Thyroid crisis mimicking clinically mild encephalitis/encephalopathy with a reversible splenial lesion: A pediatric case report.

BACKGROUND: Reversible lesions in the splenium of the corpus callosum (SCC) with viral infections are associated mainly with clinically mild encephalitis/encephalopathy with a reversible splenial lesion (MERS). We report a pediatric patient in thyroid crisis with reversible SCC lesions. CASE DESCRIPTION: We diagnosed a 9-year-old girl with thyroid crisis. She had presented with fever, tachycardia, and impaired consciousness. Magnetic resonance imaging revealed hyperintense signals in the splenium and genu of the corpus callosum and a white matter lesion of the left hemisphere in diffusion-weighted imaging. The initial, tentative diagnosis was clinically mild encephalitis/encephalopathy with a reversible splenial lesion (MERS). We initiated intravenous methylprednisolone pulse therapy; thereafter, her level of consciousness rapidly improved. On admission, thyroid function studies revealed elevation of free thyroxine and a low level of thyroid stimulating hormone with thyroid-related autoantibodies. She was begun on thiamazole and was discharged without neurological sequelae. CONCLUSION: Thyroid crisis is similar to acute encephalitis or encephalopathy associated with viral infection, especially with MERS, because the clinical and radiological features resemble those of thyroid crisis; therefore, thyroid diseases should be considered as a possible cause of reversible lesions in the splenium of the corpus callosum.

Therapeutic plasma exchange for control of thyroid storm.

Therapeutic plasma exchange (TPE) for thyroid storm has recently been upgraded to a category II indication after decades though its recommendation level still remains at Grade 2C according to the American Society for Apheresis (ASFA). In the absence of prospective randomized controlled trials due to the rarity of thyroid storm, retrospective data from case series continue to elevate the clinical evidence supporting TPE as a life-saving modality for complicated thyroid storm patients. We report three cases of life-threatening thyroid storm from Graves' disease rescued by TPE via rapid reduction in circulating thyroid hormones. Each patient underwent TPE when it was judged that other thyroid storm treatment options were futile or unsafe. The first patient received 4 cycles of TPE while the second patient received 9 cycles of TPE, and the third patient received 2 cycles of TPE with satisfactory clinical improvement. Plasma FT4 and TSH receptor antibody levels of the first case declined by 41.3% and >50% respectively right after the first round of TPE; plasma FT4 of the second patient dropped by up to 31.6% during the course of TPE; plasma FT4 and TSH receptor antibody of the third patient declined by 66% and 56.2% respectively after the first cycle of TPE. This demonstrates the safety, efficacy, and feasibility of TPE in thyroid storm especially when other therapeutic interventions are contraindicated. TPE operates via the elimination of serum proteins-bound thyroid hormones, thyroid autoantibodies, cytokines, and catecholamines in addition to increasing unsaturated binding sites for thyroid hormones.

Thyroid storm presenting as septic shock in the intensive care unit: A Case Series.

Thyroid storm is a rare endocrine emergency that rarely presents with septic shock. It occurs in thyrotoxic patients and is manifested by decompensation of multiple organs, triggered by severe stress. The diagnosis and response to treatment is made by Burch-Wartofsky point scale or Japanese thyroid association criteria due to lack of pathophysiology of thyroid storm. We reported series of patients that presented with altered sensorium, cough, fever, palpitation, shortness of breath and shock. Patient were treated initially for septic shock, later diagnosed as thyroid storm and was treated with oral carbimazole, propanolol and digoxin. From this, we want to emphasize that thyroid storm can have any presentation that should be kept in differential diagnosis of septic shock not responding to usual treatment; early diagnosis and treatment with oral medication can decrease morbidity and mortality in rural setting where intravenous form of antithyroid drug are not available for thyroid storm. Keywords: sepsis; septic shock; thyroid storm.

Atypical presentation of thyroid storm: a diagnostic dilemma.

Thyroid storm (accelerated hyperthyroidism) is an uncommon life-threatening emergency. The diagnosis is difficult and at times delayed owing to atypical presentation. Early diagnosis is the key to its successful management. We came across a patient who had presentations of acute abdomen but later diagnosed in thyroid storm. Multiorgan involvement leads all resuscitative measures futile and prevented us to salvage the patient.

Thyroid storm and warm autoimmune hemolytic anemia.

