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ACG Case Reports Journal

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name="wkhealth_article_publication_date" content="November 16, 2024" /><meta name="wkhealth_volume" content="11" /><meta name="wkhealth_ovid_accession_number" content="02075970-202411000-00013" /><meta name="wkhealth_issn" content="2326-3253" /><meta name="wkhealth_title" content="Orofacial Granulomatosis and Crohn&#39;s Disease: A Case Series" /><meta name="wkhealth_authors" content="Urquhart, Siri A.;Kim, Grace Y.;Anderson, Katelyn R.;Chedid, Victor G." /><meta name="wkhealth_doi" content="10.14309/crj.0000000000001559" /><meta name="wkhealth_abstract_html_url" content="https://journals.lww.com/acgcr/abstract/2024/11000/orofacial_granulomatosis_and_crohn_s_disease__a.13.aspx" /><meta name="wkhealth_fulltext_html_url" content="https://journals.lww.com/acgcr/fulltext/2024/11000/orofacial_granulomatosis_and_crohn_s_disease__a.13.aspx" /><meta name="wkhealth_toc_section" content="CASE REPORT" /><meta name="wkhealth_toc_sub_section" content="INFLAMMATORY BOWEL DISEASE" /><meta 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The most common treatments included intralesional corticosteroids (4 patients), oral corticosteroids (3 patients), and anti-TNF agents, specifically infliximab (3 patients). Intralesional corticosteroid administration was followed by partial response in 3 patients, but recurrences were common following initial injection. One patient had no response to topical or oral corticosteroids, antihistamines, or oral antibiotics, but partial response to intralesional corticosteroids and infliximab. None of the patients had complete response to any therapies directed at this condition. OFG may be characterized by partial response, often requiring long-term therapy. 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user-menu__item--popup-container user-menu__item--login-popup-container dib"> <button class="user-menu__link user-menu__link--login js-ejp-login-btn" type="button" data-config="{&quot;eventName&quot;:&quot;login&quot;,&quot;eventDetail&quot;:{&quot;source&quot;:&quot;Masthead Dropdown&quot;},&quot;isNull&quot;:false}"> <i class="fa fa-sign-in"></i> <span>Log in</span> </button> </li> <li id="ctl00_ctl51_Header_UserActionTools_liOr" class="dib or-spacing"><span>or</span></li> <li id="ctl00_ctl51_Header_UserActionTools_liRegister2" class="user-menu__item dib"> <a id="ctl00_ctl51_Header_UserActionTools_RegisterUserPopupLnk" class="user-menu__link js-register-link" data-config="{&quot;eventName&quot;:&quot;Register&quot;,&quot;eventDetail&quot;:{&quot;source&quot;:&quot;Masthead Dropdown&quot;},&quot;isNull&quot;:false}" href="https://journals.lww.com/acgcr/pages/register.aspx?ContextUrl=%2facgcr%2ffulltext%2f2024%2f11000%2forofacial_granulomatosis_and_crohn_s_disease__a.13.aspx">Register</a> </li> <li id="ctl00_ctl51_Header_UserActionTools_liIssueAlerts" class="user-menu__item dib"> <i class="fa fa-envelope"></i> <a rel="nofollow" title href="javascript:javascript: alerts_ShowSubscribeeTOCPopup(&#39;ACG Case Reports Journal&#39;,&#39;acgcr&#39;,&#39;eTOC&#39;, &#39;eTOC&#39;);;" onclick="javascript: alerts_ShowSubscribeeTOCPopup(&#39;ACG Case Reports Journal&#39;,&#39;acgcr&#39;,&#39;eTOC&#39;, &#39;eTOC&#39;);" class="user-menu__link"><span id="ctl00_ctl51_Header_UserActionTools_SubscribeToAlertsActionToolLink_defaultText" class="hideOnFixed">Get new issue alerts</span><span id="ctl00_ctl51_Header_UserActionTools_SubscribeToAlertsActionToolLink_stickyText" class="showOnFixed">Get alerts</span></a> </li> </ul> </section> <span id="ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_dummyTargetControl"></span> <div id="ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_pnlunSubscribeToAlerts" style="display: none;"> <div tabindex="-1" role="dialog" class="wk-modal horizontally-centered small wk-modal-with-overlay wk-modal-small ejp-modal"> <div id="ej-box-modal-style-1"> <div id="ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_pnlMain"> <div id="tocUnsubscribeModal" > <section class="wk-modal-content"> <header class="wk-modal-header ejp-modal-header"> <div class="wk-modal-title ejp-modal-title"></div> <button aria-label="close dialog" class="js-close primary-button wk-button-icon wk-modal-close ejp-modal-close "><i class="fa fa-times"></i></button> </header> <div class="wk-modal-body"> <div class="wk-field-item"> <p> <span id="ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_lblUnsubscribeMessage"></span> </p> <div class="wk-button-bar"> <div class="wk-button-group-left"> <p id="ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_pUnsubscribeButtons" Style="display: none;"> <input type="submit" name="ctl00$ctl51$Header$UserActionTools$ucUnsubscribeToAlertsUserControl$btnContinue" value="Yes" onclick="javascript:WebForm_DoPostBackWithOptions(new WebForm_PostBackOptions(&quot;ctl00$ctl51$Header$UserActionTools$ucUnsubscribeToAlertsUserControl$btnContinue&quot;, &quot;&quot;, true, &quot;&quot;, &quot;&quot;, false, false))" id="ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_btnContinue" class="primary-button" /> <input type="button" value="No" id="btnCancel" aria-label="close dialog" class="js-close secondary-button" /> </p> </div> </div> </div> </div> </section> </div> </div> </div> </div> </div> <div id="ej-clear-float"> </div> <input type="hidden" name="ctl00$ctl51$Header$UserActionTools$ucUnsubscribeToAlertsUserControl$hiddenUnSubscribeAlertType" id="ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnSubscribeAlertType" /> <input type="hidden" name="ctl00$ctl51$Header$UserActionTools$ucUnsubscribeToAlertsUserControl$hiddenUnsubscribeJournalId" id="ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnsubscribeJournalId" /> <input type="hidden" name="ctl00$ctl51$Header$UserActionTools$ucUnsubscribeToAlertsUserControl$hiddenUnsubscribeMessage" id="ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnsubscribeMessage" /> <input type="hidden" name="ctl00$ctl51$Header$UserActionTools$ucUnsubscribeToAlertsUserControl$hiddenTopicalCollectionId" id="ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenTopicalCollectionId" /> <input type="hidden" name="ctl00$ctl51$Header$UserActionTools$ucUnsubscribeToAlertsUserControl$hiddenOvidId" id="ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenOvidId" /> <input type="hidden" name="ctl00$ctl51$Header$UserActionTools$ucUnsubscribeToAlertsUserControl$hiddenJournalId" id="ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenJournalId" /> <script type="text/javascript"> function alerts_ShowUnSubscribeeTOCPopup(title, alertType, journalId) { var unSubscribeeTocPopup = $find('unSubscribeToAlerts'); var lblUnsubscribeMessage = document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_lblUnsubscribeMessage'); lblUnsubscribeMessage.innerHTML = title; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnSubscribeAlertType').value = alertType; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnsubscribeMessage').value = title; $('#' + 'ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_pUnsubscribeButtons').show(); if (document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenJournalId').value == null || document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenJournalId').value == '') { document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenJournalId').value = journalId; } if (unSubscribeeTocPopup != null) { unSubscribeeTocPopup.show(); } } function alerts_ShowUnSubscribeeTOCPopup_Root(title, journalId, alertType) { var unSubscribeeTocPopup = $find('unSubscribeToAlerts'); var lblUnsubscribeMessage = document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_lblUnsubscribeMessage'); lblUnsubscribeMessage.innerHTML = title; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnsubscribeMessage').value = title; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnSubscribeAlertType').value = alertType; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnsubscribeJournalId').value = journalId; $('#' + 'ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_pUnsubscribeButtons').show(); if (unSubscribeeTocPopup != null) { unSubscribeeTocPopup.show(); } } function alerts_ShowUnSubscribeeTOCPopup_Citation(title, journalId, ovidId) { var unSubscribeeTocPopup = $find('unSubscribeToAlerts'); var lblUnsubscribeMessage = document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_lblUnsubscribeMessage'); lblUnsubscribeMessage.innerHTML = title; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnsubscribeMessage').value = title; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnSubscribeAlertType').value = 'Citation'; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnsubscribeJournalId').value = journalId; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenOvidId').value = ovidId; $('#' + 'ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_pUnsubscribeButtons').show(); if (unSubscribeeTocPopup != null) { unSubscribeeTocPopup.show(); } } function alerts_ShowUnSubscribeeTOCPopup_Topical(title, journalId, topicalCollectionId) { var unSubscribeeTocPopup = $find('unSubscribeToAlerts'); var lblUnsubscribeMessage = document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_lblUnsubscribeMessage'); lblUnsubscribeMessage.innerHTML = title; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnsubscribeMessage').value = title; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnSubscribeAlertType').value = 'Topical'; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenUnsubscribeJournalId').value = journalId; document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenTopicalCollectionId').value = topicalCollectionId; $('#' + 'ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_pUnsubscribeButtons').show(); if (document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenJournalId').value == null || document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenJournalId').value == '') { document.getElementById('ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_hiddenJournalId').value = journalId; } if (unSubscribeeTocPopup != null) { unSubscribeeTocPopup.show(); } } function alerts_ShowUnSubscribeeTOCSuccessPopup() { var unSubscribeeTocPopup = $find('unSubscribeToAlerts'); if (unSubscribeeTocPopup != null) { unSubscribeeTocPopup.show(); } } $('.js-close').on('click', function() { var unSubscribeeTocPopup = $find('unSubscribeToAlerts'); if (unSubscribeeTocPopup != null) { unSubscribeeTocPopup.hide(); } return false; }); </script> <span id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_dummyTargetControl"></span> <input type="hidden" name="ctl00$ctl51$Header$UserActionTools$SubscribeToEtocPopupControl$hiddenSubscribeToEtocPopupState" id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_hiddenSubscribeToEtocPopupState" value="showemail" /> <input type="hidden" name="ctl00$ctl51$Header$UserActionTools$SubscribeToEtocPopupControl$hiddenConfirmSubscriptionPageUrl" id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_hiddenConfirmSubscriptionPageUrl" /> <input type="hidden" name="ctl00$ctl51$Header$UserActionTools$SubscribeToEtocPopupControl$hiddenJournalId" id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_hiddenJournalId" /> <input type="hidden" name="ctl00$ctl51$Header$UserActionTools$SubscribeToEtocPopupControl$hiddenSubscriptionType" id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_hiddenSubscriptionType" /> <div id="divPopupOverLay" class="Popup-Overlay" style="display: none; position: fixed; left: 0; top: 0; z-index: 3800; width: 1920px; height: 4517px;"></div> <div id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_pnlSubscribeToEtoc" class="etoc-popup ej-box-modal-drop-shadow" style="display: none;"> <div tabindex="-1" role="dialog" class="wk-modal horizontally-centered small wk-modal-with-overlay wk-modal-small ejp-modal"> <div id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_pnlMain" onkeypress="javascript:return WebForm_FireDefaultButton(event, &#39;ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_btnContinue&#39;)"> <section class="wk-modal-content"> <header class="wk-modal-header ejp-modal-header"> <div class="wk-modal-title ejp-modal-title"> <span id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_lblTitle"></span> </div> <button aria-label="close dialog" class="js-close primary-button wk-button-icon wk-modal-close ejp-modal-close "> <i class="fa fa-times"></i> </button> </header> <div class="wk-modal-body"> <div class="wk-field-item"> <div class="wk-field-body"> <strong> <span id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_lblThankYouMessage"></span></strong> </div> <div class="wk-field-body"> <span id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_cvEmailError" style="color:Black;display:none;"> <div class="ej-error-message"> <p class="ej-error-message-icon error-red"> </p> </div> </span> <label for="Email input">Enter your Email address:</label> <div class="wk-field-body"> <input name="ctl00$ctl51$Header$UserActionTools$SubscribeToEtocPopupControl$txtEmailAddress" type="text" id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_txtEmailAddress" class="ej-box-modal-style-1-input" /> </div> </div> <div class="wk-button-bar"> <div class="wk-button-group-left"> <input type="submit" name="ctl00$ctl51$Header$UserActionTools$SubscribeToEtocPopupControl$btnContinue" value="Continue" onclick="javascript:WebForm_DoPostBackWithOptions(new WebForm_PostBackOptions(&quot;ctl00$ctl51$Header$UserActionTools$SubscribeToEtocPopupControl$btnContinue&quot;, &quot;&quot;, true, &quot;&quot;, &quot;&quot;, false, false))" id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_btnContinue" class="primary-button" /> <input type="button" value="Cancel" id="btnCancel" class="js-close secondary-button" /> </div> </div> </div> <div class="wk-field-item"> <div class="wk-field-body"> <a id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_hypPrivacy" href="https://journals.lww.com/acgcr/_layouts/oaks.journals/privacy.aspx" target="_blank">Privacy Policy</a> </div> </div> </div> </section> </div> <div id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_pnlAuthenticatedUser" style="display: none;"> <section class="wk-modal-content"> <header class="wk-modal-header ejp-modal-header"> <div class="wk-modal-title ejp-modal-title"></div> <button aria-label="close dialog" class="js-close primary-button wk-button-icon wk-modal-close ejp-modal-close "><i class="fa fa-times"></i></button> </header> <div class="wk-modal-body"> <div class="wk-field-item"> <p> <strong> <span id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_lblAuthenticatedUserTitle"></span> </strong> </p> </div> <p id="ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_pAuthenticatedUserMessage"> </p> </div> </section> </div> </div> </div> <div id="ej-clear-float"> </div> <script type="text/javascript"> function alerts_SaveSubscriptionTypeInHiddenField(url, subscriptionType, journalId, yearId, issueId, articleId) { if (journalId && yearId && issueId && articleId) { logCounterDataAlertWhenCitedSynchronously(journalId, yearId, issueId, articleId); } $('#' + 'ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_hiddenSubscriptionType').val(subscriptionType); $('#' + 'ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_hiddenJournalId').val(journalId); if (url) window.location = url; else __doPostBack('hypSubscribeToAlerts'); return false; } function alerts_ShowSubscribeeTOCPopup(journalTitle, journalId, subscriptionType, displaySubscriptionType) { $('#' + 'ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_hiddenSubscriptionType').val(subscriptionType); var subscribeToEtocPopup = $find('subscribeToEtoc'); subscribeToEtocPopup.show(); document.getElementsByTagName( 'html' )[0].classList.add('no-scroll'); var modelPopupState = document.getElementById('ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_hiddenSubscribeToEtocPopupState'); var emailTextBox = document.getElementById('ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_txtEmailAddress'); var thanksPanel = document.getElementById('ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_pnlAuthenticatedUser'); var mainPanel = document.getElementById('ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_pnlMain'); var thankYouMessageUnAuthenticated = document.getElementById('ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_lblThankYouMessage'); var thankYouMessageAuthenticated = document.getElementById('ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_lblAuthenticatedUserTitle'); if (!displaySubscriptionType) { displaySubscriptionType = subscriptionType; } if (modelPopupState.value == 'showthanksmessage') { thanksPanel.style.display = "block"; mainPanel.style.display = "none"; thankYouMessageAuthenticated.innerHTML = "Thank you for subscribing to the " + displaySubscriptionType + " for " + journalTitle + "."; } if (modelPopupState.value == 'showemail') { thanksPanel.style.display = "none"; mainPanel.style.display = "block"; thankYouMessageUnAuthenticated.innerHTML = "Thank you for choosing to subscribe to the " + displaySubscriptionType + " for " + journalTitle + "."; } if ($('.etoc-popup') != null) { document.getElementById('ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_hiddenJournalId').value = journalId; if (modelPopupState.value == 'showemail') emailTextBox.focus(); } } $('.js-close').on('click', function () { document.getElementById('ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_txtEmailAddress').value = ""; document.getElementById('ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_cvEmailError').style.display = "none"; var pageUrl = document.getElementById('ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_hiddenConfirmSubscriptionPageUrl'); var subscribeeTocPopup = $find('subscribeToEtoc'); if (subscribeeTocPopup != null) { subscribeeTocPopup.hide(); document.getElementsByTagName( 'html' )[0].classList.remove('no-scroll'); if (pageUrl.value !