Case Report
SUBACUTE THYROIDITIS REVEALING A PHEOCHROMOCYTOMA Anna Furmaniuk1; Lea Demarquet1; Marc Klein, MD, PhD1; Georges Weryha, MD, PhD1; Eva Feigerlova, MD, PhD1,2 ABSTRACT Objective: Subacute thyroiditis is an inflammatory thyroid disorder, and its co-existence with pheochromocytoma is uncommon. Both diagnostic entities have similar clinical signs, which can mislead a correct diagnosis. Methods: We report a case of a patient with pheochromocytoma revealed by subacute thyroiditis and describe the clinical course and management. Results: Hyperthyroidism in our patient was characterized by association of elevated erythrocyte sedimentation rate, high serum free thyroxin and free triiodothyronine levels, low thyroid-stimulating hormone, and negativity of circulating thyroperoxidase antibodies and thyrotropin-receptor antibodies, in the presence of low thyroidal technetium uptake. Retrieval of past medical history revealed discovery of a left adrenal mass on ultrasonography performed 2 years prior for occasional hypertension. The catecholamine-induced pro-inflammatory state seems to be the result of the effects of catecholamines on humoral immunity in our patient. The therapeutic chal-
Submitted for publication April 5, 2015 Accepted for publication June 10, 2015 From the 1Centre Hospitalier Universitaire de Nancy, Service d’Endocrinologie, rue du Morvan, Vandoeuvre lès Nancy, 54400, France and 2Centre Hospitalier Universitaire de Poitiers, Médecine Interne, Diabétologie et Maladies Metaboliques, 2, rue de la Milétrie, Poitiers, 86000, France. Address correspondence to Dr. Eva Feigerlova, Centre Hospitalier Universitaire de Poitiers, Médecine Interne, Diabétologie et Maladies Métaboliques, 2, rue de la Milétrie, 86 000 Poitiers, France. E-mail:
[email protected] DOI: 10.4158/EP15764.CR To purchase reprints of this article, please visit: www.aace.com/reprints. Copyright © 2016 AACE.
Copyright © 2016 AACE
lenge concerns management of hyperthyroidism and optimal control of hypertension before surgery for pheochromocytoma. Our patient responded well to glucocorticoids and labetalol characterized by α:β-blocking action. There is no optimal medical strategy, and the choice of the treatment should be based on individualized risks and benefits. Conclusion: Our case underlines the importance of considering interactions among the adrenergic signaling pathways, thyroid hormones, and the immune system in the diagnosis and clinical workup. (AACE Clinical Case Rep. 2016;2:e161-e166) Abbreviations: α-AR and β-AR = alpha-adrenergic receptor and betaadrenergic receptor; BP = blood pressure; FT3 = free triiodothyronine; FT4 = free thyroxin; TPOAb = thyroperoxydase; TRAb = thyrotropin-receptor antibodies; TSH = thyroid-stimulating hormone; 18F-DOPA PET = 18-fluorodopa positron emission tomography INTRODUCTION The co-existence of pheochromocytoma and thyrotoxicosis is uncommon, as it has been reported only a few times in the literature. Both clinical conditions have similar symptoms; therefore, a detailed medical history and clinical and laboratory exams are necessary to make a correct diagnosis (1). In the present paper, we report a patient with pheochromocytoma revealed by subacute thyroiditis, describe the clinical management, and provide insights into the interactions between thyroid hormones, catecholamine receptor sensitivity, and the immune system. The case presented here highlights the importance of measuring catecholamine levels in patients with signs of thyrotoxicosis and intermittent abdominal pain.
AACE CLINICAL CASE REPORTS Vol 2 No. 2 Spring 2016 e161
e162 Thyroiditis and Pheochromocytoma, AACE Clinical Case Rep. 2016;2(No. 2)
CASE REPORT A 40-year-old woman was admitted to our hospital with painful swelling of the thyroid gland, weight loss, hypertension, and paroxysmal abdominal pain accompanied with flushing. Retrieval of her past medical history revealed discovery of a left adrenal mass on ultrasonography performed 2 years prior during examination in an outpatient cardiology clinic for occasional hypertension. Adrenal magnetic resonance imaging confirmed a 38-mm left hypervascular mass, which did not demonstrate any signal loss on in/out-of-phase imaging. Endocrine etiology of hypertension was suspected, and the patient was advised by her cardiologist to undergo hormonal explorations. Unfortunately, the patient was then lost to follow-up by specialist physicians, but she continued to consult the general practitioner. In the interval prior to development of subacute thyroiditis, the patient was diagnosed twice with viral gastroenteritis and once with influenza-like illness, each time with a rapid recovery. Her blood pressure (BP) taken over these visits fluctuated between 130 and 155 for the systolic component and between 85 and 100 for the diastolic component. Otherwise, the patient was free of symptoms. A few months prior to the initial visit, she started to suffer from tachycardia, headaches, weight lost, and anxiety. Three weeks prior, she had developed neck tenderness and paroxysmal abdominal pain with flushing. At the time of admission, physical examination revealed a slim female (body mass index, 19 kg/m2) with tachycardia (120 beats per minute), hypertension (190/115 mm Hg), and thyroid tenderness. The patient suffered from headaches, paroxysmal epigastric pain, and extreme anxiety. Biologic findings demonstrated low thyroid-stimulating hormone (TSH) (0.01 mUI/L), high free thyroxin (FT4) (62.6 pmol/L; reference value, 7.9 to 14.4 pmol/L), high free triiodothyronine (FT3) (18.1 pmol/l ; reference value, 4.0 to 6.7 pmol/l), negative thyrotropin-receptor antibodies (TRAb) and thyroperoxidase antibodies (TPOAb), in association with elevated erythrocyte sedimentation rate (81/111 mm/hour), and high C-reactive protein (59 mg/L). Thyroid ultrasonography (US) showed a hypoechogenic, heterogeneous gland, with no uptake on a technetium scan (Fig. 1 A). The patient underwent an abdominal computed tomography scan, which showed a left, hyper-dense (35 Hounsfield units) adrenal tumor that measured 50 mm, compatible with a pheochromocytoma. The plasma and urinary levels of normetanephrine were extremely high 216 nmol/L (reference value, 0.5 to 9.8 nmol/L) and 43,614 nmol/24 hours (reference value, 110 to 2,620 nmol/24 hours), respectively. The chromogranin A level was increased (250 ng/mL, reference value
