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1.
J Pak Med Assoc ; 74(5): 998-999, 2024 May.
Article in English | MEDLINE | ID: mdl-38783456

ABSTRACT

Fever is usually thought to be of an infectious or inflammatory etiology. In this brief communication, we explore the multifaceted connections between fever and endocrine dysfunction. Impaired resistance to infection often leads to fever in conditions like diabetes and Cushing's syndrome. Additionally, several endocrine disorders, including hyperthyroidism, subacute thyroiditis, carcinoid syndrome, and pheochromocytoma, can manifest as fever. Furthermore, fever can be an adverse effect of various endocrine treatments, such as bisphosphonates and antithyroid drugs. We refer to these scenarios as 'endocrine fever.' Increased awareness of these clinical associations can aid in prompt diagnosis and management of these conditions.


Subject(s)
Endocrine System Diseases , Fever , Humans , Fever/etiology , Endocrine System Diseases/therapy , Endocrine System Diseases/diagnosis , Hyperthyroidism/therapy , Hyperthyroidism/diagnosis , Cushing Syndrome/diagnosis , Cushing Syndrome/therapy , Pheochromocytoma/therapy , Pheochromocytoma/diagnosis , Pheochromocytoma/complications , Adrenal Gland Neoplasms/therapy , Adrenal Gland Neoplasms/diagnosis , Adrenal Gland Neoplasms/complications , Antithyroid Agents/therapeutic use , Antithyroid Agents/adverse effects , Diphosphonates/therapeutic use , Diphosphonates/adverse effects
2.
Pan Afr Med J ; 47: 88, 2024.
Article in French | MEDLINE | ID: mdl-38737225

ABSTRACT

Ectopic ACTH-secreting pheochromocytoma is a very rare cause of Cushing´s syndrome, posing diagnostic and therapeutic challenges. We here report the case of a female patient with suspected severe Cushing´s syndrome associated with melanoderma, arterial hypertension resistant to triple therapy and unbalanced diabetes treated with insulin therapy. Biologically, urinary ethoxylated, 24-hour urinary free cortisol and ACTH were very high. Imaging showed a 3.5 cm left adrenal mass. The patient underwent left adrenalectomy after medical preparation, with good clinico-biological outcome. Anatomopathological examination confirmed the diagnosis of pheochromocytoma. This case study highlights the importance of measuring methoxylated derivatives in any patient with ACTH-dependent Cushing´s syndrome associated with an adrenal mass. The aim is to ensure early treatment and avoid life-threatening complications.


Subject(s)
Adrenal Gland Neoplasms , Adrenalectomy , Adrenocorticotropic Hormone , Cushing Syndrome , Pheochromocytoma , Humans , Pheochromocytoma/diagnosis , Pheochromocytoma/metabolism , Pheochromocytoma/complications , Female , Adrenal Gland Neoplasms/diagnosis , Adrenal Gland Neoplasms/metabolism , Adrenal Gland Neoplasms/complications , Cushing Syndrome/etiology , Cushing Syndrome/diagnosis , Adrenalectomy/methods , Adrenocorticotropic Hormone/metabolism , Hydrocortisone/metabolism , Hypertension/etiology , Middle Aged , Adult
3.
Front Endocrinol (Lausanne) ; 15: 1385808, 2024.
Article in English | MEDLINE | ID: mdl-38808113

ABSTRACT

Background: A recent cross-sectional study showed that both comorbidities and mortality in patients with adrenal incidentaloma (AI) are tied to sex. However, few longitudinal studies evaluated the development of arterial hypertension, hyperglycemia, dyslipidemia and bone impairment in patients with AI. The aim of this study is to analyze the impact of sex in the development of these comorbidities during long-term follow-up. Methods: We retrospectively evaluated 189 patients (120 females, 69 males) with AI, from four referral centers in Italy and Croatia. Clinical characteristics, comorbidities and cortisol after 1-mg dexamethasone suppression test (1-mg DST) were assessed at baseline and at last follow-up visit (LFUV). Median follow-up was 52 (Interquartile Range 25-86) months. Results: The rates of arterial hypertension and hyperglycemia increased over time both in females (65.8% at baseline versus 77.8% at LFUV, p=0.002; 23.7% at baseline versus 39.6% at LFUV, p<0.001; respectively) and males (58.0% at baseline versus 69.1% at LFUV, p=0.035; 33.8% at baseline versus 54.0% at LFUV, p<0.001; respectively). Patients were stratified in two groups using 1.8 µg/dl as cut-off of cortisol following 1-mg DST: non-functional adrenal tumors (NFAT) and tumors with mild autonomous cortisol secretion (MACS). In the NFAT group (99 patients, females 62.6%), at baseline, we did not observe any difference in clinical characteristics and comorbidities between males and females. At LFUV, males showed a higher frequency of hyperglycemia than females (57.6% versus 33.9%, p=0.03). In the MACS group (89 patients, females 64.0%), at baseline, the prevalence of hypertension, hyperglycemia and dyslipidemia was similar between sexes, despite females were younger (60, IQR 55-69 versus 67.5, IQR 61-73, years; p=0.01). Moreover, females presented higher rates of bone impairment (89.3% versus 54.5%, p=0.02) than males. At LFUV, a similar sex-related pattern was observed. Conclusion: Patients with AI frequently develop arterial hypertension and hyperglycemia and should be periodically checked for these comorbidities, regardless of sex. In patients with MACS, the lack of difference between sexes in the frequency of cardiometabolic comorbidities despite that females are younger, and the higher frequency of bone impairment in females, suggest a sex-specific effect of cortisol.


