Brief report
Sciatic and median nerve neurolymphomatosis as initial presentation of B-cell lymphoma
Neurolinfomatosis de los nervios ciático y mediano como presentación inicial del linfoma de linfocitos B
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Barahona, I. Adlerstein, J. Donoso, F. Mercado" "autores" => array:4 [ 0 => array:4 [ "nombre" => "D." "apellidos" => "Barahona" "email" => array:1 [ 0 => "danibarahona@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "I." "apellidos" => "Adlerstein" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "J." "apellidos" => "Donoso" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "F." "apellidos" => "Mercado" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Departamento de Imagenología, Facultad de Medicina, Clínica Alemana-Universidad del Desarrollo, Vitacura, Santiago, Chile" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Departamento de Hematología, Facultad de Medicina, Clínica Alemana-Universidad del Desarrollo, Vitacura, Santiago, Chile" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Neurolinfomatosis de los nervios ciático y mediano como presentación inicial del linfoma de linfocitos B" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1598 "Ancho" => 1500 "Tamanyo" => 214016 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(A) MIP image with partial response to treatment with persistence of some bone marrow uptake. PET/CT with complete response of the adrenal thickening (B) and resolution of the right sciatic nerve FDG uptake (C).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Neurolymphomatosis (NL) is the term use to describe direct infiltration of cranial nerves and/or nerves and roots from the peripheral nervous system by lymphoma, usually by B-cell non-Hodgkin’s lymphoma.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> NL is considered primary if the disease involves exclusively the peripheral nervous system and secondary if it derives of the spread from another organ,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> being the former less frequent.</p><p id="par0010" class="elsevierStylePara elsevierViewall">It is uncommon as initial presentation of the disease and can lead to extremely heterogeneous clinical manifestations as painful radiculopathies, cranial nerves neuropathies, mononeuropathies and polyradiculopathies. There are multiple other diseases that can be confused with these manifestations like paraneoplastic syndromes, nerve root compression, vasculitic neuropathies, neurosarcoidosis, among others. It is important to diagnose and start early treatment in all cases because its prognosis is poor, due also to a frequent late diagnosis. 18F-FDG PET/CT can help making and earlier diagnosis and staging. We present the case of a patient with NL as initial manifestation of a diffuse large B-cell lymphoma which was recognized on PET/CT.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0015" class="elsevierStylePara elsevierViewall">We report the case of a 72-year old male who presented with numbness of the right hand, progressive weakness in both lower limbs, night sweats and weight loss of approximately 10 kg in the last 6 weeks. At the moment of the initial evaluation the patient needed a cane to walk and presented bilateral paresis of hamstrings as well as hypoesthesia of the index and middle finger of the right hand. A 18F-FDG PET/CT was requested suspecting lymphoma, which showed bilateral hypermetabolic adrenal and renal masses, gastric ulcer, some small hypermetabolic adenopathies, multiple focal bone marrow uptake and intense uptake in both sciatic nerves and right median nerve (<a class="elsevierStyleCrossRef" href="#fig0005">Fig.1</a>). The hypermetabolic peripheral nerves were thickened and showed enhancement with ionidated contrast media on computed tomography. A supraclavicular node and gastric biopsy confirmed diffuse large-B-cell lymphoma, activated B cell type, stage IVb with high risk Central Nervous System International Prognostic Index (CNS-IPI). After 2 cycles of R-CHOP (rituximab, cyclophosphamide, doxorubicin, vincristine and prednisolone) and intrathecal methotrexate, an interim 18F-FDG PET/CT showed partial response with resolution of the peripheral nerves, adrenal and gastric uptake and lower uptake of the bone marrow and renal lesions (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). The sciatic nerves showed resolution of the hyperenhancement and thickening, improving his neurological symptoms. After 6 cycles of R-CHOP a new PET/CT evaluation showed progressive disease with greater extension of the peripheral nerves involvement, parotid lymphadenopathies and a right periorbital tumor (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">The diagnosis of NL may be difficult and delayed after the first onset of symptoms, for these reasons its true incidence is unknown. Baehring et al.