Evidence map›Paper›PMID 28257650›Full record

ArticleJournal of medical case reports2017

Subcutaneous immunoglobulins for the treatment of a patient with antisynthetase syndrome and secondary chronic immunodeficiency after anti-CD20 treatment: a case report.

Patrick Cherin, Christophe de Jaeger, Jean-Charles Crave, Jean-Christophe Delain, Abir Tadmouri, Zahir Amoura

Open access · goldAbstract readCase Reports
In one paragraph

Article in Journal of medical case reports, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
0.7field-weighted citation impact, top 29% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

0 citing papers in PubMed, 5 citations in OpenAlex.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

6 authors at 4 institutions in 3 countries.

Patrick CherinDepartment of Internal Medicine, Pitié-Salpetrière Hospital Group, 47-83 Boulevard de l'Hôpital, 75013, Paris, France. patrick.cherin@aphp.fr.
Christophe de JaegerInstitut Galliera, 4 rue de Galliera, 75016, Paris, France.
Jean-Charles CraveOctapharma France SAS, Boulogne Billancourt, France.
Jean-Christophe DelainOctapharma France SAS, Boulogne Billancourt, France.
Abir TadmouriClinSearch, Malakoff, France.
Zahir AmouraDepartment of Internal Medicine, Pitié-Salpetrière Hospital Group, 47-83 Boulevard de l'Hôpital, 75013, Paris, France.
Octapharma (France) · FRSorbonne Université · FRMonal Group (France) · FRUniversity of St. Gallen · CH

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundAntisynthetase syndrome is a rare and debilitating multiorgan disease characterized by inflammatory myopathy, interstitial lung disease, cutaneous involvement, and frequent chronic inflammation of the joints. Standard treatments include corticosteroids and immunosuppressants. In some cases, treatment resistance may develop. Administration of immunoglobulins intravenously is recommended in patients with drug-resistant antisynthetase syndrome. CASE PRESENTATION: Here, we describe the case of a 56-year-old woman of Algerian origin. She is the first case of a patient with multidrug-resistant antisynthetase syndrome featuring pulmonary involvement and arthropathy, and chronic secondary immune deficiency with recurrent infections, after anti-CD20 treatment, in which her primary antisynthetase syndrome-related symptoms and secondary immune deficiency were treated successfully with subcutaneous administration of immunoglobulin. The administration of immunoglobulin subcutaneously was introduced at a dose of 2 g/kg per month and was well tolerated. Clinical improvement was observed within 3 months of initiation of subcutaneous administration of immunoglobulin. After 22 months of treatment, she showed a significant improvement in terms of muscle strength, pulmonary involvement, arthralgia, and immunodeficiency. Her serum creatine phosphokinase and C-reactive protein levels remained normal. Finally, she was compliant and entirely satisfied with the treatment.

conclusionsTaken together, these observations suggest that administration of immunoglobulin subcutaneously may be a useful therapeutic approach to tackle steroid-refractory antisynthetase syndrome while ensuring minimal side effects and improved treatment compliance. This treatment also allowed, in our case, for the regression of the chronic immunodeficiency secondary to rituximab treatment.

Indexed as

AgammaglobulinemiaFemaleHumansImmunoglobulinsImmunologic Deficiency SyndromesImmunosuppressive AgentsLung Diseases, InterstitialMagnetic Resonance ImagingMethotrexateMiddle AgedMyositisSubcutaneous AbsorptionImmunoglobulinsImmunosuppressive AgentsMethotrexateAnti-Jo-1 antibodyAntisynthetase syndromeAutoimmune diseaseCase reportMyositisSecondary immunodeficiencySubcutaneous human immunoglobulin

Identifiers

PMID28257650
PMCPMC5336681
OpenAlexW2589767394

What OpenQuestion holds

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LicenceCC BY
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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.