Klinik Araştırma
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Yıl 2023, Cilt: 5 Sayı: 1, 59 - 64, 15.01.2023

Serhat Çelik Zeynep Tuğba Güven Onur Aşık Muzaffer Keklik Leylagül Kaynar Bülent Eser Mustafa Çetin Ali Ünal

https://doi.org/10.37990/medr.1159055

Öz

Kaynakça

  • Referans1. AKDEMİR O, ÇOLAK A. Langerhans hücresi histiositozisi. Turkiye Klinikleri Journal of Surgical Medical Sciences. 2007;3(51):1-4.
  • Referans2. Alston RD, Tatevossian R, McNally RJ, Kelsey A, Birch JM, Eden TO. Incidence and survival of childhood Langerhans cell histiocytosis in Northwest England from 1954 to 1998. Pediatric blood & cancer. 2007;48(5):555-60.
  • Referans3. McClain K. Treatment of langerhans cell histiocytosis. UpToDate. Waltham, MA 2013.
  • Referans4. Weitzman S, Braier J, Donadieu J, Egeler RM, Grois N, Ladisch S, et al. 2′‐chlorodeoxyadenosine (2‐CdA) as salvage therapy for Langerhans cell histiocytosis (LCH). results of the LCH‐S‐98 protocol of the histiocyte society. Pediatric blood & cancer. 2009;53(7):1271-6.
  • Referans5. Gadner H, Minkov M, Grois N, Pötschger U, Thiem E, Arico M, et al. Therapy prolongation improves outcome in multisystem Langerhans cell histiocytosis. Blood, The Journal of the American Society of Hematology. 2013;121(25):5006-14.
  • Referans6. Allen CE, Flores R, Rauch R, Dauser R, Murray JC, Puccetti D, et al. Neurodegenerative central nervous system Langerhans cell histiocytosis and coincident hydrocephalus treated with vincristine/cytosine arabinoside. Pediatric blood & cancer. 2010;54(3):416-23.
  • Referans7. Simko SJ, Tran HD, Jones J, Bilgi M, Beaupin LK, Coulter D, et al. Clofarabine salvage therapy in refractory multifocal histiocytic disorders, including Langerhans cell histiocytosis, juvenile xanthogranuloma and Rosai–Dorfman disease. Pediatric blood & cancer. 2014;61(3):479-87.
  • Referans8. McClain KL, Picarsic J, Chakraborty R, Zinn D, Lin H, Abhyankar H, et al. CNS Langerhans cell histiocytosis: Common hematopoietic origin for LCH‐associated neurodegeneration and mass lesions. Cancer. 2018;124(12):2607-20.
  • Referans9. Sahin F, Ozen K, Vural F, Kamer S, Argin M, Anacak Y. Retrospective analysis of seven patients with adult-onset Langerhans cell histiocytosis syndromes: A single center experience. Turk J Hematol. 2007;24:75-9.
  • Referans10. Grois N, Pötschger U, Prosch H, Minkov M, Arico M, Braier J, et al. Risk factors for diabetes insipidus in langerhans cell histiocytosis. Pediatric blood & cancer. 2006;46(2):228-33.
  • Referans11. Prosch H, Grois N, Prayer D, Waldhauser F, Steiner M, Minkov M, et al. Central diabetes insipidus as presenting symptom of Langerhans cell histiocytosis. Pediatric blood & cancer. 2004;43(5):594-9.
  • Referans12. Di Iorgi N, Allegri AEM, Napoli F, Calcagno A, Calandra E, Fratangeli N, et al. Central diabetes insipidus in children and young adults: etiological diagnosis and long-term outcome of idiopathic cases. The Journal of Clinical Endocrinology & Metabolism. 2014;99(4):1264-72.
  • Referans13. Lindahl L, Fenger‐Grøn M, Iversen L. Topical nitrogen mustard therapy in patients with Langerhans cell histiocytosis. British Journal of Dermatology. 2012;166(3):642-5.
  • Referans14. McClain KL, Kozinetz CA. A phase II trial using thalidomide for Langerhans cell histiocytosis. Pediatric blood & cancer. 2007;48(1):44-9.
  • Referans15. Cantu MA, Lupo PJ, Bilgi M, Hicks MJ, Allen CE, McClain KL. Optimal therapy for adults with Langerhans cell histiocytosis bone lesions. PloS one. 2012;7(8).
  • Referans16. Duan M-H, Han X, Li J, Zhang W, Zhu T-N, Han B, et al. Comparison of vindesine and prednisone and cyclophosphamide, etoposide, vindesine, and prednisone as first-line treatment for adult Langerhans cell histiocytosis: A single-center retrospective study. Leukemia research. 2016;42:43-6.
  • Referans17. Minkov M, Steiner M, Pötschger U, Aricò M, Braier J, Donadieu J, et al. Reactivations in multisystem Langerhans cell histiocytosis: data of the international LCH registry. The Journal of pediatrics. 2008;153(5):700-5. e2.
  • Referans18. Abraham A, Alsultan A, Jeng M, Rodriguez‐Galindo C, Campbell PK. Clofarabine salvage therapy for refractory high‐risk langerhans cell histiocytosis. Pediatric blood & cancer. 2013;60(6):E19-E22.
  • Referans19. Willis B, Ablin A, Weinberg V, Zoger S, Wara WM, Matthay KK. Disease course and late sequelae of Langerhans' cell histiocytosis: 25-year experience at the University of California, San Francisco. Journal of clinical oncology. 1996;14(7):2073-82.
  • Referans20. Sivendran S, Harvey H, Lipton A, Drabick J. Treatment of Langerhans cell histiocytosis bone lesions with zoledronic acid: a case series. International journal of hematology. 2011;93(6):782-6.
  • Referans21. Heritier S, Emile J-F, Barkaoui M-A, Thomas C, Fraitag S, Boudjemaa S, et al. BRAF mutation correlates with high-risk Langerhans cell histiocytosis and increased resistance to first-line therapy. Journal of clinical oncology. 2016;34(25):3023.
  • Referans22. Váradi Z, Bánusz R, Csomor J, Kállay K, Varga E, Kertész G, et al. Effective BRAF inhibitor vemurafenib therapy in a 2-year-old patient with sequentially diagnosed Langerhans cell histiocytosis and Erdheim–Chester disease. OncoTargets and therapy. 2017;10:521.
  • Referans23. Héritier S, Jehanne M, Leverger G, Emile J-F, Alvarez J-C, Haroche J, et al. Vemurafenib use in an infant for high-risk Langerhans cell histiocytosis. JAMA oncology. 2015;1(6):836-8.
  • Referans24. Diamond EL, Durham BH, Ulaner GA, Drill E, Buthorn J, Ki M, et al. Efficacy of MEK inhibition in patients with histiocytic neoplasms. Nature. 2019;567(7749):521-4.

