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CORRESPONDENCE
Year : 2022  |  Volume : 67  |  Issue : 4  |  Page : 437-439
Extranodal NK-T-Cell lymphoma, nasal-type misdiagnosed as erythema multiforme with nasal fungal infection


1 From the Department of Dermatology, College of Medicine, Hallym University, Sacred Heart Hospital, Anyang-Si Gyeonggi-Do, South Korea
2 From the Department of Dermatology, College of Medicine, Hallym University, Sacred Heart Hospital, Anyang-Si, Gyeonggi-Do, South Korea

Date of Web Publication2-Nov-2022

Correspondence Address:
Soo Hyun Jeong
From the Department of Dermatology, College of Medicine, Hallym University, Sacred Heart Hospital, Anyang-Si, Gyeonggi-Do
South Korea
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.ijd_311_22

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How to cite this article:
Jeong SH, Oh DY, Park EJ, Kim KJ, Kim KH. Extranodal NK-T-Cell lymphoma, nasal-type misdiagnosed as erythema multiforme with nasal fungal infection. Indian J Dermatol 2022;67:437-9

How to cite this URL:
Jeong SH, Oh DY, Park EJ, Kim KJ, Kim KH. Extranodal NK-T-Cell lymphoma, nasal-type misdiagnosed as erythema multiforme with nasal fungal infection. Indian J Dermatol [serial online] 2022 [cited 2022 Dec 10];67:437-9. Available from: https://www.e-ijd.org/text.asp?2022/67/4/437/360326




Sir,

Extranodal NK-T-Cell lymphoma, nasal-type (ENKTCL-NT) is an aggressive lymphoma characterized by angiocentric and angiodestructive infiltrates of EBV positive malignant cells with an NK-cell or cytotoxic T-cell phenotype involvement.[1] The infiltrate involves tissues of the nasal cavity and may be seen in extranodal/extranasal sites including skin, soft tissue and gastrointestinal tract.[1]

A 73-year-old woman was referred to our dermatology department with erythematous patches on her whole body that appeared two months ago [Figure 1]a. We performed a punch biopsy of the trunk lesion for a differential diagnosis. Histopathologically, vacuolar degeneration of the basal layer and interface dermatitis with eosinophils were observed [Figure 1]b. We suspected erythema multiforme and treated the patient with systemic corticosteroids. Her skin lesions improved slightly. Systemic corticosteroids cannot be tapered because of the relapsing skin lesions. After 8 weeks, she complained of oral and nasal erosion, and a new bulla appeared on her arm [Figure 1]e. A biopsy of bulla with a direct immunofluorescence test was performed. Necrotic epidermis with lymphoid cell infiltration was observed, but the direct immunofluorescence test was negative. The patient's symptoms were not well controlled, and the systemic corticosteroids dose was increased. She complained of persistent nasal discomfort and was referred to the otorhinolaryngology department. A biopsy of her nasal septum identified diffuse interstitial atypical lymphoid cells with angiocentric and angiodestructive patterns [Figure 2]a and [Figure 2]b. Immunohistochemistries of CD3, CD7, CD56, granzyme B, and EBV in situ hybridization (ISH) were positive [Figure 2]c, [Figure 2]d, [Figure 2]e, confirming ENKTCL-NT diagnosis. The initial trunk and arm biopsy slides were reviewed, showing CD56-and EBV-positive lymphoid cells [Figure 1]c, [Figure 1]d and [Figure 2]c, [Figure 2]d. Erythematous patches on the trunk and bulla were clinical manifestations of ENKTCL-NT. Eight months after the diagnosis, new hyperpigmented patches developed in her extremities [Figure 2]f. They were also confirmed as ENKTCL-NT.
Figure 1: (a) Erythematous patches on trunk. (b) Trunk biopsy; vacuolar degeneration of the basal layer and interface dermatitis with eosinophils. H and E × 200. (c) Trunk biopsy; CD56 positive atypical cells. × 200. (d) Trunk biopsy; EBV ISH positive atypical cells. × 200. (e) Bulla on arm. (f) Arm biopsy; necrotic epidermis with lymphoid cell infiltration. H and E × 100. (g) Arm biopsy CD56 positive atypical cells. × 100. (h) Arm biopsy EBV ISH positive atypical cells. × 100

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Figure 2: (a) Whitish patches on nasal area in nasal endoscopy. (b) Nasal biopsy; atypical lymphoid cells with angiodestructive and angiocentric pattern. H and E × 400. (c) Nasal biopsy, CD56 positive atypical cells × 100. (d) Nasal biopsy, granzyme B positive atypical cells × 100. (e) Nasal biopsy, EBV ISH positive atypical cells × 100. (f) New hyperpigmented patch on extremities, which was confirmed as ENKTCL-NT

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The cutaneous ENKTCL-NT manifests in various ways. Subcutaneous nodules were most commonly observed (42%) in 24 primary cutaneous ENKTCL patients,[2] followed by cellulitis-like (33%) and erythematous to purpuric patches (25%). In 10 ENKTCL-NT patients,[3] ulcers were the most common (40%) skin manifestation, followed by cellulitis (30%), plaques and nodules (30%). Our patient's initial erythematous patches can be categorized as erythematous to purpuric patches, but bulla that occurred afterward and the subsequent hyperpigmented patches are rare skin manifestations. Hyperpigmented patches have not been reported as cutaneous involvement of ENKTCL-NT, and only two cases of bulla-type involvement have been reported.[4],[5] To date, no cases have been reported of a patient showing diverse cutaneous involvement patterns over time, such as in our case. Regarding nasal discomfort, antifungal agents were initially prescribed under suspicion of candidiasis in otorhinolaryngology department. The nasal lesion of ENKTCL-NT can be misdiagnosed as candidiasis due to systemic corticosteroids, widely used in dermatology; therefore, a biopsy must be considered. Detailed history-taking and appropriate multidisciplinary approaches are keys to the diagnosis and treatment of ENKTCL-NT. When a patient manifests with chronic and refractory cutaneous lesions and erosive nasal lesions, a biopsy must be performed.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
David E, Rosalie E, Misha R, George F, Adam I, Xiaowei W, et al. Lever's histopathology of the skin. 11th ed. Philadelphia: Wolters Kluwer; 2015. p. 1183.  Back to cited text no. 1
    
2.
Kim YJ, Won CH, Chang SE, Lee MW, Choi JH, Lee WJ. Expression of programmed death-1 in cutaneous extranodal natural killer/T-cell lymphoma and its effect on clinical findings and biological behaviour. J Eur Acad Dermatol Venereol 2017;31:821-7.  Back to cited text no. 2
    
3.
Choi YL, Park JH, Namkung JH, Lee JH, Yang JM, Lee ES. Extranodal NK/T-cell lymphoma with cutaneous involvement: 'nasal' vs. 'nasal-type' subgroups--a retrospective study of 18 patients. Br J Dermatol 2009;160:333-7.  Back to cited text no. 3
    
4.
Ma SH, Liu HN, Ho YH. Extranodal NK/T-cell lymphoma, nasal type, mimicking a bullous pyoderma gangrenosum. Australas J Dermatol 2021;62:e351-3.  Back to cited text no. 4
    
5.
Miyamoto T, Yoshino T, Takehisa T, Hagari Y, Mihara M. Cutaneous presentation of nasal/nasal type T/NK cell lymphoma: Clinicopathological findings of four cases. Br J Dermatol 1998;139:481-7.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2]



 

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