Gastrointestinal lesion in adult-onset Langerhans cell histiocytosis.
Adolescent
Adult
Age of Onset
Aged
Antineoplastic Combined Chemotherapy Protocols
/ therapeutic use
Asian People
Colonoscopy
Endoscopy, Gastrointestinal
Female
Gastrointestinal Diseases
/ diagnostic imaging
Histiocytosis, Langerhans-Cell
/ diagnostic imaging
Humans
Male
Methotrexate
/ administration & dosage
Middle Aged
Prednisolone
/ administration & dosage
Recurrence
Treatment Outcome
Vinblastine
/ administration & dosage
Young Adult
Adults
Gastrointestinal tract
Langerhans cell histiocytosis
Journal
International journal of clinical oncology
ISSN: 1437-7772
Titre abrégé: Int J Clin Oncol
Pays: Japan
ID NLM: 9616295
Informations de publication
Date de publication:
Nov 2020
Nov 2020
Historique:
received:
03
05
2020
accepted:
25
06
2020
pubmed:
10
9
2020
medline:
15
12
2020
entrez:
9
9
2020
Statut:
ppublish
Résumé
Langerhans cell histiocytosis (LCH) is a rare disease primarily occurring in children, and commonly involves the bone and skin; gastrointestinal tract involvement is notably rare. The incidence and significance of gastrointestinal lesions in adult LCH are unclear; thus, we aimed to investigate adult Japanese cases of LCH and clarify the features of gastrointestinal involvement. We gathered clinical information on 43 Japanese cases of adult LCH and analyzed patient backgrounds, affected organs, features of the gastrointestinal lesions, and the clinical courses. Thirteen patients underwent endoscopic examinations: an upper gastrointestinal endoscopy alone in 5, lower gastrointestinal endoscopy alone in 3, and both in 5 patients. A gastric lesion (one case), colonic lesion (one case), and both gastric and rectal lesions (one case) were detected. The three cases of gastrointestinal involvement also exhibited nongastrointestinal multisystem LCH lesions and showed no gastrointestinal symptoms or increased uptake on positron emission tomography. Endoscopy revealed small erosions without specific features; histological examinations were required for diagnosis. These three cases were treated with chemotherapy, comprising vinblastine/prednisolone, methotrexate, and daily 6-mercaptopurine, for 36 weeks; in two cases, the clinical condition remained stable for several years post-treatment. One case showed recurrence 1 year 7 months after treatment, and chemotherapy was re-administered. No case with single-system disease exhibited gastrointestinal involvement. Although gastrointestinal LCH lesions are rare, they were more common than expected in our cases of multisystem LCH. However, these lesions were relatively small and did not affect the patients' clinical courses.
Sections du résumé
BACKGROUND
BACKGROUND
Langerhans cell histiocytosis (LCH) is a rare disease primarily occurring in children, and commonly involves the bone and skin; gastrointestinal tract involvement is notably rare. The incidence and significance of gastrointestinal lesions in adult LCH are unclear; thus, we aimed to investigate adult Japanese cases of LCH and clarify the features of gastrointestinal involvement.
METHODS
METHODS
We gathered clinical information on 43 Japanese cases of adult LCH and analyzed patient backgrounds, affected organs, features of the gastrointestinal lesions, and the clinical courses.
RESULTS
RESULTS
Thirteen patients underwent endoscopic examinations: an upper gastrointestinal endoscopy alone in 5, lower gastrointestinal endoscopy alone in 3, and both in 5 patients. A gastric lesion (one case), colonic lesion (one case), and both gastric and rectal lesions (one case) were detected. The three cases of gastrointestinal involvement also exhibited nongastrointestinal multisystem LCH lesions and showed no gastrointestinal symptoms or increased uptake on positron emission tomography. Endoscopy revealed small erosions without specific features; histological examinations were required for diagnosis. These three cases were treated with chemotherapy, comprising vinblastine/prednisolone, methotrexate, and daily 6-mercaptopurine, for 36 weeks; in two cases, the clinical condition remained stable for several years post-treatment. One case showed recurrence 1 year 7 months after treatment, and chemotherapy was re-administered. No case with single-system disease exhibited gastrointestinal involvement.
CONCLUSIONS
CONCLUSIONS
Although gastrointestinal LCH lesions are rare, they were more common than expected in our cases of multisystem LCH. However, these lesions were relatively small and did not affect the patients' clinical courses.
