ABSTRACT
BACKGROUND/AIM: IgG4-related disease (IgG4RD) is a rare autoimmune proinflammatory condition that mimics other cancers and has unique pathological findings. The effects of radiotherapy in patients with IgG4RD remain unknown. CASE REPORT: A male patient in his seventies who received radiotherapy (68 Gy/39 fr) for bladder cancer 5 months prior, presented to our hospital with fatigue and swelling in both legs. The patient had a history of IgG4-related sclerosing cholangitis, a subtype of IgG4RD. Leg edema gradually worsened despite treatment with a diuretic agent. Computed tomography showed hyperdense soft-tissue lesions in the irradiated area. The serum level of IgG4 increased to 1,380 mg/dl. One month after administration of a corticosteroid (10 mg per day) as an ex juvantibus treatment for IgG4RD, leg edema disappeared. Soft-tissue lesions in the irradiated area decreased in size. The adverse event was ultimately diagnosed as the recurrence of IgG4RD in the irradiated area. To the best of our knowledge, this is the first case report of an adverse event of radiotherapy for a patient with IgG4RD. CONCLUSION: We experienced a unique adverse event of radiotherapy in a patient with IgG4RD. Caution is advised on radiotherapy administration in patients with IgG4RD.
Subject(s)
Autoimmune Diseases , Immunoglobulin G4-Related Disease , Humans , Male , Immunoglobulin G4-Related Disease/complications , Immunoglobulin G4-Related Disease/diagnosis , Immunoglobulin G4-Related Disease/radiotherapy , Autoimmune Diseases/diagnosis , Tomography, X-Ray Computed , Immunoglobulin G , EdemaABSTRACT
We report the case of a 20-year-old woman with intracranial IgG4-related disease, initially misdiagnosed as a meningioma on biopsy and treated with radiosurgery as it was in an eloquent location and not resectable. Her intracranial IgG4 disease had a near-complete response to radiosurgery and is still controlled six years later, so this case represents what we believe to be the first reported use of radiotherapy in the treatment of intracranial IgG4-related disease.
Subject(s)
Brain Diseases/radiotherapy , Immunoglobulin G4-Related Disease/radiotherapy , Radiosurgery/methods , Brain Diseases/diagnosis , Diagnosis, Differential , Female , Humans , Immunoglobulin G4-Related Disease/diagnosis , Magnetic Resonance Imaging , Young AdultABSTRACT
We describe the use of radiotherapy for parotid IgG4-related disease (IgG4-RD), initially misdiagnosed as Kimura's disease, with sustained good partial response in a 37-year-old male. To the best of our knowledge, this is the first reported case of radiation for extra-orbital IgG4-RD, albeit inadvertently.