Tuberculosis and Respiratory Diseases

The Korean Academy of Tuberculosis and Respiratory Diseases (대한결핵및호흡기학회)
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Pulmonary Langerhans Cell Histiocytosis in an Adult Male Presenting with Central Diabetes Insipidus and Diabetes Mellitus: A Case Report

  • Choi, Yeun Seoung (Department of Internal Medicine, Yonsei University Wonju College of Medicine) ;
  • Lim, Jung Soo (Department of Internal Medicine, Yonsei University Wonju College of Medicine) ;
  • Kwon, Woocheol (Department of Radiology, Yonsei University Wonju College of Medicine) ;
  • Jung, Soon-Hee (Department of Pathology, Yonsei University Wonju College of Medicine) ;
  • Park, Il Hwan (Department of Thoracic and Cardiovascular Surgery, Yonsei University Wonju College of Medicine) ;
  • Lee, Myoung Kyu (Department of Internal Medicine, Yonsei University Wonju College of Medicine) ;
  • Lee, Won Yeon (Department of Internal Medicine, Yonsei University Wonju College of Medicine) ;
  • Yong, Suk Joong (Department of Internal Medicine, Yonsei University Wonju College of Medicine) ;
  • Lee, Seok Jeong (Department of Internal Medicine, Yonsei University Wonju College of Medicine) ;
  • Jung, Ye-Ryung (Department of Internal Medicine, Yonsei University Wonju College of Medicine) ;
  • Choi, Jiwon (Department of Internal Medicine, Yonsei University Wonju College of Medicine) ;
  • Choi, Ji Sun (Department of Internal Medicine, Yonsei University Wonju College of Medicine) ;
  • Jeong, Joon Taek (Department of Internal Medicine, Yonsei University Wonju College of Medicine) ;
  • Yoo, Jin Sae (Department of Internal Medicine, Yonsei University Wonju College of Medicine) ;
  • Kim, Sang-Ha (Department of Internal Medicine, Yonsei University Wonju College of Medicine)
  • Received : 2015.08.11
  • Accepted : 2015.09.01
  • Published : 2015.10.30
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Abstract

Pulmonary Langerhans cell histiocytosis is an uncommon diffuse cystic lung disease in adults. In rare cases, it can involve extrapulmonary organs and lead to endocrine abnormalities such as central diabetes insipidus. A 42-year-old man presented with polyphagia and polydipsia, as well as a dry cough and dyspnea on exertion. Magnetic resonance imaging of the hypothalamic-pituitary system failed to show the posterior pituitary, which is a typical finding in patients with central diabetes insipidus. This condition was confirmed by a water deprivation test, and the patient was also found to have type 2 diabetes mellitus. Computed tomographic scanning of the lungs revealed multiple, irregularly shaped cystic lesions and small nodules bilaterally, with sparing of the costophrenic angles. Lung biopsy through video-assisted thoracoscopic surgery revealed pulmonary Langerhans cell histiocytosis. On a follow-up visit, only 1 year after the patient had quit smoking, clinical and radiological improvement was significant. Here, we report an uncommon case of pulmonary Langerhans cell histiocytosis that simultaneously presented with diabetes insipidus and diabetes mellitus.

