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CORRESPONDENCE
Year : 2018  |  Volume : 63  |  Issue : 5  |  Page : 428-430
Sweet's syndrome associated with Talaromyces marneffei and Mycobacterium abscessus infection due to anti-interferon-gamma autoantibodies


Department of Dermatology, The First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi Zhuang Autonomous Region 530021, PR China

Date of Web Publication31-Aug-2018

Correspondence Address:
Donghua Liu
Department of Dermatology, The First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi Zhuang Autonomous Region 530021
PR China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.IJD_362_17

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How to cite this article:
Xu H, Liu D, He X, Zheng D, Deng Y. Sweet's syndrome associated with Talaromyces marneffei and Mycobacterium abscessus infection due to anti-interferon-gamma autoantibodies. Indian J Dermatol 2018;63:428-30

How to cite this URL:
Xu H, Liu D, He X, Zheng D, Deng Y. Sweet's syndrome associated with Talaromyces marneffei and Mycobacterium abscessus infection due to anti-interferon-gamma autoantibodies. Indian J Dermatol [serial online] 2018 [cited 2020 Jun 2];63:428-30. Available from: http://www.e-ijd.org/text.asp?2018/63/5/428/240103




Sir,

A 54-year-old Chinese male presented with a 2-month history of recurrent fever and 1-month history of multiple enlarged superficial lymph nodes. He was admitted in our hospital on December 9, 2016. The history of the patient was not remarkable except that he had consumed bamboo rats a year ago. Physical examination showed enlarged left cervical, bilateral retroauricular, supraclavicular, and inguinal lymph nodes. He subsequently developed skin lesions which showed painful erythematous papules, plaques, pustules, and edematous erythema over the face, dorsa of hands, and lower extremities [Figure 1]a,[Figure 1]b,[Figure 1]c. The skin lesions progressed into erythema, blisters, and pustules distributed over the face and purple-red plaques and pustules distributed over the hands and lower extremities on December 14, 2016. Relevant investigations were carried out; meanwhile, treatment with fluconazole (0.2 g daily for 2 weeks) and levofloxacin (0.2 g b.i.d. during hospitalization) was started. Subsequently, methylprednisolone 40 mg/day was added to the therapeutic regimen and tapered over 2 weeks along with thalidomide (50 mg t.d.s. for 1 week). The skin lesions resolved gradually; however, a new painful nodule appeared on left underjaw on December 23, 2016 [Figure 1]d. Amphotericin B 10 mg/day was given at an incremental dose of 10 mg/day until 50 mg/day. After that, the dose was maintained. At the same time, fluconazole was replaced with itraconazole 0.2 g b.i.d. With modification to the treatment regimen, the skin lesions improved gradually and the patient was better. However, recurrent fever and enlarging cervical lymph nodes developed with swelling and flush over the neck on February 10, 2017. The clinical symptoms improved when therapeutic regimen was modified (levofloxacin was replaced with clarithromycin 0.5 g/day).
Figure 1: Clinical manifestations. Painful erythematous plaques, papules, and edematous erythema with pustules within lesions appeared on (a) the face, (b) extensor of hands, and (c) lower extremities; (d) a newly painful nodule appeared under left underjaw

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Blood test showed leukocyte count 29.08 × 109/L, neutrophilic granulocyte percentage 80.8%, erythrocyte sedimentation rate 108 mm/h, and C-reactive protein 118.83 mg/L. Test for anti-HIV was negative, but anti-interferon gamma (IFN-γ) autoantibodies were positive. Biopsy of bone marrow revealed no abnormal features. Cultures of blood and sputum were negative for fungi and bacteria. Histopathologically, biopsy of skin lesion taken from the left side of the face showed papillary dermal edema and extensive infiltration of neutrophils into the dermis [Figure 2]a, the biopsy was done on December 15, 2016]. The histopathological manifestation was consistent with Sweet's syndrome (SS). Histopathological examination of lymph node taken from right groin revealed yeast-like organisms under microscope [Figure 2]b, the biopsy was done on December 16, 2016]. Biopsy of the nodular lesion on the left underjaw showed both yeast-like organisms and acid-fast rods under microscope [Figure 2]c and d, the biopsy was done on December 23, 2016]. The DNA capacity of Talaromyces marneffei in the enlarged lymph node was 23.44 copies/μl, but those could not be detected in the whole blood and the nodular lesion from left underjaw (data were provided by the research group of Cao Cunwei). This result supported the diagnosis of deep mycosis caused by T. marneffei. Subsequently, culture of material from left underjaw lesion was negative for T. marneffei but positive for mycobacteria [Figure 3]a-c]. The type of mycobacteria was identified as Mycobacterium abscessus by mass spectrometric technique and molecular identification.
Figure 2: Histopathology findings. (a) Papillary dermal edema, extensive infiltration of neutrophils into the dermis without leukocytoclastic vasculitis (H and E, ×100); (b and c) Yeast-like organisms with binary fission (arrows), (PAS, ×400); (d) ingestion of Mycobacterium cells (arrow) in multinucleated giant cell (Ziehl–Neelsen, ×1000)

