Hypopigmented Lesions: An Unusual Initial Presentation of Infantile Langerhans Cell Histiocytosis

Authors

  • Suthassa Sithitool DIVISION OF DERMATOLOGY DEPARTMENT OF MEDICINE, CHULALONGKORN UNIVERSITY, BANGKOK, THAILAND.
  • Karaked Chantawarangul DIVISION OF PEDIATRIC DERMATOLOGY DEPARTMENT OF PEDIATRICS, CHULALONGKORN UNIVERSITY, BANGKOK, THAILAND.
  • Therdpong Tempark DIVISION OF PEDIATRIC DERMATOLOGY DEPARTMENT OF PEDIATRICS, CHULALONGKORN UNIVERSITY, BANGKOK, THAILAND.
  • Susheera Chatproedprai DIVISION OF PEDIATRIC DERMATOLOGY DEPARTMENT OF PEDIATRICS, CHULALONGKORN UNIVERSITY, BANGKOK, THAILAND.
  • Siriwan Wananukul DIVISION OF PEDIATRIC DERMATOLOGY DEPARTMENT OF PEDIATRICS, CHULALONGKORN UNIVERSITY, BANGKOK, THAILAND.
  • Jade Wititsuwannakul DIVISION OF DERMATOLOGY DEPARTMENT OF MEDICINE, CHULALONGKORN UNIVERSITY, BANGKOK, THAILAND.

Keywords:

Langerhans cell histiocytosis, LCH, hypopigmentation

Abstract

Langerhans cell histiocytosis is a rare neoplasm. Patients are typically presented with small, translucent, yellowish crusted papules on the trunk, intertriginous areas, and scalp. However, previous literature has reported hypopigmented lesions as an unusual cutaneous presentation, emphasizing the need for clinician awareness to aid a prompt diagnosis. Herein, we present a case of langerhans cell histiocytosis, whose cutaneous presentations were multiple hypopigmented lesions and yellowish crusts–an uncommon manifestation of langerhans cell histiocytosis.

References

Allen CE, Merad M, McClain KL. Langerhans-Cell Histiocytosis. N Engl J Med 2018;379:856-68.

Ribeiro KB, Degar B, Antoneli CB, Rollins B, Rodriguez-Galindo C. Ethnicity, race, and socioeconomic status influence incidence of Langerhans cell histiocytosis. Pediatr Blood Cancer 2015;62:982-7.

Poompuen S, Chaiyarit J, Techasatian L. Diverse cutaneous manifestation of Langerhans cell histiocytosis: a 10-year retrospective cohort study. Eur J Pediatr 2019;178:771-6.

Stein SL, Paller AS, Haut PR, Mancini AJ. Langerhans cell histiocytosis presenting in the neonatal period: a retrospective case series. Arch Pediatr Adolesc Med 2001;155:778-83.

Haupt R, Minkov M, Astigarraga I, et al. Langerhans cell histiocytosis (LCH): guidelines for diagnosis, clinical work-up, and treatment for patients till the age of 18 years. Pediatr Blood Cancer 2013;60:175-84.

Mehta B, Amladi S. Langerhans cell histiocytosis presenting as hypopigmented papules. Pediatr Dermatol 2010;27:215-7.

Krooks J, Minkov M, Weatherall AG. Langerhans cell histiocytosis in children: History, classification, pathobiology, clinical manifestations, and prognosis. J Am Acad Dermatol 2018;78:1035-44.

Feroze K, Unni M, Jayasree MG, Seethalekshmy NV. Langerhans cell histiocytosis presenting with hypopigmented macules. Indian J Dermatol Venereol Leprol 2008;74:670-2.

Kaddu S, Mulyowa G, Kovarik C. Hypopigmented scaly, scalp and facial lesions and disfiguring exopthalmus. Langerhans cell histiocytosis (LCH). Clin Exp Dermatol 2010;35:e52-3.

Mori S, Adar T, Kazlouskaya V, Alexander JB, Heilman E, Glick SA. Cutaneous Langerhans cell histiocytosis presenting with hypopigmented lesions: Report of two cases and review of literature. Pediatr Dermatol 2018;35:502-6.

Lozano Masdemont B, Gómez-Recuero Muñoz L, Villanueva Álvarez-Santullano A, Parra Blanco V, Campos Domínguez M. Langerhans cell histiocytosis mimicking lichen nitidus with bone involvement. Australas J Dermatol 2017;58:231-3.

Longaker MA, Frieden IJ, LeBoit PE, Sherertz EF. Congenital "self-healing" Langerhans cell histiocytosis: the need for long-term follow-up. J Am Acad Dermatol 1994;31:910-6.

Parimi LR, You J, Hong L, Zhang F. Congenital self-healing reticulohistiocytosis with spontaneous regression. An Bras Dermatol 2017;92:553-5.

Uaratanawong R, Kootiratrakarn T, Sudtikoonaseth P, Issara A, Kattipathanapong P. Congenital self-healing reticulohistiocytosis presented with multiple hypopigmented flat-topped papules: a case report and review of literatures. J Med Assoc Thai 2014;97:993-7.

Battistella M, Fraitag S, Teillac DH, Brousse N, de Prost Y, Bodemer C. Neonatal and early infantile cutaneous langerhans cell histiocytosis: comparison of self-regressive and non-self-regressive forms. Arch Dermatol 2010;146:149-56.

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Published

2022-03-21

How to Cite

Sithitool, S., Chantawarangul, K., Tempark, T., Chatproedprai, S., Wananukul, S., & Wititsuwannakul, J. (2022). Hypopigmented Lesions: An Unusual Initial Presentation of Infantile Langerhans Cell Histiocytosis. Thai Journal of Dermatology, 38(1), 29–35. Retrieved from https://he02.tci-thaijo.org/index.php/TJD/article/view/255006

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Section

Case Report