[CIS PIDD] [cis-pidd] Multifocal osteomyelitis in an eleven yo boy with CGD

[Boyce, Thomas G., M.D.]

I agree with James.  I usually use a scheduled NSAID and if patients have breakthrough symptoms while on NSAIDS, I will give them prednisone (2 mg/kg on day one, 1 mg/kg on day two, and 0.5 mg/kg on day three) and this will often help the symptoms.

From: Verbsky, James [mailto:jverbsky at mcw.edu]
Sent: Friday, February 21, 2014 3:22 PM
To: CIS-PIDD
Subject: RE: Re: [cis-pidd] Multifocal osteomyelitis in an eleven yo boy with CGD

Pere

Sorry for the late response…I agree with the thoughts about treating CRMO…although in this case a short course of prednisone might be helpful.  Steroids are often given to CGD while treating infections anyway…so might be worth a trial to see how responsive this is to immunosuppression.

Best

James


James W. Verbsky M.D./Ph.D.
Associate Professor of Pediatrics and Microbiology
Medical College of Wisconsin
Milwaukee, WI
414-266-6701



From: stephan.ehl at uniklinik-freiburg.de<mailto:stephan.ehl at uniklinik-freiburg.de> [mailto:stephan.ehl at uniklinik-freiburg.de]
Sent: Friday, February 21, 2014 12:54 PM
To: CIS-PIDD
Subject: AW: Re: [cis-pidd] Multifocal osteomyelitis in an eleven yo boy with CGD


There is also a recent PNAS report on anti-IL1, SE

________________________________

  Von: Arturo Borzutzky [drarturo at gmail.com]
  Gesendet: 21.02.2014 13:42 ZW3
  An: "CIS-PIDD" <cis-pidd at lists.clinimmsoc.org<mailto:cis-pidd at lists.clinimmsoc.org>>
  Betreff: Re: [cis-pidd] Multifocal osteomyelitis in an eleven yo boy with CGD

Dear Pere,

If NSAIDs fail, a reasonable idea -if still no infectious cause is found- would be to try thalidomide that has been recently reported in JACI to successfully treat inflammatory manifestations of CGD patients (no CRMO though) (http://www.jacionline.org/article/S0091-6749(13)00763-X/fulltext).
Pamidronate IV infusions have also been successfully used to treat CRMO. Immunosuppressive alternatives (methotrexate, TNF inhibitors, etc) seem out of the question given the underlying PID.

Good luck

Arturo


Dr. Arturo Borzutzky S.
Inmunología, Alergia y Reumatología Pediátrica
División de Pediatría
Pontificia Universidad Católica de Chile
Tel: (56-2) 23543753
www.saluduc.cl<http://www.saluduc.cl/>

On Thu, Feb 20, 2014 at 6:51 PM, Pere Soler Palacin <psoler at vhebron.net<mailto:psoler at vhebron.net>> wrote:
Dear Thomas, thnx for your comments. The patient doesn't have any GI symptoms. However, he had elevated fecal calprotectin months ago and we've tested it again, no results yet, to look for subclinical enteritis.
We've already started NSAIDs,

Best regards,


P.
Pere Soler Palacín, MD, PhD.
Pediatric Infectious Diseases and Immunodeficiencies Unit. Hospital Universitari Vall d'Hebron.
Assistant Professor. Universitat Autònoma de Barcelona.
Passeig de la Vall d'Hebron 119-129.
08035 Barcelona. Spain.
Tel: 0034934893140<tel:0034934893140>. Fax: 0034934893039<tel:0034934893039>.
E-mail: psoler at vhebron.net<mailto:psoler at vhebron.net>; 34660psp at comb.cat<mailto:34660psp at comb.cat>. Web: www.upiip.com<http://www.upiip.com/>.



