Epstein-Barr-associated leiomyomatosis and T-cell chimerism after haploidentical bone marrow transplantation for severe combined immunodeficiency disease

Srilatha Atluri, Kathleen Neville, Mary Davis, Kent Robertson, Francis E. Marshalleck, Dennis P. O'Malley, Rebecca H. Buckley, Robert Nelson

Research output: Contribution to journalArticle

11 Citations (Scopus)

Abstract

BACKGROUND: The clinical course of Epstein-Barr virus (EBV)-associated smooth muscle tumors is variable and there are no reports in patients with mixed T-cell chimerism after bone marrow transplantation (BMT). OBSERVATIONS: A child with X-linked severe combined immunodeficiency disease developed multiple renal and pulmonary leiomyomata 8 years after haploidentical BMT. Epstein-Barr viral DNA was detectable in the blood and in situ hybridization for EBV-encoded RNAs was positive in the tumor. The tumors have been radiographically stable, chimerism remains mixed, and plasma EBV DNA has been repeatedly negative for over 2 years after donor lymphocyte infusion. CONCLUSIONS: EBV-associated smooth muscle tumors may occur in patients who are partially reconstituted after BMT for severe combined immunodeficiency and may not require surgery or chemotherapy.

Original languageEnglish
Pages (from-to)166-172
Number of pages7
JournalJournal of Pediatric Hematology/Oncology
Volume29
Issue number3
DOIs
StatePublished - Mar 2007

Fingerprint

Leiomyomatosis
Severe Combined Immunodeficiency
Chimerism
Bone Marrow Transplantation
Human Herpesvirus 4
X-Linked Combined Immunodeficiency Diseases
Smooth Muscle Tumor
T-Lymphocytes
Viral DNA
Leiomyoma
In Situ Hybridization
Neoplasms
Tissue Donors
Lymphocytes
RNA
Kidney
Drug Therapy
Lung
DNA

Keywords

  • Donor lymphocyte infusion
  • EBV-associated leiomyomatosis
  • Mixed chimerism
  • Severe combined immunodeficiency disease

ASJC Scopus subject areas

  • Pediatrics, Perinatology, and Child Health
  • Oncology
  • Hematology

Cite this

Epstein-Barr-associated leiomyomatosis and T-cell chimerism after haploidentical bone marrow transplantation for severe combined immunodeficiency disease. / Atluri, Srilatha; Neville, Kathleen; Davis, Mary; Robertson, Kent; Marshalleck, Francis E.; O'Malley, Dennis P.; Buckley, Rebecca H.; Nelson, Robert.

In: Journal of Pediatric Hematology/Oncology, Vol. 29, No. 3, 03.2007, p. 166-172.

Research output: Contribution to journalArticle

Atluri, Srilatha ; Neville, Kathleen ; Davis, Mary ; Robertson, Kent ; Marshalleck, Francis E. ; O'Malley, Dennis P. ; Buckley, Rebecca H. ; Nelson, Robert. / Epstein-Barr-associated leiomyomatosis and T-cell chimerism after haploidentical bone marrow transplantation for severe combined immunodeficiency disease. In: Journal of Pediatric Hematology/Oncology. 2007 ; Vol. 29, No. 3. pp. 166-172.
@article{111cf5e6fa814d6f833399a0bad2ea1e,
title = "Epstein-Barr-associated leiomyomatosis and T-cell chimerism after haploidentical bone marrow transplantation for severe combined immunodeficiency disease",
abstract = "BACKGROUND: The clinical course of Epstein-Barr virus (EBV)-associated smooth muscle tumors is variable and there are no reports in patients with mixed T-cell chimerism after bone marrow transplantation (BMT). OBSERVATIONS: A child with X-linked severe combined immunodeficiency disease developed multiple renal and pulmonary leiomyomata 8 years after haploidentical BMT. Epstein-Barr viral DNA was detectable in the blood and in situ hybridization for EBV-encoded RNAs was positive in the tumor. The tumors have been radiographically stable, chimerism remains mixed, and plasma EBV DNA has been repeatedly negative for over 2 years after donor lymphocyte infusion. CONCLUSIONS: EBV-associated smooth muscle tumors may occur in patients who are partially reconstituted after BMT for severe combined immunodeficiency and may not require surgery or chemotherapy.",
keywords = "Donor lymphocyte infusion, EBV-associated leiomyomatosis, Mixed chimerism, Severe combined immunodeficiency disease",
author = "Srilatha Atluri and Kathleen Neville and Mary Davis and Kent Robertson and Marshalleck, {Francis E.} and O'Malley, {Dennis P.} and Buckley, {Rebecca H.} and Robert Nelson",
year = "2007",
month = "3",
doi = "10.1097/MPH.0b013e31803b95b3",
language = "English",
volume = "29",
pages = "166--172",
journal = "Journal of Pediatric Hematology/Oncology",
issn = "1077-4114",
publisher = "Lippincott Williams and Wilkins",
number = "3",