Subject(s)
Adrenal Gland Neoplasms , Comorbidity , Hypertension , Humans , Female , Male , Adrenal Gland Neoplasms/epidemiology , Adrenal Gland Neoplasms/blood , Adrenal Gland Neoplasms/complications , Middle Aged , Retrospective Studies , Aged , Hypertension/epidemiology , Sex Factors , Hyperglycemia/epidemiology , Hyperglycemia/blood , Dyslipidemias/epidemiology , Follow-Up Studies , Italy/epidemiology , Cross-Sectional Studies
4.
BMC Cardiovasc Disord ; 24(1): 261, 2024 May 20.
Article in English | MEDLINE | ID: mdl-38769478

ABSTRACT

BACKGROUND: Pheochromocytoma is rare in pregnant women. It presents as diverse symptoms, including hypertension and sweating. The symptoms of pregnant women with pheochromocytoma and comorbid hypertension often mimic the clinical manifestations of preeclampsia, and these women are often misdiagnosed with preeclampsia. CASE PRESENTATION: In this case, a pregnant woman presented with chest pain as the primary symptom, and a diagnosis of pheochromocytoma was considered after ruling out myocardial ischemia and aortic dissection with the relevant diagnostic tools. This patient then underwent successful surgical resection using a nontraditional management approach, which resulted in a positive clinical outcome. CONCLUSIONS: It is essential to consider pheochromocytoma as a potential cause of chest pain and myocardial infarction-like electrocardiographic changes in pregnant women, even if they do not have a history of hypertension.


Subject(s)
Adrenal Gland Neoplasms , Pheochromocytoma , Pregnancy Complications, Neoplastic , Humans , Pheochromocytoma/complications , Pheochromocytoma/diagnosis , Pheochromocytoma/surgery , Female , Pregnancy , Adrenal Gland Neoplasms/complications , Adrenal Gland Neoplasms/surgery , Adrenal Gland Neoplasms/diagnosis , Pregnancy Complications, Neoplastic/diagnosis , Pregnancy Complications, Neoplastic/surgery , Adult , Treatment Outcome , Chest Pain/etiology , Chest Pain/diagnosis , Predictive Value of Tests , Adrenalectomy , Electrocardiography
5.
Ann Cardiol Angeiol (Paris) ; 73(3): 101768, 2024 Jun.
Article in French | MEDLINE | ID: mdl-38788259

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.


Subject(s)
Adrenal Gland Neoplasms , Myocarditis , Pheochromocytoma , Recurrence , Humans , Pheochromocytoma/complications , Pheochromocytoma/diagnosis , Myocarditis/etiology , Myocarditis/diagnosis , Adrenal Gland Neoplasms/complications , Adrenal Gland Neoplasms/diagnosis , Adult , Male , Female
6.
Zhonghua Nei Ke Za Zhi ; 63(4): 378-385, 2024 Apr 01.
Article in Chinese | MEDLINE | ID: mdl-38561283

ABSTRACT

Objective: To analyze the clinical characteristics and prognosis of patients with primary aldosteronism (PA) associated with subclinical Cushing syndrome (SCS). Methods: This retrospective cohort study was conducted at the First Affiliated Hospital of Chongqing Medical University in China. Patients with PA were included between January 2014 and December 2022. According to the results of 1-mg overnight dexamethasone suppression test, the patients were divided into the PA group and PA associated with SCS (PA/SCS) group. The demographic information, hormone levels, and follow-up results were analyzed. Independent sample t-test, chi-square test and Mann-Whitney U test were used for data comparison. Results: A total of 489 PA patients were enrolled in this study, of which 109 had PA/SCS (22.3%). Patients with SCS were on average older (54.4±10.7 vs. 47.4±11.0, P<0.001); had a larger proportion of women (69.7%, 76/109 vs. 57.4%, 218/380; P=0.020); and a longer duration of hypertension [96 (36, 180) vs. 60 (12, 120) months, P=0.001] than patients without SCS. There were 215 and 51 patients in the PA group and PA/SCS group, who completed adrenalectomy and follow-up, respectively. The remission rate of autonomous cortisol secretion in the PA/SCS group was 85.3% (29/34). There was no significant difference in the remission rate of autonomous aldosterone secretion among patients between the PA/SCS and PA group (94.1%, 48/51 vs. 94.4%, 203/215; P=1.000), while the clinical remission rate in the PA/SCS group was lower than that in the PA group (39.2%, 20/51 vs. 61.9%, 133/215; P=0.003). Conclusions: SCS is common in PA patients (22.3%), and the clinical remission rate is low. Screening using the 1-mg overnight dexamethasone suppression test is recommended for all patients with PA.