Graves' disease is often associated with other autoimmune disorders, including rare associations with autoimmune hemolytic anemia (AIHA). We describe a unique presentation of thyroid storm and warm AIHA diagnosed concurrently in a young female with hyperthyroidism. The patient presented with nausea, vomiting, diarrhea and altered mental status. Laboratory studies revealed hemoglobin 3.9g/dL, platelets 171×109L-1, haptoglobin <5mg/dL, reticulocytosis, and positive direct antiglobulin test (IgG, C3d, warm). Additional workup revealed serum thyroid stimulating hormone (TSH) <0.01µIU/mL and serum free-T4 (FT4) level 7.8ng/dL. Our patient was diagnosed with concurrent thyroid storm and warm AIHA. She was started on glucocorticoids to treat both warm AIHA and thyroid storm, as well as antithyroid medications, propranolol and folic acid. Due to profound anemia and hemodynamic instability, the patient was transfused two units of uncrossmatched packed red blood cells slowly and tolerated this well. She was discharged on methimazole as well as a prolonged prednisone taper, and achieved complete resolution of the thyrotoxicosis and anemia at one month. Hyperthyroidism can affect all three blood cell lineages of the hematopoietic system. Anemia can be seen in 10-20% of patients with thyrotoxicosis. Several autoimmune processes can lead to anemia in Graves' disease, including pernicious anemia, celiac disease, and warm AIHA. This case illustrates a rarely described presentation of a patient with Graves' disease presenting with concurrent thyroid storm and warm AIHA.

Thyroid Storm Precipitated by Diabetic Ketoacidosis and Influenza A: A Case Report and Literature Review.

A 46-year-old woman with a history of Graves' disease presented with the chief complaints of appetite loss, weight loss, fatigue, nausea, and sweating. She was diagnosed with diabetic ketoacidosis (DKA), thyroid storm, and influenza A. She was treated with an intravenous insulin drip, intravenous fluid therapy, intravenous hydrocortisone, oral potassium iodine, and oral methimazole. As methimazole-induced neutropenia was suspected, the patient underwent thyroidectomy. It is important to maintain awareness that thyroid storm and DKA can coexist. Furthermore, even patients who have relatively preserved insulin secretion can develop DKA if thyroid storm and infection develop simultaneously.

Thyroid storm caused by a chinese herb contaminated with thyroid hormones.

BACKGROUND: We report a case of thyroid storm caused by consuming a Chinese herb contaminated with thyroid hormones. CASE REPORT: A 70-year-old man presented to an emergency department after 2 days of nausea, vomiting, and weakness. Three days previously, he had started taking Cordyceps powder and "Flower Man Sang Hung" as recommended by his Chinese physician. Following admission, the patient deteriorated and was eventually diagnosed with thyroid storm complicated by rapid atrial fibrillation requiring cardioversion, intubation, and intensive care admission. The analysis of the Chinese herb "Flower Man Sang Hung" was positive for levothyroxine. The patient was extubated 11 days after admission and discharged to a rehabilitation centre after 17 days of hospitalization. The Chinese medicine physician was informed of the events. CONCLUSIONS: Herbal products can be the source of illness, medication interactions, and contamination. Awareness should be raised among Chinese medicine physicians, allopathic physicians, and their patients. Clinicians should also have a low threshold of suspicion to seek laboratory analysis of suspect substances when the cause of the clinical presentation is unclear.

A case of thyroid storm with a markedly elevated level of circulating soluble interleukin-2 receptor complicated by multiple organ failure and disseminated intravascular coagulation syndrome.

Thyroid storm (TS) is a life-threatening endocrine emergency. However, the pathogenesis of TS is poorly understood. A 40-year-old man was admitted to a nearby hospital with body weight loss and jaundice. Five days after a contrasted abdominal computerized tomography (CT) scan, he exhibited high fever and disturbance of consciousness. He was diagnosed with TS originating from untreated Graves' disease and was transferred to the intensive care unit (ICU) of our hospital. The patient exhibited impaired consciousness (E4V1M4 in Glasgow coma scale), high fever (39.3°C), and atrial flutter with a pulse rate 162/min, and was complicated by heart failure, acute hepatic failure, and disseminated intravascular coagulation syndrome (DIC). His circulating level of soluble interleukin-2 receptor (sIL-2R), a serum marker of an activated immune response, was highly elevated (7,416 U/mL, reference range: 135-483). Multiple organ failure (MOF) and DIC were successfully managed by multimodality treatments using inorganized iodide, glucocorticoids, anti-thyroid drugs, beta-blockers, and diuretics as well as an anticoagulant agent and the transfusion of platelet concentrate and fresh frozen plasma. sIL-2R levels gradually decreased during the initial treatment, but were still above the reference range even after thyroidectomy. Mild elevations in serum levels of sIL-2R have previously been correlated with thyroid hormone levels in non-storm Graves' disease. The present study demonstrated, for the first time, that circulating sIL-2R levels could be markedly elevated in TS. The marked increase in sIL-2R levels was speculated to represent an inappropriate generalized immune response that plays an unknown role in the pathogenesis of TS.

