Adult pulmonary Langerhans cell histiocytosis revealed by central diabetes insipidus: A case report and literature review

Lourenço,Jorge; Ferreira,Cristina; Marado,Daniela

doi:10.3892/mco.2020.2100

Adult pulmonary Langerhans cell histiocytosis revealed by central diabetes insipidus: A case report and literature review

Authors:
- Jorge Lourenço
- Cristina Ferreira
- Daniela Marado
View Affiliations

Affiliations: Department of Internal Medicine, Coimbra University Central Hospital, 3000-075 Coimbra, Portugal, Department of Pneumology, Coimbra University Central Hospital, 3000-075 Coimbra, Portugal
Published online on: July 22, 2020 https://doi.org/10.3892/mco.2020.2100
Article Number: 30
Copyright: © Lourenço et al. This is an open access article distributed under the terms of Creative Commons Attribution License.

Metrics: Total Views: 0 (Spandidos Publications: | PMC Statistics: )

Metrics: Total PDF Downloads: 0 (Spandidos Publications: | PMC Statistics: )

Cited By (CrossRef): 0 citations Loading Articles...

Abstract

Langerhans cell histiocytosis (LCH) is a rare systemic and heterogeneous disease secondary to proliferation and diffuse infiltration of immature CD1a‑positive dendritic cells, also known as Langerhans cells. LCH affects predominantly paediatric patients and is rarely diagnosed in adulthood. Despite its worldwide prevalence, most reported cases are found in the Japanese population. There is no consensus regarding treatment strategy due to the low incidence of this disease and the diversity of symptoms that appear. An integrative literature review was conducted based on the PubMed database using MeSH terms ‘Langerhans’, ‘histiocytosis’ and ‘adult’. The present report describes a case of a successfully treated LCH‑induced central diabetes insipidus (uncommon presentation in adult patients) as well as an updated review of current evidence published on this matter.

