Imaging: Pulmonary Nodules |

Whipple Disease Revealed by Lung Involvement: A Case Report FREE TO VIEW

Aiping Song, PhD; Jiping Da, MD
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China-Japan Friendship Hospital, Beijing, China

Copyright 2016, American College of Chest Physicians. All Rights Reserved.

Chest. 2016;149(4_S):A252. doi:10.1016/j.chest.2016.02.263
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SESSION TITLE: Pulmonary Nodules

SESSION TYPE: Case Report Slide

PRESENTED ON: Saturday, April 16, 2016 at 01:00 PM - 02:00 PM

INTRODUCTION: WD is a rare multi-systemic disorder caused by Thropheryma whipplei, a Gram-positive bacillus. Gastrointestinal manifestations are the most frequent, but many other organs may be involved. We report a case of WD presenting with lung nodule in which shortness of breath and wheezing were the only manifestations.

CASE PRESENTATION: A 44-year-old woman with a history of tuberculosis 5 years ago, reported shortness of breath and wheezing, CT scan disclosed a nodule, did right pulmonary wedge resection, histological examination of which led to the diagnosis of WD. Postoperative pathology showed non-caseating granuloma with clustered periodic acid-Schiff (PAS)-positive macrophages. Immunoreactivity with anti-T whipplei antibodies confirmed the presence and suggested the involvement of T whipplei in the pathogenesis of the presenting lung disease. The patient was treated with oral Sulfonamides for 20 months to prevent recurrence.

DISCUSSION: In addition to our case, we identified nine published cases of WD with pulmonary involvement without GI symptoms. Including the present case the ten cases comprised eight men and two women, with a mean age of 49.5 years. WD and pulmonary tuberculosis have been diagnosed in this patient. Hypothesis that patients with WD are cellular immune defects could explain the association with tuberculosis, which could partially explain why women and all races are susceptible to this disease. This patient received the diagnosis before the onset of intestinal manifestations. In patients presenting with lung involvement associatedwith arthralgia and fever, sarcoidosis is a possible alternative diagnosis. In cases of suspected WD with or without pulmonary involvement, duodenal biopsy with PAS staining is indicated, even in the absence of intestinal clinical symptoms. In our case, the bronchial location of the bacterium was confirmed by immunohistochemical analysis with anti-T whipplei antibodies.

CONCLUSIONS: It is important to be aware that WD may have atypical presentations. Even with the major scientific improvements in the past decades, diagnosis of WD is still diffcult due to its rarity and nonspecific clinical presentations. It is important to be aware that PAS positivity is not specific for T. whipplei, could also be seen in mycobacterial infection. Acid‐fast stain is negative in WD and is useful to exclude tuberculosis. In conclusion, even in the absence of GI symptoms and without history of unexplained arthralgia and/or fever, a diagnosis of WD should be considered with lung nodules if they have evidence of atypical granuloma.

Reference #1: Urbanski G, Rivereau P, Artru L, Fenollar F, Raoult D, Puéchal X. Whippledisease revealed by lung involvement: a case report and literature review. Chest. 2012 Jun;141(6):1595-8. doi: 10.1378/chest.11-1812. Review. PubMed PMID:22670021

DISCLOSURE: The following authors have nothing to disclose: Aiping Song, Jiping Da

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