Adult Onset Langerhans Cell Histiocytosis: A Single Center Experience

Yıl 2023, Cilt: 5 Sayı: 1, 59 - 64, 15.01.2023

Serhat Çelik Zeynep Tuğba Güven Onur Aşık Muzaffer Keklik Leylagül Kaynar Bülent Eser Mustafa Çetin Ali Ünal

https://doi.org/10.37990/medr.1159055

Öz

Aim: Langerhans cell histiocytosis (LCH) is a proliferative disease caused by abnormal proliferation of histiocytes in the bone marrow dendritic cell structure. LCH is very rare in adults and its incidence is 1-2 cases per million. Therefore, there is still no clear management plan in adults. It was aimed to evaluate the very rare adult LCH patients.
Materials and Methods: Ten LCH patients who were followed up at Erciyes University Hematology Department between January 2010 and January 2020 were included.
Results: Eight (80%) of them were male and 2 (20%) were female. The median age of the patients was 34.5 ± 8.4 (23-52) years. Although the most common involvement at the time of diagnosis was bone in 7 patients (70%) and lung in 3 patients (30%); pituitary, tympanic membrane, liver and brain involvement were also observed. Three (30%) patients had single system involvement and 7 (70%) had multisystem. Four (40%) patients had relapsed and all had multisystemic involvement. The BRAF V600E mutation could be performed in 3 patients. It was negative in two patients and positive in 1 patient. All patients still have remission.
Conclusion: Unlike children, LCH has a better course in adults. The most important approach is to determine single or multisystem involvement. In our patients with single system involvement, we obtained response with corticosteroid and surgery alone. We have observed that vinblastin plus metilprednizolon treatment is a good option for multisystemic involvement. In relapsed patients, we obtained a significant response with clofarabine.