View Figures

View References

1	Stockschlaeder M and Ssucker C: Adult langerhans cell histiocytosis. Eur J Haematol. 76:363–368. 2006.PubMed/NCBI View Article : Google Scholar
2	WHO Classification of Tumours of Haematopoietic and Lymphoid Tissue. IARC, Lyon, pp470-472, 2017.
3	Lian C, Lu Y and Shen S: Langerhans cell histiocytosis in adults: A case report and review of literature. Oncotarget. 7:18678–18683. 2016.PubMed/NCBI View Article : Google Scholar
4	Histiocytosis syndromes in children. Writing group of the histiocyte society. Lancet. 1:208–209. 1987.PubMed/NCBI
5	Favara BE, Feller AC, Pauli M, Jaffe ES, Weiss LM, Arico M, Bucsky P, Egeler RM, Elinder G, Gadner H, et al: Contemporary classification of histiocytic disorders. The WHO committee on histiocytic/reticulum cell proliferations. Reclassification working group of the histiocyte society. Med Pediatr Oncol. 29:157–166. 1997.PubMed/NCBI View Article : Google Scholar
6	Titgemeyer C, Grois N, Minkov M, Flucher-Wolfram B, Gatterer-Menz I and Gadner H: Pattern and course of single-system disease in langerhans cell histiocytosis data from the DAL-HX 83- and 90-study. Med Pediatr Oncol. 37:108–114. 2001.PubMed/NCBI View Article : Google Scholar
7	Hutter C and Minkov M: Insights into the pathogenesis of Langerhans cell histiocytosis: The development of targeted therapies. Immunotargets Ther. 5:81–91. 2016.PubMed/NCBI View Article : Google Scholar
8	Tatsuno M, Shioda Y, Iwafuchi H, Yamazaki S, Iijima K, Takahashi C, Ono H, Uchida K, Okamura O, Matubayashi M, et al: BRAF V600 mutations in langerhans cell histiocytosis with a simple and unique assay. Diagn Pathol. 11(39)2016.PubMed/NCBI View Article : Google Scholar
9	Harmon CM and Brown N: Langerhans cell histiocytosis: A clinicopathologic review and molecular pathogenetic update. Arch Pathol Lab Med. 139:1211–1214. 2015.PubMed/NCBI View Article : Google Scholar
10	Kaltsas GA, Powles TB, Evanson J, Plowman PN, Drinkwater JE, Jenkins PJ, Monson JP, Besser GM and Grossman AB: Hypothalamo-pituitary abnormalities in adult patients with langerhans cell histiocytosis: Clinical, endocrinological and radiological features and response to treatment. J Clin Endocrinol Metab. 85:1370–1376. 2000.PubMed/NCBI View Article : Google Scholar
11	Choi YS, Lim JS, Kwon W, Jung SH, Park IH, Lee MK, Lee WY, Yong SJ, Lee SJ, Jung YR, et al: Pulmonary langerhans cell histiocytosis in an adult male presenting with central diabetes insipidus and diabetes mellitus: A case report. Tuberc Respir Dis (Seoul). 78:463–468. 2015.PubMed/NCBI View Article : Google Scholar
12	Choi JE, Lee HR, Ohn JH, Moon MK, Park J, Lee SJ, Choi MG, Yoo HJ, Kim JH and Hong EG: Adult multisystem langerhans cell histiocytosis presenting with central diabetes insipidus successfully treated with chemotherapy. Endocrinol Metab (Seoul). 29:394–399. 2014.PubMed/NCBI View Article : Google Scholar
13	Hong ES, Ohn JH, Kim JH, Hwang-Bo Y, Kim JJ, Kwon JH, Lee JW, Choi SY, Lee EK, Cho SW, et al: Clinical characteristics of langerhans cell histiocytosis with hypothalamo-pituitary involvement. Endocrinol Metab. 26:38–43. 2011.PubMed/NCBI View Article : Google Scholar
14	Wei P, Lu HW, Jiang S, Fan LC, Li HP and Xu JF: Pulmonary langerhans cell histiocytosis: Case series and literature review. Medicine (Baltimore). 93:1–7. 2014.PubMed/NCBI View Article : Google Scholar
15	Tazi A, de Margerie C, Naccache JM, Fry S, Dominique S, Jouneau S, Lorillon G, Bugnet E, Chiron R, Wallaert B, et al: The natural history of adult pulmonary langerhans cell histiocytosis: A prospective multicentre study. Orphanet J Rare Dis. 10:1–10. 2015.PubMed/NCBI View Article : Google Scholar
16	Ninaber M, Dik H and Peters E: Complete pathological resolution of pulmonary Langerhans cell histiocytosis. Respirol Case Rep. 2:76–78. 2014.PubMed/NCBI View Article : Google Scholar
17	Simko SJ, Garmezy B, Abhyankar H, Lupo PJ, Chakraborty R, Lim KPH, Shih A, Hicks MJ, Wright TS, Levy ML, et al: Differentiating skin-limited and multisystem langerhans cell histiocytosis. J Pediatr. 165:990–996. 2014.PubMed/NCBI View Article : Google Scholar