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> reported 80% detection sensitivity in nerve biopsy and just 21% for cerebrospinal fluid cytology, but nerve biopsy is invasive and carries a risk of permanent nerve damage.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Imaging can play an important role avoiding invasive procedures, Grisariu et al. reported in a series of cases 77% of positive magnetic resonance imaging (MRI) and 84% (16 of 19) positive PET/CT<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> suggesting NL. MRI can characterize the morphological alterations of nerves and plexus with better resolution than PET/CT but 18F-FDG PET/CT could be the stand-alone study to explore the whole body and detect nerves and other organs compromised by lymphoma at the same time.</p><p id="par0030" class="elsevierStylePara elsevierViewall">NL has a characteristic appearance on PET/CT, it presents as a linear or fusiform FDG uptake along a neuronal path. The uptake can be variable ranging from SUV max 1.5 up to 17.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Most commonly, NL affects brachial and lumbar plexuses, peripheral nerves of the extremities and the trigeminal nerve root.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">18F-FDG PET/CT has proved to be useful in the staging and response assessment of Hodgkin’s lymphoma and aggressive Non-Hodgkin lymphoma (NHL), being part of the actual state of the art. It has prognostic value before and after the treatment, could guide the biopsy to the higher metabolic site, can detect relapse and unusual sites of disease as in this case.</p><p id="par0040" class="elsevierStylePara elsevierViewall">In our case the clinicians requested a PET/CT for the patient weight loss and night sweats to discard lymphoma but, at the beginning, not associating these symptoms to the neurological symptoms, which were related to the same disease. The multiorgan compromise detected on PET/CT suggested lymphoma as the first diagnosis and the biopsy were taken from the less invasive and risky sites, a supraclavicular node and gastric mucosa, confirming the suspicion.</p><p id="par0045" class="elsevierStylePara elsevierViewall">The patient was treated with systemic chemotherapy and intrathecal methotrexate with good initial response, PET/CT showed partial resolution of the disease and complete response of peripheral nerves. However, most patients with NL will relapse despite directed therapy, some may have long term survival; in the cohort of Davidson et al. the longest survivor lived for 7.82 years after NL diagnosis.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">In summary NL is a rare manifestation of NHL and it can present in different ways, making its diagnosis challenging, it should be considered in patients with lymphoma and neuropathy. 18F-FDG PET/CT could provide a whole-body evaluation helping in its prompt diagnosis, besides staging, guiding the best biopsy site and evaluating the treatment response.</p></span><span id="sec1020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect1040">Authorship</span><p id="par0055" class="elsevierStylePara elsevierViewall"><ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">1.</span><p id="par0060" class="elsevierStylePara elsevierViewall">Responsible for study integrity: DB.</p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">2.</span><p id="par0065" class="elsevierStylePara elsevierViewall">Study conception: DB.</p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">3.</span><p id="par0070" class="elsevierStylePara elsevierViewall">Study design: DB.</p></li><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">4.</span><p id="par0075" class="elsevierStylePara elsevierViewall">Data acquisition: DB, IA.</p></li><li class="elsevierStyleListItem" id="lsti0025"><span class="elsevierStyleLabel">5.</span><p id="par0080" class="elsevierStylePara elsevierViewall">Data analysis and interpretation: DB, IA.</p></li><li class="elsevierStyleListItem" id="lsti0030"><span class="elsevierStyleLabel">6.</span><p id="par0085" class="elsevierStylePara elsevierViewall">Statistical processing: N/A.</p></li><li class="elsevierStyleListItem" id="lsti0035"><span class="elsevierStyleLabel">7.</span><p id="par0090" class="elsevierStylePara elsevierViewall">Literature search: DB, FM.</p></li><li class="elsevierStyleListItem" id="lsti0040"><span class="elsevierStyleLabel">8.</span><p id="par0095" class="elsevierStylePara elsevierViewall">Drafting of the manuscript: DB, IA, FM.</p></li><li class="elsevierStyleListItem" id="lsti0045"><span class="elsevierStyleLabel">9.</span><p id="par0100" class="elsevierStylePara elsevierViewall">Critical review of the manuscript with intellectually significant contributions: JD.</p></li><li class="elsevierStyleListItem" id="lsti0050"><span class="elsevierStyleLabel">10.