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Occult celiac disease in an octogenarian presenting with a small intestinal adenocarcinotna H UGH ) AMES FREEMAN MD
HJ FREEMAN. Occult celiac disease in an octogenarian presenting with a small
intestinal adenocarcinoma. Can J Gastroenterol 1994;8(6):354-357. An 85-year-old male initially presented with an obstructeJ upper gastrointestinal tract a sociated with a duodena l adenocarcinoma. Subsequent histological studie revea led occu lt celiac disease, an assoc iated condition that otherwise wo uld not have been recognized in this e lderly patient. The celiac disease responded to a gluten-free diet, even in the pre ence of an intestinal adenocarcinoma. This report suggests that celiac disease and ·mall bowel carcinoma may coexist more frequently than is appreciated. Key Words: Celiac disease , Duodenal adenocarcinoma, Gluten-free diet, Malabsorp-
t
tion syndromes, Occult celiac disease, Small bowel cancer
Maladie coeliaque occulte chez un octogenaire qui se presente avec un adenocarcinome de l'intestin grele RESUME : Un homme de 85 ans se presence initialement avec une obstruction des voies gastr -intestinales superieures as ociee a un adenocarcinome duodenal. Des epreuves histologiques subsequent s ont revele une maladie coeliaque occulte, maladie associee q ui n'aurait pu etre autrement identifiee chez ce patient aoe. La maladie coeliaque a repondu a une diete sans gluten, meme en presence d'un adenocarcinome intestinal. Ce rapport suggere que la maladie coeliaque et le carcinome de l'intestin grele peuvent coexister plus frequemment que !'on ne I penserait.
P
ATI ENTS WIT I I CELI AC DISEASE
have an increa ed risk of developing malignant complicatio ns, especially abdominal lymphoma (1 -3 ). In additio n, some epith lial malignanc ies appear to occur more commo nly in pat ients with celiac disease compared
with the genera l popul atio n; these in clude carc ino mas of the gastrointestinal trac t, inc luding the sma ll intestine ( 4 ). Although ea rlier studies have indi ca ted that ma lignancy may complicate long-sta nding celiac disease, subequent investiga tions revea led that
University Hospital (U BC Site) , Vancouver, British Columbia Correspondence and reprin Ls: Dr Hugh) Freeman , Head, Gascroenterology, ACU F- 137, University Hospital (U BC Site), 22 11 Wesbrool< Mall , Vancouver, British Columbia V6T 1W5. Telephone (604) 822-72 16 Received for Jmblicarion December 13, 199 3. Accepted A/Jril 12, 1994
354
CAN
lympho mas, parti cularly of the sma ll in testine, could be the presenting manifestati on of occul t or subc linica l celi ac disease (3 ) as we ll as latent ccliac disease (5 ). It is increas ingly recogni zed tha t carcinomas of the prox imal intestine, spec ifically the duodenum and jejunum , may also occasionall y complicate the clinical course of establi hed celi ac disease (6-34) . H oweve r, prec ise data on the true relationship between these two condition · are not ava ilabl e, in large pa rt beca use recogni t ion of eel iac di ease , particularly if sympto ms are minimal, is difficult. Diagnosis of ccliac disease re ts on at leas t two criteri a. O ne is the finding of a characteristi c 'fl at' muco a in the prox ima l small in te tine. The second is the demon ·tration of a response to a glu te n-free diet. In a clinica l di agnostic setting, a successful therapeut ic respo nse i · often based on the disappearance of sy mpto ms and normalizati on of laboratory abnorma li t ies. Howe ver, if the initi al cl ini ca l presen ta tion is occul t with few sympto ms and little or no steatorrh ea, documentation of a morpho log ica l respo nse i importa nt. ln the prese nt report, the opposite e nd o f this ce li ac di sease-ca rc ino ma re lati onship is explored. An elde rl y pat ient with an adenocarc ino ma of the duodenum was seen; subsequently, occul t ccliac disease was recognized. Although more systematic studies are needed, this repo rt further emphas izes
J GASTROENTEROL VOL 8 No 6 N OVEMBER
L994
Celiac disease and small bowel adenocarcinoma
the occult natu re of ce liac disease and suggests that the association of ce li ac disease and sma ll intestinal ca ncer is more frequent than is appreciated.
