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Ann Thorac Surg 2004;77:1839-1841
© 2004 The Society of Thoracic Surgeons


Case report

Plasmapheresis as effective treatment for thyrotoxic storm after sleeve pneumonectomy

Jurgen Petry, MDa, Paul E. Y. Van Schil, MD, PhDb, Pascale Abrams, MDc, Philippe G. Jorens, MD, PhDa*

a Department of Intensive Care Medicine, University Hospital of Antwerp, Antwerp, Belgium
b Department of Thoracic and Vascular Surgery, University Hospital of Antwerp, Antwerp, Belgium
c Department of Endocrinology, University Hospital of Antwerp, Antwerp, Belgium

Accepted for publication June 3, 2003.

* Address reprint requests to Dr Jorens, University Hospital of Antwerp, Department of Intensive Care Medicine, Wilrijkstraat 10, B-2650 Edegem, Belgium.
e-mail: philippe.jorens{at}uza.be


    Abstract
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 Abstract
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 Comment
 References
 
Th yrotoxicosis is a life-threatening disorder when encountered after a major surgical procedure. Plasma exchange is an occasionally reported alternative treatment to thyroidectomy, iodine ablative therapy, or the administration of thyreostatic drugs. We used plasmapheresis as a lifesaving treatment in a patient with thyrotoxicosis, as encountered after a left-sleeve pneumonectomy, in whom many classic therapies either failed or were not retained.


    Introduction
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 Abstract
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Thyrotoxic storm is a clinical syndrome of exaggerated thyrotoxicosis based on typical cardinal manifestations in the presence of abnormal thyroid function test results [1]. Even when diagnosed early and treated vigorously, its mortality rate ranges from 10% to 75% in hospitalized patients [1].

We report on the favorable use of plasmapheresis in a patient with a fulminating thyrotoxic storm, which occurred in the postoperative period after an elective sleeve pneumonectomy.

A 45-year-old white woman with a history of asthma bronchiale was diagnosed with an adenoid cystic carcinoma of the left main bronchus. The elective surgical procedure had to be postponed because of the concomitant diagnosis of Graves' hyperthyroidism. Laboratory values at that moment were thyroid-stimulating hormone (TSH) less than 0.02 µU/mL (reference value, 0.47 to 4.7 µU/mL), FT4 = 66.7 pmol/L (reference value, 9.7 to 23.4 pmol/L), FT3 = 28.5 pmol/L (reference value, 4.3 to 8.1 pmol/L), and TSH receptor antibodies = 71 mU/mL (reference value, < 1 mU/mL) (Table 1). She was initially treated with temazepam, the thyreostatic agent thiamazole, the ß-blocking agent propranolol, and inhalation corticosteroids.


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Table 1. Follow-Up of Thyroid Hormone Assay Values at Diagnosis, Before and After Pneumonectomy, and After Treatment (With Both Medication and Plasmapheresis)

 
One month later, the patient underwent the surgical intervention. No hyperthyroidism was observed. A left-sleeve pneumonectomy by bilateral thoracotomy was performed. Regarding the technical details of the operative procedure, a left pneumonectomy was initially performed through a posterolateral thoracotomy. The results of a frozen-section examination were negative for the aortopulmonary nodes but positive for the bronchial section margin. The bronchial stump was sutured manually. Afterward, the patient was turned on the left side, and a right posterolateral thoracotomy was performed. With the aid of high-frequency jet ventilation, the main carina was resected. The results of frozen-section analysis of the tracheal and right bronchial margins were negative. The right main bronchus was anastomized to the trachea with a continuous polypropylene 4-0 suture. Bronchoscopy at the end of the procedure showed no abnormalities at the anastomotic site. Immediately postoperatively, treatment with propranolol and thiamazole was continued at the intensive care unit (ICU). Four days postoperatively, propylthiouracil and iodide were added because of deteriorating thyroid function. Cefepime, amikacin, and fluconazole were also started for pneumonia. One day later, potassium perchlorate was further added as conservative therapy. On the 10th day, the patient was extubated without success; she was reintubated because of hypoxemia 1 day later. She also experienced progressive muscular weakness. Electromyography did not demonstrate demyelinization or polyneuropathy. Guillain-Barré syndrome was excluded by a normal protein level in the cerebrospinal fluid and the absence of typical electromyographic signs.

At that time, a thyroid storm was suspected. Clinical signs included fever and leucocytosis, atrial fibrillation, anxiety, profuse sweating, nausea and vomiting, hypoglycemia, hypercalcemia, diarrhea, and hemodynamic instability with alternating hypotension and hypertension. The laboratory values of thyroid function revealed pronounced hyperthyroidism (Table 1). The administration of the thyreostatic drug thiamazole was stopped because of its apparent inefficacy. Other classic pharmacologic therapies for thyrotoxic storm were not withheld in our patient, including higher doses of ß-adrenergic blockers to ameliorate the manifestations of thyroid hormone excess because of the patient's history of severe asthma. Thyroidectomy is a high-risk procedure immediately after sleeve pneumonectomy. Glucocorticoids, given on the basis of presumed increased glucocorticoid requirements and reduced adrenal reserve, were not administered, because they may cause and aggravate myopathy [2] or increase postoperative complications at the tracheal anastomosis, namely, infection or dishence. The last option was plasmapheresis, which also has a therapeutic window.

