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Nair RK, Parikh MA, Frishman WH, Peterson SJ. Efficacy of Beta-Blockers in Acute Management and Prevention of Recurrence in Takotsubo Syndrome. Cardiol Rev 2025:00045415-990000000-00487. [PMID: 40327812 DOI: 10.1097/crd.0000000000000926] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 05/08/2025]
Abstract
Takotsubo cardiomyopathy is a nonischemic cardiomyopathy characterized by a transient ballooning of the apical cardiac wall secondary to stress. Since its discovery and diagnosis in Japan in 1990, Takotsubo cardiomyopathy has been known as a disease with a generally favorable prognosis due to the transient nature of its apical wall abnormalities. However, complications, including arrhythmia, hemodynamic instability, heart failure, intracardiac thrombus, and rupture, have all been reported in the literature, requiring flexible management of this unique pathology. Although therapy is guided by inciting factors and complication management, beta-blockers are a widely accepted acute and long-term treatment modality. We review the current literature describing the pathogenesis, medical evaluation, and treatment options for Takotsubo cardiomyopathy. This article aims to provide a greater understanding of the pathophysiology and further evaluate the efficacy of beta-blocker therapy when considering acute and long-term treatment and prevention of Takotsubo cardiomyopathy.
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Affiliation(s)
- Ranjit K Nair
- From the Department of Medicine, New York Presbyterian Brooklyn Methodist Hospital, Brooklyn, NY
| | - Manish A Parikh
- From the Department of Medicine, New York Presbyterian Brooklyn Methodist Hospital, Brooklyn, NY
- Division of Cardiology Brooklyn, Weill Department of Medicine, Weill Cornell Medicine, New York, NY
| | | | - Stephen J Peterson
- From the Department of Medicine, New York Presbyterian Brooklyn Methodist Hospital, Brooklyn, NY
- Division of Cardiology Brooklyn, Weill Department of Medicine, Weill Cornell Medicine, New York, NY
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2
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Guo N, Liu T, Zhao P, Bai S. Long-Term Cardiovascular Complications in Patients With Pheochromocytomas and Paragangliomas After Surgery: A Large Multi-Center Study. Clin Endocrinol (Oxf) 2025; 102:273-280. [PMID: 39696830 DOI: 10.1111/cen.15181] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/25/2024] [Revised: 11/29/2024] [Accepted: 12/07/2024] [Indexed: 12/20/2024]
Abstract
OBJECTIVE The effects of pheochromocytomas and paragangliomas (PPGLs)-induced catecholamine overproduction on vascular and cardiac function are generally thought to be reversible after PPGLs removal. However, a sizable proportion of patients who were free of the recurrent disease still faced high risks of cardiovascular problems after successful surgery. We aim to identify incidence and risk factors for long-term cardiovascular complications in PPGLs patients after surgery. DESIGN We retrospectively reviewed 602 patients who underwent surgery for sporadic PPGLs at three centers between January 2012 and October 2022. Demographic characteristics and perioperative data were recorded. Multiple logistic regression was used to determine the risk factors for postoperative long-term cardiovascular complications. RESULTS Finally, a total of 602 patients were included in the analysis, comprising 460 (76.4%) patients with pheochromocytomas and 142 (23.6%) patients with paragangliomas. After a median follow-up of 64 months, 76 (12.6%) patients had developed long-term cardiovascular complications. Independent risk factors included pheochromocytomas (odds ratio [OR] = 4.13, 95% confidence interval [CI]: 1.425-11.965, p = 0.009), had low preoperative left ventricular ejection fraction (LVEF, OR = 5.659, 95% CI: 2.141-14.955, p < 0.001), experienced intraoperative hemodynamic instability (HDI, OR = 2.498, 95% CI: 1.423-4.385, p = 0.001), suffered from postoperative in-hospital cardiovascular complications (OR = 5.723, 95% CI: 2.078-15.758, p = 0.001) and long-term persistent hypertension (OR = 3.552, 95% CI: 1.580-7.988, p = 0.002). CONCLUSIONS Long-term cardiovascular complications commonly occur in patients with surgical-cured PPGLs. Pheochromocytomas, had low preoperative LVEF, experienced intraoperative HDI, suffered from postoperative in-hospital cardiovascular complications, and persistent hypertension were determined as the risk factors for long-term cardiovascular complications. These findings may help to improve follow-up management.
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Affiliation(s)
- Nan Guo
- Department of Anesthesiology, Shengjing Hospital of China Medical University, Shenyang, China
| | - Tao Liu
- Department of Urology, The First Hospital of China Medical University, Shenyang, China
| | - Ping Zhao
- Department of Anesthesiology, Shengjing Hospital of China Medical University, Shenyang, China
| | - Song Bai
- Department of Urology, Shengjing Hospital of China Medical University, Shenyang, China
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Li G, Lin M, Zhou F, Li Q. Bidirectional Ventricular Tachycardia due to Pheochromocytoma: A Case Report. Ann Noninvasive Electrocardiol 2025; 30:e70063. [PMID: 40070271 PMCID: PMC11897606 DOI: 10.1111/anec.70063] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/31/2025] [Revised: 02/16/2025] [Accepted: 02/28/2025] [Indexed: 03/15/2025] Open
Abstract
Pheochromocytoma, a type of neuroendocrine tumor, can cause numerous symptoms and signs similar to those of other clinical conditions, with the classic triad being palpitations, headache, and diaphoresis. Patients with pheochromocytoma can present with various cardiac complications, including myocarditis, acute coronary syndromes, cardiomyopathy, heart failure, and arrhythmias. Here we report a case of pheochromocytoma that first presented with bidirectional ventricular tachycardia. The patient was initially diagnosed with acute viral myocarditis and was treated accordingly. A pheochromocytoma crisis with severe blood pressure fluctuation occurred after glucocorticoid administration, leading to further diagnostic work-up, which eventually revealed the adrenal pheochromocytoma.
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Affiliation(s)
- Gui‐yang Li
- Xiamen Cardiovascular Hospital, Division of CardiologyXiamen UniversityXiamenFujianChina
| | - Man‐Xin Lin
- Xiamen Cardiovascular Hospital, Division of CardiologyXiamen UniversityXiamenFujianChina
| | - Fa‐Guang Zhou
- Xiamen Cardiovascular Hospital, Division of CardiologyXiamen UniversityXiamenFujianChina
| | - Qiang Li
- Xiamen Cardiovascular Hospital, Division of CardiologyXiamen UniversityXiamenFujianChina
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Ali NA, Calissendorff J, Falhammar H. Sex differences in presentation of pheochromocytoma and paraganglioma. Front Endocrinol (Lausanne) 2025; 16:1463945. [PMID: 39917539 PMCID: PMC11798811 DOI: 10.3389/fendo.2025.1463945] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/12/2024] [Accepted: 01/07/2025] [Indexed: 02/09/2025] Open
Abstract
Purpose The aim of the study was to investigate sex differences in presentation of pheochromocytomas and paragangliomas (PPGLs). Methods This is a retrospective cohort study including 183 patients with confirmed PPGL (females n=100, pheochromocytoma n=156) between year 2005 and 2023, attending Department of Endocrinology, Karolinska University Hospital, Stockholm. The collected data included the mode of presentation, symptoms, biochemical, genetic and histopathological test results. Results The mean age at surgery/diagnosis was 54.9 ± 17.0 years. Sweating was more common in females compared to males (44% vs 23%, p=0.003), and also takotsubo syndrome (10% vs 0% p=0.002). Males, on the other hand, were more likely to experience pallor (16% vs 4%, p=0.009), and were more often diagnosed due to investigations of a suspected PPGL (31% vs 18%, p=0.039) although no difference was found in the classic triad (sweating, palpitations and headache). Left-sided pheochromocytoma was more common among males than females (48% vs 29%, p=0.009). No differences between sexes were found in biochemical, genetical or histopathological results, or presence of metastasis at diagnosis. Conclusions The reported symptoms by patients with PPGL were generally similar between the sexes, except for pallor and sweating. Takotsubo syndrome was more common among females. More males with PPGL were found based on suspicion than females. Further research into sex differences in various aspects of PPGL should be pursued.
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Affiliation(s)
- Nora Azin Ali
- Department of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden
| | - Jan Calissendorff
- Department of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden
- Department of Endocrinology, Karolinska University Hospital, Stockholm, Sweden
| | - Henrik Falhammar
- Department of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden
- Department of Endocrinology, Karolinska University Hospital, Stockholm, Sweden
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5
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Berends AMA, Lenders JWM, Kerstens MN. Update on clinical characteristics in the evaluation of phaeochromocytoma and paraganglioma. Best Pract Res Clin Endocrinol Metab 2024; 38:101953. [PMID: 39384447 DOI: 10.1016/j.beem.2024.101953] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 10/11/2024]
Abstract
Pheochromocytomas and sympathetic paragangliomas (PPGL) are rare neuroendocrine tumors originating from chromaffin tissue of the adrenal medulla and extra-adrenal sympathetic paraganglia. Historically, many of these tumors were diagnosed postmortem, earning pheochromocytomas the moniker "great mimic" due to their diverse clinical manifestations that can resemble various other conditions. Over time, the clinical presentation of PPGL has evolved, with a shift from symptomatic or postmortem diagnoses to more frequent incidental discoveries or diagnoses through screening, with postmortem identification now being rare. The development of a clinical scoring system has improved the identification of patients at increased risk for PPGL. Notably, the proportion of PPGL patients with normal blood pressure ranges from 15 % to 40 %, varying based on the clinical context. Despite the tumor's reputation, PPGL is an exceedingly rare cause of resistant hypertension. Management of a pheochromocytoma crisis has advanced, with several classes of drugs available for treatment. However, PPGL during pregnancy remains a significant concern, associated with substantial maternal and fetal mortality rates. Additionally, PPGL can present as rare disorders, including catecholamine-induced cardiomyopathy, Cushing syndrome, and urinary bladder PGL. Given these varied presentations, heightened awareness and prompt recognition of PPGL are crucial for timely diagnosis and treatment, ultimately improving patient outcomes. In this article, we offer an in-depth analysis of the diverse clinical presentations of PPGL, highlighting their complexity and the associated diagnostic and treatment strategies.
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Affiliation(s)
- Annika M A Berends
- Department of Endocrinology, University of Groningen, University Medical Center Groningen, Groningen, the Netherlands.
| | - Jacques W M Lenders
- Department of Internal Medicine, Radboud University Medical Center, Nijmegen, the Netherlands
| | - Michiel N Kerstens
- Department of Endocrinology, University of Groningen, University Medical Center Groningen, Groningen, the Netherlands
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Bouamoud A, Zaim C, Kerrouani W, Haddioui ME, Bouissou K, Bouazaze M, Amri R. [A rare case of recurrent myocarditis revealing pheochromocytoma]. Ann Cardiol Angeiol (Paris) 2024; 73:101768. [PMID: 38788259 DOI: 10.1016/j.ancard.2024.101768] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/02/2024] [Revised: 04/05/2024] [Accepted: 04/05/2024] [Indexed: 05/26/2024]
Abstract
Pheochromocytoma is a rare neuroendocrine tumor characterized by overproduction of catecholamines. The overproduction of catecholamines leads to cardiac remodeling which manifests in several forms ranging from Takotsubo to dilated cardiomyopathy. Studies suggest that pheochromocytoma-induced cardiomyopathy can take various forms depending on the duration of catecholamine exposure. Myocarditis is a fairly rare presentation of cardiac manifestations of pheochromocytoma which are mainly dominated by Takotsubo and dilated cardiomyopathies. We report a rare case of recurrent myocarditis in a young 37-year-old patient revealing the diagnosis of adrenal pheochromocytoma. Through this case and through a review of the literature we will take stock of the epidemiology of cardiac involvement in pheochromocytoma, mainly cardiomyopathies, and we will take stock of the value of diagnosis and early management in improving the prognosis of patients.
