Tracheobronchitis with Dyspnea in a Patient with Ulcerative Colitis

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1 CASE REPORT Trcheoronchitis with Dyspne in Ptient with Ulcertive Colitis Msy Hiyoshi 1,2, Kzushige Kwi 2, Mihoko Shiuy 3, Tsuyoshi Ozw 2, Junko Kishikw 2, Tkko Nirei 2, Toshiki Tnk 2, Junichiro Tnk 2, Tomomichi Kiyomtsu 2, Tomohiro Td 2, Tkmitsu Knzw 2, Shinsuke Kzm 2, Hirofumi Shod 3, Shuji Sumitomo 3, Kne Kuo 3, Hironori Ymguchi 2, Soichiro Ishihr 2, Eiji Sunmi 2, Joji Kitym 2, Kzuhiko Ymmoto 3 nd Toshiki Wtne 1 Astrct We herein report the cse of 42-yer-old mn with one-yer history of ulcertive colitis who presented with excerted loody dirrhe, productive cough nd incresing rething difficulties. Colonoscopy reveled typicl deep ulcers in the rectosigmoid colon nd typicl multiple sucker-like ulcers in the trnsverse colon, nd computed tomogrphy of the chest demonstrted wll thickening of the trche nd ronchi. In ddition, ronchoscopy showed ulcers in the trche, nd histopthology disclosed findings of necrosis nd inflmmtion of the suepithelil tissue of the trche. Bsed on these findings, the ptient s respirtory symptoms were strongly suspected to e due to ulcertive colitis-relted trcheoronchitis. Tretment with systemic corticosteroids susequently resulted in rpid clinicl improvement. Key words: ulcertive colitis, trcheoronchitis, inflmmtory owel disese, extrintestinl mnifesttions (Intern Med 54: , 2015) () Introduction Extrintestinl mnifesttions of inflmmtory owel disese (IBD) re reltively frequent, lthough cliniclly evident pulmonry disese ssocited with IBD is uncommon. Trcheoronchitis is prticulrly rre extrintestinl mnifesttion of ulcertive colitis (UC) nd is often dignosed incorrectly nd treted s sthm (1). We herein report cse of fulminnt trcheoronchitis ssocited with excertion of UC in which colitis developed with typicl sucker-like ulcers. In this report, we descrie our difficulties in the selecting the tretment pln nd provide the detils of the ptient s complex clinicl course. Cse Report A 42-yer-old mn visited our hospitl complining of incresing loody dirrhe nd mild dominl pin. He hd een dignosed with UC y his fmily doctor yer erlier (Fig. 1), fter which time he hd received tretment with orl meslzine (3.6 g/dy). He hd lso experienced productive cough nd nose pin with purulent nsl dischrge for few dys prior to the current dmission. He ws nonsmoker nd hd no history of pulmonry disese. Computed tomogrphy (CT) indicted diffuse colonic owel wll thickening nd mild inflmmtion of the prnsl sinuses, without norml findings in the thorcic region. Blood tests showed severe inflmmtory chnges, with white lood cell count of 16,500 cells/mm 3 (82% neutrophils; 2.0% eosinophils; 6.0% monocytes; 10% lymphocytes), n erythrocyte sedimenttion rte of 94 mm/h nd C- rective protein level of mg/dl. Due to the highly elevted inflmmtory rection, the ptient ws hospitlized with dignosis of excertion of UC complicted y sinusitis. Deprtment of Surgery, Tokyo Metropolitn Bokutoh Hospitl, Jpn, Deprtment of Surgicl Oncology, Fculty of Medicl Sciences, the University of Tokyo, Jpn nd Deprtment of Allergy nd Rheumtology, Fculty of Medicl Sciences, the University of Tokyo, Jpn Received for puliction April 24, 2014; Accepted for puliction August 24, 2014 Correspondence to Dr. Msy Hiyoshi, mhiyoshi-tky@umin.c.jp 749

