Orofacial Granulomatosis in Children can be the Initial Manifestation of Systemic Disease: A Presentation of Two Cases
Identifieur interne : 002184 ( Pmc/Corpus ); précédent : 002183; suivant : 002185Orofacial Granulomatosis in Children can be the Initial Manifestation of Systemic Disease: A Presentation of Two Cases
Auteurs : Anne Birgitte Simonsen ; Mette DeleuranSource :
- Dermatology Reports [ 2036-7392 ] ; 2014.
Abstract
Orofacial granulomatosis is a chronic granulomatous condition characterized by relapsing and remitting lip swelling and oral involvement that may include deep ulcers, tags and cobblestone formation. It occurs as an independent entity but also in conjunction with systemic diseases such as tuberculosis, sarcoidosis and Crohn’s disease. The clinical presentation is not indicative of concomitant systemic disease. To highlight the importance of thorough examination to rule out systemic disease, we present two childhood cases of orofacial granulomatosis, one of which was associated to Crohn’s disease.
Url:
DOI: 10.4081/dr.2014.5039
PubMed: 25386323
PubMed Central: 4223999
Links to Exploration step
PMC:4223999Le document en format XML
<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Orofacial Granulomatosis in Children can be the Initial Manifestation of Systemic Disease: A Presentation of Two Cases</title><author><name sortKey="Simonsen, Anne Birgitte" sort="Simonsen, Anne Birgitte" uniqKey="Simonsen A" first="Anne Birgitte" last="Simonsen">Anne Birgitte Simonsen</name></author><author><name sortKey="Deleuran, Mette" sort="Deleuran, Mette" uniqKey="Deleuran M" first="Mette" last="Deleuran">Mette Deleuran</name></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">25386323</idno><idno type="pmc">4223999</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4223999</idno><idno type="RBID">PMC:4223999</idno><idno type="doi">10.4081/dr.2014.5039</idno><date when="2014">2014</date><idno type="wicri:Area/Pmc/Corpus">002184</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">002184</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Orofacial Granulomatosis in Children can be the Initial Manifestation of Systemic Disease: A Presentation of Two Cases</title><author><name sortKey="Simonsen, Anne Birgitte" sort="Simonsen, Anne Birgitte" uniqKey="Simonsen A" first="Anne Birgitte" last="Simonsen">Anne Birgitte Simonsen</name></author><author><name sortKey="Deleuran, Mette" sort="Deleuran, Mette" uniqKey="Deleuran M" first="Mette" last="Deleuran">Mette Deleuran</name></author></analytic><series><title level="j">Dermatology Reports</title><idno type="ISSN">2036-7392</idno><idno type="eISSN">2036-7406</idno><imprint><date when="2014">2014</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Orofacial granulomatosis is a chronic granulomatous condition characterized by relapsing and remitting lip swelling and oral involvement that may include deep ulcers, tags and cobblestone formation. It occurs as an independent entity but also in conjunction with systemic diseases such as tuberculosis, sarcoidosis and Crohn’s disease. The clinical presentation is not indicative of concomitant systemic disease. To highlight the importance of thorough examination to rule out systemic disease, we present two childhood cases of orofacial granulomatosis, one of which was associated to Crohn’s disease.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Sanderson, J" uniqKey="Sanderson J">J Sanderson</name></author><author><name sortKey="Nunes, C" uniqKey="Nunes C">C Nunes</name></author><author><name sortKey="Escudier, M" uniqKey="Escudier M">M Escudier</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Campbell, H" uniqKey="Campbell H">H Campbell</name></author><author><name sortKey="Escudier, M" uniqKey="Escudier M">M Escudier</name></author><author><name sortKey="Patel, P" uniqKey="Patel P">P Patel</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Tuxen, Aj" uniqKey="Tuxen A">AJ Tuxen</name></author><author><name sortKey="Orchard, D" uniqKey="Orchard D">D Orchard</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Tilakaratne, Wm" uniqKey="Tilakaratne W">WM Tilakaratne</name></author><author><name sortKey="Freysdottir, J" uniqKey="Freysdottir J">J Freysdottir</name></author><author><name sortKey="Fortune, F" uniqKey="Fortune