Subject(s)
Adrenal Gland Neoplasms , Cushing Syndrome , Hyperaldosteronism , Humans , Female , Cushing Syndrome/complications , Cushing Syndrome/diagnosis , Adrenal Gland Neoplasms/complications , Retrospective Studies , Hyperaldosteronism/complications , Hyperaldosteronism/diagnosis , Prognosis , Dexamethasone/therapeutic use , Aldosterone
7.
Am J Case Rep ; 25: e943005, 2024 Apr 07.
Article in English | MEDLINE | ID: mdl-38582958

ABSTRACT

BACKGROUND 21-hydroxylase deficiency, an essential enzyme for glucocorticoid and mineralocorticoid synthesis, is the cause of congenital adrenal hyperplasia (CAH) in more than 95% of cases. It is an autosomal recessive disorder encoded by the CYP21A2 gene, categorized into classical forms, which encompass the salt-wasting (SW) and simple virilizing (SV) forms, as well as the nonclassical form (NC). The aim of medical treatment is to replace missing glucocorticoids and, if necessary, mineralocorticoids, while also reducing elevated adrenal androgens. CASE REPORT We present the case of a 42-year-old woman with CAH who discontinued therapy during adolescence and was admitted to hospital with fatigue, nausea, and severe abdominal pain. A CT scan showed an extreme enlargement of the adrenal glands. Laboratory tests revealed elevated levels of 17-hydroxyprogesterone and other adrenal androgens, along with normal plasma metanephrine levels. Decreased morning cortisol levels suggested partial adrenal insufficiency requiring glucocorticoid replacement therapy. Due to the development of several serious complications and clinical deterioration, the multidisciplinary team recommended bilateral removal of masses measuring 300×250×200 mm on the right side and 250×200×200 mm on the left side. Histological and immunochemical examination confirmed the presence of giant myelolipomas with adrenal cortex hyperplasia. CONCLUSIONS Adrenal tumors, particularly myelolipomas, have a higher prevalence in patients with CAH. Our case report provides further evidence of the suspected link between non-compliant CAH therapy and the development of myelolipomas, along with promotion of their pronounced growth.


Subject(s)
Adrenal Gland Neoplasms , Adrenal Hyperplasia, Congenital , Lipoma , Myelolipoma , Adult , Female , Humans , Adrenal Gland Neoplasms/complications , Adrenal Gland Neoplasms/diagnosis , Adrenal Glands , Adrenal Hyperplasia, Congenital/complications , Adrenal Hyperplasia, Congenital/diagnosis , Adrenal Hyperplasia, Congenital/genetics , Glucocorticoids/therapeutic use , Myelolipoma/diagnosis , Myelolipoma/surgery , Myelolipoma/complications , Steroid 21-Hydroxylase/genetics
8.
Front Endocrinol (Lausanne) ; 15: 1370525, 2024.
Article in English | MEDLINE | ID: mdl-38596218

ABSTRACT

Introduction: Endocrine hypertension (EHT) due to pheochromocytoma/paraganglioma (PPGL), Cushing's syndrome (CS), or primary aldosteronism (PA) is linked to a variety of metabolic alterations and comorbidities. Accordingly, patients with EHT and primary hypertension (PHT) are characterized by distinct metabolic profiles. However, it remains unclear whether the metabolomic differences relate solely to the disease-defining hormonal parameters. Therefore, our objective was to study the association of disease defining hormonal excess and concomitant adrenal steroids with metabolomic alterations in patients with EHT. Methods: Retrospective European multicenter study of 263 patients (mean age 49 years, 50% females; 58 PHT, 69 PPGL, 37 CS, 99 PA) in whom targeted metabolomic and adrenal steroid profiling was available. The association of 13 adrenal steroids with differences in 79 metabolites between PPGL, CS, PA and PHT was examined after correction for age, sex, BMI, and presence of diabetes mellitus. Results: After adjustment for BMI and diabetes mellitus significant association between adrenal steroids and metabolites - 18 in PPGL, 15 in CS, and 23 in PA - were revealed. In PPGL, the majority of metabolite associations were linked to catecholamine excess, whereas in PA, only one metabolite was associated with aldosterone. In contrast, cortisone (16 metabolites), cortisol (6 metabolites), and DHEA (8 metabolites) had the highest number of associated metabolites in PA. In CS, 18-hydroxycortisol significantly influenced 5 metabolites, cortisol affected 4, and cortisone, 11-deoxycortisol, and DHEA each were linked to 3 metabolites. Discussions: Our study indicates cortisol, cortisone, and catecholamine excess are significantly associated with metabolomic variances in EHT versus PHT patients. Notably, catecholamine excess is key to PPGL's metabolomic changes, whereas in PA, other non-defining adrenal steroids mainly account for metabolomic differences. In CS, cortisol, alongside other non-defining adrenal hormones, contributes to these differences, suggesting that metabolic disorders and cardiovascular morbidity in these conditions could also be affected by various adrenal steroids.