2 Figure 1. Endoscopic () nd histologicl findings () of the rectum t the initil dignosis. The iopsy specimen showed crypt scess. Figure 2. Colonoscopy of the trnsverse colon. Sucker-like ulcers in trnsverse colon (). Typicl deep ulcertion ws oserved continuously from the sigmoid colon to the rectum (). Figure 3. No norml findings were detected on n X-ry exmintion, even when the ptient s senstion of dyspne nd stridor worsened. Colonoscopy reveled oth typicl deep ulcers in the rectosigmoid colon nd typicl multiple round ulcers in the trnsverse colon (Fig. 2). A microscopic exmintion of the iopsy specimens showed cute inflmmtion with crypt scesses, lthough neither cytomeglovirus nor dysentery moes were detected on immunostining. Initilly, the ptient ws treted with empiricl ntiiotics; however, the dministrtion of this therpy for one week did not improve the inflmmtory rection, s oserved on lood tests. Insted, the ptient s respirtory symptoms worsened, with the ppernce of dyspne nd horseness. Although the chest X-ry findings remined within the norml limits (Fig. 3), reexmintion using chest CT showed wll thickening of the trche nd ronchi (Fig. 4). Flexile ronchoscopy disclosed the presence of mucosl irregulrities nd ulcers in the trche, findings indictive of ulcertive colitis (Fig. 5). A trchel iopsy showed necrosis nd mild inflmmtion of the suepithelil tissue, with no infectious fetures (Fig. 6). Hence, the ptient s respirtory symptoms were strongly suspected to e extrintestinl mnifesttions of UC rther thn infectious ronchitis. The ptient ws treted with 1,000 mg of intrvenous methylprednisolone for three dys followed y 30 mg of methylprednisolone. The corticosteroids quickly improved the ptient s respirtory sttus, with mrked regression of the trchel wll nd ronchi thickening (Fig. 4). The digestive symptoms relted to UC lso improved simultneously. Follow-up colonoscopy performed eight months lter showed the disppernce of the sucker-like ulcers in the trnsverse colon, without scrs. At tht time, the deep ulcers in the sigmoid colon nd rectum hd lso heled with residul scrs nd inflmmtory polyps. The dose of corticoste- 750

3 c d Figure 4. Chest CT imges otined efore (, ) nd fter (c, d) corticosteroid tretment. Wll thickening of the trche nd ronchi (white rrows) ws relieved y tretment. Figure 5. Bronchoscopy performed five dys fter steroid pulse therpy. Irregulr thickening of the mucos, edem nd ulcer-like lesions were oserved in the trche nd ronchi. roids ws therefore grdully tpered, without recurrence of trcheoronchitis. The ptient is currently free of oth gstrointestinl nd respirtory symptoms one yer fter the episode. Discussion Extrintestinl mnifesttions my develop in 21% to 41% of ptients with IBD; this incidence is higher in sujects with Crohn s disese (CD) thn in those with UC (2, 3). However, pulmonry involvement s n extrintestinl mnifesttion of UC is rre, occurring in only Figure 6. A iopsy of the trche showed necrosis nd mild inflmmtion of the suepithelil tissue (Hemtoxylin nd Eosin stining). No infectious etiology ws found. 0.21% of IBD ptients (4). The colonic nd ronchil epitheli my exhiit common inflmmtory response, s oth tissues re derived from the primitive gut (5). Therefore, ny prt of the respirtory system my e involved in irwy-ssocited diseses, such s suglottic-glottic stenosis, trcheitis, trcheoronchitis, chronic ronchitis, ronchiectsis nd pulmonry disorders, including ronchiolitis with orgnizing pneumoni, interstitil lung disese nd serositis (6, 7). Trcheoronchitis is chrcterized y the presence of sumucosl inflmmtion of lrge irwys nd the prolifertion of the reserve cell lyer (7), usully s result of virl or cteril infection with underlying lung disese (7). This condition is rre pulmonry compliction of UC, with only 16 reported cses, including the present cse (nine men 751