F">F Fortune</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kemmler, N" uniqKey="Kemmler N">N Kemmler</name></author><author><name sortKey="Pfannschmidt, N" uniqKey="Pfannschmidt N">N Pfannschmidt</name></author><author><name sortKey="Strohal, R" uniqKey="Strohal R">R Strohal</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Grave, B" uniqKey="Grave B">B Grave</name></author><author><name sortKey="Mccullough, M" uniqKey="Mccullough M">M McCullough</name></author><author><name sortKey="Wiesenfeld, D" uniqKey="Wiesenfeld D">D Wiesenfeld</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Campbell, He" uniqKey="Campbell H">HE Campbell</name></author><author><name sortKey="Escudier, Mp" uniqKey="Escudier M">MP Escudier</name></author><author><name sortKey="Patel, P" uniqKey="Patel P">P Patel</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Al Azri, Ar" uniqKey="Al Azri A">AR Al-Azri</name></author><author><name sortKey="Logan, Rm" uniqKey="Logan R">RM Logan</name></author><author><name sortKey="Goss, An" uniqKey="Goss A">AN Goss</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Ramesh, V" uniqKey="Ramesh V">V Ramesh</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kruschinski, C" uniqKey="Kruschinski C">C Kruschinski</name></author><author><name sortKey="Welkoborsky, Hj" uniqKey="Welkoborsky H">HJ Welkoborsky</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Saalman, R" uniqKey="Saalman R">R Saalman</name></author><author><name sortKey="Mattsson, U" uniqKey="Mattsson U">U Mattsson</name></author><author><name sortKey="Jontell, M" uniqKey="Jontell M">M Jontell</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Khouri, Jm" uniqKey="Khouri J">JM Khouri</name></author><author><name sortKey="Bohane, Td" uniqKey="Bohane T">TD Bohane</name></author><author><name sortKey="Day, As" uniqKey="Day A">AS Day</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Sainsbury, Cp" uniqKey="Sainsbury C">CP Sainsbury</name></author><author><name sortKey="Dodge, Ja" uniqKey="Dodge J">JA Dodge</name></author><author><name sortKey="Walker, Dm" uniqKey="Walker D">DM Walker</name></author><author><name sortKey="Aldred, Mj" uniqKey="Aldred M">MJ Aldred</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Rana, Ar" uniqKey="Rana A">AR Rana</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Smith, Vm" uniqKey="Smith V">VM Smith</name></author><author><name sortKey="Murphy, R" uniqKey="Murphy R">R Murphy</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Girlich, C" uniqKey="Girlich C">C Girlich</name></author><author><name sortKey="Bogenrieder, T" uniqKey="Bogenrieder T">T Bogenrieder</name></author><author><name sortKey="Palitzsch, Kd" uniqKey="Palitzsch K">KD Palitzsch</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kolho, Kl" uniqKey="Kolho K">KL Kolho</name></author><author><name sortKey="Heiskanen, K" uniqKey="Heiskanen K">K Heiskanen</name></author><author><name sortKey="Verkasalo, M" uniqKey="Verkasalo M">M Verkasalo</name></author><author><name sortKey="Pitkaranta, A" uniqKey="Pitkaranta A">A Pitkaranta</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Singhal, P" uniqKey="Singhal P">P Singhal</name></author><author><name sortKey="Chandan, Gd" uniqKey="Chandan G">GD Chandan</name></author><author><name sortKey="Das, Um" uniqKey="Das U">UM Das</name></author><author><name sortKey="Singhal, A" uniqKey="Singhal A">A Singhal</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kaarthikeyan, G" uniqKey="Kaarthikeyan G">G Kaarthikeyan</name></author><author><name sortKey="Arvind, M" uniqKey="Arvind M">M Arvind</name></author><author><name sortKey="Jayakumar, N" uniqKey="Jayakumar N">N Jayakumar</name></author><author><name sortKey="Khakar, M" uniqKey="Khakar M">M Khakar</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Howell, Jl" uniqKey="Howell J">JL Howell</name></author><author><name sortKey="Bussell, Rm" uniqKey="Bussell R">RM Bussell</name></author><author><name sortKey="Hegarty, Am" uniqKey="Hegarty A">AM Hegarty</name></author><author><name sortKey="Zaitoun, H" uniqKey="Zaitoun H">H Zaitoun</name></author></analytic></biblStruct></listBibl></div1></back></TEI><pmc article-type="case-report"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">Dermatol Reports</journal-id><journal-id