Recurrence of non-cardiogenic pulmonary edema and sustained hypotension shock in cystic pheochromocytoma

Abstract

Pheochromocytoma is a rare neuroendocrine tumor which derives from chromaffin cells of the adrenal gland or relevant to sympathetic nerves and ganglia. The clinical features of pheochromocytoma are various. Paroxysmal episodes of serious hypertension, headache, palpitation, and diaphoresis are the typical manifestations (Bravo, 2004). Hypotension shock, pulmonary edema, and acute coronary syndrome induced by pheochromocytoma are uncommon (Malindretos et al., 2008;Batisse-Lignier et al., 2015). In this study, we present a rare case of cystic pheochromocytoma causing recurrent hypotension shock, non-cardiogenic pulmonary edema, and acute coronary syndrome, and the possible mechanisms are discussed.A 54-year-old woman presented to the emergency department with recurrent chest pain and dyspnea for 2 years, which have been aggravated for 4 h. She had no complaints of headache, cough, or palpitation. She had a history of pollen allergy but no other disease. On physical examination, the patient was afebrile. Her heart rate was 95 beats/min, blood pressure was 105/78 mmHg, respiratory rate was 21 breaths per minute, and oxygen saturation was 93% in ambient air. Much moist rale was audible, and examination of heart and abdomen had no abnormal findings. Emergency troponin I increased (2.34 µg/L; normal range 0-0.04 µg/L). An initial electrocardiogram (ECG) showed V4-V6 ST depression. Coronary angiography and computed tomography (CT) pulmonary angiogram were normal. Contrast-enhanced CT of the chest showed diffuse lesions in the lungs, large numbers of alveoli with inflammatory exudation (Fig. S1). IgE was positive, and influenza A and B virus antigens were negative. Brain natriuretic peptide was normal (62.7 pg/ml; normal range 0-100 pg/ml). The transthoracic echocardiogram was normal and left ventricular ejection fraction was 71.7%. She developed shock with hypotension (68/34 mmHg), and was stabilized with dopamine and norepinephrine for about 30 h. The initial diagnosis was hypersensitivity pneumonitis. After treatment with methylprednisolone and antibiotics, the patient got well and was discharged. One week later, serious chest pain and dyspnea attacked again, and abdomen B ultrasound showed weak echo mass (about 4 cm×3.5 cm×3.5 cm) in the retroperitoneum. Contrast-enhanced magnetic resonance imaging showed a cystic lesion of the left adrenal gland, which was considered as pheochromocytoma (Fig. 1). The patient recovered after surgery, and histological analysis (Fig. 2) confirmed the diagnosis of pheochromocytoma. At 1 year of follow-up, there was no recurrence of chest pain.Pheochromocytoma has an estimated incidence of 0.005%-0.100% in the worldwide population and 0.1%-0.6% in the hypertension population (Omura et al.,

Left ventricular depression and pulmonary edema in rats after short-term normobaric hypoxia: effects of adrenergic blockade and reduced fluid load

“…All experiments were performed on 147 female Sprague-Dawley rats supplied by Charles River (Sulzfeld, Germany). The body weight was 225 ± 23 g at the beginning of the study corresponding to an age of about 12…”

“…High plasma levels of catecholamines (CA) may also elicit pulmonary injury with PE and inflammation as demonstrated in experiments with intravenous (i.v.) CA administration [ 27 , 36 ] or observed in clinical conditions such as pheochromocytoma [ 12 , 48 ] or neurogenic PE [ 34 , 42 ]. In rats, infusion of adrenergic agonists induced development of PE and a significant increase of IL-1 and IL-6 mRNA in the lung [ 37 , 39 ].…”

Pheochromocytoma crisis with refractory Acute Respiratory Distress Syndrome (ARDS), Takotsubo syndrome, emergency adrenalectomy, and need for Extracorporeal Membrane Oxygenation (ECMO) in a previously undiagnosed and asymptomatic patient, due to the use of metoclopramide

“…In most patients, pulmonary edema is cardiogenic and caused by cardiomyopathy, myocardial infarction, and severe arrhythmias. Noncardiogenic pulmonary edema was thought to be the result of a catecholamine-induced transient increase in pulmonary capillary pressure caused by pulmonary venoconstriction and altered pulmonary capillary permeability [ 19 , 20 ]. Sukoh reported a patient with pheochromocytoma complicated by acute noncardiogenic pulmonary edema.…”

Hypoxia-Induced Pulmonary Injury—Adrenergic Blockade Attenuates Nitrosative Stress and Inflammation but Not Pulmonary Edema

“…However, strong sympathetic activation may induce PE even in normoxic conditions. In human pathology, neurogenic PE or PE as a complication of pheochromocytoma are well-known examples of catecholamine-induced PE [34][35][36][37]. In rats infused with norepinephrine (NE) or other adrenergic agonists, we observed development of moderate interstitial edema after 6-8 h of infusion, which was accompanied by histological signs of inflammation in the lungs and increased concentrations of proinflammatory cytokines in serum and pleural fluid.…”