A case of postural urticaria in a 14-year-old girl
Sofianne Gabrielli, MSc1, Michelle Le,
MD2, Elena Netchiporouk, MD, MSc2,
Moshe Ben-Shoshan, MD, MSc1
1Division of Allergy, Immunology and Dermatology,
Montreal Children’s Hospital, Montreal,
QC, Canada.
2Division of Dermatology, McGill University, Montreal,
QC, Canada.
Keywords: urticaria, postural urticaria, antihistamines, omalizumab
To the Editor,
Chronic urticaria (CU) is defined by the presence of wheals and/or
angioedema for more than six weeks1. CU is classified
as spontaneous (CSU) or inducible (CIndU) urticaria, based on the
ability to identify a trigger1. CIndU, which can be
classified into physical and non-physical forms, is less common than CSU
and can be diagnosed by provocation test. We describe the case of a
14-year-old female patient with postural urticaria induced by changing
from a seating to standing position. To our knowledge, there is only one
other case of postural urticaria described in a teenage
boy2.
A 14-year-old female patient presented with a two-year history of
intermittent hives on her bilateral legs which developed 20 minutes
after standing. The patient also described symptoms of tingling in her
legs prior to the development of the hives. The symptoms usually
resolved after a few hours. In addition, the patient reported
spontaneous hives almost daily not related to an identifiable trigger.
The patient was otherwise healthy and was not taking any medication. The
patient’s father reported a history of urticaria in childhood which
resolved, although he still rarely experiences episodes of hives.
Upon examination, the patient developed prominent vasculature (livedo
reticularis) and urticaria on her bilateral legs 20 minutes after
changing from a seated to a standing position (Figure 1A and 1B). When
seated, the patient’s blood pressure was 113/77 and her heart rate was
102. Upon standing, the patient’s blood pressure was 101/82 and her
heart rate was 118. All laboratory tests, which included a complete
blood count, thyroid stimulating hormone, anti-thyroid peroxidase
Immunoglobulin (Ig) G, tryptase, and total IgE, were within the normal
limits. The patient’s weekly Urticaria Activity Score (UAS7) in the week
prior to exam was 9.
Given poor response to standard doses of second-generation
anti-histamines, the patient was treated with bilastine 40 mg twice
daily. After one year, due to poor response to treatment, the patient
was placed on 300 mg of omalizumab administered once per month. Over the
period of one year, the patient did experience improvement in her
spontaneous urticaria symptoms, with her UAS7 decreasing from 38 to 9
over a period of two years. However, there was no improvement in the
symptoms related to change in posture.
The mechanism of action of postural urticaria in this patient is
unknown. It was previously hypothesized that change in venous pressure
or blood flow volume from sitting to standing may induce the release of
mediators such as acetylcholine that may induce hive
development2.
The management of postural urticaria in our patient was not clear as she
did not improve with antihistamines, nor with the addition of omalizumab
treatment. It possible that the patient may gain better control of her
hives with other management strategies, such as a higher dose of
omalizumab3 or a different biologic, such as
ligelizumab4. Clinicians should be made aware of this
presentation of postural urticaria given that it may reflect a more
recalcitrant form of CIndU. We suggest that a provocation test
consisting of standing for 10 to 20 minutes should be used to confirm
the diagnosis of postural urticaria.