[CIS PIDD] [cis-pidd] reactions to SCIG

[Seppänen Mikko]

My suggestions echo Nacho's. Tolerization scheme has also been published a long time ago by Lennart Hammarstrom and colleagues. See also Tales from the Listserve. 
Does the patient produce IgE? This sounds like delayed hypersensitivity though.

And I would start daily antihistamines a week before the next attempt.

I would also actively look for B-, T- cell clone and MM, though she sounds like a CVID. And  I would definitely check anti-PnP responses. 

Mikko Seppänen Finland


________________________________________
Lähettäjä: Richard Wasserman [drrichwasserman at gmail.com]
Lähetetty: 21. kesäkuuta 2014 15:09
Vastaanottaja: CIS-PIDD
Aihe: [cis-pidd] reactions to SCIG

Colleagues,

I am posting this patient for a colleague.

She is a 49 yo with a history of sinopulmonary infections but no evidence of bronchiectasis. Her IgG ranges from 250 to 350/dL, her IgM is normal her IgA is in the 40s. She has never been treated with IGIV products.

She tolerated the first Hyzentra infusion without a problem and the second one (given three weeks later) was completed but she then developed itching, tongue swelling and wheezing 4 hours after the completion of the infusion. She was treated in the emergency department but did not receive epinephrine.

Prior to the second infusion ACE inhibitor treatment was begun. She had similar reactions around that time to inactivated influenza vaccine and an antibiotic. The ACE inhibitor was discontinued.

A few weeks later, she was treated with Gamunex-C (15g subq). Again, there were no problems during the infusion but she developed itching, wheezing and tongue swelling several hours later.

Several weeks later she was given 1g of Gammagard subq and within minutes she developed generalized and tongue pruritis and wheezing.

Another Gammagard subq infusion was attempted. Her PFTs were normal before the infusion began.  Her FVC was actually 77%, but her FEV1 was 85% and FEF25-75 was 116%.  Her chest was clear and she did not get a bronchodilator treatment before the infusion began.  She received no pre-meds before the infusion was started. The plan was to give her 5 grams of Gammagard.  After 2 grams, she developed itching of the mouth and end expiratory wheeze.  The infusion was stopped and she felt a bit better after 60 minutes so the infusion was restarted. Wheezing worsened as did the itching.  At that time a serum tryptase was drawn which came back as 2.  When she was clearly wheezing, there was no change in her PFTs.

Suggestions for a way to treat her?

Thanks,
Richard Wasserman
Dallas
--
Richard L. Wasserman, MD, PhD
DallasAllergyImmunology
7777 Forest Lane, Suite B-332
Dallas, Texas 75230
Office (972) 566-7788
Fax (972) 566-8837
Cell (214) 697-7211

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