Oral Manifestations of T-Cell Large Granular Lymphocytic Leukemia: a Case Report

Abstract

BackgroundT-cell large granular lymphocytic (T-LGL) leukemia is a rare, chronic, often indolent lymphoproliferative disorder of mature T cells (CD3+). Severe neutropenia and other cytopenias are common features in patients with T-LGL leukemia and may cause infections, thus representing a major cause of morbidity in this disease. Immunosuppressive therapy with low-dose regimes of methotrexate, cyclophosphamide, corticosteroids or cyclosporine A is the treatment of choice. Amongst the variety of T-LGL leukemia complications, oral manifestations such as ulcers have been rarely reported. The purpose of this paper is to report a case of T-cell large granular lymphocyte leukemia with oral manifestations and to discuss their pathogenesis and management.MethodsIn the present case, a 65 year old female with a two-month history of diagnosed T-LGL leukemia presented with oral lesions, including ulcerations on the ventral tongue and soft palate as well as swollen, erythematous and ulcerated gingiva. The patient was under treatment with methotrexate, granulocyte colony-stimulating factor (G-CSF) and erythropoietin.ResultsConsidering patients' medical history and clinical appearance of the lesions, a clinical diagnosis of a neutropenic ulcer of the tongue was established. The oral lesions resolved after treatment with antibiotics, topical steroids and antiseptics combined with improvement of the hematological condition. The pertinent literature related to T-LGL leukemia ethiopathology, diagnostics and treatment was discussed.ConclusionsAlthough rare, T-cell large granular lymphocytic leukemia should be included in the list of lymphoproliferative disorders, which may present with oral manifestations as a result of the disease and its treatment complications.

Gingival Ulcerations in a Patient with Acute Myeloid Leukemia: A Case Report and Literature Review

“…In 2000, Copete and Sheridan 3 concluded that it was still unclear whether ulcerations in the oral cavity of patients undergoing leukemia treatment were caused by infective etiology or they were manifestations of neutropenia. The same was concluded by Arvanitidou et al 4 in 2011. It seems that the degree of susceptibility to infection is roughly proportional to the blood neutrophil count.…”

“…To prevent abnormal increases in such bacteria on the gingiva, scaling and/or root planing together with local antiseptic and local/systemic antibiotic may contribute to infection control in such patients. This is sometimes impossible as patients may have severe neutropenia or thrombocytopenia, as seen in our case, as well as in cases reported by Arvanitidou et al 4 .…”

“…Oral neutropenic ulcerations have been so far described in patients suffering from neutropenia, multiple myeloma, and acute as well as T-large cell leukemia [1][2][3][4] . Barrett and Buckley 2 report that neutropenic ulceration and herpes simplex infection were the major causes of morbidity in three patients following administration of high-dose melphalan for the treatment of multiple myeloma.…”

“…It is well known that chemotherapy agents such as methotrexate might induce oral ulcerations and that these might be dose dependent. It is well recognized that patients may benefit from temporary discontinuation of methotrexate 4 ; and 3) infectious etiology of oral ulcerations.…”

Paraneoplastic pyoderma gangrenosum‐like ulceration in T‐cell large granular lymphocytic leukemia

“…Außerdem gibt es zahlreiche paraneoplastische Symptome bei der LGL-Leukämie, darunter B-Zell-Krankheiten (zum Beispiel die Monoklonale Gammopathie unklarer Signifikanz [MGUS]) und Zytopenien (hauptsächlich Neutropenie) [5]. Wenn auch selten berichtet, können Ulzerationen der Haut oder der Mundhöhle mit einem Pyoderma-gangraenosum-artigen Erscheinungsbild auftreten [5,[10][11][12]. Klinische, histologische und immunhistologische Untersuchungen bei unserem und einem früheren Fall [10] legen nahe, dass der ulzerative Prozess möglicherweise auf eine mikrovaskuläre Schädigung, verursacht durch eine intravaskuläre Ansammlung von T-LGLs in der Dermis, zurückzuführen ist.…”