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A Case of a Patient with Medical-Refractory Thyroid Storm Treated Successfully by Surgical Resection
Int J Thyroidol 2023;16(2):200-204
Published online November 30, 2023;  https://doi.org/10.11106/ijt.2023.16.2.200
© 2023 Korean Thyroid Association.

Sang Yun Lee1, Jaehoon Kim1, Seongjun Won1,2 and Jung Je Park1,2

Department of Otorhinolaryngology, Gyeongsang National University College of Medicine and Gyeongsang National University Hospital1, Institute of Health Sciences, Gyeongsang National University2, Jinju, Korea
Correspondence to: Jung Je Park, MD, PhD, Department of Otorhinolaryngology, Gyeongsang National University Hospital, 79 Gangnam-ro, Jinju 52727, Korea
Tel: 82-55-750-8177, Fax: 82-55-759-0613, E-mail: capetown@hanmail.net
Received October 23, 2023; Revised November 10, 2023; Accepted November 10, 2023.
This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (https://creativecommons.org/licenses/by-nc/4.0), which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Abstract
Thyroid storm is a life-threatening condition caused by elevated thyroid hormone levels. While multidrug treatments, encompassing antithyroid agents, inorganic iodine, and corticosteroids, are typically employed, they may be ineffective in certain cases. We present the case of a 41-year-old woman whose thyroid storm did not respond to standard medical treatments or therapeutic plasma pheresis, which led to a deterioration in her condition. Ultimately, an emergency total thyroidectomy was performed with no postoperative complications, such as recurrent laryngeal nerve injury or bleeding. This case underscores the potential efficacy of emergency thyroidectomy for the management of refractory thyroid storm.
Keywords : Thyroid storm, Thyroidectomy, Thyrotoxicosis, Thyroid crisis
Introduction

Hyperthyroidism can be defined as suppressed levels of pituitary thyroid-stimulating hormone (TSH) and elevated levels of thyroxine (T4) and triiodothyronine (T3). Thyroid storm is a rare, clinical condition caused by the exacerbation of hyperthyroidism1) that can lead to life-threatening multi-organ failure.1) Antithyroid drugs (ATDs) are the mainstay treatment for the control of thyrotoxicosis, and are effective.2,3) However, in some cases, ATDs can have rare but severe side effects or fail to control disease progression.4) In such cases, other treatments, such as therapeutic plasma exchange (TPE), radioiodine therapy, or thyroidectomy are required. Here, we report a case of thyroid storm that could not be managed by ATDs.