== '' && pageUrl.value !== window.location.pathname) window.location = pageUrl.value; } return false; }); </script> <div id="ctl00_ctl51_Header_UserActionTools_eTocTopContainer"> <div id="eTocTop" style="display: none;"> <a rel="nofollow" title href="javascript:javascript: alerts_ShowSubscribeeTOCPopup(&#39;ACG Case Reports Journal&#39;,&#39;acgcr&#39;,&#39;eTOC&#39;, &#39;eTOC&#39;);;" onclick="javascript: alerts_ShowSubscribeeTOCPopup(&#39;ACG Case Reports Journal&#39;,&#39;acgcr&#39;,&#39;eTOC&#39;, &#39;eTOC&#39;);"><span id="ctl00_ctl51_Header_UserActionTools_alertLinkControlTop_defaultText" class="hideOnFixed">Subscribe to eTOC</span></a> </div> </div> <script> $(function () { //find recent searches link and move container var recentSearchesControl = $("a[id$='RecentSearches']"); if (recentSearchesControl.length) { recentSearchesControl.click(function (e) { e.stopPropagation(); if ($("#ResentSearchContainer").is(":hidden")) { $("#ResentSearchContainer").slideDown("slow"); recentSearchesControl.addClass('open'); } else { $("#ResentSearchContainer").hide(); recentSearchesControl.removeClass('open'); } }); } var eTocAccountLink = $("ul.js-user-action-toolbar__dropdown>li>a[id='SubscribeToEToc']"); if (eTocAccountLink.length) { eTocAccountLink.parent().html($("#eTocTop>a")); } }); </script> <a class="logo-secondary black" href="http://wolterskluwer.com/" target="_blank"> <h3 class="logo-secondary-text">Secondary Logo</h3> </a> <a href="#" class="sticky-search"><i class="fa fa-search"></i></a> </div> <div class="header__layout-bottom wk-grid-page-container"> <a id="hypJournals" class="logo-main" href="https://journals.lww.com/acgcr/pages/default.aspx"> <h3 class="logo-main-text">Journal Logo </h3> </a> <div class="search-v2"> <div class="search-wrapper"> <select name="ctl00$ctl51$Header$SearchTopBoxControl$ddlContentScope" id="ctl00_ctl51_Header_SearchTopBoxControl_ddlContentScope"> <option value="All">All</option> <option selected="selected" value="Articles">Articles</option> <option value="Images">Images</option> <option value="Videos">Videos</option> <option value="Podcasts">Podcasts</option> <option value="Blogs">Blogs</option> </select> <div class="input-group" id="globalSearch"> <input name="ctl00$ctl51$Header$SearchTopBoxControl$txtKeywords" type="text" value="Search" id="ctl00_ctl51_Header_SearchTopBoxControl_txtKeywords" class="form-control searchText" /> <span class="input-group-btn"> <button id="ctl00_ctl51_Header_SearchTopBoxControl_btnGlobalSearchMagnifier" type="submit" class="btn btn-default"><i class="fa fa-search"></i></button> </span> <input type="hidden" name="ctl00$ctl51$Header$SearchTopBoxControl$IsAutoCompleteEnabled" id="ctl00_ctl51_Header_SearchTopBoxControl_IsAutoCompleteEnabled" value="false" /> <input type="hidden" name="ctl00$ctl51$Header$SearchTopBoxControl$AutocompleteApiUrl" id="ctl00_ctl51_Header_SearchTopBoxControl_AutocompleteApiUrl" value="https://platformservices.wolterskluwerhealth.com/autocomplete/ACGCRJ/shingle/" /> </div> </div> <div class="advance-search" style=""> <a id="ctl00_ctl51_Header_SearchTopBoxControl_lnkAdvanceSearch" href="https://journals.lww.com/acgcr/pages/advancedsearch.aspx">Advanced Search</a> <input type="submit" name="ctl00$ctl51$Header$SearchTopBoxControl$btnGlobalSearch" value="" id="ctl00_ctl51_Header_SearchTopBoxControl_btnGlobalSearch" disabled="disabled" class="aspNetDisabled btnGlobalSearch" style="display: none;" /> <input name="ctl00$ctl51$Header$SearchTopBoxControl$hfldKeywords" type="hidden" id="ctl00_ctl51_Header_SearchTopBoxControl_hfldKeywords" class="hfldKeywords" value="disableButton" /> </div> </div> <script> $(function() { try { var objSearchBox = { btnGlobalSearchMagnifierId: 'ctl00_ctl51_Header_SearchTopBoxControl_btnGlobalSearchMagnifier', searchButtonId: 'ctl00_ctl51_Header_SearchTopBoxControl_btnGlobalSearch', txtKeywordsId: 'ctl00_ctl51_Header_SearchTopBoxControl_txtKeywords', hfldKeywordsHiddenId: 'ctl00_ctl51_Header_SearchTopBoxControl_hfldKeywords', defaultSearchText: 'Search' }; new Searchbox(objSearchBox).Init(); } catch (e) { console.error(e); } }); //autocomplete functionality for basic search //this functionality will call the SBA url and start suggesting after typing 3rd character var textKeywords = null; var hiddenAutocompleteApiUrl = null; var buttonGlobalSearch = null; $(function () { try { var 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Granulomatosis and Crohn\'s Disease: A Case Series', '11162024', 'Siri A. Urquhart, Grace Y. Kim, Katelyn R. Anderson, et al', '11', '11', '\u00A9 2024 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of The American College of Gastroenterology.', '02075970-202411000-00013', '', true, 'CCBY-NC-ND'); } </script> <div id="ArticleContainer"> <article id="ej-article-view" class="js-article-view ej-article-view" xmlns:ssr="http://ssr-eus-go-csi.cloudapp.net/v1/assets" xmlns:mml="http://www.w3.org/1998/Math/MathML" xmlns:xlink="http://www.w3.org/1999/xlink" xmlns:xhtml="http://www.w3.org/1999/xhtml"><section class="ejp-r-article-subsection"><div class="ejp-r-article-subsection__text">CASE REPORT: INFLAMMATORY BOWEL DISEASE</div></section><header class="ejp-article-header"><h1 class="ejp-article-title">Orofacial Granulomatosis and Crohn's Disease: A Case Series</h1></header><section id="ejp-article-authors"><p id="P7"><i class="wki icon-orcid" onclick="javascript:ToolTip_Orcid('Urquhart, Siri A. MD', 'https://orcid.org/0000-0002-2337-0480');" onmouseover="javascript:ToolTip_Orcid('Urquhart, Siri A. MD', 'https://orcid.org/0000-0002-2337-0480');"></i>Urquhart, Siri A. MD<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">1</sup>; Kim, Grace Y. MD<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">2</sup>; Anderson, Katelyn R. MD<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">2</sup>; Chedid, Victor G. MD, MS<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">1</sup></p><section><a href="#" id="ejp-article-authors-link"> Author Information <i class="wk-icon-arrow-down-r"></i></a></section></section><section id="ejp-article-authors-info" class="ejp-article-authors-info ejp-article-authors-info--hidden"><div class="ejp-article-authors-info-holder"> <p id=""><sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">1</sup>Division of Gastroenterology and Hepatology, Mayo Clinic, Rochester, MN</p> <p id=""><sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">2</sup>Department of Dermatology, Mayo Clinic, Rochester, MN</p> <p id="cor1"><strong xmlns:mrws="http://webservices.ovid.com/mrws/1.0">Correspondence:</strong> Siri A. Urquhart, MD (<span><a href="/cdn-cgi/l/email-protection#e489858d88908bde919695918c859690ca978d968da489859d8bca818091" onclick="javascript:window.open('mailto:mailto:urquhart.siri@mayo.edu');return false"><span class="__cf_email__" data-cfemail="f7828586829f968583d9849e859eb79a968e98d9929382">[email&#160;protected]</span></a></span>).</p> <p id="">This is an open access article distributed under the terms of the <span><a href="http://creativecommons.org/licenses/by-nc-nd/4.0/" onclick="javascript:window.open('http://creativecommons.org/licenses/by-nc-nd/4.0/');return false">Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND)</a></span>, where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal.</p> </div></section><section id="article-journal-info"><div class="ej-journal-info"><span class="ej-journal-name">ACG Case Reports Journal </span><span id="ej-journal-date-volume-issue-pg">11(11):p e01559, November 2024.