CASE PRESENTATION An 85-year-old male was first ho pitalized in Octobe r 1989 becau e of sudden onset of severe postprandial abdom inal pain and vomiting of fluids and solids. Prior to this acute pre entation he had intermittent postprandial abdominal discomfort and bloating fo r approximately e ight months with an estimated weight loss of 10 kg. Medical hi to ry was unremarkable and, specificall y, there was no prior diarrhea, Fever or known anemia. Examination revealed abdom inal .IRER
l 994
Cncl1:1c disease , glut en -fret' d1e1, and m:1l1gnancy. C ut 1976;17:6 12-1.J. 28. (~run M, Sch ul. A, Kraus J, Leinweber B. lieu, ,y111pt,11m du e rn 1hrcc dis1incti w ly d,tfcrcntiarcd s111a ll 111tes11ne ca rci11111n:1, 111 nn11trop1c:1I sprue. Disch McJ Wochenschr 1989; 114:709- 1I. 29. Maurano A, C irill n Ll ', Nllv1c ll ll A. Ccli:ic dbl'~l',e a... a ri sk
30.
wo1nan with
no111mrical sprue. Ga,trncmerolog\ 1961;44: 330-4. 18. 1lolmes GKT, Dunn GI, Cockcl R, Brookes VS. Adenocarcinoma of the upper sma ll howel com pli ca ting cocl i:1c disease. (,uL 1980;2 l: IOL0-6. 19. O'Brien CJ, Saverymuuu S, I lodgson I IJ F,
20.
Th is malignan cy has been commo nl y observed in pat ients with a va riety of diseases hav ing some form of immu no logical alte ratio n ( ic, S jogre n 's syn drome , syste mi c lurus c rythematosis, sarcoidosis, post-transp lantation), patient s with diseases havi ng we ll -doc umented dcfic icnc ie:. in humoral (ic, h ypoga mmaglobu linem ia) or ce llular immune function (ic, acq uired immunodeficiency sy ndrome) as well ,is in pat ients rece iving immunosupprcssivc drugs. A gluten -free di et restores the mucosa! architecture and mucosa ! function towards normal in ce liac disease and, as a result, has been hypothesized (2) to reduce its malignant po tenti a l.
31.
32.
31.
34.
15.
16.
17.
fact(W
for digestive
neophisms. Rndiol Med (Torino) 1987;73: 174-7. J:1cnh,en MB, F:wsa 0, Elgjo K. Sch rumpf E. I Jepat ic lesion, in adult cncl 1nc dise;ise. Scand J Gas troenrerol i 990;25:656-62. Marsch SC, Iker M, Su lser 11, I Jan y A. Adcnllcarc inomn nt' the sma ll intestine 111 celhic Jisease. Case report :ind li1cra1urc rev iew. Schwciz Med Wochcnsc hr 1990; 120: I 15-4 1. OT)riswl l BRC, Stevens FM, O'Corman TA, ct al. I ILA type of patient, with coc lin c di,easc and malignancy in rhc west of Ireland. Cut 1982;2 l:662-5. Ashk cn:m A. Bar:m M. M.il,gnan cy comp licm ing cc li ac di sease. In : Branski D, Ro:en r, Kagnoff MF, eJs. l,l utcn-Sens irive En1crnpnthy. Basel: Ka rger, 1992: 184-91. Blackwell JR. Two c:ises llf carci ,wma ,,f the ,mal l bnwe l with mal:ih,orplllln. C,111 196 1;2: 177. Wolher R, Owe n D, Belhuono L, Appelm,m 11 , Freeman I IJ . Lymphocytic gastritis in 11:nie111s w11h eel inc sprue or spruclik c int esun:1 1disease. Ci,istmcmernlng\ 1990;98: l l 0-5. I lolmes GKT, Pnnr r, Lane MR, P11pe l), A ll an RN. Malignancy in ccli:ic disc:ise effeCI of :, glute n-free d1cl. C ut 1989; 10: 331-H. Bruni PW, S ircus W, Maclea n N. Nenplasia and di e coclmL ~yndromc 111
adults. Lan cet 1969;1: 180-4. 18. Barry RE, Read AE. Cnel ,ac di,e:1,c and malignancy. Q J Med J971;42:665-75. N. l, da1 T , Fischel B, l\ 11111 J, Lcwenthal M. M,1rpholng\ llf ,ma ll hnwcl 11111cns:1 in malignancy. Digc,t11111 1971;7: 147 -55. 40. Cooke WT. Thompslln 11, WilliamsJA. Malign:incy and adu lt clle l, ac disease. Gui 1969; 10: 108- 11 .
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