However, we report herein the first case to our knowl-

edge of eruptive nevi in a patient receiving cyclosporine

A monotherapy for a dermatologic condition. The use of
this drug without any other drug might show a relation-
ship between the development of eruptive melanocytic
nevi and cyclosporine. Given the uncertain nature of erup-
tive nevi and the increased susceptibility of immunosup-
pressed patients to skin cancers, it is essential that we
perform regular screenings of our immunosuppressed pa-
tients to detect dysplastic nevi or melanoma.
Verónica López, MD
Inmaculada Molina, MD
José M. Martı́n, MD
Nuria Santonja, MD
Marı́a José Forner, MD
Esperanza Jordá, MD, PhD Figure 1. Erosions of the vermillion portion of the lower lip and the tongue.

Author Affiliations: Departments of Dermatology (Drs

López, Molina, Martı́n, and Jordá), Pathology (San-
tonja), and Internal Medicine (Forner), Hospital Clı́nico
Universitario de Valencia, Valencia, Spain
Correspondence: Dr López, Hospital Clı́nico Universi-
tario de Valencia, Avda Blasco Ibáñez, nº 17, 46010 Va-
lencia, Spain (veronica_17@live.com).
Financial Disclosure: None reported.
1. Gelfer A, Rivers JK. Long-term follow-up of a patient with eruptive melano-
cytic nevi after Stevens-Johnson syndrome. Arch Dermatol. 2007;143(12):
1555-1557.
2. Bovenschen HJ, Tjioe M, Vermaat H, et al. Induction of eruptive benign me-
lanocytic naevi by immune suppressive agents, including biologicals. Br J
Dermatol. 2006;154(5):880-884.
3. Piaserico S, Alaibac M, Fortina AB, Peserico A. Clinical and dermatoscopic
fading of post-transplant eruptive melanocytic nevi after suspension of im-
munosuppressive therapy. J Am Acad Dermatol. 2006;54(2):338-340.
4. Cardones AR, Grichnik J. ␣-Melanocyte-stimulating hormone–induced erup-
tive nevi. Arch Dermatol. 2009;145(4):441-444. Figure 2. Erosions of the hard palate.
5. Scope A, Marghoob AA, Chen CS, Lieb JA, Weinstock MA, Halpern AC; SONIC
Study Group. Dermoscopic patterns and subclinical melanocytic nests in nor-
mal-appearing skin. Br J Dermatol. 2009;160(6):1318-1321.
nopathy nor hepatosplenomegaly was found. The re-
mainder of the physical examination findings were un-
remarkable. Diagnoses of herpes virus infection and
paraneoplastic pemphigus were considered. Treatment
Oral Manifestation of T-Cell with valacyclovir was immediately started but was inef-
Leukemia/Lymphoma fective, and results for a herpetic infection were nega-
tive on the Tzanck test and viral cultures of the lips and
Report of a Case. A 34-year-old woman from Guinea with hard palate. A biopsy specimen of the lower mucosal lip
no medical history was diagnosed in 2007 with acute adult showed a massive infiltrate of lobulated lymphoid
T-cell leukemia/lymphoma (ATL/L) when she was seen cells with hyperlobulated nuclei in the epithelium and
with fatigue, lymphocytosis with cerebriform cells, and Pautrier-like microabscesses (Figure 3). Immunohis-
multiple osteolytic lesions. Immunologic typing showed tochemical analyses showed that these abnormal cells were
that these cells strongly expressed CD3, CD4, and CD25. positive for CD3 and CD25. Cutaneous direct immuno-
Human T-cell lymphotropic virus type 1 (HTLV-1) se- fluorescence findings were negative, and no circulating
rologic test results were positive by enzyme-linked im- antibodies directed against the cell surface of keratino-
munosorbent assay and Western blot analysis. Thora- cytes were detected. A bone marrow smear confirmed the
coabdominopelvic computed tomography and bone medullar involvement with 58% tumoral cells. Specific
marrow biopsy specimens revealed no abnormalities. Six meningoencephalitis developed, and other chemothera-
cycles of CHOP chemotherapy (cyclophosphamide, doxo- peutic regimens were ineffective: the oral lesions wors-
rubicin, vincristine, and prednisone) with bortezomib re- ened. At last follow-up only supportive care was being
sulted in partial response. given.
In January 2009 (8 months after the last cycle of che-
motherapy), the patient sought medical advice for recent- Comment. Adult T-cell leukemia/lymphoma is an ag-
onset multiple painful erosions of the oral cavity involv- gressive malignant condition associated with HTLV-1 pro-
ing the dorsal aspect of the tongue, the hard palate, and virus integrated into the DNA of tumor cells.1,2 The vi-
the lips (Figure 1 and Figure 2). Neither lymphade- rus is endemic mainly in Japan, Africa, South America,

(REPRINTED) ARCH DERMATOL/ VOL 146 (NO. 7), JULY 2010 WWW.ARCHDERMATOL.COM
804

©2010 American Medical Association. All rights reserved.