On the 12th day, after the diagnosis of increasing thyroid hormones and the exclusion of Guillain-Barré syndrome, plasmapheresis was performed on 9 consecutive days. On day 19, the administration of potassium perchlorate and propylthiouracil was stopped. The clinical manifestations of the thyroid storm ameliorated, and the patient left the ICU 39 days after the procedure. She was discharged on day 59 to a revalidation center. After a follow-up of 17 months, no new periods of thyrotoxicosis have been observed.


    Comment
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 Abstract
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 Comment
 References
 
The term thyrotoxic storm is applied to a specific clinical syndrome marked by the exaggerated manifestation of thyrotoxicosis [1]. Typical manifestations include fever, tachycardia, atrial fibrillation out of proportion of the fever, central nervous system signs, gastrointestinal dysfunction, hypoglycemia, and hypercalcemia, which were all observed in our patient. Moreover, our patient presented with severe muscular weakness without polyneuropathy, which has been reported occasionally as a clinical manifestation of a thyrotoxic storm [3].

Most patients with thyrotoxic storms have histories of precipitating events that initiated hyperthyroidism into the clinical syndrome of thyrotoxic storm. Events associated with precipitation include sepsis, ketoacidosis, embolism, major surgical procedures, trauma, and the administration of iodinated contrast dyes [1]. In our patient, the precipitating events were sepsis (although treated and not important at the moment of clinical exacerbation) and mainly a recent major surgical procedure, that is, sleeve pneumonectomy. Amiodarone, known to influence thyroid function, had not been used to treat atrial fibrillation. Thyrotoxicosis has been reported occasionally after a thoracic surgical procedure [4]. The risk for a postoperative thyrotoxic crisis can be minimized by adequate preoperative medical treatment. The sleeve pneumonectomy in our patient was performed once a stable clinical condition and acceptable thyroid values were obtained. Apparently, this major nonthyroid surgical procedure precipitated the thyroid storm. The pathogenesis of the operation-related precipitation of a thyrotoxic storm may be related to the appearance of circulating inhibitors of hormone binding to their circulating binding proteins [1].

The administration of the appropriate doses of antithyroid drugs did not result in the amelioration of thyroid hormone levels or in clinical improvement.

In addition, both thyroid hormone and circulating inhibitors of hormone binding may be removed rapidly from the peripheral circulation by plasmapheresis [5]. During plasmapheresis, plasma separated from cellular components in blood is filtered by passing through a second filter, which removes the desired components. Although debatable, plasmapheresis has been used occasionally to treat patients with thyrotoxic storms. However, plasmapheresis increases the risk for serious infections related to the removal of antibodies and immune complexes, which may be essential in host defenses [5].

Plasmapheresis was used successfully in our patient, resulting in a decline of thyroid hormone levels (both FT3 and FT4) and improvements of both the general well-being and the myopathy of the patient, with apparently no major side effects.

Therapeutic plasmapheresis has been used rarely in the general postoperative period, because both its immunosuppressive effect, with an increased risk for infection, and its hemodynamic instability compromise its widespread usage. It has been used successfully to treat thyrotoxic storm in the postoperative period after thyroid surgical procedures [5], but not after nonthyroid procedures. Plasmapheresis is used occasionally in the postoperative period for other indications, such as after thymectomy for myasthenia gravis [6] and during cardiopulmonary bypass for removing antibodies in presensitized cardiac transplantation patients [7]. The use of plasmapheresis has been reported only once after a noncardiac thoracic surgical procedure, namely, in a 62-year-old patient for the treatment of ataxia after right middle lobe resection for a small cell carcinoma [8]. This treatment not only improved the ataxia related to pathogenic paraneoplastic antibody production but apparently also removed antitumoral factors, which could have been partly responsible for the observed early tumor reoccurrence.

Plasmapheresis might be considered a valid alternative to treat the potentially lethal condition of thyrotoxic storm in nonthyroid cardiothoracic postoperative patients. In our patient, who had undergone a sleeve pneumonectomy, it was used successfully when other treatment options were either contraindicated or ineffective.


    References
 Top
 Abstract
 Introduction
 Comment
 References
 

  1. Stathatos N., Wartofsky L. Thyrotoxic storm. Analytic review. J Intensive Care Med 2002;17:1-7.[Abstract]
  2. Hund E. Myopathy in critical ill patients. Crit Care Med 1999;27:2544-2547.[Medline]
  3. Gonzalez-Trevino O., Rosas-Guzman J. Normokalemic thyrotoxic periodic paralysis: a new therapeutic strategy. Thyroid 1999;9:61-63.[Medline]
  4. Miyakawa M., Sato K., Hasegawa M., et al. Severe thyrotoxicosis induced by thyroid metastasis of lung carcinoma: a case report and review of the literature. Thyroid 2001;11:883-888.[Medline]
  5. Martin C., Auffray J.P., Rocca B., Dumas D., Gouin F. Post-operative thyroid storm treated by plasmapheresis. Anesth Analg 1981;38:713-715.
  6. Batocchi A.P., Evoli A., Di Schino C., Tonali P. Therapeutic apheresis in myasthenia gravis. Ther Apher 2000;4:275-279.[Medline]
  7. Larson D.F., Elkund D.K., Arabia F., Copeland J.G. Plasmapheresis during cardiopulmonary bypass: proposed treatment for presensitized cardiac transplantation patients. J Extracorpor Technol 1999;31:177-183.[Medline]
  8. Miyamoto K., Kato T., Watanabe H., Miyamoto E., Suzuki S. A case of paraneoplastic syndrome accompanied by two types of cancer. J Neurol Neurosurg Psychiatry 2002;72:408-409.[Abstract/Free Full Text]




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