Anahtar Kelimeler

Adult BRAF V600E mutation clofarabin histiocytosis langerhans cell Radiotherapy Treatment vinblastin plus methylprednisolone Adult, BRAF V600E mutation, clofarabin, histiocytosis, langerhans cell, Radiotherapy, Treatment, vinblastin plus methylprednisolone

Kaynakça

  • Referans1. AKDEMİR O, ÇOLAK A. Langerhans hücresi histiositozisi. Turkiye Klinikleri Journal of Surgical Medical Sciences. 2007;3(51):1-4.
  • Referans2. Alston RD, Tatevossian R, McNally RJ, Kelsey A, Birch JM, Eden TO. Incidence and survival of childhood Langerhans cell histiocytosis in Northwest England from 1954 to 1998. Pediatric blood & cancer. 2007;48(5):555-60.
  • Referans3. McClain K. Treatment of langerhans cell histiocytosis. UpToDate. Waltham, MA 2013.
  • Referans4. Weitzman S, Braier J, Donadieu J, Egeler RM, Grois N, Ladisch S, et al. 2′‐chlorodeoxyadenosine (2‐CdA) as salvage therapy for Langerhans cell histiocytosis (LCH). results of the LCH‐S‐98 protocol of the histiocyte society. Pediatric blood & cancer. 2009;53(7):1271-6.
  • Referans5. Gadner H, Minkov M, Grois N, Pötschger U, Thiem E, Arico M, et al. Therapy prolongation improves outcome in multisystem Langerhans cell histiocytosis. Blood, The Journal of the American Society of Hematology. 2013;121(25):5006-14.
  • Referans6. Allen CE, Flores R, Rauch R, Dauser R, Murray JC, Puccetti D, et al. Neurodegenerative central nervous system Langerhans cell histiocytosis and coincident hydrocephalus treated with vincristine/cytosine arabinoside. Pediatric blood & cancer. 2010;54(3):416-23.
  • Referans7. Simko SJ, Tran HD, Jones J, Bilgi M, Beaupin LK, Coulter D, et al. Clofarabine salvage therapy in refractory multifocal histiocytic disorders, including Langerhans cell histiocytosis, juvenile xanthogranuloma and Rosai–Dorfman disease. Pediatric blood & cancer. 2014;61(3):479-87.
  • Referans8. McClain KL, Picarsic J, Chakraborty R, Zinn D, Lin H, Abhyankar H, et al. CNS Langerhans cell histiocytosis: Common hematopoietic origin for LCH‐associated neurodegeneration and mass lesions. Cancer. 2018;124(12):2607-20.
  • Referans9. Sahin F, Ozen K, Vural F, Kamer S, Argin M, Anacak Y. Retrospective analysis of seven patients with adult-onset Langerhans cell histiocytosis syndromes: A single center experience. Turk J Hematol. 2007;24:75-9.
  • Referans10. Grois N, Pötschger U, Prosch H, Minkov M, Arico M, Braier J, et al. Risk factors for diabetes insipidus in langerhans cell histiocytosis. Pediatric blood & cancer. 2006;46(2):228-33.
  • Referans11. Prosch H, Grois N, Prayer D, Waldhauser F, Steiner M, Minkov M, et al. Central diabetes insipidus as presenting symptom of Langerhans cell histiocytosis. Pediatric blood & cancer. 2004;43(5):594-9.
  • Referans12. Di Iorgi N, Allegri AEM, Napoli F, Calcagno A, Calandra E, Fratangeli N, et al. Central diabetes insipidus in children and young adults: etiological diagnosis and long-term outcome of idiopathic cases. The Journal of Clinical Endocrinology & Metabolism. 2014;99(4):1264-72.
  • Referans13. Lindahl L, Fenger‐Grøn M, Iversen L. Topical nitrogen mustard therapy in patients with Langerhans cell histiocytosis. British Journal of Dermatology. 2012;166(3):642-5.
  • Referans14. McClain KL, Kozinetz CA. A phase II trial using thalidomide for Langerhans cell histiocytosis. Pediatric blood & cancer. 2007;48(1):44-9.
  • Referans15. Cantu MA, Lupo PJ, Bilgi M, Hicks MJ, Allen CE, McClain KL. Optimal therapy for adults with Langerhans cell histiocytosis bone lesions. PloS one. 2012;7(8).
  • Referans16. Duan M-H, Han X, Li J, Zhang W, Zhu T-N, Han B, et al. Comparison of vindesine and prednisone and cyclophosphamide, etoposide, vindesine, and prednisone as first-line treatment for adult Langerhans cell histiocytosis: A single-center retrospective study. Leukemia research. 2016;42:43-6.
  • Referans17. Minkov M, Steiner M, Pötschger U, Aricò M, Braier J, Donadieu J, et al. Reactivations in multisystem Langerhans cell histiocytosis: data of the international LCH registry. The Journal of pediatrics. 2008;153(5):700-5. e2.
  • Referans18. Abraham A, Alsultan A, Jeng M, Rodriguez‐Galindo C, Campbell PK. Clofarabine salvage therapy for refractory high‐risk langerhans cell histiocytosis. Pediatric blood & cancer. 2013;60(6):E19-E22.
  • Referans19. Willis B, Ablin A, Weinberg V, Zoger S, Wara WM, Matthay KK. Disease course and late sequelae of Langerhans' cell histiocytosis: 25-year experience at the University of California, San Francisco. Journal of clinical oncology. 1996;14(7):2073-82.
  • Referans20. Sivendran S, Harvey H, Lipton A, Drabick J. Treatment of Langerhans cell histiocytosis bone lesions with zoledronic acid: a case series. International journal of hematology. 2011;93(6):782-6.
  • Referans21. Heritier S, Emile J-F, Barkaoui M-A, Thomas C, Fraitag S, Boudjemaa S, et al. BRAF mutation correlates with high-risk Langerhans cell histiocytosis and increased resistance to first-line therapy. Journal of clinical oncology. 2016;34(25):3023.
  • Referans22. Váradi Z, Bánusz R, Csomor J, Kállay K, Varga E, Kertész G, et al. Effective BRAF inhibitor vemurafenib therapy in a 2-year-old patient with sequentially diagnosed Langerhans cell histiocytosis and Erdheim–Chester disease. OncoTargets and therapy. 2017;10:521.
  • Referans23. Héritier S, Jehanne M, Leverger G, Emile J-F, Alvarez J-C, Haroche J, et al. Vemurafenib use in an infant for high-risk Langerhans cell histiocytosis. JAMA oncology. 2015;1(6):836-8.
  • Referans24. Diamond EL, Durham BH, Ulaner GA, Drill E, Buthorn J, Ki M, et al. Efficacy of MEK inhibition in patients with histiocytic neoplasms. Nature. 2019;567(7749):521-4.
Toplam 24 adet kaynakça vardır.

