Follicular Eruption With Folliculotropic Lymphocytic Infiltrates Associated With Iatrogenic Immunosuppression: Report and Study of 3 Cases, and Review of the Literature.
Journal
The American Journal of dermatopathology
ISSN: 1533-0311
Titre abrégé: Am J Dermatopathol
Pays: United States
ID NLM: 7911005
Informations de publication
Date de publication:
Jul 2020
Jul 2020
Historique:
pubmed:
4
12
2019
medline:
17
4
2021
entrez:
3
12
2019
Statut:
ppublish
Résumé
Several cases of folliculotropic mycosis fungoides, associated with immunosuppressive therapy, including calcineurin inhibitors, have been reported in solid organ transplant patients. We have encountered 3 patients on immunosuppressive therapy who developed follicular eruptions with folliculocentric infiltrates of nonatypical lymphocytes. To characterize these follicular eruptions and review the literature. Three patients, aged 7-15 years, who were treated with systemic immunosuppressive therapy developed follicular eruptions characterized histopathologically by folliculocentric lymphocytic infiltrates. These were studied clinically, histopathologically, immunophenotypically, and molecularly for T-cell receptor (TCR) gene rearrangement. All 3 cases were characterized histopathologically by folliculocentric infiltrates of nonatypical CD3 T lymphocytes with variable follicular exocytosis. Two cases also showed follicular mucinosis. Marked reduction in CD7 staining, and marked predominance of CD4 cells over CD8 cells was observed in all 3 cases. The TCR gene rearrangement studies were monoclonal in 2 cases. Oral calcineurin inhibitors (2 cyclosporine A and 1 tacrolimus) were part of the therapeutic regimen in all 3 patients. Their cessation along with local corticosteroid creams in 2 patients, and phototherapy with oral acitretin in one patient, was associated with complete clinical remission. Patients undergoing systemic immunosuppressive therapy that includes calcineurin inhibitors might develop follicular eruption with some immunophenotypical variations and a monoclonal TCR gene rearrangement but lack sufficient cytomorphological features of folliculotropic mycosis fungoides. Altering the immunosuppressive agent including calcineurin inhibitors may result in regression of the eruptions.
Sections du résumé
BACKGROUND
BACKGROUND
Several cases of folliculotropic mycosis fungoides, associated with immunosuppressive therapy, including calcineurin inhibitors, have been reported in solid organ transplant patients. We have encountered 3 patients on immunosuppressive therapy who developed follicular eruptions with folliculocentric infiltrates of nonatypical lymphocytes.
OBJECTIVE
OBJECTIVE
To characterize these follicular eruptions and review the literature.
METHODS
METHODS
Three patients, aged 7-15 years, who were treated with systemic immunosuppressive therapy developed follicular eruptions characterized histopathologically by folliculocentric lymphocytic infiltrates. These were studied clinically, histopathologically, immunophenotypically, and molecularly for T-cell receptor (TCR) gene rearrangement.
RESULTS
RESULTS
All 3 cases were characterized histopathologically by folliculocentric infiltrates of nonatypical CD3 T lymphocytes with variable follicular exocytosis. Two cases also showed follicular mucinosis. Marked reduction in CD7 staining, and marked predominance of CD4 cells over CD8 cells was observed in all 3 cases. The TCR gene rearrangement studies were monoclonal in 2 cases. Oral calcineurin inhibitors (2 cyclosporine A and 1 tacrolimus) were part of the therapeutic regimen in all 3 patients. Their cessation along with local corticosteroid creams in 2 patients, and phototherapy with oral acitretin in one patient, was associated with complete clinical remission.
CONCLUSIONS
CONCLUSIONS
Patients undergoing systemic immunosuppressive therapy that includes calcineurin inhibitors might develop follicular eruption with some immunophenotypical variations and a monoclonal TCR gene rearrangement but lack sufficient cytomorphological features of folliculotropic mycosis fungoides. Altering the immunosuppressive agent including calcineurin inhibitors may result in regression of the eruptions.
Identifiants
pubmed: 31789839
doi: 10.1097/DAD.0000000000001547
pii: 00000372-202007000-00005
doi:
Substances chimiques
Immunosuppressive Agents
0
Types de publication
Case Reports
Journal Article
Review
Langues
eng
Sous-ensembles de citation
IM
Pagination
498-505Références
Seçkin D, Barete S, Euvrard S, et al. Primary cutaneous posttransplant lymphoproliferative disorders in solid organ transplant recipients: a multicenter European case series. Am J Transpl. 2013;13:2146–2153.
Spence-Shishido A, Streicher JL, George RP, et al. Folliculotropic mycosis fungoides as a post transplant lymphoproliferative disorder. Pediatrics. 2015;136:e701–e705.
