title: Omalizumab in Severe Refractory Vernal Keratoconjunctivitis in Children: Case Series and Review of the Literature.
abstract: BACKGROUND
  Vernal keratoconjunctivis (VKC) is a severe form of pediatric ocular allergy, characterized by acute and chronic corneoconjunctival inflammation that may lead to visual sequelae. Although topical immunosuppressive drugs such as cyclosporine are usually effective, some severe forms may be refractory and require prolonged steroid therapy. Very few papers report the use of omalizumab in VKC in the literature. In the present study, we describe our clinical experience with omalizumab in severe VKC children.
  
  
  METHODS
  We retrospectively reviewed the files of four boys treated with omalizumab because of severe VKC, defined as persistent corneal inflammation despite continuous topical 2% cyclosporine and steroid eye drops. We also performed a literature review.
  
  
  RESULTS
  Four boys, aged 7-13 years old, were treated. All children had asthma and one had severe lid eczema. Two patients had required intrapalpebral depot-steroid injections. Omalizumab was administered every 2 weeks by subcutaneous injections, at doses varying from 450 to 600 mg per injection. Three patients out of four responded to the treatment, with a decrease in global symptoms (median symptom rating decreasing from 89 to 29 on a 100-mm visual analog scale), frequency and in duration of the inflammatory flares, and also a decreased need for topical steroid. Their median clinical grade decreased from 4 to 3 (Bonini grading). However, the response was incomplete and they still had inflammatory corneoconjunctival flares despite continuous topical cyclosporine. On the other hand, asthma and lid eczema were completely controlled in these three patients. The fourth child did not respond to omalizumab and needed oral steroids for his VKC and his asthma. Noticeably, this latter patient did not have detectable sensitization to any allergen, contrary to the other cases. The treatment was stopped in this refractory case, but is still ongoing in all other cases, with a median duration of 33 months (range 16-42 months). In the literature (four cases), omalizumab may have a more complete efficacy in some cases, but the results are still variable.
  
  
  CONCLUSIONS
  Omalizumab is an interesting treatment in severe refractory forms of VKC, but its efficacy is incomplete in these very severe cases.
authors.affiliation: Ophthalmology Department of Fondation A de Rothschild and Hôpital Bichat, Paris, France. serge.doan@gmail.com.|Université Paris Diderot, Sorbonne Paris Cité, Paris, France. serge.doan@gmail.com.
authors.forename: Serge
authors.initials: S
authors.lastname: Doan
authors.affiliation: APHP-Hôpital Armand Trousseau, Centre de l'asthme et des allergies, Paris, France.|Université Pierre et Marie Curie, Paris, France.|Equipe EPAR, Institut Pierre Louis d'Epidémiologie et de Santé Publique, UMR_S1136, INSERM, Paris, France.
authors.forename: Flore
authors.initials: F
authors.lastname: Amat
authors.affiliation: Ophthalmology Department of Fondation A de Rothschild and Hôpital Bichat, Paris, France.|Université Paris Diderot, Sorbonne Paris Cité, Paris, France.
authors.forename: Eric
authors.initials: E
authors.lastname: Gabison
authors.affiliation: APHP-Hôpital Armand Trousseau, Centre de l'asthme et des allergies, Paris, France.|Université Pierre et Marie Curie, Paris, France.|Equipe EPAR, Institut Pierre Louis d'Epidémiologie et de Santé Publique, UMR_S1136, INSERM, Paris, France.
authors.forename: Sarah
authors.initials: S
authors.lastname: Saf
authors.affiliation: Ophthalmology Department of Fondation A de Rothschild and Hôpital Bichat, Paris, France.|Université Paris Diderot, Sorbonne Paris Cité, Paris, France.
authors.forename: Isabelle
authors.initials: I
authors.lastname: Cochereau
authors.affiliation: APHP-Hôpital Armand Trousseau, Centre de l'asthme et des allergies, Paris, France.|Université Pierre et Marie Curie, Paris, France.|Equipe EPAR, Institut Pierre Louis d'Epidémiologie et de Santé Publique, UMR_S1136, INSERM, Paris, France.
authors.forename: Jocelyne
authors.initials: J
authors.lastname: Just
keywords: Children; Cyclosporine; IgE; Immunomodulation; Severe allergy; Steroids; VKC