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Letters to the Editor
Evolution of a patient with submaxillitis secondary to azathioprine, 4 years later
Evolución de paciente con submaxilitis secundaria a azatioprina, 4 años después
Adriana R. Guerra Romero
Corresponding author
adrianaguerra17@gmail.com

Corresponding author.
, Pilar López Serrano, José Lázaro Pérez Calle
Servicio de Digestivo del Hospital Universitario Fundación Alcorcón, Alorcón, Madrid, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We read with interest an opinion piece by Dr Blasco Pati&#241;o et al&#46; recently published in <span class="elsevierStyleSmallCaps">Gastroenterolog&#237;a y Hepatolog&#237;a</span> under the title &#8220;Acute submandibular sialadenitis in a patient with Crohn&#39;s disease being treated with infliximab&#8221;&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The authors reported the case of a patient with penetrating ileal impairment meeting the criteria for treatment with tumour necrosis factor &#40;TNF&#41; inhibitors&#46; This patient started treatment with infliximab and&#44; six weeks later&#44; developed bilateral acute submaxillitis&#44; which followed a good course after steroids were started&#46; Their piece referred to a letter previously published in the same journal in which we reported the same complication in a patient with Crohn&#39;s disease of the ileum and colon&#44; but in our case after starting treatment with azathioprine&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">This complication relapsed after the drug was suspended and subsequently switched to mercaptopurine&#46; Therefore&#44; according to the score on the Naranjo scale&#44; we determined that thiopurines were likely the origin of the patient&#39;s submaxillitis&#44; possibly due to an idiosyncratic&#44; dose-independent mechanism&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">We agree with the authors on the difficulty of determining a direct pathophysiological mechanism accounting for the development of this condition&#46; However&#44; we disagree with Blasco et al&#46; that the most plausible hypothesis was reactivation of an indeterminate virus with a special tropism for the salivary glands due to pharmacological immunosuppression &#40;in their case due to infliximab and in ours due to azathioprine&#41;&#46; For this reason&#44; we would like to report the course of our case since November 2017&#44; when it was published&#44; following suspension of azathioprine&#47;mercaptopurine&#46; The patient&#44; despite maintaining immunosuppressant treatment with TNF inhibitors&#44; has not presented any further episodes of clinical submaxillitis&#46; At present&#44; he remains on maintenance biologic therapy&#44; without having required any dose escalation&#44; in clinical remission with mucosal healing demonstrated on his most recent colonoscopy&#44; a year ago&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">This course resembles that reported by Alves da Silva et al&#46;&#44; who recently published another case of submaxillitis possibly linked to azathioprine&#44; also resolved after switching to adalimumab&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Finally&#44; perhaps we should reflect on the origin of submaxillitis in patients with inflammatory bowel disease and even raise another possibility in addition to those discussed above &#40;adverse drug effect versus immunosuppression&#41;&#44; which would be that of an extraintestinal manifestation not reported to date in relation to disease activity&#59; however&#44; that would require a larger number of available cases for purposes of measuring statistically significant associations that account for the onset of this condition in these patients&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0035" class="elsevierStylePara elsevierViewall">No funding was received for this study&#46;</p></span></span>"
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