Case for diagnosis
Submandibular Cutaneous Fistula
Fístula cutánea submandibular
C. Prada García
, M.Á. Rodríguez Prieto
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Servicio de Dermatología, Complejo Asistencial Universitario de León, León, Spain
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Prada García, M.Á. Rodríguez Prieto" "autores" => array:2 [ 0 => array:4 [ "nombre" => "C." "apellidos" => "Prada García" "email" => array:1 [ 0 => "caminoprada@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "M.Á." 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The lesion had been present for 6 months. Of interest in her medical history were hypertension, type 2 diabetes mellitus, dyslipidemia, and osteoporosis. She had been receiving treatment with nimodipine, metformin, fluvastatin, and alendronate for the last 3 years, and had undergone a tooth extraction 2 years earlier.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical Examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">The left perimandibular region was swollen, erythematous, and indurated to palpation, with a submandibular, ulcerated, fistulous area of retracted skin, from which a seropurulent exudate drained. Within this area was a fleshy excrescent papule (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Examination of the oral cavity revealed teeth in poor condition.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Histopathology</span><p id="par0015" class="elsevierStylePara elsevierViewall">Hematoxylin-eosin staining showed an area of ulceration and acute inflammation, and the presence of large multinucleated cells without any histologic signs of malignancy.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Additional Tests</span><p id="par0020" class="elsevierStylePara elsevierViewall">Orthopantomography showed an area of osteolysis of the cortical bone in the left mandibular arch, as well as other radiolucent points indicative of general mandibular involvement (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleBold">What Is Your Diagnosis?</span></p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Diagnosis</span><p id="par0030" class="elsevierStylePara elsevierViewall">Bisphosphonate-induced osteonecrosis of the jaw.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Course and Treatment</span><p id="par0035" class="elsevierStylePara elsevierViewall">Alendronate was discontinued. After culture of the exudate, which was positive for <span class="elsevierStyleItalic">Staphylococcus aureus</span>, the patient was managed conservatively with antibiotic therapy with ciprofloxacin 750<span class="elsevierStyleHsp" style=""></span>mg every 12<span class="elsevierStyleHsp" style=""></span>hours for 3 weeks. The skin lesion improved markedly. The seropurulent exudate and inflammation were no longer present, but the fistula persisted (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Comment</span><p id="par0040" class="elsevierStylePara elsevierViewall">Biphosphonates are potent inhibitors of osteoclastic bone resorption and angiogenesis. They are used in the treatment of diseases such as lytic bone metastases, malignant hypercalcemia, multiple myeloma, osteoporosis, and Paget disease.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–3</span></a> Osteonecrosis of the jaw appears to be caused by poor blood supply and lack of bone remodeling and regeneration.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> This condition was first described in 2003.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,5</span></a> The incidence of this complication in oral bisphosphonate therapy is estimated to range approximately from 0.01% to 0.04%.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,5</span></a> Osteonecrosis of the jaw develops after chronic inflammation in an environment of deficient vascularization, leading to changes in bone microstructure and ultimately to collapse.