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Affiliation(s)
- A Bouamoud
- Service de cardiologie B, centre hospitalier universitaire IBN SINA, Rabat, Maroc.
| | - C Zaim
- Service de cardiologie B, centre hospitalier universitaire IBN SINA, Rabat, Maroc.
| | - W Kerrouani
- Service de cardiologie B, centre hospitalier universitaire IBN SINA, Rabat, Maroc.
| | - M El Haddioui
- Service de cardiologie B, centre hospitalier universitaire IBN SINA, Rabat, Maroc.
| | - K Bouissou
- Service de cardiologie B, centre hospitalier universitaire IBN SINA, Rabat, Maroc.
| | - M Bouazaze
- Service de cardiologie B, centre hospitalier universitaire IBN SINA, Rabat, Maroc.
| | - R Amri
- Service de cardiologie B, centre hospitalier universitaire IBN SINA, Rabat, Maroc.
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Xu M, Guan Q, Liu T, Huang Y, Pan C, Luo L, Tang W, Xu J, Huang H, Xiao L, Liu K, Chen J. In-Hospital Adverse Events of Pheochromocytoma-Induced Takotsubo Syndrome: A Literature Review and Cluster Analysis of 172 Cases. Rev Cardiovasc Med 2024; 25:216. [PMID: 39076320 PMCID: PMC11270063 DOI: 10.31083/j.rcm2506216] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/31/2023] [Revised: 03/08/2024] [Accepted: 03/13/2024] [Indexed: 07/31/2024] Open
Abstract
Background Pheochromocytoma-induced takotsubo syndrome (Pheo-TTS) significantly increases the risk of adverse events for inpatient. The early identification of risk factors at admission is crucial for effective risk stratification and minimizing complications in Pheo-TTS patients. Methods We conducted a systematic review combined with hierarchical cluster and feature importance analysis of demographic, clinical and laboratory data upon admission, alongside in-hospital complication data for Pheo-TTS patients. We analyzed cases published in PubMed and Embase from 2 May 2006 to 27 April 2023. Results Among 172 Pheo-TTS patients, cluster analysis identified two distinct groups: a chest pain dominant (CPD) group (n = 86) and a non-chest pain dominant (non-CPD) group (n = 86). The non-CPD group was characterized by a younger age (44.0 ± 15.2 vs. 52.4 ± 14.4, p < 0.001), a higher prevalence of neurological/psychiatric disorders (53.5% vs. 32.6%), and increased presentation of dyspnea (87.2% vs. 17.4%), pulmonary rales (59.3% vs. 8.1%), and tachycardia (77.9% vs. 30.2%). Additionally, they exhibited more atypical takotsubo syndrome (TTS) imaging phenotypes (55.8% vs. 36.5%, all p < 0.05). The non-CPD group experienced more than a 2-fold increase for in-hospital adverse events compared to the CPD group (70.9% vs. 30.2%, p < 0.001). After adjusting for confounding factors, the absence of chest pain (odds ratio [OR] = 0.407, 95% confidence interval [CI] 0.169-0.979, p = 0.045), the presence of abdominal symptoms (OR = 3.939, 95% CI 1.770-8.766, p = 0.001), pulmonary rales (OR = 4.348, 95% CI 1.857-10.179, p = 0.001), and atypical TTS imaging phenotype (OR = 3.397, 95% CI 1.534-7.525, p = 0.003) remained as independent predictors of in-hospital complications. Conclusions Clinical manifestations and imaging features at admission help to predict in-hospital complications for Pheo-TTS patients.
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Affiliation(s)
- Mei Xu
- Department of Cardiovascular Medicine, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
| | - Qianglin Guan
- Department of Cardiovascular Medicine, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
| | - Tianmin Liu
- Department of Cardiovascular Medicine, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
| | - Yuxi Huang
- Department of Cardiovascular Medicine, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
| | - Cunxue Pan
- Department of Radiology, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
| | - Liyun Luo
- Department of Cardiovascular Medicine, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
- Center for Interventional Medicine, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
| | - Wenyi Tang
- Department of Cardiovascular Medicine, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
| | - Junwei Xu
- Department of Cardiovascular Medicine, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
| | - Hsi Huang
- Department of Cardiovascular Medicine, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
| | - Li Xiao
- Department of Cardiovascular Medicine, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
| | - Kan Liu
- Division of Cardiology, Heart and Vascular Center, Washington University in St Louis, Barnes-Jewish Hospital, St Louis, MO 63110, USA
| | - Jian Chen
- Department of Cardiovascular Medicine, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
- Center for Interventional Medicine, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
- Guangdong Provincial Engineering Research Center of Molecular Imaging, The Fifth Affiliated Hospital of Sun Yat-sen University, 519000 Zhuhai, Guangdong, China
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8
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Madias JE. Frequent POCUS and auscultation for an earlier diagnosis of takotsubo syndrome and unraveling of its pathophysiology: The possible crucial role of LVOTO. Curr Probl Cardiol 2024; 49:102482. [PMID: 38401826 DOI: 10.1016/j.cpcardiol.2024.102482] [Citation(s) in RCA: 10] [Impact Index Per Article: 10.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/21/2024] [Accepted: 02/21/2024] [Indexed: 02/26/2024]
Abstract
There is ample literature associating LVOTO with hypertension, AMI, LV hypertrophy, sigmoid septum, HCM, and TTS, particularly in midde aged/elderly/postmenopausal women, suggestive of a causal role for LVOTO in the pathophysiology of TTS. Although there is significant evidence that TTS is triggered by a sudden autonomic sympathetic nervous system surge and/or elevated blood-ridden catecholamines, the exact pathophysiologic trajectory leading to the clinical expression of the disease is still being debated. This review expounds on the possibility that LVOTO is a causal early component of this trajectory, and proposes that TTS is a malady within the broad spectrum of the myocardial ischemic injury/stunned myocardium states. The postulated underlying mechanism by which LVOTO causes TTS is a sudden abterload rise, with resultant oxygen/energy supply/demand mismatch, leading to a transient myocardial ischemia/injury myocardial stunning state. This needs to be explored painstakingly, and this review includes some suggestions for such undertaking. Ellucidation of the pathophysiology of TTS, and possible proof about a mechanistic role of LVOTO, may ensure that our current pharmacological and device panoply is adequate for the management of TTS.
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Affiliation(s)
- John E Madias
- Icahn School of Medicine at Mount Sinai, New York, NY, United States; Division of Cardiology, Elmhurst Hospital Center, Elmhurst, NY, United States.
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Manhaeghe L, Goethals AM, Van Aerde N, Frederiks P, Sinnaeve P, Janssens S, Willems R. Pheochromocytoma leading to Takotsubo and Ogilvie syndrome, a pathophysiological triad. Acta Cardiol 2024; 79:109-113. [PMID: 37823390 DOI: 10.1080/00015385.2023.2268438] [Citation(s) in RCA: 2] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/05/2023] [Accepted: 10/03/2023] [Indexed: 10/13/2023]
Abstract
Takotsubo syndrome (TTS) is a transient left ventricle dysfunction usually caused by a stressful trigger (emotional or physical). We report the case of a 77 year-old female patient who presented with TTS caused by a pheochromocytoma, a catecholamine-producing neuroendocrine tumour. Diagnosis was facilitated by acute kidney injury prompting renal ultrasound, recurrence of TTS and symptoms of episodic palpitations, profuse sweating and labile blood pressure. Furthermore, during her hospitalisation the patient also developed an Ogilvie syndrome, an acute colonic pseudo-obstruction, due to the catecholamine-excess. Treatment consisted of betablocker and angiotensin-converting enzyme inhibitor for TTS, neostigmine for Ogilvie syndrome, in combination with alpha-blocker and surgical removal of the tumour after recuperation of left ventricular function and colonic pseudo-obstruction. To our knowledge, this is the first case report of the pathophysiological triad of pheochromocytoma leading to Takotsubo and Ogilvie syndrome in a single patient.
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Affiliation(s)
- Lynn Manhaeghe
- Department of Cardiovascular Diseases, University Hospitals Leuven, Leuven, Belgium
| | | | - Nathalie Van Aerde
- Department of Cardiovascular Diseases, University Hospitals Leuven, Leuven, Belgium
| | - Pascal Frederiks
- Department of Cardiovascular Diseases, University Hospitals Leuven, Leuven, Belgium
| | - Peter Sinnaeve
- Department of Cardiovascular Diseases, University Hospitals Leuven, Leuven, Belgium
- Department of Cardiovascular Sciences, KU Leuven - University of Leuven, Leuven, Belgium
| | - Stefan Janssens
- Department of Cardiovascular Diseases, University Hospitals Leuven, Leuven, Belgium
- Department of Cardiovascular Sciences, KU Leuven - University of Leuven, Leuven, Belgium
| | - Rik Willems
- Department of Cardiovascular Diseases, University Hospitals Leuven, Leuven, Belgium
- Department of Cardiovascular Sciences, KU Leuven - University of Leuven, Leuven, Belgium
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10
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Carolino D'Araujo S, Dendievel R. Coronary artery compression by a mediastinal paraganglioma - clinical case report. Acta Cardiol 2024; 79:85-86. [PMID: 37882607 DOI: 10.1080/00015385.2023.2273083] [Citation(s) in RCA: 1] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/28/2023] [Accepted: 09/01/2023] [Indexed: 10/27/2023]
Affiliation(s)
| | - Renaud Dendievel
- Department of Cardiology, Erasme University Hospital, Brussels, Belgium
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11
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Szatko A, Glinicki P, Gietka-Czernel M. Pheochromocytoma/paraganglioma-associated cardiomyopathy. Front Endocrinol (Lausanne) 2023; 14:1204851. [PMID: 37522121 PMCID: PMC10374018 DOI: 10.3389/fendo.2023.1204851] [Citation(s) in RCA: 13] [Impact Index Per Article: 6.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/12/2023] [Accepted: 06/26/2023] [Indexed: 08/01/2023] Open
Abstract
Pheochromocytoma/paraganglioma (PPGL) are neuroendocrine tumors that frequently produce and release catecholamines. Catecholamine excess can manifest in several cardiovascular syndromes, including cardiomyopathy. PPGL-induced cardiomyopathies occur in up to 11% of cases and are most often associated with an adrenal pheochromocytoma (90%) and rarely with a paraganglioma derived from the sympathetic ganglia (10%). PPGL-associated cardiomyopathies can be chronic or acute, with takotsubo cardiomyopathy being the most often reported. These two types of PPGL-induced cardiomyopathy seem to have different pathophysiological backgrounds. Acute catecholaminergic stress inundates myocardial β-adrenoceptors and leads to left ventricle stunning and slight histological apoptosis. In chronic cardiomyopathy, prolonged catecholamine exposure leads to extended myocardial fibrosis, inflammation, and necrosis, and ultimately it causes dilated cardiomyopathy with a low ejection fraction. Sometimes, especially in cases associated with hypertension, hypertrophic cardiomyopathy can develop. The prognosis appears to be worse in chronic cases with a higher hospital mortality rate, higher cardiogenic shock rate at initial presentation, and lower left ventricular recovery rate after surgery. Therefore, establishing the correct diagnosis at an early stage of a PPGL is essential. This mini-review summarizes current data on pathophysiological pathways of cardiac damage caused by catecholamines, the clinical presentation of PPGL-induced cardiomyopathies, and discusses treatment options.