journal-id-type="iso-abbrev">Dermatol Reports</journal-id><journal-id journal-id-type="publisher-id">DR</journal-id><journal-title-group><journal-title>Dermatology Reports</journal-title></journal-title-group><issn pub-type="ppub">2036-7392</issn><issn pub-type="epub">2036-7406</issn><publisher><publisher-name>PAGEPress Publications, Pavia, Italy</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">25386323</article-id><article-id pub-id-type="pmc">4223999</article-id><article-id pub-id-type="doi">10.4081/dr.2014.5039</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Orofacial Granulomatosis in Children can be the Initial Manifestation of Systemic Disease: A Presentation of Two Cases</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Simonsen</surname><given-names>Anne Birgitte</given-names></name><xref ref-type="corresp" rid="cor1"></xref></contrib><contrib contrib-type="author"><name><surname>Deleuran</surname><given-names>Mette</given-names></name></contrib></contrib-group><aff>Department of Dermatology,<institution>Aarhus University Hospital</institution>,<addr-line>Aarhus, Denmark</addr-line></aff><author-notes><corresp id="cor1">Department of Dermatology, Aarhus University Hospital, Norrebrogade 44, 8000 Aarhus C, Denmark. <phone>+45.784.50000</phone><email>anbsim@rm.dk</email></corresp><fn fn-type="con"><p>Contributions: the authors contributed equally.</p></fn><fn fn-type="conflict"><p>Conflict of interests: the authors declare no potential conflict of interests.</p></fn></author-notes><pub-date pub-type="epub"><day>12</day><month>5</month><year>2014</year></pub-date><pub-date pub-type="collection"><day>17</day><month>2</month><year>2014</year></pub-date><volume>6</volume><issue>1</issue><elocation-id>5039</elocation-id><history><date date-type="received"><day>20</day><month>8</month><year>2013</year></date><date date-type="rev-recd"><day>17</day><month>11</month><year>2013</year></date><date date-type="accepted"><day>20</day><month>12</month><year>2013</year></date></history><permissions><copyright-statement>©Copyright A.B. Simonsen and M. Deleuran</copyright-statement><copyright-year>2014</copyright-year><copyright-holder>Licensee PAGEPress, Italy</copyright-holder><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc/3.0/"><license-p>This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (<uri xlink:href="http://creativecommons.org/licenses/by-nc/3.0/">http://creativecommons.org/licenses/by-nc/3.0/</uri>) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p></license></permissions><abstract><p>Orofacial granulomatosis is a chronic granulomatous condition characterized by relapsing and remitting lip swelling and oral involvement that may include deep ulcers, tags and cobblestone formation. It occurs as an independent entity but also in conjunction with systemic diseases such as tuberculosis, sarcoidosis and Crohn’s disease. The clinical presentation is not indicative of concomitant systemic disease. To highlight the importance of thorough examination to rule out systemic disease, we present two childhood cases of orofacial granulomatosis, one of which was associated to Crohn’s disease.</p></abstract><kwd-group><title>Key words</title><kwd>orofacial granulomatosis</kwd><kwd>children</kwd><kwd>Chron's disease</kwd></kwd-group><counts><fig-count count="2"></fig-count><table-count count="1"></table-count><equation-count count="0"></equation-count><ref-count count="20"></ref-count><page-count count="2"></page-count></counts></article-meta></front><body><sec sec-type="intro" id="sec1-1"><title>Introduction</title><p>Orofacial granulomatosis (OFG) is a chronic granulomatous condition characterized by relapsing and remitting lip swelling and oral involvement affecting the buccal mucosa, gingivae and floor of the mouth.<sup><xref rid="ref1" ref-type="bibr">1</xref>,<xref rid="ref2" ref-type="bibr">2</xref></sup> Histologically, the condition is characterized by non-caseating granulomas that block lymphatics causing lymphoedema. Eventually, fibrosis may develop, making the swelling permanent.<sup><xref rid="ref3" ref-type="bibr">3</xref></sup> Differential diagnoses include granulomatous cheilitis, where lesions are limited to the lips, and Melkersson-Rosenthal Syndrome, which consists of the triad of persistent lip or facial swelling, recurrent facial paralysis and fissured tongue (<xref ref-type="table" rid="table001">Table l</xref>).