Subject(s)
Adrenal Gland Neoplasms , Cortisone , Cushing Syndrome , Diabetes Mellitus , Hypertension , Paraganglioma , Pheochromocytoma , Female , Humans , Middle Aged , Male , Hydrocortisone/metabolism , Retrospective Studies , Cushing Syndrome/complications , Steroids , Adrenal Gland Neoplasms/complications , Hypertension/complications , Pheochromocytoma/complications , Paraganglioma/complications , Catecholamines , Dehydroepiandrosterone
9.
J Am Vet Med Assoc ; 262(6): 1-6, 2024 Jun 01.
Article in English | MEDLINE | ID: mdl-38608654

ABSTRACT

OBJECTIVE: Thromboelastography (TEG) is a whole blood assay that yields global assessment of hemostasis, as it evaluates clot time, strength, and kinematics of clot formation and lysis. The main objective was to describe preoperative TEG findings in dogs that had an adrenalectomy performed and, secondarily, to describe TEG findings in the dogs with or without hyperadrenocorticism (HAC). ANIMALS: 30 dogs that had preoperative TEG and adrenalectomy performed. METHODS: Medical records between 2018 and 2022 were reviewed. Signalment, diagnostic data, and perioperative treatment were abstracted. RESULTS: 53% (16/30) of the dogs were hypercoagulable, and none were hypocoagulable. Based on histopathology, 6 of 9 dogs with adenocarcinoma were hypercoagulable, 4 of 8 with pheochromocytoma were hypercoagulable, and 6 of 10 with adenoma were hypercoagulable. None of the 3 dogs with other histopathologic diagnoses or combinations of diagnoses (adrenocortical hyperplasia, poorly differentiated sarcoma, and both adrenocortical adenocarcinoma and pheochromocytoma) were hypercoagulable. Of the 14 dogs tested preoperatively for HAC, 4 of 8 HAC dogs were hypercoagulable and 2 of 6 non-HAC dogs were hypercoagulable. CLINICAL RELEVANCE: The present report describes for the first time TEG findings for dogs undergoing adrenalectomy and suggests that the majority of dogs with adrenal neoplasia are hypercoagulable based on TEG results.


Subject(s)
Adrenalectomy , Adrenocortical Hyperfunction , Dog Diseases , Thrombelastography , Thrombophilia , Animals , Dogs , Thrombelastography/veterinary , Adrenalectomy/veterinary , Dog Diseases/surgery , Dog Diseases/diagnosis , Dog Diseases/blood , Male , Female , Thrombophilia/veterinary , Thrombophilia/diagnosis , Adrenocortical Hyperfunction/veterinary , Adrenocortical Hyperfunction/complications , Retrospective Studies , Adrenal Gland Neoplasms/veterinary , Adrenal Gland Neoplasms/surgery , Adrenal Gland Neoplasms/complications , Pheochromocytoma/veterinary , Pheochromocytoma/surgery
10.
Curr Probl Cardiol ; 49(7): 102578, 2024 Jul.
Article in English | MEDLINE | ID: mdl-38657719

ABSTRACT

INTRODUCTION: Pheochromocytoma is a rare cause of hypertensive emergency. The objective of this analysis was to compare the clinical characteristics, comorbidities, and in-hospital outcomes of patients admitted with hypertensive emergencies with and without co-existing pheochromocytoma. METHODS: A retrospective analysis of the National Inpatient Sample (NIS) Database from 2016 to 2020 was conducted, encompassing 640,395 patients hospitalized for hypertensive emergencies, including 2535 patients diagnosed with pheochromocytoma. We compared demographics, comorbidities, in-hospital outcomes and resource utilization metrics in patients with and without pheochromocytoma. Propensity-score matching was utilized to account for potential confounders and risk of complications was compared. RESULTS: Among the pheochromocytoma cohort (51.9% female), a significant portion (35.7%) were under 50 years of age, with the majority being Caucasian (47.9%). Comorbid conditions such as obesity, diabetes, and smoking were prevalent, with notable differences in cancer (7.5% vs. 2.3%, p < 0.001) and peripheral vascular disease (17% vs. 8.2%, p < 0.001) rates compared to the non-pheochromocytoma cohort. Pheochromocytoma patients had a longer hospital stay (7.5 vs. 6 days, p = 0.002) and higher odds of acute kidney injury (AKI) (1.54, 1.18-2, p=0.001) but lower odds of requiring hemodialysis (0.52, 0.32-0.79, p < 0.001) or experiencing major cardiovascular events (0.5, 0.36-0.69, p < 0.001). No significant difference in inflation-adjusted hospitalization costs was found between the groups. CONCLUSIONS: Patients with hypertensive emergencies and pheochromocytoma had a higher incidence of AK, certain comorbidities (cancer, peripheral vascular disease), and more complex hospital courses suggested by longer length of stay. However, the overall cost of hospitalization did not significantly differ between the two cohorts.