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Affiliation(s)
- Alicja Szatko
- Department of Endocrinology, Centre of Postgraduate Medical Education, Warsaw, Poland
- EndoLab Laboratory, Centre of Postgraduate Medical Education, Warsaw, Poland
| | - Piotr Glinicki
- Department of Endocrinology, Centre of Postgraduate Medical Education, Warsaw, Poland
- EndoLab Laboratory, Centre of Postgraduate Medical Education, Warsaw, Poland
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12
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Falhammar H. Specialty grand challenge in adrenal endocrinology. Front Endocrinol (Lausanne) 2023; 14:1237733. [PMID: 37484952 PMCID: PMC10358978 DOI: 10.3389/fendo.2023.1237733] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/09/2023] [Accepted: 06/26/2023] [Indexed: 07/25/2023] Open
Affiliation(s)
- Henrik Falhammar
- Department of Endocrinology, Karolinska University Hospital, Stockholm, Sweden
- Department of Molecular Medicine and Surgery, Karolinska Intitutet, Stockholm, Sweden
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13
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Akhtar MM, Cammann VL, Templin C, Ghadri JR, Lüscher TF. Takotsubo syndrome: getting closer to its causes. Cardiovasc Res 2023:7161872. [PMID: 37183265 DOI: 10.1093/cvr/cvad053] [Citation(s) in RCA: 14] [Impact Index Per Article: 7.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/08/2022] [Revised: 01/18/2023] [Accepted: 02/07/2023] [Indexed: 05/16/2023] Open
Abstract
Takotsubo syndrome (TTS) accounts for between 1 and 4% of cases presenting clinically as an acute coronary syndrome. It typically presents as a transient cardiac phenotype of left ventricular dysfunction with spontaneous recovery. More dramatic presentations may include cardiogenic shock or cardiac arrest. Despite progress in the understanding of the condition since its first description in 1990, considerable questions remain into understanding underlying pathomechanisms. In this review article, we describe the current published data on potential underlying mechanisms associated with the onset of TTS including sympathetic nervous system over-stimulation, structural and functional alterations in the central nervous system, catecholamine secretion, alterations in the balance and distribution of adrenergic receptors, the additive impact of hormones including oestrogen, epicardial coronary or microvascular spasm, endothelial dysfunction, and genetics as potentially contributing to the cascade of events leading to the onset. These pathomechanisms provide suggestions for novel potential therapeutic strategies in patients with TTS including the role of cognitive behavioural therapy, beta-blockers, and endothelin-A antagonists. The underlying mechanism of TTS remains elusive. In reality, physical or emotional stressors likely trigger through the amygdala and hippocampus a central neurohumoral activation with the local and systemic secretion of excess catecholamine and other neurohormones, which exert its effect on the myocardium through a metabolic switch, altered cellular signalling, and endothelial dysfunction. These complex pathways exert a regional activation in the myocardium through the altered distribution of adrenoceptors and density of autonomic innervation as a protective mechanism from myocardial apoptosis. More research is needed to understand how these different complex mechanisms interact with each other to bring on the TTS phenotype.
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Affiliation(s)
- Mohammed Majid Akhtar
- Royal Brompton and Harefield Hospitals, Imperial College and King's College, London SW3 6NP, UK
| | - Victoria L Cammann
- University Heart Center, Department of Cardiology, University Hospital Zürich, Zürich 8091, Switzerland
| | - Christian Templin
- University Heart Center, Department of Cardiology, University Hospital Zürich, Zürich 8091, Switzerland
| | - Jelena R Ghadri
- University Heart Center, Department of Cardiology, University Hospital Zürich, Zürich 8091, Switzerland
| | - Thomas F Lüscher
- Royal Brompton and Harefield Hospitals, Imperial College and King's College, London SW3 6NP, UK
- Center for Molecular Cardiology, University of Zürich, Zürich 8952, Switzerland
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A systematic cohort review of pheochromocytoma-induced typical versus atypical Takotsubo cardiomyopathy. Int J Cardiol 2023; 371:287-292. [PMID: 36055473 DOI: 10.1016/j.ijcard.2022.08.053] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/16/2022] [Revised: 08/21/2022] [Accepted: 08/24/2022] [Indexed: 12/14/2022]
Abstract
BACKGROUND A rare presentation of pheochromocytoma (PCC) is catecholamine-induced-cardiomyopathy, or Takotsubo cardiomyopathy (TCM). PCC-induced TCM(PCC-TCM) can present as a typical or atypical type, based on the location of cardiac wall motion abnormalities. In this review, we sought to assess features and outcomes for PCC-TCM, and to compare typical and atypical subtypes. METHODS A search was conducted on two databases (PubMed and Embase) for case series or reports on PCC-TCM from 2006 to 2020. RESULTS One-hundred-and-two papers with a total of 104 cases of PCC-TCM were retrieved: 67(64.4%) typical and 37(35.6%) atypical subtypes. Overall median age was 50[range:23-86] years, the atypical group about a decade younger(p < 0.001). A female preponderance was seen for either subtype (∼75%). The most common presentations were chest pain(n = 60;58%), dyspnoea(n = 46;44%), and headache(n = 41;39.4%). Those with atypical subtype more often presented with fluid overload (typical:3% versus atypical:60%); acute pulmonary oedema (35% versus 60%); and cardiogenic shock (22% versus 43%) (all p < 0.05). Six patients (6%) died pre-operatively (typical:8% versus atypical:3%; p = 0.32). Non-fatal pre-operative complications occurred more among those with atypical TCM(p < 0.001), specifically cardiac arrest (typical:5% versus atypical:32%) and respiratory failure (9% versus 24%; both p < 0.05). Overall, 98 underwent surgery, majority undergoing laparoscopic adrenalectomy (81%); similar among the subtypes(p = 0.71). No robust data was provided on short-term outcomes, although two patients suffered from post-operative complications. CONCLUSION Although quite similar in presentation to either standalone TCM or PCC, PCC-TCM seems to be associated with a higher degree of morbidity and mortality. The atypical PCC-TCM subgroup seems to have a more severe course with possibly a poorer outcome. Further research is needed to make more reliable inferences.
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15
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Gevaert SA, Halvorsen S, Sinnaeve PR, Sambola A, Gulati G, Lancellotti P, Van Der Meer P, Lyon AR, Farmakis D, Lee G, Boriani G, Wechalekar A, Okines A, Asteggiano R, Combes A, Pfister R, Bergler-Klein J, Lettino M. Evaluation and management of cancer patients presenting with acute cardiovascular disease: a Clinical Consensus Statement of the Acute CardioVascular Care Association (ACVC) and the ESC council of Cardio-Oncology—part 2: acute heart failure, acute myocardial diseases, acute venous thromboembolic diseases, and acute arrhythmias. EUROPEAN HEART JOURNAL. ACUTE CARDIOVASCULAR CARE 2022; 11:865-874. [DOI: 10.1093/ehjacc/zuac107] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Received: 08/19/2022] [Accepted: 08/29/2022] [Indexed: 12/05/2022]
Abstract
Abstract
Advances in treatment, common cardiovascular (CV) risk factors and the ageing of the population have led to an increasing number of cancer patients presenting with acute CV diseases. These events may be related to cancer itself or cancer treatment. Acute cardiac care specialists must be aware of these acute CV complications and be able to manage them. This may require an individualized and multidisciplinary approach. The management of acute coronary syndromes and acute pericardial diseases in cancer patients was covered in part 1 of a clinical consensus document. This second part focusses on acute heart failure, acute myocardial diseases, venous thromboembolic diseases and acute arrhythmias.
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Affiliation(s)
- Sofie A Gevaert
- Department of Cardiology, Ghent University Hospital , Gent , Belgium
| | - Sigrun Halvorsen
- Department of Cardiology, Oslo University Hospital Ulleval, and University of Oslo , Oslo , Norway
| | - Peter R Sinnaeve
- Department of Cardiology, University Hospital Leuven , Leuven , Belgium
| | - Antonia Sambola
- Department of Cardiology, University Hospital Vall d’Hebron, Universitat Autonòma, CIBER-CV , Barcelona , Spain
| | - Geeta Gulati
- Department of Cardiology, Oslo University Hospital Ulleval, and University of Oslo , Oslo , Norway
| | - Patrizio Lancellotti
- University of Liège Hospital, GIGA Cardiovascular Science, Department of Cardiology , CHU Sart Tilman, Liège , Belgium
| | - Peter Van Der Meer
- Department of Cardiology, University of Groningen, University Medical Center Groningen , Groningen , The Netherlands
| | - Alexander R Lyon
- Cardio-Oncology Clinic at Royal Brompton Hospital and Imperial College London , London , UK
| | | | - Geraldine Lee
- Florence Nightingale Faculty of Nursing, Midwifery and Palliative Care, King’s College , London , UK
| | - Giuseppe Boriani
- Cardiology Division, Department of Biomedical, Metabolic and Neural Sciences, University of Modena and Reggio Emilia , Policlinico Di Modena, Modena , Italy
| | - Ashutosh Wechalekar
- Department of Haematology, University College London/University College London Hospitals , London , UK
| | - Alicia Okines
- Department of Medicine, The Royal Marsden NHS Foundation Trust , London , UK
| | - Riccardo Asteggiano
- Insubria University , Varese , Italy
- LARC (Laboratorio Analisi e Ricerca Clinica) , Turin , Italy
| | - Alain Combes
- Medical-Surgical ICU, Hôpital Pitié–Salpêtrière , Paris , France
- Sorbonne University, Institute of Cardiometabolism and Nutrition , Paris , France
| | - Roman Pfister
- Department III of Internal Medicine, Heart Center, University of Cologne , Cologne , Germany
| | - Jutta Bergler-Klein
- Department of Cardiology, University Clinic of Internal Medicine II, Medical University of Vienna , Vienna , Austria
| | - Maddalena Lettino
- Department of Cardiology, San Gerardo Hospital, ASST-Monza , Monza , Italy
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16
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A Rare Case of a Right Atrial Paraganglioma in an Individual with the SHDB Mutation. Case Rep Cardiol 2022; 2022:1140976. [PMID: 36226034 PMCID: PMC9550431 DOI: 10.1155/2022/1140976] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/15/2022] [Revised: 07/13/2022] [Accepted: 09/13/2022] [Indexed: 11/06/2022] Open
Abstract
Paragangliomas are extra-adrenal chromaffin cell tumors. A small percentage of these tumors can be found in the thoracic cavity and, when in the heart, are typically in the left atrium. In this case report, we discuss the case of an individual with a history of several paragangliomas with the SHDB mutation who was found to have two cardiac paragangliomas in the right atrium.