<sup><xref rid="ref4" ref-type="bibr">4</xref></sup></p><p>The precise etiology of OFG is unknown, although allergies, infections, genetic predisposition and immunological mechanisms have been suggested as causative agents.<sup><xref rid="ref5" ref-type="bibr">5-7</xref></sup>Furthermore, OFG is seen as an independent entity, but also in conjunction with systemic conditions such as tuberculosis, sarcoidosis and Crohn’s disease.<sup><xref rid="ref8" ref-type="bibr">8-10</xref></sup> To highlight the association between childhood OFG and systemic disease, we present two cases of OFG in children.</p></sec><sec id="sec1-2"><title>Case Report #1</title><p>Case 1 was a 12-year old boy referred to us because of persistent swelling of the lips and gingivae, along with an uncharacteristic facial rash (<xref ref-type="fig" rid="fig001">Figure 1</xref>). He had a history of atopic dermatitis, but was otherwise previously healthy and had no predisposition to skin- or gastrointestinal disease. He presented with pronounced perioral edema, severe fissuring of the lips, angular cheilitis, gingival edema, and cobblestone formation of the buccal mucosa. During flare-ups the boy experienced severe abdominal pain. Suspecting inflammatory bowel disease, the patient was thoroughly examined with blood tests, biopsies from the gingival and buccal mucosa, and endoscopy of the gastrointestinal tract. The histopathological examination of the mucosal biopsies revealed chronic granulomatous inflammation consistent with OFG and oral Crohn’s disease. Serum calprotectin was elevated to 409 mg/kg. CRP and blood sedimentation rate were normal. Gastroscopy showed duodenal inflammation and by subsequent endoscopy ileac aphtous ulcers were detected. The patient was diagnosed with Crohn’s disease. Infliximab treatment was initiated with a good clinical response on both abdominal symptoms and mucosal lesions.</p></sec><sec id="sec1-3"><title>Case Report #2</title><p>Case 2 was a 9-year old boy who presented chronic swelling of the upper lip, redness, swelling and hypertrophy of the gingivae, and cobblestone formation of the buccal mucosa (<xref ref-type="fig" rid="fig002">Figure 2</xref>). This patient had IgE-mediated allergy to grass, birch, and alternaria, but was otherwise healthy and was not predisposed to any skin or systemic diseases. He had no history of abdominal pain or stool alteration and no respiratory symptoms. As in case 1, the mucosal biopsy from the upper-lip revealed granulomatous inflammation. All blood tests were normal. The patient was further conferred with a pediatric gastroenterologist. Serum and fecal calprotectin were normal. As this patient had no history of abdominal problems, no further examination was indicated at the time. The oral lesions were treated with topical fluticasone propionate 50 g/dose. The initial dosage was 1 spray unit twice daily. After 6 weeks this was reduced to once daily for another 6 weeks, after which the treatment was given pro necessitate. The lesions responded well to this treatment and after 12 weeks only mild infiltration and swelling of the gingivae remained without noticeable inflammatory activity. Regular check-ups every 3 months ensure that the condition is stable, and that possible gastrointestinal symptoms are detected.</p></sec><sec sec-type="discussion" id="sec1-4"><title>Discussion</title><p>Whether OFG should be considered a separate entity or a manifestation of systemic disease remains a topic of discussion.<sup><xref rid="ref4" ref-type="bibr">4</xref></sup> In particular, several case reports have suggested an association to Crohn’s disease. The two entities share a number of clinical and histological features, but the exact relationship has not yet been established.<sup><xref rid="ref4" ref-type="bibr">4</xref></sup> Some authors suggest that patients with OFG may have subclinical Crohn’s disease.<sup><xref rid="ref1" ref-type="bibr">1</xref>,<xref rid="ref11" ref-type="bibr">11</xref></sup> Furthermore, several authors have observed that children are more likely to have onset of OFG preceding symptoms of Crohn’s disease,<sup><xref rid="ref3" ref-type="bibr">3</xref>,<xref rid="ref11" ref-type="bibr">11-13</xref></sup> and speculate that childhood onset carries a higher risk of developing Crohn’s disease.