</span><span> | </span><span class="ej-journal-doi">DOI: </span>10.14309/crj.0000000000001559<a id="ej-article-indicators-ccl" onmouseover="javascript:Tooltip_CCL('This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal.')" onmouseout="javascript:Tooltip_MouseOut()" href="http://creativecommons.org/licenses/by-nc-nd/4.0/" target="_blank"><span class="ejp-article-indicators__ccl"></span></a></div></section><section id="ejp-article-indicators" class="ejp-article-indicators"><ul class="ejp-article-indicators__list"> <li id="ej-article-indicators-open" class="ejp-article-indicators__list-item" onmouseover="javascript:Tooltip_Open()" onmouseout="javascript:Tooltip_MouseOut()"><span class="ejp-indicator">Open</span></li> </ul><button id="openArticleMetricsPopupButton" type="button" class="wk-button wk-button-ghost wk-button-icon-left ejp-article-metrics-btn" data-articleMetricsEndpoint="//services.journals.lww.com/IntegrationServices/ArticleMetricsService.svc" data-articleMetricsDetailsUrl="https://wolterskluwer.altmetric.com/details/" data-doi="10.14309/crj.0000000000001559" data-suiteId="vrs_woltere_journalsglobalvrs" data-articlePublicationDate="2024-11-16" data-articlePublicationDateString="November 16, 2024" data-receivedDate="2024-09-09" data-revisionReceivedDate="" data-acceptedDate="2024-10-17" data-crossrefRefLinkRoot="https://doi.org/" data-medlineRefLinkRoot="http://www.ncbi.nlm.nih.gov/pubmed/" data-pubMedId=""><i class="wk-icon-filled-chart-r"></i><i class="wk-icon-filled-chart-white-r"></i> Metrics </button></section><section class="ejp-article-tabs" id="abstractWrap"><h2 class="ejp-article-tabs-heading">Abstract</h2> <div class="ejp-article-tabs__tabpanel" id="panel1" role="tabpanel" aria-labelledby="tab1"> <div id="article-abstract-content1"> <div class="ejp-article-text-abstract"> <p>Orofacial granulomatosis (OFG) is a rare syndrome that can occur in association with Crohn's disease (CD). The electronic medical record was searched for “OFG” and “CD.” A total of 297 patients were identified, and relevant data were abstracted. Five patients met inclusion criteria. Most (80%) had ileocolonic CD with nonstricturing/nonpenetrating phenotype. The most common treatments included intralesional corticosteroids (4 patients), oral corticosteroids (3 patients), and anti-TNF agents, specifically infliximab (3 patients). Intralesional corticosteroid administration was followed by partial response in 3 patients, but recurrences were common following initial injection. One patient had no response to topical or oral corticosteroids, antihistamines, or oral antibiotics, but partial response to intralesional corticosteroids and infliximab. None of the patients had complete response to any therapies directed at this condition. OFG may be characterized by partial response, often requiring long-term therapy. Additional investigations into novel treatments may improve future clinical outcomes.</p> </div> </div> </div></section><div class="ejp-fulltext-content js-ejp-fulltext-content"><section id="ArticleBody"><h2 class="ejp-article-outline-heading" data-level="1" id="s1">INTRODUCTION</h2> <p id="JCL-P-1">Inflammatory bowel disease (IBD), specifically Crohn's disease (CD), is a systemic condition that manifests not only in the gastrointestinal (GI) tract but also in extraintestinal organs in many patients. The quality of life for patients with IBD can be substantially affected by these extraintestinal manifestations (EIMs), which can occur in up to 24% of patients with IBD before the onset of intestinal symptoms.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R1">1</a></sup></sup> Cutaneous EIMs have been reported in 5%–15% of patients with IBD.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R2">2</a></sup></sup> It may be disputed whether oral lesions represent an EIM or are rather a manifestation of IBD. The prevalence of oral lesions is reported in a range from 5% to 50%.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R3 R4 R5">3–5</a></sup></sup> Oral lesions are reported to be more common in patients with CD and more prevalent in children.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R1">1</a></sup></sup></p> <p id="JCL-P-2">Orofacial granulomatosis (OFG) is an uncommon condition characterized by the histologic finding of noncaseating granulomas within the face and oral mucosa on tissue biopsy, resulting in persistent swelling.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R6 R7 R8">6–8</a></sup></sup> OFG is found in less than 1% of patients with CD, and biopsy is the most effective means of establishing the diagnosis.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R8 R9">8,9</a></sup></sup> Some may consider OFG a distinct condition without a systemic granulomatous disorder, while others consider this to be a broader diagnosis encompassing isolated OFG and a possible manifestation of CD or sarcoidosis. OFG often follows a chronic and disfiguring course, making it exceptionally difficult to manage and ultimately affecting the quality of life in affected patients.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R8 R9">8,9</a></sup></sup></p> <p id="JCL-P-3">OFG has been described in the literature as an independent entity and a subtype of CD. In a systematic review from Gavioli et al, OFG often presented as a distinct, independent condition, but carried a higher risk of developing CD, especially in the pediatric population.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R10">10</a></sup></sup> OFG may occur before CD and those with concurrent CD may be more likely to have buccal sulcus involvement. In a systematic review from Lazzerini et al, there is a high prevalence of CD in pediatric patients with OFG, with 40.4% diagnosed with both conditions. OFG may therefore be a subtype of CD as OFG often precedes the diagnosis of CD. Perianal disease and family history of CD were significantly associated with a higher risk of CD in pediatric patients with OFG.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R11">11</a></sup></sup> Patients with CD and coexistent OFG may also have a distinct phenotype characterized by a higher incidence of perianal disease, intestinal granulomas, and greater need for extensive therapy including perianal surgery and combination immunomodulatory treatments to achieve remission.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R12 R13">12,13</a></sup></sup></p> <p id="JCL-P-4">OFG and CD share several pathophysiologic features, suggesting a potential relationship between these 2 conditions. One key aspect of the pathophysiologic relationship is the immune environment. Freysdottir et al described a Th1-dominant immune response in OFG tissue, similar to that observed in CD. Specifically, increased levels of CD4<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">+</sup> T cell, interferon gamma, and other Th1 cytokines have been noted in OFG, mirroring the immunopathological features of CD.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R14">14</a></sup></sup> Genetic factors also play a significant role. Variants in NOD2 gene, which are associated with CD, have been found in patients with OFG, particularly in those with concurrent intestinal disease.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R15">15</a></sup></sup></p> <p id="JCL-P-5">Given the rarity of this syndrome and discrepancies in the literature as to if OFG is an independent clinical entity or a subtype of CD with a distinct phenotype, we aimed to retrospectively review our institutional experience to better describe the clinical features, presentation, and outcomes of OFG in patients with CD.</p> <h2 class="ejp-article-outline-heading" data-level="1" id="s2">METHODS</h2> <h3 class="ejp-article-outline-heading" data-level="2" id="s2-1">Patient population</h3> <p id="JCL-P-6">This retrospective study was approved by the Mayo Clinic Institutional Review Board. All patients were identified based on a search of <em xmlns:mrws="http://webservices.ovid.com/mrws/1.0">International Classification of Diseases-9/10</em> diagnostic codes and clinical notes search through an institutional bioinformatic tool to include diagnoses of OFG (including granulomatous cheilitis, cheilitis granulomatosa, and Melkersson-Rosenthal syndrome) and CD between January 1992 and May 2023. This was followed by manual review of individual patient charts as permitted by Minnesota Health Records Act, 144.295, which allows patients to opt out of retrospective research.