Initiation of levothyroxine in a patient with hypothyroidism inducing adrenal crisis requiring VA ECMO: a tale of preventable disaster

Kang, Mandip; Sandhu, Charnjeet; Singh, Natasha Ramroop; Evans, Timothy

doi:10.1136/bcr-2019-230601

Cited by 12 publications

(11 citation statements)

References 7 publications

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“…Kang et al . 17 reported a case of a 31-year-old man with hypothyroidism inducing adrenal crisis who was also successfully salvaged by VA-ECMO support and whose subsequent autoimmune work-up confirmed the diagnosis of autoimmune polyglandular syndrome type 2 with positive antiperoxidase antibodies, supporting the diagnosis of Hashimoto’s thyroiditis. Additionally, Krishnamoorthy et al .…”

Section: Discussionmentioning

confidence: 87%

Venoarterial extracorporeal membrane oxygenation as bridge to effective treatment in a 19-year-old woman with acute adrenal crisis: a case report

Lüsebrink

Krieg

Maßberg

et al. 2021

European Heart Journal - Case Reports

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Background Adrenal crisis is an acute life-threatening exacerbation of the Addison’s disease or primary adrenal insufficiency (PAI) and is associated with a high mortality rate. It can be the first manifestation of adrenal insufficiency and is caused by a critical lack of glucocorticoids. Case summary Here, we report the case of a 19-year-old woman presenting to the emergency room with unspecific symptoms, i.e. lethargy, fatigue, arthralgia, dyspnoea, and hypotension. The patient’s examination showed major pericardial effusion resulting in cardiac tamponade requiring immediate pericardiocentesis. In the further course, acute right heart failure and progressive cardiogenic shock occurred. Due to recurrent bradycardia and finally asystole, the patient had to be resuscitated for 15 min in total until return of spontaneous circulation. However, non-invasive haemodynamic stabilization of the patient was not successful. Hence, venoarterial extracorporeal membrane oxygenation (VA-ECMO) was used as salvage intervention to provide temporary circulatory support. We diagnosed an Addison crisis as first manifestation of her previously unknown Addison’s disease. An appropriate substitution therapy with hydrocortisone and fludrocortisone was immediately initiated and the patient’s condition rapidly improved. After a total in-hospital stay of 4 weeks, she recovered completely and could be discharged from hospital. Discussion An Addison crisis requires rapid diagnosis and immediate treatment to end a life-threatening condition caused by critical glucocorticoid deficiency. In patients with non-specific symptoms, such as fatigue, hypotension, weight loss, and hyponatraemia, adrenocortical insufficiency should be considered as differential diagnosis. If patients suffer from an Addison crisis, clinical suspicion requires immediate substitution of hydrocortisone as this is essential for patient’s survival. Venoarterial extracorporeal membrane oxygenation therapy can serve as a bridge to diagnosis and effective treatment in patients requiring temporary cardiopulmonary support, especially as salvage intervention for patients in cardiogenic shock. To our knowledge, this is the first case of a young patient with acute Addison crisis and cardiogenic shock, who was successfully salvaged by VA-ECMO support.

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Section: Discussionmentioning

confidence: 87%

Venoarterial extracorporeal membrane oxygenation as bridge to effective treatment in a 19-year-old woman with acute adrenal crisis: a case report

Lüsebrink

Krieg

Maßberg

et al. 2021

European Heart Journal - Case Reports

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“…Primary hypothyroidism and type 2 diabetes may also contribute to the development of an AC, albeit that it remains elusive, how osteoporosis may culminate in an AC. Either initiation or overtreatment of hypothyroidism has been shown to precipitate an AC in vulnerable persons for example, autoimmune polyglandular syndrome where hypothyroidism and AD can coexist, raising the cortisol demands [27]. In a Swedish national in-patient cohort, coexistence of diabetes and AD represented a 4-fold increased risk of all-cause mortality, compared with matched controls of diabetes alone and although cardiovascular risk was the most common cause of death, infections and potentially an AC, were more common than in diabetics alone [28].…”

Section: Discussionmentioning

confidence: 99%

Knowledge of crisis prevention strategies are highly effective, whereas vulnerability scores may elicit Addison’s disease crises

Ross

Anderson

Demeke

et al. 2022

Preprint

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Objective: The knowledge in patients with Addison’s disease (AD) to prevent an adrenal crisis and the degree to which patients feel vulnerable. Design: Telephonic interview of patients with AD in 47 patients, assessing knowledge of prevention of a crisis and vulnerability using a vulnerability scale. Methods: Knowledge of adrenal crisis (AC) and vulnerability score completed. Patients were compared based on having had a crisis or not. Factors associated with AC and the impact of knowledge, vulnerability were determined. Results: (64%) had an adrenal crisis in the previous five years, compared with 34% who never had a crisis. Crisis was associated with poorer knowledge prevention of an AC, especially having a cold (31.2% vs 71%; p=0.002) fever (44.7% vs 90.3%; p<0.001) infection (50% vs 93.5%; p<0.001) vomiting (50% vs 80.6%; p=0.007), and diarrhoea (37.5% vs 71%; p<0.001), respectively. The median proportion with vulnerability for each question was 41.5%. Disease duration was negatively correlated with vulnerability; (r=-0.426, p=0.003). Number of comorbidities was correlated with vulnerability (r=0.290, p=0.048). A negative correlation between knowledge score and number of crises in the previous five years (r=-0.530, p<0.001). Logistic regressions showed the only predictor for a crisis in the past year or past 5 years was patients’ knowledge (p<0.001). Predictors of knowledge were younger patients, more comorbidities, patients who had AC, scored higher on vulnerability questionnaire. Variables accounted for 52% of the variance (F=9.751, p<0.001). Conclusions: Education is critical in preventing AC, specifically cold, fever, infection, vomiting and diarrhoea. Health-care providers should provide psychological support, considering significant vulnerability burden.

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“…Other endocrinopathies of APS-1 are usually managed with hormonal replacement treatment. Initiation of levothyroxine before glucocorticoid replacement in APS-1 or APS-2 hypothyroid patients with AD can lead to the precipitation of adrenal crisis [ 38 ]. If asplenism is found, vaccinations against encapsulated bacteria ( Haemophilus influenzae type b, Neisseria meningitidis , and Streptococcus pneumoniae ) should be administered [ 39 ].…”

Section: Autoimmune Polyendocrine Syndromesmentioning

confidence: 99%

Autoimmune Polyendocrine Syndromes in the Pediatric Age

et al. 2023

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Autoimmune polyendocrine syndromes (APSs) encompass a heterogeneous group of rare diseases characterized by autoimmune activity against two or more endocrine or non-endocrine organs. Three types of APSs are reported, including both monogenic and multifactorial, heterogeneous disorders. The aim of this manuscript is to present the main clinical and epidemiological characteristics of APS-1, APS-2, and IPEX syndrome in the pediatric age, describing the mechanisms of autoimmunity and the currently available treatments for these rare conditions.

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Initiation of levothyroxine in a patient with hypothyroidism inducing adrenal crisis requiring VA ECMO: a tale of preventable disaster

Cited by 12 publications

References 7 publications

Venoarterial extracorporeal membrane oxygenation as bridge to effective treatment in a 19-year-old woman with acute adrenal crisis: a case report

Venoarterial extracorporeal membrane oxygenation as bridge to effective treatment in a 19-year-old woman with acute adrenal crisis: a case report

Knowledge of crisis prevention strategies are highly effective, whereas vulnerability scores may elicit Addison’s disease crises

Autoimmune Polyendocrine Syndromes in the Pediatric Age

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