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Correspondence
Association of systemic characteristics and histological variations in a case study of adult-onset asthma and periocular xanthogranuloma
  1. Toshinobu Kubota1,
  2. Suzuko Moritani2,
  3. Shu Ichihara2,
  4. Hiroko Terasaki3
  1. 1Department of Ophthalmology, National Hospital Organization, Nagoya Medical Center, Nagoya, Japan
  2. 2Department of Pathology, National Hospital Organization, Nagoya Medical Center, Nagoya, Japan
  3. 3Department of Ophthalmology, Nagoya University Medical School, Nagoya, Japan
  1. Correspondence to Dr Toshinobu Kubota, Department of Ophthalmology, National Hospital Organization, Nagoya Medical Center, 4-1-1, Sannomaru, Naka-ku, Nagoya-shi, Aichi-ken 460-0001, Japan; ganiky{at}nnh.hosp.go.jp

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Introduction

Ocular adnexal histiocytic disorders, including adult-onset asthma and periocular xanthogranuloma (AAPOX), necrobiotic xanthogranuloma and Erdheim-Chester disease, have common histological characteristics, such as the presence of foamy histiocytes (xanthoma cells) and Touton giant cells.1–3 Additionally, AAPOX presents lymphoid aggregation with prominent T cells and other histological variations with prominent B cells and plasma cells.2–5 AAPOX is also associated with unique systemic disorders, including adult-onset asthma, systemic lymphadenopathy, salivary gland enlargement and elevated serum levels of IgG.1–6 The causal relationships between the systemic characteristics and histological variations have not been determined.

To investigate the relationship between the systemic and histological associations of AAPOX in more detail, we studied four patients with AAPOX. We compared the clinicopathological features of AAPOX with those of ocular adnexal IgG4-related lymphoplasmacytic infiltrative disorder (IgG4-related ophthalmic disease).7

Case reports

Procedures used in this study conformed to the tenets of the Declaration of Helsinki, and were approved by the Ethics Committee at Nagoya Medical Center, Nagoya, Japan.

Case 1

A 38-year-old man presented with bilateral swollen eyelids that had gradually worsened over 9 years (figure 1). He had had autoimmune pancreatitis 10 years earlier and asthma 4 years earlier. He had received oral prednisolone, and 5 mg of oral prednisolone was given every other day before our initial examination. The laboratory data showed that the serum level of IgG4 was 525 mg/dL (reference range, 4.8–105 mg/dL). CT imaging showed that the lesions had bilateral periocular and …

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