PURPOSE: Paraneoplastic autoimmune multiorgan syndrome (PAMS) is a rare disorder that manifests with severe mucocutaneous blistering and erosions accompanied by underlying malignancy. Constrictive bronchiolitis (CB) can occur in a subset of patients. We sought to characterize the particular group of PAMS patients with CB by comparing the underlying tumor in these patients versus all reported PAMS patients. METHODS: We performed a systematic review of the Medline/Pubmed and compared the incidence of the most frequent tumor type in the CB cohort with the previously reported incidences of tumors in all documented PAMS patients. RESULTS: There is an increased occurrence of Castleman's tumor in patients with mucocutaneous and pulmonary complications of PAMS OR 3.86, 95 % CI [2.2-6.7]. CONCLUSIONS: As PAMS is a syndrome with a heterogeneous presentation and autoantibody profile, the particular pathogenic pathway seen in Castleman's tumor may provide additional insight into the antigenic sites involved in PAMS-related CB.
PURPOSE:Paraneoplastic autoimmune multiorgan syndrome (PAMS) is a rare disorder that manifests with severe mucocutaneous blistering and erosions accompanied by underlying malignancy. Constrictive bronchiolitis (CB) can occur in a subset of patients. We sought to characterize the particular group of PAMS patients with CB by comparing the underlying tumor in these patients versus all reported PAMS patients. METHODS: We performed a systematic review of the Medline/Pubmed and compared the incidence of the most frequent tumor type in the CB cohort with the previously reported incidences of tumors in all documented PAMS patients. RESULTS: There is an increased occurrence of Castleman's tumor in patients with mucocutaneous and pulmonary complications of PAMS OR 3.86, 95 % CI [2.2-6.7]. CONCLUSIONS: As PAMS is a syndrome with a heterogeneous presentation and autoantibody profile, the particular pathogenic pathway seen in Castleman's tumor may provide additional insight into the antigenic sites involved in PAMS-related CB.
Authors: G J Anhalt; S C Kim; J R Stanley; N J Korman; D A Jabs; M Kory; H Izumi; H Ratrie; D Mutasim; L Ariss-Abdo Journal: N Engl J Med Date: 1990-12-20 Impact factor: 91.245
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Authors: Hanan Rashid; Aniek Lamberts; Gilles F H Diercks; Hendri H Pas; Joost M Meijer; Maria C Bolling; Barbara Horváth Journal: Am J Clin Dermatol Date: 2019-12 Impact factor: 7.403