Rituximab: a novel treatment for refractory Riedel’s thyroiditis

Hunt, Leanne; Harrison, Barney; Bull, Matthew; Stephenson, Tim; Allahabadia, Amit

doi:10.1530/edm-17-0132

Cited by 10 publications

(15 citation statements)

References 23 publications

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“…Continued monitoring can be accomplished using symptomatic and radiologic assessment. Similar to the cases described by Soh et al (1) and Hunt et al (9), our patient had a normal baseline IgG4 level and IgG4 could not be used as a biologic marker for improvement.…”

Section: Discussionsupporting

confidence: 86%

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Successful Use of Rituximab in a Case of Riedel Thyroiditis Resistant to Treatment with Prednisone and Tamoxifen

Mammen¹,

Gordon²

2019

AACE Clinical Case Reports

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Objective: Riedel thyroiditis (RT) is a rare disorder with high morbidity and limited treatment options. We describe a case resistant to conventional treatment with corticosteroids and tamoxifen, which subsequently responded to rituximab. Methods: Surgical pathology with hematoxylin and eosin staining was initially performed to confirm diagnosis, followed by IgG4 immunostaining and IgG4 serology in the setting of refractory RT, given its association with IgG4-related disease. Response to treatment was monitored subjectively as well as objectively with serial computed tomography scans. Results: A 51-year-old female with history of Hashimoto thyroiditis presented with compressive neck symptoms. Imaging was suggestive of a multinodular goiter with a large right thyroid nodule. Total thyroidectomy was planned, however intraoperative findings of a densely adherent thyroid with disruption of resection planes led to early termination of surgery. Biopsies obtained during surgery showed benign thyroid tissue with chronic lymphocytic thyroiditis, dense fibrous scar tissue, and benign lymph nodes, confirming the diagnosis of RT. The patient had minimal symptomatic improvement with chronic prednisone as high as 60 mg daily with tamoxifen at 30 mg twice a day. She subsequently received 4 doses of intravenous rituximab at 375 mg/m 2 every 3 weeks, resulting in significant subjective improvement of her compressive symptoms as well as an objective decrease in size of the thyroid mass as seen on a subsequent computed tomography scan. Conclusion: Evidence regarding etiology and management of RT is limited. We present a case of refractory RT treated with rituximab with resultant symptomatic improvement, thus providing further evidence for use of rituximab in resistant cases. (AACE Clinical Case Rep. 2019;5:e218-e221) Abbreviations: IgG4-RD = IgG4-related disease; RT = Riedel thyroiditis

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Section: Discussionsupporting

confidence: 86%

“…In 2018, Hunt et al (9) reported a second case of refractory RT successfully treated with rituximab. This patient received 3 monthly infusions of 1,000 mg of rituximab resulting in symptomatic and radiologic improvement that persisted at 30 months post treatment.…”

Section: Discussionmentioning

confidence: 99%

Successful Use of Rituximab in a Case of Riedel Thyroiditis Resistant to Treatment with Prednisone and Tamoxifen

Mammen¹,

Gordon²

2019

AACE Clinical Case Reports

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“…The optimal duration and doses of these agents for maintenance therapy have not been evaluated in detail. In our cases, RT responded well to GC and TMX, and rituximab can be saved for more aggressive and refractory disease [21,22].…”

Section: Discussionmentioning

confidence: 51%

Long-Term Outcomes of Tamoxifen Citrate Therapy and Histo- and Immunopathological Properties in Riedel Thyroiditis

et al. 2020

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Background: Riedel thyroiditis (RT) is a rare form of thyroiditis; thus, data about the disease course and treatment options are limited. Therefore, we aimed to assess the clinical, serological, radiological, and histopathological features, as well as short- and long-term follow-up of RT patients under glucocorticoid (GC) and tamoxifen citrate (TMX). Parameters related to IgG4-related diseases (IgG4-RD) were also investigated. Methods: Eight patients with RT diagnosed between 2000 and 2019 were enrolled. Data were collected in a retrospective and prospective manner. The diagnosis was confirmed with histopathological features in all patients. Results of the treatment with GCs on short- to mid-term, followed by TMX in the long term, were evaluated. Results: The mean age at diagnosis was 40.5 ± 6.8 years; female predominance was observed (F/M:7/1). Parameters related to IgG4-RD, like increase in IgG4 serum levels, total plasmablast counts, and IgG4+ plasmablasts, were negative in most of our patients in both active and inactive states of the disease. Likewise, an increased ratio of IgG4/IgG-positive plasma cells >40% could only be observed in 2 cases. GCs followed by TMX were given to the patients with an over-all median follow-up time of 67 (8–216) months. All the patients considerably improved clinically and had a reduction in the size of the mass lesion on GCs, followed by TMX therapy. None of the patients had a recurrence under TMX therapy for a median period of 18.5 (7–96) months. Conclusion: Even though RT is suggested to be a member of IgG4-RD, serologic or histological evidence of IgG4 elevation or positivity is only useful for diagnosis and follow-up of RT. The diagnosis should be based on clinical and radiological evidence and confirmed by histopathology. GCs are effective for initial treatment, and TMX is a successful and safe therapeutic option for long-term maintenance therapy.

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“…However, this drug was quickly discontinued from the patient's line-up because it was poorly tolerated. Mycophenolate mofetil is associated with several serious side effects, including pancytopenia and renal failure [ 17 ]. A drug gaining popularity in the treatment of refractory Riedel's thyroiditis is Rituximab, a monoclonal antibody against the CD20 protein found mainly on B lymphocytes.…”

Section: Discussionmentioning

confidence: 99%

Riedel’s Thyroiditis: Pitfalls in Diagnosis and Subsequent Complications

Pandev

Khan

Ratheesh

2023

Case Reports in Endocrinology

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Riedel’s thyroiditis is a rare disease of chronic inflammation with fibrotic infiltration of the thyroid gland and its surrounding vital structures. Due to its low incidence, there are often delays in diagnosis as it is commonly mistaken for other thyroid diseases. We report the case of a 34-year-old female patient who presented with a firm, enlarged mass in the neck, compression symptoms, and hypothyroidism. Lab tests showed elevated A-TG (thyroglobulin antibodies) and A-TPO (thyroid peroxidase antibodies) levels. Based on the disease presentation and supporting lab findings, the patient was misdiagnosed with Hashimoto’s thyroiditis and treated accordingly. Yet the patient’s symptoms grew progressively worse. She was discovered to have severe tracheal compression and bilateral RLN (recurrent laryngeal nerve) palsy. Tracheotomy became a necessary surgical intervention after the development of respiratory failure, but this procedure was complicated by the development of an intraoperative pneumothorax. After an open biopsy, histology revealed Riedel’s thyroiditis. A new treatment was introduced, with which the patient’s condition improved. However, she continued to suffer from the open tracheocutaneous fistula left by the tracheostomy, which adversely affected her everyday life. A follow-up operation was performed to close the fistula. In this case report, we discuss the consequences of misdiagnosing the patient and delaying the appropriate treatment for her disease.

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Rituximab: a novel treatment for refractory Riedel’s thyroiditis

Cited by 10 publications

References 23 publications

Successful Use of Rituximab in a Case of Riedel Thyroiditis Resistant to Treatment with Prednisone and Tamoxifen

Successful Use of Rituximab in a Case of Riedel Thyroiditis Resistant to Treatment with Prednisone and Tamoxifen

Long-Term Outcomes of Tamoxifen Citrate Therapy and Histo- and Immunopathological Properties in Riedel Thyroiditis

Riedel’s Thyroiditis: Pitfalls in Diagnosis and Subsequent Complications

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