Herpes virus (KSHV) associated kaposi sarcoma in a 3-year-old child with non-HIV-induced immunodeficiency

Kusenbach, G.; Rübben, Albert; Schneider, E. M.; Barker, Michael; Büssing, Arndt; Lassay, Lisa; Skopnik, H.; Heiman, G.

doi:10.1007/s004310050633

Cited by 10 publications

(4 citation statements)

References 20 publications

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“…17,18,21 In these patients, KS lesions appeared shortly after the introduction of the immunosuppressive drugs and disappeared after these drugs were withdrawn. Similar observations have been made regarding organ transplantations, 20 as well as nontransplant immunocompromised situations, such as recurrent leukemia, 23 congenital immune deficiency, 24 and immunosuppressive treatment for rheumatoid arthritis 25 or bullous pemphigoid. 26 However, all these reports were either descriptional or were based on circumstantial evidence.…”

Section: Discussionsupporting

confidence: 62%

Classical Kaposi sarcoma

Brenner

Weissmann-Brenner

Rakowsky

et al. 2002

Cancer

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BACKGROUNDClassical Kaposi sarcoma (CKS) is a rare indolent neoplasm that is particularly prevalent among Jews of Ashkenazi and Mediterranean origin. Data regarding prognostic factors for CKS are scarce. The aim of the current retrospective analysis was to better define prognostic subgroups among patients with CKS.METHODSBetween 1960 and 1995, 248 consecutive patients with CKS were treated at the Rambam and Rabin Medical Centers in Israel. Although treatment options included local excision, radiotherapy, and chemotherapy, observation alone was used for 31% of patients. For prognostic factor analysis, disease progression was classified as any progression and dissemination, and progression‐free survival was calculated for each.RESULTSAt a median follow‐up of 20 months, four patients (1.6%) died of CKS. Of the patients eligible for analysis, 94 of 220 (39%) had any progression and 23 of 120 (18%) had dissemination. Only 8 of 202 (4%) had visceral spread. On univariate analysis, age was a statistically significant prognostic factor for any progression (P = 0.04), whereas immunosuppression and visceral involvement at presentation had only borderline significance. Immunosuppression was the only prognostic factor for dissemination (P = 0.003). On multivariate analysis, both age and immunosuppression were significant prognostic factors for any progression (P = 0.001 and 0.01, respectively). Immunosuppression was also predictive of dissemination (P = 0.006).CONCLUSIONSImmunosuppression and older age (50 years and older) are strongly associated with poorer outcome among CKS patients. The two end points used in this study may be used for future prognostic factor analyses. Cancer 2002;95:1982–7. © 2002 American Cancer Society.DOI 10.1002/cncr.10907

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Section: Discussionsupporting

confidence: 62%

Classical Kaposi sarcoma

Brenner

Weissmann-Brenner

Rakowsky

et al. 2002

Cancer

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“…Our patient had received systemic steroid treatment for 1 month and a first blood transfusion. Only 1 month after, KS associated with PID has been previously reported in only two children with PIDs, one child with complete IFNγR1 deficiency and mycobacterial coinfection [5] and in one child with an ill-defined PID associated with profound CD4 lymphopenia [15]. Why is KS so uncommon in children with PIDs, and why are PIDs so rare among children with KS?…”

Section: Discussionmentioning

confidence: 96%

Kaposi’s sarcoma in a child with Wiskott-Aldrich syndrome

Pïcard

Mellouli²,

Duprez

et al. 2006

Eur J Pediatr

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“…The histological features of the four forms of KS are identical, with spindle-shaped cells, inflammation, and angioproliferation with erythrocyte extravasation, suggesting a similar pathogenic mechanism. With the exception of epidemic KS, KS is very rare in children, suggesting that affected patients may suffer from an inherited immunodeficiency (182,183).…”

Section: Hhv8 Immunitymentioning

confidence: 99%

Human inborn errors of immunity to herpes viruses

Jouanguy

Béziat

Mogensen

et al. 2020

Current Opinion in Immunology

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Infections with any of the nine human herpes viruses (HHV) can be asymptomatic or lifethreatening. The study of patients with severe diseases caused by HHVs, in the absence of overt acquired immunodeficiency, has led to the discovery or diagnosis of various inborn errors of immunity. The related inborn errors of adaptive immunity disrupt α/β T-cell rather than B-cell immunity. Affected patients typically develop HHV infections in the context of other infectious diseases. However, this is not always the case, as illustrated by inborn errors of SAP-dependent Tcell immunity to EBV-infected B cells. The related inborn errors of innate immunity disrupt leukocytes other than T and B cells, non-hematopoietic cells, or both. Patients typically develop only a single type of infection due to HHV, although, again, this is not always the case, as illustrated by inborn errors of TLR3 immunity resulting in HSV-1 encephalitis in some patients and influenza pneumonitis in others. Most severe HHV infections in otherwise healthy patients remains unexplained. The forward human genetic dissection of isolated and syndromic HHV-

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Herpes virus (KSHV) associated kaposi sarcoma in a 3-year-old child with non-HIV-induced immunodeficiency

Abstract: The case supports the hypothesis that KSHV is involved in the aetiology of KS. Bone marrow transplantation is possibly a therapeutic option for KS in patients with immunodeficiency not related to human immunodeficiency virus infection.

Cited by 10 publications

References 20 publications

Classical Kaposi sarcoma

Classical Kaposi sarcoma

Kaposi’s sarcoma in a child with Wiskott-Aldrich syndrome

Human inborn errors of immunity to herpes viruses

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