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Rituximab in refractory sarcoidosis: a single centre experience.

Cinetto F, Compagno N, Scarpa R, Malipiero G, Agostini C - Clin Mol Allergy (2015)

Bottom Line: However, refractory disease is still a great challenge.Rituximab is an anti-CD20 chimeric monoclonal antibody, currently used for the treatment of B cell malignancies and systemic autoimmune diseases.There are few case reports describing the successful use of Rituximab in refractory sarcoidosis with lung, eye, lymph nodes and skin involvement.

View Article: PubMed Central - PubMed

Affiliation: Clinical Immunology, Department of Medicine-DIMED, Padova University Hospital, via Giustiniani 2, 35128 Padua, Italy.

ABSTRACT
Sarcoidosis is a granulomatous disease whose outcome varies from spontaneous remission to chronic refractory disease. Provided that steroids represent the gold standard as a first line treatment, many immune suppressants drugs are currently used in the disease management. However, refractory disease is still a great challenge. Rituximab is an anti-CD20 chimeric monoclonal antibody, currently used for the treatment of B cell malignancies and systemic autoimmune diseases. There are few case reports describing the successful use of Rituximab in refractory sarcoidosis with lung, eye, lymph nodes and skin involvement. In this paper we described three different case reports in which Rituximab has been used to treat refractory sarcoidosis and we reviewed the existing literature.

No MeSH data available.


Related in: MedlinePlus

Lung volumes and DLco in patient 1.
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Fig1: Lung volumes and DLco in patient 1.

Mentions: Eight months later patient complained of a new worsening of dyspnoea and a CT scan showed an interstitial pulmonary involvement, prevalent in the upper lobes, with recurrence of lymphadenopathy. Azathioprine was started and suspended in few weeks, due to intolerance. Cyclophosphamide was then administered every 2 weeks in our outpatient clinic for 6 times; the initial dose was 600 mg, later escalated up to 750 mg. A chest CT scan performed at the end of the treatment showed no progression of the disease, but a new assessment after 6 months revealed new radiological signs of active interstitial lung disease, extending from upper to lower lobes, with increasing evidence of parenchymal and peri-bronchial fibrosis. A worsening was registered in terms of dyspnoea and lung function (Fig. 1). Patient refused infliximab as a treatment option, due to the long term administration schedule. Thus, rituximab 375 mg/m2 every 2 weeks for six times was administered. Treatment was well tolerated and three further evaluations, respectively 6, 12 and 18 months after the end of the treatment, highlighted an arrest in lung disease progression. At present patient only has radiological evidence of inactive fibrotic lesions, prevalently located at upper lobes, and a stable restrictive disease (Fig. 1); he is under maintenance treatment with hydroxychloroquine.Fig. 1


Rituximab in refractory sarcoidosis: a single centre experience.

Cinetto F, Compagno N, Scarpa R, Malipiero G, Agostini C - Clin Mol Allergy (2015)

Lung volumes and DLco in patient 1.
© Copyright Policy - OpenAccess
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4556310&req=5

Fig1: Lung volumes and DLco in patient 1.
Mentions: Eight months later patient complained of a new worsening of dyspnoea and a CT scan showed an interstitial pulmonary involvement, prevalent in the upper lobes, with recurrence of lymphadenopathy. Azathioprine was started and suspended in few weeks, due to intolerance. Cyclophosphamide was then administered every 2 weeks in our outpatient clinic for 6 times; the initial dose was 600 mg, later escalated up to 750 mg. A chest CT scan performed at the end of the treatment showed no progression of the disease, but a new assessment after 6 months revealed new radiological signs of active interstitial lung disease, extending from upper to lower lobes, with increasing evidence of parenchymal and peri-bronchial fibrosis. A worsening was registered in terms of dyspnoea and lung function (Fig. 1). Patient refused infliximab as a treatment option, due to the long term administration schedule. Thus, rituximab 375 mg/m2 every 2 weeks for six times was administered. Treatment was well tolerated and three further evaluations, respectively 6, 12 and 18 months after the end of the treatment, highlighted an arrest in lung disease progression. At present patient only has radiological evidence of inactive fibrotic lesions, prevalently located at upper lobes, and a stable restrictive disease (Fig. 1); he is under maintenance treatment with hydroxychloroquine.Fig. 1

Bottom Line: However, refractory disease is still a great challenge.Rituximab is an anti-CD20 chimeric monoclonal antibody, currently used for the treatment of B cell malignancies and systemic autoimmune diseases.There are few case reports describing the successful use of Rituximab in refractory sarcoidosis with lung, eye, lymph nodes and skin involvement.

View Article: PubMed Central - PubMed

Affiliation: Clinical Immunology, Department of Medicine-DIMED, Padova University Hospital, via Giustiniani 2, 35128 Padua, Italy.

ABSTRACT
Sarcoidosis is a granulomatous disease whose outcome varies from spontaneous remission to chronic refractory disease. Provided that steroids represent the gold standard as a first line treatment, many immune suppressants drugs are currently used in the disease management. However, refractory disease is still a great challenge. Rituximab is an anti-CD20 chimeric monoclonal antibody, currently used for the treatment of B cell malignancies and systemic autoimmune diseases. There are few case reports describing the successful use of Rituximab in refractory sarcoidosis with lung, eye, lymph nodes and skin involvement. In this paper we described three different case reports in which Rituximab has been used to treat refractory sarcoidosis and we reviewed the existing literature.

No MeSH data available.


Related in: MedlinePlus