</p> <p id="JCL-P-7">In patients with a confirmed OFG and CD diagnosis, relevant demographic and clinical outcomes were abstracted. The date of first visit for CD and/or OFG diagnosis (index) was recorded. We excluded patients without confirmed OFG or CD diagnosis on record, absence of pathology report on file confirming OFG, and absence of endoscopic procedure on file confirming CD diagnosis.</p> <h3 class="ejp-article-outline-heading" data-level="2" id="s2-2">Crohn's disease characteristics</h3> <p id="JCL-P-8">CD was diagnosed based on clinical diagnostic criteria as per the treating gastroenterologist and review of the medical record for the confirmation of characteristic endoscopic, radiographic, and/or histologic findings. The date of CD diagnosis was defined by the earliest pathologic confirmation at our institution. Related complications such as upper GI involvement or perianal disease and former or current tobacco use were abstracted. CD characteristics included extent of disease involvement, disease behavior, and prior medical therapies trialed for CD.</p> <h3 class="ejp-article-outline-heading" data-level="2" id="s2-3">OFG characteristics</h3> <p id="JCL-P-9">OFG was diagnosed based on the presence of typical clinical features including lip erythema, edema, and/or ulceration of oral mucosa in addition to histologic features consistent with the diagnosis including lymphocytic inflammatory infiltrate with noncaseating (non-necrotizing) granulomas. The date of OFG diagnosis was defined by the earliest pathologic confirmation at our institution. Prior treatment strategies for OFG were recorded including treatment duration, patient response, and time to response.</p> <h3 class="ejp-article-outline-heading" data-level="2" id="s2-4">Statistical analysis</h3> <p id="JCL-P-10">Patient characteristics and clinical data were presented as mean with SD, median with range, or frequency with percentages. Descriptive statistics were used to report findings. Categorical variables were reported as a unique count and percentage of the sample.</p> <h2 class="ejp-article-outline-heading" data-level="1" id="s3">RESULTS</h2> <p id="JCL-P-11">The initial data search identified 297 patients with suspected cases of OFG and CD. After manual review, 5 patients were included in the final analysis, and 292 were excluded (<a href="javascript:void(0)" onclick="javascript:showSlideShowByImageID('F1', '02075970-202411000-00013');">Figure 1</a>).</p><section class="ejp-r-article-images"><figure class="ejp-r-article-images__figure"><a href="javascript:void(0)" class="ejp-r-article-images__image-link" onclick="showSlideShowByImageID('F1', '02075970-202411000-00013')"><img class="ejp-r-article-images__img js-lazy-load lazy-load" src="javascript:void(0);" data-src="https://images.journals.lww.com/acgcr/ArticleViewerPreview.02075970-202411000-00013.F1.jpeg" data-srcset="https://images.journals.lww.com/acgcr/ArticleViewerPreview@2.02075970-202411000-00013.F1.jpeg 2x" srcset="" alt="F1"></a><figcaption class="ejp-r-article-images__figcaption"><a href="javascript:void(0)" class="ejp-r-article-images__figcaption-link" onclick="showSlideShowByImageID('F1', '02075970-202411000-00013')">Figure 1.: </a><div class="ejp-r-article-images__figcaption-text">Screening of patients for study inclusion. Using an institutional bioinformatics search tool, all patients were identified using a combination of <em xmlns:mrws="http://webservices.ovid.com/mrws/1.0">International Classification of Diseases-9/ICD-10</em> codes and search terms including “orofacial granulomatosis,” “granulomatous cheilitis,” “cheilitis granulomatosa,” “Melkersson-Rosenthal syndrome,” and “Crohn's disease” who had a valid research authorization on file. A total of 292 patients were excluded.</div></figcaption></figure></section><h3 class="ejp-article-outline-heading" data-level="2" id="s3-1">Crohn's disease characteristics</h3> <p id="JCL-P-12">Five patients met inclusion criteria (40% female), with a median age of 15 years at the time of CD diagnosis. Most (80%) had ileocolonic distribution of CD. Two (40%) had upper GI involvement, and 1 (20%) had perianal involvement. Nonstricturing/penetrating (80%) was the most common CD phenotype. OFG was diagnosed in 1 (20%) before CD, 2 (40%) concurrently with CD, and 2 (40%) after CD diagnosis. At the time of OFG diagnosis, CD was active in 2 (40%) and quiescent in 2 patients (40%).</p> <p id="JCL-P-13">Previous treatments of CD included 5-aminosalicylate only (20%), oral corticosteroids (20%), anti-tumor necrosis factor (anti-TNF) +/− immunomodulator (60%), and ustekinumab (20%). Only one patient had prior surgical resection for CD (<a href="#T1">Table 1</a>).</p> <div class="wk-clearfix-article-table"></div> <div class="article-table"><a id="T1"></a><div class="article-table-title"><span>Table 1. - </span> Demographic and clinical characteristics of patients with Crohn's disease and orofacial granulomatosis<em class="fa fa-arrows-alt expand-button js-expand-table-event" aria-label="expand table"></em><div class="clearfix"></div> </div> <div class="scroll-message"></div> <div class="dummy-scroll"> <div></div> </div> <div class="article-table-body"> <div class="article-table-container"> <table> <thead xmlns="http://www.w3.org/1999/xhtml"> <tr> <td rowspan="1" colspan="1">Characteristic</td> <td rowspan="1" colspan="1">Patients (N = 5)</td> </tr> </thead> <tbody xmlns="http://www.w3.org/1999/xhtml"> <tr> <td rowspan="1" colspan="1">Age, median (range), y</td> <td rowspan="1" colspan="1" /> </tr> <tr> <td rowspan="1" colspan="1"> At CD diagnosis</td> <td rowspan="1" colspan="1">15 (9–26)</td> </tr> <tr> <td rowspan="1" colspan="1"> At OFG diagnosis</td> <td rowspan="1" colspan="1">14 (9–59)</td> </tr> <tr> <td rowspan="1" colspan="1">Sex, female to male ratio</td> <td rowspan="1" colspan="1">2:3</td> </tr> <tr> <td rowspan="1" colspan="1">CD distribution, n (%)</td> <td rowspan="1" colspan="1" /> </tr> <tr> <td rowspan="1" colspan="1"> Ileocolonic</td> <td rowspan="1" colspan="1">4 (80.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Ileal</td> <td rowspan="1" colspan="1">1 (20.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Colonic, isolated</td> <td rowspan="1" colspan="1">0 (0.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Upper GI involvement</td> <td rowspan="1" colspan="1">2 (40.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Perianal involvement</td> <td rowspan="1" colspan="1">1 (20.0)</td> </tr> <tr> <td rowspan="1" colspan="1">CD phenotype, n (%)</td> <td rowspan="1" colspan="1" /> </tr> <tr> <td rowspan="1" colspan="1"> Nonstricturing/penetrating</td> <td rowspan="1" colspan="1">4 (80.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Stricturing</td> <td rowspan="1" colspan="1">0 (0.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Penetrating</td> <td rowspan="1" colspan="1">1 (20.0)</td> </tr> <tr> <td rowspan="1" colspan="1">Tobacco use history, n (%)</td> <td rowspan="1" colspan="1" /> </tr> <tr> <td rowspan="1" colspan="1"> Current or former</td> <td rowspan="1" colspan="1">0 (0.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Never</td> <td rowspan="1" colspan="1">5 (100.0)</td> </tr> <tr> <td rowspan="1" colspan="1">Timing of OFG diagnosis in relation to CD diagnosis, n (%)<sup><sup xmlns=""><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="tblfn1">a</a></sup></sup> </td> <td rowspan="1" colspan="1" /> </tr> <tr> <td rowspan="1" colspan="1"> Before CD diagnosis</td> <td rowspan="1" colspan="1">1 (20.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Concurrently with CD diagnosis</td> <td rowspan="1" colspan="1">2 (40.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> After CD diagnosis</td> <td rowspan="1" colspan="1">2 (40.0)</td> </tr> <tr> <td rowspan="1" colspan="1">CD activity at the time of OFG diagnosis, n (%)</td> <td rowspan="1" colspan="1" /> </tr> <tr> <td rowspan="1" colspan="1"> Active</td> <td rowspan="1" colspan="1">2 (40.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Quiescent</td> <td rowspan="1" colspan="1">2 (40.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> CD not yet diagnosed</td> <td rowspan="1" colspan="1">1 (20.0)</td> </tr> <tr> <td rowspan="1" colspan="1">OFG diagnosis, n (%)</td> <td rowspan="1" colspan="1" /> </tr> <tr> <td rowspan="1" colspan="1"> Clinically</td> <td rowspan="1" colspan="1">5 (100.