Human T-Lymphotropic Virus
Type-1 Related Adult T-cell
Leukemia/Lymphoma Presenting
as a Parotid Mass Diagnosed By
Fine-Needle Aspiration Biopsy
Guo-Xia Tong, M.D., Ph.D., Osvaldo Hernandez, M.D.,
Herman T. Yee, M.D., Ph.D., Shyun-Hua Zheng, M.D., Ph.D., and
Grace C.H. Yang, M.D., F.I.A.C.
*
A 48-yr-old black woman with a history of blood transfusions for
menorrhagia secondary to uterine fibroids but no known Carib-
bean association presented with a 6-wk history of a rapidly en-
larging right parotid mass. At the time of presentation, she could
not close her right eye. An aspiration biopsy showed small, me-
dium, and large lymphoma cells with angulated nuclei, red ma-
cronucleoli, and basophilic cytoplasm with fine vacuoles. Flow
cytometry indicated a (CD25
+
/CD7
-
) T-cell lineage, suggesting
an human T-lymphotropic virus (HTLV) 1-related T-cell leukemia/
lymphoma, which was confirmed by polymerase chain reaction
(PCR)-based amplification on DNA extracted from fresh tissue
with specific oligonucleotide primers for HTLV-1 DNA sequence.
Histology showed interstitial infiltration and destruction of the
parotid parenchyma by lymphoma cells without involvement of
adjacent lymph nodes. Total body CT scan and magnetic reso-
nance imaging (MRI) studies were negative for lymphadenopathy
but showed liver metastasis. To our knowledge, this is the first
reported case of HTLV-1-related primary parotid lymphoma as the
initial presentation of adult T-cell leukemia/lymphoma. Diagn.
Cytopathol. 2004;31:333–337. © 2004 Wiley-Liss, Inc.
Key Words: primary parotid lymphoma; T-cell lymphoma;
HTLV-1; adult T-cell lymphoma/leukemia; salivary gland neo-
plasms
Fine-needle aspiration (FNA) biopsy is used in the preop-
erative evaluation of parotid masses to triage benign from
malignant epithelial neoplasms.
1,2
Primary lymphoma of the
salivary glands accounts for only 1–2% of neoplasms and
10% of malignancies of salivary glands and 5% of extran-
odal lymphomas.
3
The majority of primary lymphomas of
salivary glands are of B-cell lineage and they usually arise
in the background of lymphoproliferation associated with
Sjo ¨gren’s syndrome. The most common subtype is low-
grade B-cell lymphoma of mucosa-associated lymphoid tis-
sue (MALT) type.
3,4
In a previous study from our Cytopa-
thology Laboratory,
5
all 18 cases of salivary gland
lymphomas were B-cell lymphomas. Primary T-cell lym-
phoma of the salivary gland is extraordinarily rare. Only 15
cases have been reported in the English literature, including
6 cases in the parotid gland, 5 cases in the submandibular
gland, 2 cases in the sublingual gland, and 2 cases in which
the salivary gland was unspecified.
6
Of the 15 cases, 9 cases
were from Chinese patients from mainland China or Hong
Kong,
7
4 cases were from Western population, and in the
remainder, the patients’ nationalities were not specified.
Many of these reported cases were positive for Epstein-Barr
virus (EBV) or human immunodeficiency virus (HIV) in-
fection.
7,8
We report here a primary T-cell lymphoma of the
parotid gland in a black woman who was negative for both
EBV and HIV infection but positive for human T-lympho-
tropic virus type-1 (HTLV-1). To the best of our knowl-
edge, it is the first reported case of HTLV-1-related primary
parotid T-cell lymphoma and the fifth case of primary T-cell
lymphoma of the salivary gland in the Western population.
Clinical History
A 48-yr-old previously healthy black woman noticed a
small painless nodule in her right cheek 6 wk before her
presentation at Bellevue Hospital Center. The nodule grew
rapidly, and 4 wk later, she experienced excruciating pain
radiating to her right forehead, eye, and retroauricular area.
Department of Pathology, New York University School of Medicine,
New York, New York
*Correspondence to: Grace C.H. Yang, M.D., Cytopathology Labora-
tory, NB 4S-16, New York University Medical Center, 462 First Ave.,
New York, NY 10016. E-mail: grace.yang@nyu.edu
Received 23 October 2003; Accepted 27 April 2004
DOI 10.1002/dc.20121
Published online in Wiley InterScience (www.interscience.wiley.com).
© 2004 WILEY-LISS, INC. Diagnostic Cytopathology, Vol 31, No 5 333