4 Tle. Literture Dt on Trcheoronchitis in Ptients with Ulcertive Colitis Sex Smoking sttus Durtion of UC, yrs. UC ctivity Tretment Outcome [Ref.] M(55) Ex-smoker 30 remission steroids, prtil improvement [1] zthioprine F (44) Ex-smoker 5 remission steroids improvement [6] M(50) N/A 0 ctive steroids mrked improvement [7] F (24) Never 10 ctive steroids rpid improvement [9] F (51) Never 20 ctive steroids rpid improvement [9] F (45) Never 11 colectomy, N/A N/A [10] 1yr prior F (54) Never 27 colectomy, N/A N/A [10] 12yr prior M(52) Never 21 remission N/A N/A [10] M(50) None for 22 colectomy, steroids improvement [11] 12 yr 8yr prior M(64) None for 5 colectomy, steroids improvement [11] 10 yr 4yr prior F (23) Never 8 remission steroids improvement [12] F (29) N/A 0 ctive meslzine, steroids, improvement [13] ntiiotics M(47) None for 12 ctive steroids, ntiiotics rpid improvement [14] 3yr M(57) Never 30 remission steroids, prtil improvement [15] zthioprine M(42) Never 1 ctive steroids rpid improvement [present cse] Dt in curved prentheses re ptient ge. UC: ulcertive colitis, F: femle, M: mle, N/A: dt not ville nd seven women; ge: yers, verge: 46 yers) (Tle) (1, 6-15). In most of these cses, pulmonry involvement nd colonic excertion developed simultneously, s in our ptient, wheres trcheoronchitis developed fter colectomy in few cses (8, 10, 11) or fter UC remission of more thn 30 yers (1, 15). The reltionship etween smoking nd IBD is well estlished (16), lthough the role of smoking in the onset of pulmonry mnifesttions of IBD remin uncler. In the present cse, mucosl irregulrities nd UC-like ulcers in the trche were oserved on ronchoscopy, without the detection of grnulom formtion or infection in the iopsy specimens. A wide vriety of ronchoscopic findings hve een reported, including severe trchel nrrowing, erythemtous, irregulr nd ulcerted mucos (13), colestone ppernce (14) nd exuernt pus (11). Active inflmmtion with mrked lymphoplsmcytic infiltrtion within the epithelium nd lmin propri without evidence of grnulom formtion re typicl histopthologicl findings (15). In the 16 previously reported cses, including ours, ll ptients were treted with corticosteroids, with n immedite response to therpy in ll ut one cse (8), in which only limited improvement ws chieved with systemic steroids. Although ronchil diltion ws ttempted in tht cse, the procedure resulted in trchel rupture nd deth (8). While gstrointestinl nd respirtory symptoms were relieved simultneously in most cses (9, 13), the clinicl courses of the upper irwy nd digestive diseses were not lwys entirely prllel (17). For exmple, in one cse, the ptient s respirtory sttus improved mrkedly with corticosteroid therpy, wheres the excertion of UC worsened, resulting in emergency colectomy for toxic megcolon (7). Infectious ronchitis, drug-relted disese nd grnulomtosis with polyngitis should e considered s differentil dignoses. Becuse tretment with corticosteroids is essentil for UC-ssocited trcheoronchitis nd cn lso promote infection, ruling out infectious diseses is prticulrly importnt. In the present cse, n infectious cuse of ronchitis ws excluded sed on the findings of repeted sputum nd lood cultures with no evidence of infection. Meslzine, the principl drug for IBD tretment, is lso responsile for respirtory complictions, such s interstitil disese nd eosinophilic pleuritis or pneumoni (18). However, the present ptient s respirtory symptoms remitted without interruption of this drug. Becuse the iopsy of the ronchil mucos otined vi ronchoscopy showed no grnuloms, there ws little possiility of the presence of grnulomtosis with polyngiitis, leding to dignosis of trcheoronchitis s n extrintestinl mnifesttion of UC. The current ptient hd originlly een dignosed with proctitis type UC; however, y the time of the current episode, the UC hd progressed to the pncolitis type. The mnifesttion of ulcers in the rectosigmoid colon ws typicl of UC, wheres tht in the trnsverse colon ws typicl, including sucker-like ulcers tht ppered to line up with the intestinl xis with norml mucos interposed etween the lesions. We initilly considered the coexistence of moeic colitis. However, iopsy specimen otined from the edge of the sucker-like ulcer showed only mildly inflmed colonic mucos, nd the findings for moes were negtive, even on PAS stining. We lso suspected cytomeglovirus 752

