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1例由类鼻疽引起的Sweet综合征罕见病例。

A rare case of Sweet syndrome secondary to melioidosis.

作者信息

Vithoosan Sahathevan, Thanushah Balendran, Piranavan Paramarajan, Gamlaksha Dayal, Karunatilake Harindra, Jayanaga Ananda

机构信息

National Hospital of Sri Lanka, Colombo, Sri Lanka.

Internal Medicine, Saint Vincent Hospital, Worcester, MA, 01608, UK.

出版信息

BMC Dermatol. 2019 Dec 2;19(1):16. doi: 10.1186/s12895-019-0096-2.

Abstract

BACKGROUND

Melioidosis is an emerging infection in South Asia caused by Burkholderia pseudomallei with various clinical presentations that include pneumonia, bacteraemia, arthritis, and deep-seated abscesses. Various cutaneous manifestations have been described in association with melioidosis. However Sweet Syndrome secondary to melioidosis has not been reported in the literature. Herein we describe the first case of Sweet syndrome secondary to melioidosis.

CASE PRESENTATION

A 53-year-old previously healthy Sri Lankan female presented with high-grade fever, painful oral ulcers, odynophagia and multiple bilateral cervical lymphadenopathies for 1 month. She also had a loss of appetite and weight. She had oral ulcers and bilateral blepharitis. Dermatological examination revealed multiple tender papules with a mamillated appearance and targetoid lesions with a yellowish centre over the face, upper trunk and upper limbs. She also had multiple tender subcutaneous nodules over the extensor aspect of upper limbs. Her inflammatory markers were significantly elevated. Aspirate from a submental lymph node abscess revealed the growth of Burkholderia pseudomallei. Melioidosis antibody titer was > 10,240. The histology of the skin lesions of the face and left forearm showed a prominent neutrophilic infiltrate in the dermis and the morphological features were in favour of Sweet syndrome with panniculitis. She was started on intravenous meropenem 2 g daily and showed rapid clinical improvement with the disappearance of skin lesions as well as a reduction in inflammatory markers.

CONCLUSION

Sweet syndrome is an uncommon inflammatory disorder known to be associated with upper respiratory tract and gastrointestinal infections, malignancies and the use of certain drugs. Melioidosis is an emerging infection with various cutaneous manifestations. This is the first case of melioidosis causing the secondary sweet syndrome. It emphasizes the importance of considering melioidosis as a potential aetiology in patients with Sweet syndrome.

摘要

背景

类鼻疽是由类鼻疽伯克霍尔德菌引起的一种在南亚地区新出现的感染性疾病,临床表现多样,包括肺炎、菌血症、关节炎和深部脓肿。与类鼻疽相关的各种皮肤表现已有报道。然而,文献中尚未报道过类鼻疽继发Sweet综合征的病例。在此,我们报告首例类鼻疽继发Sweet综合征的病例。

病例介绍

一名53岁、既往健康的斯里兰卡女性,出现高热、疼痛性口腔溃疡、吞咽困难和双侧多发颈部淋巴结肿大1个月。她还伴有食欲减退和体重下降。她有口腔溃疡和双侧睑缘炎。皮肤科检查发现面部、上躯干和上肢有多个质地柔软的乳头状丘疹、中央呈淡黄色的靶形皮损。上肢伸侧也有多个质地柔软的皮下结节。她的炎症指标显著升高。颏下淋巴结脓肿穿刺抽吸物培养出类鼻疽伯克霍尔德菌。类鼻疽抗体滴度>10240。面部和左前臂皮肤病变的组织学检查显示真皮内有显著的中性粒细胞浸润,形态学特征符合伴脂膜炎的Sweet综合征。开始每天静脉滴注美罗培南2g治疗后,患者临床症状迅速改善,皮肤病变消失,炎症指标下降。

结论

Sweet综合征是一种罕见的炎症性疾病,已知与上呼吸道和胃肠道感染、恶性肿瘤以及某些药物的使用有关。类鼻疽是一种新出现的感染性疾病,有多种皮肤表现。这是首例类鼻疽引起继发Sweet综合征的病例。它强调了在Sweet综合征患者中考虑类鼻疽作为潜在病因的重要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c34d/6886164/5632ffdfc153/12895_2019_96_Fig1_HTML.jpg

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