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Granulomas on histology</td> <td rowspan="1" colspan="1">5 (100.0)</td> </tr> <tr> <td rowspan="1" colspan="1">Clinical presentation associated with OFG, ratio to total</td> <td rowspan="1" colspan="1" /> </tr> <tr> <td rowspan="1" colspan="1"> Erythema</td> <td rowspan="1" colspan="1">1 (20.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Lip edema</td> <td rowspan="1" colspan="1">4 (80.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Ulceration of oral mucosa</td> <td rowspan="1" colspan="1">1 (20.0)</td> </tr> <tr> <td rowspan="1" colspan="1">Previous treatments for luminal CD, n (%)</td> <td rowspan="1" colspan="1" /> </tr> <tr> <td rowspan="1" colspan="1"> No medications</td> <td rowspan="1" colspan="1">1 (20.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> 5-ASA only</td> <td rowspan="1" colspan="1">1 (20.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Oral corticosteroids</td> <td rowspan="1" colspan="1">1 (20.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Anti-TNF +/− immunomodulator<sup><sup xmlns=""><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="tblfn2">b</a></sup></sup> </td> <td rowspan="1" colspan="1">3 (60.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Ustekinumab</td> <td rowspan="1" colspan="1">1 (20.0)</td> </tr> <tr> <td rowspan="1" colspan="1"> Previous surgical resection<sup><sup xmlns=""><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="tblfn3">c</a></sup></sup> </td> <td rowspan="1" colspan="1">1 (20.0)</td> </tr> </tbody> </table> <div class="article-table-descriptions"> <div class="article-table-descriptions-content"><span id=""><div class="dib">Percentages were calculated on the basis of those with data available.</div></span><span id=""><div class="dib">5-ASA, 5-aminosalicylate; anti-TNF, anti-tumor necrosis factor; CD, Crohn's disease; GI, gastrointestinal; OFG, orofacial granulomatosis.</div></span><span id="tblfn1"><div class="dib"><sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">a</sup>In the patient with a diagnosis of OFG before CD, duration between diagnoses was 24 months. In the 2 patients with a diagnosis of OFG after CD, duration between diagnoses was 22 months to 39 years.</div></span><span id="tblfn2"><div class="dib"><sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">b</sup>Anti-TNF agents included infliximab and adalimumab. Immunomodulator agents included methotrexate.</div></span><span id="tblfn3"><div class="dib"><sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">c</sup>One patient underwent ileal pouch-anal anastomosis 10 years after CD diagnosis.</div></span></div> </div> </div> </div> </div><br><h3 class="ejp-article-outline-heading" data-level="2" id="s3-2">OFG characteristics</h3> <p id="JCL-P-14">The median age was 14 years at the time of OFG diagnosis. OFG was confirmed histologically in all patients. The most common presenting symptom associated with OFG was lip swelling (80%) (<a href="#T1">Table 1</a>).</p> <p id="JCL-P-15">The most common treatment strategies for OFG included intralesional corticosteroids (4 patients), oral corticosteroids (3 patients), and anti-TNF agents, specifically infliximab (3 patients). Although intralesional corticosteroid administration was followed by partial response in 3 patients, recurrences were common after the initial injection. One patient had no response to topical corticosteroids, oral corticosteroids, antihistamines, or oral antibiotics, but partial response to intralesional corticosteroids and infliximab. None of the patients had complete response to any therapies directed at OFG (<a href="#T2">Table 2</a>).</p> <div class="wk-clearfix-article-table"></div> <div class="article-table"><a id="T2"></a><div class="article-table-title"><span>Table 2. - </span> Treatments and responses in 5 patients with Crohn's disease and orofacial granulomatosis<em class="fa fa-arrows-alt expand-button js-expand-table-event" aria-label="expand table"></em><div class="clearfix"></div> </div> <div class="scroll-message"></div> <div class="dummy-scroll"> <div></div> </div> <div class="article-table-body"> <div class="article-table-container"> <table> <thead xmlns="http://www.w3.org/1999/xhtml"> <tr> <td rowspan="2" colspan="1">Treatment</td> <td rowspan="2" colspan="1">No. of patients</td> <td rowspan="2" colspan="1">Treatment duration, median (range), mo</td> <td colspan="3" rowspan="1">Response</td> <td rowspan="2" colspan="1">Time to response, median (range), mo</td> </tr> <tr> <td rowspan="1" colspan="1">NR<sup><sup xmlns=""><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="tblfn4">a</a></sup></sup> </td> <td rowspan="1" colspan="1">PR<sup><sup xmlns=""><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="tblfn5">b</a></sup></sup> </td> <td rowspan="1" colspan="1">CR<sup><sup xmlns=""><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="tblfn6">c</a></sup></sup> </td> </tr> </thead> <tbody xmlns="http://www.w3.org/1999/xhtml"> <tr> <td rowspan="1" colspan="1">Topical tacrolimus</td> <td align="char" char="." rowspan="1" colspan="1">1</td> <td rowspan="1" colspan="1">30</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td align="char" char="." rowspan="1" colspan="1">1</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td rowspan="1" colspan="1">2</td> </tr> <tr> <td rowspan="1" colspan="1">Topical corticosteroids</td> <td align="char" char="." rowspan="1" colspan="1">1</td> <td rowspan="1" colspan="1">N/A</td> <td align="char" char="." rowspan="1" colspan="1">1</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td rowspan="1" colspan="1">N/A</td> </tr> <tr> <td rowspan="1" colspan="1">Intralesional corticosteroids</td> <td align="char" char="." rowspan="1" colspan="1">4</td> <td rowspan="1" colspan="1">14 (2–21)</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td align="char" char="." rowspan="1" colspan="1">3</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td rowspan="1" colspan="1">1 (27 d to 2 mo)</td> </tr> <tr> <td rowspan="1" colspan="1">Oral corticosteroids</td> <td align="char" char="." rowspan="1" colspan="1">3</td> <td rowspan="1" colspan="1">2</td> <td align="char" char="." rowspan="1" colspan="1">1</td> <td align="char" char="." rowspan="1" colspan="1">2</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td rowspan="1" colspan="1">16 d</td> </tr> <tr> <td rowspan="1" colspan="1">Antihistamines</td> <td align="char" char="." rowspan="1" colspan="1">1</td> <td rowspan="1" colspan="1">N/A</td> <td align="char" char="." rowspan="1" colspan="1">1</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td rowspan="1" colspan="1">N/A</td> </tr> <tr> <td rowspan="1" colspan="1">Oral antibiotics</td> <td align="char" char="." rowspan="1" colspan="1">2</td> <td rowspan="1" colspan="1">3</td> <td align="char" char="." rowspan="1" colspan="1">2</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td rowspan="1" colspan="1">3</td> </tr> <tr> <td rowspan="1" colspan="1">Anti-TNF<sup><sup xmlns=""><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="tblfn7">d</a></sup></sup> </td> <td align="char" char="." rowspan="1" colspan="1">3</td> <td rowspan="1" colspan="1">10 (7–29)</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td align="char" char="." rowspan="1" colspan="1">3</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td rowspan="1" colspan="1">7 (2–10)</td> </tr> <tr> <td rowspan="1" colspan="1">Dapsone</td> <td align="char" char="." rowspan="1" colspan="1">1</td> <td rowspan="1" colspan="1">6</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td align="char" char="." rowspan="1" colspan="1">1</td> <td align="char" char="." rowspan="1" colspan="1">0</td> <td rowspan="1" colspan="1">3</td> </tr> </tbody> </table> <div class="article-table-descriptions"> <div class="article-table-descriptions-content"><span id=""><div class="dib">Treatment duration and time to response were calculated on the basis of those with data available.</div></span><span id=""><div class="dib">anti-TNF, anti-tumor necrosis factor; CR, complete response; N/A, not applicable; NR, no response; PR, partial response.</div></span><span id="tblfn4"><div class="dib"><sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">a</sup>No response was defined by the terms no response, recurrence, poor, weak, worsened, and/or no change.</div></span><span id="tblfn5"><div class="dib"><sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">b</sup>Partial response was defined by the terms favorable response, responding, decreased swelling, controlled, improved, much improved, greatly improved, and/or markedly better.