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CORRESPONDENCE
Year : 2021  |  Volume : 39  |  Issue : 1  |  Page : 63-64

A case of neutrophilic dermatosis clinically resembling actinomycosis


Department of Dermatology, Osaka City University Graduate School of Medicine, Osaka, Japan

Date of Submission15-Jul-2020
Date of Decision01-Sep-2020
Date of Acceptance29-Sep-2020
Date of Web Publication24-Mar-2021

Correspondence Address:
Dr. Koji Sugawara
Department of Dermatology, Osaka City University Graduate School of Medicine, 1-4-3 Asahimachi, Abeno-ku, Osaka 545-8585
Japan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ds.ds_49_20

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How to cite this article:
Yukawa K, Sugawara K, Fukunaga Y, Tsuruta D. A case of neutrophilic dermatosis clinically resembling actinomycosis. Dermatol Sin 2021;39:63-4

How to cite this URL:
Yukawa K, Sugawara K, Fukunaga Y, Tsuruta D. A case of neutrophilic dermatosis clinically resembling actinomycosis. Dermatol Sin [serial online] 2021 [cited 2021 May 7];39:63-4. Available from: https://www.dermsinica.org/text.asp?2021/39/1/63/311837



Dear Editor,

Neutrophilic dermatosis (ND) is a group of heterogeneous disorders characterized by severe inflammation primarily caused by neutrophils without infection. Several subtypes of ND have been suggested, including Sweet Syndrome, ND of the dorsal hands, and pyoderma gangrenosum.[1] Cutaneous findings vary and many ND patients have overlapping features.[1] We herein report a case of ND that clinically resembles actinomycosis. Informed consent was obtained from the patient.

A 64-year-old man was referred to our department with painful and indurated erythema of the left jaw [Figure 1]a. He had a medical history of cryptogenic organizing pneumonia and had been treated by the long-term oral administration of prednisolone (5 mg daily). The patient had already been treated by antibiotics due to a suspected cutaneous infection before referral to our department. A histopathological examination of erythema revealed diffuse inflammatory cell infiltration, including neutrophils. Although infection was not detected by a culture inspection, an apical abscess on the left back tooth was observed on a computed tomography scan. Therefore, we suspected actinomycosis of the face. His skin symptoms gradually improved with combination therapy using antibacterial agents (the infusion of ceftriaxone, followed by amoxicillin and minocycline) [Figure 1]b.
Figure 1: (a) Clinical appearance of an erythematous plaque on the left jaw in the first visit. (b) Clinical image of the improved eruption after a treatment with antibiotics.

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The patient was referred again to our department approximately 2 years later with painful skin eruptions on the face. A clinical examination revealed indurated erythema with scales, pustules, crusts, and ulcers on his left cheek [Figure 2]a and [Figure 2]b. Blood tests showed a high white blood cell count of 14,800/μL and the number of neutrophils was also elevated to 14,000/μL. His C-reactive protein level (18.84 mg/dL) was also increased. His hemoglobin level (9.9 g/dL) and platelet count (11.9 × 104/μL) were decreased. We initially suspected the recurrence of actinomycosis. A histopathological examination showed an abscess composed of various types of inflammatory cells; however, there were no obvious components of bacteria, fungi, or Mycobacterium tuberculosis detected by periodic acid–Schiff, Giemsa, Grocott's, or Ziehl–Neelsen histochemistry. A culture inspection using skin samples obtained by skin biopsy showed no signs of infection. Although he was treated by the infusion of ampicillin–sulbactam and cefepime dihydrochloride hydrate, skin symptoms worsened and also appeared on the left hand [Figure 2]c. A further histopathological examination of erythema on the left hand showed diffuse inflammatory cell infiltration, predominantly with neutrophils [Figure 2]d,[Figure 2]e,[Figure 2]f. However, there were no signs of infection. Based on the data obtained, we diagnosed the present case as ND. Bone marrow biopsy performed by a hematologist confirmed myelodysplastic syndromes (MDS). Therefore, the oral administration of prednisolone (20 mg daily) was initiated. Skin symptoms gradually improved. No other malignancy was detected by computed tomography throughout the body.
Figure 2: (a) Clinical appearance of an erythematous plaque with crusts and ulcers. (b) Clinical image of the erythematous plaque at higher magnification. (c) Clinical image of erythema on the left dorsal hand. (d and e) Histopathological images of the erythematous plaque on the left dorsal hand (H and E, original magnification [d] ×40, [e] ×200). The dense infiltration of inflammatory cells was observed throughout the dermis. (f) Dense infiltrates with a predominance of neutrophils were detected at a higher magnification (H and E, ×400).

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ND is characterized by neutrophilic hyperfunction without any infections.[2] Gammoudi et al. previously reported a rare case of recurrent ND of the face.[3] There have only been seven possible cases reported in the literature.[3] However, in contrast to the present case, none of these cases had associated conditions, such as myeloproliferative disorders or infectious diseases.[3] Since skin lesions may extend from the face to other sites or change to a typical presentation of Sweet's syndrome, the follow-up of patients is strongly recommended.[4] Our patient showed the gradual development of skin lesions to the hand. ND of the hands is associated with malignancy, such as lung cancer, laryngeal cancer, and MDS.[5],[6] The presence of skin symptoms in other lesions, except for the face, may be related to associated conditions, including MDS.

Actinomycosis is regarded as a rare type of granulomatous suppurative infection caused by anaerobic actinomyces that commonly occurs as cervicofacial, thoracic, abdominal, or pelvic lesions;[7] however, we did not find any signs of infection even though culture tests were performed several times.

Since cutaneous findings of ND were previously reported to vary,[1] it is also important to note that skin symptoms of ND may resemble infectious diseases, including actinomycosis, as observed in the present case.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Filosa A, Filosa G. Neutrophilic dermatoses: A broad spectrum of disease. G Ital Dermatol Venereol 2018;153:265-72.  Back to cited text no. 1
    
2.
Arita T, Nomiyama T, Asai J, Takenaka H, Fukuoka M, Katoh N. Unusual presentation of relapsing neutrophilic dermatosis limited to the face. J Dermatol 2014;41:661-2.  Back to cited text no. 2
    
3.
Gammoudi R, Ghariani Fetoui N, Ghariani N, Boussofara L, Sriha B, Denguezli M. Recurrent neutrophilic dermatosis of the face. Clin Exp Dermatol 2019;44:716-7.  Back to cited text no. 3
    
4.
Maki T, Yanaba K, Ishiuji Y, Umezawa Y, Asahina A, Nakagawa H. Recurrent neutrophilic dermatosis of the face: A report of two cases and review of the publish work. J Dermatol 2016;43:811-814.  Back to cited text no. 4
    
5.
Weenig RH, Bruce AJ, McEvoy MT, Gibson LE, Davis MD. Neutrophilic dermatosis of the hands: Four new cases and review of the literature. Int J Dermatol 2004;43:95-102.  Back to cited text no. 5
    
6.
Yang MY, Kim JM, Kim GW, Song M, Kim HS, Ko HC, et al. Neutrophilic dermatosis of the palms in association with myelodysplastic syndrome. Ann Dermatol 2017;29:495-7.  Back to cited text no. 6
    
7.
Cocuroccia B, Gubinelli E, Fazio M, Girolomoni G. Primary cutaneous actinomycosis of the forehead. J Eur Acad Dermatol Venereol 2003;17:331-3.  Back to cited text no. 7
    


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