Surviving the storm: two cases of thyroid storm successfully treated with plasmapheresis.

Thyroid storm is a rare, but critical, illness that can lead to multiorgan failure and carries a high death rate. The following case series describes two adult men with Graves' disease who presented in thyroid storm and either failed or could not tolerate conventional medical management. However, both patients responded well to plasmapheresis, which resulted in clinical and biochemical stabilisation of their disease processes. The treatment option of plasmapheresis should be considered as a stabilising measure, especially when patients have failed or cannot tolerate conventional therapy. Plasmapheresis leads to amelioration of symptoms and a significant decline in thyroid hormone levels, providing a window to treat definitively with thyroidectomy.

Significant attenuation of stimulated cortisol in early Graves disease without adrenal autoimmunity.

OBJECTIVE: To investigate cortisol responses to adrenocorticotropic hormone during thyrotoxic (G1) and euthyroid (G2) phases in patients with Graves disease (GD) who were without adrenal autoimmunity. METHODS: Fifteen patients with GD, who were thyrotropin receptor antibody positive and 21-hydroxylase antibody negative, were recruited to this prospective pilot study. A modified short Synacthen test (SST) was performed, in which cortisol was measured every 30 minutes for 2 hours during G1 and G2. RESULTS: The median times to SST were 3 weeks (G1) and 27 weeks (G2) after diagnosis of GD. Integrated stimulated cortisol levels were significantly lower at G1 in comparison with G2: mean ± standard error of the mean for area under the curve was 78,091.6 ± 4,462.1 nmol/L (G1) versus 89,055 ± 4,434 nmol/L at 120 minutes (G2), P = .017; and for delta area under the curve was 36,309.9 ± 3,526 nmol/L (G1) versus 44,041.7 ± 2,147 nmol/L at 120 minutes (G2), P = .039. Mean cortisol levels were significantly lower for G1 versus G2 at 60, 90, and 120 minutes of the SST (P = .001 to .013). The cortisol level was abnormal in 2 patients (13%) at 30 minutes during G1 but in none during G2. There was no correlation of integrated cortisol with free thyroxine or thyrotropin receptor antibody. There was no significant difference in median adrenocorticotropic hormone level (17 versus 20.4 ng/mL at G1 and G2, respectively; P = .14). CONCLUSION: Significant attenuation of stimulated cortisol occurs in the early thyrotoxic phase in comparison with the euthyroid phase in patients with GD without adrenal autoimmunity. Clinicians treating patients with GD should have a low threshold for investigating symptoms suggestive of hypoadrenalism at times of "stress."

Hepatic failure resulting from thyroid storm with normal serum thyroxine and triiodothyronine concentrations.

A 63-year-old male presented with productive cough, fever, palpitation, shortness of breath, and mental confusion. Fulminant hepatic failure occurred with high aminotransferase and severe jaundice. Thyroid function tests showed thyroid-stimulating hormone level of 0.7 microU/mL (normal range, < 6.2 microU/mL), with normal serum thyroxine and triiodothyronine levels. He died on post-admission day 5. Autopsy revealed diffuse hyperplasia of the thyroid gland, centrilobular necrosis of the liver, and congestion of the gastrointestinal tract, spleen, and adrenal glands. These pathological findings were compatible with decompensated hyperthyroidism. Thyroid storm may present with normal serum triiodothyronine, thyroxine, and hepatic failure. Such presentation is rare.

A case of atypical thyroid storm with hypoglycemia and lactic acidosis.

We describe herein a case of thyroid storm with hypoglycemia and lactic acidosis-a rare complication of thyroid storm. The patient was a 50-year-old Japanese woman who suffered cardiopulmonary arrest an hour after hospitalization. Analysis of a blood sample obtained before her cardiopulmonary arrest yielded surprising results: Her plasma glucose level was 14 mg/dL and her lactic acid concentration had increased to 6.238 mM. Thus, if atypical thyroid storm presents with normothermic hypoglycemia, and lactic acidosis, we believe it is necessary to consider a diagnosis of thyroid storm earlier, because this condition requires emergency treatment. Moreover, it is very important to apply standard principles in the treatment of atypical cases of thyroid storm.

An association between Takotsubo cardiomyopathy and thyroid storm.