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> These events result from the inability of the bone to increase the regeneration process in response to chewing or dental manipulation or infections; in such situations a greater functional bone reserve is required.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The risk of this complication is greater when the treatment duration exceeds 3 years, and 70% of cases are triggered by tooth extractions.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3,5</span></a> Other factors such as corticosteroid use, diabetes, advanced age, insufficient dental hygiene, smoking, and excessive alcohol consumption increase the risk of the disease and accelerate progression.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,5</span></a> Overall, 60% of cases are reported in women.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The half-life the bisphosphonate that has accumulated in bone can be as long as 11 years.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">When a mandibular fistula is present, the differential diagnosis should include not only odontogenic cysts, foreign-body reaction, pyogenic granuloma, tumors, and infectious processes, but also bisphosphonate-induced osteonecrosis of the jaw if the patient is receiving biphosphonates, particularly if multiple fistulas are present.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Diagnosis is based largely on the clinical manifestations. Imaging studies are the main additional tests and allow the severity and extent of mandibular involvement to be determined.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">No effective treatment has been established, although a conservative approach seems the most recommendable. Aggressive debridement should be avoided due to the risk of recurrence and sequelae, as bone involvement is generalized and there is a risk of increasing the size of the lesion.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> Systematic cultures are recommended.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> According to Marx et al.,<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> the approach should consist of oral hygiene and rinsing with chlorhexidine 0.12%, long-term continuous or intermittent antibiotic treatment, and avoidance of surgery or extensive debridement.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:8 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Medical History" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Physical Examination" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Histopathology" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Additional Tests" ] 4 => array:2 [ "identificador" => "sec0025" "titulo" => "Diagnosis" ] 5 => array:2 [ "identificador" => "sec0030" "titulo" => "Course and Treatment" ] 6 => array:2 [ "identificador" => "sec0035" "titulo" => "Comment" ] 7 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Prada García C, Rodríguez Prieto MÁ. Fístula cutánea submandibular. Actas Dermosifiliogr. 2013;104:629–30.</p>" ] ] "multimedia" => array:3 [ 0 => array:6 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1447 "Ancho" => 1583 "Tamanyo" => 200754 ] ] ] 1 => array:6 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 903 "Ancho" => 1583 "Tamanyo" => 161631 ] ] ] 2 => array:6 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1290 "Ancho" => 1583 "Tamanyo" => 201142 ] ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Fístulas mandibulares en una mujer de 75 años" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "R. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 16 | 10 | 26 |
| 2024 Octubre | 108 | 58 | 166 |
| 2024 Septiembre | 115 | 46 | 161 |
| 2024 Agosto | 150 | 64 | 214 |
| 2024 Julio | 141 | 45 | 186 |
| 2024 Junio | 118 | 56 | 174 |
| 2024 Mayo | 113 | 40 | 153 |
