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Int J Clin Exp Pathol 2013;6(12):3014-3018

Case Report
Adult T-cell leukemia/lymphoma accompanying follicular mucinosis: a case report with review of
the literature

Mitsuaki Ishida, Muneo Iwai, Keiko Yoshida, Akiko Kagotani, Hidetoshi Okabe

Department of Clinical Laboratory Medicine and Division of Diagnostic Pathology, Shiga University of Medical Science, Shiga, Japan

Received September 30, 2013; Accepted October 23, 2013; Epub November 15, 2013; Published December 1, 2013

Abstract: Follicular mucinosis is recognized as one of the histopathological reaction patterns characterized by the accumulation of mucin
within follicular epithelium. It is induced by various causes including inflammatory diseases, and more than half of the cases are associated
with malignant lymphoma, mainly mycosis fungoides. Herein, we describe the third documented case of adult T-cell leukemia/lymphoma
(ATLL) accompanying follicular mucinosis. A 72-year-old Japanese male presented with persistent erythema in his arm and neck. Laboratory
tests demonstrated positivity for human T-cell leukemia virus (HTLV)-1 antibodies. Histopathological study of the biopsy specimen from the
neck revealed superficial perivascular, nodular, and intrafollicular lymphocytic infiltrations. These lymphocytes were small- to medium-sized
and had convoluted nuclei. Mucoid material deposition was observed within the hair follicles, and it was digested by hyaluronidase.
Immunohistochemically, these lymphocytes were positive for CD3, CD4, CD25, and Foxp3. Accordingly, an ultimate diagnosis of ATLL
accompanying follicular mucinosis was made. The skin is the most common extralymphatic site of involvement of ATLL. The present case
clearly demonstrated that albeit extremely rare, ATLL can cause follicular mucinosis. Therefore, ATLL should be included in the differential
diagnostic consideration of follicular mucinosis. (IJCEP1309078).

Keywords: Adult T-cell leukemia/lymphoma, follicular mucinosis, skin

Address correspondence to: Dr. Mitsuaki Ishida, Department of Clinical Laboratory Medicine and Division of Diagnostic Pathology, Shiga
University of Medical Science, Tsukinowa-cho, Seta, Otsu, Shiga, 520-2192, Japan. Tel: +81-77-548-2603; Fax: +81-77-548-2407; E-mail:
mitsuaki@belle.shiga-med.ac.jp