Subject(s)
Adrenal Gland Neoplasms , Hospital Mortality , Hypertension , Pheochromocytoma , Propensity Score , Humans , Pheochromocytoma/complications , Pheochromocytoma/epidemiology , Female , Male , Middle Aged , United States/epidemiology , Hypertension/epidemiology , Retrospective Studies , Adrenal Gland Neoplasms/complications , Adrenal Gland Neoplasms/epidemiology , Hospital Mortality/trends , Aged , Adult , Comorbidity , Emergencies , Hypertensive Crisis
11.
BMJ Case Rep ; 17(4)2024 Apr 25.
Article in English | MEDLINE | ID: mdl-38670572

ABSTRACT

Pheochromocytoma is a chromaffin cell-derived adrenal medullary tumour and usually presents with paroxysms of hypertension, palpitations, sweating and headache due to excessive catecholamine release. These tumours can also secrete a variety of bioactive neuropeptides and hormones other than catecholamines, resulting in unusual clinical manifestations. We report a female in her mid-30s who presented with fever, anaemia, thrombocytosis and markedly elevated inflammatory markers. The fever profile, including cultures, was negative. Contrast-enhanced CT of abdomen showed a large solid-cystic right adrenal lesion with elevated plasma-free normetanephrine levels suggestive of pheochromocytoma. The fever persisted despite empirical antibiotics and antipyretics. Interleukin-6 (IL-6) levels were elevated (41.2 pg/mL (3-4 pg/mL)). She was initiated on naproxen (NPX) at a dose of 250 mg two times per day. The patient responded to NPX, and after stabilisation, she underwent an adrenalectomy. There was a complete resolution of fever with normalisation of IL-6 levels postoperatively.


Subject(s)
Adrenal Gland Neoplasms , Adrenalectomy , Interleukin-6 , Pheochromocytoma , Humans , Pheochromocytoma/complications , Pheochromocytoma/surgery , Pheochromocytoma/blood , Female , Adrenal Gland Neoplasms/complications , Adrenal Gland Neoplasms/surgery , Adrenal Gland Neoplasms/diagnosis , Adrenal Gland Neoplasms/blood , Interleukin-6/blood , Adult , Naproxen/therapeutic use , Fever/etiology , Anti-Inflammatory Agents, Non-Steroidal/therapeutic use , Anti-Inflammatory Agents, Non-Steroidal/administration & dosage , Tomography, X-Ray Computed
12.
J Pediatr Endocrinol Metab ; 37(5): 482-485, 2024 May 27.
Article in English | MEDLINE | ID: mdl-38618883

ABSTRACT

OBJECTIVES: Childhood cancer survivors are at risk for premature ovarian insufficiency, especially after treatment with alkylating agents. The objective of this report is to highlight a case in which this phenomenon caused a false-positive pregnancy test. CASE PRESENTATION: A workup was performed in a 14-year-old girl with a positive pregnancy test. She was diagnosed with stage IV neuroblastoma of the left adrenal gland at the age of 4 years. She received extensive treatment, including alkylating agents, and had been diagnosed with premature ovarian insufficiency. An LH/hCG suppression test was performed using high dose 17 bèta-estradiol: hCG levels normalized. CONCLUSIONS: The pregnancy test was false-positive due to production of low amounts of hCG by the pituitary gland as a result of high LH concentrations following premature ovarian insufficiency. It may be helpful to perform the LH/hCG suppression test to prove pituitary origin of the hCG overproduction.


Subject(s)
Primary Ovarian Insufficiency , Humans , Female , Primary Ovarian Insufficiency/diagnosis , Primary Ovarian Insufficiency/pathology , Adolescent , Pregnancy , Pregnancy Tests , Neuroblastoma/complications , Neuroblastoma/pathology , Neuroblastoma/drug therapy , Adrenal Gland Neoplasms/complications , Adrenal Gland Neoplasms/pathology , Adrenal Gland Neoplasms/diagnosis , False Positive Reactions , Luteinizing Hormone/blood , Prognosis
13.
Ned Tijdschr Geneeskd ; 1682024 03 19.
Article in Dutch | MEDLINE | ID: mdl-38546048

ABSTRACT

Spontaneous retroperitoneal hematoma refers to bleeding of the retroperitoneal space without prior trauma or iatrogenic injury. Its diagnosis is often delayed due to a nonspecific clinical presentation. This case-series presents three patients with spontaneous retroperitoneal bleeding. Patient A presented with a spontaneous bleeding around the right iliopsoas muscle due to antiplatelet medication and was managed conservatively. Patient B had a spontaneous arterial bleeding near the right kidney, subsequently determined to be associated with a pheochromocytoma. Endovascular treatment successfully stopped the bleeding. The pheochromocytoma was resected during follow-up. Patient C had a spontaneous rupture of the left iliac vein with a concomitant deep vein thrombosis. She underwent emergent laparotomy, but unfortunately did not survive. This case-series emphasize the diverse presentation, varied causes, and different treatment strategies for spontaneous retroperitoneal hematoma. Timely diagnosis is hard, but crucial to enable an individualized treatment.


