2018
DOI: 10.1186/s13052-018-0578-6
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Anaphylaxis in atypical cold urticaria: case report and review of literature

Abstract: BackgroundCold-induced urticaria is a kind of physical urticaria characterized by the appearance of wheals after exposure to cold. The atypical form is a rare sub-type characterized by appearance of hives even in areas not directly exposed to the cold and by a negative cold stimulation test. Its diagnosis is often challenging because of the lack of specific tests and it is usually based on the patient’s clinical history. Hypotension due to generalized exposure to the cold is described both in the typical and t… Show more

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Cited by 10 publications
(18 citation statements)
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“…Definitely, enzyme-mediated correction of GAG levels can improve some, but not all cellular functions, as dysregulation of important genes may result in various changes in cell physiology even in the absence of GAG storage. One might speculate that such irreversible changes in gene expression regulation can explain a failure of enzyme replacement therapy to correct all MPS symptoms, especially if the treatment is initiated at advanced stages of the disease, the phenomenon that was described previously [ 5 , 6 ].…”
Section: Discussionmentioning
confidence: 98%
See 1 more Smart Citation
“…Definitely, enzyme-mediated correction of GAG levels can improve some, but not all cellular functions, as dysregulation of important genes may result in various changes in cell physiology even in the absence of GAG storage. One might speculate that such irreversible changes in gene expression regulation can explain a failure of enzyme replacement therapy to correct all MPS symptoms, especially if the treatment is initiated at advanced stages of the disease, the phenomenon that was described previously [ 5 , 6 ].…”
Section: Discussionmentioning
confidence: 98%
“…Accumulation of GAGs in lysosomes is the primary cause of MPS, and due to continuous production of these compounds and their impaired degradation, these diseases are progressive and severe, with expected average life span between one and two decades [ 4 ]. Unfortunately, despite incredible progress of molecular genetic studies and extensive medical investigations, only a few therapeutic options are available for MPS patients while none of them being able solve all, or even most, clinical problems [ 5 , 6 ].…”
Section: Introductionmentioning
confidence: 99%
“…In contrast, the atypical form also presents symptoms in areas not directly exposed to the cold and the specific stimulus test is negative. In both forms, hypotension due to generalized exposure to cold is described 1 .…”
Section: Classificationmentioning
confidence: 99%
“…Cold-induced urticaria (CU) is characterized by the appearance of wheals 1 or angioedema 2 after exposure to cold. It is an uncommon subtype of physical urticaria that can present with mild symptoms (localized reaction) and severe reactions, leading to anaphylaxis 3 .…”
Section: Introductionmentioning
confidence: 99%
“…Systemic symptoms rarely occur in CIndUs; acute respiratory involvement, hypotension, lipothymia, and anaphylaxis are occasionally reported in association with cold, cholinergic, or contact urticaria [ 8 , 9 , 10 ]. Although very rarely, these systemic symptoms may also be elicited by CTs; hence, CTs must be conducted only in health centers with resuscitation equipment and trained personnel [ 6 , 7 ].…”
Section: Introductionmentioning
confidence: 99%