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Conjunctival myeloid sarcoma in acute myeloblastic leukemia-M1
Sarcoma granulocítico conjuntival en leucemia mieloblástica aguda M1
P. Méndez-Cepedaa,
Corresponding author
, A.C. Millán-Rodrígueza, E. Diosa, C. Alvarezb, C. Sevillanoa
a Servicio de Oftalmología, Complexo Hospitalario de Pontevedra, Pontevedra, Spain
b Servicio de Anatomía Patológica, Complexo Hospitalario de Pontevedra, Pontevedra, Spain
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It can predict or express together with acute or chronic myeloblastic leukemia or with myelodisplasic syndromes&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Clinic case</span><p id="par0015" class="elsevierStylePara elsevierViewall">A male&#44; aged 69&#44; presented with a personal history of AML M1 &#40;myeloblastic without differentiation&#41; in complete remission &#40;CR&#41; which started 11 months earlier after chemotherapy treatment according to the PETHEMA-LAM 99 protocol&#46; In February 2008 the patient was admitted due to breathing condition with catarrh and significant nasal congestion without response to antibiotics&#46; In addition&#44; the patient exhibited erithematous papules in the back&#44; supraclavicular and sub-maxillary adenopathies&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The patient was referred by the hematologist due to red eye with 2 days evolution and without any other ocular symptoms&#46; The exploration gave a corrected visual acuity of 20&#47;20 in right eye &#40;RE&#41; and 18&#47;20 in left eye &#40;LE&#41;&#46; Ocular reflexes&#44; motility and tonometry were within normal ranges&#46; Anterior pole biomicroscopy showed in the RE a fleshy&#44; salmon-colored raised lesion in the upper &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41; and lower tarsus&#46; In the LE we found a similar lesion in the lower tarsal conjunctiva &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; The ocular fundus assessment did not reveal alterations&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">The possibility of leukemic involvement of the conjunctiva was considered and a biopsy of the lesion was carried out&#44; the histological study of which revealed a diffuse proliferation of myeloid habit cells arranged under the conjunctival epithelium which were positive at the immunohistochemical level for myeloperoxidase and CD43 &#40;<a class="elsevierStyleCrossRefs" href="#fig0015">Figs&#46; 3 and 4</a>&#41;&#44; and negative for B and T cell markers &#40;CD20&#44; CD3&#44; CD5&#41; as well as for TdT&#44; CD34 and CD117&#46; Accordingly&#44; the case was diagnosed as granulocytic sarcoma&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">New biopsies confirmed the involvement of the nasal mucosa and the skin&#46; With these findings&#44; the diagnosis was of MS involving the conjunctiva&#44; the skin and the nasal mucosa&#46; However&#44; in a peripheral blood and a medulla ossea sample relapse was not observed and therefore it was concluded that at this level the CR persisted&#46; Lumbar puncture as well as the rest of explorations gave normal results&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">After the diagnosis of extra-medullar relapse in the form of AML MS&#44; second-line treatment was initiated according to the FLAG-IDA protocol with a good response by the infiltrating lesions&#46; The patient died in May 2008&#44; 3 months after the MS diagnosis due to a multiorgan failure&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0040" class="elsevierStylePara elsevierViewall">Ocular involvement by leukemia is well known&#44; with the most frequent being retinal hemorrhage<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> usually due to the systemic effects of the disease such as leukocytosis&#44; hyperviscosity or anemia&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> In turn&#44; the retina is the most frequently involved ocular structure due to it being directly affected by leukemia cells&#44; up to 30&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">In AML&#44; the infiltration of the conjunctiva rarely occurs&#46; It is more frequent in the myelomonocytic M4 and monocytic M5 forms&#46; In the other types of AML&#44; only 4 cases of conjunctival infiltration have been described&#44; all of them located in the bulbar conjunctiva&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Two cases occurred in adults and a further 2 in children&#44; with 50&#37; of these cases being bilateral&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The appearance of the lesion was nodular although one was described as a &#8220;salmon-colored fleshy mass&#8221;&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> As far as we know&#44; this is the first case in which MS is diagnosed as a salmon-colored mass in the tarsal conjunctiva with bilateral expression in an adult in CR of M1 AML&#44; even though our patients exhibited MS simultaneously in other locations&#44; with the most frequent being the periosteum and the skin including the subcutaneous cellular tissue according to other studies&#46; Up to 25&#37; of MS are found in the orbit&#44; which is the most frequent location in children with AML&#46; Occasionally&#44; it appears in several areas at the same time as in the case presented here&#46; It usually indicates that the disease is at an advanced stage&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">By way of conclusion&#44; we emphasize that leukemic infiltration of the conjunctiva in the context of acute leukemia is an ominous prognostic sign&#46; In our case&#44; matching the mean descriptions of the literature&#44; survival did not exceed 3 months&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Said infiltration can be a sign of presentation of leukemia or relapse thereof and usually does not involve the eyesight&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conflict of interests</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors have not declared any conflict of interests&#46;</p></span></span>"
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        "resumen" => "<span class="elsevierStyleSectionTitle">Clinical case</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A 69-year-old man with a history of acute myeloblastic leukemia-M1 presented with bilateral conjunctival injection&#46; Ophthalmological examination revealed lesions located at the upper tarsal conjunctiva of the right eye and lower tarsal conjunctiva of both eyes&#46; Histological and immunohistochemical studies confirmed conjunctival myeloid sarcoma &#40;MS&#41;&#46; The patient died due to multiorgan failure three months later&#46;</p> <span class="elsevierStyleSectionTitle">Discussion</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Extramedullary recurrence of leukemia can appear as an ocular manifestation that brings about a recurrence of the leukemia&#44; leading to a poor prognosis&#46;</p>"
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        "resumen" => "<span class="elsevierStyleSectionTitle">Caso cl&#237;nico</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Var&#243;n de 69 a&#241;os diagnosticado de leucemia mielobl&#225;stica aguda M1 en remisi&#243;n completa&#44; remitido a Oftalmolog&#237;a por cuadro de ojo rojo no doloroso&#46; Biomicrosc&#243;picamente se observan lesiones sobreelevadas asalmonadas en conjuntiva tarsal superior de ojo derecho e inferior de ambos ojos&#46; Mediante estudios histol&#243;gicos e inmunohistoqu&#237;micos se confirma recidiva extramedular en forma de sarcoma mieloide en conjuntiva&#46; El paciente fallece a los 3 meses de un fallo multiorg&#225;nico&#46;</p> <span class="elsevierStyleSectionTitle">Discusi&#243;n</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">La recidiva extramedular de una leucemia puede presentarse como una manifestaci&#243;n ocular que conlleva una recurrencia de la leucemia&#44; siendo esta de mal pron&#243;stico&#46;</p>"
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