Infantile Hemorrhagic Angiodysplasia

Odell, J. Michael; Haas, Joel E.; Tapper, David; Nugent, Diane J.

doi:10.3109/15513818709161426

Cited by 18 publications

(8 citation statements)

References 18 publications

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“…An excised lesion was referred to as ''cavernous hemangioma'' but was not illustrated [21]. The third report was that of a newborn with widely disseminated cutaneous and gastrointestinal vascular ectasias, termed ''infantile hemorrhagic angiodysplasia,'' who developed chronic gastrointestinal hemorrhage and thrombocytopenia [22]. Histopathology showed nests of dilated capillaries, arterioles, and postcapillary venules.…”

Section: Discussionmentioning

confidence: 99%

Cutaneovisceral Angiomatosis with Thrombocytopenia

et al. 2005

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We describe 10 children with multiple vascular lesions of the skin and gastrointestinal tract associated with sustained, minor thrombocytopenia. In some children, there was involvement of the lung (n = 5), bone (n = 2), liver (n = 1), spleen (n = 1), and muscle (n = 1). The cutaneous lesions were congenital, multifocal, discrete, red-brown and variably blue macules and papules; in 3 children, a large dominant plaque was also present. All children developed hematemesis and/or melena and endoscopic evaluation revealed several to numerous small mucosal lesions that involved all levels of the gastrointestinal tract. Three of 5 children with pulmonary nodules had cough and 1 also had hemoptysis. Biopsies of cutaneous, gastrointestinal, and pulmonary lesions showed thin-walled, blood-filled vascular channels and variable endothelial hyperplasia. The endothelial nuclei were elongated, round, crescentic, or hobnailed. Cytoplasmic and extracellular periodic acid-Schiff positive deposits were often present in the zones of endothelial hyperplasia. The platelets were small in some children, suggesting a primary defect, possibly accounting for the thrombocytopenia. Gastrointestinal hemorrhage and hemoptysis required antiangiogenic therapy. The constellation of findings defines a congenital proliferative disorder of blood vessels with a distinctive microscopic appearance. We have termed this relatively indolent or slowly progressive disorder cutaneovisceral angiomatosis with thrombocytopenia because this designation incorporates its major clinical and histopathologic features.

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Section: Discussionmentioning

confidence: 99%

Cutaneovisceral Angiomatosis with Thrombocytopenia

et al. 2005

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“…This lesion, also known as multifocal lymphangioendotheliomatosis with thrombocytopenia or infantile haemorrhagic angiodysplasia, is characterized by generalized multiple, red brown to blue, discrete papules, macules, plaques and nodules ranging in size from millimetres to several centimetres involving the trunk and extremities 53–55 . The lesions arise congenitally and new lesions continually appear throughout childhood.…”

Section: Recently Described Vascular Tumoursmentioning

confidence: 99%

Cutaneous vascular tumours: an update

Goh

Calonje

2008

Histopathology

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This review evaluates changes in vascular nomenclature particularly in the category of vascular tumours of intermediate malignancy that includes the various haemangioendotheliomas, Kaposi's sarcoma and giant cell angioblastoma. Recently described entities in this category, including the latter tumour and composite haemangioendothelioma, are described. Several newly characterized vascular tumours not included in recent classifications of vascular tumours are also summarized. These include acquired elastotic haemangioma, symplastic haemangioma, cutaneous epithelioid angiomatous nodule and cutaneovisceral angiomatosis with thrombocytopenia. The newly introduced clinical classification of congenital haemangiomas into non-involuting and rapidly involuting variants and radiation-induced atypical vascular lesions are also reviewed.

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“…Odell et al 4 have reported on a female infant who presented at birth with an unusual syndrome of disseminated cutaneous and gastrointestinal malformations associated with severe thrombocytopenia and chronic gastrointestinal hemorrhage. Her biopsy revealed that the cutaneous lesion was composed of a collection of tortuous, anastomosing ectatic vessels resembling post‐capillary venules and extending from the papillary dermis through the deep reticular dermis into subcutaneous fat.…”

Section: Discussionmentioning

confidence: 99%