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Scientific letter
Disseminated Mycobacterium scrofulaceum infection in a patient in treatment with golimumab
Infección diseminada por
Clara González Ojea
Corresponding author
clara.gonzalez.ojea@sergas.es

Corresponding author.
, Rebeca Longueira Suárez, Adrián Sousa Dominguez, Manuel Crespo Casal
Unidad de Enfermedades Infecciosas, Servicio de Medicina Interna, Hospital Álvaro Cunqueiro, Vigo, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Mycobacterium scrofulaceum &#40;M&#46; scrofulaceum&#41;</span> belongs to the nontuberculous mycobacteria group&#44; with common presentation as cervical lymphadenopathy in infancy&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> There are only a few cases of disseminated infection described to date&#44; for which reason we present below the case of an immunocompromised adult&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 38-year-old male is diagnosed with psoriatic arthropathy and had been being treated with golimumab for 2 years &#40;he had previously received etanercept and adalimumab&#41;&#46; He attended due to general malaise&#44; headache&#44; cough and a fever of 39&#176; with no response to outpatient antibiotics&#46; In the physical examination he presented with nuchal rigidity for which reason&#44; in light of a normal brain computed tomography &#40;CT&#41;&#44; a lumbar puncture was conducted with cerebrospinal fluid compatible with lymphocytic meningitis&#44; with 12 leukocytes&#47;mm<span class="elsevierStyleSup">3</span> &#40;100&#37; mononuclear&#41;&#44; glucose 67<span class="elsevierStyleHsp" style=""></span>mg&#47;dl&#44; proteins 28<span class="elsevierStyleHsp" style=""></span>mg&#47;dl and ADA 1&#46;7<span class="elsevierStyleHsp" style=""></span>u&#47;l&#46; In ordinary culture&#44; Lowenstein and multiple PCR of the cerebrospinal fluid were negative&#46; In light of mediastinal widening on the x-ray&#44; a CT was performed&#44; with pulmonary micronodules&#44; mediastinal and mesenteric lymphadenopathies&#44; and splenomegaly&#44; findings suggestive of miliary tuberculosis &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Following a bronchoscopy with biopsy of right paratracheal adenopathy empiric antituberculosis treatment was started &#40;isoniazid&#44; rifampicin&#44; pyrazinamide and ethambutol&#41; pending microbiological results&#46; The node biopsy revealed necrotising epithelioid granulomas with presence of acid-fast bacilli&#44; but in the culture material <span class="elsevierStyleItalic">M&#46; Scrofulaceum</span> was isolated sensitive to all tested antibiotics&#44; so antibiotics were adjusted to clarithromycin&#44; rifampicin and ethambutol&#46; This germ was not isolated in blood or cerebrospinal fluid&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The patient presented with good tolerance to treatment&#46; A CT at 6 months demonstrated practical resolution of the lymphadenopathies and pulmonary micronodules&#44; and after completing 12 months of treatment the patient is currently asymptomatic&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Atypical or nontuberculous mycobacteria are frequently found in the environment&#44; especially in water&#46; They are usually transmitted by inhalation or direct inoculation&#44; they do not spread from person to person and they do not usually cause disease in immunocompetent subjects&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> More than 150 species have been described&#44; and within these <span class="elsevierStyleItalic">M&#46; scrofulaceum</span> represents just 2&#46;2&#37; of these infections&#46; Their typical form of presentation is cervical lymphadenitis or scrofula in infancy&#44; followed by slowly growing cavitary pneumonia&#44; more common in the elderly and those with chronic lung disease&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Disseminated <span class="elsevierStyleItalic">M&#46; scrofulaceum</span>infection is exceptional&#46; Since the first report in 1971<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> we have found only 10 cases described in the literature&#44; 6 of these in adults&#58; one patient had an HIV infection&#44;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> 2 leukaemia&#44;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;5</span></a> in another the immunological situation was unknown<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> and the other 2 did not have known immunodeficiency&#44; except mild lymphopaenia in one of them&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;7</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Our patient was also immunosuppressed due to chronic treatment with golimumab&#46; This is an anti-tumour necrosis factor &#40;anti-TNF&#41; monoclonal antibody&#44; which acts by decreasing the immune response through blocking this proinflammatory cytokine&#46; It is employed in diseases such as rheumatoid arthritis&#44; psoriatic arthritis or ankylosing spondylitis&#46; However&#44; due to its action on the immune system&#44; it can increase the risk of bacterial infections&#44; infections by mycobacteria &#40;mainly tuberculosis but also atypical&#41;&#44; or opportunistic&#47;invasive fungal infections&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> As with other anti-TNFs cases of reactivation of latent tuberculosis have been described with the use of golimumab&#44;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> so it is essential to rule out this possibility and treat it if necessary&#44; before starting treatment with this drug&#46; In our patient&#44; latent tuberculosis infection was ruled out by a Mantoux test &#40;0<span class="elsevierStyleHsp" style=""></span>mm&#41; before starting treatment&#46; The risk of infection by nontuberculous mycobacteria is also heightened in patients treated with anti-TNF&#44; most often by <span class="elsevierStyleItalic">M&#46; avium complex</span>&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> However&#44; we have not found any described cases of infection by <span class="elsevierStyleItalic">M&#46; scrofulaceum</span> in the literature to date associated with the use of golimumab&#44; nor with other anti-TNFs&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Regarding treatment&#44; just as with TB&#44; discontinuation of the anti-TNF drug is recommended while treating the infection&#46; Regarding antibiotic treatment&#44; the regimen to be followed is not protocolised and neither is its duration&#46; Given that <span class="elsevierStyleItalic">M&#46; scrofulaceum</span> demonstrates higher rates of resistance that other mycobacteria<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> combination therapy guided by antibiogram is recommended for at least a year&#44; individualised according to the immunocompetence level of the patient&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interest</span><p id="par0035" class="elsevierStylePara elsevierViewall">There are no conflicts of interest and this paper has not been funded by an institution&#46;</p></span></span>"
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