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Microsporidial myositis in adult-onset immunodeficiency: case-based review

  • T. G. Sundaram
  • Amita Aggarwal
  • Sujata Ganguly
  • Elgiva Khristie Iangngap
  • Rungmei S. K. Marak
  • Latika GuptaEmail author
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Abstract

Polymyositis is a diagnosis of exclusion. In patients with odd features, it can be of infective etiology. A high index of suspicion is required for diagnosis. A 55-year-old gentleman presented with gradual-onset proximal muscle weakness. Examination revealed mild distal weakness but no rash. Muscle enzymes were raised and tests for autoantibodies were negative. Biopsy revealed microsporidiosis. In view of this unusual infection, immunodeficiency was considered and he was found to have lymphopenia which antedated his illness. Later, he developed cranial nerve palsies due to multiple lesions in the pons. In addition, he had Cytomegalovirus viremia. Literature was reviewed to identify 20 cases of microsporidial myositis, its presentation, underlying immunodeficient state, and clinical course. Infective polymyositis should be considered in a patient with paucity of clinical and serological autoimmune features. Lymphopenia can point to underlying immunodeficiency. CMV infection could be the contributor to or bystander-effect of idiopathic lymphopenia.

Keywords

Myositis Microsporidiosis Immunodeficiency Lymphopenia 

Abbreviations

ILD

Interstitial lung disease

CMV

Cytomegalovirus

H. nana

Hymenolepis nana

MRC

Medical research council

MMT

Manual muscle testing

ANA

Anti-nuclear antibody

FDG PET-CT

Fluorodeoxy glucose positron emission tomography–computed tomography

PAS

Periodic acid–Schiff

GMS

Gomori methenamine silver

AFB

Acid fast bacillus

CSF

Cerebrospinal fluid

PCR

Polymerase chain reaction

HIV

Human immunodeficiency virus

AIDS

Acquired immunodeficiency syndrome

P. jiroveci

Pneumocystis jiroveci

LAP

Lymphadenopathy

AmpB

Amphotericin B

TNF

Tumour necrosis factor

Notes

Acknowledgements

We would like to thank the Departments of Microbiology at Post-Graduate Institute of Medical Education and Research (PGIMER), Chandigarh for helping with the diagnosis and management of this challenging case.

Author contributions

Substantial contributions to the management of the case and design of this case-based review were made by all the authors. EKI and RSKM contributed in identifying the pathogenic organism. Drafting of the work and revising it critically was done by TGS, AA, SG and LG. Final approval of the version to be published was done by LG. All the authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.

Funding

None.

Compliance with ethical standards

Conflict of interest

The authors declare that they have no conflict of interest.

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Copyright information

© Springer-Verlag GmbH Germany, part of Springer Nature 2019

Authors and Affiliations

  1. 1.Department of Clinical Immunology and RheumatologySanjay Gandhi Post Graduate Institute of Medical SciencesLucknowIndia
  2. 2.Department of PathologySanjay Gandhi Post Graduate Institute of Medical SciencesLucknowIndia
  3. 3.Department of MicrobiologySanjay Gandhi Post Graduate Institute of Medical SciencesLucknowIndia

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