Psychology and hereditary angioedema: A systematic review

Savarese, Livia; Mormile, Ilaria; Bova, María; Petraroli, Angelica; Maiello, Assunta; Spadaro, Giuseppe; Freda, Maria Francesca

doi:10.2500/aap.2021.42.200073

Cited by 21 publications

(20 citation statements)

References 46 publications

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“…The diagnostic delay caused by misdiagnosing HAE-nC1INH-UNK prolongs disease burden, as patients will not be free of angioedema attacks. Recurrent angioedema attacks have been shown to greatly impact the quality of many aspects of patients' lives, not only physically, but also because of their effects on mental health, reproductive choices, social relationships, productivity, and work performance (26)(27)(28)(29)(30). Furthermore, on demand and prophylactic treatments for HAE-nC1INH (25) are very costly.…”

Section: Discussionmentioning

confidence: 99%

Case report: Recurrent angioedema: Diagnosing the rare and the frequent

et al. 2022

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Hereditary angiodema with normal C1 inhibitor and unknown mutation (HAE-nC1INH-UNK), an exceedingly rare subtype of HAE, appears to be often diagnosed in patients who do not have this condition, but have mast cell-mediated angioedema. Here, we report two patients diagnosed with HAE-nC1INH-UNK by their physicians, who referred them to our center for treatment continuation with costly kallikrein-kinin-system targeted therapies. We describe how we established the correct diagnosis of recurrent mast cell-mediated angioedema after thorough investigation of both patients and initiated effective treatment with omalizumab. Also, we present and discuss the consensus criteria for diagnosing the very rare condition HAE-nC1INH in light of recent research and based on our own clinical experience. In conclusion, HAE-nC1INH-UNK should only be considered after more common differential diagnoses, i.e., mast cell-mediated angioedema, have thoroughly been investigated and ruled out. This approach reduces both the patients’ disease burden and healthcare costs and contributes to meaningful research.

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Section: Discussionmentioning

confidence: 99%

Case report: Recurrent angioedema: Diagnosing the rare and the frequent

et al. 2022

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“…Psychological stress was most commonly reported as a trigger factor of attacks in all three angioedema types. This trigger is well known in HAE and CSU 6,9,18,40 , whereas the triggering factors of ACE‐AE are far from being clear.…”

Section: Discussionmentioning

confidence: 99%

Identification of novel biomarkers to distinguish bradykinin‐mediated angioedema from mast cell‐/histamine‐mediated angioedema

Bindke

Gehring

Wieczorek

et al. 2021

Allergy

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Background The pathophysiology of the underlying paroxysmal permeability disturbances in angioedema (AE) is not well understood. Methods To identify clinical and laboratory parameters specific for a certain AE subtype, 40 AE patients were prospectively enrolled: 15 hereditary (HAE), 13 ACE‐inhibitor induced (ACE‐AE), and 12 mast cell‐mediated without wheals in chronic spontaneous urticaria (CSU‐AE). Ten healthy subjects served as controls. Serum levels of markers indicating activation of the ficolin‐lectin pathway, of endothelial cells, or those indicating impairment of vascular integrity or inflammation were assessed by enzyme‐linked immunosorbent assay. Results New routine clinical diagnostic criteria could not be identified, not even for distinguishing bradykinin‐mediated (BK‐) AE (ie, HAE and ACE‐AE) from mast cell‐/histamine‐mediated CSU‐AE. However, FAP‐α and tPA were significantly increased in all AE compared to controls. In HAE, FAP‐ α, tPA, uPAR, pentraxin‐3, Tie‐2, sE‐selectin, and VE‐cadherin were significantly increased compared to controls. In HAE compared to CSU‐AE and ACE‐AE, sE‐Selectin, Tie‐2, and VE‐Cadherin were significantly increased, whereas for Ang‐2 the difference was significant compared to CSU‐AE only. Tie‐2 correlated strongly negatively with C4, C1‐INH activity, and C1‐INH function. Conclusions This study is the first to compare HAE, ACE‐AE, and CSU‐AE. Although significance is limited by small sample size, Tie‐2 was identified as a new promising biomarker candidate for HAE. FAP‐ α and tPA might serve as a marker for AE in general, whereas sE‐selectin and Ang‐2 were increased in BK‐AE only. Our results add information to the role of endothelial dysfunction and serine proteases in different AE subtypes.

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“…HAE not only cause of short-term disability associated with attacks, but also may lead to the development of persistent anxiety between attacks. Savarese et al examined 21 articles on HAE by using the keywords psychology, anxiety, stress, and depression [14]. Although the articles confirm that stress can induce a variety of physical diseases, the authors concluded that multiple factors contribute to HAE expression.…”

Section: Discussionmentioning

confidence: 99%

Depression and anxiety may be cause or effect of attacks in hereditary angioedema

Özden

Bankır

2021

Family Practice and Palliative Care

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Introduction:The study aims to determine the depression and anxiety levels of patients with hereditary angioedema using the Beck depression inventory(BDI), Beck anxiety inventory (BAI) and to compare the results with the patients' demographic characteristics, the number of attacks, attack types, and healthy controls. Methods: 60 hereditary angioedema patients and 60 healthy controls were included in the study. The demographic characteristics of the patients, the total number of attacks/months before treatment, the number of attacks according to the localization of the attacks, and the number of attacks/month after treatment were filled in retrospectively from their hospital records. BDI and BAI were used for anxiety and depression levels. Results: Median BAI (11 vs 5; p=0.001) and BDI (11 vs 7; p=0.024) in HAE patients, the ratio of patients with moderate anxiety (21.7% vs 1.7%; p=0.001) and the ratio of patients with severe anxiety (8.3% vs. 0%; p=0.001) were compared to the control group, was high. There was no statistically significant difference between the number of attacks under treatment and the BAI and BDI scores. A positive correlation was found between the number of untreated attacks and the number of attacks under treatment and the BAI score (r=0.759; p=0.001) and BDI score (r=0.599; p=0.001). Conclusions: Due to the high prevalence of depression and anxiety in HAE patients, health care providers should be attentive of this comorbidity and refer patients to mental health specialists, when needed.

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Psychology and hereditary angioedema: A systematic review

Cited by 21 publications

References 46 publications

Case report: Recurrent angioedema: Diagnosing the rare and the frequent

Case report: Recurrent angioedema: Diagnosing the rare and the frequent

Identification of novel biomarkers to distinguish bradykinin‐mediated angioedema from mast cell‐/histamine‐mediated angioedema

Depression and anxiety may be cause or effect of attacks in hereditary angioedema

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