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

In: Pulmonary function

8 Mar 2014

The anemia continued to worsen with the hemoglobin level dropping to 70 g/L. Direct and indirect antiglobulin tests, negative prior to phenytoin treatment, were positive with anticomplement C3d and negative with anti-IgG. Saline-reactive cold agglutinins widi anti-I specificity of low-titer, high thermal amplitude were present, reacting in saline with adult RBCs at a titer of 1:512 at 4°C and 1:4 at 30°C; with albumin, the titer was 1:8 at 30°C. Nucleated RBCs, occasional cell fragments and spherocytes, and 1 to 2% atypical lymphocytes were found on a smear. Transfusion of packed RBCs following crossmatch by prewarm technique failed to show the appropriate rise in hemoglobin and was followed by further transfusion. The coagulopathy worsened (prothrombin time, 20 s; partial thromboplastin time, 35 s; international normalized ratio, 3.0) and was unresponsive to vitamin K, but did improve following administration of fresh-frozen plasma. Factor analysis showed hepatocellular injury.
An open-lung biopsy specimen showed myxoid organizing granulation tissue present in bronchioles, which was consistent with BOOP (Fig 2, tap). A mild increase in eosinophils within a chronic mononuclear inflammatory infiltrate was present in the alveolar exudate (Fig 2, bottom,). Lung tissue was negative for acid-fast bacilli, fungi, P carinii pneumonia, vasculitis, or granu-lomata. Electron microscopy was negative for viral inclusions. BAL culture later grew rhinovims. Methylprednisolone sodium succinate (Solu-Medrol), 60 mg, was administered intravenously every 6 h for 48 h and then followed by a slow intravenous taper over 1 week. This was followed by high-dose treatment with prednisone (1 mg/kg) for 3 months, followed by a very slow taper for a total steroid course of 1 year. Pulmonary function tests done 10, 21, and 42 days after steroid administration showed a resolving restrictive lung defect and reduced diffusing capacity of carbon monoxide. A chest x-ray film, liver function, and all hematologic abnormalities resolved. cialis professional

Figure 2. Top: histopathologic findings of open-lung biopsy specimen showing myxoid granulation tissues in bronchioles, extending to alveolar ducts and alveoli with confluent areas (arrowheads) consistent with BOOP. Both the pleura (p) and lobular septum (s) are congested and edematous (hematoxylin-eosin, original X25). Bottom: higher magnification of Figure 2, top, showing chronic inflammatory cellular infiltrate with a few eosinophils (arrowheads). Asterisk indicates myxoid connective tissue background (original X250).
Figure 2. Top: histopathologic findings of open-lung biopsy specimen showing myxoid granulation tissues in bronchioles, extending to alveolar ducts and alveoli with confluent areas (arrowheads) consistent with BOOP. Both the pleura (p) and lobular septum (s) are congested and edematous (hematoxylin-eosin, original X25). Bottom: higher magnification of Figure 2, top, showing chronic inflammatory cellular infiltrate with a few eosinophils (arrowheads). Asterisk indicates myxoid connective tissue background (original X250).


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