Subject(s)
Adrenal Gland Neoplasms , Pheochromocytoma , Female , Humans , Pheochromocytoma/complications , Hematoma/diagnosis , Hematoma/etiology , Hematoma/surgery , Gastrointestinal Hemorrhage , Platelet Aggregation Inhibitors , Rupture, Spontaneous , Adrenal Gland Neoplasms/complications
14.
Int J Mol Sci ; 25(5)2024 Feb 29.
Article in English | MEDLINE | ID: mdl-38474067

ABSTRACT

Severe obesity in young children prompts for a differential diagnosis that includes syndromic conditions. Rapid-Onset Obesity with Hypothalamic Dysfunction, Hypoventilation, and Autonomic Dysregulation (ROHHAD) syndrome is a potentially fatal disorder characterized by rapid-onset obesity associated with hypoventilation, neural crest tumors, and endocrine and behavioral abnormalities. The etiology of ROHHAD syndrome remains to be established, but recent research has been focusing on autoimmunity. We report on a 2-year-old girl with rapid-onset obesity during the first year of life who progressed to hypoventilation and encephalitis in less than four months since the start of accelerated weight gain. The patient had a high titer of anti-ZSCAN1 antibodies (348; reference range < 40), and the increased values did not decline after acute phase treatment. Other encephalitis-related antibodies, such as the anti-NDMA antibody, were not detected. The rapid progression from obesity onset to central hypoventilation with encephalitis warns about the severe consequences of early-onset ROHHAD syndrome. These data indicate that serial measurements of anti-ZSCAN1 antibodies might be useful for the diagnosis and estimation of disease severity. Further research is needed to determine whether it can predict the clinical course of ROHHAD syndrome and whether there is any difference in antibody production between patients with and without tumors.


Subject(s)
Adrenal Gland Neoplasms , Autonomic Nervous System Diseases , Encephalitis , Hypothalamic Diseases , Pediatric Obesity , Female , Humans , Child, Preschool , Hypoventilation/complications , Hypoventilation/diagnosis , Pediatric Obesity/complications , Adrenal Gland Neoplasms/complications , Syndrome , Encephalitis/complications
15.
J Med Case Rep ; 18(1): 161, 2024 Mar 19.
Article in English | MEDLINE | ID: mdl-38500192

ABSTRACT

BACKGROUND: This report presents a case of cannabinoid-induced hyperemesis syndrome causing repeated violent retching in a patient with a large (8 cm) adrenal pheochromocytoma resulting in hypertensive urgency. CASE PRESENTATION: A 69-year-old white male patient with a previously diagnosed pheochromocytoma presented to the emergency department with nausea and vomiting and was found to have hypertensive urgency. Computed tomography scan did not show any acute abdominal pathology and history was inconsistent with a gastrointestinal etiology. Patient had a history of daily cannabinoid use for many years and repeated self-limited hyperemesis episodes, and thus a diagnosis of cannabinoid-induced hyperemesis syndrome was made. It was concluded that the likely explanation for the hypertensive urgency was from physical compression of his adrenal tumor during the episodes of retching resulting in a catecholamine surge. The patient was given antiemetics and admitted to the intensive care unit for blood pressure management. Blood pressure was initially controlled with phentolamine and a clevidipine infusion, then transitioned to oral doxazosin and phenoxybenzamine. Hyperemesis and abdominal pain resolved after 24 hours, and his blood pressure returned to baseline. The patient was discharged with the recommendation to stop all cannabis use. On follow-up, his blood pressure remained well controlled, and he subsequently underwent adrenalectomy for tumor removal. CONCLUSION: Hyperemesis can cause hypertensive events in patients with pheochromocytoma by increasing abdominal pressure, leading to catecholamine release.