Case Report

A 41-year-old woman visited an emergency room (ER) complaining of palpitation. Palpitation started 2 weeks before presentation, and the patient complained of intermittent chest pain. The patient’s vital signs at arrival to the ER were a systolic blood pressure of 148 mmHg, a diastolic blood pressure of 112 mmHg, a heart rate (HR) of 202 bpm, a respiratory rate of 20 breaths per minute, and a body temperature of 36.2°C. She had a huge, soft, and non-tender mass in the anterior cervical region with a dilated vessel (Fig. 1). Electrocardiography (ECG) revealed atrial fibrillation with a rapid ventricular response. Laboratory tests revealed total bilirubin (T. bilirubin) 1.78 mg/dL, alkaline phosphatase (ALP) 199 U/L, alanine transferase (ALT) 30 U/L, aspartate aminotransferase (AST) 45 U/L, Troponin-T 30 ng/L, and N-terminal prohormone of brain natriuretic peptide (NT-proBNP) 2368 pg/mL. In the cardioechogram, four chambers were enlarged, and the left ventricle ejection fraction was 24%. Her thyroid function tests (TFT) showed triiodothyronine (T3) 333 ng/dL, free thyroxine (free T4) 7.06 ng/dL, thyroid-stimulating hormone (TSH) 0.01 uIU/mL, thyroglobulin antibody 13.80 IU/mL, anti-thyroid peroxidase (anti-TPO) 332.00 IU/mL, TSH receptor antibody (TSH-R Ab) >40.00 IU/L. She complained of persistent palpitations, and was administered a beta-blocker and hydrocortisone (100 mg) intravenously and propylthiouracil (PTU) 200 mg and inorganic iodine orally. Her clinical status deteriorated, and her systolic blood pressure dropped to 55 mmHg. Thus, she underwent synchronized cardioversion together with intravenous norepinephrine and oro-tracheal intubation. Her Bruch-Wartofsky point score was 70, strongly suggesting that her symptoms were caused by a thyroid storm (Fig. 2). Then, the patient was hospitalized in an intensive care unit (ICU) with extubation. She was treated with PTU 600 mg/day, inorganic iodide, hydrocortisone 300 mg/day, and amiodarone infusion for 3 days to control tachycardia. Due to decreased hepatic function, we changed PTU 600 mg to methimazole (MMI) 30 mg. Despite being maintained on this regimen, her condition deteriorated further. Her laboratory findings showed hepatic failure (ALP 220 U/L, AST 5031 U/L, ALT 1628 U/L, T. bilirubin 4.8 mg/dL) and prothrombin time (PT INR) 5.76, activated partial thromboplastin time (aPTT) 35.9 sec, thrombin time 24.7 sec, fibrinogen degradation product (FDP) 105.05 μg/mL, fibrinogen 95 mg/dL, D-dimer >20 μg/mL FEU, indicating ongoing disseminated intravascular coagulation (DIC). Due to liver failure and DIC, we attempted TPE for 3 days. However, thyrotoxicosis symptoms continued to exacerbate. Finally, we decided that she should undergo surgery. The patient underwent total thyroidectomy (Fig. 3). During surgery, huge thyroid was wet condition, and higher bleeding tendency than others. So, we conducted surgery with cauterization carefully. Three days after surgery, her TFT function test revealed T3 84.9 ng/dL, TSH 0.01 uIU/mL, and free T4 2.05 ng/dL (Fig. 4) and biopsy reported diffuse toxic goiter. On the same day, the patient complained of dyspnea and continued to complain even after receiving supplemental oxygen. As a result, the ICU doctor attempted oro-tracheal intubation, but it failed twice; thus, we conducted an emergency tracheostomy through the previous operation site (Fig. 5). After tracheostomy, we performed a chest X-ray, which revealed total atelectasis of the right lung. A day later, ECG indicated torsade de pointes, and cardiac arrest occurred. Thus, advanced cardiovascular life support (ACLS) was provided, and 200 joules were delivered for defibrillation. After 12 min, her heart rhythm returned to spontaneous circulation (ROSC). The patient’s electrolyte imbalance was corrected during further hospitalization, the tracheostomy tube was removed, and the patient was discharged at 1 month post operation. The patient is still under observation on an outpatient basis.

Fig. 1. Gross photograph of anterior huge neck mass.

Fig. 2. Bruch-Wartofsky point.

Fig. 3. (A) Intra-operative specimen, resected thyroid specimen. (B) Post-operative operation field, saved both recurrent laryngeal nerve. (C) The pathology of the resected specimen showed an increased follicular component, Hematoxylin and Eosin stain (H&E stain), ×40.

Fig. 4. Thyroid hormone level change trend.

Fig. 5. Emergency tracheostomy though previous operation site.
Discussion

Thyroid storm is the most severe type of thyrotoxicosis. The disease has a high mortality rate if not treated promptly. The mortality rate is about 20% to 30%.1,5) Thyroid hormone is the primary hormone regulating the body’s metabolic systems. It increases the activity of the sodium/potassium pump in tissues, which increases thermogenesis and basal metabolic rate.6) Thyroid storm leads to tachycardia, hypertension, and hyperthermia.7)

The diagnostic criteria for thyroid storm are based on a clinical symptom scoring system, the Burch and Wartofsky’s scoring system. A score of 45 or above is diagnostic of thyroid storm, whereas a score below 25 points suggests thyroid storm is unlikely.8) The patient in this case study had a Burch and Wartofsky score of 70 (tachycardia 25 points, atrial fibrillation 10 points, congestive heart failure 15 points, jaundice 20 points), indicating she was suffering from thyroid storm.

