[CIS PIDD] [cis-pidd] XLA with arthritis
CIS-PIDD
cis-pidd at lists.clinimmsoc.org
Wed Mar 29 14:54:27 EDT 2017
The mycoplasmas we described in XLA are very difficult to culture and require antibiotics that are not routinely used, eg, doxycycline. Our approach is when cultures appear sterile, think mycoplasma
Stuckey M, Quinn PA and Gelfand EW. Identification of ureaplasma urealyticum (T-stain mycoplasma) in a patient with polyarthritis. Lancet 2:917-920, 1978.
Roifman CM, Rao CP, Lederman HM, Lavi S, Quinn P and Gelfand EW. Increased susceptibility to mycoplasma infection in patients with hypogammaglobulinemia . Amer J Med 80:590-594, 1986.
Mohiuddin AA, Corren J, Harbeck RJ, Teague JL, Volz M and Gelfand EW. Ureaplasma urealyticum chronic osteomyelitis in a patient with hypogammaglobulinemia. J Allergy Clin Immunol 87:104-107, 1991.
Gelfand EW. Unique susceptibility to mycoplasma infection in patients with antibody deficiency. Clin Infect Dis 17:250-253, 1993
Erwin W. Gelfand, M.D.
Department of Pediatrics
National Jewish Health
1400 Jackson Street
Denver, CO 80206
Ph: 303-398-1196
Fax: 303-270-2105
E-mail: gelfande at njhealth.org<mailto:gelfande at njhealth.org>
From: CIS-PIDD <cis-pidd at lists.clinimmsoc.org<mailto:cis-pidd at lists.clinimmsoc.org>>
Reply-To: CIS-PIDD <cis-pidd at lyris.dundee.net<mailto:cis-pidd at lyris.dundee.net>>
Date: Wednesday, March 29, 2017 at 11:37 AM
To: CIS-PIDD <cis-pidd at lyris.dundee.net<mailto:cis-pidd at lyris.dundee.net>>
Subject: Re: [cis-pidd] XLA with arthritis
Thank you Robert - the comments are very helpful. He is also HLA B27(-). He has not responded to chronic NSAID therapy. I will certainly push harder to rule out infectious issues - we only did some routine cultures of synovial fluid in the past.
Dave Buchbinder
CHOC Children's
________________________________
From: CIS-PIDD <cis-pidd at lists.clinimmsoc.org<mailto:cis-pidd at lists.clinimmsoc.org>>
Sent: Tuesday, March 28, 2017 8:04 PM
To: CIS-PIDD
Subject: Re: [cis-pidd] XLA with arthritis
Several years ago Joe Church reported a case of HLA B27(-) enthesitis in a young man with XLA which responded beautifully to chronic NSAID therapy. I encountered a very similar case recently: recurrent hospital admissions for suspected septic arthritis involving the knees and ankles, cultures and Ureaplasma PCR uniformly negative. No recurrences since starting daily naproxen 6 months ago.
Hope that helps.
Robert
Robert W. Sugerman, M.D.
[Description: Description: Description: Description: cid:image001.jpg at 01CD31EB.7E39DF90]
of North Texas
7777 Forest Lane, Suite B-332
Dallas, TX 75230
Tel: 972 566-7788<tel:972%20566-7788> Fax: 972 566-8837<tel:972%20566-8837>
www.dallasallergy.net<http://www.dallasallergy.net/>
On Tue, Mar 28, 2017 at 4:46 PM, CIS-PIDD <cis-pidd at lists.clinimmsoc.org<mailto:cis-pidd at lists.clinimmsoc.org>> wrote:
Dear Colleagues,
I have a now 5 year old young man with XLA. He originally had a history of recurrent OM and C.diff as well as occasional neutropenia. He was referred for right knee swelling suggestive of JIA. Based on his history additional studies documented data diagnostic of XLA. No B cells. Pan-hypogammaglobulinemia. BTK expression studies and genetic analyses were done as well. A hemizygous variant in BTK was documented. I placed him on Hizentra and he has done wonderfully. His arthritis also seemed to get better - he also had some corticosteroid injections. More recently, he has developed a more significant flare of his JIA - more joints (wrists, knee, ankle, etc.). We were going to use a anti-TNF agent (e.g. Humira or Enbrel). I was wondering if anyone has any words of wisdom / caution in this setting given the risk of enterovirus issues, neutropenia.... any other suggested agents that would be excellent.
Thanks as always!
Dave
David Buchbinder, MD
CHOC Children's Hospital
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