Steroid-responsive Alveolitis Associated with Ulcerative Colitis

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9 Mir-Sepasi MH, Cazzanigu AB, Bartlett RH . Surgical treatment of primary sternal osteomyelitis. Ann Thorne Surg 1975; 19:69.'l-

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1977; 137:1036-40 12 Roca RP, \hshikawa TT. Primary skeletal infectious in heroin users . Clin Orthop 1979 ; 144:238-48 13 Chandrasekar PH, Narula AP. Bone and joint infections in intravenous drug abusers. Rev Infect Dis 1986; 8:904-11 14 Whimbey E , Gold JWM, Polsky B, Dryjanski J. Hawkins C.

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patients with the acquired immune deficiency syndrome (AIDS) and AIDS-related complex . Am J Med 19.'l7; 82 :900-06

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Steroid-responsive Alveolitis Associated with Ulcerative Colitis Jolin M. S!III/'erson, D .M.t

Two patients with ulcerative colitis are reported in whom widespread alveolitis developed rapidly and responded promptly and completely to administration of prednisolone, (Chest 1992; 101:585-86)

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wide variety of systemic manifestations of ulcerative colitis have been recognized, but little attention has been paid to its pulmonary complications. These are thought to be due to immunologic abnormalities associated with the colitis. Bronchial inflammation is much more common than alveolar disorders, but the two subjects of this report developed an extensive, steroid-responsive alveolitis. CASE REPORTS CASt:

longt'r hreathh-ss and his prednisolom- dos,' coutiuu..d to 1)(' reduced .

In Decemher, W1l:3 . tlu- pntient d"vl'lop,'d s('n·....hlood\ diarrl...a and a histologiC' diaJ!nosis of ulcerativ» ('olitis was lIIad,- on n-etul hiopsy. 11(' li,il('d to respond 10 inteusive n...dk-al In'alln"nl iucluding an inl'n'asc' in pn-dnisolom- and n-quin«] a lolal '"01,'( '10111\', At no tune dnring this illnl'ss did his d u-st film show ahnonuallilldinJ!s or his respirutorv function dt'1(·rioralt· . Ilis pn-dniso]...... was t1isl"ntillllt,d in F('hruar}~ 1986 and h,· n-maim-d asvmptonuui« with a normal chest film findings and unt'hangt'd spirouu-try during 3'/! years of lollow-up. CASE

2

A 64-yt'ar-old retired t'ngineer presented in March, 191i7 with a four-month history of l"ngh and incn-asiru; hreathlessness. 11(' had contracted pulmonary tuberculosis in 1942 which n-lapsr-d in 1955 and 196.'3. at which time lit' received a full course of d ...moll ...rapy, 11(' had developed ulcerative l"litis in 1972. This diagnosb was established hy a rectal hiopsy and barium "IIt'ma which showed inllammntory changes in the- descending ('olon and n-etum . lit ' had received sulfasalaziue 2 g daily t"ntinuonsly from W76 . hul was taking no other medications.

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A 50-year-old communications consultant presented in Fehruary 19.'l2 with a three-week history of a febrile illness with headaches.

sweating am! increased breathlessness. Ill' had previously been well and was a nonsmoker and on no medication. Ill' was short of breath on slight exertion, and bilateral basal inspiratory crack It's were audible. There was no cyanosis or finger clubbing, Arterial Po, was 6 .0 kPa; Pco, 4.4 kPa; and FEV,IFVC 1.7/1.9 L (predicted 3 .75/ 4 .9). Chest radiograph revealed homogeneous hilateral hasal shadowing. Complete blood count showed no eosinophils. The ESH was R4 mm/h , blood cultures were sterile, and ANF. RA latex and viral and mycoplasma antibodies negative. Fiberoptic bronchoscopy result was normal, hut transhronchial biopsies showed cellular interstitial infiltration with little collagen formation and extensive cellular desquamation (Fig 1). in keeping with a diagnosis of cryptogenic fihrosing alveolitis. Ill' was treated initially with 60 mg prednisolone daily and within two weeks his FEV,IFVC had risen to 2.413.35 L and his chest xray appearance had improved considerably The dose was gradually reduced . After two months Ill' treatment. his FEV,IFVC was 2 .9/ ·Consultant Phvsician, Newmarket General Hospital, Suffolk, England. . Re/Jrint requests: Dr. Slineerson, Newmarket Generlll Hospital,

Neunnarket, Suffolk. f:ll{!,llInd

FIGllHE 2, Chest radiograph of case 2. CHEST I 101 I 2 I FEBRUARY, 1992

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On examination, there was no finger clubbing or central cyanosis, but be had bilateral basal late inspiratory crackles. His initial FEV,I FVC was 3.114.4 (predicted 3.314.9) L with a TLC of6.7 L (predicted 7.2), RV of 2.3 L (predicted 2.9) and KCO of 1.26 (predicted 1.11.6). His chest x-ray film showed patchy shadowing in both lower zones (F ig 2). He had no blood eosinophilia and ANF and RA latex test results were negative. Sputum was negative for acid-fast bacilli on three occasions. Fiberoptic bronchoscopy findin~ were normal , and transbronchial biopsies were nondtagncstic. He underwent an npen lung biopsy which showed interstitial fibrosis with fibroblasts and chronic inRammatory cells in the alveolar walls with type 2 cell hyperplasia and areas of fibro-myxoid connective tissue within the alveoli . There was no vasculitis or granuloma. He was treated with prednisolone 40 mg daily, and after one month , his chest x-ray film findin~s had returned to normal. His FEV,IFVC was then 3 .115.2 L with a TLC of7.5 L. RV 2.3 Land KCO 1.17. His prednisolone dose was gradually reduced until it was discontinued in July, 1988. He has remained asymptomatic with normal chest x-ray findings during two years of follow-up, during which time he has continued to take sulfasalazine 2 g daily. He has had no exacerbations of his ulcerative colitis since his chest symptoms appeared. DISCUSSION