Various treatments for thyroid storm patients have been developed, including beta-blocker, inorganic iodide, and corticosteroid therapies.2,9) ATDs can change thyroid hormone status by inhibiting the organification of iodide and coupling of iodothyronine.10) However, ATDs have limited effects in treating thyroid storm because it takes several weeks to reach euthyroidism.11) Antithyroid medications and inorganic iodine therapy cannot control thyroid hormone levels within 3 days, so we employed TPE, which is known to improve thyrotoxicosis rapidly. TPE can be considered as a treatment option if thyrotoxicosis symptoms do not improve within 24-48 hours of initial treatment since thyrotoxic symptoms typically fade away within 12-24 hours of proper initial therapy.2) But the thyrotoxicosis symptoms of our patient continued to deteriorate despite performing TPE three times and multidrug therapy; this forced us to choose a definite treatment therapy, such as surgery or radioiodine therapy. Radioiodine therapy is an option for the treatment of hyperthyroidism. However, it is unsuitable for the treatment of acute thyroid storm because it can worsen symptoms.12)

The patient in this case study had large goiters and did not respond to medical treatment or TPE. Therefore, total thyroidectomy was required. However, to perform thyroidectomy successfully, the patient must be in a euthyroid state,9) which was not the case for our patient. Nevertheless, we performed thyroidectomy despite the patient not being in an euthyroid state. As a result, we got good results, which included no complications, recurrent laryngeal nerve injury, or postoperative bleeding after surgery.

In conclusion, if medical treatment fails to control a thyroid storm, an urgent total thyroidectomy can be a highly effective solution, regardless of whether the patient is in a euthyroid state or not.

Conflicts of Interest

No potential conflict of interest relevant to this article was reported.

References
  1. Tietgens ST, Leinung MC. Thyroid storm. Med Clin North Am 1995;79(1):169-84.
    Pubmed CrossRef
  2. Satoh T, Isozaki O, Suzuki A, Wakino S, Iburi T, Tsuboi K, et al. 2016 Guidelines for the management of thyroid storm from The Japan Thyroid Association and Japan Endocrine Society (First edition). Endocr J 2016;63(12):1025-64.
    Pubmed CrossRef
  3. Aulet RM, Wein RO, Siegel RD. Surgical management of an atypical presentation of a thyroid storm. Int J Endocrinol Metab 2014;12(2):e13539.
    Pubmed KoreaMed CrossRef
  4. Chiu HHC, Sarsagat JPD, Dominguez HB, Lucero JAC, Uy ABC, Paz-Pacheco E. Therapeutic plasma exchange in thyroid storm refractory to conventional treatment. Acta Med Philipp 2022;56(5):157-60.
    CrossRef
  5. Alfadhli E, Gianoukakis AG. Management of severe thyrotoxi-cosis when the gastrointestinal tract is compromised. Thyroid 2011;21(3):215-20.
    Pubmed CrossRef
  6. Uchida N, Suda T, Ishiguro K. Thyroidectomy in a patient with thyroid storm: report of a case. Surg Today 2015;45(1):110-4.
    Pubmed CrossRef
  7. Gavin LA. Thyroid crises. Med Clin North Am 1991;75(1):179-93.
    Pubmed CrossRef
  8. Burch HB, Wartofsky L. Life-threatening thyrotoxicosis. Thyroid storm. Endocrinol Metab Clin North Am 1993;22(2):263-77.
    Pubmed CrossRef
  9. Ross DS, Burch HB, Cooper DS, Greenlee MC, Laurberg P, Maia AL, et al. 2016 American Thyroid Association guidelines for diagnosis and management of hyperthyroidism and other causes of thyrotoxicosis. Thyroid 2016;26(10):1343-421.
    Pubmed CrossRef
  10. Migneco A, Ojetti V, Testa A, De Lorenzo A, Gentiloni Silveri N. Management of thyrotoxic crisis. Eur Rev Med Pharmacol Sci 2005;9(1):69-74.
    Pubmed CrossRef
  11. Schaaf L, Greschner M, Paschke R, Kusterer K, Teuber J, Huck K, et al. Thyrotoxic crisis in Graves' disease: indication for immediate surgery. Klin Wochenschr 1990;68(21):1037-41.
    Pubmed CrossRef
  12. Shafer RB, Nuttall FQ. Thyroid crisis induced by radioactive iodine. J Nucl Med 1971;12(5):262-4.
    Pubmed


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