| 2024 Abril | 107 | 31 | 138 |
| 2024 Marzo | 106 | 43 | 149 |
| 2024 Febrero | 89 | 33 | 122 |
| 2024 Enero | 75 | 37 | 112 |
| 2023 Diciembre | 81 | 21 | 102 |
| 2023 Noviembre | 83 | 38 | 121 |
| 2023 Octubre | 68 | 44 | 112 |
| 2023 Septiembre | 70 | 36 | 106 |
| 2023 Agosto | 46 | 23 | 69 |
| 2023 Julio | 64 | 44 | 108 |
| 2023 Junio | 76 | 32 | 108 |
| 2023 Mayo | 90 | 26 | 116 |
| 2023 Abril | 85 | 28 | 113 |
| 2023 Marzo | 82 | 28 | 110 |
| 2023 Febrero | 67 | 24 | 91 |
| 2023 Enero | 44 | 27 | 71 |
| 2022 Diciembre | 53 | 65 | 118 |
| 2022 Noviembre | 39 | 33 | 72 |
| 2022 Octubre | 27 | 24 | 51 |
| 2022 Septiembre | 40 | 39 | 79 |
| 2022 Agosto | 29 | 27 | 56 |
| 2022 Julio | 30 | 34 | 64 |
| 2022 Junio | 39 | 29 | 68 |
| 2022 Mayo | 104 | 36 | 140 |
| 2022 Abril | 131 | 28 | 159 |
| 2022 Marzo | 140 | 68 | 208 |
| 2022 Febrero | 129 | 45 | 174 |
| 2022 Enero | 188 | 59 | 247 |
| 2021 Diciembre | 105 | 54 | 159 |
| 2021 Noviembre | 123 | 68 | 191 |
| 2021 Octubre | 123 | 65 | 188 |
| 2021 Septiembre | 84 | 55 | 139 |
| 2021 Agosto | 87 | 42 | 129 |
| 2021 Julio | 74 | 41 | 115 |
| 2021 Junio | 80 | 41 | 121 |
| 2021 Mayo | 85 | 49 | 134 |
| 2021 Abril | 145 | 62 | 207 |
| 2021 Marzo | 132 | 39 | 171 |
| 2021 Febrero | 98 | 41 | 139 |
| 2021 Enero | 73 | 25 | 98 |
| 2020 Diciembre | 57 | 25 | 82 |
| 2020 Noviembre | 44 | 25 | 69 |
| 2020 Octubre | 39 | 10 | 49 |
| 2020 Septiembre | 37 | 21 | 58 |
| 2020 Agosto | 23 | 26 | 49 |
| 2020 Julio | 25 | 15 | 40 |
| 2020 Junio | 35 | 29 | 64 |
| 2020 Mayo | 28 | 15 | 43 |
| 2020 Abril | 35 | 27 | 62 |
| 2020 Marzo | 23 | 13 | 36 |
| 2020 Febrero | 3 | 1 | 4 |
| 2020 Enero | 4 | 0 | 4 |
| 2019 Diciembre | 8 | 6 | 14 |
| 2019 Noviembre | 4 | 6 | 10 |
| 2019 Octubre | 0 | 9 | 9 |
| 2019 Septiembre | 10 | 12 | 22 |
| 2019 Agosto | 4 | 14 | 18 |
| 2019 Julio | 2 | 17 | 19 |
| 2019 Junio | 4 | 30 | 34 |
| 2019 Mayo | 5 | 28 | 33 |
| 2019 Abril | 0 | 32 | 32 |
| 2019 Marzo | 0 | 15 | 15 |
| 2019 Febrero | 2 | 3 | 5 |
| 2019 Enero | 1 | 1 | 2 |
| 2018 Diciembre | 2 | 5 | 7 |
| 2018 Noviembre | 2 | 0 | 2 |
| 2018 Octubre | 1 | 0 | 1 |
| 2018 Septiembre | 4 | 0 | 4 |
| 2018 Mayo | 0 | 3 | 3 |
| 2018 Abril | 0 | 1 | 1 |
| 2018 Marzo | 5 | 1 | 6 |
| 2018 Febrero | 36 | 3 | 39 |
| 2018 Enero | 37 | 5 | 42 |
| 2017 Diciembre | 49 | 4 | 53 |
| 2017 Noviembre | 45 | 5 | 50 |
| 2017 Octubre | 32 | 5 | 37 |
| 2017 Septiembre | 44 | 3 | 47 |
| 2017 Agosto | 52 | 4 | 56 |
| 2017 Julio | 45 | 4 | 49 |
| 2017 Junio | 63 | 14 | 77 |
| 2017 Mayo | 54 | 11 | 65 |
| 2017 Abril | 41 | 9 | 50 |
| 2017 Marzo | 40 | 33 | 73 |
| 2017 Febrero | 34 | 6 | 40 |
| 2017 Enero | 45 | 13 | 58 |
| 2016 Diciembre | 60 | 7 | 67 |
| 2016 Noviembre | 84 | 12 | 96 |
| 2016 Octubre | 127 | 20 | 147 |
| 2016 Septiembre | 175 | 10 | 185 |
| 2016 Agosto | 88 | 13 | 101 |
| 2016 Julio | 32 | 8 | 40 |
| 2016 Junio | 8 | 7 | 15 |
| 2016 Mayo | 8 | 9 | 17 |
| 2016 Abril | 3 | 16 | 19 |
| 2016 Marzo | 3 | 3 | 6 |
| 2016 Febrero | 4 | 3 | 7 |
| 2016 Enero | 7 | 2 | 9 |
| 2015 Diciembre | 12 | 2 | 14 |
| 2015 Noviembre | 22 | 2 | 24 |
| 2015 Octubre | 15 | 1 | 16 |
| 2015 Septiembre | 6 | 7 | 13 |
| 2015 Agosto | 5 | 2 | 7 |
| 2015 Julio | 89 | 7 | 96 |
| 2015 Junio | 46 | 5 | 51 |
| 2015 Mayo | 48 | 4 | 52 |
| 2015 Abril | 39 | 3 | 42 |
| 2015 Marzo | 34 | 4 | 38 |
| 2015 Febrero | 55 | 6 | 61 |
| 2015 Enero | 38 | 4 | 42 |
| 2014 Diciembre | 47 | 7 | 54 |
| 2014 Noviembre | 29 | 7 | 36 |
| 2014 Octubre | 53 | 12 | 65 |
| 2014 Septiembre | 33 | 9 | 42 |
| 2014 Agosto | 60 | 21 | 81 |
| 2014 Julio | 35 | 11 | 46 |
| 2014 Junio | 45 | 8 | 53 |
| 2014 Mayo | 56 | 15 | 71 |
| 2014 Abril | 42 | 5 | 47 |
| 2014 Marzo | 44 | 7 | 51 |
| 2014 Febrero | 27 | 5 | 32 |
| 2014 Enero | 34 | 4 | 38 |
| 2013 Diciembre | 20 | 5 | 25 |
| 2013 Noviembre | 23 | 6 | 29 |
| 2013 Octubre | 20 | 3 | 23 |
| 2013 Septiembre | 131 | 5 | 136 |
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