Case Reports
Multiple milia formation following refractory bullous pemphigoid
Masato Ishikawa*, Tatuhiko Mori, Tomoko Hiraiwa, Takenobu Ohashi, Nobuyuki Kikuchi, Yuka Hanami, and Toshiyuki Yamamoto
Corresponding Author: Dr. Masato Ishikawa, Department of Dermatology, Fukushima Medical University, Hikarigaoka 1, Fukushima 960-1295, Japan E-mail:ishimasa@fmu.ac.jp
Received: May 11, 2015; Accepted: May 25, 2015; Published: Aug 28, 2015;
Citation: Ishikawa et al (2015) Multiple milia formation following bullous pemphigoid. Dermatol Clin Res, 1(2): 34-36
Copyrights: ©2015 Ishikawa et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
 

Formation of millium after bullous pemphigoid (BP) is not so rare, but the cases presenting numerous number of milia like our case is rare. We report a 70-year-old male who developed a remarkable number a millium after BP improved.  The number of millium in our case might be more than previous reports.  One of the cause of a remarkable number of millium appeared in our case might be his chronic clinical course.  In his 6 months of hospitalization, erythema, blister and erosion relapsed many times over.  Every time erosions cured, keratin might be entrapped, and it might lead a remarkable number of milia.

CASE REPORT

A 70-year-old male was referred to our department, complaining of generalized blisters and erosions.  He had been suspected of bullous pemphigoid (BP) and treated with oral prednisolone (PSL) (30 mg/day), however, his eruptions got worsened.  Physical examination revealed erythema and a number of scattered, tense blisters and erosions on the trunk and extremities (Figure 1).  Laboratory data by ELISA showed elevated anti-BP180 antibody titer (2040; normal < 9). A biopsy specimen taken from blister showed subepidermal bulla, and inflammatory cell infiltration, mainly composed of eosinophils (Figure 2).  Direct immunofluorescence showed linear deposit of IgG in epidermal side of dermo-epidermal junction (Figure 3).  We treated him with immunosuppressant (ciclosporin and methotrexate), and plasma exchange in addition to oral PSL.  Erosions and blisters were slowly cured.  After erosions were cured, a numbers of milium-like eruptions that was more than 100 appeared scattered on trunk and extremities (Figure 4, 5). A biopsy specimen showed a cystic nodule with epithelial wall. The border of nodules was clear, and lumen of the cyst was filled with keratin (Figure 6).

 

DISCUSSION

Milium is a common skin cyst which is formed at the base of a hair follicle or sweat gland, and could be categorized as primary or secondary. Primary milia are formed from entrapped keratin, and are usually found on the faces. Secondary milium develops after an injury, burn, skin graft, or bullous diseases such as BP [1]. One of the treatment for milium is to puncture it and pressing out the keratin inside the milium.

Formation of millium after BP is not so rare, but the cases presenting numerous number of milia like our case is rare. In a recent literature, Tsuruta et al. reported multiple large milia appeared on BP patients [2]. In our case, remarkable number of millium appeared, and it might be more than previous reports. One of the cause of remarkable number of millium appeared in our case might be his chronic clinical course.  In his 6 months of hospitalization, erythema, blister and erosion relapsed many times over.  Every time erosions cured, keratin might be entrapped, and it might lead remarkable number of milia.  However there may be other causes of multiple milia formation, and further studies are necessary.

REFERENCES

1. Hisa T, Goto Y, Taniguchi S, Nakanishi T, Kakudo K, Takigawa M (1996) Post-bullous milia. Australas J Dermatol 37: 153-154.

2. Tsuruta D, Brzezinski P, Koga H, Ohata C, Furumura M, Hashimoto T (2013) Bullous pemphigoid with prominent milium formation.Acta Dermatovenerol Croat 21: 35-38.