• Jingen Hu and Caibao Hu.
• Department of Orthopedics, the First Affiliated Hospital, School of Medicine, Zhejiang University, No. 79 Qingchun Road, Hangzhou 310003, Zhejiang, China.
• Medicine (Baltimore). 2022 Mar 4; 101 (9): e28928e28928.

RationaleMcCune-Albright syndrome (MAS) is a rare heterogeneous clinical disease caused by sporadic, somatic, and postzygotic mutations. Thyroid crisis is even rare in patients with MAS, and we report the clinical outcomes of the first case of a MAS patient with atypical triiodothyronine (T3) hyperthyroidism who developed thyroid crisis after orthopedic surgery.Patient ConcernsThe patient with MAS and atypical T3 hyperthyroidism was an 11-year-old man who had undergone surgery for a right femur fracture and shepherd bending deformity. His main symptoms were dizziness, nausea, and vomiting with elevated body temperature because of developed thyroid crisis. Thyroid function tests showed high T3 and remarkably high free T3 levels, and remarkably increased thyrotropin level, but unchanged thyroxine and free thyroxine levels.DiagnosisThe patient was diagnosed with postoperative thyroid crisis following surgery for a right femur fracture, shepherd bending deformity, and MAS with atypical T3 hyperthyroidism.InterventionsPropranolol was intravenously administered. The therapy included intravenous hydrocortisone, a saturated solution of potassium iodine and propylthiouracil, and continuous physical cooling.OutcomesThe patient was discharged after achieving a stable condition with normal thyroid and liver function after surgery because of active anti-thyroid crisis treatment.LessonsThe operation of such patients should focus on the pre-operative heart rate, platelet level, and thyroid hormone levels. Abnormal values should be adjusted to the normal range, and such patients should achieve complete hemostasis and transfuse with blood following surgery anemia.Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.

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