Anti-Jo-1 Antibody-positive Interstitial Pneumonia in an Elderly Patient with Congenital Rubella Syndrome

A 71-year-old woman with congenital rubella syndrome (CRS) presented with prolonged cough. No physical findings suggested the presence of any connective tissue diseases. Chest computed tomography showed ground-glass opacities and consolidations in the bilateral lower lobes. She had elevated serum Krebs von den Lungen-6, hypoxemia and positive serum anti-Jo-1 antibody. Bronchoalveolar lavage fluid revealed lymphocytosis with a decreased CD4/CD8 ratio. A transbronchial lung biopsy specimen revealed organizing pneumonia. Based on a diagnosis of interstitial pneumonia with autoimmune features (IPAF), systemic corticosteroids were administered, and a good outcome was obtained. A possible relationship between CRS and IPAF is herein discussed.

[1]  P. Lang,et al.  Mechanisms of lymphatic system‐specific viral replication and its potential role in autoimmune disease , 2019, Clinical and experimental immunology.

[2]  M. Arcangeletti,et al.  A Paradigmatic Interplay between Human Cytomegalovirus and Host Immune System: Possible Involvement of Viral Antigen-Driven CD8+ T Cell Responses in Systemic Sclerosis , 2018, Viruses.

[3]  D. Hashimoto,et al.  Anti-PL-7 Antisynthetase Syndrome with Eosinophilic Pleural Effusion , 2018, Internal medicine.

[4]  Satoshi Suzuki,et al.  Congenital rubella syndrome with death from interstitial pneumonia , 2016, Pediatrics international : official journal of the Japan Pediatric Society.

[5]  A. Kai,et al.  Shedding of Rubella Virus among Infants with Congenital Rubella Syndrome Born in Tokyo, Japan, 2013-2014. , 2016, Japanese journal of infectious diseases.

[6]  D. Lynch,et al.  An official European Respiratory Society/American Thoracic Society research statement: interstitial pneumonia with autoimmune features , 2015, European Respiratory Journal.

[7]  I. Noth,et al.  “ERS/ATS Task Force onUndifferentiated Forms of CTD-ILD”. An official European RespiratorySociety/American Thoracic Society research statement: interstitial pneumonia withautoimmunefeatures , 2015 .

[8]  S. Katow,et al.  Epidemiological characteristics of rubella and congenital rubella syndrome in the 2012-2013 epidemics in Tokyo, Japan. , 2015, Japanese journal of infectious diseases.

[9]  M. Hashizume,et al.  Mortality Associated With Pulmonary Hypertension in Congenital Rubella Syndrome , 2014, Pediatrics.

[10]  S. Ueno,et al.  Characterization of dermatomyositis with coexistence of anti-Jo-1 and anti-SRP antibodies. , 2012, Internal medicine.

[11]  A. B. Chang,et al.  Congenital rubella pneumonitis complicated by Pneumocystis jiroveci infection with positive long term respiratory outcome: A case report and literature review , 2009, Pediatric pulmonology.

[12]  T. Español,et al.  Congenital rubella syndrome, hyper‐IgM syndrome and autoimmunity in an 18‐year‐old girl , 2007, Journal of paediatrics and child health.

[13]  N. Mogulkoc,et al.  An unusual presentation of anti-Jo-1 syndrome, mimicking lung metastases, with massive pleural and pericardial effusions. , 2006, Journal of clinical rheumatology : practical reports on rheumatic & musculoskeletal diseases.

[14]  C. Briani,et al.  Polymyositis–dermatomyositis and infections , 2006, Autoimmunity.

[15]  S. Franklin,et al.  Congenital Rubella and Interstitial Pneumonitis , 2001, Clinical pediatrics.

[16]  R. Kasukawa,et al.  Two patients with polymyositis or dermatomyositis complicated with massive pleural effusion. , 1998, Internal medicine.

[17]  I. Caragol,et al.  Circumscribed scleroderma in congenital rubella syndrome with hypogammaglobulinemia , 1998, Allergy.

[18]  P Rubinstein,et al.  Diabetes mellitus and autoimmunity in patients with the congenital rubella syndrome. , 1985, Reviews of infectious diseases.

[19]  W. Clarke,et al.  Autoimmunity in congenital rubella syndrome. , 1984, The Journal of pediatrics.

[20]  D. Jeffries,et al.  Desquamative interstitial pneumonia and antigen-antibody complexes in two infants with congenital rubella. , 1983, Pediatrics.