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Open access

Waralee Chatchomchaun, Yotsapon Thewjitcharoen, Karndumri Krittadhee, Veekij Veerasomboonsin, Soontaree Nakasatien, Sirinate Krittiyawong, Sriurai Porramatikul, Ekgaluck Wanathayanoroj, Auchai Kanchanapituk, Pairoj Junyangdikul and Thep Himathongkam

Summary

In this case report, we describe a 37-year-old male who presented with fever and tender neck mass. Neck ultrasonography revealed a mixed echogenic multiloculated solid-cystic lesion containing turbid fluid and occupying the right thyroid region. Thyroid function tests showed subclinical hyperthyroidism. The patient was initially diagnosed with thyroid abscess and he was subsequently treated with percutaneous aspiration and i.v. antibiotics; however, his clinical symptoms did not improve. Surgical treatment was then performed and a pathological examination revealed a ruptured epidermoid cyst with abscess formation. No thyroid tissue was identified in the specimen. The patient was discharged uneventfully. However, at the 3-month and 1-year follow-ups, the patient was discovered to have developed subclinical hypothyroidism. Neck ultrasonography revealed a normal thyroid gland. This report demonstrates a rare case of epidermoid cyst abscess in the cervical region, of which initial imaging and abnormal thyroid function tests led to the erroneous diagnosis of thyroid abscess.

Learning points:

  • Epidermoid cyst abscess at the cervical region can mimic thyroid abscess.
  • Neck ultrasonography cannot distinguish thyroid abscess from epidermoid cyst abscess.
  • Thyroid function may be altered due to the adjacent soft tissue inflammation.
Open access

Hiroto Minamino, Hidefumi Inaba, Hiroyuki Ariyasu, Hiroto Furuta, Masahiro Nishi, Takashi Yoshimasu, Akinori Nishikawa, Masanori Nakanishi, Shigeki Tsuchihashi, Fumiyoshi Kojima, Shin-ichi Murata, Gen Inoue and Takashi Akamizu

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.