Hypovitaminosis C as an unexpected cause of bleeding in a cirrhotic patient

Sogbe, Miguel; Iñarrairaegui, Mercedes; Alegre, Félix

doi:10.1016/j.medcle.2021.12.007

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Partially thrombosed false lumen." ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Joaquín Valle Alonso, Leandro Noblia, Ana Esther Perez Diaz" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Joaquín Valle" "apellidos" => "Alonso" ] 1 => array:2 [ "nombre" => "Leandro" "apellidos" => "Noblia" ] 2 => array:2 [ "nombre" => "Ana Esther Perez" "apellidos" => "Diaz" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020622002789?idApp=UINPBA00004N" "url" => "/23870206/0000015800000012/v1_202206230718/S2387020622002789/v1_202206230718/en/main.assets" ] "en" => array:14 [ "idiomaDefecto" => true "cabecera" => "Letter to the Editor" "titulo" => "Hypovitaminosis C as an unexpected cause of bleeding in a cirrhotic patient" "tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "e17" "paginaFinal" => "e18" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Miguel Sogbe, Mercedes Iñarrairaegui, Félix Alegre" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Miguel" "apellidos" => "Sogbe" "email" => array:1 [ 0 => "msogbe@unav.es" ] "referencia" => array:3 [ 0 => array:2 [ "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Mercedes" "apellidos" => "Iñarrairaegui" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "b" "identificador" => "aff0010" ] 1 => array:2 [ "etiqueta" => "c" "identificador" => "aff0015" ] ] ] 2 => array:3 [ "nombre" => "Félix" "apellidos" => "Alegre" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "b" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Internal Medicine, Clínica Universidad de Navarra, Pamplona, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Liver Unit, Clínica Universidad de Navarra, Pamplona, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Centro de Investigación Biomédica en Red de Enfermedades Hepáticas y Digestivas (CIBEREHD) – IDISNA, Pamplona, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Hipovitaminosis C como causa inesperada de hemorragia en un paciente con cirrosis hepática" ] ] "textoCompleto" => "Hypovitaminosis C, or scurvy, is historically associated with sailors on lengthy voyages. Currently, it is a rare cause of bleeding in developed countries, although it may be underestimated in patients with risk factors such as alcoholism and cirrhosis.<a class="elsevierStyleCrossRefs" href="#bib0030">1,2</a> Based on clinical presentation and nutritional assessment, a scurvy diagnosis should be suspected, and low serum levels confirm the diagnosis.<a class="elsevierStyleCrossRef" href="#bib0040">3</a>The objective of this letter to the editor is to describe a rare cause of bleeding in a cirrhotic patient with risk factors for scurvy.A 65-year-old male with active alcoholism and Child-Pugh class B9 cirrhosis with a past medical history of heavy smoking habit and mild COPD was referred to our hospital because of a 2-month history of recurrent oropharyngeal bleeding. Bleeding was spontaneous and manifested when coughing or vomiting. The patient had undergone multiple outpatient and inpatient consultations, lab tests, imaging techniques (Angio-CT scans), and endoscopic studies (both ENT (Ear-Nose-Throat) fiberscopes and upper gastrointestinal endoscopies). No active bleeding point was observed. Esophageal varices III/IV were detected, and prophylactic banding was performed. Despite this procedure, recurrent oropharyngeal bleeding persisted, requiring frequent blood transfusions, a total of 15, due to severe anemia. At admission, the patient was conscious and had stable vital signs. Oral cavity examination showed an edentulous patient with hematic residues in the oropharynx without active bleeding. An abdominal exam revealed hepatomegaly without palpable splenomegaly. Mild jaundice and hematomas in the arms and legs were evident. Laboratory findings at admission showed hemoglobin 8.6g/dL, the mean corpuscular volume of 100fL, Platelets 92·10E9/L, and white blood cell count 3.5·10E9/L. Total bilirubin 3.41mg/dL, direct bilirubin 1.6mg/dL, albumin 3.67g/dL, prothrombin time 28s, INR 2.1 and fibrinogen 103mg/dL were also observed. Iron studies, vitamin B12, and folate were within normal ranges. During admission, he presented several episodes of oropharyngeal bleeding (≤100cc) without hemodynamic repercussion or need of blood transfusion. Angio-CT