5 colitis; however, cytomeglovirus immunostining ws negtive. Nevertheless, we were unle to completely exclude the potentil of infectious enteritis nd thus continued the tretment with ntiiotics nd meslzine. Consequently, the initition of corticosteroid therpy due to the need to tret the ptient s rpidly deteriorting irwy symptoms lso grdully stopped the loody stools. Furthermore, colonoscopy performed four dys fter the initition of corticosteroids confirmed tht the ctive leeding in the rectosigmoid colon hd stopped nd tht the ulcers in the trnsverse colon hd decresed in size long with reduction in redness nd the findings of depressed ulcer mrgin. Hence, corticosteroid therpy ws effective for these ulcers. On follow-up colonoscopy performed eight months lter, the ulcers in the trnsverse colon hd heled without ny scrring. Therefore, we suggest tht the sucker-like ulcers reflected specific sutype of ulcertive colitis or nother form of utoimmune colitis. In conclusion, lrge irwy involvement, such s tht due to trcheoronchitis, is rre ut potentilly criticl extrintestinl mnifesttion of UC. This compliction should e kept in mind when dignosing respirtory symptoms in ptients with UC, prticulrly ecuse tretment with corticosteroids cn quickly improve these symptoms. The uthors stte tht they hve no Conflict of Interest (COI). References 1. Kr S, Thoms SG. A cse of trcheoronchitis in ulcertive colitis: review of literture. Clin Respir J 3: 51-54, Veloso FT, Crvlho J, Mgro F. Immune-relted systemic mnifesttions of inflmmtory owel disese. A prospective study of 792 ptients. J Clin Gstroenterol 23: 29-34, Veloso FT. Extrintestinl mnifesttions of inflmmtory owel disese: do they influence tretment nd outcome? World J Gstroenterol 17: , Rogers BH, Clrk LM, Kirsner JB. The epidemiologic nd demogrphic chrcteristics of inflmmtory owel disese: n nlysis of computerized file of 1400 ptients. J Chronic Dis 24: , McDermott RP, Nsh GS, Nhm MH. Antiody secretion y humn intestinl mononucler cells from norml controls nd inflmmtory owel disese ptients. Immunol Investi 18: , Byrktroglu S, Bsoglu O, Ceyln N, Aydın A, Tuncel S, Svs R. A rre extrintestinl mnifesttion of ulcertive colitis: trcheoronchitis ssocited with ulcertive colitis. J Crohns Colitis 4: , Shd JA, Shrieff GQ. Trcheoronchitis s n initil presenttion of ulcertive colitis. J Clin Gstroenterol 33: , Wilcox P, Miller R, Miller G, et l. Airwy involvement in ulcertive colitis. Chest 92: 18-22, Cmus P, Pird F, Ashcroft T, Gl AA, Coly TV. The lung in inflmmtory owel disese. Medicine 72: , Grg K, Lynch DA, Newell JD. Inflmmtory irwys disese in ulcertive colitis: CT nd high-resolution CT fetures. J Thorc Imging 8: , Vsisht S, Wood JB, McGinty F. Ulcertive trcheoronchitis yers fter colectomy for ulcertive colitis. Chest 106: , Rickli H, Fretz C, Hoffmn M, Wlser A, Knoluch A. Severe inflmmtory upper irwy stenosis in ulcertive colitis. Eur Respir J 7: , Cross DL, Scudder DD. Airwy ostruction in ulcertive colitis. Southern Medicl Journl 90: , Jnssen WJ, Bierig LN, Beuther DA, Miller YE. Stridor in 47- yer-old mn with inflmmtory owel disese. Chest 129: , Ymmoto AK, Br JL. Cse 184: ulcertive trcheoronchitis. Rdiology 264: , Thoms GA, Rhodes J, Green JT. Inflmmtory owel disese nd smoking: review. Am J Gstroenterol 93: , Asmi T, Koym S, Wtne Y, et l. Trcheoronchitis in ptient with Crohn s disese. Intern Med 48: , Csell G, Villncci V, Di Bell C, Antonelli E, Bldini V, Bssotti G. Pulmonry diseses ssocited with inflmmtory owel diseses. J Crohns Colitis 4: , The Jpnese Society of Internl Medicine 753