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An Interesting Case of Dyspnea: Recurrence of Disseminated Whipple’s Disease
Description
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A3607 - An Interesting Case of Dyspnea: Recurrence of Disseminated Whipple’s Disease
Author Block: J. Virk1, K. Yashi1, A. Ghimire2; 1Internal Medicine, SUNY Upstate Medical University, Syracuse, NY, United States, 2Pulmonary and Critical Care, SUNY Upstate Medical University, Syracuse, NY, United States.
Introduction
Whipple’s disease is a rare multi-system disease and usually requires a long treatment with anti-microbial agents. We present a case of neurological Whipple’s disease with pulmonary recurrence 12 years after completion of treatment.
Case
A 35 year old male with history of neurological whipple’s disease adequately treated 12 years back presents with new onset shortness of breath and generalized fatigue. He does not have fever or chills but has a dry cough. He has also experienced a 10 pounds weight loss. His vital signs are normal and pulmonary exam reveled diffuse fine crackles bilaterally. Initial lab work shows White blood cell count: 6500/uL, Creatinine: 0.8mg/dL, Calcium: 9.3mg/dL and normal inflammatory markers. A CT thorax was obtained that showed mediastinal lymphadenopathy. A lymph node biopsy was obtained and showed non necrotizing granulomatous lymphadenitis with no organism seen on acid fast stain or fungal stains. Blood culture and tissue cultures were negative for any bacterial/ fungal/ mycobacterial organisms. A Periodic Acid-Schiff stained macrophages were present but Tropheryma whipplei PCR (polymerase chain reaction) was negative. Pulmonary function test showed a restrictive ventilatory defect with decrease in diffusing capacity. He was started on intravenous (IV) ceftriaxone as empiric coverage and he showed a marked improvement in symptoms within 2 weeks. He received 4 weeks of IV ceftriaxone followed by oral doxycycline and hydoxychloroquine for 1 year and his symptoms completely resolved. A repeat CT thorax was obtained and showed complete resolution of mediastinal lymphadenopathy. Pulmonary function tests were repeated and a complete resolution of restrictive ventilatory defect was seen. The patient was followed for 2 years after the completion of treatment and he had no recurrence of symptoms.
Discussion
This case is unique as it shows that recurrence of Whipple’s disease can occur even after appropriate therapy and after extended periods of remission (12 years in our case). Furthermore, this case depicts that PCR can be unreliable in this setting and an empiric treatment should be started to determine symptoms resolution. Hence, a high degree of suspicion should be maintained to diagnose and treat recurrence Whipple’s disease.
Author Block: J. Virk1, K. Yashi1, A. Ghimire2; 1Internal Medicine, SUNY Upstate Medical University, Syracuse, NY, United States, 2Pulmonary and Critical Care, SUNY Upstate Medical University, Syracuse, NY, United States.
Introduction
Whipple’s disease is a rare multi-system disease and usually requires a long treatment with anti-microbial agents. We present a case of neurological Whipple’s disease with pulmonary recurrence 12 years after completion of treatment.
Case
A 35 year old male with history of neurological whipple’s disease adequately treated 12 years back presents with new onset shortness of breath and generalized fatigue. He does not have fever or chills but has a dry cough. He has also experienced a 10 pounds weight loss. His vital signs are normal and pulmonary exam reveled diffuse fine crackles bilaterally. Initial lab work shows White blood cell count: 6500/uL, Creatinine: 0.8mg/dL, Calcium: 9.3mg/dL and normal inflammatory markers. A CT thorax was obtained that showed mediastinal lymphadenopathy. A lymph node biopsy was obtained and showed non necrotizing granulomatous lymphadenitis with no organism seen on acid fast stain or fungal stains. Blood culture and tissue cultures were negative for any bacterial/ fungal/ mycobacterial organisms. A Periodic Acid-Schiff stained macrophages were present but Tropheryma whipplei PCR (polymerase chain reaction) was negative. Pulmonary function test showed a restrictive ventilatory defect with decrease in diffusing capacity. He was started on intravenous (IV) ceftriaxone as empiric coverage and he showed a marked improvement in symptoms within 2 weeks. He received 4 weeks of IV ceftriaxone followed by oral doxycycline and hydoxychloroquine for 1 year and his symptoms completely resolved. A repeat CT thorax was obtained and showed complete resolution of mediastinal lymphadenopathy. Pulmonary function tests were repeated and a complete resolution of restrictive ventilatory defect was seen. The patient was followed for 2 years after the completion of treatment and he had no recurrence of symptoms.
Discussion
This case is unique as it shows that recurrence of Whipple’s disease can occur even after appropriate therapy and after extended periods of remission (12 years in our case). Furthermore, this case depicts that PCR can be unreliable in this setting and an empiric treatment should be started to determine symptoms resolution. Hence, a high degree of suspicion should be maintained to diagnose and treat recurrence Whipple’s disease.
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