Xanthoma disseminatum with respiratory tract involvement and fatal outcome

Davies, Christopher W. H.; Marren, P.; Juniper, Mark; Gray, Winifred; Wojnorowska, Fenella; Benson, Matthew A.

doi:10.1136/thorax.55.2.170

Cited by 34 publications

(21 citation statements)

References 10 publications

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“…Prognosis of XD is related to the mucous membrane manifestations and involvement of the upper respiratory tract. 4 The response to any form of therapy in XD is unsatisfactory and resistant to treatment. 3 Probucol, cholesterol synthesis enzyme inhibitors and glucocorticoid medication did not influence the xanthomas.…”

Section: Discussionmentioning

confidence: 99%

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A Rare Case of Sessile Polypoidal Xanthoma of Vocal Cord

Shah¹,

Katarkar²,

Modh³

et al. 2012

International Journal of Phonosurgery &Amp; Laryngology

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We report a rare case of sessile polypoidal xanthoma of vocal cord in a 38-year-old female, presented with complaint of hoarseness of voice for 3 months. Hopkins telescopic examination showed a sessile polyp arising from the anterior aspect of laryngeal vocal cord. With clinical diagnosis of vocal cord polyp, microlaryngeal surgery was done and tissue was sent for histopathological and culture examination. Histopathological findings showed features of polypoidal xanthoma. Culture was negative.

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Section: Discussionmentioning

confidence: 99%

“…1 The common sites are skin and subcutis but occasionally involve the tendon or synovium. 1 Only few studies of xanthoma involving the larynx have been recorded in English literature [1][2][3][4] and sessile polypoidal xanthoma of larynx is extremely rare.…”

Section: Introductionmentioning

confidence: 99%

A Rare Case of Sessile Polypoidal Xanthoma of Vocal Cord

Shah¹,

Katarkar²,

Modh³

et al. 2012

International Journal of Phonosurgery &Amp; Laryngology

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“…Peripheral airway involvement is very rare compared to upper respiratory tract involvement, which is the most recognized cause of respiratory failure in XD [2, 7]. Recently, a few cases with involvement of small-sized bronchi, which causes a fatal outcome, have been reported [8, 9]. XD is usually benign and is often self-limiting, but pulmonary involvement suggests that the prognosis is poor.…”

Section: Discussionmentioning

confidence: 99%

Xanthoma disseminatum with Large Plaques Confined to the Back, Pulmonary Involvement and Multiple Intestinal Xanthomas

Hisanaga

Akaike²,

Kuroda³

2004

Dermatology

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Xanthoma disseminatum (XD) is a rare benign mucocutaneous xanthomatosis that is classified as a benign non-Langerhans cell histiocytosis. We report a 68-year-old man who presented with peculiar, large plaques confined to the back 7 years after the onset of cranial diabetes insipidus. Histopathological features of the cutaneous lesions were typical of XD. The patient had lower respiratory tract involvement with histiocytic infiltrates, which was unresponsive to various treatments and resulted in a fatal outcome. Gastrointestinal endoscopies revealed multiple xanthomas in the sigmoid colon and the rectum. To our knowledge, this is the first reported case of intestinal xanthomas associated with XD.

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“…However, when the lower respiratory tract was involved, such as in this case, the prognosis is poor. 104 There are rare reports of associations between XD and plasma cell dyscrasias and monoclonal gammopathy. 96 Histology.…”

Section: Xanthoma Disseminatummentioning

confidence: 98%