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AUTHOR'S PROOF
Metadata of the article that will be visualized in OnlineFirst
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Article Title
Bronchiolitis Obliterans Associated w ith Stev ens-Johnson Syndrome and Response to Azathioprine
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Article Sub- Title
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Article Copyright Year
Dr. K C Chaudhuri Foundation 2013 (This w ill be the copyright line in the final PDF)
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Journal Name
The Indian Journal of Pediatrics
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Family Name
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Given Name
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Singh Surj it
Suffix Corresponding Author
Organization
Post Graduate Institute of Medical Education and Research
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Division
Department of Pediatrics, Advanced Pediatrics Centre
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Address
Chandigarh 160012, India
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e-mail
[email protected]
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Family Name
Dogra
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Given Name
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Shiv ani
Organization
Post Graduate Institute of Medical Education and Research
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Division
Department of Pediatrics, Advanced Pediatrics Centre
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Address
Chandigarh 160012, India
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e-mail
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Author
Author
Saini Arushi G.
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Organization
Post Graduate Institute of Medical Education and Research
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Division
Department of Pediatrics, Advanced Pediatrics Centre
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Address
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e-mail
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Chandigarh 160012, India Suri Deepti
Organization
Post Graduate Institute of Medical Education and Research
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Division
Department of Pediatrics, Advanced Pediatrics Centre
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Address
Chandigarh 160012, India
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e-mail
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Family Name
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Author
Raw at Amit
Organization
Post Graduate Institute of Medical Education and Research
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Division
Department of Pediatrics, Advanced Pediatrics Centre
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Address
Chandigarh 160012, India
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e-mail
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Given Name
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Author
Sodhi K. S.
Organization
Post Graduate Institute of Medical Education and Research
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Department of Pediatrics, Advanced Pediatrics Centre
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Address
Chandigarh 160012, India
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e-mail
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Received
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Schedule
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Revised Accepted
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Abstract
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Keywords separated by ' - '
29 May 2013 31 July 2013
AUTHOR'S PROOF 58
Foot note information
AUTHOR'S PROOF
JrnlID 12098_ArtID 1204_Proof# 1 - 03/08/2013
Indian J Pediatr DOI 10.1007/s12098-013-1204-7
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SCIENTIFIC LETTER
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Shivani Dogra & Arushi G. Saini & Deepti Suri & Amit Rawat & K. S. Sodhi & Surjit Singh
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Received: 29 May 2013 / Accepted: 31 July 2013 # Dr. K C Chaudhuri Foundation 2013
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To the Editor: Stevens-Johnson syndrome (SJS) is an acute, self-limiting, immune-complex mediated hypersensitivity disorder of skin and mucus membranes. Though acute pulmonary complications are well described, persistent pulmonary sequelae associated with SJS are rare [1]. We describe severe progressive form of bronchiolitis obliterans (BO) following SJS and good response to azathioprine. A 9-y-old boy developed SJS following oral ibuprofen prescribed for febrile illness. A week later, he developed persistent cough, wheezing and progressive respiratory distress. At admission, he had tachypnea, retractions, extensive wheezing and crepitations with hyperpigmented scars, cicatrical ectropion in the left eye and dystrophic changes in nails. Oxygen-saturation was 87 % in room-air. Pulmonary function test showed severe obstruction with no response to bronchodilators. Chest-radiograph and high-resolution computerized tomography were consistent with BO (Figs. 1 and 2). He received supplemental oxygen, β2-agonist and budesonide inhalations, chest-physiotherapy and prednisolone. Azithromycin three times a week was also added. At 2-mo follow-up, he had persistent cough, exertional dyspnea and wheeze for which azathioprine was added as a steroid sparing agent. At 1-y follow-up, the patient improved clinically and was able to do his daily activities at home. Oxygenation and PFT improved. Although, he had dyspnea on moderate exertion, intermittent cough with wheeze, the symptoms had not worsened and the disease appeared to be controlled.
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Bronchiolitis Obliterans Associated with Stevens-Johnson Syndrome and Response to Azathioprine
N C O R R EC TE D
BO may occur within days after SJS even when the initial pulmonary involvement is not severe [1]. The association of non-steroidal anti-inflammatory drugs like ibuprofen with SJS is well known [2]. The findings of mosaic perfusion, vascular attenuation and central bronchiectasis on HRCT are diagnostic of BO obviating the need for lung-biopsy in a consistent clinical setting, as in index case [3]. The anti-inflammatory effects of corticosteroids justify their use in the early phases. We added immune-modulator azathioprine which suppresses lymphocyte proliferation and decreases CD8+ lymphocyte subpopulation, as a corticosteroid sparing agent for control of inflammation. Previous anaecdotal reports in adults have suggested that azathioprine might be useful as a corticosteroidsparing agent for the treatment of organizing pneumonia [4]. Moreover, drugs like mycophenolate mofetil and azathioprine have also been used for prevention of bronchiolitis obliterans
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S. Dogra : A. G. Saini : D. Suri : A. Rawat : K. S. Sodhi : S. Singh (*) Department of Pediatrics, Advanced Pediatrics Centre, Post Graduate Institute of Medical Education and Research, Chandigarh 160012, India e-mail:
[email protected] S. Singh e-mail:
[email protected]
Fig. 1 Chest radiograph showing marked bilateral hyperinflation with diffusely decreased attenuation and relatively decreased lung markings
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AUTHOR'S PROOF
JrnlID 12098_ArtID 1204_Proof# 1 - 03/08/2013
Indian J Pediatr
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1. Bakirtas A, Harmanci K, Toyran M, Razi CH, Turktas I. Bronchiolitis obliterans: a rare chronic pulmonary complication associated with Stevens-Johnson syndrome. Pediatr Dermatol. 2007;24:E22–5. 2. Neuman M, Nicar M. Apoptosis in ibuprofen-induced StevensJohnson syndrome. Transl Res. 2007;149:254–9. 3. Moonnumakal SP, Fan LL. Bronchiolitis obliterans in children. Curr Opin Pediatr. 2008;20:272–8. 4. Laszlo A, Espolio Y, Auckenthaler A, Michel JP, Janssens JP. Azathioprine and low-dose corticosteroids for the treatment of cryptogenic organizing pneumonia in an older patient. J Am Geriatr Soc. 2003;51: 433–4. 5. McNeil K, Glanville AR, Wahlers T, Knoop C, Speich R, Mamelok RD, et al. Comparison of mycophenolate mofetil and azathioprine for prevention of bronchiolitis obliterans syndrome in de novo lung transplant recipients. Transplantation. 2006;81:998–1003.
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References
N C O R R EC TE D
syndrome in de novo lung transplant recipients [5]. Since the course of post-SJS bronchiolitis obliterans is progressive and
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Fig. 2 Computerized tomography chest revealing bilateral hyperinflation with bronchiectasis (right side affected more than left side). Bronchial wall showed thickening. There was mosaic perfusion in right middle lobe
irreversible in contrast to post-infectious BO, immunemodulators like azathioprine can be tried in refractory cases.
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