Chronic bilateral panuveitis as first manifestation of Whipple's disease

Arriola-Villalobos, Pedro; Ruiz-Medrano, Jorge; Diaz-Valle, David; Benitez del Castillo, Jose M.

doi:10.1016/j.medcle.2016.06.041

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"apellidos" => "Benitez del Castillo" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "b" "identificador" => "aff0010" ] 1 => array:2 [ "etiqueta" => "c" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Servicio de Oftalmología, Hospital Clínico San Carlos, Instituto de Investigación Sanitaria del Hospital Clínico San Carlos (IdISSC), Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Cooperative Research Network on Age-Related Ocular Pathology, Visual and Life Quality, Instituto de Salud Carlos III, Madrid, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Oftalmología, Departamento de Oftalmología y Otorrinolaringología, Hospital Clínico San Carlos, Facultad de Medicina, Universidad Complutense de Madrid, Instituto de Investigación Sanitaria del Hospital Clínico San Carlos (IdISSC), Madrid, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Panuveítis crónica bilateral como primera manifestación de la enfermedad de Whipple" ] ] "textoCompleto" => "Ocular involvement in Whipple's disease is rare (2–5% of patients, depending on the series),<a class="elsevierStyleCrossRef" href="#bib0050">1</a> and it mainly manifests as uveitis, optic neuropathy and macular oedema.<a class="elsevierStyleCrossRefs" href="#bib0055">2–4</a> Ocular problems can also be secondary to a central nervous system (CNS) impairment, and may manifest as oculomotor palsy, nystagmus and ophthalmoplegia.<a class="elsevierStyleCrossRef" href="#bib0065">4</a> Ocular involvement usually occurs late in the disease and it is associated with gastrointestinal<a class="elsevierStyleCrossRef" href="#bib0070">5</a> and/or neurological manifestations. Ocular involvement non-secondary to CNS disorder in the absence of intestinal manifestations is very rare, with few cases described in literature.<a class="elsevierStyleCrossRefs" href="#bib0075">6–9</a> Despite being very unusual, it may be the first manifestation of Whipple's disease, making the already complex diagnosis of this entity even more difficult.We report the case of a 59-year-old male without systemic symptoms and an 18-month history of anterior uveitis, peripheral retinal vasculitis and bilateral macular oedema with poor response to multiple anti-inflammatory and immunosuppressive treatments at local and systemic level. A complete etiologic study was conducted, including serology, autoimmunity tests and imaging tests, but it was not sufficient to establish a causal diagnosis. After a month of mild gastrointestinal symptoms, the patient was referred to internal medicine due to severe diarrhoea as well as fatigue and weight loss of 3kg. An endoscopic capsule was performed, reported as lymphangiectasis, affecting the entire small intestine. Days later, by scheduled jejunoscopy, 3 samples of jejunal mucosa were taken. The histopathologic study was suggestive of Whipple’ disease, as there were numerous macrophages with negative PAS+ and Ziehl–Neelsen staining in the small intestine mucosa layer. The combination of compatible clinical and pathological anatomy confirmed the diagnosis of Whipple's disease, so antibiotic therapy was started with intramuscular ceftriaxone (2g daily for 2 weeks) and cefixime (400mg/day, orally, for one year). During the subsequent months, there was an overall improvement, with weight gain and gradual disappearance of gastrointestinal and joint symptoms. A new capsule endoscopy was performed, showing a significant improvement, with almost complete disappearance of the intestinal lymphangiectasis. The retinal vasculitis also improved; that wasn’t the case of the macular oedema, which persisted due to the development of an epiretinal membrane.The pathogenesis of ocular manifestations in Whipple's disease remains unclear.<a class="elsevierStyleCrossRefs" href="#bib0060">3,5</a> Some authors uphold an immune mechanism, which assumes that the ocular manifestations are due to a hypersensitivity phenomenon. Another plausible theory is that there is a bacterial invasion in the eye, as the presence of the bacterium was detected by PCR in vitreous samples.<a class="elsevierStyleCrossRefs" href="#bib0075">6–9</a> Even then, the presence of bacterial products, as determined by PCR, does not demonstrate that the bacteria are responsible for the ocular signs and symptoms.Late diagnosis is very common in Whipple's disease due to the large number of non-specific signs and symptoms with which it manifests itself. And it is even more delayed in patients with an unusual presentation of the disease. Ocular involvement is not usually the first manifestation of Whipple's disease.<a class="elsevierStyleCrossRefs" href="#bib0075">6,7</a> Nevertheless, in the case we are reporting, the first symptoms of Whipple's disease were ocular manifestations. The diagnosis of the disease was done almost 2 years after the onset of ocular symptoms.In short, this case illustrates, on one hand, the need to include Whipple's disease in the differential diagnosis of chronic bilateral panuveitis, given its harmful progression (it is life threatening) and good response to antibiotic treatment of systemic and ocular manifestations. This consideration is even more important in long term cases which are refractory to immunosuppressive treatment. In these cases, an infectious cause should always be ruled out. On the other hand, this case also shows that Whipple's disease can start with unusual symptoms, far from the typical insidious pattern of joint pain, diarrhoea and weight loss.<a class="elsevierStyleCrossRef" href="#bib0050">1</a>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "Please cite this article as: Arriola-Villalobos P, Ruiz-Medrano J, Diaz-Valle D, Benitez del Castillo JM. Panuveítis crónica bilateral como primera manifestación de la enfermedad de Whipple. Med Clin (Barc). 2016;146:467–468." ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0050" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Whipple's disease" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "T. 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ISSN: 2387-0206

Launched in 1943, Medicina Clínica is a fortnightly journal aimed at the promotion of clinical research and practice among internal medicine and other specialists. The key characteristics of Medicina Clínica are the scientific and methodological rigor of its manuscripts, the topicality of its contents, and, especially, its practical focus with highly useful information for clinical practice. Medicina Clínica is predominantly interested in publishing original research manuscripts, which are rigorously selected according to their quality, originality, and interest, and also in continued medical education-oriented manuscripts, which are commissioned by the journal to relevant authors (Editorials, Reviews, and Diagnosis and Treatment). These manuscripts contain updated topics with a major clinical or conceptual relevance in modern medicine. The journal adheres to the standards of academic research publications in all aspects including peer-review and ethical principles. Medicina Clínica is included in the General and Internal Medicine category of Thomson Reuters.

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Current Contents/Clinical Medicine, Journal Citation Reports, SCI-Expanded, Index Medicus/Medline, Excerpta Medica/EMBASE, IBECS, IME, MEDES, PASCAL, SCOPUS, SciVerse ScienceDirect

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Impact factor

The Impact Factor measures the average number of citations received in a particular year by papers published in the journal during the two preceding years.

© Clarivate Analytics, Journal Citation Reports 2022

Impact factor 2023

2.6

Citescore

CiteScore measures average citations received per document published.

Citescore 2023

3.1

SJR

SRJ is a prestige metric based on the idea that not all citations are the same. SJR uses a similar algorithm as the Google page rank; it provides a quantitative and qualitative measure of the journal's impact.

SJR 2023

0.371

SNIP

SNIP measures contextual citation impact by wighting citations based on the total number of citations in a subject field.

SNIP 2023

0.511

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Journal Information

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Vol. 146. Issue 10.

Pages 467-468 (May 2016)

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