</p><p>The prevalence of Crohn’s disease in children with OFG is not well-established and could be underestimated since our current knowledge is based on case reports and case studies.<sup><xref rid="ref3" ref-type="bibr">3</xref></sup> The clinical presentation of the two patients presented here was almost identical. The history revealed that one of the patients had intermittent abdominal pain, which led to further examination, revealing an underlying Crohn’s disease. This illustrates an important point, to be remembered when encountering children with OFG. The clinical presentation is not indicative of concomitant systemic disease and symptoms of this may be few. Regardless of the clinical presentation, systemic disease should always be ruled out. OFG has been reported to be a manifestation of tuberculosis and sarcoidosis,<sup><xref rid="ref8" ref-type="bibr">8-10</xref></sup> however, the vast majority of the literature focuses on the possible link between OFG and Crohn’s disease (Supplementary Table 1).<sup><xref rid="ref3" ref-type="bibr">3</xref>,<xref rid="ref11" ref-type="bibr">11</xref>,<xref rid="ref12" ref-type="bibr">12</xref>,<xref rid="ref14" ref-type="bibr">14-20</xref></sup> In our opinion, the caring for pediatric patients with OFG should focus mainly on monitoring for signs, symptoms, and laboratory evidence of Crohn’s disease. A thorough patient history and clinical examination is necessary and we recommend blood screening including CRP, blood sedimentation rate, complete blood count, and serum calprotectin. If serum calprotectin is elevated, the patient should be referred to a pediatric gastroenterologist. Any specific symptoms or abnormal blood screening should lead to further examination of relevance, <italic>i.e.</italic> a history of pulmonary symptoms should lead to X-ray examination of the chest and further blood tests depending on the symptoms and patient history. We recommend that patients be followed closely until the oral symptoms stabilize. When the condition is stable follow up at regular intervals ensures that the condition remains stable and any new symptom of systemic disease is detected.</p></sec></body><back><ref-list><title>References</title><ref id="ref1"><label>1.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Sanderson</surname><given-names>J</given-names></name><name><surname>Nunes</surname><given-names>C</given-names></name><name><surname>Escudier</surname><given-names>M</given-names></name><etal></etal></person-group><article-title>Oro-facial granulomatosis: Crohn’s disease or a new inflammatory bowel disease?</article-title><source>Inflamm Bowel Dis</source><year>2005</year>;<volume>11</volume>:<fpage>840</fpage>-<lpage>6</lpage>.<pub-id pub-id-type="pmid">16116319</pub-id></mixed-citation></ref><ref id="ref2"><label>2.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Campbell</surname><given-names>H</given-names></name><name><surname>Escudier</surname><given-names>M</given-names></name><name><surname>Patel</surname><given-names>P</given-names></name><etal></etal></person-group><article-title>Distinguishing orofacial granulomatosis from Crohn’s disease: two separate disease entities?</article-title><source>Inflamm Bowel Dis</source><year>2011</year>;<volume>17</volume>:<fpage>2109</fpage>-<lpage>15</lpage>.<pub-id pub-id-type="pmid">21910172</pub-id></mixed-citation></ref><ref id="ref3"><label>3.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Tuxen</surname><given-names>AJ</given-names></name><name><surname>Orchard</surname><given-names>D</given-names></name></person-group><article-title>Childhood and adolescent orofacial granulomatosis is strongly associated with Crohn’s disease and responds to intralesional corticosteroids</article-title>. <source>Australas J Dermatol</source><year>2010</year>;<volume>51</volume>:<fpage>124</fpage>-<lpage>7</lpage>.<pub-id pub-id-type="pmid">20546219</pub-id></mixed-citation></ref><ref id="ref4"><label>4.