Vlassova N, Frankel Y, Patel MJ, et al. Folliculotropic mycosis fungoides following renal transplantation. J Am Acad Dermatol. 2011;65:e96–e98.
Rodríguez-Gil Y, Palencia SI, López-Ríos F, et al. Mycosis fungoides after solid-organ transplantation: report of 2 new cases. Am J Dermatopathol. 2008;30:150–155.
Laffitte E, Venetz JP, Aubert JD, et al. Mycosis fungoides in a lung transplant recipient with advanced ciclosporin nephropathy: management with mechlorethamine and subsequent renal transplantation. Dermatology. 2008;217:87–88.
Amin A, Burkhart C, Groben P, et al. Primary cutaneous T-cell lymphoma following organ transplantation in a 16-year-old boy. Pediatr Dermatol. 2009;26:112–113.
Ravat FE, Spittle MF, Russell-Jones R. Primary cutaneous T-cell lymphoma occurring after organ transplantation. J Am Acad Dermatol. 2006;54:668–675.
Pomerantz RG, Campbell LS, Jukic DM, et al. Posttransplant cutaneous T-cell lymphoma: case reports and review of the association of calcineurin inhibitor use with posttransplant lymphoproliferative disease risk. Arch Dermatol. 2010;146:513–516.
Ward HA, Russo GG, McBurney E, et al. Posttransplant primary cutaneous T-cell lymphoma. J Am Acad Dermatol. 2001;44:675–680.
De Nisi MC, D'Amuri A, Lalinga AV, et al. Posttransplant primary cutaneous CD30 (Ki-1)-positive anaplastic large T-cell lymphoma. A case report. Br J Dermatol. 2005;152:1068–1070.
Rogers TS, McGevna L, Cook DL. Pediatric cutaneous T-cell post-transplant lymphoproliferative disorder: case report and review of the literature. J Cutan Pathol. 2018;45:858–863.
van Doorn R, Scheffer E, Willemze R. Follicular mycosis fungoides, a distinct disease entity with or without associated follicular mucinosis: a clinicopathologic and follow-up study of 51 patients. Arch Dermatol. 2002;138:191–198.
Pimpinelli N, Olsen EA, Santucci M, et al. Defining early mycosis fungoides. J Am Acad Dermatol. 2005;53:1053–1063.
Hodak E, Amitay-Laish I, Atzmony L, et al. New insights into folliculotropic mycosis fungoides (FMF): a single-center experience. J Am Acad Dermatol. 2016;75:347–355.
Ritz N, Sahar D, Bergman R. T-cell receptor gene rearrangement studies using the GeneScan technique as an adjunct to the histopathological diagnosis of mycosis fungoides. Am J Dermatopathol. 2015;37:210–213.
Kirby B, Owen CM, Blewitt RW, et al. Cutaneous t-cell lymphoma developing in a patient on cyclosporin therapy. J Am Acad Dermatol. 2002;47:S165–S167.
Mougel F, Dalle S, Balme B, et al. Aggressive CD30 large cell lymphoma after cyclosporin given for putative atopic dermatitis. Dermatology. 2006;213:239–241.
Wood GS, Hong SR, Sasaki DT, et al. Leu8/CD7 antigen expression by CD3+ T cells: comparative analysis of skin and blood in mycosis fungoides/Sézary syndrome relative to normal blood values. J Am Acad Dermatol. 1990;22:602–607.
Nuckols JD, Shea CR, Horenstein MG, et al. Quantitation of intraepidermal T-cell subsets in formalin-fixed, paraffin-embedded tissue helps in the diagnosis of mycosis fungoides. J Cutan Pathol. 1999;26:169–175.
Cotta AC, Cintra ML, de Souza EM, et al. Reassessment of diagnostic criteria in cutaneous lymphocytic infiltrates. Sao Paulo Med J. 2004;122:161–165.
Tirumalae R, Panjwani PK. Origin use of CD4, CD8, and CD1a immunostains in distinguishing mycosis fungoides from its inflammatory mimics: a pilot study. Indian J Dermatol. 2012;57:424–427.
Machan, Salma MD, El Shabrawi-Caelen, et al. White goose bumps all over the body: answer. Am J Dermatopathol. 2017;39:871–872.
Dias-Polak D, Bergman R, Avitan-Hersh E. Mycophenolate mofetil therapy in adult patients with recalcitrant atopic dermatitis. J Dermatolog Treat. 2019;30:49–51.
Magro CM, Daniels BH, Crowson AN. Drug induced pseudolymphoma. Semin Diagn pathol. 2018;35:247–259.