|Year : 2021 | Volume
| Issue : 4 | Page : 206-207
Sweet's syndrome occurred in a case with tuberculous cervical lymphadenitis
Chieh-Hsun Chen1, Ting-Ya Yang2, Feng-Jie Lai3
1 Department of Dermatology, Kaohsiung Medical University Hospital, Kaohsiung; Medical Education Center, Chi Mei Medical Center, Tainan, Taiwan
2 Medical Education Center; Department of Family Medicine, Chi Mei Medical Center, Tainan, Taiwan
3 Department of Dermatology, Chi Mei Medical Center; Center for General Education, Southern Taiwan University of Science and Technology, Tainan, Taiwan
|Date of Submission||04-Mar-2021|
|Date of Decision||12-Jul-2021|
|Date of Acceptance||31-Jul-2021|
|Date of Web Publication||29-Dec-2021|
Dr. Feng-Jie Lai
Department of Dermatology, Chi Mei Medical Center, No. 901, Zhonghua Road, Yongkang District, Tainan City 710
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Chen CH, Yang TY, Lai FJ. Sweet's syndrome occurred in a case with tuberculous cervical lymphadenitis. Dermatol Sin 2021;39:206-7
Sweet's syndrome (SS), also known as “acute febrile neutrophilic dermatosis,” is an uncommon disease characterized by cutaneous eruption of tender erythematous papules, plaques, or nodules, presenting diffuse infiltrates of predominantly neutrophils in histopathology. Infection, malignancies, and drugs are the often mentioned reasons, and SS is usually caused by preceding upper respiratory tract (e.g., viral and streptococcal) or gastrointestinal tract (e.g., Yersinia More Details) infection. However, Mycobacterium tuberculosis-associated SS is extremely rare.,,,,, Herein, we reported a case of tuberculous cervical lymphadenitis who developed SS lesions.
A 58-year-old male presented with right cervical granulomatous lymphadenopathy [Figure 1]a and M. tuberculosis was isolated from sputum culture. After 3-month course of antituberculous therapy (ATD), he developed coalescing papules and nodules with central ulceration at the right buttock [Figure 1]b. In the following 6 weeks, cutaneous eruption of multiple tender erythematous bumps with varying sizes was found in an asymmetric distribution on the face, neck, trunk, and upper extremities [Figure 1]c and [Figure 1]d, accompanied with headache, arthralgia, and myalgia. The bumps grew quickly in size, spreading into painful clusters up to an inch or so in diameter. Laboratory investigations revealed high total leukocyte count 17,400/μL (normal range: 3400–9100/μL) with 86.7% polymorphonuclear cells, elevated erythrocyte sedimentation rate 65 mm/h and C-reactive protein 131.9 mg/L (normal range: <5 mg/L). Biochemical examinations including renal profile and liver function test were within normal limits. Tests for tumor marker (carcinoembryonic antigen) and antinuclear antibody were unremarkable. Brain, chest, and abdomen computed tomography revealed no obvious internal malignancy. The diagnosis of SS was confirmed on skin biopsy [Figure 1]e. ATD was temporarily withdrew. Significant improvement was noticed in cutaneous lesions within 3 days after methylprednisolone administration (40 mg twice daily). However, headache and arthralgia persisted until colchicine (1 mg/day) was added on. The disease initially improved but flared up 1 month later after tapering the medication to prednisolone (5 mg three times daily). Accordingly, we initiated methylprednisolone pulse therapy (500 mg/day) for 3 days, shifting to methylprednisolone (40 mg every 8 h) for 10 days, and gradually tapered to prednisolone (5 mg/day) for 8 months. ATD regimens were simultaneously reintroduced for completion of tuberculous treatment. There was so far no recurrence.
|Figure 1: (a) Right cervical lymphadenopathy was found on chest computed tomography (shown by arrow), which was diagnosed as tuberculous cervical lymphadenitis with biopsy. (b) Coalescing papules and nodules with central ulceration were found at the right buttock. (c) Multiple tender erythematous nodules and plaques were located at the face, neck, and trunk in an asymmetric distribution. (d) “Pustular vasculitis of the dorsal hand,” which was presented as erythematous-based, purple pustules with central necrosis at the left dorsal hand. (e) The skin biopsy revealed neutrophilic dermatosis with leukocytoclasis and neutrophilic lobular panniculitis, which suggested Sweet's syndrome (H and E stain, ×300).|
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SS has been identified as a reactive dermatosis related to various factors and also been considered a cutaneous manifestation of underlying diseases. It is usually characterized by a constellation of symptoms and signs, including fever, painful erythematous papules or nodules, and diffuse infiltrates consisting predominantly of neutrophils in histopathology. There are three clinical settings to classify SS, which are classical (or idiopathic) SS, malignancy-associated SS, and drug-induced SS, respectively. Classical SS may be associated with infection, inflammatory bowel disease, or pregnancy while malignancy-associated SS is most often related to acute myelogenous leukemia, but solid-organ cancers were also reported. Drug-induced SS is most frequently observed in patients with administration of granulocyte-colony stimulating factor, but other agents have been reported.
In our case, we currently did not find any evidence of malignancy nor autoimmune disease. The possibility of relationship between SS and ATD could also be ruled out. Although we temporarily discontinued ATD since SS was confirmed, no recurrence was noted after ATD reintroduction. Moreover, several literatures,,,,, have reported disease resolution by immunosuppressive therapy without interruption of ATD, which strongly goes against the hypothesis of ATD-induced SS. Therefore, M. tuberculosis infection is the most suspect reason for our case. We thoroughly reviewed cases of M. tuberculosis-associated SS, and only a few were found.,,,,, These cases are summarized in [Supplementary Table 1]. We could find the variable onset time of SS ranging from 1 to 3 months, which is very different from other common pathogens, such as streptococcal or Yersinia infection (usually 1–3 weeks). However, there has been no literature mentioning about the cause of delayed onset, and it is hypothesized that an immune reaction to infection or certain cytokines/chemokines may induce SS through stimulating the inflammatory response that promotes neutrophil activation and infiltration. Further study is necessary to clarify the mechanisms that M. tuberculosis contributes to the development of SS.
For management of SS, all cases were in good response to systemic corticosteroids, which is “gold standard” for treatment.,,,,,, However, some cases relapsed after tapering or discontinuing the medication. Other first-line therapy, including potassium iodide and colchicine, may also induce rapid remission.,,, Besides, if SS is caused by medication, the disease frequently improves following withdrawal of the culprit drug.
In conclusion, we should recognize that M. tuberculosis infection may be one of the causes of SS.
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 identity, but anonymity cannot be guaranteed.
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Conflicts of interest
There are no conflicts of interest.
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