Identifiants
pubmed: 32901339
doi: 10.1007/s10147-020-01739-1
pii: 10.1007/s10147-020-01739-1
doi:
Substances chimiques
Vinblastine
5V9KLZ54CY
Prednisolone
9PHQ9Y1OLM
Methotrexate
YL5FZ2Y5U1
Types de publication
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
1945-1950Références
Egeler RM, D'Angio GJ (1995) Langerhans cell histiocytosis. J Pediatr 127(1):1–11. https://doi.org/10.1016/s0022-3476(95)70248-2
doi: 10.1016/s0022-3476(95)70248-2
pubmed: 7608790
Beverley PC, Egeler RM, Arceci RJ et al (2005) The Nikolas symposia and histiocytosis. Nat Rev Cancer 5(6):488–494. https://doi.org/10.1038/nrc1632
doi: 10.1038/nrc1632
pubmed: 15928676
Lian C, Lu Y, Shen S (2016) Langerhans cell histiocytosis in adults: a case report and review of the literature. Oncotarget 7(14):18678–18683. https://doi.org/10.18632/oncotarget.7892
doi: 10.18632/oncotarget.7892
pubmed: 26942568
pmcid: 4951319
Badalian-Very G, Vergilio JA, Degar BA et al (2012) Recent advances in the understanding of Langerhans cell histiocytosis. Br J Haematol 156(2):163–172. https://doi.org/10.1111/j.1365-2141.2011.08915.x
doi: 10.1111/j.1365-2141.2011.08915.x
pubmed: 22017623
Swerdlow SH, Campo E, Harris NL et al (2017) WHO classification of tumours of haematopoietic and lymphoid tissues, vol 2. World Health Organization Classification of Tumours. International Agency for Research on Cancer (IARC). 69008 Lyon, France. https://publications.iarc.fr/Book-And-Report-Series/Who-Classification-Of-Tumours/WHO-Classification-Of-Tumours-Of-Haematopoietic-And-Lymphoid-Tissues-2017
Chang KL, Snyder DS (2008) Langerhans cell histiocytosis. Cancer Treat Res 142:383–398. https://doi.org/10.1007/978-0-387-73744-7_17
doi: 10.1007/978-0-387-73744-7_17
pubmed: 18283796
Howarth DM, Gilchrist GS, Mullan BP et al (1999) Langerhans cell histiocytosis: diagnosis, natural history, management, and outcome. Cancer 85(10):2278–2290. https://doi.org/10.1002/(sici)1097-0142(19990515)85:10<2278:aid-cncr25>3.0.co;2-u
doi: 10.1002/(sici)1097-0142(19990515)85:10<2278::aid-cncr25>3.0.co;2-u
pubmed: 10326709
(1996) A multicentre retrospective survey of Langerhans’ cell histiocytosis: 348 cases observed between 1983 and 1993. The French Langerhans’ Cell Histiocytosis Study Group. Arch Dis Child 75(1):17–24. https://doi.org/10.1136/adc.75.1.17
Morimoto A, Shimazaki C, Takahashi S et al (2013) Therapeutic outcome of multifocal Langerhans cell histiocytosis in adults treated with the Special C regimen formulated by the Japan LCH Study Group. Int J Hematol 97(1):103–108. https://doi.org/10.1007/s12185-012-1245-0
doi: 10.1007/s12185-012-1245-0
pubmed: 23243004
Groisman GM, Rosh JR, Harpaz N (1994) Langerhans cell histiocytosis of the stomach. A cause of granulomatous gastritis and gastric polyposis. Arch Pathol Lab Med 118(12):1232–1235
pubmed: 7979922
Yadav SP, Kharya G, Mohan N et al (2010) Langerhans cell histiocytosis with digestive tract involvement. Pediatr Blood Cancer 55(4):748–753. https://doi.org/10.1002/pbc.22663
doi: 10.1002/pbc.22663
pubmed: 20535829
Singhi AD, Montgomery EA (2011) Gastrointestinal tract langerhans cell histiocytosis: a clinicopathologic study of 12 patients. Am J Surg Pathol 35(2):305–310. https://doi.org/10.1097/PAS.0b013e31820654e4
doi: 10.1097/PAS.0b013e31820654e4
pubmed: 21263252
Terracciano L, Kocher T, Cathomas G et al (1999) Langerhans cell histiocytosis of the stomach with atypical morphological features. Pathol Int 49(6):553–556. https://doi.org/10.1046/j.1440-1827.1999.00909.x
doi: 10.1046/j.1440-1827.1999.00909.x
pubmed: 10469399
Nozaki Y, Oshiro H, Nakajima A (2010) Image of the month. Langerhans cell histiocytosis of the stomach mimicking early gastric cancer. Clin Gastroenterol Hepatol 8(9):A18. https://doi.org/10.1016/j.cgh.2010.01.009
doi: 10.1016/j.cgh.2010.01.009
pubmed: 20117242
Wada R, Yagihashi S, Konta R et al (1992) Gastric polyposis caused by multifocal histiocytosis X. Gut 33(7):994–996. https://doi.org/10.1136/gut.33.7.994
doi: 10.1136/gut.33.7.994
pubmed: 1644344
pmcid: 1379420
Heritier S, Emile JF, Barkaoui MA et al (2016) BRAF mutation correlates with high-risk Langerhans cell histiocytosis and increased resistance to first-line therapy. J Clin Oncol 34(25):3023–3030. https://doi.org/10.1200/JCO.2015.65.9508
doi: 10.1200/JCO.2015.65.9508
pubmed: 27382093
pmcid: 5321082
Berres ML, Lim KP, Peters T et al (2015) BRAF-V600E expression in precursor versus differentiated dendritic cells defines clinically distinct LCH risk groups. J Exp Med 212(2):281. https://doi.org/10.1084/jem.2013097701202015c
doi: 10.1084/jem.2013097701202015c
pubmed: 25646268
pmcid: 4322054
Cortazar JM, Kim AS (2017) Incidental Langerhans cell histiocytosis of the colon with BRAF p.V600E mutation. Blood 130(16):1870. https://doi.org/10.1182/blood-2017-07-796607
doi: 10.1182/blood-2017-07-796607
pubmed: 29051154
Gadner H, Grois N, Arico M et al (2001) A randomized trial of treatment for multisystem Langerhans’ cell histiocytosis. J Pediatr 138(5):728–734. https://doi.org/10.1067/mpd.2001.111331
doi: 10.1067/mpd.2001.111331
pubmed: 11343051
Kobayashi M, Tojo A (2018) Langerhans cell histiocytosis in adults: advances in pathophysiology and treatment. Cancer Sci 109(12):3707–3713. https://doi.org/10.1111/cas.13817
doi: 10.1111/cas.13817
pubmed: 30281871
pmcid: 6272080
Kobayashi M, Ando S, Kawamata T et al (2020) Clinical features and outcomes of adult Langerhans cell histiocytosis: a single-center experience. Int J Hematol. https://doi.org/10.1007/s12185-020-02892-z
doi: 10.1007/s12185-020-02892-z
pubmed: 33025461