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17
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Januszewicz A, Mulatero P, Dobrowolski P, Monticone S, Van der Niepen P, Sarafidis P, Reincke M, Rexhaj E, Eisenhofer G, Januszewicz M, Kasiakogias A, Kreutz R, Lenders JW, Muiesan ML, Persu A, Agabiti-Rosei E, Soria R, Śpiewak M, Prejbisz A, Messerli FH. Cardiac Phenotypes in Secondary Hypertension. J Am Coll Cardiol 2022; 80:1480-1497. [DOI: 10.1016/j.jacc.2022.08.714] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/15/2022] [Revised: 08/03/2022] [Accepted: 08/04/2022] [Indexed: 11/06/2022]
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18
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Curtiaud A, Delmas C, Gantzer J, Zafrani L, Siegemund M, Meziani F, Merdji H. Cardiogenic shock among cancer patients. Front Cardiovasc Med 2022; 9:932400. [PMID: 36072868 PMCID: PMC9441759 DOI: 10.3389/fcvm.2022.932400] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/29/2022] [Accepted: 07/04/2022] [Indexed: 11/13/2022] Open
Abstract
Sophisticated cancer treatments, cardiovascular risk factors, and aging trigger acute cardiovascular diseases in an increasing number of cancer patients. Among acute cardiovascular diseases, cancer treatment, as well as the cancer disease itself, may induce a cardiogenic shock. Although increasing, these cardiogenic shocks are still relatively limited, and their management is a matter of debate in cancer patients. Etiologies that cause cardiogenic shock are slightly different from those of non-cancer patients, and management has some specific features always requiring a multidisciplinary approach. Recent guidelines and extensive data from the scientific literature can provide useful guidance for the management of these critical patients. Even if no etiologic therapy is available, maximal intensive supportive measures can often be justified, as most of these cardiogenic shocks are potentially reversible. In this review, we address the major etiologies that can lead to cardiogenic shock in cancer patients and discuss issues related to its management.
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Affiliation(s)
- Anais Curtiaud
- Université de Strasbourg (UNISTRA), Faculté de Médecine, Hôpitaux Universitaires de Strasbourg, Nouvel Hôpital Civil, Service de Médecine Intensive-Réanimation, Strasbourg, France
| | - Clement Delmas
- Intensive Cardiac Care Unit, Cardiology Department, University Hospital of Rangueil, Toulouse, France
| | - Justine Gantzer
- Department of Medical Oncology, Strasbourg-Europe Cancer Institute (ICANS), Strasbourg, France
| | - Lara Zafrani
- Medical Intensive Care Unit, Saint-Louis Hospital, Assistance Publique des Hôpitaux de Paris, University of Paris, Paris, France
| | - Martin Siegemund
- Intensive Care Unit, Department of Acute Medicine, University Hospital, Basel, Switzerland
- Department of Clinical Research, University of Basel, Basel, Switzerland
| | - Ferhat Meziani
- Université de Strasbourg (UNISTRA), Faculté de Médecine, Hôpitaux Universitaires de Strasbourg, Nouvel Hôpital Civil, Service de Médecine Intensive-Réanimation, Strasbourg, France
- INSERM (French National Institute of Health and Medical Research), UMR 1260, Regenerative Nanomedicine (RNM), FMTS, Strasbourg, France
| | - Hamid Merdji
- Université de Strasbourg (UNISTRA), Faculté de Médecine, Hôpitaux Universitaires de Strasbourg, Nouvel Hôpital Civil, Service de Médecine Intensive-Réanimation, Strasbourg, France
- INSERM (French National Institute of Health and Medical Research), UMR 1260, Regenerative Nanomedicine (RNM), FMTS, Strasbourg, France
- *Correspondence: Hamid Merdji
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19
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Madias JE. Inquiries Arising From a Proposed Coronary Vasospasm-Induced Pathophysiologic Mechanism of Takotsubo Syndrome. Tex Heart Inst J 2022; 49:481099. [PMID: 35511783 DOI: 10.14503/thij-22-7875] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/30/2022]
Affiliation(s)
- John E Madias
- Icahn School of Medicine at Mount Sinai, New York, New York.,Division of Cardiology, Elmhurst Hospital Center, Elmhurst, New York
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20
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Nakamura M, Imamura T, Fukui T, Oshima A, Ueno H, Kinugawa K. Successful Management of Pheochromocytoma Crisis with Cardiogenic Shock by Percutaneous Left Ventricular Assist Device. J Cardiovasc Dev Dis 2022; 9:jcdd9030071. [PMID: 35323619 PMCID: PMC8949284 DOI: 10.3390/jcdd9030071] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/12/2022] [Revised: 02/22/2022] [Accepted: 02/25/2022] [Indexed: 11/23/2022] Open
Abstract
Therapeutic strategy utilizing mechanical circulatory supports in patients with pheochromocytoma-related cardiogenic shock remains unestablished. We had a 51-year-old man with acute decompensated heart failure due to pheochromocytoma crisis. He received a percutaneous left ventricular assist device-supported alpha-blocker and intensive fluid infusion therapy, which ameliorated impaired end-organ dysfunction, maintaining hemodynamics and achieving cardiac recovery, followed by the successfully scheduled adrenalectomy. Early suspicion of pheochromocytoma and Impella-supported disease-specific medical management might be a promising bridge to surgery strategy.
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21
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Pheochromocytomas and Abdominal Paragangliomas: A Practical Guidance. Cancers (Basel) 2022; 14:cancers14040917. [PMID: 35205664 PMCID: PMC8869962 DOI: 10.3390/cancers14040917] [Citation(s) in RCA: 27] [Impact Index Per Article: 9.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/14/2021] [Revised: 02/02/2022] [Accepted: 02/08/2022] [Indexed: 02/07/2023] Open
Abstract
Simple Summary Pheochromocytomas and abdominal paragangliomas (PPGLs) are rare. They can be discovered incidentally by imaging with computed tomography or magnetic resonance imaging and during hormonal surveillance in patients with known genetic variants that are associated with PPGLs. As most PPGLs are functioning, a hormonal work-up evaluating for catecholamine excess is recommended. Classical symptoms, such as tachycardia, hypertension and headache, can be present, but when the PPGL is discovered as an incidentaloma, symptoms may be lacking or be more discrete. PPGLs carry malignant potential, and patients should undergo close surveillance, as recurrence of disease or metastasis may develop. Genetic susceptibility for multifocal disease has gained more attention, and germline variants are commonly detected, thus facilitating detection of hereditary cases and afflicted family members. Any patient with a PPGL should be managed by an expert multidisciplinary team consisting of endocrinologists, radiologists, surgeons, pathologists and clinical geneticists. Abstract Pheochromocytomas and abdominal paragangliomas (PPGLs) are rare tumors arising from the adrenal medulla or the sympathetic nervous system. This review presents a practical guidance for clinicians dealing with PPGLs. The incidence of PPGLs has risen. Most cases are detected via imaging and less present with symptoms of catecholamine excess. Most PPGLs secrete catecholamines, with diffuse symptoms. Diagnosis is made by imaging and tests of catecholamines. Localized disease can be cured by surgery. PPGLs are the most heritable of all human tumors, and germline variants are found in approximately 30–50% of cases. Such variants can give information regarding the risk of developing recurrence or metastases as well as the risk of developing other tumors and may identify relatives at risk for disease. All PPGLs harbor malignant potential, and current histological and immunohistochemical algorithms can aid in the identification of indolent vs. aggressive tumors. While most patients with metastatic PPGL have slowly progressive disease, a proportion of patients present with an aggressive course, highlighting the need for more effective therapies in these cases. We conclude that PPGLs are rare but increasing in incidence and management should be guided by a multidisciplinary team.
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22
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Wang Y, Yu X, Huang Y. Predictive Factors for Catecholamine-Induced Cardiomyopathy in Patients with Pheochromocytoma and Paraganglioma. Front Endocrinol (Lausanne) 2022; 13:853878. [PMID: 35355563 PMCID: PMC8959126 DOI: 10.3389/fendo.2022.853878] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/13/2022] [Accepted: 02/08/2022] [Indexed: 12/23/2022] Open
Abstract
OBJECTIVE To investigate possible predictive factors of catecholamine-induced cardiomyopathy in pheochromocytoma and paraganglioma (CICMPP) patients. METHODS In all, 50 CICMPP patients and 152 pheochromocytoma and paraganglioma (PPGL) patients without CICMPP who were treated in our institution between August 2012 and April 2018 were included in this retrospective study to assess predictors of CICMPP. RESULTS Patients with CICMPP reported younger onset age, more clinical symptoms and signs, more family history of hypertension, and higher maximum systolic, diastolic, and mean BP and maximum HR. Medical evaluation also showed higher level of blood hematocrit, blood glucose, 24-h urine catecholamines, larger diameter of the tumor and more comorbidities, von Hippel-Lindau syndromes, and metastatic tumors in these patients. Multivariable analysis identified maximum resting HR over 115 beats/min (OR 10.05, 95% CI 3.71-27.20), maximum resting systolic BP over 180 mmHg (OR 7.17, 95% CI 2.22-23.23), blood glucose over 8.0 mmol/L (OR 6.52, 95% CI 2.25-18.86), more than 3 symptoms and signs (OR 6.05, 95% CI 1.86-19.64), and onset age under 40 years (OR 3.74, 95% CI 1.37-10.20) as independent predictors of CICMPP. Female sex (OR 5.06, 95% CI 1.19-21.54), complaint of chest pain (OR 5.84, 95% CI 1.27-26.90), and extra-adrenal tumor (OR 8.64, 95% CI 1.82-40.94) were independent predictors of Takotsubo cardiomyopathy in CICMPP. CONCLUSION Maximum resting HR ≥115 beats/min, maximum resting systolic BP ≥180 mmHg, blood glucose ≥8.0 mmol/L, number of symptoms and signs ≥3, and onset age ≤40 years were found to be predictive factors for CICMPP.