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Tilakaratne</surname><given-names>WM</given-names></name><name><surname>Freysdottir</surname><given-names>J</given-names></name><name><surname>Fortune</surname><given-names>F</given-names></name></person-group><article-title>Orofacial granulomatosis: review on aetiology and pathogenesis</article-title>. <source>J Oral Pathol Med</source><year>2008</year>;<volume>37</volume>:<fpage>191</fpage>-<lpage>5</lpage>.<pub-id pub-id-type="pmid">18321344</pub-id></mixed-citation></ref><ref id="ref5"><label>5.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Kemmler</surname><given-names>N</given-names></name><name><surname>Pfannschmidt</surname><given-names>N</given-names></name><name><surname>Strohal</surname><given-names>R</given-names></name></person-group><article-title>Orofacial granulomatosis as first manifestation of Crohn’s disease: successful treatment of both conditions with a combination of infliximab and dapsone</article-title>. <source>Acta Derm Venereol</source><year>2012</year>;<volume>92</volume>:<fpage>406</fpage>-<lpage>7</lpage>.<pub-id pub-id-type="pmid">22113308</pub-id></mixed-citation></ref><ref id="ref6"><label>6.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Grave</surname><given-names>B</given-names></name><name><surname>McCullough</surname><given-names>M</given-names></name><name><surname>Wiesenfeld</surname><given-names>D</given-names></name></person-group><article-title>Orofacial granulomatosis - a 20-year review</article-title>. <source>Oral Dis</source><year>2009</year>;<volume>15</volume>:<fpage>46</fpage>-<lpage>51</lpage>.<pub-id pub-id-type="pmid">19076470</pub-id></mixed-citation></ref><ref id="ref7"><label>7.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Campbell</surname><given-names>HE</given-names></name><name><surname>Escudier</surname><given-names>MP</given-names></name><name><surname>Patel</surname><given-names>P</given-names></name><etal></etal></person-group><article-title>Review article: cinnamon- and benzoate-free diet as a primary treatment for orofacial granulomatosis</article-title>. <source>Aliment Pharmacol Ther</source><year>2011</year>;<volume>34</volume>:<fpage>687</fpage>-<lpage>701</lpage>.<pub-id pub-id-type="pmid">21815899</pub-id></mixed-citation></ref><ref id="ref8"><label>8.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Al-Azri</surname><given-names>AR</given-names></name><name><surname>Logan</surname><given-names>RM</given-names></name><name><surname>Goss</surname><given-names>AN</given-names></name></person-group><article-title>Oral lesion as the first clinical presentation in sarcoidosis: a case report</article-title>. <source>Oman Med J</source><year>2012</year>;<volume>27</volume>:<fpage>243</fpage>-<lpage>5</lpage>.<pub-id pub-id-type="pmid">22811777</pub-id></mixed-citation></ref><ref id="ref9"><label>9.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Ramesh</surname><given-names>V</given-names></name></person-group><article-title>Orofacial granulomatosis due to tuberculosis</article-title>. <source>Pediatr Dermatol</source><year>2009</year>;<volume>26</volume>:<fpage>108</fpage>-<lpage>9</lpage>.<pub-id pub-id-type="pmid">19250428</pub-id></mixed-citation></ref><ref id="ref10"><label>10.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Kruschinski</surname><given-names>C</given-names></name><name><surname>Welkoborsky</surname><given-names>HJ</given-names></name></person-group><article-title>Tuberculosis of the larynx associated with orofacial granulomatosis in childhood</article-title>. <source>Otolaryngol Head Neck Surg</source><year>2005</year>;<volume>132</volume>:<fpage>967</fpage>-<lpage>9</lpage>.<pub-id pub-id-type="pmid">15944576</pub-id></mixed-citation></ref><ref id="ref11"><label>11.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Saalman</surname><given-names>R</given-names></name><name><surname>Mattsson</surname><given-names>U</given-names></name><name><surname>Jontell</surname><given-names>M</given-names></name></person-group><article-title>Orofacial granulomatosis in childhood-a clinical entity that may indicate Crohn’s disease as well as food allergy</article-title>. <source>Acta Paediatr</source><year>2009</year>;<volume>98</volume>:<fpage>1162</fpage>-<lpage>7</lpage>.<pub-id pub-id-type="pmid">19397547</pub-id></mixed-citation></ref><ref id="ref12"><label>12.