Tekkeşin F, Okur A, Derinkuyu BE, Akyürek N, Oğuz A, Karadeniz C, Pınarlı FG. Multisystem langerhans cell histiocytosis with thymic involvement diagnosed with anterior mediastinal mass in a 2 month-old boy. Turk J Pediatr 2016; 58: 424-428.Thymus gland involvement in Langerhans cell histiocytosis (LCH) is usually part of multi-system disease and may be more common than previously recognized. However, thymic involvement causing an anterior mediastinal mass is an extremely rare presentation of multisystem LCH. Here we report a 2-month-old-boy admitted to hospital with a giant anterior mediastinal mass with multisystem LCH involving the thymus, lungs, liver and skin. The differential diagnosis of mediastinal mass in children should also include LCH, especially multisystem disease. LCH should also be kept in mind in the differential diagnosis of skin lesions in infants, even if spontaneous regression occurs.
___
1. Siddaiah R, Weinblatt M, Roberts J, Cataletto M. Anterior mediastinal bone-eroding mass with disseminated lung lesions. Chest 2011; 140; 1371-1376.
2. Pizzo PA, Poplack DG. Principles and Practice of Pediatric Oncology, 6th ed. Philedelphia: Lippincott Williams and Wilkins 2011: 703-716.
3. Junewick JJ, Fitzgerald NE. The thymus in Langerhans cell histiocytosis. Pediatr Radiol 1999; 29: 904-907.
4. Hatakeyama N, Hori T, Yamamoto M, et al. An infant with self-healing cutaneous Langerhans cell histiocytosis followed by isolated thymic relapse. Pediatr Blood Cancer 2009: 53; 229-231.
5. Ducassou S, Seyrig F, Thomas C, et al. Thymus and mediastinal node involvement in childhood langerhans cell histicytosis: Long-term follow-up from the French National Cohort. Pediatr Blood Cancer 2013; 60: 1759- 1765.
6. Elliott M, Kokai GK, Abernethy LJ, Pizer BL. Spontaneous resoluton of isolated thymic langerhans cell histiocytosis. Med Pediatr Oncol 2002; 38: 274- 276.
7. Weitzman S, Egeler RM. Langerhans cell histiocytosis: update for the pediatrician. Current Opin in Pediatr 2008; 20: 23-29.
8. Lucioni M, Beluffi G, Bandiera L, et al. Congenital aggressive variant of Langerhans Cells histiocytosis with CD56+/E-Cadherin- phenotype. Pediatr Blood Cancer 2009; 53: 1107-1110.
9. Abramson SJ, Berdon WE, Reilly BJ, Kuhn JP. Cavitation of anterior mediastinal masses in children with histiocytosis-X. Report of four cases with radiographic, pathologic finding and clinical follow-up. Pediatr Radiol 1987; 17: 10-14.
10. Crawley AJ, Guillerman RP. Langerhans cell histiocytosis with intrathymic calcifications and cavitation. Pediatr Radiol 2010; 40(Suppl 1): S62.
11. Heller GD, Haller JO, Berdon WE, Sane S, Kleinman PK. Punctate thymic calcification in infants with untreated langerhans' cell histiocytosis: report of four new cases. Pediatr Radiol 1999; 29: 813-815.
12. Orle J, Mosca AM, Sousa MAJ, Lima CM, Adriano AR, Rezende PM. Congenital self healing reticulohistiocytosis in a newborn (Hashimato Pritzker). An Bras Dermatol 2011; 86: 785-788.
13. Kapur P, Erickson C, Rakheja D, Carder KR, Hoang MP. Congenital self-healing reticulohistiocytosis (Hashimato-Pritzker disease): ten year experience at Dallas Children's Medical Center. J Am Acad Dermatol 2007; 56: 290-294
14. Walia M, Paul P, Mishra S, Mehta R. Congenital Langerhans cell histicytosis: The self-healing variety. J Pediatr Hematol Oncol 2004; 26: 398-402.
15. Jensen MLS, Bygum A, Clemmensen O, Fenger-Gron J. Congenital self-healing reticulohistiocytosis-an important diagnostic challenge. Acta Paediatr 2011; 100; 784-786.
16. Aggarwal V, Seth A, Jain M, Krishnamurthy S, Chandra V, Aneja S. Congenital Langerhans Cell histiocytosis with skin and lung involvement: Spontaneous regression. Indian J Pediatr 2010; 77: 811-812.
17. Minkov M, Prosch H, Steiner M, et al. Langerhans cell histiocytosis in neonates. Pediatr Blood Cancer 2005; 45: 802-807.
18. Picarsic J, Egeler RM, Chikwava K, Patterson K and Jaffe R. Histologic patterns of thymic involvement in Langerhans cell proliferations: A clinicopathologic study and review of the literature. Pediatr Dev Pathol 2015; 18: 127-138.
19. Ronceray L, Pötschger U, Janka G, Gadner H, Minkov M; German Society for Pediatric Hematology and Oncology, Langerhans Cell Histiocytosis Study Group. Pulmonary involvement in pediatric-onset multisystem Langerhans cell histiocytosis: effect on course and outcome. J Pediatr 2012; 161: 129-133. e1-e3.