Bronchiolitis Obliterans With Organizing Pneumonia and Cold Agglutinin Disease Associated With Phenytoin Hypersensitivity Syndrome

In: Pulmonary function

6 Mar 2014

Bronchiolitis Obliterans With Organizing Pneumonia and Cold Agglutinin Disease Associated With Phenytoin Hypersensitivity SyndromeProvided here is the first description of histopathologic bronchiolitis obliterans with organizing pneumonia (BOOP) in a patient with severe phenytoin hypersensitivity syndrome (PHS). The presence of cold hemagglutinin disease was also documented for the first time. The patient showed dramatic improvement with high-dose steroid therapy. www.mycanadianpharmacy.com

Case Report
In September 1994, a 40-year-old black man underwent craniotomy and started receiving therapy with phenytoin sodium (Dilantin; Parke-Davis; Morris Plains, NJ), 300 mg daily. A medical history revealed chronic use of tobacco, crack cocaine, and alcohol. Results of a lung examination and a chest x-ray film showed no abnormalities. Four weeks following phenytoin therapy, clear rhinorrhea, myalgia, high fever, diffusely spreading pruritic maculopapular rash, and dry cough developed. The cough became productive and was associated with dyspnea.
Six weeks after phenytoin therapy was initiated, the patient was readmitted to the hospital. Vital signs were as follows: respiratory rate, 24 breaths per minute; heart rate, 112 beats per minute; BP, 100/60 mm Hg; and temperature, 38°C. Examination revealed diffuse skin exfoliation; conjunctivitis; facial edema; tender, enlarged lymph nodes; decreased breath sounds bilaterally with diffuse inspiratory crackles; mild right upper quadrant tenderness; and hepatomegaly.
Laboratory findings included a fall in hemoglobin level from 155 g/L (noted 6 weeks earlier prior to phenytoin treatment) to 133 g/L; WBC count, 29X109/L (35% eosinophils); subtherapeu-tic phenytoin level; mildly elevated liver function tests; while breathing room air, arterial blood gas values showed mild hypoxia; and chest radiograph, diffuse, patchy, asymmetric, re-ticulonodular infiltrates. Administration of broad-spectrum antibiotics and prednisone, 50 mg daily, was started. Phenytoin treatment was discontinued. Tests for cold agglutinins were positive. The Mantoux test and tests for HIV, heterophile agglutination, antinuclear antibody, rheumatoid factor, Legionella, and sputum cultures for bacteria, fungi, acid-fast bacilli, and Pneumocystis carinii pneumonia were negative. A skin biopsy specimen showed a drug eruption. Serological tests for Mycoplasma, adenovirus, influenza A and B, herpes simplex, cytomegalovirus, and hepatitis A, B, and C were negative. Spiking fevers, marked eosinophilia, and deterioration of liver function tests continued (aspartate aminotransferase, 214 U/L [normal, <40 U/L]; alanine aminotransferase, 247 U/L [normal, <35 U/L]; lactate dehydrogenase, 435 U/L [normal, <260 U/L]; alkaline phosphatase, 237 U/L [normal, <35 to 125 U/L]). The admission hemoglobin level of 133 g/L dropped to 101 g/L within 48 h of hospitalization, and a mild coagulopathy developed. Respiratory insufficiency culminated in ICU admission and intubation. A chest x-ray film revealed asymmetric patchy infiltrates at lung bases (Fig 1). Sepsis-like hemodynamics developed with hypotension; tachycardia (heart rate, 120 to 140 beats per minute); cardiac output, 10 to 17.5 L/min; and systemic vascular resistance, 300 to 500 dyne#s*cm~ (normal, 770 to 1,500 dyne scm~) requiring inotropic and fluid support. Bronchoscopy, BAL stains, cultures, and cytologic studies were negative for organisms. Urine was treated for a Staphylococcus aureus infection. All other cultures remained negative for organisms.

Figure 1. Chest x-ray film showing status after intubation in ICU; of note are diffuse patchy interstitial and alveolar infiltrates at bilateral (right greater than left) lung bases.

Figure 1. Chest x-ray film showing status after intubation in ICU; of note are diffuse patchy interstitial and alveolar infiltrates at bilateral (right greater than left) lung bases.


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