{"doi":"10.2337/dc24-2322","title":"An Atypical Presentation of Cytokine Release Syndrome With Signs of Arthritis During Treatment With Teplizumab in a Pediatric Patient","abstract":"Teplizumab is an anti-CD3 monoclinal antibody that was recently approved by the U.S. Food and Drug Administration to delay the onset of clinical (stage 3) type 1 diabetes in those with stage 2 (preclinical with dysglycemia) type 1 diabetes (1,2).Common side effects that peak around day 5, as CD3 saturation is reached, include lymphopenia, rash, and fatigue (3).Rare major adverse effects include cytokine release syndrome (CRS) (typically including fever, nausea, headache, myalgia, arthralgia, and organ involvement), serious infections, and hypersensitivity reactions, including anaphylaxis, angioedema, and serum sickness (3).Here, we describe an atypical reaction and our approach, with ultimate successful completion of therapy.An 11-year-old boy with autoimmune hypothyroidism presented with stage 2 type 1 diabetes.He had elevated HbA 1c (5.9%, 41 mmol/mol), glutamic acid decarboxylase autoantibody (>250 IU/mL; normal is <5 IU/mL), and insulin autoantibody (2.3 units/mL; normal is <0.4 IU/mL).Family history was significant for autoimmune Graves disease, lupus, and vitiligo.The patient started daily teplizumab infusions, with dosing per label, at Boston Children's Hospital Outpatient Infusion Center, with recommended pretreatment medications acetaminophen and diphenhydramine.His clinical course proceeded as follows.","journal":"Diabetes Care","year":2025,"id":522605,"datarank":0.0,"base_score":0.0,"endowment":0.0,"self_citation_contribution":0.0,"citation_network_contribution":0.0,"self_endowment_contribution":0.0,"citer_contribution":0.0,"corpus_percentile":null,"corpus_rank":null,"citation_count":4,"citer_count":0,"citers_with_citation_signal":0,"citers_with_endowment":0,"datacite_reuse_total":0,"is_dataset":false,"is_dataset_confidence":0.9545,"is_data_producer":false,"deposit_databanks":null,"is_oa":true,"file_count":0,"downloads":0,"has_version_chain":false,"published_date":"2025-01-01","fair_score":null,"fair_percentile":null,"algorithm_id":"datarank_citation_only_1hop_v6","ranking_scope":"data_only","authors":[{"id":1015976,"name":"Raymond J. Kreienkamp","orcid":"0000-0002-1683-323X","position":1,"is_corresponding":false},{"id":1394766,"name":"Evan C. Rieger","orcid":null,"position":2,"is_corresponding":false},{"id":274165,"name":"Thinh H. Nguyen","orcid":"0000-0001-6660-6556","position":3,"is_corresponding":false},{"id":336606,"name":"Jason L. Gaglia","orcid":"0000-0003-2904-312X","position":4,"is_corresponding":false},{"id":430036,"name":"Belinda Lennerz","orcid":"0000-0002-8021-7984","position":5,"is_corresponding":false},{"id":750134,"name":"Courtney J. Duckworth","orcid":"0000-0002-5724-3471","position":0,"is_corresponding":true}],"reference_count":3,"raw_metadata":null,"created_at":"2026-07-19T02:49:54.058858Z","pmid":"39841557","pmcid":null,"fwci":null,"citation_percentile":null,"influential_citations":0,"oa_status":null,"license":null,"views":0,"total_file_size_bytes":0,"version_count":0,"fair_f":null,"fair_a":null,"fair_i":null,"fair_r":null,"fair_zscore":null,"fair_rationale":null,"fair_model":null,"fair_agent_version":null,"fair_fulltext_source":null,"fair_has_llm":null,"fair_computed_at":null,"clinical_trials":[],"software_tools":[],"db_accessions":[],"linked_datasets":[],"topics":[]}