[CIS PIDD] [cis-pidd] cis-pidd digest: September 22, 2014

Tue Sep 23 15:49:52 EDT 2014

Re: Chronic Norovirus Infection

I follow a 12 y old with hematologic malignancy who came into 2nd
transplant with norovirus exposure and lived through it (and beyond) with
GVHD diarrhea and adenoviral illness, now all resolved. He was excreting
norovirus genotype II 32 months from initial detection when we stopped
checking.

-Rakesh Goyal
Children's Hospital of Pittsburgh

On Tue, Sep 23, 2014 at 12:01 AM, CIS-PIDD digest <
cis-pidd at lists.clinimmsoc.org> wrote:

> CIS-PIDD Digest for Monday, September 22, 2014.
>
> 1. Re: Histiocytosis and persistant disseminated atypical mycobacteria
> 2. Chronic Norovirus Infection
> 3. Re: Chronic Norovirus Infection
> 4. RE: Chronic Norovirus Infection
> 5. RE: Chronic Norovirus Infection
>
> ----------------------------------------------------------------------
>
> Subject: Re: Histiocytosis and persistant disseminated atypical
> mycobacteria
> From: Laia Alsina Manrique de Lara <lalsina at hsjdbcn.org>
> Date: Mon, 22 Sep 2014 08:15:46 +0200
> X-Message-Number: 1
>
> Hola Nacho,
> Coincido con lo sugerido.
> Quería comentarte que tengo un FIS destinado a estudiar pacientes con
> sospecha de MSMD. Así que si te interesa podría realizar una evaluación
> completa de la vía en este paciente tuyo. Necesitaría 15ml de sangre
> fresca, que podrías enviar lunes o martes de la semana que viene. Si te
> interesa, te doy las especificaciones concretas de tubos, CI y dirección
> envió.
> Un abrazo,
>
> Laia Alsina
>
> El 20/09/2014, a las 11:07, "Nacho Gonzalez" <nachgonzalez at gmail.com
> <mailto:nachgonzalez at gmail.com>> escribió:
>
> Thanks everyone for your help. I´ll keep you updated.
> Best regards,
>
> Luis Ignacio Gonzalez-Granado
> Immunodeficiencies Division
> Hematology & Oncology Division
> Pediatrics. Hospital 12 octubre. Madrid. Spain
>
> 2014-09-19 14:56 GMT+02:00 Dewton Vasconcelos <dmvascon at usp.br<mailto:
> dmvascon at usp.br>>:
> Dear Nacho, good morning
>
> I agree with the previous comments and I would add that we have seen a
> patient presenting IL12RB1 deficiency with a similar evolution and
> histopathological confusion with malignant histiocytosis. This patient was
> initially treated as LCH leading to dissemination of a BCG infection. After
> intensive anti-mycobacterial therapy he improved a lot but the parents
> interrupted the therapy after one year of rifampin, ethambutol and
> isoniazid and the infection relapsed. He was again biopsied and the
> pathology of his town misdiagnosed Hodgkin lymphoma this time. We asked for
> the piece and it was really ganglionar mycobacteriosis, starting another
> anti-mycobacterial therapy with rifampin, isoniazid, ethambutol and
> clarithromycin.
>
> In the lesions of these patients there are sometimes extensive sinusal
> histiocytosis in the periphery of the granulomatous lesions that
> occasionally are confounded at the first sight.
> Moreover, lymphnodes are difficult to evaluate and we have also seen
> confusion between "sarcoidosis" and Hodgkin lymphoma (the correct diagnosis
> in the case).
>
> I would try to do a STAT4 phosphorylation assay stimulated by IL-12 and
> IFN-alpha and the expression of CD212 in T cell blasts (PHA+IL2 stimulated)
> to exclude CD212 deficiency.
> Moreover, patients presenting with severe mycobacteriosis (and also severe
> deep mycoses) present lymphopenia, mainly of CD4 but also CD8, B and NK
> (see Antas PR et al, J Allergy Clin Immunol. 2006 Apr;117(4):916-23.)
>
> All the best,
>
> Dewton
>
> Dewton de Moraes Vasconcelos, MD, PhD
> Primary Immunodeficiencies Outpatient Unit ADEE3003
> Lab. of Medical Investigation Unit 56
> University of São Paulo School of Medicine
>
> Nacho Gonzalez wrote:
> Dear colleagues,
>
> I would like to hear any input regarding diagnosis and treatment for a 3
> 1/2  yo male with LCH and disseminated M. genavense infection.
>
> At 6 mo of age weight < P3 & height P10. Multisystemic Langerhans
> histiocytosis (LCH)  treated with vinblastin, steroids and clofarabin (this
> drug from Nov 2012 to 22 june 2013, 8 courses) finished in April 2013.
