Han Soo Park1, Su Kyoung Kwon1 and Ye Na Kim2 Department of Internal Medicine, Division of Endocrinology and Metabolism  Division of Nephrology, Kosin University College of Medicine, Busan Republic of Korea
Thyroid storm is a rare and potentially life-threatening medical emergency. We experienced a case of thyroid storm associated with sepsis caused by pneumonia, which had a catastrophic course including recurrent cardiac arrest and subsequent multiple organ failure (MOF). A 22-year-old female patient with a 10-year history of Graves’ disease was transferred to our emergency department (ED). She had a cardiac arrest at her home and a second cardiac arrest at the ED. Her heart recovered after 20 min of cardiac resuscitation. She was diagnosed with thyroid storm associated with hyperthyroidism complicated by pneumonia and sepsis. Although full conventional medical treatment was given, she had progressive MOF and hemodynamic instability consisting of hyperthermia, tachycardia and hypotension. Because of hepatic and renal failure with refractory hypotension, we reduced the patient’s dose of beta-blocker and antithyroid drug, and she was started on continuous veno-venous renal replacement therapy (CRRT) with intravenous albumin and plasma supplementation. Subsequently, her body temperature and pulse rate began to stabilize within 1 h, and her blood pressure reached 120/60 mmHg after 6 h. We discontinued antithyroid drug 3 days after admission because of aggravated hyperbilirubinemia. The patient exhibited progressive improvement in thyroid function even after cessation of antithyroid drug, and she successfully recovered from thyroid storm and MOF. This is the first case of thyroid storm successfully treated by CRRT in a patient considered unfit for antithyroid drug treatment.
The presenting manifestations of thyroid storm vary and can include cardiac arrest with multiorgan failure in rare cases.
In some patients with thyroid storm, especially those with severe complications, conventional medical treatment may be ineffective or inappropriate.
During thyroid storm, the initiation of CRRT can immediately lower body temperature and subsequently stabilize vital signs.
Early initiation of CRRT can be life-saving in patients with thyroid storm complicated by MOF, even when used in combination with suboptimal medical treatment.
Thyroid storm is a rare and potentially life-threatening medical emergency. The presenting manifestations of thyroid storm are varied, and some cases may go unrecognized at first. Therefore, prompt recognition and proper treatment of thyroid storm is fundamental to saving patients with condition (1). In this report, we describe our experience with a rare case of thyroid storm that presented as recurrent cardiac arrest with subsequent multiple organ failure (MOF) and was successfully treated with continuous veno-venous renal replacement therapy (CRRT).
A 22-year-old female patient was transferred to our hospital for a cardiac arrest after a history of several days of dyspnea. The patient had a cardiac arrest at her home just after the arrival of emergency medical technicians and recovered after cardiac resuscitation. She had a second cardiac arrest immediately after arriving at the emergency department (ED) and recovered again after 20 min of cardiac resuscitation. Her husband indicated she had a 10-year history of hyperthyroidism and irregular adherence to medication, and she suffered from symptoms of cough, fever and anorexia about 1 week and dyspnea for 2 days.
On initial assessment after cardiac resuscitation, her body temperature was 38.5°C, heart rate was 180 beats/min (Fig. 1) and blood pressure was 80/40 mmHg. Findings on physical examination revealed anterior neck swelling and rales in the area of the left lower lung.
Initial biochemical tests showed neutrophilia with thrombocytopenia, elevated total bilirubin, aspartate aminotransferase (AST), alanine aminotransferase (ALT) and evidence of coagulopathy. Blood tests for thyroid function were as follows: free thyroxine (FT4): 4.19 ng/dL (reference value: 0.78–1.54 ng/dL), triiodothyronine (T3) >800 ng/dL (reference value: 65–150 ng/dL) and thyroid-stimulating hormone (TSH): 0.007 μU/mL (reference value: 0.55–4.78 μU/mL). Detailed laboratory data were presented as a table (Table 1). She had no history of alcohol intake, drug abuse, blood transfusion or exposure to hepatitis A, B or C. Extrahepatic obstruction was ruled out with an abdominal computed tomography (CT) scan. Chest CT scan revealed peribronchial consolidation of the left lung and diffuse enlargement of the thyroid (Fig. 2). Echocardiography revealed normal-sized cardiac chambers and preserved left ventricular systolic function with moderate pulmonary hypertension. The patient had a Burch and Wartofsky score of 110/140, which is highly suggestive of thyroid storm (2) and was finally diagnosed with thyroid storm complicated by pneumonia and sepsis.