</div></span><span id="tblfn6"><div class="dib"><sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">c</sup>Complete response was defined by the terms improved completely, no ongoing symptoms, not active, controlled with no recurrence, cleared, asymptomatic, resolved, largely resolved, complete control, clinical remission, and/or normal.</div></span><span id="tblfn7"><div class="dib"><sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0">d</sup>All 3 patients who received anti-TNF therapy received infliximab.</div></span></div> </div> </div> </div> </div><br><h2 class="ejp-article-outline-heading" data-level="1" id="s4">DISCUSSION</h2> <p id="JCL-P-16">While aphthous ulcerations of the mouth are well-recognized manifestations of active disease, OFG is a rare syndrome that may occur in association with CD.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R7">7</a></sup></sup> In our study, OFG associated with CD may occur before, concurrently with, or after the diagnosis of CD and most often occurs in younger patients. Ileocolonic distribution of CD is a common association, in addition to the presence of upper GI involvement and perianal disease.</p> <p id="JCL-P-17">The treatment of OFG in patients with CD involves a combination of therapies targeting both the oral and GI manifestations of the disease. Given the shared pathophysiologic mechanisms, treatment strategies often overlap but require tailored approaches based on the severity and specific manifestations of each condition. Spontaneous remission is rare, and with lack of controlled trials, different therapeutic approaches have been used for treatment. Several drugs have been used with variable results, and there is no consensus regarding optimal treatment.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R16 R17">16,17</a></sup></sup></p> <p id="JCL-P-18">First-line treatments for OFG typically include topical and systemic corticosteroids. However, these often provide only short-term relief and are not suitable for long-term management due to potential side effects.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R18">18</a></sup></sup> Intralesional corticosteroids, such as triamcinolone acetonide, have shown long-term effectiveness in reducing orofacial swelling and maintaining remission in OFG. In a study by Mignogna et al, 57.9% of patients achieve complete clinical remission with intralesional triamcinolone acetonide alone, while those partially responsive to triamcinolone acetonide achieved remission with the addition of topical pimecrolimus.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R19">19</a></sup></sup> Fedele et al also reported 63.6% of patients did not experience disease recurrence after one course of triamcinolone acetonide, with mean disease-free period of 28.9 months.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R20">20</a></sup></sup> However, systemic corticosteroids are generally used as a bridge to more sustainable therapies.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R18 R19 R20 R21">18–21</a></sup></sup></p> <p id="JCL-P-19">Immunosuppressive agents such as azathioprine have shown the efficacy, particularly in patients with concurrent OFG and CD. Mentzer et al reported 59% of patients with CD, and OFG responded to azathioprine at 12 months, compared with 21% of those with OFG alone.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R22">22</a></sup></sup> The use of immunomodulators in combination with biologics is often necessary for severe or refractory diseases. Malmquist et al found 36% of patients with OFG and CD were on combination therapy at the end of a 5-year observation period, indicating a need for extensive therapy to manage these concomitant conditions.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R12">12</a></sup></sup></p> <p id="JCL-P-20">Biologic therapies have shown significant efficacy in managing both OFG and CD. Anti-TNF agents, such as infliximab and adalimumab, are commonly used and have been effective in inducing remission in patients with both conditions. In a multicenter case series supported by the European Crohn's and Colitis Organization, anti-TNF agents were used successfully in 9 patients with OFG associated with CD.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R13 R23">13,23</a></sup></sup> Infliximab has been reported to have a 71% short-term response rate, but a significant proportion of patients may lose response over time.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R24">24</a></sup></sup> Other biologics, including vedolizumab and ustekinumab, have also been used successfully in some cases. Phillips et al noted remission was achieved in 23 of 28 patients using various biologics, including anti-TNFs, vedolizumab, and ustekinumab.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R13">13</a></sup></sup> The American Gastroenterological Association recommends anti-TNF agents for treating severely active CD, which can include OFG manifestations.<sup xmlns:mrws="http://webservices.ovid.com/mrws/1.0"><sup><a class="ejp-citation-link js-ejp-citation-link" data-reference-links="R25">25</a></sup></sup></p> <p id="JCL-P-21">In our study, patients required similar treatment modalities in comparison with those as described in the literature. These included intralesional corticosteroids (4 patients), oral corticosteroids (3 patients), and anti-TNF agents, specifically infliximab (3 patients). Although intralesional corticosteroid administration was followed by partial response in 3 patients, recurrences were common after the initial injection. One patient had no response to topical corticosteroids, oral corticosteroids, antihistamines, or oral antibiotics, but partial response to intralesional corticosteroids and infliximab. None of the patients had complete response to any therapies directed at OFG. This may reflect inherent limitations in our study, including it being performed at a single tertiary referral center, which may result in bias, limiting generalizability. This was also a retrospective study, and as a result, there was significant reliance on documentation within the electronic medical record. However, we used a large patient database and identified all possible patients with OFG and CD at our institution.</p> <p id="JCL-P-22">Despite the availability of different treatment modalities, the management of OFG remains challenging. None of the patients in this series achieved complete remission, highlighting the need for ongoing management and patient counseling regarding partial responses. Future research into novel therapies is essential to improve outcomes for OFG in CD.</p> <h2 class="ejp-article-outline-heading" data-level="1" id="s5">DISCLOSURES</h2> <p id="JCL-P-23">Author contributions: SA Urquhart: literature search, data collection and interpretation, formation of figures and tables, and drafting of manuscript. GY Kim: data analysis and interpretation, and critical review of manuscript. KR Anderson: data analysis and interpretation, and critical review of manuscript. VG Chedid: literature search, study design, data interpretation, and critical review of manuscript, and is the article guarantor.</p> <p id="JCL-P-24">Financial disclosure: None to report.</p> <p id="JCL-P-25">Previous presentation: This study was presented at the Advances in Inflammatory Bowel Disease conference; December 14, 2023; Orlando, Florida.</p> <p id="JCL-P-26">Informed consent could not be obtained for this case report.</p></section><section id="article-references" class="article-references js-article-references"><h2 class="ejp-article-outline-heading" data-level="1" id="JCL-L-0">REFERENCES</h2> <div id="R1" class="article-references__item js-article-reference">1. Rogler G, Singh A, Kavanaugh A, Rubin DT. Extraintestinal manifestations of inflammatory bowel disease: Current concepts, treatment, and implications for disease management. 