}

TY - JOUR

T1 - Epstein-Barr-associated leiomyomatosis and T-cell chimerism after haploidentical bone marrow transplantation for severe combined immunodeficiency disease

AU - Atluri, Srilatha

AU - Neville, Kathleen

AU - Davis, Mary

AU - Robertson, Kent

AU - Marshalleck, Francis E.

AU - O'Malley, Dennis P.

AU - Buckley, Rebecca H.

AU - Nelson, Robert

PY - 2007/3

Y1 - 2007/3

N2 - BACKGROUND: The clinical course of Epstein-Barr virus (EBV)-associated smooth muscle tumors is variable and there are no reports in patients with mixed T-cell chimerism after bone marrow transplantation (BMT). OBSERVATIONS: A child with X-linked severe combined immunodeficiency disease developed multiple renal and pulmonary leiomyomata 8 years after haploidentical BMT. Epstein-Barr viral DNA was detectable in the blood and in situ hybridization for EBV-encoded RNAs was positive in the tumor. The tumors have been radiographically stable, chimerism remains mixed, and plasma EBV DNA has been repeatedly negative for over 2 years after donor lymphocyte infusion. CONCLUSIONS: EBV-associated smooth muscle tumors may occur in patients who are partially reconstituted after BMT for severe combined immunodeficiency and may not require surgery or chemotherapy.

AB - BACKGROUND: The clinical course of Epstein-Barr virus (EBV)-associated smooth muscle tumors is variable and there are no reports in patients with mixed T-cell chimerism after bone marrow transplantation (BMT). OBSERVATIONS: A child with X-linked severe combined immunodeficiency disease developed multiple renal and pulmonary leiomyomata 8 years after haploidentical BMT. Epstein-Barr viral DNA was detectable in the blood and in situ hybridization for EBV-encoded RNAs was positive in the tumor. The tumors have been radiographically stable, chimerism remains mixed, and plasma EBV DNA has been repeatedly negative for over 2 years after donor lymphocyte infusion. CONCLUSIONS: EBV-associated smooth muscle tumors may occur in patients who are partially reconstituted after BMT for severe combined immunodeficiency and may not require surgery or chemotherapy.

KW - Donor lymphocyte infusion

KW - EBV-associated leiomyomatosis

KW - Mixed chimerism

KW - Severe combined immunodeficiency disease

UR - http://www.scopus.com/inward/record.url?scp=34247643283&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=34247643283&partnerID=8YFLogxK

U2 - 10.1097/MPH.0b013e31803b95b3

DO - 10.1097/MPH.0b013e31803b95b3

M3 - Article

VL - 29

SP - 166

EP - 172

JO - Journal of Pediatric Hematology/Oncology

JF - Journal of Pediatric Hematology/Oncology

SN - 1077-4114

IS - 3

ER -