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Khouri</surname><given-names>JM</given-names></name><name><surname>Bohane</surname><given-names>TD</given-names></name><name><surname>Day</surname><given-names>AS</given-names></name></person-group><article-title>Is orofacial granulomatosis in children a feature of Crohn’s disease?</article-title><source>Acta Paediatr</source><year>2005</year>;<volume>94</volume>:<fpage>501</fpage>-<lpage>4</lpage>.<pub-id pub-id-type="pmid">16092468</pub-id></mixed-citation></ref><ref id="ref13"><label>13.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Sainsbury</surname><given-names>CP</given-names></name><name><surname>Dodge</surname><given-names>JA</given-names></name><name><surname>Walker</surname><given-names>DM</given-names></name><name><surname>Aldred</surname><given-names>MJ</given-names></name></person-group><article-title>Orofacial granulomatosis in childhood</article-title>. <source>Br Dent J</source><year>1987</year>;<volume>163</volume>:<fpage>154</fpage>-<lpage>7</lpage>.<pub-id pub-id-type="pmid">3478055</pub-id></mixed-citation></ref><ref id="ref14"><label>14.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Rana</surname><given-names>AR</given-names></name></person-group><article-title>Orofacial granulomatosis: a case report with review of literature</article-title>. <source>J Indian Soc Periodontol</source><year>2012</year>;<volume>16</volume>:<fpage>469</fpage>-<lpage>74</lpage>.<pub-id pub-id-type="pmid">23162350</pub-id></mixed-citation></ref><ref id="ref15"><label>15.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Smith</surname><given-names>VM</given-names></name><name><surname>Murphy</surname><given-names>R</given-names></name></person-group><article-title>Orofacial granulomatosis: three case reports illustrating the spectrum of disease and overlap with Crohn’s disease</article-title>. <source>Clin Exp Dermatol</source><year>2013</year>;<volume>38</volume>:<fpage>33</fpage>-<lpage>5</lpage>.<pub-id pub-id-type="pmid">22548302</pub-id></mixed-citation></ref><ref id="ref16"><label>16.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Girlich</surname><given-names>C</given-names></name><name><surname>Bogenrieder</surname><given-names>T</given-names></name><name><surname>Palitzsch</surname><given-names>KD</given-names></name><etal></etal></person-group><article-title>Orofacial granulomatosis as initial manifestation of Crohn’s disease: a report of two cases</article-title>. <source>Eur J Gastroenterol Hepatol</source><year>2002</year>;<volume>14</volume>:<fpage>873</fpage>-<lpage>6</lpage>.<pub-id pub-id-type="pmid">12172408</pub-id></mixed-citation></ref><ref id="ref17"><label>17.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Kolho</surname><given-names>KL</given-names></name><name><surname>Heiskanen</surname><given-names>K</given-names></name><name><surname>Verkasalo</surname><given-names>M</given-names></name><name><surname>Pitkaranta</surname><given-names>A</given-names></name></person-group><article-title>Orofacial granulomatosis in children: a challenge for diagnosis and treatment</article-title>. <source>Int J Pediatr Otorhinolaryngol</source><year>2011</year>;<volume>75</volume>:<fpage>864</fpage>-<lpage>7</lpage>.<pub-id pub-id-type="pmid">21458863</pub-id></mixed-citation></ref><ref id="ref18"><label>18.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Singhal</surname><given-names>P</given-names></name><name><surname>Chandan</surname><given-names>GD</given-names></name><name><surname>Das</surname><given-names>UM</given-names></name><name><surname>Singhal</surname><given-names>A</given-names></name></person-group><article-title>A rare case report of orofacial granulomatosis in a pediatric patient</article-title>. <source>J Indian Soc Pedod Prev Dent</source><year>2012</year>;<volume>30</volume>:<fpage>262</fpage>-<lpage>6</lpage>.<pub-id pub-id-type="pmid">23263433</pub-id></mixed-citation></ref><ref id="ref19"><label>19.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Kaarthikeyan</surname><given-names>G</given-names></name><name><surname>Arvind</surname><given-names>M</given-names></name><name><surname>Jayakumar</surname><given-names>N</given-names></name><name><surname>Khakar</surname><given-names>M</given-names></name></person-group><article-title>Idiopathic orofacial granulomatosis in a young patient: a rare entity</article-title>. <source>J Oral Maxillofac Pathol</source><year>2012</year>;<volume>16</volume>:<fpage>432</fpage>-<lpage>4</lpage>.