10.5826/dpc.1302a78
text/xml
2160-9381
2628
en
Viktoryia Kazlouskaya
https://creativecommons.org/licenses/by-nc/4.0
Dermatology Practical & Conceptual
immune reconstitution inflammatory syndrome
Eosinophilic Pustular Folliculitis in the Setting of Solid Organ Transplant Immunosuppression
Text.Serial.Journal
https://dpcj.org/index.php/dpc
Research Letter
2023-04-29
2022-06-02
2023-04-29
2023-06-05
e2023078
Dermatol Pract Concept
Viktoryia Kazlouskaya
2023/04/29
10.5826/dpc.1302a78
e2023078
University of Pittsburgh Department of Dermatology/Dermatopathology, Pittsburgh, PA, United States
2160-9381
immune reconstitution inflammatory syndrome
en
e2023078
Sasaki Y, Kishi A, Takagi S, Uchida N, Hayashi N. Eosinophilic pustular folliculitis after hematopoietic stem cell transplantation: A study of 11 cases. J Dermatol. 2021;48(5):e231-e233. DOI: 10.1111/1346-8138.15846. PMID: 33715192. PMCID: PMC8251622.
Eosinophilic Pustular Folliculitis in the Setting of Solid Organ Transplant Immunosuppression
https://dpcj.org/index.php/dpc/article/download/2628/2092
       Eosinophilic Pustular Folliculitis in the Setting
       of Solid Organ Transplant Immunosuppression
              Jeffrey Chen1, Colleen J Beatty2, Lauryn M Falcone3, Joseph C English III3,
                                        Viktoryia Kazlouskaya2


1 University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania, United States
2 University of Pittsburgh Department of Dermatology/Dermatopathology, Pittsburgh, Pennsylvania, United States
3 University of Pittsburgh Department of Dermatology, Pittsburgh, Pennsylvania, United States



Key words: Eosinophilic pustular folliculitis, Ofuji disease, hematopoietic stem cell transplantation, immunosuppression,
immune reconstitution inflammatory syndrome
Citation: Chen J, Beatty CJ, Falcone LM, English JC, Kazlouskaya V. Eosinophilic Pustular Folliculitis in the Setting of Solid Organ
Transplant Immunosuppression. Dermatol Pract Concept. 2023;13(2):e2023078. DOI: https://doi.org/10.5826/dpc.1302a78
Accepted: June 20, 2023; Published: April 2023
Copyright: ©2023 Chen et al. This is an open-access article distributed under the terms of the Creative Commons
Attribution-NonCommercial License (BY-NC-4.0), https://creativecommons.org/licenses/by-nc/4.0/, which permits unrestricted
noncommercial use, distribution, and reproduction in any medium, provided the original authors and source are credited.
Funding: None.
Competing interests: None.
Authorship: All authors have contributed significantly to this publication.
Corresponding author: Viktoryia Kazlouskaya, MD, PhD, University of Pittsburgh, Department of Dermatology and Dermatopathology,
Medical Arts Building, 3708 5th Avenue, Suite 500.68, Pittsburgh, Pennsylvania 15213 E-mail: viktoriakozlovskaya@yahoo.com




Introduction                                                             Case Presentation
Eosinophilic pustular folliculitis (EPF) is an inflammatory              A 69-year-old male on long-term immunosuppression for
dermatosis that presents as follicular papulopustules with               cardiac transplantation secondary to non-ischemic cardio-
pronounced eosinophils. Subtypes include classic EPF,                    myopathy was evaluated for a diffuse pruritic rash that
infancy-associated EPF, and immunosuppression-associated                 began several weeks after switching medications from
EPF (IS-EPF). IS-EPF can be sub-divided into HIV-associated              tacrolimus to sirolimus. Skin examination revealed multi-
(IS/HIV) and non-HIV-associated (IS/non-HIV), with the                   ple erythematous to purpuric papules involving the torso,
latter associated with hematologic malignancy, particularly              upper back, and upper extremities (Figure 1). Exanthem-
after hematopoietic stem cell transplantation (HSCT) [1].                atous drug eruption was favored as the initial diagnosis
Herein, we describe a case of IS-EPF in the setting of chronic           due to temporal correlation, but graft-versus-host disease,
immunosuppression due to cardiac transplantation. This is                neutrophilic dermatitis, and infectious process were also
the second reported case of IS-EPF associated with solid                 considered.
­organ transplant, with the first reported case in 2014 by                    Histopathological examination revealed mid-dermal
Kim et al [2].                                                           perifollicular collections of lymphocytes, histiocytes, and




Research Letter | Dermatol Pract Concept. 2023;13(2):e2023078                                                                          1
                                Figure 1. Erythematous swollen papulopustules on the back.




prominent eosinophils (Figure 2). GMS, Gram, AFB, and             that the expression patterns of IL-36β, IL-36γ, and IL36Ra
PAS stains were negative. Biopsy findings were consistent         contribute partially to the eosinophil migration in EPF [4].
with EPF, likely associated with the patient’s immunosup-             Unlike classic EPF, the lesions observed in IS-EPF are less
pression. The patient was treated with triamcinolone aceton-      strongly associated with the formation of pustules and do
ide cream and showed gradual resolution.                          not show the same predilection for the face, as they tend to
                                                                  affect the upper body and extremities [1]. Moreover, IS-EPF
                                                                  responds more favorably to topical or systemic steroids com-

Conclusions                                                       pared to classic EPF suggesting different pathogenesis [5].
                                                                  The clinicopathological features of this case are similar to
While the pathophysiology of EPF is unknown, prostaglan-          those observed in IS/non-HIV patients, specifically, cases of
din D2 (PGD2) and the IL-36 cytokine family are speculated        IS-EPF that developed after HSCT. A study by Sasaki et al
to be involved in the folliculotropic infiltration of eosino-     likened the lesions of IS-EPF following HSCT to the devel-
phils [3,4]. Increases in PGD2 production in EPF have been        opment of IS-EPF in HIV patients caused by immune recon-
demonstrated to increase mRNA expression of eotaxin-3,            stitution inflammatory syndrome (IRIS) [6]. In our case, the
which is an eosinophilic chemotactic factor produced by se-       patient developed EPF after his tacrolimus was switched to
bocytes [3]. IL36α, IL-36β, IL-36γ, and IL36Ra are known          sirolimus, which is a decrease in immunosuppression and a
modulators of the cutaneous inflammatory response in-             step closer to immune reconstitution. Accordingly, this case
volved in eosinophil recruitment. Previous studies showed         provides preliminary evidence to suggest a relationship be-
that IL-36β, IL-36γ, and IL36Ra are upregulated in EPF.           tween IRIS and IS-EPF in the setting of solid organ trans-
Interestingly, the expression of IL36Ra was negatively cor-       plant immunosuppression, however, further accumulation of
related with IL-36β and IL-36γ in normal skin, suggesting         cases is needed to investigate this potential association.




2                                                                     Research Letter | Dermatol Pract Concept. 2023;13(2):e2023078
       Figure 2. (A) Perifollicular granulomatous inflammation (H&E, x200). (B) High power demonstrating numerous eosinophils in
       the infiltrate (H&E stain, x400). (C) Another focus with keratin remnants surrounded by granulomatous inflammation (H&E
       stain, x200). (D) High power demonstrating numerous eosinophils in the infiltrate (H&E stain, x400).




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Research Letter | Dermatol Pract Concept. 2023;13(2):e2023078                                                                                3
