Literature DB >> 3823704

[Crow-Fukase syndrome (POEMS syndrome) and osseous mastocytosis secondary to Castleman's angiofollicular lymphoid hyperplasia].

J Lapresle, C Lacroix-Ciaudo, M Reynès, P Madoulé.   

Abstract

In a case of Crow-Fukase (POEMS) syndrome there was a chronic, progressive, and eventually lethal polyradiculoneuropathy. In addition, adenomegaly, oedema and pleural effusions, gonadic atrophy, serum monoclonal IgA, and skin pigmentation were present. Plain x-rays and CT scan of the pelvis and lower vertebrae showed multiple poorly defined lesions. At postmortem there was no myeloma and a bone mastocytosis was found. In addition, next to T11, there was an abdominal nodule, 2 cm in diameter, with histological characteristics of Castleman's angiofollicular lymphoid hyperplasia. Immunohistochemical studies showed that plasmocytes of this lesion secreted polyclonal immunoglobulins with a high prevalence of IgA. Thus, the primary interest of this case lies in the association of bone mastocytosis with a POEMS syndrome. Also, one single localisation of Castleman angiofollicular lymphoid hyperplasia was found, assumed to be in this case the cause of the POEMS syndrome. Therefore, a minute and benign hyperplasic lesion, which was only discovered at autopsy, secreted the protein responsible for the symptoms and signs, and eventually the patient's death.

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Year:  1986        PMID: 3823704

Source DB:  PubMed          Journal:  Rev Neurol (Paris)        ISSN: 0035-3787            Impact factor:   2.607


  4 in total

Review 1.  A case of POEMS syndrome associated with essential thrombocythaemia and dermal mastocytosis.

Authors:  A Jackson; I E Burton
Journal:  Postgrad Med J       Date:  1990-09       Impact factor: 2.401

2.  POEMS syndrome in a 15-year-old boy: radiological findings.

Authors:  Cecilia Lanza; Marco Misericordia; Giancarlo Fabrizzi
Journal:  Pediatr Radiol       Date:  2007-09-18

3.  Pathological findings in three non-Japanese patients with the POEMS syndrome.

Authors:  R Gherardi; M Baudrimont; M Kujas; D Malapert; F Lange; F Gray; J Poirier
Journal:  Virchows Arch A Pathol Anat Histopathol       Date:  1988

4.  Uncompacted myelin lamellae in polyneuropathy, organomegaly, endocrinopathy, M-protein and skin changes syndrome. Ultrastructural study of peripheral nerve biopsy from 22 patients.

Authors:  C Vital; R Gherardi; A Vital; N Kopp; J F Pellissier; M Soubrier; P Clavelou; R Bellance; M B Delisle; M M Ruchoux
Journal:  Acta Neuropathol       Date:  1994       Impact factor: 17.088

  4 in total

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