open access

Vol 82, No 5 (2014)
CASE REPORTS
Submitted: 2014-08-18
Accepted: 2014-08-18
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Granulomatosis with polyangiitis (Wegener’s granulomatosis) with hard palate and bronchial perforations treated with rituximab — a case report

Joanna Kosałka, Stanisława Bazan-Socha, Anna Zugaj, Maria Ignacak, Joanna Żuk, Barbara Sokołowska, Jacek Musiał
DOI: 10.5603/PiAP.2014.0059
·
Pneumonol Alergol Pol 2014;82(5):454-457.

open access

Vol 82, No 5 (2014)
CASE REPORTS
Submitted: 2014-08-18
Accepted: 2014-08-18

Abstract

We present a case of a 57-year-old woman suffering from granulomatosis with polyangiitis (GPA), who in the seventh months of immunosuppressive treatment (cyclophosphamide) progressed with new pulmonary changes and perforations of the hard palate and bronchi. Rituximab was introduced resulting in B-cell depletion and disappearance of anti-PR3 antibody. Palatal holes have substantially diminished and all bronchial perforations disappeared, covered by fibrous tissue. In the fourth month after rituximab administration, large scarring obstruction of the right main bronchus with upper and middle lobes atelectasis emerged. Because of increasing dyspnoea, stenotic bronchus was re-opened by bronchoscopy. Intervention was complicated by bilateral pneumothorax and later, on the seventh day, by fatal pulmonary bleeding. To our knowledge, this is the first report of GPA refractory to cyclophosphamide complicated by palatal and bronchial perforations.

Abstract

We present a case of a 57-year-old woman suffering from granulomatosis with polyangiitis (GPA), who in the seventh months of immunosuppressive treatment (cyclophosphamide) progressed with new pulmonary changes and perforations of the hard palate and bronchi. Rituximab was introduced resulting in B-cell depletion and disappearance of anti-PR3 antibody. Palatal holes have substantially diminished and all bronchial perforations disappeared, covered by fibrous tissue. In the fourth month after rituximab administration, large scarring obstruction of the right main bronchus with upper and middle lobes atelectasis emerged. Because of increasing dyspnoea, stenotic bronchus was re-opened by bronchoscopy. Intervention was complicated by bilateral pneumothorax and later, on the seventh day, by fatal pulmonary bleeding. To our knowledge, this is the first report of GPA refractory to cyclophosphamide complicated by palatal and bronchial perforations.

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Keywords

rituximab, granulomatosis with polyangiitis, vasculitis, Wegener’s granulomatosis, immunosuppression, palatal perforation, bronchial perforation

About this article
Title

Granulomatosis with polyangiitis (Wegener’s granulomatosis) with hard palate and bronchial perforations treated with rituximab — a case report

Journal

Advances in Respiratory Medicine

Issue

Vol 82, No 5 (2014)

Pages

454-457

DOI

10.5603/PiAP.2014.0059

Bibliographic record

Pneumonol Alergol Pol 2014;82(5):454-457.

Keywords

rituximab
granulomatosis with polyangiitis
vasculitis
Wegener’s granulomatosis
immunosuppression
palatal perforation
bronchial perforation

Authors

Joanna Kosałka
Stanisława Bazan-Socha
Anna Zugaj
Maria Ignacak
Joanna Żuk
Barbara Sokołowska
Jacek Musiał

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