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----- Mensaje original -----
De: "Thomas G. Boyce, M.D." <Boyce.Thomas at mayo.edu<mailto:Boyce.Thomas at mayo.edu>>
Para: "CIS-PIDD" <cis-pidd at lists.clinimmsoc.org<mailto:cis-pidd at lists.clinimmsoc.org>>
Enviados: Jueves, 20 de Febrero 2014 17:35:47
Asunto: RE: [cis-pidd] Multifocal osteomyelitis in an eleven yo boy with CGD

Does he have any GI symptoms?  CRMO is more common in patients with Crohn’s disease.  I usually treat CRMO with scheduled indomethacin or naprosyn (while continuing to be vigilant for possible infectious causes, especially in a patient with CGD).  Clinical response to NSAIDS is presumptive evidence for CRMO (although it waxes and wanes on its own as well).


Thomas G. Boyce, MD, MPH
Pediatric Infectious Diseases and Immunology
Mayo Clinic
email: boyce.thomas at mayo.edu<mailto:boyce.thomas at mayo.edu>
phone: 507-255-8464
fax: 507-255-7767


From: Pere Soler Palacin [mailto:psoler at vhebron.net<mailto:psoler at vhebron.net>]
Sent: Thursday, February 20, 2014 10:28 AM
To: CIS-PIDD
Cc: Mai Figueras
Subject: [cis-pidd] Multifocal osteomyelitis in an eleven yo boy with CGD


Dear all we have a 11 yo male with XL-CGD with multifocal ostemyelitis unresponsive to antibiotic therapy.

The patient presented with mild pain of his right pelvic area almost two months ago (he was doing previously well). At that point MRI, Tc-scintigraphy and bone biopsy were performed: MRI was compatible with acute osteomyelitis, scintigraphy showed two focus (right pelvis and right knee) and bone biopsy showed PMN infiltration but cultures (B, MB, fungus) and both panfungal and 16sRNA PCRs were negative. IV meropenem was instituted at that point and the patient clinically improved with acute phase reactants lowering too.

However, 3 weeks later APR increased again and the patient presented with knee pain. Once again, a MRI was performed and showed a new focus on the contralateral acetabulum. Bone biopsy was performed with negative results again. Tc- scintigraphy was compatible with multifocal osteomielytis affecting the jaw and several metaphyseal bones of both the upper and lower limbs despite the patient did not described any simptoms at that level. Scintigraphy with marked leucocytes revealed signs of inflammation at the initial focus but showed cold lesions in all the other affected bones. Ecocardiogram ruled IE out.

Serological studies to Bartonella spp, Coxiella burnetti, Leishmania spp and Brucella spp were negative. Long-term cultures are still on going but no microorganism has grew yet.



Do you have any experience with aseptic meningitis in CGD patients? CRMO?  Any other suggestion?



Thanks in advance.



Pere.



Pere Soler Palacín, MD, PhD.
Pediatric Infectious Diseases and Immunodeficiencies Unit. Hospital Universitari Vall d'Hebron.
Assistant Professor. Universitat Autònoma de Barcelona.
Passeig de la Vall d'Hebron 119-129.
08035 Barcelona. Spain.
Tel: 0034934893140<tel:0034934893140>. Fax: 0034934893039<tel:0034934893039>.
E-mail: psoler at vhebron.net<mailto:psoler at vhebron.net>; 34660psp at comb.cat<mailto:34660psp at comb.cat>. Web: www.upiip.com<http://www.upiip.com/>.



No imprimir aquest correu ajudarà a preservar el medi ambient.
Si vostè no és el destinatari del missatge, o l'ha rebut per error, si us plau notifiqui-ho al remitent i destrueixi el missatge amb tot el seu contingut. Està prohibida la distribució no autoritzada del contingut d'aquest missatge.

No imprimir este correo ayudará a preservar el medio ambiente.
Si usted no es el destinatario del mensaje, o lo ha recibido por error, notifíquelo por favor al remitente y destruya el mensaje con todo su contenido. Está prohibida la distribución no autorizada del contenido de este mensaje.


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