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CASE REPORT
Year : 2019  |  Volume : 37  |  Issue : 1  |  Page : 50-52

Idiopathic lymphoplasmacellular mucositis of the vulva in a patient with partial interferon-γ receptor 1 deficiency


1 Department of Dermatology, Chang Gung Memorial Hospital, Taipei; Department of School of Medicine, College of Medicine, Chang Gung University, Taoyuan, Taiwan
2 Department of Pathology, Chang Gung Memorial Hospital, Taipei, Taiwan

Correspondence Address:
Ya-Hui Chuang
Department of Dermatology, Chang Gung Memorial Hospital, 199, Tung-Hwa North Road, Taipei 105
Taiwan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ds.ds_18_18

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We report a case of idiopathic lymphoplasmacellular mucositis (ILPM) of the vulva in a 48-year-old woman with partial interferon-γ receptor 1 (IFN-γR1) deficiency. The lesion had an unusual ulcerovegetative presentation. Remarkable response was observed with oral and topical steroids in the first 3 weeks. However, the lesion recurred after tapering oral steroids and continuous low-dose oral steroids were required to suppress recurrence. To the best of our knowledge, this is the first case report of ILPM in a patient with partial IFN-γR1 deficiency. ILPM should be included in the differential diagnosis of persistent vulvar ulcerovegetative lesions.


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