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23
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Zhao L, Meng X, Mei Q, Fan H, Liu Y, Zhou X, Zhu H, Zhang S. Risk Factors for Cardiac Complications in Patients With Pheochromocytoma and Paraganglioma: A Retrospective Single-Center Study. Front Endocrinol (Lausanne) 2022; 13:877341. [PMID: 35721724 PMCID: PMC9199364 DOI: 10.3389/fendo.2022.877341] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/16/2022] [Accepted: 04/12/2022] [Indexed: 11/13/2022] Open
Abstract
BACKGROUND Catecholamine excess arising from pheochromocytomas and paragangliomas (PPGLs) can cause a wide spectrum of cardiac manifestations. Although there are reviews of reported cases, these reviews lack detailed data, which makes it impossible to perform an accurate analysis. In this study, we conducted a comprehensive analysis of cardiovascular complications (CCs), including PPGL-related myocardial injury, cardiogenic shock, and arrhythmias requiring antiarrhythmic therapy, in a large cohort of patients with PPGL. METHODS We retrospectively analyzed the clinical data of consecutive patients with PPGL admitted between January 2018 and June 2020. The prevalence and the characteristics of patients with CCs were investigated. Moreover, comparisons were made between patients with and without CCs. RESULTS Compared with the non-CC group, the percentage of men was significantly lower (14/41 vs.92/175, 34.1% vs. 52.6%, p = 0.034) and the proportion of patients with paroxysmal hypertension was significantly higher (13/41 vs.29/173, 31.7% vs.16.8%, p = 0.03) in the CC group. More patients showed excessive sweating (19/41 vs 64/175, 46.3% vs. 24.0%, p = 0.004) and PPGL crisis (7/41 vs. 10/175, 17.1% vs.5.7%, p=0.035) in the CC group. In terms of laboratory findings, higher white blood cell [7.36 (6.49, 20.23) vs. 5.95 (5.1, 6.97)×109/L, p<0.001] and platelet [339.28 ± 108.54 vs. 250.66 ± 70.83(×109/L), p = 0.021] counts were more common in the CC group. There was also a higher prevalence of combination-producing PPGL in the CC group (13/24 vs.20/149, 54.2% vs.13.4%, p<0.001). However, the tumor size, invasive behavior on histology, and hemorrhage or necrosis on histology did not differ between the two groups. Platelet count [odds ratio (OR): 1.009; 95% confidence interval (CI) 1.001-1.016; p=0.023] and combination-secreting PPGL (OR: 5.009; 95% CI 1.365-18.38; p=0.015) are independent risk factors for CCs in patients with PPGL. CONCLUSIONS In patients with PPGL, even in the absence of signs and symptoms of CCs, a work up of cardiology should be strongly considered. Importantly, if patients with PPGLs have higher platelet counts and the combination-secreting pattern, they are more likely to have CCs. Thus, a careful cardiac evaluation should be performed.
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Affiliation(s)
- Lin Zhao
- Department of Cardiology, Fuwai Hospital, National Center for Cardiovascular Disease, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China
| | - Xu Meng
- Department of Cardiology, Fuwai Hospital, National Center for Cardiovascular Disease, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China
| | - QiMin Mei
- Department of Emergency, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, China
| | - Hua Fan
- Department of Urology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, China
| | - YeCheng Liu
- Department of Emergency, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, China
- *Correspondence: YeCheng Liu, ; XianLiang Zhou, ; HuaDong Zhu, ; ShuYang Zhang,
| | - XianLiang Zhou
- Department of Cardiology, Fuwai Hospital, National Center for Cardiovascular Disease, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China
- *Correspondence: YeCheng Liu, ; XianLiang Zhou, ; HuaDong Zhu, ; ShuYang Zhang,
| | - HuaDong Zhu
- Department of Emergency, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, China
- *Correspondence: YeCheng Liu, ; XianLiang Zhou, ; HuaDong Zhu, ; ShuYang Zhang,
| | - ShuYang Zhang
- Department of Cardiology, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Science and Peking Union Medical College, Beijing, China
- *Correspondence: YeCheng Liu, ; XianLiang Zhou, ; HuaDong Zhu, ; ShuYang Zhang,
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Keramida K, Farmakis D, Filippatos G. Cancer and Takotsubo syndrome: from rarity to clinical practice. ESC Heart Fail 2021; 8:4365-4369. [PMID: 34873869 PMCID: PMC8712822 DOI: 10.1002/ehf2.13741] [Citation(s) in RCA: 16] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/09/2021] [Indexed: 11/12/2022] Open
Affiliation(s)
- Kalliopi Keramida
- Cardiology Department, General Anti-Cancer, Oncological Hospital, Agios Savvas, Athens, Greece
| | | | - Gerasimos Filippatos
- Department of Cardiology, National and Kapodistrian University of Athens School of Medicine, University Hospital Attikon, Athens, Greece
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25
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Madias JE. Blood norepinephrine/epinephrine/dopamine measurements in 108 patients with takotsubo syndrome from the world literature: pathophysiological implications. Acta Cardiol 2021; 76:1083-1091. [PMID: 33300464 DOI: 10.1080/00015385.2020.1826703] [Citation(s) in RCA: 24] [Impact Index Per Article: 6.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
OBJECTIVES Release of norepinephrine (NE) from neuronal cardiac nerve endings and/or blood-borne catecholamines (CATs), mainly epinephrine (EPI), may mediate TTS. The aim of this study was to document the levels of NE, EPI, and dopamine (DA) in patients with TTS. MATERIALS AND METHODS A qualitative/quantitative meta-analysis of CATs and their relationship to age, gender, and triggers, was carried out, employing the world literature on TTS, published in PubMed. RESULTS NE/EPI/DA in108 patients with TTS, 65.2 ± 16.4 years old, 89 (82.4%) women, revealed that: NE was measured more frequently than EPI, and EPI than DA; the timing of the measurements was variable; CATs were reported variably (qualitatively/quantitatively/with/without upper limits of normal); NE/EPI or NE/EPI/DA rose to the same degree; CATs were normal, or mildly/moderately elevated, with only 6 patients showing markedly elevated NE/EP/DA; NE, EPI, and DA were similar in patients with physical triggers and NE was similar in patients with physical, emotional, or no triggers (p = 0.47); EPI was higher than NE in patients with emotional triggers and EPI was higher in patients with emotional than physical triggers (p = 0.012); NE, EPI, and DA rose to the same proportion in men and women; types of TTS triggers were distributed proportionally in men and women. CONCLUSION NE, EPI, and DA rise proportionally in patients with TTS; CATs are mildly/moderately, and rarely markedly elevated; measurements of CATs should become more systematised; although CATs may not be essential for TTS diagnosis, they may contribute to prognosis and elucidation of the pathophysiology of TTS.
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Affiliation(s)
- John E. Madias
- Icahn School of Medicine at Mount Sinai, New York, NY, USA
- Division of Cardiology, Elmhurst Hospital Center, Elmhurst, NY, USA
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26
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Martínez A, Gallo-Bernal S, Acosta SC, Calixto CA, Isaza N, Isaza D, Jaimes C. Biventricular Takotsubo Cardiomyopathy as the Initial Manifestation of a Pheochromocytoma. CASE 2021; 5:363-367. [PMID: 34993364 PMCID: PMC8712975 DOI: 10.1016/j.case.2021.09.007] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Indexed: 11/25/2022]
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Y-Hassan S, Sörensson P, Ekenbäck C, Lundin M, Agewall S, Brolin EB, Caidahl K, Cederlund K, Collste O, Daniel M, Jensen J, Hofman-Bang C, Lyngå P, Maret E, Sarkar N, Spaak J, Winnberg O, Ugander M, Tornvall P, Henareh L. Plasma catecholamine levels in the acute and subacute stages of takotsubo syndrome: Results from the Stockholm myocardial infarction with normal coronaries 2 study. Clin Cardiol 2021; 44:1567-1574. [PMID: 34490898 PMCID: PMC8571561 DOI: 10.1002/clc.23723] [Citation(s) in RCA: 12] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/23/2021] [Revised: 07/13/2021] [Accepted: 08/27/2021] [Indexed: 12/30/2022] Open
Abstract
AIMS It is well-accepted that takotsubo syndrome (TS) is characterized by a massive surge of plasma catecholamines despite lack of solid evidence. The objective of this study was to examine the hypothesis of a massive catecholamine elevation in TS by studying plasma-free catecholamine metabolites in patients participating in the Stockholm myocardial infarction (MI) with normal coronaries 2 (SMINC-2) study where TS constituted more than one third of the patients. METHODS AND RESULTS The patients included in the SMINC-2 study were classified, according to cardiac magnetic resonance (CMR) imaging findings (148 patients), which was performed at a median of 3 days after hospital admission. Plasma-free catecholamine metabolites; metanephrine, normetanephrine, and methoxy-tyramine were measured on day 2-4 after admission. Catecholamine metabolite levels were available in 125 patients. One hundred and ten (88%) of the 125 patients included in SMINC-2 study, and 38 (86.4%) of the 44 patients with TS had completely normal plasma metanephrine and normetanephrine levels. All patients had normal plasma methoxy-tyramine levels. Fourteen (11.2%) of the 125 patients included in SMINC-2 study, and 5 (11.6%) of the 43 patients with TS had mild elevations (approximately 1.2 times the upper normal limits) of either plasma metanephrine or normetanephrine. One patient with pheochromocytoma-triggered TS had marked elevation of plasma metanephrine and mild elevation of plasma normetanephrine. There were no significant differences between the number or degree of catecholamine metabolite elevations between the different groups of patients with CMR imaging diagnosis included in SMINC-2 study. CONCLUSION There was no evidence of massive catecholamine elevations in the acute and subacute stages of TS apart from one patient with pheochromocytoma-induced TS. Most of the TS patients had normal catecholamine metabolites indicating that blood-borne catecholamines do not play a direct role in the pathogenesis of TS.