</span><p id="par0105" class="elsevierStylePara elsevierViewall">Approval of the final version: DB.</p></li></ul></p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Declarations of interest</span><p id="par1055" class="elsevierStylePara elsevierViewall">None.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres1726705" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1524924" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1726706" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1524925" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Case report" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec1020" "titulo" => "Authorship" ] 8 => array:2 [ "identificador" => "sec0020" "titulo" => "Declarations of interest" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-09-14" "fechaAceptado" => "2020-10-08" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1524924" "palabras" => array:4 [ 0 => "Neurolymphomatosis" 1 => "PET" 2 => "Neuropathy" 3 => "Diffuse large-B-cell lymphoma" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1524925" "palabras" => array:4 [ 0 => "Neurolinfomatosis" 1 => "PET" 2 => "Neuropatía" 3 => "Linfoma difuso de linfocitos B grandes" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Neurolymphomatosis (NL) is the infiltration of cranial nerves or nerves and roots from the peripheral nervous system by lymphoma, usually by B-cell non-Hodgkin’s lymphoma. It is uncommon as initial presentation of the disease and can lead to extremely heterogeneous clinical manifestations. We report the case of a 72-year old male who presented with numbness of the right hand, progressive weakness in both lower limbs and weight loss. 18F-FDG PET/CT showed bilateral hypermetabolic adrenal masses, gastric ulcer, small hypermetabolic adenopathies, multiple focal bone marrow uptake and intense uptake in both sciatic nerves and right median nerve. A node and gastric biopsy confirmed diffuse large-B-cell lymphoma, activated B cell type, with posterior resolution of peripheral nerves uptake after beginning chemotherapy.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">La neurolinfomatosis es la infiltración de los nervios craneales o de los nervios y raíces del sistema nervioso periférico por un linfoma, generalmente un linfoma no Hodgkin de linfocitos B. Es poco frecuente como presentación inicial de la enfermedad y puede dar lugar a manifestaciones clínicas extremadamente heterogéneas. Informamos del caso de un hombre de 72 años que presentaba entumecimiento de la mano derecha, debilidad progresiva en ambas extremidades inferiores y pérdida de peso. La PET/TC con <span class="elsevierStyleSup">18</span>F-FDG mostró masas suprarrenales hipermetabólicas bilaterales, úlcera gástrica, pequeñas adenopatías hipermetabólicas, captación de médula ósea de focalización múltiple y captación intensa tanto en los nervios ciáticos como en el nervio mediano derecho. La biopsia gástrica y de los ganglios confirmó un linfoma difuso de linfocitos B grandes, del tipo de linfocitos B activados, con una resolución posterior de la captación de los nervios periféricos después de iniciar la quimioterapia.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Barahona D, Adlerstein I, Donoso J, Mercado F. Neurolinfomatosis de los nervios ciático y mediano como presentación inicial del linfoma de linfocitos B. Radiología. 2022;64:266–269.</p>" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 963 "Ancho" => 900 "Tamanyo" => 115082 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) 18F-FDG PET/CT Maximum intensity projection (MIP) pretreatment. (B) Hypermetabolic gastric ulcer (white arrow), thickened and hypermetabolic adrenal glands (red arrows) and right median nerve uptake (white arrowhead). (C) High FDG uptake in both sciatic nerves (red arrows).</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1598 "Ancho" => 1500 "Tamanyo" => 214016 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(A) MIP image with partial response to treatment with persistence of some bone marrow uptake. PET/CT with complete response of the adrenal thickening (B) and resolution of the right sciatic nerve FDG uptake (C).</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1270 "Ancho" => 1500 "Tamanyo" => 141200 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">(A) MIP view displays peripheral nerves FDG uptake in arms and thighs. (B) PET/CT shows right hypermetabolic periorbital tumor (red arrow) and (C) thickened hypermetabolic sciatic nerves (white arrows).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Neurolymphomatosis: diagnosis, management, and outcomes in patients treated with rituximab" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "H.K. Gan" 1 => "A. Azad" 2 => "L. Cher" 3 => "P.L.R. 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