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Figure 3: Culture of skin lesion. (a) Colonies on brain heart infusion media appeared smooth or rough, white or grayish; (b) Mycobacterium cells were Gram-positive (Gram, ×1000); (c) Mycobacterium cells appeared acid-fast rods. (Ziehl–Neelsen, ×1000)

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The patient was diagnosed as SS with T. marneffei and M. abscessus infections due to anti-IFN-γ autoantibodies. According to our practice, low-dose corticosteroid treatment for a short time was good at reducing the aseptic inflammatory reaction in SS caused by the pathogens. Thalidomide was used in the treatment to reduce the dose of corticosteroid gradually. Then, the effect of antimicrobial agents produced excellent results, as the clinical symptoms of our patient improved gradually. After hospital discharge, he remained well with the administration of long-term antimicrobial therapy at the follow-up so far.

To the best of our knowledge, similar cases were rarely reported in the literatures.[1],[2] Moreover, this was the first reported case of a patient developed SS associated with T. marneffei and M. abscessus infection due to anti-IFN-γ autoantibodies.

Chan et al.[1] reported a 57-year-old Chinese male with anti-IFN-γ autoantibodies positivity developed SS with T. marneffei infection. The hallmark edema in SS was not seen in that case but was present in our case. SS was described as a kind of reactive dermatosis in patients with nontubercular mycobacterial infection.[3],[4] Though neutrophilic infiltration was present in the dermis in the case reported by Liu et al.[3] but dermal edema was absent. The phenomenon of absent edema in histopathological examination of SS in these reported cases was interesting. We speculate that it might be associated with varied virulence of pathogens and varied immune status of patients. There was limited knowledge to clarify this interesting phenomenon, especially in patients with deficiency of anti-IFN-γ autoantibodies.

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 name and initial will not be published, and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Acknowledgment

This work was supported by the National Nature Science Foundation of China (grant number 81360238). We acknowledge the detection of DNA capacity of T. marneffei in samples of our patient conducted by the research group of Cao Cunwei.

Financial support and sponsorship

This work was supported by the National Nature Science Foundation of China (grant number 81360238).

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Chan JF, Trendell-Smith NJ, Chan JC, Hung IF, Tang BS, Cheng VC, et al. Reactive and infective dermatoses associated with adult-onset immunodeficiency due to anti-interferon-gamma autoantibody: Sweet's syndrome and beyond. Dermatology 2013;226:157-66.  Back to cited text no. 1
    
2.
Pruetpongpun N, Khawcharoenporn T, Damronglerd P, Suthiwartnarueput W, Apisarnthanarak A, Rujanavej S, et al. Disseminated Talaromyces marneffei and Mycobacterium abscessus in a patient with anti-interferon-γ autoantibodies. Open Forum Infect Dis 2016;3:ofw093.  Back to cited text no. 2
    
3.
Liu TT, Weng SW, Wang MC, Huang WT. Nontuberculous mycobacterial infection with concurrent IgG4-related lymphadenopathy. APMIS 2016;124:216-20.  Back to cited text no. 3
    
4.
Mahaisavariya P, Chaiprasert A, Manonukul J, Khemngern S. Scrofuloderma and Sweet's syndrome. Int J Dermatol 2002;41:28-31.  Back to cited text no. 4
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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