Infection DOI 10.1007/s15010-015-0814-6

CASE REPORT

Acne inversa complicated by Actinomyces neuii Jakob Nedomansky1 · Doris Weiss2 · Birgit Willinger3 · Stefanie Nickl1 · Christoph Steininger4 

Received: 2 May 2015 / Accepted: 18 June 2015 © Springer-Verlag Berlin Heidelberg 2015

Abstract  Introduction  Acne inversa (AI) is a chronic and recurrent inflammatory skin disease. It occurs in intertriginous areas of the skin and causes pain, drainage, malodor and scar formation. While supposedly caused by an autoimmune reaction, bacterial superinfection is a secondary event in the disease process. Methods  A unique case of a 43-year-old male patient suffering from a recurring AI lesion in the left axilla was retrospectively analysed. Results  A swab revealed Actinomyces neuii as the only agent growing in the lesion. The patient was then treated with Amoxicillin/Clavulanic Acid 3 × 1 g until he was cleared for surgical excision. The intraoperative swab was negative for A. neuii. Antibiotics were prescribed for another 4 weeks and the patient has remained relapse free for more than 12 months now. Conclusion  Primary cutaneous Actinomycosis is a rare entity and the combination of AI and Actinomycosis has never been reported before. Failure to detect superinfections of AI lesions with slow-growing pathogens like Actinomyces spp. might contribute to high recurrence rates

* Christoph Steininger [email protected] 1

Division of Plastic and Reconstructive Surgery, Department of Surgery, Medical University of Vienna, Vienna, Austria

2

Division of Immunology, Department of Dermatology, Allergy and Infectious Diseases, Medical University of Vienna, Vienna, Austria

3

Division of Clinical Microbiology, Department of Laboratory Medicine, Medical University of Vienna, Vienna, Austria

4

Division of Infectious Diseases, Department of Medicine 1, Medical University of Vienna, Vienna, Austria





after immunosuppressive therapy of AI. The present case underlines the potentially multifactorial pathogenesis of the disease and the importance of considering and treating potential infections before initiating immunosuppressive regimens for AI patients. Keywords  Acne inversa · Hidradenitis suppurativa · Soft tissue infection · Actinomycosis · Actinomyces neuii

Acne inversa (AI), also known as Hidradenitis suppurativa, is a chronic and recurrent inflammatory skin disease affecting 0.5–4.1 % of the population [1]. It occurs in intertriginous areas of the skin and typical clinical presentations are nodules, sinus tracts and abscesses causing pain, drainage, malodor and scar formation [2]. Although the complete pathogenesis of AI is still unknown, it is supposedly caused by an autoimmune reaction leading to occlusion and consequently inflammation of the pilosebaceous unit of the sebaceous glands [3]. Bacterial superinfection is considered a secondary event in the disease process [4]. A wide range of different aerobic and anaerobic bacteria has been detected in AI lesions before [5]. We report for the first time a case where AI was complicated by an isolate of Actinomyces spp. A 43-year-old male patient presented with a recurring, painfully swollen abscess in the left axilla (see Fig.  1), without general symptoms like fever or weight loss. According to the patient pus was draining at times. His medical history included obesity and chronic nicotine abuse (25 pack years). The clinical presentation lead us to the tentative diagnosis of Acne inversa, while possible differential diagnoses included Furunculosis, Lymphogranuloma venereum, Acne vulgaris, Crohn disease, Tuberculosis and Erysipelas. The lesion was incised and a swab for cultivation was taken. Actinomyces neuii was