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Figure 3. Biopsy specimen from the lower mucosal lip, Note the massive
infiltrate of hyperlobulated lymphocytic cells with nuclear atypia in the
stroma and mucosal epithelium (hematoxylin-eosin, original magnification
⫻20).
and the Caribbean basin, and 3% to 5% of seropositive
patients will develop ATL/L. Diagnosis relies on cyto-
logic findings of “flower cells” and on an immunologi-
cal phenotype of neoplastic T cells positive for CD3, CD4,
CD5, and CD25 but that do not express CD7 or CD8 in
peripheral blood or in tissues.1,2
Acute, chronic, and smoldering lymphoma are dis-
tinguished based on the presentation, progression, and
response to treatment. Lymphadenopathy, hepatospleno-
megaly, and hypercalcemia are usually present in ATL/L.
Specific cutaneous lesions are a common finding, ob-
served in 50% to 70% of patients, including erythema-
tous papules, nodules, plaques, and erythroderma.1,2 Con-
versely, mucosal lesions are rare. Indeed, in a series of
195 patients with ATL/L, only 4 patients with the lym-
phoma type had a tumor mass of the palate.1 Four other
cases have been well described with oral lesions as the
presenting sign of the disease, consisting of a nodule in
the lingual dorsal portion in a lymphoma type,3 a severe
ulcerative stomatitis in an acute type,4,5 and an ulcer-
ated swelling involving the hard palate and the gingival
mucosa in a lymphoma type.6 Usually chemotherapy has
limited efficacy on ATL/L, but in 2 cases, improvement
of oral lesions was observed under treatment.5,6 Oral le-
sions present in patients with ATL/L are indications of
the disease or its progression.
Edouard Bégon, MD
Laurent Blum, MD
Riad Benramdane, MD
Christelle Leroux, MD
Francis Pascal, MD
Annie Lévy, MD
Claude Bachmeyer, MD
Author Affiliations: Unité Fonctionnelle de Dermatolo-
gie (Drs Bégon and Blum) and Unité Fonctionnelle
d’Hématologie (Dr Benramdane), Centre Hospitalier
Général René Dubos, Pontoise, France; Service de Der-
matologie, Centre de référence des dermatoses bulle-
uses auto-immunes (Drs Leroux and Pascal), and Anato-
mopathologie (Dr Lévy), CHU Avicenne, Bobigny, France;
(REPRINTED) ARCH DERMATOL/ VOL 146 (NO. 7), JULY 2010
805
©2010 American Medical Association. All rights reserved.
Downloaded From: http://archderm.jamanetwork.com/ by a New York University User on 05/11/2015
and Service de Médecine Interne, CHU Tenon, AP-HP,
Paris, France (Dr Bachmeyer).
Correspondence: Dr Bachmeyer, Service de Médecine In-
terne, CHU Tenon (AP-HP), 4 rue de la Chine, 75020
Paris, France (claude.bachmeyer@tnn.aphp.fr).
Financial Disclosure: None reported.
1. Pombo De Oliveira MS, Loureiro P, Bittencourt A, et al; The Brazilian ATLL
Study Group. Geographic diversity of adult T-cell leukemia/lymphoma in Brazil.
Int J Cancer. 1999;83(3):291-298.
2. Setoyama M, Katahira Y, Kanzaki T. Clinicopathologic analysis of 124 cases
of adult T-cell leukaemia/lymphoma with cutaneous manifestations: the smol-
dering type with skin manifestations has a poorer prognosis than previously
thought. J Dermatol. 1999;26(12):785-790.
3. Segami N, Hosoda M, Fukuda M, Sugihara T, Yahata Y. Adult T-cell leukaemia/
lymphoma of the tongue. Oral Surg Oral Med Oral Pathol. 1990;70(2):206-
209.
4. Matsui R, Yamaguchi K, Tabata M, Soejima K, Sugihara K. A case of acute
adult T-cell leukemia with severe ulcerative stomatitis considered as an early
symptom [in Japanese]. Japanese J Oral Maxillofacial Surg. 2004;50(4):251-
254.
5. Owatari S, Uozumi K, Tokunaga M, et al. Adult T-cell leukaemia/lymphoma
in a 21-year-old man. Clin Lab Haematol. 2006;28(2):141-144.
6. Albuquerque MA, Migliari DA, Sugaya NN, et al. Adult T-cell leukemia/
lymphoma with predominant bone involvement, initially diagnosed by its oral
manifestation: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod.
2005;100(3):315-320.
Durable Complete Remission
of Therapy-Refractory, Tumor-Stage
Cutaneous T-Cell Lymphoma Under
Radioimmunotherapy With Electron Beam
Irradiation and Denileukin Diftitox
C utaneous T-cell lymphoma treated in the early
stages has an excellent prognosis. However, in
advanced stages, the prognosis is fatal; thera-
peutic response rates are low; and quality of life is se-
verely reduced. Hence, a therapy that achieves a sub-
stantial and long-lasting down-staging of tumor burden
in the advanced stages is warranted.
Report of a Case. An 87-year-old man was seen with ul-
cerative skin tumors of the foot and erythematous
plaques and patches. Histologic analysis revealed cuta-
neous T-cell lymphoma (mycosis fungoides) in tumor
stage with a high proliferation index and a CD30 trans-
formation. Further staging examinations were not in-
dicative for systemic lymphoma. Subsequently, local-
ized radiotherapy for the tumors in combination with
methotrexate treatment, 20 mg/wk, achieved a com-
plete remission.
However, after 4 months, the disease recurred, mani-
festing in disseminated plaques. During the following
months, different therapeutic strategies including irra-
diation and systemic therapy with bexarotene, lipo-
somal doxorubicin, the histone deacetylase inhibitor dep-
sipeptide, and anti–CD4 antibody did not produce
sufficient disease control (Figure, A and B). With re-
gard to this heavily pretreated elderly patient in a re-
duced physical condition showing rapidly progressive dis-
ease, we initiated a compassionate-use therapy of
denileukin diftitox (Ontak; Eisai Inc, Woodcliff Lake, New
Jersey) in combination with a modified total skin elec-
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