads

Published

2023-05-25

Issue

Section

Case Reports