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Fig. 1  Acne inversa lesion in the left axilla before the surgery

the only agent growing in the culture. Susceptibility testing revealed sensitivity of the agent to Amoxicillin/Clavulanic Acid, Meropenem and Vancomycin, while Clindamycin and Metronidazole were ineffective. The patient was treated with Amoxicillin/Clavulanic Acid 3 × 1 g. Over the course of several weeks the local condition improved significantly and the patient was cleared for surgical excision under ongoing antimicrobial therapy. Excision and primary closure of the AI lesion was then performed without any complications. Histopathological examination of the resected tissue confirmed the diagnosis of AI. The intraoperative swab was negative for A. neuii this time. The patient was discharged on day 8 after surgery. Antibiotics were prescribed for another 4 weeks and the patient has remained relapse free for more than 12 months now. Actinomyces spp. are slow-growing and facultatively anaerobic bacteria which are a natural commensal of the mucous membranes of the mouth, digestive and urinary tract. When it comes to impaired barrier function of the mucous membrane, however, it can lead to endogenous cervicofacial or intra-abdominal infections, called Actinomycosis [6]. Primary cutaneous Actinomycosis, like in this case, is a rare entity and the combination of Acne inversa and Actinomycosis has never been reported before. The incidence of Actinomycosis in patients with AI may be underestimated. Direct Gram staining is very insensitive with detection rates as low as 17–21 % [7]. Detection of pathognomonic sulfur granules on histological examination requires high quantity and quality of clinical samples [8]. Successful culture of these bacteria may be compromised by empiric antimicrobial pre-treatment. In this case, the preoperative swab obtained from the abscess was incubated on blood agar, chocolate agar

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and Sabouraud Dextrose Agar (Thermo Fisher-Oxoid, London, UK) at 37 °C in a 5 % CO2 atmosphere and on Brucella agar (Thermo Fisher-Oxoid, London, UK) under anaerobic conditions. After several days, cultures showed growth of Gram-positive rods, which were identified as A. neuii using matrix-assisted laser desorption ionisation time-of-flight mass spectrometry (MALDITOF MS, Bruker Daltonik, Bremen, Germany). Still, the same bacterial pathogen could not be cultivated with the same approach in the second, intraoperative swab. In our opinion, it is most likely that the preoperative antimicrobial treatment compromised detection of A. neuii by culture rather than eliminated all pathogens from the still extensive lesions. AI therapy is currently based on several modalities. Antibiotic long-term treatment, followed by surgical excision in severe cases, used to be the first-line therapy. Nowadays therapeutic options have been supplemented with immunosuppressive therapies such as TNF-α blockers. A recent review outlines high response rates in AI patients treated with TNF-α blockers (up to 90 % for Infliximab) but unfortunately also high recurrence rates after discontinuation of this therapy [9]. Two cases of cutaneous Actinomycosis as complication of TNF-α blocker therapy already have been previously reported [10]. Failure to detect superinfections of AI lesions with slow-growing pathogens like Actinomyces spp. might contribute to high recurrence rates after immunosuppressive therapy of AI. The present case underlines the potentially multifactorial pathogenesis of AI and the importance of considering and treating potential infections in AI before initiating immunosuppressive regimens. Conflict of interest  On behalf of all authors, the corresponding author states that there is no conflict of interest.

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Acne inversa complicated by Actinomyces neuii 7. Gomez-Garces JL, Burillo A, Gil Y, Saez-Nieto JA. Soft tissue infections caused by Actinomyces neuii, a rare pathogen. J Clin Microbiol. 2010;48(4):1508–9. 8. Bennhoff DF. Actinomycosis: diagnostic and therapeutic considerations and a review of 32 cases. Laryngoscope. 1984;94(9):1198–217. 9. Blok JL, van Hattem S, Jonkman MF, Horvath B. Systemic therapy with immunosuppressive agents and retinoids in

hidradenitis suppurativa: a systematic review. Br J Dermatol. 2013;168(2):243–52. 10. Breton AL, Lamblin G, Pariset C, Jullien D. Cutaneous actinomycosis associated with anti-TNF-alpha therapy: report of two cases. Dermatology. 2014;228(2):112–4.

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Acne inversa complicated by Actinomyces neuii.

Acne inversa (AI) is a chronic and recurrent inflammatory skin disease. It occurs in intertriginous areas of the skin and causes pain, drainage, malod...
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