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Affiliation(s)
- Shams Y-Hassan
- Coronary Artery Disease Area, Heart and Vascular Theme, Karolinska Institute and Karolinska University Hospital, Stockholm, Sweden
| | - Peder Sörensson
- Department of Medicine Solna, Karolinska Institutet, and Department of Cardiology, Karolinska University Hospital, Stockholm, Sweden
| | - Christina Ekenbäck
- Division of Cardiovascular Medicine, Karolinska Institutet, Department of Clinical Sciences, Danderyd Hospital, Stockholm, Sweden
| | - Magnus Lundin
- Department of Clinical Physiology, Karolinska University Hospital, and Karolinska Institutet, Stockholm, Sweden
| | - Stefan Agewall
- Institute of Clinical Medicine, University of Oslo, Oslo, Norway
| | - Elin Bacsovics Brolin
- Department of Clinical Science, Division of Medical Imaging and Technology, Intervention and Technology at Karolinska Institutet, Stockholm, Sweden.,Department of Radiology, Capio S:t Görans Hospital, Stockholm, Sweden
| | - Kenneth Caidahl
- Department of Clinical Physiology, Karolinska University Hospital, and Karolinska Institutet, Stockholm, Sweden
| | - Kerstin Cederlund
- Department of Clinical Science, Division of Medical Imaging and Technology, Intervention and Technology at Karolinska Institutet, Stockholm, Sweden.,Department of Radiology, Södertälje Hospital, Södertälje, Sweden
| | - Olov Collste
- Department of Clinical Science and Education, Södersjukhuset, Karolinska Institutet, and Cardiology Unit, Södersjukhuset, Stockholm, Sweden
| | - Maria Daniel
- Department of Clinical Science and Education, Södersjukhuset, Karolinska Institutet, and Cardiology Unit, Södersjukhuset, Stockholm, Sweden
| | - Jens Jensen
- Department of Clinical Science and Education, Södersjukhuset, Karolinska Institutet, and Department of Cardiology, Capio St: Görans Hospital, Stockholm, Sweden
| | - Claes Hofman-Bang
- Division of Cardiovascular Medicine, Karolinska Institutet, Department of Clinical Sciences, Danderyd Hospital, Stockholm, Sweden
| | - Patrik Lyngå
- Department of Clinical Science and Education, Södersjukhuset, Karolinska Institutet, and Cardiology Unit, Södersjukhuset, Stockholm, Sweden
| | - Eva Maret
- Department of Clinical Physiology, Karolinska University Hospital, and Karolinska Institutet, Stockholm, Sweden
| | - Nondita Sarkar
- Department of Cardiology, Karolinska University Hospital, Stockholm, Sweden
| | - Jonas Spaak
- Division of Cardiovascular Medicine, Karolinska Institutet, Department of Clinical Sciences, Danderyd Hospital, Stockholm, Sweden
| | - Oscar Winnberg
- Department of Clinical Science and Education, Södersjukhuset, Karolinska Institutet, and Department of Cardiology, Capio St: Görans Hospital, Stockholm, Sweden
| | - Martin Ugander
- Department of Clinical Physiology, Karolinska University Hospital, and Karolinska Institutet, Stockholm, Sweden.,Kolling Institute, Royal North Shore Hospital, and Charles Perkins Centre, Faculty of Medicine and Health, University of Sydney, Sydney, Australia
| | - Per Tornvall
- Department of Clinical Science and Education, Södersjukhuset, Karolinska Institutet, and Cardiology Unit, Södersjukhuset, Stockholm, Sweden
| | - Loghman Henareh
- Coronary Artery Disease Area, Heart and Vascular Theme, Karolinska Institute and Karolinska University Hospital, Stockholm, Sweden
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Madias JE. Takotsubo Cardiomyopathy: Current Treatment. J Clin Med 2021; 10:3440. [PMID: 34362223 PMCID: PMC8347171 DOI: 10.3390/jcm10153440] [Citation(s) in RCA: 24] [Impact Index Per Article: 6.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/08/2021] [Revised: 07/21/2021] [Accepted: 07/26/2021] [Indexed: 02/07/2023] Open
Abstract
Management of takotsubo syndrome (TTS) is currently empirical and supportive, via extrapolation of therapeutic principles worked out for other cardiovascular pathologies. Although it has been emphasized that such non-specific therapies for TTS are consequent to its still elusive pathophysiology, one wonders whether it does not necessarily follow that the absence of knowledge of TTS' pathophysiological underpinnings should prevent us for searching, designing, or even finding, therapies efficacious for its management. Additionally, it is conceivable that therapy for TTS may be in response to pathophysiological/pathoanatomic/pathohistological consequences (e.g., "myocardial stunning/reperfusion injury"), common to both TTS and coronary artery disease, or other cardiovascular disorders). The present review outlines the whole range of management principles of TTS during its acute phase and at follow-up, including considerations pertaining to the recurrence of TTS, and commences with the idea that occasionally management of TTS should consist of mere observation along the "first do no harm" principle, while self-healing is under way. Finally, some new therapeutic hypotheses (i.e., large doses of insulin infusions in association with the employment of intravenous short- and ultrashort-acting β-blockers) are being entertained, based on previous extensive animal work and limited application in patients with neurogenic cardiomyopathy and TTS.
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Affiliation(s)
- John E. Madias
- Icahn School of Medicine at Mount Sinai, New York, NY 10029, USA; ; Tel.: +1-(718)-334-5005; Fax: +1-(718)-334-5990
- Division of Cardiology, Elmhurst Hospital Center, Elmhurst, NY 11373, USA
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Granberg D, Juhlin CC, Falhammar H. Metastatic Pheochromocytomas and Abdominal Paragangliomas. J Clin Endocrinol Metab 2021; 106:e1937-e1952. [PMID: 33462603 PMCID: PMC8063253 DOI: 10.1210/clinem/dgaa982] [Citation(s) in RCA: 54] [Impact Index Per Article: 13.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/18/2020] [Indexed: 12/20/2022]
Abstract
CONTEXT Pheochromocytomas and paragangliomas (PPGLs) are believed to harbor malignant potential; about 10% to 15% of pheochromocytomas and up to 50% of abdominal paragangliomas will exhibit metastatic behavior. EVIDENCE ACQUISITION Extensive searches in the PubMed database with various combinations of the key words pheochromocytoma, paraganglioma, metastatic, malignant, diagnosis, pathology, genetic, and treatment were the basis for the present review. DATA SYNTHESIS To pinpoint metastatic potential in PPGLs is difficult, but nevertheless crucial for the individual patient to receive tailor-made follow-up and adjuvant treatment following primary surgery. A combination of histological workup and molecular predictive markers can possibly aid the clinicians in this aspect. Most patients with PPGLs have localized disease and may be cured by surgery. Plasma metanephrines are the main biochemical tests. Genetic testing is important, both for counseling and prognostic estimation. Apart from computed tomography and magnetic resonance imaging, molecular imaging using 68Ga-DOTATOC/DOTATATE should be performed. 123I-MIBG scintigraphy may be performed to determine whether 131I-MIBG therapy is a possible option. As first-line treatment in patients with metastatic disease, 177Lu-DOTATATE or 131I-MIBG is recommended, depending on which shows best expression. In patients with very low proliferative activity, watch-and-wait or primary treatment with long-acting somatostatin analogues may be considered. As second-line treatment, or first-line in patients with high proliferative rate, chemotherapy with temozolomide or cyclophosphamide + vincristine + dacarbazine is the therapy of choice. Other therapies, including sunitinib, cabozantinib, everolimus, and PD-1/PDL-1 inhibitors, have shown modest effect. CONCLUSIONS Metastatic PPGLs need individualized management and should always be discussed in specialized and interdisciplinary tumor boards. Further studies and newer treatment modalities are urgently needed.
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Affiliation(s)
- Dan Granberg
- Department of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden
- Department of Breast, Endocrine Tumors and Sarcoma, Karolinska University Hospital, Stockholm, Sweden
| | - Carl Christofer Juhlin
- Department of Oncology-Pathology, Karolinska Institutet, Karolinska University Hospital, Stockholm, Sweden
- Department of Pathology and Cytology, Karolinska University Hospital, Stockholm, Sweden
| | - Henrik Falhammar
- Department of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden
- Department of Breast, Endocrine Tumors and Sarcoma, Karolinska University Hospital, Stockholm, Sweden
- Department of Endocrinology, Metabolism and Diabetes, Karolinska University Hospital, Stockholm, Sweden
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30
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Maffè S, Dellavesa P, Paffoni P, Bergamasco L, Arrondini M, Valentini S, Facchini E, Prenna E, Baduena L, Careri G, Franchetti Pardo N, Parravicini U. Takotsubo syndrome and pheochromocytoma: an insidious combination. Monaldi Arch Chest Dis 2021; 91. [PMID: 33691391 DOI: 10.4081/monaldi.2021.1711] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/30/2020] [Accepted: 03/02/2021] [Indexed: 11/23/2022] Open
Abstract
Pheochromocytoma is a rare adrenal tumor characterized by the secretion of catecholamines and vasoactive peptides. It can cause a catecholaminergic storm and lead to acute coronary syndromes. We present the case of a 53-year-old man, without any medical history, who arrived to the hospital following a spinal trauma due a fall. He presents back and retrosternal pain, with a clinical status of acute pulmonary edema, sinus tachycardia with left bundle branch block, left ventricular apical ballooning with depressed systolic function. Blood tests show a very important increase of Troponin and transaminases. A contrast chest-abdomen CT highlighted a right adrenal solid mass, with a diameter of 78mm, partial capsular laceration, compression of the inferior vena cava and the hepatic parenchyma. The clinical condition of the patient rapidly worsens from a respiratory and hemodynamic point of view, with cardiogenic shock, anuria and sepsis, refractory to all the medical treatments, until the patient died. The autopsy confirmed that the abdominal mass was a pheochromocytoma, broken after the trauma suffered. The resulting catecholaminergic storm caused a myocardial ischemia with Takotsubo syndrome, with cardiogenic shock. This unfortunate case confirms the pheochromocytoma as important risk factor for the onset of Takotsubo syndrome, and the how dramatic and severe a catecholaminergic storm can be.
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Affiliation(s)
- Stefano Maffè
- Division of Cardiology, SS Trinità Borgomanero Hospital, ASL NO, Novara.
| | | | - Paola Paffoni
- Division of Cardiology, SS Trinità Borgomanero Hospital, ASL NO, Novara.
| | - Luca Bergamasco
- Division of Cardiology, SS Trinità Borgomanero Hospital, ASL NO, Novara.
| | - Marisa Arrondini
- Division of Surgical Pathology, SS Trinità Borgomanero Hospital, ASL NO, Novara.
| | - Stefano Valentini
- Division of Surgical Pathology, SS Trinità Borgomanero Hospital, ASL NO, Novara.
| | - Emanuela Facchini
- Division of Cardiology, SS Trinità Borgomanero Hospital, ASL NO, Novara.
| | - Eleonora Prenna
- Division of Cardiology, SS Trinità Borgomanero Hospital, ASL NO, Novara.
| | - Lara Baduena
- Division of Cardiology, SS Trinità Borgomanero Hospital, ASL NO, Novara.
| | - Giulia Careri
- Division of Cardiology, SS Trinità Borgomanero Hospital, ASL NO, Novara.
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31
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Shi F, Sun LX, Long S, Zhang Y. Pheochromocytoma as a cause of repeated acute myocardial infarctions, heart failure, and transient erythrocytosis: A case report and review of the literature. World J Clin Cases 2021; 9:951-959. [PMID: 33585644 PMCID: PMC7852630 DOI: 10.12998/wjcc.v9.i4.951] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/22/2020] [Revised: 11/25/2020] [Accepted: 12/10/2020] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Pheochromocytoma is a rare catecholamines-secreting tumor arising from chromaffin cells in the adrenal medulla. It classically presents with paroxysmal hypertension, headaches, palpitations, sweating, and metabolic disorders. Atypical presentations such as acute myocardial infarction, heart failure, cardiomyopathy, stroke, and transient erythrocytosis have been infrequently documented.
CASE SUMMARY We describe the case of a 72-year-old man diagnosed with pheochromocytoma presenting with non-ST segment elevation myocardial infarction, heart failure, and transient erythrocytosis with nonobstructed coronary arteries. This was his second heart attack. The patient was previously diagnosed with myocardial infarction, and an immense mass was found on the left adrenal gland 3 years prior. Based on clinical and laboratory findings, a diagnosis of pheochromocytoma was confirmed. His coronary angiogram showed nonobstructed coronary arteries except for a myocardial bridge in the left anterior descending branch. This was a form of type-2 myocardial infarction. The myocardial cell lesions were caused by sudden secretion of catecholamines by the pheochromocytoma. Even more atypically, his hemoglobin level was obviously elevated at admission, but after a few days of treatment with an alpha-adrenergic receptor blocker, it dropped to normal levels without additional treatment.