> Maintenance chemo (MTX+MCP & steroids) stopped 8 months ago. LCH is
> considered as non-active disease after several reevaluations.
>
> Disseminated atypical mycobacteria was diagnosed in Jan 2014. After 1
> month under iv etambutol+levo+azithro treatment and 1 1/2  months po, fever
> relapsed and bone marrow was again full of atypical mycobacteria. Then we
> used iv linezolid, amikacin, ethambutol, azithromycin and levo. In June PCR
> revealed NTM was M. genavense. Since then, he is under iv levo +
> clarithromycin + ethambutol + rifampicin.
> For the last 2 1/2 months fever subsided and night sweating dissapeared.
> However in the last 10 days he has daily fever and night sweating.
> Extensive infectious disease work up is negative.
> He is TPN dependent because of chronic diarrhea, malnutrition and protein
> losing enteropathy (PLE has resolved lately). Two gut endoscopies revealed
> NTM within macrophages covering the lamina propria. Microbiologists cannot
> ensure they are not viable. All cultures (BM, gut) have been negative for
> NTM. Abdominal MRI (March 2013) suggested sclerosing mesenteritis. A trial
> of colchicine turned to be ineffective. Follow up ultrasounds show
> improvement of this.
> In the past he had prolonged viral shedding with viral infections (but not
> clinically severe) after Paraflu, Flu, noro and rota infections in the last
> two years w or w/o chemo.
> The patient had myelodisplasia in the last two BM samples. He has no
> cytopenias now.
>
> IFNg/IL12 axis assessment is normal, with normal production of TNFa and
> IFNg after PBMC stimulation).
> Lymph subsets: ALC 1300 - 45 00/uL CD3 (216 - 432 CD4, 1000 - 3000 CD8+.
> Almost all of them have memory phenotype. Decreased thymic output, CD31+
> 10% ),  40 - 176 /uL CD19 with normal IgG levels. Decreased T cell
> proliferation with PHA, PMA/iono, antiCD3/CD28. (see follow-up
> immunological tests attached)
> [cid:]
> Genes sequenced: IFNgR1, RAG1/2,GATA2. All wt
>
> Questions:
> Is this PID primary or secondary ? Is there any way to know it?
> How to balance benefit/risk ratio of sc IFNg, as LCH eventually may recurr
> ? Anyone has experience on this?
> HSCT? When?
>
> Any input will be welcomed.
> Best regards,
>
> Luis Ignacio Gonzalez-Granado
> Immunodeficiencies Division
> Hematology & Oncology Division
> Pediatrics. Hospital 12 octubre. Madrid. Spain
>
> ---
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> ----------------------------------------------------------------------
>
> Subject: Chronic Norovirus Infection
> From: "Church, Joseph" <JChurch at chla.usc.edu>
> Date: Mon, 22 Sep 2014 23:36:43 +0000
> X-Message-Number: 2
>
> Colleagues:
>
> I am trying to get some idea of the extent of chronic norovirus infection
> in patients with primary or acquired immunodeficiency.
>
> I would very much appreciate your letting me know how many such patients
> you follow and their underlying immune deficiencies.
>
> Thank you for your help.
>
> Joe Church
> Children's Hospital Los Angeles
>
>
>
> ---------------------------------------------------------------------
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>
> ----------------------------------------------------------------------
>
> Subject: Re: Chronic Norovirus Infection
> From: "Patel, Niraj C" <Niraj.Patel at carolinashealthcare.org>
> Date: Mon, 22 Sep 2014 23:59:59 +0000
> X-Message-Number: 3
>
> I have followed 2 pediatric patients, both status post BMT for hematogic
> malignancy. One had chronic norovirus, and another had chronic infection
> with sapovirus, a close relative of norovirus.
>
> Niraj Patel, MD, MS
> Carolinas Medical Center
> Charlotte, NC
>
>
> On 22 September 2014 19:37 PM, "Church, Joseph" <JChurch at chla.usc.edu>
> wrote:
> Colleagues:
>
> I am trying to get some idea of the extent of chronic norovirus infection
> in patients with primary or acquired immunodeficiency.
>
> I would very much appreciate your letting me know how many such patients
> you follow and their underlying immune deficiencies.
>
> Thank you for your help.
>
> Joe Church
> Children’s Hospital Los Angeles
