Selective immunoglobulin M deficiency in an adult with miliary tuberculosis: A clinically interesting coexistence. A case report and review of the literature

Hassanein, Hassan A.; El-Badry, Mahmoud

doi:10.1016/j.ijmyco.2015.11.002

Cited by 15 publications

(8 citation statements)

References 8 publications

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“…The most common clinical manifestation of sIgMD is recurrent infections such as upper and lower respiratory tract infections (otitis media, chronic sinusitis, bronchitis, bronchiectasis and pneumonia), urinary tract infections and less frequently meningitis, sepsis, diarrhoea, cholangitis, hepatitis, chronic candidiasis, deep tissue and liver abscess 9–159 autoimmune diseases and malignancies.…”

Section: Introductionmentioning

confidence: 99%

Autoimmunity and immunodeficiency at the crossroad: autoimmune disorders as the presenting feature of selective IgM deficiency

et al. 2019

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Selective immunoglobulin M deficiency (sIgMD) is an immunodeficiency with undefined pathogenesis and commonly presenting with recurrent infections. 1 The European Society for Immunodeficiencies Registry defines sIgMD as a serum IgM level repeatedly below 2 SD of normal with normal levels of serum IgA, IgG and IgG subclasses, normal vaccination responses, absence of T-cell defects and absence of causative external factors. Rarely it can also be associated with autoimmune diseases. 2–7 Here we describe a patient with primary sIgMD; who presented with multiple autoimmune diseases without a history of recurrent infections and we provide a short literature review on sIgMD and autoimmune diseases.

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Section: Introductionmentioning

confidence: 99%

Autoimmunity and immunodeficiency at the crossroad: autoimmune disorders as the presenting feature of selective IgM deficiency

et al. 2019

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“…of normal with normal levels of serum IgA, IgG and IgG subclasses, normal vaccination responses, absence of T cell defects and absence of causative external factors (http://www.esid.org). Many previously published papers that report on ‘IgM deficiency’ do not fulfil these criteria .…”

Section: Introductionmentioning

confidence: 99%

Truly selective primary IgM deficiency is probably very rare

Janssen

Macken

Creemers

et al. 2017

Clinical and Experimental Immunology

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Isolated decreased serum-immunoglobulin (Ig)M has been associated with severe and/or recurrent infections, atopy and autoimmunity. However, the reported high prevalence of clinical problems in IgM-deficient patients may reflect the skewed tertiary centre population studied so far. Also, many papers on IgM deficiency have included patients with more abnormalities than simply IgM-deficiency. We studied truly selective primary IgM deficiency according to the diagnostic criteria of the European Society for Immunodeficiencies (ESID) (true sIgMdef) by reviewing the literature (261 patients with primary decreased serum-IgM in 46 papers) and analysing retrospectively all patients with decreased serum-IgM in a large teaching hospital in 's-Hertogenbosch, the Netherlands [1 July 2005-23 March 2016; n = 8049 IgM < 0·4 g/l; n = 2064 solitary (IgG+IgA normal/IgM < age-matched reference)]. A total of 359 of 2064 (17%) cases from our cohort had primary isolated decreased serum-IgM, proven persistent in 45 of 359 (13%) cases; their medical charts were reviewed. Our main finding is that true sIgMdef is probably very rare. Only six of 261 (2%) literature cases and three of 45 (7%) cases from our cohort fulfilled the ESID criteria completely; 63 of 261 (24%) literature cases also had other immunological abnormalities and fulfilled the criteria for unclassified antibody deficiencies (unPAD) instead. The diagnosis was often uncertain (possible sIgMdef): data on IgG subclasses and/or vaccination responses were lacking in 192 of 261 (74%) literature cases and 42 of 45 (93%) cases from our cohort. Our results also illustrate the clinical challenge of determining the relevance of a serum sample with decreased IgM; a larger cohort of true sIgMdef patients is needed to explore fully its clinical consequences. The ESID online Registry would be a useful tool for this.

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“…Since then, about 160 SIgMD cases have been reported, occasionally in association to other immune Metastatic Infection by Methicillin-Sensitive Staphylococcus aureus and its Potential Association to an Unusual Humoral Immunodeficiency diseases [3,8,9]. Prevalence is estimated around 0.03-0.37% in general population, 0.07-0.26% in Immunology-&-Allergy Clinics, 0.1-3.8% in hospitalized patients, and up to 6% within primary-immunodeficiency patients [2][3][4][5]10]. Apparently, there is no difference in SIgMD sex-distribution [2], however it tends to be more frequently symptomatic in men, finding a male-to-female ratio of even 11:2 [3,5,9,11,12].…”

Section: Discussionmentioning

confidence: 99%

Metastatic Infection by Methicillin-Sensitive Staphylococcus aureus and its Potential Association to an Unusual Humoral Immunodeficiency

Yubini¹,

Contreras²,

Cerdá³

et al. 2018

iid

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Multifocal metastatic infection by Staphylococcus aureus is severe and uncommon. We describe the case of a previously asymptomatic 63-year-old man diagnosed with complicated staphylococcal endocarditis of the mitral valve along with cervical and lumbar spondylodiscitis, and multiple pre/para-vertebral abscesses; establishing a persistent flaccid tetraparesis, neurogenic bladder and anal sphincter dysfunction. The patient overcame infection after an 8-week Cloxacillin/ Rifampicin treatment associated to multiple surgical procedures. Thereafter, immunological assessment was performed, driving to the diagnosis of selective IgM deficiency. We propose a physiopathological relationship between this rare dysgammaglobulinemia and such fulminant staphylococcal invasive infection; that would, perhaps, justify humoral immunity assessment in patients undergoing staphylococcemia.

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Selective immunoglobulin M deficiency in an adult with miliary tuberculosis: A clinically interesting coexistence. A case report and review of the literature

Cited by 15 publications

References 8 publications

Autoimmunity and immunodeficiency at the crossroad: autoimmune disorders as the presenting feature of selective IgM deficiency

Autoimmunity and immunodeficiency at the crossroad: autoimmune disorders as the presenting feature of selective IgM deficiency

Truly selective primary IgM deficiency is probably very rare

Metastatic Infection by Methicillin-Sensitive Staphylococcus aureus and its Potential Association to an Unusual Humoral Immunodeficiency

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