CONCLUSION Pheochromocytoma may be a cause of acute myocardial infarction, heart failure, and transient erythrocytosis.
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Affiliation(s)
- Fei Shi
- Department of Cardiology, Affiliated Hospital of Chengde Medical University, Chengde 067000, Hebei Province, China
| | - Li-Xian Sun
- Department of Cardiology, Affiliated Hospital of Chengde Medical University, Chengde 067000, Hebei Province, China
| | - Sen Long
- Department of Traditional Chinese Medicine, Affiliated Hospital of Chengde Medical University, Chengde 067000, Hebei Province, China
| | - Ying Zhang
- Department of Cardiology, Affiliated Hospital of Chengde Medical University, Chengde 067000, Hebei Province, China
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32
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Elliott PF, Berhane T, Ragnarsson O, Falhammar H. Ectopic ACTH- and/or CRH-Producing Pheochromocytomas. J Clin Endocrinol Metab 2021; 106:598-608. [PMID: 32710791 DOI: 10.1210/clinem/dgaa488] [Citation(s) in RCA: 24] [Impact Index Per Article: 6.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/27/2020] [Indexed: 12/14/2022]
Abstract
CONTEXT The characteristics of catecholamine-secreting pheochromocytomas have been well studied. However, less is known about the characteristics, management and outcome in patients with ectopic adrenocorticotropic hormone (ACTH) and/or corticotrophin-releasing hormone (CRH)-secreting pheochromocytomas. OBJECTIVE To review the characteristics and outcomes of ACTH- and/or CRH-secreting pheochromocytomas. DATA SOURCE A systematic search of PubMed/MEDLINE and Web of Science, identifying relevant reports published up to 10 February 2020. STUDY SELECTION Original articles, including case reports and case series, reporting individual patient data from patients with ACTH- and/or CRH-secreting pheochromocytomas. DATA EXTRACTION Information on sex, age, symptoms at presentation, comorbidities, biochemistry, imaging, histopathology, and outcomes was extracted. DATA SYNTHESIS We identified 91 articles reporting on 99 cases of ACTH- and/or CRH-secreting pheochromocytomas (CRH-secreting n = 4). Median age at diagnosis was 49 years (interquartile range 38-59.5) with a 2:1 female to male ratio. Most patients presented with clinical Cushing syndrome (n = 79; 81%), hypertension (n = 87; 93%), and/or diabetes (n = 50; 54%). Blood pressure, glucose control, and biochemical parameters improved in the vast majority of patients postoperatively. Infections were the most common complication. Most cases (n = 70, 88%) with reported long-term outcome survived to publication (median follow-up 6 months). CONCLUSION Ectopic ACTH- and/or CRH-secreting pheochromocytoma should be considered in patients presenting with ACTH-dependent Cushing syndrome and adrenal mass. Despite the challenge in diagnosis, patient outcomes appear favorable.
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Affiliation(s)
| | - Thomas Berhane
- Department of Medicine, Royal Darwin Hospital, Darwin, Australia
| | - Oskar Ragnarsson
- Department of Internal Medicine and Clinical Nutrition, Institute of Medicine at Sahlgrenska Academy, University of Gothenburg, Göteborg, Sweden
- Department of Endocrinology, Sahlgrenska University Hospital, Göteborg, Sweden
| | - Henrik Falhammar
- Department of Endocrinology, Royal Darwin Hospital, Darwin, Australia
- Department of Endocrinology, Metabolism and Diabetes, Karolinska University Hospital, Stockholm, Sweden
- Department of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden
- Menzies School of Health Research, Darwin, Australia
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33
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Berends AMA, Kerstens MN, Lenders JWM, Timmers HJLM. Response to Letter to the Editor: "Approach to the Patient: Perioperative Management of the Patient With Pheochromocytoma or Sympathetic Paraganglioma". J Clin Endocrinol Metab 2020; 105:5902590. [PMID: 32895722 PMCID: PMC7553242 DOI: 10.1210/clinem/dgaa602] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/19/2020] [Accepted: 09/06/2020] [Indexed: 11/19/2022]
Affiliation(s)
- Annika M A Berends
- Department of Endocrinology, University of Groningen, University Medical Center Groningen, Groningen, the Netherlands
| | - Michiel N Kerstens
- Department of Endocrinology, University of Groningen, University Medical Center Groningen, Groningen, the Netherlands
| | - Jacques W M Lenders
- Department of Internal Medicine, Radboud University Medical Center, Nijmegen, the Netherlands
- Department of Medicine III, University Hospital Carl Gustav Carus, Technical University Dresden, Dresden, Germany
| | - Henri J L M Timmers
- Department of Internal Medicine, Radboud University Medical Center, Nijmegen, the Netherlands
- Correspondence and Reprint Requests: Henri J.L.M. Timmers, MD, PhD, Radboud University Medical Center Nijmegen, Department of Internal Medicine, Section of Endocrinology, Geert Grooteplein Zuid 10, 6525 GA Nijmegen, P.O. Box 9101, 6500 HB, the Netherlands. E-mail:
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Y-Hassan S, Falhammar H. Cardiovascular Manifestations and Complications of Pheochromocytomas and Paragangliomas. J Clin Med 2020; 9:jcm9082435. [PMID: 32751501 PMCID: PMC7465968 DOI: 10.3390/jcm9082435] [Citation(s) in RCA: 69] [Impact Index Per Article: 13.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/20/2020] [Revised: 07/14/2020] [Accepted: 07/28/2020] [Indexed: 12/13/2022] Open
Abstract
Pheochromocytomas and paragangliomas (PPGLs) are rare neuro-endocrine tumors. The catecholamine surge causes paroxysmal or chronic secondary hypertension. PPGLs may present as hypertensive- or PPGL-crisis with severe life-threatening cardiac and cerebrovascular complications. PPGLs-induced cardiac manifestations have been reported with diagnoses as PPGLs-induced electrocardiogram (ECG) changes “mimicking acute myocardial infarction”, arrhythmias, myocarditis, acute coronary syndrome, dilated cardiomyopathy, and lately as takotsubo syndrome. Critical analysis of these reports reveals that most of these cardiac manifestations have certain features in common. They have a dramatic clinical presentation and are reversible if the disease is treated with appropriate medical therapy and surgical resection of the PPGL tumor. They may have the same repolarization ECG changes irrespective of the clinical cardiac diagnosis, usually associated with mild to moderate elevations of myocardial biomarkers as troponins and normal coronary arteries. The histopathological findings are usually focal or multifocal in the form hypercontracted sarcomeres and contraction band necrosis (myofibrillar degeneration) with subsequent secondary mononuclear cell infiltration. Evidences argue the PPGL caused surge of catecholamines triggers hyperactivation of the sympathetic nervous system with cardiac sympathetic nerve terminal disruption with norepinephrine spillover causing the cardiac complications. A comprehensive review of various reported cardiovascular manifestations and complications of PPGLs are presented.
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Affiliation(s)
- Shams Y-Hassan
- Coronary Artery Disease Area, Heart and Vascular Theme, Karolinska Institutet and Karolinska University Hospital, 141 86 Stockholm, Sweden;
| | - Henrik Falhammar
- Department of Endocrinology, Metabolism and Diabetes, Karolinska University Hospital, 141 86 Stockholm, Sweden
- Department of Molecular Medicine and Surgery, Karolinska Institutet, 171 76 Stockholm, Sweden
- Correspondence:
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35
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Y-Hassan S. Autonomic neurocardiogenic syndrome is stonewalled by the universal definition of myocardial infarction. World J Cardiol 2020; 12:231-247. [PMID: 32774776 PMCID: PMC7383352 DOI: 10.4330/wjc.v12.i6.231] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/30/2019] [Revised: 04/27/2020] [Accepted: 05/14/2020] [Indexed: 02/06/2023] Open
Abstract
Myocardial infarction (MI) is defined as myocardial cell death due to prolonged myocardial ischemia. Clinically, troponin rise and/or fall have become the “defining feature of MI” according to the universal definition of MI (UD-MI). Takotsubo syndrome (TS) and TS-related disease conditions also cause troponin elevation with typical rise and/or fall pattern but through a mechanism other than coronary ischemia. By strict application of the clinical diagnostic criteria for type-1 MI, type-2 MI, type-3 MI, and MI with non-obstructive coronary arteries according to the UD-MI including the fourth one published recently, TS and most of the 26 other causes of troponin elevation mentioned in the fourth UD-MI may erroneously be classified as MI. The existing evidence argues for the case that TS by itself is not a MI. Hyper-activation of the autonomic-sympathetic nervous system including local cardiac sympathetic hyper-activation and disruption with nor-epinephrine churn and spillover is the most probable cause of TS. This autonomic neuro-cardiogenic (ANCA) mechanism results in myocardial “cramp” (stunning), the severity and duration of which depend on the degree of the sympathetic-hyperactivation and nor-epinephrine spillover. The myocardial cramp may squeeze the cytosolic free troponin pools causing mild to moderate troponin elevation in TS and TS-related disease conditions. This ANCA syndrome, which has hitherto been enveloped by the UD-MI over more than one decade, may occur in acute, recurrent, and chronic forms. In this critical review, the controversies of UD-MI, evidence for ANCA syndrome, and a hypothetical mechanism for the troponin elevation in ANCA syndrome are provided.
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Affiliation(s)
- Shams Y-Hassan
- Coronary Artery Disease Area, Heart and Vascular Theme, Karolinska Institutet and Karolinska University Hospital, Stockholm S-141 86, Sweden
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36
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Chen M, Zhao T, Chen G, Hu S. A rare long-term undetected pheochromocytoma leading to Takotsubo syndrome in an older male patient: a case report. BMC Endocr Disord 2020; 20:93. [PMID: 32576166 PMCID: PMC7313138 DOI: 10.1186/s12902-020-00578-5] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/21/2019] [Accepted: 06/16/2020] [Indexed: 04/20/2023] Open
Abstract
BACKGROUND Takotsubo syndrome is an uncommon, acute, and reversible cardiomyopathy that occurs primarily in postmenopausal females. The clinical presentation of the syndrome resembles acute coronary syndrome, but coronary angiography reveals no obstructive coronary artery disease. Rarely, a catecholamine surge due to pheochromocytoma may induce Takotsubo syndrome. The clinical features of pheochromocytoma include paroxysmal hypertension, headache, palpitations, and profuse sweating. However, owing to the episodic, rather than continued, symptoms and signs of pheochromocytoma, its timely diagnosis poses a challenge for clinicians. Here, we report a rare case of long-term undetected pheochromocytoma leading to Takotsubo syndrome in an older male patient. CASE PRESENTATION A 70-year-old man presented with paroxysmal chest distress and chest pain. Examinations revealed acute coronary syndrome with normal coronary arteries, heart failure, reversible left ventricular regional wall motion abnormalities, labile blood pressure, a giant left adrenal mass, and extremely high levels of metanephrine and normetanephrine. Clinical manifestations, laboratory reports, and imaging findings suggested a diagnosis of Takotsubo syndrome caused by pheochromocytoma. Supportive therapy, administration of alpha- adrenergic receptor blockers, and left adrenal mass resection resolved the patient's symptoms. A histological examination confirmed the presence of pheochromocytoma. We reviewed his history of midbrain hemorrhage 6 years prior and found a mass in the left adrenal region by reviewing the computed tomography images of the lung that were also taken 6 years prior, on which the pheochromocytoma was evident. CONCLUSIONS Our case illustrates the importance of understanding the link between pheochromocytoma and Takotsubo syndrome. A diagnosis of pheochromocytoma-induced Takotsubo syndrome should be considered during the differential diagnosis of acute coronary syndrome, especially in patients with labile blood pressure and normal coronary angiography findings; meanwhile, assessments of catecholamines and its metabolites and abdominal computed tomography scan should be performed at the right time. Clinicians should also be alert to potential pheochromocytoma in patients with unexplained cerebral hemorrhage, even in the absence of symptoms of catecholamine excess.
