Langerhans Cell Histiocytosis Presented as Generalized Vesiculo-Pustules in a Neonate-A Case Report

蘭格罕氏細胞組織球增生病在一新生兒以全身性水膿樣病灶表現－病例報告

李瑜芬(Yu-Fen Lee) ； 李定達(Ding-Dar Lee) ； 劉漢南(Han-Nan Liu)

《中華皮膚科醫學雜誌》 27卷3期 (2009/09) Pp. 176-180

https://doi.org/10.29784/DS.200909.0005

摘要

一位女嬰自出生後一週起，在頭上，臉上，軀幹及四肢出現了許多的水及膿，因為疑似疹病毒感染在滿月時住院並接受靜脈注射acyclovir。雖然單純疹病毒和水痘帶狀疹病毒的血清檢查與病毒培養的結果都是陰性，但是病灶的直接抗體螢光檢查呈現第一型單純疹病毒。治療之後，仍有許多新病灶出現，於皮膚科會診後進行Tzanck抹片以及皮膚切片檢查。皮膚切片的病理報告為蘭格罕氏細胞組織球增生病。之後的檢查結果包括：頭部X光檢查發現在左側頂骨處有擴張性的蝕骨病灶；胸部X光攝影顯示在二側肺野均有網狀結節狀浸潤增加的情形；長骨X光檢查與腹部超音波掃描皆無異常。總而言之，我們強調以新生兒的全身性水膿樣病灶所表現的蘭格罕氏細胞組織球增生病，在臨床上必須與疹病毒感染作鑑別診斷。

並列摘要

A one-month-old female baby had been noted to have multiple vesicles and pustules over the head, face, trunk and four extremities since she was one-week-old. Under the impression of disseminated herpes virus infection, intravenous acyclovir was given after admission. Serum herpes simplex virus and varicella zoster virus enzyme-linked immunosorbent assay test and virus culture were all fruitless, although herpes simplex virus-1 was vaguely identified in direct fl uorescent staining of lesion scrapings. Unfortunately, new lesions still continued popping out despite treatment. Dermatologist was then consulted, and Tzanck smear as well as skin biopsy were performed. The histopathological exam showed Langerhans cell histiocytosis. Skull x-ray film showed expansile osteolytic lesion over the left parietal area and the chest x-ray increased reticulonodular infiltration in both lung fields. There was no abnormal finding in the long bone survey or abdominal sonography. In summary, we report a case of neonatal Langerhans cell histiocytosis presenting as generalized vesiculo-pustules. It should be differentiated from herpes virus infection in the clinical setting.

並列關鍵字

Langerhans cell histiocytosis ； Vesiculo-pustules ； Tzanck smear