>
>
> ---------------------------------------------------------------------
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> ----------------------------------------------------------------------
>
> Subject: RE: Chronic Norovirus Infection
> From: Maite de la Morena <Maite.delaMorena at UTSouthwestern.edu>
> Date: Tue, 23 Sep 2014 00:56:43 +0000
> X-Message-Number: 4
>
> Joe
> Have a girl with hypogammaglobulinemia and unknown molecular defect that I
> follow.
> Family hx significant for sister with hypogammaglobulinemia who died at
> age 3 of respiratory complications
> No consanguinity, healthy brother
> Maite
>
>
> Maite de la Morena, MD
> Professor of Pediatrics  and Internal Medicine
> Division of Allergy and Immunology
> University of Texas Southwestern Medical Center in Dallas
> 5323 Harry HInes Blvd
> Dallas, Texas 75390-9063
> Phone 214 456-5161
> Fax: 214 456-8317
> Email: maite.delamorena at utsouthwestern.edu
> From: Church, Joseph [mailto:JChurch at chla.usc.edu]
> Sent: Monday, September 22, 2014 6:37 PM
> To: CIS-PIDD
> Subject: [cis-pidd] Chronic Norovirus Infection
>
> Colleagues:
>
> I am trying to get some idea of the extent of chronic norovirus infection
> in patients with primary or acquired immunodeficiency.
>
> I would very much appreciate your letting me know how many such patients
> you follow and their underlying immune deficiencies.
>
> Thank you for your help.
>
> Joe Church
> Children's Hospital Los Angeles
>
>
> ---------------------------------------------------------------------
> CONFIDENTIALITY NOTICE: This e-mail message, including any attachments,
> is for the sole use of the intended recipient(s) and may contain
> confidential
> or legally privileged information. Any unauthorized review, use, disclosure
> or distribution is prohibited. If you are not the intended recipient,
> please
> contact the sender by reply e-mail and destroy all copies of this original
> message.
>
> ---------------------------------------------------------------------
>
> ---
>
> The CIS-PIDD listserv is supported by:
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>
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> ________________________________
>
> UT Southwestern Medical Center
> The future of medicine, today.
>
> ----------------------------------------------------------------------
>
> Subject: RE: Chronic Norovirus Infection
> From: "Sriaroon, Panida" <psriaroo at health.usf.edu>
> Date: Tue, 23 Sep 2014 03:08:43 +0000
> X-Message-Number: 5
>
> Hi Joe,
>
> We have a teenage boy with CVID and lymphopenia. I posted his case on the
> listserv few months ago and there were several responses from colleagues in
> US and Europe. Maybe you could find more info there, too.
>
> Panida
> University of South Florida
> ________________________________________
> From: Church, Joseph [JChurch at chla.usc.edu]
> Sent: Monday, September 22, 2014 7:36 PM
> To: CIS-PIDD
> Subject: [cis-pidd] Chronic Norovirus Infection
>
> Colleagues:
>
> I am trying to get some idea of the extent of chronic norovirus infection
> in patients with primary or acquired immunodeficiency.
>
> I would very much appreciate your letting me know how many such patients
> you follow and their underlying immune deficiencies.
>
> Thank you for your help.
>
> Joe Church
> Children’s Hospital Los Angeles
>
>
> ---------------------------------------------------------------------
> CONFIDENTIALITY NOTICE: This e-mail message, including any attachments,
> is for the sole use of the intended recipient(s) and may contain
> confidential
> or legally privileged information. Any unauthorized review, use, disclosure
> or distribution is prohibited. If you are not the intended recipient,
> please
> contact the sender by reply e-mail and destroy all copies of this original
> message.
>
> ---------------------------------------------------------------------
>
> ---
>
> The CIS-PIDD listserv is supported by:
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>
>
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> END OF DIGEST
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