18	Stull MA, Kransdorf MJ and Devaney KO: Langerhans cell histiocytosis of bone. Radiographics. 12:801–823. 1992.PubMed/NCBI View Article : Google Scholar
19	Kim SH and Choi MY: Langerhans cell histiocytosis of the rib in an adult: A case report. Case Rep Oncol. 9:83–88. 2016.PubMed/NCBI View Article : Google Scholar
20	Song YS, Lee IS, Yi JH, Cho KH, Kim DK and Song JW: Radiologic findings of adult pelvis and appendicular skeletal langerhans cell histiocytosis in nine patients. Skeletal Radiol. 40:1421–1426. 2010.PubMed/NCBI View Article : Google Scholar
21	Christopher Z, Binitie O, Henderson-Jackson E, Perno J and Makanji RJ: Langherhans cell histiocytosis of bone in an adult: A case report. Radiol Case Rep. 13:310–314. 2018.PubMed/NCBI View Article : Google Scholar
22	Aricò M, Girschikofsky M, Généreau T, Klersy C, McClain K, Grois N, Emile JF, Lukina E, Juli ED and Danesino C: Langerhans cell histiocytosis in adults: Report from the international registry of the histiocyte society. Eur J Cancer. 39:2341–2348. 2003.PubMed/NCBI View Article : Google Scholar
23	Do J, Lee J and Kim Y: Successful treatment of cutaneous langerhans' cell histiocytosis with targeted narrowband ultraviolet B phototherapy in an infant. Clin Exp Dermatol. 34(e280-e281)2009.PubMed/NCBI View Article : Google Scholar
24	Vogel C, Aughenbaugh W and Sharata H: Excimer laser as adjuvant therapy for adult cutaneous langerhans cell histiocytosis. Arch Dermatol. 144:1287–1290. 2008.PubMed/NCBI View Article : Google Scholar
25	Satter E and High W: Langerhans cell histiocytosis: A review of current recommendations of the histiocyte society. Pediatr Dermatol. 25:291–295. 2008.PubMed/NCBI View Article : Google Scholar
26	Gadner H: Treatment of adult-onset langerhans cell histiocytosis-is it different from the pediatric approach? Ann Oncol. 21:1141–1142. 2010.PubMed/NCBI View Article : Google Scholar
27	Gadner H, Minkov M, Grois N, Pötschger U, Thiem E, Aricò M, Astigarraga I, Braier J, Donadieu J, Henter JI, et al: Therapy prolongation improves outcome in multisystem langerhans cell histiocytosis. Blood. 121:5006–5004. 2013.PubMed/NCBI View Article : Google Scholar
28	Simko S, McClain K and Allen C: Up-front therapy for LCH: Is it time to test an alternative to vinblastine/prednisone? Br J Haematol. 169:299–301. 2015.PubMed/NCBI View Article : Google Scholar
29	Cantu MA, Lupo PJ, Bilgi M, Hicks MJ, Allen CE and McClain KL: Optimal therapy for adults with langerhans cell histiocytosis bone lesions. PLoS One. 7(e43257)2012.PubMed/NCBI View Article : Google Scholar
30	NassEr M, Traclet J and Cottin V: Effect of cladribine therapy on lung cysts in pulmonary langerhans cell histiocytosis. ERJ Open Res. 4:00089–2017. 2018.PubMed/NCBI View Article : Google Scholar
31	Haroche J, Cohen-Aubart F, Emile JF, Arnaud L, Maksud P, Charlotte F, Cluzel P, Drier A, Hervier B, Benameur N, et al: Dramatic efficacy of vemurafenib in both multisystemc and refractory erdheim-chester disease and langerhans cell histiocytosis harbouring the BRAF V600E mutation. Blood. 121:1495–1500. 2013.PubMed/NCBI View Article : Google Scholar
32	Abla O and Weitzman S: Treatment of langerhans cell histiocytosis: Role of BRAF/MAPK inhibition. Hematology Am Soc Hematol Educ Program. 2015:565–570. 2015.PubMed/NCBI View Article : Google Scholar
33	Steiner M, Matthes-Martin S, Attarbaschi A, Minkov M, Grois N, Unger E, Holter W, Vormoor J, Wawer A, Ouachee M, et al: Improved outcome of treatment-resistant high-risk langerhans cell histioytosis after allogenic stem cell transplantation with reduced-intensity conditioning. Bone Marrow Transplant. 36:215–225. 2005.PubMed/NCBI View Article : Google Scholar
34	Kesik V, Citak C, Kismet E, Koseoglu V and Akyuz C: Hematopoietic stem cell transplantation in langerhans cell histiocytosis: Case report and review of the literature. Pediatr Transplant. 13:371–374. 2009.PubMed/NCBI View Article : Google Scholar