Langerhans Cell Histiocytosis: A Case Report with Unusual Cutaneous Manifestation

Document Type : CASE REPORT

Authors
Fariba Iraji 1
Nazila Poostiyan 1
Parvin Rajabi Dehnavi 2
Mojgan Soghrati 3
1 Department of Dermatology, Skin Diseases and Leishmaniasis Research Center, Isfahan University of Medical Sciences, Isfahan, Iran
2 Department of Pathology, Isfahan University of Medical Sciences, Isfahan, Iran
3 Alzahra Research Institute, Isfahan, Iran
Abstract
Langerhans cell histiocytosis (LCH) is characterized by idiopathic monoclonal infiltration of Langerhans cells in different organs such as the skeleton, skin, pituitary gland, liver, spleen, lungs, and the hematopoietic system. Skin lesions are common in LCH and affect about 40% of cases. It is reported that skin lesions are usually the first manifestation of LCH in 80% of patients. Usually, cutaneous presentations of LCH in adults are generalized or seborrhea-like lesions and it is often the first manifestation of disease. Here, we describe a 45-year old female who was known case of hypothyroidism, systemic lupus erythematosus, and diabetes insipidus. In our patient, cutaneous involvement was unusual. It was single and presented in unusual site (ankle) and before developing such lesion, she had diabetes insipidus for several years due to the involvement of pituitary gland.

Keywords

1.
Blazewicz I, Biernat W, Kowalczyk A, Baranska-Rybak W, Nowicki R, Stawczyk-Macieja M, et al. Adult onset of multisystem Langerhans cell histiocytosis with skin and lymph node involvement. Postepy Dermatol Alergol 2015;32:225-8.  Back to cited text no. 1
    
2.
Jeunon T, Sousa MA, Santos-Rodrigues N, Lopes R. Langerhans cell histiocytosis - A case report. Dermatol Pract Concept 2012;2:25-9.  Back to cited text no. 2
[PUBMED]    
3.
Reddy IS, Gowrishankar S, Somani VK, Murthy DB. Adult onset Langerhans cell histiocytosis: Report of two patients. Indian J Dermatol Venereol Leprol 2014;80:560-2.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Abla O, Egeler RM, Weitzman S. Langerhans cell histiocytosis: Current concepts and treatments. Cancer Treat Rev 2010;36:354-9.  Back to cited text no. 4
[PUBMED]    
5.
Lai JY, Tang MM, Priya G, Rajasuriar JS, Suganthi T. Langerhans Cell Histiocytosis in an Adult - a rare, life-threatening and not to be missed. Med J Malaysia 2014;69:95-7.  Back to cited text no. 5
[PUBMED]    
6.
Chai D, Tao Y, Bao Z, Yang L, Feng Z, Ma L, et al. The adverse prognostic hallmarks in identical twins with Langerhans cell histiocytosis: A clinical report and literature review. Tohoku J Exp Med 2013;230:219-25.  Back to cited text no. 6
[PUBMED]    
7.
Erdem H, Kadioglu N, Uzunlar AK, Yücel I, Oktay M, Besir FH. Langerhans cell histiocytosis mimicking osteomyelitis in an infant. APSP J Case Rep 2013;4:27.  Back to cited text no. 7
    
8.
Azreen AB, Kwan CY, Prepagaren N. Langerhans cell histiocytosis of maxillary sinus. Med J Malaysia 2012;67:627-8.  Back to cited text no. 8
[PUBMED]    
9.
Stockschlaeder M, Sucker C. Adult Langerhans cell histiocytosis. Eur J Haematol 2006;76:363-8.  Back to cited text no. 9
[PUBMED]    
10.
Histiocyte Society 2009. Langerhans Cell Histiocytosis Evaluation and Treatment Guidelines. Histocyte Society Online; 2009. Available from: http://www.histiocytesociety.org/site/c.mqISL2PIJrH/b. 4442715/k.A339/Treatment_Plans.htm. [Last accessed on 2009 Apr].  Back to cited text no. 10
    
11.
Gadner H, Grois N, Arico M, Broadbent V, Ceci A, Jakobson A, et al. Arandomized trial of treatment for multisystem Langerhans' cell histiocytosis. J Pediatr 2001;138:728-34.  Back to cited text no. 11
[PUBMED]    
12.
Sun WG, Zhong LS, Chen H. A case of adult generalized cutaneous langerhans cell histiocytosis. Ann Dermatol 2016;28:262-4.  Back to cited text no. 12
[PUBMED]    
13.
Liu YH, Fan XH, Fang K. Langerhans' cell histiocytosis with multisystem involvement in an adult. Clin Exp Dermatol 2007;32:765-8.  Back to cited text no. 13
[PUBMED]    
14.
Wong LS. An unusual case of neonatal langehrans cell histiocytosis presenting with diffuse hemorrhagic nodules and a cutaneous abdominal mass. Indian J Dermatol 2015;60:424.  Back to cited text no. 14
[PUBMED]  [Full text]  
Advanced Biomedical Research
Volume 2018, june
June 2018
Pages 1-4