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Case Reports
. 2018 Jan 23:12:801.
doi: 10.3332/ecancer.2018.801. eCollection 2018.

An unusual presentation of adult T-cell leukemia/lymphoma

Mohammed Tag-Adeen  1   2 , Keiichi Hashiguchi  1 , Yuko Akazawa  1 , Ken Ohnita  1 , Sawayama Yasushi  3 , Niino Daisuke  4 , Kazuhiko Nakao  1
Affiliations
Case Reports

An unusual presentation of adult T-cell leukemia/lymphoma

Mohammed Tag-Adeen et al. Ecancermedicalscience. .

Abstract

Adult T-cell Leukemia/Lymphoma (ATL) is a rare disease, related to human T-lymphotropic virus-1 (HTLV-1) and presented mainly in adulthood by generalised lymphadenopathy, hepatosplenomegaly, skin lesions and hypercalcaemia, with rare gastrointestinal and/or oral manifestations. We reported this case to raise awareness and demonstrate the therapeutic challenges of this rare disease. A 49-year-old Japanese female presented with skin papules on both forearms, painful mouth ulcers and multiple neck swellings since early February 2017. Initial clinical examination and laboratory investigations were misleading and her condition was diagnosed as candidiasis. Because of un-improvement of the case, a screening upper endoscopy was requested 2 months later and revealed characteristic oropharyngeal ulcers which were biopsied, and its pathologic examination confirmed smouldering type ATL. This case report should raise awareness of doctors and endoscopists about this disease especially in HTLV-1 endemic areas to avoid late diagnosis and help achieve earlier therapeutic decisions.

Keywords: adult T-Cell leukaemia/lymphoma; endoscopic findings; human T-lymphotropic virus; lymphoma; oral ulcers; oropharyngeal ulcers.

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Figures

Figure 1.
Figure 1.. Endoscopic picture of ulcers at the soft palate and gingiva with irregular margins, raised edges and erythematous floors.
Figure 2.
Figure 2.. Ulcer at the uvula with white light endoscopy (left) and NBI (right).
Figure 3.
Figure 3.. Thickened aryepiglottic fold (yellow arrow) suspicious of infiltration.
Figure 4.
Figure 4.. Multiple submucosal swellings at the second part of the duodenum as seen by white light endoscopy (upper left), NBI (lower left) and magnified view (right).
Figure 5.
Figure 5.. Histopathologic sections from oropharyngeal mucosa, stained by hematoxylin & eosin with different magnification powers (×4: left side, ×8: right side); showing malignant lymphocytic infiltration with hyperchromatic enlarged nuclei and scanty cytoplasm.
Figure 6.
Figure 6.. Histopathologic sections stained by hematoxylin & eosin from the same specimen with higher magnification powers (×20: left side, ×40: right side).
Figure 7.
Figure 7.. Immune-histochemical staining of the same sections showing CD3-positive cells (×20: left side, ×40: right side).
Figure 8.
Figure 8.. Immune-histochemical staining of the same sections showing CD4-positive cells (×20: left side, ×40: right side).
Figure 9.
Figure 9.. Immune-histochemical staining of the same sections showing CD8-negative cells (x20: left side, x40: right side).
See this image and copyright information in PMC

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