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Diagnosis and Treatment > Signs and Symptoms

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Hiroto Minamino The First Department of Medicine, Wakayama Medical University, 811-1, Kimiidera, Wakayama, 641-8509, Japan
Department of Diabetes and Endocrinology, Wakayama Red Cross Hospital, Wakayama, Japan

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Hidefumi Inaba The First Department of Medicine, Wakayama Medical University, 811-1, Kimiidera, Wakayama, 641-8509, Japan

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Hiroyuki Ariyasu The First Department of Medicine, Wakayama Medical University, 811-1, Kimiidera, Wakayama, 641-8509, Japan

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Hiroto Furuta The First Department of Medicine, Wakayama Medical University, 811-1, Kimiidera, Wakayama, 641-8509, Japan

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Masahiro Nishi The First Department of Medicine, Wakayama Medical University, 811-1, Kimiidera, Wakayama, 641-8509, Japan

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Takashi Yoshimasu Department of Dermatology, Wakayama Medical University, Wakayama, Japan

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Akinori Nishikawa Department of Hematology, Wakayama Medical University, Wakayama, Japan

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Masanori Nakanishi Department of Respiratory Medicine & Medical Oncology, Wakayama Medical University, Wakayama, Japan

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Shigeki Tsuchihashi Department of Otolaryngology, Wakayama Medical University, Wakayama, Japan

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Fumiyoshi Kojima Department of Human Pathology, Wakayama Medical University, Wakayama, Japan

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Shin-ichi Murata Department of Human Pathology, Wakayama Medical University, Wakayama, Japan

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Gen Inoue Department of Diabetes and Endocrinology, Wakayama Red Cross Hospital, Wakayama, Japan

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Takashi Akamizu The First Department of Medicine, Wakayama Medical University, 811-1, Kimiidera, Wakayama, 641-8509, Japan

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Summary

A 73-year-old man with Hashimoto's thyroiditis (HT) suffered from purpura on the lower legs. He was diagnosed with IgG4-related disease (IgG4-RD) with serum IgG4 elevation and dacryo-sialadenitis confirmed histologically. Serum Th2 and Treg cytokines, interleukin 7 (IL7), IL8 and Th2 chemokine levels were elevated, while skewed Th1 balance was seen in fluorescence-activated cell sorting (FACS). Therefore, preferential Th1 balance in HT appeared to be followed by IgG4-RD characterized with Th2 and Treg polarization. The commencement of steroid therapy dramatically exacerbated clinical manifestations including IgG4-RD-associated HT. The measurement of cytokine and chemokine levels as well as FACS analysis in the development of IgG4-RD seemed to be beneficial. In conclusion, an innovative association of HT, IgG4-RD and vasculitis was observed. This report also offers novel diagnostic and therapeutic approaches for IgG4-RD.

Learning points

  • Recently, a subtype of HT has been considered to be a thyroid manifestation of IgG4-RD, although the etiology of IgG4-RD is not established yet.

  • Immunologically a close association between HT and vasculitis was reported.

  • Leukocytoclastic vasculitis is a rare skin presentation of IgG4-RD.

  • In the current case, during the course of HT, IgG4-RD and leukocytoclastic vasculitis occurred; thus, innate immunity and acquired immunity seem to be involved in the development of IgG4-RD.

  • The measurement of cytokine and chemokines appeared to be beneficial in the development of IgG4-RD.

  • Remarkably, effectiveness of steroid therapy for HT suggested presence of IgG4-RD-associated HT. Therefore, this report highlights the pathogenesis of IgG4-RD and proposes novel therapeutic mechanisms. Clinicians should pay attention to the development of IgG4-RD and vasculitis during long course of HT.

Taxonomy:
Clinical Overview, Gland/Organ, Thyroid, Topic, Thyroid, Hormone, Thyroxine (T4), Triiodothyronine (T3), TSH, Condition/ Syndrome, Hashimoto’s disease, IgG4–related systemic disease, Leukocytoclastic vasculitis, Lymphadenopathy, Thyroiditis, Patient Demographics, Age, Adult, Geriatric, Gender, Male, Ethnicity, Asian - Japanese, Country of Treatment, Japan, Diagnosis and Treatment, Signs and Symptoms, Eyelid swelling, Salivary gland swelling, Skin pigmentation - spotty, Investigation, Antinuclear antibody, Biopsy, C3 complement, C4 complement, Chemokines, C-reactive protein, CT scan, Cytokines, FT4, Haematoxylin and eosin staining, Haemoglobin, Histopathology, IgG4/IgG ratio, Immunoglobulin G4, Skin biopsy, Thyroid antibodies, Thyroid ultrasonography, TSH, White blood cell differential count, X-ray, Medication, Glucocorticoids, Levothyroxine, Prednisolone, Related Disciplines, Dermatology, Publication Details, Case Report Type, Insight into disease pathogenesis or mechanism of therapy
DOI:
https://doi.org/10.1530/EDM-16-0004
Volume/Issue:
Volume 2016: Issue 1
Open access