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Case study
Linear IgA Bullous Dermatosis With Laryngeal Involvement
Dermatosis ampollosa lineal IgA con afectación laríngea
Emilio Vives Ricomaa,
Corresponding author
emilio_vives89@hotmail.com

Corresponding author.
, Mahfoud El Uali Abeidaa, María Jesús Viso Sorianob, Rafael Fernández Liesaa
a Servicio de Otorrinolaringología, Hospital Universitario Miguel Servet, Zaragoza, Spain
b Servicio de Anatomía Patológica, Hospital Universitario Miguel Servet, Zaragoza, Spain
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combined in recent months with sulfasalazine 1&#8239;g&#47;8&#8239;h&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Despite this&#44; the patient was seen on numerous occasions in the emergency department for acute dyspnoea and stridor and treated with high-dose intravenous cortico-therapy and support measures&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">On examination &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#44; erosive blistering lesions&#44; some haemorrhagic&#44; were observed in the oropharynx&#44; larynx and hypopharynx&#46; The presence of erosive lesions in the nasal cavities was noteworthy&#44; with significant dryness of the mucous membranes and formation of scabs&#46; We also observed generalised atrophy of the turbinates and presence of intranasal synechiae&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">The abovementioned lesions were seen at the level of the larynx&#44; as well as some areas with fibrin deposit in the supraglottis&#44; in the arytenoid region and pyriform sinuses&#46; A moderate reduction of the laryngeal vestibule was also observed due to mucosal oedema and formation of synechia in the arytenoid region</p><p id="par0030" class="elsevierStylePara elsevierViewall">To date&#44; the patient has not required tracheotomy&#44; although due to the progression of the lesions after each outbreak and the worsening of his baseline dyspnoea&#44; it seems likely that it will be necessary in the future&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">IgA linear dermatosis is a chronic acquired bullous disease&#44; recognised as an entity in its own right since 1979 by Jablonska and Chorzelski&#44;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> differentiating it from dermatitis herpetiformis and bullous pemphigoid&#46; It has an annual incidence in Europe of &#46;5 cases per million inhabitants&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Its clinical presentation consists of the appearance of vesiculobullous lesions with a predilection for the extensor surfaces and with mucosal involvement in up to 70&#37; of cases and particularly oral and conjunctival involvement in the head and neck&#44; extension to the larynx is very rare&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">There are 2 age peaks&#46; There is a child form in the first years of life&#44; between 5 months and 6 years&#44; which usually disappears after puberty and more frequently affects the face&#44; genitals and thighs&#46; There is another peak in adulthood&#44; between the fourth and fifth decades of life&#44; with a predilection for the face&#44; trunk and limbs&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">In most cases it is of idiopathic origin&#44; although it has been associated with some drugs&#44; such as vancomycin&#44; NSAIDs&#44; captopril and amiodarone&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Gluten-sensitive enteropathy has been associated in 25&#37; and 33&#37; of cases&#44; but in a milder form than that observed in 90&#37; of patients with dermatitis herpetiformis&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">IgA autoantibodies target the proteins LAD-1 &#40;120&#8239;kD&#41; and LABD97 &#40;97&#8239;kD&#41;&#44; proteolytic fragments of glycoprotein BP 180 &#40;type XVII collagen&#41;&#44; located in the lamina lucida&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Its clinical presentation is heterogeneous&#44; usually in the form of tight blisters of varying size&#44; although it can also manifest itself in the form of papules and clear or haemorrhagic vesicles&#46; The lesions sit on skin of normal appearance or erythematous base&#44; with a generally symmetrical distribution&#46; They are usually located on the extensor surfaces of the limbs&#44; the trunk&#44; the buttocks and the face&#46; The mucous membranes can be involved in up to 70&#37; of cases&#44; particularly with oral and conjunctival involvement&#44; although on very rare occasions&#44; as in this patient&#44; the laryngeal mucosa may be affected&#44; even compromising the upper airway&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Although there are numerous cases described with involvement of the oral mucosa&#44;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#8211;8</span></a> we find few published studies of patients with IgA dermatitis with involvement of the oral mucosa&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">The histological study &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41; shows the presence of blisters in the subepidermal space that contain abundant neutrophils and a variable number of eosinophils&#44; as well as non-specific inflammatory infiltration in the dermis&#46; Less frequently&#44; micro-abscesses of neutrophils are observed in the dermal papillae&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0070" class="elsevierStylePara elsevierViewall">Direct immunofluorescence is the key to diagnosis&#44; observing the linear deposit of IgA along the basement membrane&#44; and enables a differential diagnosis with other bullous diseases&#46; IgA autoantibodies target proteins LAD-1 &#40;120&#8239;kD&#41; and LABD97 &#40;97&#8239;kD&#41;&#44; proteolytic fragments of the glycoprotein BP 180 &#40;type XVII collagen&#41;&#44; at the level of the lamina lucida&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">In some cases&#44; IgM and C3 deposits can also be detected along with IgA&#46; Indirect immunofluorescence is positive in adults in only 30&#37; of cases and at low titres&#44; which increases to 75&#37; in children&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">A differential diagnosis must be made with other dermatoses such as bullous or cicatricial pemphigoid&#44; dermatitis herpetiformis&#44; epidermolysis bullosa acquisita or even toxic epidermal necrolysis&#44; among others</p><p id="par0085" class="elsevierStylePara elsevierViewall">The treatment of choice for IgA linear dermatitis is dapsone or sulphonamides&#44; and systemic corticosteroids can be combined&#46; Other therapeutic options are immunosuppressants such as colchicine&#44; mycophenolate mofetil or cyclosporine&#46; In drug-induced cases&#44; discontinuation is usually sufficient&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of Interests</span><p id="par0090" class="elsevierStylePara elsevierViewall">The authors have no conflict of interests to declare&#46;</p></span></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Vives Ricoma E&#44; El Uali Abeida M&#44; Viso soriano MJ&#44; Fern&#225;ndez Liesa R&#46; Dermatosis ampollosa lineal IgA con afectaci&#243;n lar&#237;ngea&#46; Acta Otorrinolaringol Esp&#46; 2020&#59;71&#58;190&#8211;192&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Vesiculobullous&#44; erosive and haemorrhagic lesions&#44; oedema and areas with fibrin deposits in the nasal mucosa and laryngopharyngeal area are observed&#46; A&#41; Oropharynx&#46; B&#41; Right nostril&#46; C&#41; Lingual face of epiglottis&#46; D&#41; Oedema and interarytenoid synechia&#44; reduction of laryngeal vestibule&#46;</p>"
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">The patient&#8217;s skin biopsy&#46; A&#41; A subepidermal blister can be seen&#44; with epidermis of normal appearance and unaltered keratinisation&#46; The blister cavity contains fibrin with nuclear dust and neutrophils&#46; The floor of the blister corresponds to the papillary dermis showing perivascular infiltration of lymphocytes and interstitial infiltration of neutrophils&#44; with practically no eosinophils &#40;haematoxylin-eosin stain &#215;4&#41;&#46; B&#41; Direct immunofluorescence in which a linear band of IgA is observed in the basement membrane &#40;&#215;400 magnification&#41;&#46;</p>"
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