Abstract
Purpose
To present a case of ulcerative colitis associated with teprotumumab treatment for thyroid eye disease.
Observations
A 46-year-old Indian female was treated with teprotumumab (Tepezza) for severe thyroid eye disease within 9 months of diagnosis. The patient noted progression of her disease on oral prednisone and demonstrated severe, debilitating proptosis accompanied by eye ache and dry eyes. After 5 infusions of teprotumumab over a four-month period, the patient developed bloody diarrhea and fecal urgency. These symptoms progressively worsened and after two additional treatments, she underwent a colonoscopy. This confirmed the diagnosis of ulcerative colitis (UC). Treatment with teprotumumab was halted prior to the administration of the 8th infusion; however, the patient continued to have severe gastrointestinal symptoms two months after her last treatment.
Conclusions and importance
Teprotumumab is an insulin-like growth factor-1 receptor (IGF-1R) inhibitor demonstrated to improve proptosis in patients with active thyroid eye disease. Most adverse events reported are mild or moderate in severity; however, inflammatory bowel disease (IBD) is a serious adverse event that can develop as a result of treatment.
1
Introduction
The etiology of thyroid eye disease has not been fully elucidated. Patients with active disease have been shown to have elevated levels of the insulin-like growth factor-1 receptor (IGF-1R) and orbital fibroblasts have been implicated in the pathogenesis of the disease. Multiple treatment options have been well studied, including various courses of corticosteroid therapy, orbital radiotherapy, and monoclonal antibodies, including rituximab and tocilizumab. , However, teprotumumab, an inhibitor of the IGF-IR, is the sole monoclonal antibody approved by the US Food and Drug Administration for the medical treatment of thyroid eye disease. Published studies of teprotumumab have documented adverse events, including muscle spasms, nausea, diarrhea, stomatitis and hearing loss; however, though inflammatory bowel disease (IBD) is a contraindication to its use, it has not been reported previously as an adverse event.
2
Case report
A 46-year-old Indian female was referred to our clinic two months after being diagnosed with hyperthyroid Graves’ disease. She was initiated on oral methimazole therapy (5 mg daily) by her primary care provider. Her past ocular history was significant for high myopia with astigmatism. She was a non-smoker and occasionally consumed alcohol.
The patient’s best corrected visual acuity was 20/20 in her right eye and 20/30 in her left eye. Her extraocular movements were intact bilaterally. Her CAS was 2. Thyroid stimulating immunoglobulin (TSI) was 404 (<140% normal). Her exam was significant for severe eyelid retraction bilaterally with the MRD1 measuring 5 mm in both eyes. Hertel measurements were 22 mm and 23 mm in her right eye and left eye, respectively. She was started on selenium 200 mcg daily, offered intravenous or oral prednisone, continued on daily methimazole, and asked to return to clinic in 6 weeks.
The patient returned for follow up 12 weeks later with complaints of worsening proptosis, dry eyes and ocular ache. MRD1 measured 6 mm in both eyes and Hertel measurements had increased to 26 mm in both right and left eyes. The patient had been placed on oral prednisone (30 mg daily) by her primary care physician without improvement after 8 weeks. Teprotumumab (Tepezza) treatment was discussed in detail. Her TSI was 449.
At the time of teprotumumab initiation, the patient demonstrated diplopia and restriction of up gaze bilaterally. Hertel measurements were 28 mm and 26 mm in her right eye and left eye, respectively. Visual acuity in both eyes was stable Her CAS was 6. She started on an initial dose of 10 mg/kg of body weight for the first dose, followed by 20 mg/kg for the remaining doses.
The patient experienced tolerable muscle spasms, subjective hearing impairment and gastrointestinal symptoms during each treatment. However after the 5th teprotumumab treatment, she developed hematochezia and fecal urgency. Her abdominal symptoms progressively worsened after two additional infusions. She underwent a CT scan ( Fig. 1 ) and colonoscopy which established the diagnosis of ulcerative colitis (UC) ( Fig. 2 ) Teprotumumab infusions were discontinued while the ulcerative colitis was managed. Of note, after the third and each subsequent treatment, the patient demonstrated gradual improved proptosis, extraocular motility and diplopia. Her TSI declined to 360. Improvement from presentation through treatment is demonstrated in Fig. 3 . Despite discontinuation of teprotumumab, the patient continued to have severe gastrointestinal symptoms two months after the 7th treatment. She was treated with infliximab and prednisone. Her subjective hearing loss ultimately resolved..
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