|Laboratory test||Reference value||At arrival||Day 1||Day 2|
|Aspartate aminotransferase (U/L)||5–40||390||493||180|
|Alanine aminotransferase (U/L)||5–40||315||192||123|
|Alkaline phosphatase (U/L)||25–100||205|
|Lactate dehydrogenase (U/L)||200–450||1932|
|Prothrombin time (s)||11–15||27.1||33.5||35.5|
|Activated partial thromboplastin time (s)||27–45||43.2||50.3||180|
|International normalized ratio (INR)||0.8–1.2||2.57||3.39||2.86|
|White blood cells (×109/L)||43–9.4||2.57||3.09||1.36|
|Platelet count (×109/L)||188||80||34|
|Sputum||Streptococcus agalactiae, Candida albicans|
The patient was provided with prompt and full conventional medical treatment for thyroid storm (oral propylthiouracil, Lugol solution, intravenous beta-blocker and steroids) after emergent cardiac resuscitation and was given general supportive management (intravenous propacetamol, hydration, antibiotics and dopamine and norepinephrine); however, the patient had sustained tachycardia, hyperpyrexia and progressive multiorgan dysfunction including acute liver failure, coagulopathy, heart failure and rapid deterioration of renal function. Because of progressive hepatic dysfunction and refractory hypotension below 70/40 mmHg, we reduced the patient’s dose of beta-blocker and propylthiouracil and prepared her for therapeutic plasma exchange (TPE) for rapid direct removal of thyroid hormones (1). However, prior to initiating TPE, we started her on veno-venous CRRT with intravenous albumin and plasma supplementation for treatment of sepsis and acute renal failure as defined by urine output criteria of anuria over 6 h (3). After initiation of CRRT, her body temperature and pulse rate began to stabilize within 1 h, and her blood pressure increased to 100/60 mmHg 2 h later and 120/60 mmHg within 6 h (Fig. 3). Serum levels of triiodothyronine (T3) and alanine aminotransferase examined the following day showed moderate improvement, but her serum bilirubin level and aspartate aminotransferase (AST) remained elevated. In response, we reduced oral propylthiouracil from 200 mg every 6 h to 100 mg every 8 h on post-admission day 2 and stopped this treatment 3 days after admission because of aggravated hyperbilirubinemia and concerns regarding hepatic dysfunction. The patient exhibited progressive improvement of thyroid function even after cessation of propylthiouracil and successfully recovered from hyperthyroxinemia and hepatic dysfunction without TPE (Figs 4 and 5). We restarted antithyroid drug 13 days after admission.
The patient was discharged from the hospital with alert mental status and normal renal and liver function tests and coagulation parameters 1 month after admission.
Thyroid storm is a potentially fatal disease with a reported hospital mortality rate of 10–75% (1). The laboratory results for thyroid function during thyroid storm are not significantly different from those associated with thyrotoxicosis. Therefore, the diagnosis of thyroid storm is predominantly based on clinical manifestations including fever, cardiovascular dysfunction, gastrointestinal dysfunction and change in mental status, and clinical suspicion is essential for the diagnosis (2).
There have been several reports of unusual presentations of thyroid storm, including status epilepticus, complete heart block, rhabdomyolysis, stroke and MOF (4). In this case, the patient presented with recurrent cardiac arrest and subsequent MOF in the background of sepsis associated with pneumonia, which is an extremely rare presentation.
The patient in our case had refractory hyperthermia, tachycardia and other clinical deteriorations despite appropriate medical care for several hours. Plasmapheresis and TPE are alternative therapies for direct removal of free and protein-bound thyroid hormones, cytokines and autoantibodies used for severe cases of thyroid storm that are not effectively treated with antithyroid drugs alone or in patients unable to take antithyroid drugs because of hepatic dysfunction or medication side effects (5). Dialysis is generally regarded as ineffective for the removal of thyroid hormones because thyroid hormones are bound to plasma proteins; however, peritoneal dialysis (6), hemoperfusion through a resin (7) or charcoal column (8) and albumin dialysis (9) have shown some clinical benefits.
There have been no clinical reports describing improvement of thyroid storm with CRRT when it was not combined with TPE in patients unfit for antithyroid drug treatment. In the present case, we initiated CRRT before TPE because of the onset of acute kidney injury. Compared to intermittent hemodialysis, CRRT involves continuous dialysis and filtration over 24 h to facilitate slower removal of fluid and solute, which is better tolerated and results in fewer metabolic changes with improved removal of solute and immunomodulatory substances in critically ill patients. Therefore, CRRT is preferred to renal replacement therapy for hemodynamically unstable patients (10).
Prior to CRRT, the patient was treated with IV propacetamol and external cooling with an ice blanket and repeated alcohol swabs for several hours; however, her body temperature did not respond and remained between 39°C and 40°C for 10 h after admission. The most striking change after initiation of CRRT was rapid control of body temperature, which was accompanied by a slowed pulse rate and elevation of blood pressure (Fig. 3). The exact mechanism for the clinical improvement achieved by CRRT in this case is unclear; however, two possible mechanisms can be suggested. First, CRRT utilizes large volumes room-temperature fluids comprising dialysate and replacement fluids, and their administration can cause hypothermia. This well-known adverse effect of large volumes room-temperature fluids may have been useful for treating the patient’s hyperthermia. Second, intravenous infusion of albumin and plasma in CRRT can increase the ability of proteins to bind free thyroid hormone (5). Given these possibilities, further investigation regarding the effects and prompt initiation of CRRT in hemodynamically unstable thyrotoxicosis is needed.
In conclusion, this is the first case of thyroid storm successfully treated by CRRT not combined with TPE in a patient unfit for full dosing with antithyroid drug without surgery. Furthermore, our patient had a rare and severe presentation of thyroid storm involving repeated cardiac arrest and subsequent multiorgan dysfunction that was treated successfully. Our experience suggests that prompt recognition of thyroid storm and early initiation of CRRT combined with conventional medical treatment may be a useful approach when treating patients with thyroid storm associated with hemodynamically unstable MOF.
The authors declare that there is no conflict of interest that could be perceived as prejudicing the impartiality of the research reported.
This research did not receive any specific grant from any funding agency in the public, commercial or not-for-profit sector.
Written informed consent has been obtained from the patient for publication of the submitted article and accompanying images.
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