A 65-year-old woman presented to the hospital with weight loss and diarrhea. A diagnosis of thyroid storm was established by the presence of fever, lethargy, tachycardia, heart failure (HF), and abnormal thyroid function tests. An acute coronary syndrome (ACS) was suspected because of anteroseptal ST-segment elevations on electrocardiogram and severe left ventricular (LV) dysfunction with apical dilation on echocardiogram. The ejection fraction (EF) was 25%. Treatment for ACS and HF was begun. Coronary arteriography revealed normal coronaries. Propylthiouracil and corticosteroids were added to treat thyrotoxicosis. On Day 4, repeat echocardiography confirmed an EF of 65% and complete resolution of LV dysfunction and cavity dilation. Reversible LV dysfunction and apical cavity dilation is consistent with Takotsubo cardiomyopathy. While commonly associated with psychological trauma, its association with thyroid storm has only been reported twice before. Knowledge of this possible association is of importance in establishing the diagnosis and instituting a proper treatment plan.

Thyrotoxicosis with coronary spasm that required coronary artery bypass surgery.

We describe a 47-year-old woman with severe coronary vasospasm induced by hyperthyroidism. The patient complained of anginal chest pain without specific characteristics of thyrotoxicosis. Coronary arteriography was performed and revealed 90% stenosis of both the left and right coronary os. She was treated with emergent coronary artery bypass graft surgery. Postoperatively, she exhibited a comatose mentality. Severe thyrotoxicosis was indicated on thyroid function tests and thyrotoxic storm was diagnosed. Nineteen days after the surgery and following the initiation of propylthiouracil treatment, coronary arteriography revealed entirely normal coronary arteries.

Two cases of thyroid storm-associated cholestatic jaundice.

OBJECTIVE: To describe the association of the rare and serious complication of jaundice with severe thyrotoxicosis, a potentially lethal endocrine disorder. METHODS: We report the clinical, laboratory, and pathologic findings of 2 cases of severe jaundice (total bilirubin levels: 35.2 mg/dL in case 1 and 42 mg/dL in case 2) associated with thyroid storm in the absence of a history of liver disease, thionamide exposure, or congestive heart failure. We also present other relevant reports available in the literature. RESULTS: Case 1 was a 38-year-old woman who presented with nausea, vomiting, fatigue, pruritus, and frequent nonbloody diarrhea. She was transferred to our institution because of worsening hyperbilirubinemia. Case 2 was a 35-year-old woman admitted to a community hospital with thyroid storm and jaundice. Upon transfer to our institution, the patient was unconscious, mechanically ventilated, and in atrial fibrillation. In case 2, liver biopsy results revealed diffuse hepatocellular ballooning with intrahepatic cholestasis with mild portal lymphocytic infiltration. Both patients presented with severe cholestatic jaundice in the absence of congestive heart failure; underlying liver disease (infectious or autoimmune); or previous exposure to thionamides, other hepatotoxic agents, or complementary and alternative medications. In both cases, jaundice responded to therapy with antithyroid medications. Both patients eventually underwent thyroidectomy with complete resolution of the jaundice. CONCLUSION: The data strongly suggest that in these patients, the hepatic dysfunction was primarily due to hyperthyroidism. These cases indicate that the mere presence of hyperbilirubinemia during severe thyrotoxicosis should not per se delay the use of potentially life-saving thionamides once a thorough evaluation for other causes of liver disease has been completed.

Thyroid storm induced by trauma due to spear fishing-gun trident impaction in the neck.

A rare case of thyroid storm induced by thyroid gland injury because of penetrating neck trauma is reported. The injury was because of a spear fishing-gun trident impaction in the neck. The diagnosis of thyroid gland injury was suspected by preoperative clinical examination and established during neck exploration in the theatre. The gland injury led to thyroid storm owing to the rupture of acini and liberation of T4 into the bloodstream. Withdrawal of the impacted trident along with subtotal thyroid lobectomy and repair of soft tissue damage in addition to supported treatments, which corrected the hyperthyroid state, led to uneventful recovery.

Thyrotoxicosis and thyroid storm.

Thyroid storm represents the extreme manifestation of thyrotoxicosis as a true endocrine emergency. Although Grave's disease is the most common underlying disorder in thyroid storm, there is usually a precipitating event or condition that transform the patient into life-threatening thyrotoxicosis. Treatment of thyroid storm involves decreasing new hormone synthesis, inhibiting the release of thyroid hormone, and blocking the peripheral effects of thyroid hormone. This multidrug, therapeutic approach uses thionamides, iodine, beta-adrenergic receptor antagonists, corticosteroids in certain circumstances, and supportive therapy. Certain conditions may warrant the use of alternative therapy with cholestyramine, lithium carbonate, or potassium perchlorate. After the critical illness of thyroid storm subsides, definitive treatment of the underlying thyrotoxicosis can be planned.