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IPH is rare in adults, predominantly affecting children. Co-occurrence with celiac disease (CD) is rare and is called Lane-Hamilton syndrome.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> A significant improvement can be obtained with a gluten-free diet (GFD), both from the point of view of intestinal and pulmonary symptoms.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of a 52-year-old patient with history of dyslipidemia, arterial hypertension, smoking, microcytic chronic anemia secondary to internal hemorrhoids and a deep vein thrombosis after a bone fracture surgery. He was on rivaroxaban treatment.</p><p id="par0015" class="elsevierStylePara elsevierViewall">He consulted in the emergency department with dyspnea and mild hemoptysis. A CT scan was performed showing bilateral alveolar hemorrhage. Bronchoscopy and bronchoalveolar lavage were also compatible with alveolar hemorrhage. Treatment with methylprednisolone was started during admission, with a subsequent tapering regimen with oral prednisone at discharge.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Bleeding was limited after the withdrawal of anticoagulation, and blood tests showed iron deficiency, so treatment with oral iron was started.</p><p id="par0025" class="elsevierStylePara elsevierViewall">One year later, the patient was admitted again for a new episode of hemoptysis. Treatment with methylprednisolone and a tapering regimen of oral prednisone was started again. A new bronchoscopy was performed. It was consistent with alveolar hemorrhage, with 100% of the macrophages with positive Perls stain. The complete study including autoimmunity was negative and no other organs were affected, so a lung biopsy was finally performed, which showed diffuse alveolar hemorrhage without evidence of inflammation or neoplasia.</p><p id="par0030" class="elsevierStylePara elsevierViewall">During follow-up, a CD study was performed. Serum IgA antibodies to tissue transglutaminase and antiendomisin <span class="elsevierStyleItalic">antibodies were</span> positive. An intestinal biopsy showed partial villous atrophy and profuse intraepithelial lymphocytosis CD3+ over 50 per 100 epithelial cells, resulting in a type 3a Marsh celiac disease. Therefore, the diagnosis of Lane-Hamilton syndrome was established and a GFD was started with good tolerance and without immediate relapses. Ferropenic anemia, folate, vitamin B12 and D deficiency progressively improved too.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Again, still on treatment with oral prednisone, the patient went to the emergency department for a new episode of hemoptysis that was finally diagnosed as a recurrence of the alveolar hemorrhage. The patient reported not being on the GFD in the last two months before the episode. At the present time, he has restarted the GFD and has not suffered new episodes of alveolar hemorrhage during follow-up.</p><p id="par0040" class="elsevierStylePara elsevierViewall">IPH is a clinical picture of unknown etiology characterized by hemoptysis, diffuse alveolar infiltrates and iron deficiency anemia.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> It is diagnosed by ruling out other possible causes and when the aforementioned clinical triad is present.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The association of IPH with CD is known as Lane-Hamilton syndrome and was first described in 1971.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> It is most common in children under 15 years of age; however, cases of Lane-Hamilton syndrome have also been reported in adults.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1–3</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Due to this association, it's been proposed that patients with idiopathic prolonged hemosiderosis should be screened for CD, even in the absence of gastrointestinal symptoms<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> as it happened to our patient.</p><p id="par0055" class="elsevierStylePara elsevierViewall">There is not much evidence on the correct treatment of Lane-Hamilton syndrome. Due to its rare occurrence, treatment is based on anecdotal case reports and case series. However, they all have in common the importance of a strict GFD.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> Patients presenting with Lane-Hamilton syndrome show satisfactory improvement of pulmonary and gastrointestinal symptoms with a GFD.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> In addition, especially in adult patients, high-dose corticosteroids have shown to reduce morbidity and mortality and delay or halt disease progression.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> Alveolar hemorrhage could be explain as an immune-mediated condition more than a coagulation impairment due to malabsorption of vitamin K.</p><p id="par0060" class="elsevierStylePara elsevierViewall">Although many previous studies suggest that treatment of CD may lead to remission of IPH, it should be noted that remission of both IPH and CD achieved with a GFD in CD patients is unclear, as the patients also received concomitant corticosteroids.