Bronchial in8ammation' and bullae' are well recognized in association with ulcerative colitis, but diffuse chest x-ray film shadowing is much less common. It may be due to vasculitis" or an eosinophilic infiltrate which may' or may not' be caused by sulfasalazine. The few previous case reports resembling cryptogenic fibrosing alveolitis either do not include histologic confirmation of colitis' or alveolitis,7 or involve predominantly the upper zones." In case I, the diagnosis of alveolitis was established histologically and the illness took an acute form. The patient responded unusually quickly and completely to a course of prednisolone and did not suffer a relapse when his ulcerative colitis appeared almost two years later or during subsequent follow-up. Patient 2 differed slightly in that the chest x-ray film shadowing was more patchy, although it was also mainly in the lower zones. Sulfasalazine is unlikely to have been the cau se of this shadowing since he had no eosinophilia, the drug had been taken for 11 years, and administration continued following his chest illness without it recurring. His symptoms, chest x-ray film findings and respiratory function improved promptly after prednisolone was given, In neither of these patients did the appearance of the alveolitis coincide with an exacerbation of the colitis. The natural history of the alveolitis is uncertain, but it was extremely sensitive to prednisolone and did not show relapse after treatment was discontinued . Complete radiologic resolution of "cryptogenic" fibrosing alveolitis is unusual and these patients suggest that if this does occur, the alveolitis may be associated with the presence or subsequent development of ulcerative colitis. REFERENCES

Butland RJA, Cole P, Citron KM, Turner-Warwick M. Chronic bronchial suppuration and inflammatory bowel disease. Quart J Med 1981; 50:63-75 2 Shneerson JM. Lung bullae, bronchiectasis and Hashimotos disease associated with ulcerative colitis treated by colectomy. Thorax 1981; 36:313-14 3 Isenberg }1, Goldstein H, Korn AR, Ozeran RS, Rosen V.

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Pulmonary vasculitis-an uncommon complication of ulcerative colitis. Report of a case. N Engl J Med 1968; 279:1376-77 Jones GR , Malone DNS . Sulphasalazine-induced lung disease. Thorax 1972; 27:713-17 Trewby PN , Connolly CK. Reversible pulmonary disease and eosinophilia associated with sulphasalazine. J Royal Soc Med 1988; 81:558 Turner-Warwick M. Fibrosin~ alveoli tis and chronic liver disease. Quart J Med 1968; 37:133-49 Snook lA, de Silva HJ, Jewell DP. The association of autoimmune disorders with inRammatory bowel disease. Quart J Med 1989; 72:835-40 Heinrich R, Riedel L, Schomerus H . Bronchopulmonale infiltrate bei chronisch entzundlicher darmerkrankung, Dtsch med Wschr 1983; 108:1106-10

Complex Cryptococcal Empyema* Michael F. Tenholder; M.D., F.C.C.P.;t

Frank W Ewald,Jr., M.D ., F.C.C .P.;:j: Nayereh K. Khankhan/an, M.D .;§ and john H. Crosby, M.D .§

CryptocOCCWl

ne%rmDnlI continues to present diagnostic and treatment challenges in patients with underlying malignant neoplasms. Cryptococcal empyema is a relatively rare complication of cryptococcal disease. It is important to distinguish whether uncontrolled malignancy or cryptococcal infection is responsible for the effusion. We used traditional diagnostic approaches, bronchoscopy and transthoracic fine needle aspiration, to verify the presence of the organism but continued to have treatment failure until adequate drainage was established. (Chest 1992; 101:586-88)

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=transthoracic Hne needle aspiration

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is a ubiquitous fungus found in the soil. On inhalation, a complex, incompletely understood series of host responses begins that determines whether the infection will be controlled or will progress to local or disseminated disease.' The frequency of cryptococcaldisease in steroid-treated individuals, allograft recipients, and acquired immunodeficiency syndrome (AIDS) victims highlights the importance ofT lymphocyte-dependent host defenses.' We are witnessing an evolution in pulmonary cryptococcal infection, both in clinical presentations' and advances in diagnostic laboratory testing, as illustrated by the following case. ryptococcus ne%rowns

CASE REPORT

A 46-year-old man was admitted to the hospital with a chest roentgenogram with multiple pleural-based nodules considered suspicinus for metastatic cancer. A year earlier, he had undergone total laryngectomy and left radical neck dissection for sq uamous cell carcinoma of the larynx . He was not otherwise immunocompro-

mised. Bronchial brushin~ and a transhronchial biops y specimen from ·From the Medical College of Georgia and the VA Medical Center, Augusta, t Associate Professor of Medicine. :j:Assistant Professor of Medicine. §Associate Professor of Pathology. This work was supported by the Research Service of the Veterans Affairs Medical Center, Augusta, Ceorgia. ComplexCryptoooccal Empyema (Tenholderel aI)