10.5826/dpc.1203a71
text/xml
2160-9381
1878
en
Sedat Kiraz
https://creativecommons.org/licenses/by-nc/4.0
Dermatology Practical & Conceptual
kaposi sarcoma
Unusual Presentation of Kaposi Sarcoma During Adalimumab Therapy: A Case Report
Text.Serial.Journal
https://dpcj.org/index.php/dpc
Research Letter
2022-07-28
2021-07-09
2022-07-28
2022-09-15
e2022071
Dermatol Pract Concept
Sedat Kiraz
2022/07/28
10.5826/dpc.1203a71
e2022071
Hacettepe University, School of Medicine, Department of Rheumatology
2160-9381
kaposi sarcoma
en
e2022071
Unusual Presentation of Kaposi Sarcoma During Adalimumab Therapy: A Case Report
https://dpcj.org/index.php/dpc/article/download/1878/1650
                    Dermatology Practical & Conceptual


     Unusual Presentation of Kaposi Sarcoma During
          Adalimumab Therapy: a Case Report
       Gulsun-Hazan Tabak1, Basak Yalici-Armagan1, Burkay-Adem Sahin1, Ozay Gokoz2,
                                        Sedat Kiraz3


1 Hacettepe University, School of Medicine, Department of Dermatology and Venereology, Ankara, Turkey
2 Hacettepe University, School of Medicine, Department of Pathology, Ankara, Turkey
3 Hacettepe University, School of Medicine, Department of Rheumatology, Ankara, Turkey



Key words: psoriasis, biologics, kaposi sarcoma, TNF antagonist
Citation: Tabak GH, Yalici-Armagan B, Sahin BA, Gokoz O, Kiraz S. Unusual Presentation of Kaposi’s Sarcoma During Adalimumab
Therapy: A Case Report. Dermatol Pract Concept. 2022;12(3):e2022071. DOI: https://doi.org/10.5826/dpc.1203a71
Accepted: October 11, 2021; Published: July 2022
Copyright: ©2022 Tabak et al. This is an open-access article distributed under the terms of the Creative Commons Attribution 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: Basak Yalici-Armagan, MD, Hacettepe University, School of Medicine, Department of Dermatology and
Venereology, 06100, Ankara, Turkey, Telephone: 00903123051704, Fax: 0 3123114340, E-mail: basakarmagan@gmail.com




Introduction                                                             hyperkeratotic crust and fibrin just under the fourth toe nail
                                                                         (Figure 1, A and B). Excisional biopsy was performed with
Kaposi sarcoma (KS) is a rare angioproliferative malignancy              provisional diagnoses of amelanotic malignant melanoma,
associated with Human Herpesvirus-8 (HHV-8) that in-                     PG and cutaneous squamous cell carcinoma. Histopatho-
volves skin and visceral organs. It typically manifests with             logical examination showed nodular lesion in the dermis
magenta-colored macules, papules, and nodules on the skin.               made up of atypical spindle shaped cells showing whorled
Herein, we present a patient who developed pyogenic granu-               structures and slit like spaces filled with erythrocytes. Im-
loma (PG)-like KS during adalimumab therapy for psoriatic                munohistochemical examination indicated HHV-8 positivity
arthritis.                                                               and confirmed the diagnosis of KS (Figure 1E). In detailed
                                                                         dermatological examination, purple-colored patches on the
                                                                         lateral aspects of both feet were noticed (Figure 1, C and D).
Case presentation
                                                                         HIV serology was negative. Adalimumab treatment was
A 48-year-old male applied to our dermatology outpatient                 ceased immediately and the patient was addressed to oncol-
clinic for a bleeding lesion on the right forth toe. The lesion          ogy department for remaining lesions.
occurred seven-months before and grew rapidly in last one-
month. He has been diagnosed with psoriatic arthritis 4 years
                                                                         Conclusions
ago and treated with adalimumab, a tumor necrosis factor-α
inhibitor (TNFi), for the last one year. Dermatological ex-              The potential of TNFis to increase the risk of malignancy
amination revealed a 1cm x1 cm diameter of reddish, ulcer-               including melanoma and non-melanoma skin cancer is still
ated, protruding hemorrhagic nodule partially covered with               controversial. Although iatrogenic KS has been described in



Research Letter | Dermatol Pract Concept. 2022;12(3):e2022071                                                                          1
    Figure 1. (A, B) Reddish-purplish, firm, easy-bleed-
    ing nodule covered with hyperkeratotic crust and
    fibrine just under the right forth toe nail. (C, D) Pur-
    ple-colored patches on the lateral sides of both feet.
    (E) Spindle cells, slit like spaces and erythrocytes,
    HHV8 positivity (inlet) are shown in histopatho-
    logic examination (H&E x200).




2   Research Letter | Dermatol Pract Concept. 2022;12(3):e2022071
organ transplant recipients receiving immunosuppressive            We present this rare case of PG-like KS that can be easily
therapy, there are only a few reports of KS during TNFi ther-      confused with other skin tumors in a patient under biologic
apy that were presenting as typically purplish-red patches         treatment.
and papulonodular lesions on the lower leg [1]. Classic KS            If characteristic purple-colored patches of KS on the lat-
is typically seen in older men (64–72 years) of Mediterra-         eral sides of feet were not overlooked on physical examina-
nean, Eastern European (Ashkenazi) Jewish, or South Amer-          tion in the current case, it could have been possible to include
ican origin whereas endemic KS is limited to sub-Saharan           KS in the clinical pre-diagnoses. Detailed whole body skin
Africa and is typically seen in young (25–40 years), black,        examination is crucial for dermatologists to provide correct
HIV-negative men. On the other hand, KS and psoriasis spec-        clinical diagnosis for early management. Further evidence is
ulated to share common pathogenesis particularly related           needed to clarify the relationship between biologics and ma-
with interleukin-6 cytokine pathway or the same human leu-         lignancy development.
kocyte antigen alleles. Although we cannot explain the exact
mechanism of this association whether it is a co-existence
                                                                   References
of KS and psoriasis or it is triggered by adalimumab, HIV
negativity and the early age of onset compared to classical         1. Ursini F, Naty S, Mazzei V, Spagnolo F, Grembiale RD.
KS suggested a relationship with adalimumab in the present             Kaposi’s sarcoma in a psoriatic arthritis patient treated with
                                                                       infliximab. Int Immunopharmacol. 2010;10(7):827-828.
case. Unlike reported KS cases induced by TNFi treatment,
                                                                       DOI: 10.1016/j.intimp.2010.04.016. PMID: 20433950.
PG-like presentation with classical purple-colored patches
                                                                    2. Megaly M, Boshra N. Pyogenic granuloma-like Kaposi’s sar-
was a remarkable finding of the current case. PG-like KS is a          coma. Lancet. 2022;399(10335):e38. DOI: 10.1016/S0140-
rare variant of KS that is difficult to diagnose clinically [2].       6736(15)00467-5. PMID: 26498707.




Research Letter | Dermatol Pract Concept. 2022;12(3):e2022071                                                                      3