Ayrıntılar

Birincil Dil İngilizce
Konular Sağlık Kurumları Yönetimi
Bölüm Özgün Makaleler
Yazarlar

Serhat Çelik 0000-0002-1052-9800

Zeynep Tuğba Güven 0000-0003-1600-9731

Onur Aşık 0000-0002-9422-2944

Muzaffer Keklik 0000-0002-6426-5249

Leylagül Kaynar 0000-0002-2035-9462

Bülent Eser 0000-0002-4513-3486

Mustafa Çetin 0000-0003-4512-1124

Ali Ünal 0000-0001-7011-3412

Erken Görünüm Tarihi 15 Ocak 2023
Yayımlanma Tarihi 15 Ocak 2023
Kabul Tarihi 6 Ekim 2022
Yayımlandığı Sayı Yıl 2023 Cilt: 5 Sayı: 1

Kaynak Göster

AMA Çelik S, Güven ZT, Aşık O, Keklik M, Kaynar L, Eser B, Çetin M, Ünal A. Adult Onset Langerhans Cell Histiocytosis: A Single Center Experience. Med Records. Ocak 2023;5(1):59-64. doi:10.37990/medr.1159055
 Kapak Resmi İndir

         

Chief Editors
Assoc. Prof. Zülal Öner
Address: İzmir Bakırçay University, Department of Anatomy, İzmir, Turkey

Assoc. Prof. Deniz Şenol
Address: Düzce University, Department of Anatomy, Düzce, Turkey

E-mail: medrecsjournal@gmail.com

Publisher:
Medical Records Association (Tıbbi Kayıtlar Derneği)
Address: Düzce / Türkiye


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