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> However, the reappearance of IPH symptoms in a patient who stopped GFD, despite continued corticosteroid therapy, supports a gluten-dependent immune mechanism. This was previously suggested in a pediatric case series<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> and also described in another cause of IPH such as Heiner syndrome.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">In conclusion, this case illustrates that IPH with CD, which constitutes the Lane-Hamilton syndrome, can appear in adult patients even in absence of a previous history of digestive disease, and a poor compliance with the GFD can result in clinical recurrence of both malabsorptive and immune-mediated diseases such as alveolar hemorrhage.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical considerations</span><p id="par0070" class="elsevierStylePara elsevierViewall">Written informed consent was obtained from the patient included in the publication.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Informed consent</span><p id="par0075" class="elsevierStylePara elsevierViewall">Written informed consent was obtained from all individual participants included in the study.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Funding</span><p id="par0080" class="elsevierStylePara elsevierViewall">No funding was required to carry out the study.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conflict of interest</span><p id="par0085" class="elsevierStylePara elsevierViewall">All authors declare that they have no conflict of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:5 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Ethical considerations" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Informed consent" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Funding" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflict of interest" ] 4 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Idiopathic pulmonary haemosiderosis with celiac disease (Lane-Hamilton syndrome) in an adult – a case report" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "N. Berger" 1 => "J. Nichols" 2 => "D. Datta" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/crj.12258" "Revista" => array:6 [ "tituloSerie" => "Clin Respir J" "fecha" => "2016" "volumen" => "10" "paginaInicial" => "661" "paginaFinal" => "665" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/25586081" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0035" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Lane-Hamilton syndrome: association or coincidence" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "S. Paksu" 1 => "M.S. Paksu" 2 => "A.G. Kalayci" 3 => "R. Sancak" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Indian Pediatr" "fecha" => "2012" "volumen" => "49" "paginaInicial" => "243" "paginaFinal" => "244" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/22484746" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0040" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Idiopathic steatorrhoea and idiopathic pulmonary haemosiderosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "D.J. Lane" 1 => "W.S. Hamilton" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1136/bmj.2.5753.89" "Revista" => array:6 [ "tituloSerie" => "Br Med J" "fecha" => "1971" "volumen" => "2" "paginaInicial" => "89" "paginaFinal" => "90" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/1276823" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Screening for celiac disease in idiopathic pulmonary hemosiderosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "M. Khemiri" 1 => "M. Ouederni" 2 => "F. Khaldi" 3 => "S. Barsaoui" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.gcb.2008.05.010" "Revista" => array:6 [ "tituloSerie" => "Gastroenterol Clin Biol" "fecha" => "2008" "volumen" => "32" "paginaInicial" => "745" "paginaFinal" => "748" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/18603390" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "A different clinical presentation of Heiner syndrome: the case of diffuse alveolar hemorrhage causing massive hemoptysis and hematemesis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "A.S. Koc" 1 => "A. Sucu" 2 => "U. Celik" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.rmcr.2019.01.019" "Revista" => array:5 [ "tituloSerie" => "Respir Med Case Rep" "fecha" => "2019" "volumen" => "23" "paginaInicial" => "206" "paginaFinal" => "208" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/00257753/0000016000000001/v1_202212290520/S0025775322004559/v1_202212290520/en/main.assets" "Apartado" => array:4 [ "identificador" => "66430" "tipo" => "SECCION" "es" => array:2 [ "titulo" => "Cartas al Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "es" ] "PDF" => "https://static.elsevier.es/multimedia/00257753/0000016000000001/v1_202212290520/S0025775322004559/v1_202212290520/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0025775322004559?idApp=UINPBA00004N" ]
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