Subject(s)
Adrenal Gland Neoplasms , Cannabinoid Hyperemesis Syndrome , Cannabinoids , Hypertensive Crisis , Pheochromocytoma , Aged , Humans , Male , Adrenal Gland Neoplasms/complications , Adrenal Gland Neoplasms/diagnostic imaging , Adrenal Gland Neoplasms/surgery , Cannabinoids/adverse effects , Catecholamines , Pheochromocytoma/complications , Pheochromocytoma/diagnostic imaging , Pheochromocytoma/surgery , Vomiting/chemically induced
16.
Curr Probl Cardiol ; 49(6): 102521, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38492617

ABSTRACT

Fulminant myocarditis (FM) is a rare illness characterized by abrupt and severe widespread cardiac inflammation, which frequently results in mortality due to cardiogenic shock, ventricular arrhythmias, or multiorgan system failure. Pheochromocytoma is an uncommon and difficult-to-diagnose cause of FM, and it is associated with a significant risk of recurrent acute myocarditis. There is, however, little information on reoccurring acute FM. Herein, we report a rare case of recurrent acute FM due to pheochromocytoma. We present the case of a 22-year-old woman who was admitted to our hospital three days previously with acute dyspnea. Five months prior, the patient was diagnosed with post-acute myocarditis, and a massive tumor on the right adrenal gland was discovered, which lead to pheochromocytoma diagnosis. In this present admission, following the exclusion of infection, autoimmune, and metabolic derangements, pheochromocytoma was presumed to be the reason for the recurrence and more severe acute FM during the current hospitalization. The patient responded favorably to high-dose steroids combined with heart failure therapy regimens. To detect recurrent acute myocarditis related to pheochromocytoma, a multidisciplinary approach was used, including several laboratory biomarkers and imaging findings. Following pheochromocytoma removal and biopsy, the patient recovered satisfactorily. Our findings may provide beneficial contributions to the literature as pheochromocytoma is an uncommon but important cause of recurrent acute myocarditis. A multidisciplinary approach is essential in identifying acute FM and determining the underlying causes of this malady.


Subject(s)
Adrenal Gland Neoplasms , Myocarditis , Pheochromocytoma , Recurrence , Humans , Pheochromocytoma/diagnosis , Pheochromocytoma/complications , Myocarditis/diagnosis , Myocarditis/etiology , Myocarditis/therapy , Female , Adrenal Gland Neoplasms/diagnosis , Adrenal Gland Neoplasms/complications , Young Adult , Acute Disease , Tomography, X-Ray Computed , Adrenalectomy/methods
17.
J Clin Hypertens (Greenwich) ; 26(4): 416-424, 2024 Apr.
Article in English | MEDLINE | ID: mdl-38459755

ABSTRACT

This study aimed to elucidate the clinical diagnostic value of plasma catecholamines and their metabolites for pheochromocytoma and paraganglioma (PPGL)-induced secondary hypertension using ultraperformance liquid chromatography-mass spectrometry (UPLC-MS/MS). The study population included 155 patients with PPGL that were divided into the PPGL with hypertension (n = 79) and a PPGL without hypertension (n = 76) groups, and 90 healthy volunteers and 90 patients with primary hypertension as the control groups. UPLC-MS/MS was performed to detect plasma levels of catecholamines and their metabolites, including dopamine, vanillylmandelic acid (VMA), norepinephrine, metanephrine, and normetanephrine. Receiver operating characteristic curves were generated to analyze the diagnostic value of the plasma levels of catecholamines and their metabolites in PPGL-induced secondary hypertension. Patients in the primary hypertension and PPGL without hypertension groups had higher levels of dopamine, VMA, norepinephrine, metanephrine, and normetanephrine than patients in the normal group (all p < .05). On the other hand, patients in the PPGL with hypertension group had higher levels of dopamine, VMA, norepinephrine, metanephrine, and normetanephrine than patients in the normal, primary hypertension, and PPGL without hypertension groups (all p < .05). Collectively, our findings showed that dopamine, VMA, norepinephrine, metanephrine, and normetanephrine are all effective biomarkers for the diagnosis of PPGL and PPGL-induced secondary hypertension.


Subject(s)
Adrenal Gland Neoplasms , Hypertension , Mandelic Acids , Paraganglioma , Pheochromocytoma , Humans , Catecholamines , Pheochromocytoma/complications , Pheochromocytoma/diagnosis , Metanephrine , Normetanephrine , Dopamine , Chromatography, Liquid/methods , Liquid Chromatography-Mass Spectrometry , Hypertension/diagnosis , Tandem Mass Spectrometry/methods , Paraganglioma/complications , Paraganglioma/diagnosis , Norepinephrine , Adrenal Gland Neoplasms/complications , Adrenal Gland Neoplasms/diagnosis , Essential Hypertension/diagnosis
18.
Medicine (Baltimore) ; 103(5): e36127, 2024 Feb 02.
Article in English | MEDLINE | ID: mdl-38306535