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Affiliation(s)
- Min Chen
- Department of Endocrinology, the First Affiliated Hospital, College of Medicine, Zhejiang University, #79, Qingchun Road, Zhejiang, 310003, Hangzhou, China.
| | - Tong Zhao
- Department of Endocrinology, the First Affiliated Hospital, College of Medicine, Zhejiang University, #79, Qingchun Road, Zhejiang, 310003, Hangzhou, China
| | - Guoping Chen
- Department of Endocrinology, the First Affiliated Hospital, College of Medicine, Zhejiang University, #79, Qingchun Road, Zhejiang, 310003, Hangzhou, China
| | - Shenjiang Hu
- Department of Cardiology, the First Affiliated Hospital, College of Medicine, Zhejiang University, #79, Qingchun Road, Zhejiang, 310003, Hangzhou, China
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Cornu E, Motiejunaite J, Belmihoub I, Vidal-Petiot E, Mirabel M, Amar L. Acute Stress Cardiomyopathy: Heart of pheochromocytoma. ANNALES D'ENDOCRINOLOGIE 2020; 82:201-205. [PMID: 32988608 DOI: 10.1016/j.ando.2020.03.011] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/29/2023]
Abstract
Stress cardiomyopathy (SCM) is a syndrome characterized by transient regional systolic dysfunction of the left ventricle in the absence of angiographic evidence of coronaropathy. This abnormality is associated with high levels of catecholamines. Stress cardiomyopathy is also called Takotsubo (TS) cardiomyopathy. Pheochromocytoma crisis can occur spontaneously or can be precipitated by manipulation of the tumor, trauma, certain medications or stress for example during non-adrenal surgery. The main drugs leading to pheochromocytoma crisis include D2 dopamine receptor antagonists, noncardioselective β-adrenergic receptor blockers, tricyclic antidepressants and related neurotransmitter uptake blockers, sympathomimetics, certain peptide and steroid hormones and several agents used during induction of anesthesia. Patients can develop symptoms of heart failure associated with tachyarrhythmia, cardiogenic shock with hypotension and collapse, or apparent acute coronary syndromes. This review describes pathophysiology, epidemiology, diagnosis criteria and management of SCM.
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Affiliation(s)
- Erika Cornu
- Hypertension Unit, Hôpital Européen Georges Pompidou, Assistance Publique - Hôpitaux de Paris, 75015 Paris, France; Université de Paris, Paris, France
| | - Justina Motiejunaite
- Université de Paris, Paris, France; Department of Physiology, Hôpital Bichat, Assistance Publique - Hôpitaux de Paris, 75018 Paris, France
| | - Ines Belmihoub
- Hypertension Unit, Hôpital Européen Georges Pompidou, Assistance Publique - Hôpitaux de Paris, 75015 Paris, France; Université de Paris, Paris, France
| | - Emmanuelle Vidal-Petiot
- Université de Paris, Paris, France; Department of Physiology, Hôpital Bichat, Assistance Publique - Hôpitaux de Paris, 75018 Paris, France
| | - Mariana Mirabel
- Université de Paris, Paris, France; Cardio-oncology unit, Hôpital Européen Georges Pompidou, Assistance Publique - Hôpitaux de Paris, 75015 Paris, France
| | - Laurence Amar
- Hypertension Unit, Hôpital Européen Georges Pompidou, Assistance Publique - Hôpitaux de Paris, 75015 Paris, France; Université de Paris, Paris, France; Université de Paris, PARCC, INSERM, Equipe Labellisée par la Ligue contre le Cancer, 75015 Paris, France.
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38
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Acute Heart Failure as a First Presentation of Pheochromocytoma Complicated with "Inverted" Takotsubo Syndrome. Case Rep Endocrinol 2020; 2020:2521046. [PMID: 32257461 PMCID: PMC7103034 DOI: 10.1155/2020/2521046] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/05/2020] [Accepted: 02/27/2020] [Indexed: 12/23/2022] Open
Abstract
Takotsubo syndrome is a rare but emerging form of acute reversible myocardial injury characterized by transient systolic LV dysfunction, often related to emotional or physical stress. Pheochromocytoma is increasingly recognised as another possible trigger. Pheochromocytoma is a rare catecholamine-secreting tumour arising from chromaffin cells within the adrenal medulla or extra-adrenal paraganglia. The pathognomonic quartet of paroxysmal hypertension, palpitations, headache, and diaphoresis is rarely present, and diagnosis is often delayed. We describe a 43-year-old formerly healthy patient with an adrenal pheochromocytoma, presenting as an "inverted" takotsubo syndrome complicated with acute heart failure and pulmonary oedema.
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Y-Hassan S, Falhammar H. Clinical features, complications, and outcomes of exogenous and endogenous catecholamine-triggered Takotsubo syndrome: A systematic review and meta-analysis of 156 published cases. Clin Cardiol 2020; 43:459-467. [PMID: 32125009 PMCID: PMC7244299 DOI: 10.1002/clc.23352] [Citation(s) in RCA: 28] [Impact Index Per Article: 5.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/21/2019] [Accepted: 02/19/2020] [Indexed: 12/25/2022] Open
Abstract
Innumerable physical stress factors including externally administered catecholamines, and pheochromocytomas and paragangliomas (PPGLs) have been reported to trigger Takotsubo syndrome (TS). A systematic search of PubMed/MEDLINE identified 156 patients with catecholamine‐induced TS up to December 2017. Data were compared within the catecholamine‐induced TS cohort, but some comparisons were also done to a previously published large all‐TS cohort (n = 1750). The mean age was 46.4 ± 16.4 years (72.3% women). The clinical presentation was dramatic with high complication rates in (68.2%, n = 103; multiple complications 34.6%, n = 54). The most common TS ballooning pattern was apical or mid‐apical (45.2%, n = 69), followed by basal pattern (28.8%, n = 45), global pattern (16.0%, n = 25), mid‐ventricular (8.3%, n = 13), focal (0.6%, n = 1), and unidentified pattern (1.9%, n = 3). There was an increase in the prevalence of apical sparing ballooning pattern compared to all‐TS population (37.7% vs 18.3%, P < .00001). Higher complication rates were observed in TS with global ballooning pattern compared to apical ballooning pattern (23/25, 92% vs 38/65, 58.5%; P = .0022). Higher complication rates were observed in patients with age < 50 years than patients >50 years (73/92, 79.3% vs 29/56, 51.8%, P = 0.0009). Recurrence occurred exclusively in patients with PPGL‐induced TS (18/107 patients, 16.8%). PPGL‐induced TS was characterized by more global ballooning's pattern (22/104, 21.2% vs 3/49, 6.1%, P = 0.02), and lower left ventricular ejection fraction (25.54 ± 11.3 vs 31.82 ± 9.93, P = 0.0072) compared to exogenous catecholamine‐induced TS. In conclusion, catecholamine‐induced TS was characterized by a dramatic clinical presentation with extensive left ventricular dysfunction, and high complication rate.
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Affiliation(s)
- Shams Y-Hassan
- Coronary Artery Disease Area, Heart and Vascular Theme, Karolinska Institutet and Karolinska University Hospital, Stockholm, Sweden
| | - Henrik Falhammar
- Department of Endocrinology, Metabolism and Diabetes, Karolinska University Hospital, Stockholm, Sweden.,Departement of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden
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Y-Hassan S, Falhammar H. Stumbling broke the spleen and unveiled pheochromocytoma, which in turn broke the heart. Endocrine 2020; 67:727-728. [PMID: 31883060 PMCID: PMC7054229 DOI: 10.1007/s12020-019-02169-4] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/30/2019] [Accepted: 12/18/2019] [Indexed: 01/29/2023]
Affiliation(s)
- Shams Y-Hassan
- Coronary Artery Disease Area, Heart and Vascular Theme, Karolinska Institutet and Karolinska University Hospital, Stockholm, Sweden
| | - Henrik Falhammar
- Department of Endocrinology, Metabolism and Diabetes, Karolinska University Hospital, Stockholm, Sweden.
- Departement of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden.
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Falcetta P, Orsolini F, Molinaro E, Vitti P, Tonacchera M. Tako-tsubo Syndrome as First Manifestation in a Case of Pheochromocytoma Developed From a Non-functional Adrenal Incidentaloma. Front Endocrinol (Lausanne) 2020; 11:51. [PMID: 32117073 PMCID: PMC7033429 DOI: 10.3389/fendo.2020.00051] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/30/2019] [Accepted: 01/28/2020] [Indexed: 12/16/2022] Open
Abstract
Background: Pheochromocytoma is a catecholamine secreting tumor that, in extremely rare cases, may develop over time from a non-functional adrenal adenoma. Catecholamine excess can lead to a kind of cardiomyopathy similar to that seen in tako-tsubo syndrome (TTS). Case report: A 69 years old female with a history of type 2 diabetes, hypertension, and a non-functional right adrenal adenoma diagnosed 3 years earlier was referred to our center for further investigations. During the evaluation, she had a hypertensive crisis with chest pain, tachycardia, and diaphoresis. Suspecting an acute coronary syndrome, she underwent coronary angiography, which showed the typical features of TTS. The high 24 h-urinary metanephrines excretion and abdominal MRI findings were suggestive of pheochromocytoma. Right laparoscopic adrenalectomy was performed, with the resolution of all symptoms. Pathology findings confirmed the diagnosis of pheochromocytoma. After 12 months, the patient was still asymptomatic, with the echocardiography displaying a complete recovery of the left-ventricular function. Conclusions: The development of a pheochromocytoma from an adrenal non functional adenoma is an extremely rare event, but potentially life-threating because of the catecholamine-associated cardiovascular toxicity. In particular, TTS is a form of cardiomyopathy that has been increasingly described as associated with catecholamine-secreting tumors. The exclusion of pheochromocytoma in a patient with TTS has important therapeutic implications, since the administration of β-blockers may be extremely harmful in patients with catecholamine surge in the absence of adequate α-blockage.
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