A case of IgG4-related disease with severe hypereosinophilia

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  • YAMAMOTO Kana
    Department of Pathology, Noguchi Thyroid Hospital and Foundation
  • MARUTA Junko
    Department of Pathology, Noguchi Thyroid Hospital and Foundation
  • ITO Yukiko
    Department of Pathology, Noguchi Thyroid Hospital and Foundation
  • YOKOYAMA Shigeo
    Department of Pathology, Noguchi Thyroid Hospital and Foundation

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Other Title
  • 高度の好酸球増多症を伴うIgG4関連疾患の1例
  • コウド ノ コウサンキュウ ゾウ タショウ オ トモナウ IgG4 カンレン シッカン ノ 1レイ

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Abstract

<p>We report a case of IgG4-related disease (IgG4-RD) with severe hypereosinophilia. A woman in her 70s with suspected IgG4-RD noticed swellings on both submandibular regions. She was referred to our hospital for further examination. Laboratory data revealed a high level of serum IgG4 (3,420 mg/dL) and leukocytosis (115.2 × 109/L), but neutrophils and eosinophils could not be calculated on an automated hematology analyzer. By visual inspection, we found that 96.9% of the total leukocytes were eosinophils, many of which showed abnormal nuclear lobulations and decreased counts of intracytoplasmic granules. She underwent a surgical biopsy of the submandibular gland and supraclavicular lymph nodes, and was eventually diagnosed as having IgG4-RD (definite) according to the comprehensive diagnostic criteria of IgG4-RD. In the imprinted smears of lymph node specimens subjected to May–Giemsa staining, eosinophils were easily recognized together with numerous plasma cells and lymphocytes. However, in the imprinted smears of lymph node subjected to Papanicolaou staining, eosinophils were not distinguishable from neutrophils because the acidophilic granules were not stained. Every additional test for determining the cause of hypereosinophilia showed negative results. Thereafter, steroid administration for IgG4-RD treatment improved her symptoms and laboratory data, including hypereosinophilia. On the basis of these findings, hypereosinophilia was judged to be a secondary response associated with IgG4-RD. On a practical level of medical technologists, it should be recognized that automated blood cell analyzers may not distinguish between neutrophils and eosinophils in patients with severe hypereosinophilia. Thus, for observing cytological smears with suspected eosinophilic infiltration, May–Giemsa staining, together with Papanicolaou staining, is advisable.</p>

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