ABSTRACT

INTRODUCTION: To investigate the clinical features, pregnancy care, timing, and approaches of pregnancy termination as well as the perinatal management of pregnant women with ectopic pheochromocytomas (EPCC) (paragangliomas, PGL). METHODS: We report the diagnosis and treatment of a pregnant women with EPCC which was confirmed in the third trimester in our hospital. Literature in relation to EPCC during pregnancy both in and outside China was searched for data analysis such as maternal clinical features and maternal and fetal prognosis. RESULTS: A total of 20 papers including 21 cases (plus ours) were retrieved. The average age of pregnant patients was 28 years old (from 21 to 37). Two patients presented no hypertension. Nineteen had hypertension in various extent with the accompany of headache (11 cases, 57.9%), palpitations (8 cases, 42.1%), sweating (6 cases, 31.6%), nausea (6 cases), abdominal pain (2 cases), etc. The tumor was found in the chest in 3 patients, in the upper abdomen in 1 patient, in the middle abdomen in 10 patients, between the lower abdomen and pelvic cavity in 3 patients and in the pelvic cavity in 3 patients. Five patients had a surgical removal of the tumor before delivery, 3 during cesarean section and 10 after giving birth. CONCLUSION: EPCC (PGL) during pregnancy is a rare extra-adrenal tumor, whose manifestations are often confused with those of pregnancy-induced hypertension. It is extremely hard to diagnosis the disease before surgery. Patients still have an opportunity of undergoing spontaneous delivery if their tumors have been removed before labor. However, for patients whose pheochromocytomas is localized before labor, it is better to terminate their pregnancy via cesarean section in a proper time according to their obstetric conditions, while under the supervision of multidisciplinary specialists. The preparations of both α and ß adrenergic receptor blocker treatment that is normally carried out before PGL removal surgery are unnecessary to be overemphasized before the cesarean section.


Subject(s)
Adrenal Gland Neoplasms , Hypertension , Labor, Obstetric , Pheochromocytoma , Pregnancy , Humans , Female , Adult , Pregnancy Trimester, Third , Cesarean Section , Pheochromocytoma/complications , Pheochromocytoma/diagnosis , Pheochromocytoma/surgery , Adrenal Gland Neoplasms/complications , Adrenal Gland Neoplasms/diagnosis , Adrenal Gland Neoplasms/surgery , Retrospective Studies
19.
Hipertens Riesgo Vasc ; 41(1): 62-65, 2024.
Article in Spanish | MEDLINE | ID: mdl-38402070

ABSTRACT

Post-COVID-19 syndrome is a set of symptoms and signs that persist for more than 12 weeks after COVID-19 infection and currently lacks a standardised clinical definition. Only one case has been reported in which a pheochromocytoma was mistaken for post-COVID-19 syndrome. The symptomatology of this syndrome is variable and ranges from headache and fatigue to persistent dyspnoea and neurocognitive disturbances. In addition, SARS-CoV-2 can affect the autonomic nervous system, contributing to symptoms resembling those of pheochromocytoma. The importance and need to discern between COVID-19-related symptoms and other conditions is emphasised, as the specificity of the clinical manifestations of post-COVID-19 syndrome is very low and can be confused with other vital pathologies. A case is presented in which a pheochromocytoma was mistaken for post-COVID-19 syndrome in a patient with no medical history.


Subject(s)
Adrenal Gland Neoplasms , COVID-19 , Hypertension , Pheochromocytoma , Humans , COVID-19/complications , COVID-19/diagnosis , Pheochromocytoma/complications , Pheochromocytoma/diagnosis , Post-Acute COVID-19 Syndrome , SARS-CoV-2 , Adrenal Gland Neoplasms/complications , Adrenal Gland Neoplasms/diagnosis , Headache/etiology , Hypertension/diagnosis , Hypertension/etiology
20.
BMJ Case Rep ; 17(2)2024 Feb 06.
Article in English | MEDLINE | ID: mdl-38320821

ABSTRACT

We report the case of a middle-aged hypertensive woman presenting to the neurology department with short-lasting episodic headaches for 4 years. She was initially diagnosed and treated with cluster headaches for one year. Following this, she presented with right lower limb arterial claudication. Arterial Doppler of lower limbs showed thrombosis of the bilateral common femoral arteries. Further computed tomography (CT) angiogram of the lower limbs confirmed extensive arterial thrombosis in bilateral lower limbs. The CT angiogram incidentally detected a left adrenal lesion. She had elevated urinary vanillylmandelic Acid and 24-hour metanephrines suggesting the presence of a pheochromocytoma. She was initially medically managed and later underwent left open adrenalectomy. Histopathology examination of the sections proved pheochromocytoma. Postsurgery, the patient's symptoms improved remarkably. This case highlights the importance of diagnosing pheochromocytoma when you encounter a patient with refractory short-lasting headaches, hypertension and hypercoagulability.


Subject(s)
Adrenal Gland Neoplasms , Hypertension , Pheochromocytoma , Thrombophilia , Thrombosis , Middle Aged , Female , Humans , Pheochromocytoma/complications , Pheochromocytoma/diagnosis , Pheochromocytoma/surgery , Hypertension/etiology , Hypertension/surgery , Adrenal Gland Neoplasms/complications , Adrenal Gland Neoplasms/diagnosis , Adrenal Gland Neoplasms/surgery , Adrenalectomy , Headache/etiology , Headache/surgery , Thrombophilia/complications , Thrombophilia/diagnosis , Thrombosis/surgery
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