<pub-id pub-id-type="pmid">23248481</pub-id></mixed-citation></ref><ref id="ref20"><label>20.</label><mixed-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Howell</surname><given-names>JL</given-names></name><name><surname>Bussell</surname><given-names>RM</given-names></name><name><surname>Hegarty</surname><given-names>AM</given-names></name><name><surname>Zaitoun</surname><given-names>H</given-names></name></person-group><article-title>Service evaluation of patients with orofacial granulomatosis and patients with oral Crohn’s disease attending a paediatric oral medicine clinic</article-title>. <source>Eur Arch Paediatr Dent</source><year>2012</year>;<volume>13</volume>:<fpage>191</fpage>-<lpage>6</lpage>.<pub-id pub-id-type="pmid">22883358</pub-id></mixed-citation></ref></ref-list></back><floats-group><fig id="fig001" orientation="portrait" position="float"><label>Figure 1.</label><caption><p>Perioral edema, severe fissuring of the lips, angular cheilitis, gingival edema, and cobblestone formation of the mucosa.</p></caption><graphic xlink:href="dr-2014-1-5039-g001"></graphic></fig><fig id="fig002" orientation="portrait" position="float"><label>Figure 2.</label><caption><p>Chronic swelling of the upper lip. Redness, swelling and hypertrophy of the gingivae.</p></caption><graphic xlink:href="dr-2014-1-5039-g002"></graphic></fig><table-wrap id="table001" orientation="portrait" position="float"><label>Table 1.</label><caption><p>Differential diagnoses of orofacial granulomatosis.</p></caption><table frame="box" rules="all"><thead><tr><th rowspan="1" colspan="1"></th><th align="center" rowspan="1" colspan="1">Granulomatous swelling of lips</th><th align="center" rowspan="1" colspan="1">Cobblestone formation of oral mucosa</th><th align="center" rowspan="1" colspan="1">Mucosal swelling</th><th align="center" rowspan="1" colspan="1">Oral ulcers</th><th align="center" rowspan="1" colspan="1">Facial nerve paralysis</th><th align="center" rowspan="1" colspan="1">Plicated tongue</th></tr></thead><tbody><tr><td align="left" rowspan="1" colspan="1">Orofacial granulomatosis</td><td align="center" rowspan="1" colspan="1">+</td><td align="center" rowspan="1" colspan="1">+</td><td align="center" rowspan="1" colspan="1">+</td><td align="center" rowspan="1" colspan="1">(+)</td><td align="center" rowspan="1" colspan="1"><bold>-</bold></td><td align="center" rowspan="1" colspan="1">-</td></tr><tr><td align="left" rowspan="1" colspan="1">Granulomatous cheilitis</td><td align="center" rowspan="1" colspan="1">+</td><td align="center" rowspan="1" colspan="1"><bold>-</bold></td><td align="center" rowspan="1" colspan="1"><bold>-</bold></td><td align="center" rowspan="1" colspan="1"><bold>-</bold></td><td align="center" rowspan="1" colspan="1"><bold>-</bold></td><td align="center" rowspan="1" colspan="1">-</td></tr><tr><td align="left" rowspan="1" colspan="1">Melkersson-Rosenthal Syndrome</td><td align="center" rowspan="1" colspan="1">+</td><td align="center" rowspan="1" colspan="1"><bold>-</bold></td><td align="center" rowspan="1" colspan="1"><bold>-</bold></td><td align="center" rowspan="1" colspan="1"><bold>-</bold></td><td align="center" rowspan="1" colspan="1">+</td><td align="center" rowspan="1" colspan="1">+</td></tr></tbody></table></table-wrap></floats-group></pmc></record>Pour manipuler ce document sous Unix (Dilib)
EXPLOR_STEP=$WICRI_ROOT/Wicri/Sante/explor/LymphedemaV1/Data/Pmc/Corpus
HfdSelect -h $EXPLOR_STEP/biblio.hfd -nk 002184 | SxmlIndent | more
Ou
HfdSelect -h $EXPLOR_AREA/Data/Pmc/Corpus/biblio.hfd -nk 002184 | SxmlIndent | more
Pour mettre un lien sur cette page dans le réseau Wicri
{{Explor lien
|wiki= Wicri/Sante
|area= LymphedemaV1
|flux= Pmc
|étape= Corpus
|type= RBID
|clé= PMC:4223999
|texte= Orofacial Granulomatosis in Children can be the Initial Manifestation of Systemic Disease: A Presentation of Two Cases
}}
Pour générer des pages wiki
HfdIndexSelect -h $EXPLOR_AREA/Data/Pmc/Corpus/RBID.i -Sk "pubmed:25386323" \
| HfdSelect -Kh $EXPLOR_AREA/Data/Pmc/Corpus/biblio.hfd \
| NlmPubMed2Wicri -a LymphedemaV1
| This area was generated with Dilib version V0.6.31. |  |