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一种慢性致残性鼻畸形,诊断为皮肤黏膜利什曼病,延误诊断。

A chronic mutilating rhinopathy with a delayed diagnosis of mucocutaneous leishmaniasis.

机构信息

Department of Dermatology, Venereology & Leprosy, Kottayam Medical College, Kottayam, India.

出版信息

Int J Dermatol. 2010 Apr;49(4):426-9. doi: 10.1111/j.1365-4632.2010.04361.x.

DOI:10.1111/j.1365-4632.2010.04361.x
PMID:20465699
Abstract

BACKGROUND

Mucocutaneous leishmaniasis is a granulomatous disease clinically characterized by ulcerated skin and mucosal lesions. Mucocutaneous leishmaniasis is very rare in India and to our knowledge, only two cases have been reported, and this is the first case of mucocutaneous leishmaniasis presenting with mutilating rhinopathy reported from the Indian subcontinent.

CASE REPORT

A 64-year-old man presented with a destructive ulceration of the central face of 23 years' duration, who was diagnosed to have mucocutaneous leishmaniasis, and showed dramatic response to intramuscular injections of sodium stibogluconate.

RESULTS

Histopathologic examination of skin biopsy revealed a granulomatous infiltrate with the presence of leishmania donovani (LD) bodies. The clinical picture, plus the pathologic findings, and the response to sodium stibogluconate confirmed mucocutaneous leishmaniasis.

CONCLUSION

Mucocutaneous leishmaniasis is a rare disease in the Indian subcontinent, and clinicians from this region should have a high index of suspicion on encountering mucocutaneous ulcerative lesions.

摘要

背景

黏膜皮肤利什曼病是一种临床特征为溃疡性皮肤和黏膜损伤的肉芽肿性疾病。黏膜皮肤利什曼病在印度非常罕见,据我们所知,仅报告过两例,而这是首例来自印度次大陆的具有破坏性鼻畸形的黏膜皮肤利什曼病病例。

病例报告

一名 64 岁男性,因中央面部进行性破坏性溃疡就诊,病程 23 年,被诊断为黏膜皮肤利什曼病,对肌肉内注射葡萄糖酸锑钠有明显反应。

结果

皮肤活检的组织病理学检查显示存在利什曼原虫(LD)体的肉芽肿性浸润。临床图片加上病理发现以及对葡萄糖酸锑钠的反应证实了黏膜皮肤利什曼病的诊断。

结论

黏膜皮肤利什曼病在印度次大陆较为罕见,来自该地区的临床医生在遇到黏膜溃疡性病变时应高度怀疑该病。

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