Keywords

References

  1. Vassallo R, Ryu JH, Colby TV, Hartman T, Limper AH. Pulmonary Langerhans'-cell histiocytosis. N Engl J Med 2000;342: 1969-78. https://doi.org/10.1056/NEJM200006293422607
  2. Vassallo R, Ryu JH, Schroeder DR, Decker PA, Limper AH. Clinical outcomes of pulmonary Langerhans'-cell histiocytosis in adults. N Engl J Med 2002;346:484-90. https://doi.org/10.1056/NEJMoa012087
  3. Kim C, Jeong SH, Shim JJ, Cha SI, Son C, Chung MP, et al. Clinical features of pulmonary Langerhans cell histiocytosis in Korea. Tuberc Respir Dis 2009;66:98-103. https://doi.org/10.4046/trd.2009.66.2.98
  4. Howarth DM, Gilchrist GS, Mullan BP, Wiseman GA, Edmonson JH, Schomberg PJ. Langerhans cell histiocytosis: diagnosis, natural history, management, and outcome. Cancer 1999; 85:2278-90. https://doi.org/10.1002/(SICI)1097-0142(19990515)85:10<2278::AID-CNCR25>3.0.CO;2-U
  5. Girschikofsky M, Arico M, Castillo D, Chu A, Doberauer C, Fichter J, et al. Management of adult patients with Langerhans cell histiocytosis: recommendations from an expert panel on behalf of Euro-Histio-Net. Orphanet J Rare Dis 2013;8:72. https://doi.org/10.1186/1750-1172-8-72
  6. Mogulkoc N, Veral A, Bishop PW, Bayindir U, Pickering CA, Egan JJ. Pulmonary Langerhans' cell histiocytosis: radiologic resolution following smoking cessation. Chest 1999;115:1452-5. https://doi.org/10.1378/chest.115.5.1452
  7. Alexandraki KI, Makras P, Protogerou AD, Dimitriou K, Stathopoulou A, Papadogias DS, et al. Cardiovascular risk factors in adult patients with multisystem Langerhans-cell histiocytosis: evidence of glucose metabolism abnormalities. QJM 2008;101:31-40.
  8. Thai AC, Sng I, Suri R, Cheah JS. Disseminated histiocytosis X with diabetes insipidus and diabetes mellitus in an adult female (histiocytosis with DI and DM). Ann Acad Med Singapore 1988;17:294-8.
  9. Rowntree LG, Poppiti RJ. Diabetes inspidius, diabetes mellitus, and insulin resistance with histiocytosis. J Am Med Assoc 1954;156:310-2. https://doi.org/10.1001/jama.1954.02950040016005
  10. Hong ES, Ohn JH, Kim JH, Hwang-Bo YK, Kim JJ, Kwon JH, et al. Clinical characteristics of Langerhans cell histiocytosis with hypothalamo-pituitary involvement. Endocrinol Metab 2011;26:38-43. https://doi.org/10.3803/EnM.2011.26.1.38
  11. Choi JE, Lee HR, Ohn JH, Moon MK, Park J, Lee SJ, et al. Adult multisystem langerhans cell histiocytosis presenting with central diabetes insipidus successfully treated with chemotherapy. Endocrinol Metab 2014;29:394-9. https://doi.org/10.3803/EnM.2014.29.3.394
  12. Makras P, Samara C, Antoniou M, Zetos A, Papadogias D, Nikolakopoulou Z, et al. Evolving radiological features of hypothalamo-pituitary lesions in adult patients with Langerhans cell histiocytosis (LCH). Neuroradiology 2006;48:37-44. https://doi.org/10.1007/s00234-005-0011-x
  13. Grois N, Prayer D, Prosch H, Minkov M, Potschger U, Gadner H. Course and clinical impact of magnetic resonance imaging findings in diabetes insipidus associated with Langerhans cell histiocytosis. Pediatr Blood Cancer 2004;43:59-65. https://doi.org/10.1002/pbc.20003
  14. Arico M, Girschikofsky M, Genereau T, Klersy C, McClain K, Grois N, et al. Langerhans cell histiocytosis in adults: reportfrom the International Registry of the Histiocyte Society. Eur J Cancer 2003;39:2341-8. https://doi.org/10.1016/S0959-8049(03)00672-5
  15. Moore AD, Godwin JD, Muller NL, Naidich DP, Hammar SP, Buschman DL, et al. Pulmonary histiocytosis X: comparison of radiographic and CT findings. Radiology 1989;172:249-54. https://doi.org/10.1148/radiology.172.1.2787035
  16. Hansen NJ, Hankins JH. Pulmonary langerhans cell histiocytosis: PET/CT for initial workup and treatment response evaluation. Clin Nucl Med 2015;40:153-5. https://doi.org/10.1097/RLU.0000000000000506
  17. Lee HJ, Ahn BC, Lee SW, Lee J. The usefulness of F-18 fluorodeoxyglucose positron emission tomography/computed tomography in patients with Langerhans cell histiocytosis. Ann Nucl Med 2012;26:730-7. https://doi.org/10.1007/s12149-012-0635-y

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