scan and new endoscopic procedures (ENT fibroscopy, upper gastrointestinal endoscopy, and bronchoscopy) were performed without identifying an active bleeding point. In upper GI endoscopy, hematic residues at the hypopharynx and friable mucosa were observed. Given the clinical presentation with diffuse superficial hematomas and easy mucosal bleeding, the diagnosis of hypovitaminosis C was suspected, in addition to other additional factors such as coagulopathy associated with his advanced liver disease. Scurvy was confirmed by a reduced serum vitamin C level (0.27mg/dL, normal range: 0.4–2mg/dL). A detailed review of the patient's dietary habits revealed a low fruit and vegetable intake. Intravenous vitamin C infusion was started (1g/d for 3 days followed by oral intake of 1g once daily). Oropharyngeal bleeding resolved within a few days after vitamin C supplementation. In the follow-up visit after one month, improvement in hemoglobin and vitamin C (0.7mg/dL) values were observed.Humans are among the rare vertebrates who do not synthesize vitamin C. The only source of vitamin C is diet. Hypovitaminosis C may be secondary to lack of intake, decreased absorption, and/or increased demand (such as oxidative states, like smoking). Alcoholic and cirrhotic patients are at risk for scurvy because of low intake and decreased intestinal vitamin C absorption.<a class="elsevierStyleCrossRefs" href="#bib0035">2,4</a> Vitamin C contributes to the maintenance of the soft tissue through collagen synthesis and elastic fiber stabilization. Hypovitaminosis C produces blood vessel fragility and poor wound healing, classifying scurvy as a disease of soft tissue.<a class="elsevierStyleCrossRefs" href="#bib0030">1,5</a> The diagnosis of scurvy is generally based on dietary history, and clinical features such as gingival bleeding, perifollicular hemorrhage, follicular hyperkeratosis with corkscrew hairs, and blood vessel fragility (which manifests as ecchymoses). A rapid resolution of signs and symptoms is seen after vitamin supplementation.<a class="elsevierStyleCrossRef" href="#bib0050">5</a> Laboratory investigations may be necessary to confirm less typical cases.<a class="elsevierStyleCrossRef" href="#bib0030">1</a> In addition, serum levels correlate with the risk of developing clinical symptoms.<a class="elsevierStyleCrossRef" href="#bib0040">3</a> The classic signs of hypovitaminosis C, such as gingival changes, are not always present (as in our patient, who was edentulous and received a soft-food diet), and the presence of other comorbidities may delay the diagnosis.<a class="elsevierStyleCrossRef" href="#bib0050">5</a>This case illustrates the need for a high index of suspicion for scurvy in patients with cirrhosis and coagulopathy who present mucosal bleeding, even when the classic signs of scurvy are missing.Source of fundingNone declared.Conflicts of interestNone declared." 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Zuily" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.amjmed.2017.03.016" "Revista" => array:6 [ "tituloSerie" => "Am J Med" "fecha" => "2017" "volumen" => "130" "paginaInicial" => "e387" "paginaFinal" => "e388" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/28389311" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Revisiting the pathobiology of scurvy: a review of the literature in the context of a challenging case" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "H. Deirawan" 1 => "J.W. Fakhoury" 2 => "M. Zarka" 3 => "M.H. Bluth" 4 => "M. Moossavi" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1111/ijd.14832" "Revista" => array:6 [ "tituloSerie" => "Int J Dermatol" "fecha" => "2020" "volumen" => "59" "paginaInicial" => "1450" "paginaFinal" => "1457" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/32154584" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Adult scurvy" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "J.V. Hirschmann" 1 => "G.J. Raugi" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/s0190-9622(99)70244-6" "Revista" => array:6 [ "tituloSerie" => "J Am Acad Dermatol" "fecha" => "1999" "volumen" => "41" "paginaInicial" => "895" "paginaFinal" => "910" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/10570371" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] "agradecimientos" => array:1 [ 0 => array:4 [ "identificador" => "xack613806" "titulo" => "Acknowledgements" "texto" => "None." 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