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To keep this Web Part, click Cancel.';var lblColleaguesEmail = $get('ctl00_ctl29_g_c168c423_9055_40ab_abd6_309bb94ef67f_ctl00_ucArticleTools_emailToColleagueControl_lblColleaguesEmail'); var Page_ValidationActive = false; if (typeof(ValidatorOnLoad) == "function") { ValidatorOnLoad(); } function ValidatorOnSubmit() { if (Page_ValidationActive) { return ValidatorCommonOnSubmit(); } else { return true; } } //]]> </script> <script type='text/javascript'>SP.SOD.set_prefetch(0);</script> <script type="text/javascript"> //<![CDATA[ WPSC.Init(document); var varPartWPQ1 = WPSC.WebPartPage.Parts.Register('WPQ1','bcb44f38-efa3-4ed6-ac8b-6cab786dbcc2',document.getElementById('WebPartWPQ1')); var varPartWPQ2 = WPSC.WebPartPage.Parts.Register('WPQ2','fdd90111-81d0-4047-974e-23626b17d1fc',document.getElementById('WebPartWPQ2')); var varPartWPQ3 = WPSC.WebPartPage.Parts.Register('WPQ3','af3d8d3d-a08b-4f7b-ab76-45691226be72',document.getElementById('WebPartWPQ3')); var varPartWPQ4 = WPSC.WebPartPage.Parts.Register('WPQ4','a734cb32-fafa-4681-8bc5-f8bed7a521c9',document.getElementById('WebPartWPQ4')); var varPartWPQ5 = WPSC.WebPartPage.Parts.Register('WPQ5','a8bc5309-7129-453a-8924-805a4edc1f60',document.getElementById('WebPartWPQ5')); var varPartWPQ6 = WPSC.WebPartPage.Parts.Register('WPQ6','a06f7896-92e8-4b52-916e-d4f46ebca7ea',document.getElementById('WebPartWPQ6')); WPSC.WebPartPage.WebURL = 'https:\u002f\u002fjournals.lww.com\u002facgcr'; WPSC.WebPartPage.WebServerRelativeURL = '\u002facgcr'; var g_clientIdDeltaPlaceHolderMain = "DeltaPlaceHolderMain"; var g_clientIdDeltaPlaceHolderUtilityContent = "DeltaPlaceHolderUtilityContent"; WebForm_AutoFocus('ctl00_ctl29_g_c168c423_9055_40ab_abd6_309bb94ef67f_ctl00_emailToColleague_txtYourName');Sys.Application.add_init(function() { $create(AjaxControlToolkit.ModalPopupBehavior, {"BackgroundCssClass":"Popup-Overlay","DropShadow":true,"PopupControlID":"ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_pnlunSubscribeToAlerts","dynamicServicePath":"/acgcr/pages/articleviewer.aspx","id":"unSubscribeToAlerts"}, null, null, $get("ctl00_ctl51_Header_UserActionTools_ucUnsubscribeToAlertsUserControl_dummyTargetControl")); }); Sys.Application.add_init(function() { $create(AjaxControlToolkit.ModalPopupBehavior, {"BackgroundCssClass":"Popup-Overlay","DropShadow":true,"PopupControlID":"ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_pnlSubscribeToEtoc","dynamicServicePath":"/acgcr/pages/articleviewer.aspx","id":"subscribeToEtoc"}, null, null, $get("ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_dummyTargetControl")); }); document.getElementById('ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_cvEmailError').dispose = function() { Array.remove(Page_Validators, document.getElementById('ctl00_ctl51_Header_UserActionTools_SubscribeToEtocPopupControl_cvEmailError')); } g_QuickLaunchControlIds.push("zz1_TopNavigationMenu");_spBodyOnLoadFunctionNames.push('QuickLaunchInitDroppable'); var g_zz1_TopNavigationMenu = null; function init_zz1_TopNavigationMenu() { if (g_zz1_TopNavigationMenu == null) g_zz1_TopNavigationMenu = $create(SP.UI.AspMenu, null, null, null, $get('zz1_TopNavigationMenu')); } if(g_spPreFetchKeys) {{ g_spPreFetchKeys.push('SP.Core.js');}}EnsureScriptFunc('SP.Core.js', 'SP.UI.AspMenu', init_zz1_TopNavigationMenu); Sys.Application.add_init(function() { $create(AjaxControlToolkit.ModalPopupBehavior, {"BackgroundCssClass":"Popup-Overlay","PopupControlID":"ctl00_ctl29_g_c168c423_9055_40ab_abd6_309bb94ef67f_ctl00_ucArticleTools_emailToColleagueControl_pnlEmailToColleague","dynamicServicePath":"/acgcr/pages/articleviewer.aspx","id":"emailToColleaguePopupExtender_1859226663"}, null, null, $get("ctl00_ctl29_g_c168c423_9055_40ab_abd6_309bb94ef67f_ctl00_ucArticleTools_emailToColleagueControl_hypEmailToColleague")); }); Sys.Application.add_init(function() { $create(AjaxControlToolkit.ModalPopupBehavior, {"BackgroundCssClass":"Popup-Overlay","PopupControlID":"ctl00_ctl29_g_c168c423_9055_40ab_abd6_309bb94ef67f_ctl00_ucArticleTools_exportToCitationManager_pnlExportTo","dynamicServicePath":"/acgcr/pages/articleviewer.aspx","id":"OpenExportToPopupExtender__195590522"}, null, null, $get("ctl00_ctl29_g_c168c423_9055_40ab_abd6_309bb94ef67f_ctl00_ucArticleTools_exportToCitationManager_hypExportToCitationManager")); }); Sys.Application.add_init(function() { $create(AjaxControlToolkit.ModalPopupBehavior, {"BackgroundCssClass":"Popup-Overlay","PopupControlID":"ctl00_ctl29_g_c168c423_9055_40ab_abd6_309bb94ef67f_ctl00_emailToColleague_pnlEmailToColleague","dynamicServicePath":"/acgcr/pages/articleviewer.aspx","id":"emailToColleaguePopupExtender_1258665720"}, null, null, $get("ctl00_ctl29_g_c168c423_9055_40ab_abd6_309bb94ef67f_ctl00_emailToColleague_hypEmailToColleague")); }); //]]> </script> </form> <span id="DeltaPlaceHolderUtilityContent"> </span> <script type="text/javascript">// <![CDATA[ var g_Workspace = "s4-workspace"; // ]]> </script> <div id="ctl00_ctl64_divAdConfig" data-adconfig="{&quot;adTitle&quot;:&quot;Advertisement&quot;,&quot;slotClass&quot;:&quot;ad-slot-container&quot;,&quot;topBanner&quot;:{&quot;sticky&quot;:&quot;false&quot;,&quot;stickyDuration&quot;:&quot;5000&quot;},&quot;lazyLoadingEnabled&quot;:false,&quot;npiHash&quot;:&quot;&quot;,&quot;pageTargeting&quot;:{&quot;ArticleId&quot;:&quot;02075970-202411000-00013&quot;}}" data-videoadconfig="{&quot;serverURL&quot;:&quot;https://pubads.g.doubleclick.net/gampad/ads&quot;,&quot;sizes&quot;:&quot;320x180|640x360&quot;,&quot;overlayUnitPath&quot;:&quot;/15726702761/Desktop/acgcr/Video/overlay&quot;,&quot;prerollUnitPath&quot;:&quot;/15726702761/Desktop/acgcr/Video/pre-roll&quot;,&quot;postrollUnitPath&quot;:&quot;/15726702761/Desktop/acgcr/Video/post-roll&quot;}" class="js-ad-config"> </div> <div id="ctl00_ctl64_divScripts"><script type="text/plain" async="async" src="https://securepubads.g.doubleclick.net/tag/js/gpt.js" class="optanon-category-C0004"></script> <script type="text/javascript" src="https://lww.com/_layouts/15/oaks.journals/core/ads.js?v=05.24.00.023" defer="true"> </script> </div> <!-- Control to add scripts depending on settings from Configuration DB like --> <script type="text/javascript"> (function($) { if (false) { $(".top-microbar-container").addClass('top-microbar-container-moved').insertAfter(".advertisement-content_top"); } $(function () { if (false) { $( '<div class="top-banner-verify-email">Be sure to check your email to verify your new user account! Thanks!</div>') .insertAfter(".top-microbar-container"); } }); })(jQuery); </script> <script type="text/javascript"> window.twttr = (function(d, s, id) { var js, fjs = d.getElementsByTagName(s)[0], t = window.twttr || {}; if (d.getElementById(id)) return t; js = d.createElement(s); js.id = id; js.src = "https://platform.twitter.com/widgets.js"; fjs.parentNode.insertBefore(js, fjs); t._e = []; t.ready = function(f) { t._e.push(f); }; return t; }(document, "script", "twitter-wjs")); </script> <script type="text/javascript" src="https://lww.com/_layouts/15/oaks.journals/core/ee.js?v=05.24.00.023" defer="true"> </script> <script type="text/javascript"> function OTScriptExecutedEventHandler(e) { if (window.WKTracker && window.TrackingSetup) { window.TrackingSetup.api = window.TrackingSetup.api || {}; window.TrackingSetup.api.baseUrl = 'https://apim-hlrp-ejp-prod.azure-api.net/ee'; window.TrackingSetup.api.log = window.TrackingSetup.api.log || {}; window.TrackingSetup.api.resolve = window.TrackingSetup.api.resolve || {}; window.TrackingSetup.gdpr = window.TrackingSetup.gdpr || {}; window.TrackingSetup.gdpr.cookieUsageConsent = window.cookieUsageConsent || false; window.WKTracker.configure(window.TrackingSetup); window.WKTracker.track(); // Remove the event listener window.removeEventListener('OTScriptExecuted', OTScriptExecutedEventHandler); } } window.addEventListener('OTScriptExecuted', OTScriptExecutedEventHandler); </script> <script>(function(){function c(){var b=a.contentDocument||a.contentWindow.document;if(b){var d=b.createElement('script');d.innerHTML="window.__CF$cv$params={r:'8e73abc66f6581b4',t:'MTczMjM5MTI0Ny4wMDAwMDA='};var a=document.createElement('script');a.nonce='';a.src='/cdn-cgi/challenge-platform/scripts/jsd/main.js';document.getElementsByTagName('head')[0].appendChild(a);";b.getElementsByTagName('head')[0].appendChild(d)}}if(document.body){var a=document.createElement('iframe');a.height=1;a.width=1;a.style.position='absolute';a.style.top=0;a.style.left=0;a.style.border='none';a.style.visibility='hidden';document.body.appendChild(a);if('loading'!==document.readyState)c();else if(window.addEventListener)document.addEventListener('DOMContentLoaded',c);else{var e=document.onreadystatechange||function(){};document.onreadystatechange=function(b){e(b);'loading'!==document.readyState&&(document.onreadystatechange=e,c())}}}})();</script></body> </html>

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