Primary gastric actinomycosis: report of a case diagnosed in a gastroscopic biopsy

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1 Al-Oidy et l. BMC Clinicl Pthology (2015) 15:2 DOI /s CASE REPORT Open Access Primry gstric ctinomycosis: report of cse dignosed in gstroscopic iopsy Khleel Al-Oidy 1, Ftimh Alruwii 1, Areej Al Nemer 1, Red Alsulimn 2, Zin Alruwii 3 nd Mohmed A Shwry 1* Astrct Bckground: Primry gstric ctinomycosis is extremely rre, the ppendix nd ileocecl region eing the most commonly involved sites in dominopelvic ctinomycosis. Herein, we report cse of primry gstric ctinomycosis. The dignosis ws mde on microscopic evlution of gstroscopic iopsy specimens. To the est of our knowledge, this is the third cse to e reported in the literture, in which the dignosis ws mde in gstroscopic iopsy rther thn resection specimen. Cse presenttion: An 87-yer-old Sudi mle on mediction for crdiomyopthy, premture ventriculr contrctions, renl impirment, hypertension, nd dyslipidemi, presented to the emergency deprtment with cute diffuse dominl pin, dominl distension, constiption nd vomiting for two dys, with no history of fever, dominl surgery or trum. The ptient ws dmitted to the hospitl with n impression of gstric outlet ostruction. Bsed on rdiologic nd gstroscopic findings, non-infectious etiology ws suspected, possily denocrcinom or lymphom. Gstroscopic iopsies showed n ctively inflmed, foclly ulcerted trophic fundic mucos long with frgments of firinopurulent exudte contining rownish, iron negtive pigment nd undnt filmentous cteri, morphologiclly consistent with Actinomyces. Conclusion: Althuogh extremely rre, primry gstric ctinomycosis should e considered in the differentil dignosis of rdiologic nd gstroscopic diffuse gstric wll thickening nd sumucosl tumor-like or infiltrtive lesions, prticulrly in ptients with history of dominl surgery or trum, or those receiving extensive mediction. A high level of suspicion is required y the pthologist to chieve dignosis in gstroscopic iopsies. Sutle chnges such s the presence of pigmented inflmmtory exudte should lert the pthologist to perform pproprite specil stins to revel the custive orgnism. Keywords: Actinomycosis, Gstric, Grocott s, Grm, PAS Bckground Actinomycosis is chronic suppurtive grnulomtous inflmmtion cused y neroic, filmentous, Grmpositive cteri of Actinomyces species, most often Actinomyces isrelii. There re three min forms of ctinomycosis, nmely, cervicofcil (31%-65%), dominopelvic (20%-36%) nd thorcic (15%-30%) [1-4]. In dominopelvic ctinomycosis, the ppendix nd ileocecl region re the most commonly involved sites (65%) [2,3,5-7]. Primry gstric ctinomycosis is extremely rre, with only 23 cses reported to dte [5,6,8-20]. Herein, we report cse of primry gstric * Correspondence: melshwry46@hotmil.com 1 Pthology Deprtment, College of Medicine, University of Dmmm, P.O. Box 1982, Dmmm 31441, Sudi Ari Full list of uthor informtion is ville t the end of the rticle ctinomycosis. The dignosis ws mde on microscopic evlution of gstroscopic iopsy specimens. To the est of ourknowledge,thisisthethirdcsetoereportedintheliterture, in which the dignosis ws mde in gstroscopic iopsy rther thn resection specimen [6,8]. Cse presenttion Clinicl nd lortory findings An 87-yer-old Sudi mle on mediction for non-ischemic crdiomyopthy, frequent premture ventriculr contrctions, renl impirment, hypertension, nd dyslipidemi, presented to the emergency deprtment with cute diffuse dominl pin, dominl distension, constiption nd vomiting of two dys durtion, with no history of fever, dominl surgery or trum. Medictions received y the 2015 Al-Oidy et l.; licensee BioMed Centrl. This is n Open Access rticle distriuted under the terms of the Cretive Commons Attriution License ( which permits unrestricted use, distriution, nd reproduction in ny medium, provided the originl work is properly credited. The Cretive Commons Pulic Domin Dediction wiver ( pplies to the dt mde ville in this rticle, unless otherwise stted.

2 Al-Oidy et l. BMC Clinicl Pthology (2015) 15:2 Pge 2 of 5 ptient for the lst four yers included, minly, dily cetyl slicylic cid 81 mg, torvsttin 40 mg, iresrtn 300 mg nd hydrochlorothizide 25 mg. Adominl exmintion reveled stle vitl signs long with positive findings of dominl distension nd mild epigstric tenderness. Lortory investigtions showed leucocytosis (16.6 k/μl with 89% segmented cells), mild normocytic normochromic nemi (Hg 11.5 g/dl, MCV 93.7 fl, MCH 31.7 pg), elevted serum lipse (1123 U/L), mylse (269 U/L), nd cretinine (1.4 mg/dl), nd low potssium (3.1 meq/l). Plin dominl X-ry showed mrkedly dilted stomch (Figure 1). The ptient ws dmitted to the hospitl with n impression of gstric outlet ostruction. NGT ws inserted & spirtion yielded lrge mount of greenish fluid. The ptient ws then immeditely put on empiric ntiiotic coverge for 5 dys with 2 doses of IV levofloxcin nd 3 doses of IV metronidzole dministered. Contrst CT-scn, performed to rule out n orgnic cuse for the gstric outlet ostruction, showed significntly distended stomch with thickened wll nd norml configurtion, nd single ir-fluid level (Figure 1). Two gstroscopies were then performed, 1 week prt, nd reveled deformed stomch with hrd mss infiltrting the greter curvture in the fundic re, covered y necrotic greenish rown mteril, long with sent peristltic movement nd no pprent orgnic ostruction to the gstric outlet (Figure 2). Bsed on the rdiologic nd gstroscopic findings, non-infectious etiology ws suspected, possily denocrcinom or lymphom. Biopsies otined from the edge nd the centre of the fundic mss during oth gstroscopies were sent for pthologicl exmintion. Histologic exmintion showed n ctively inflmed, foclly ulcerted, trophic fundic mucos with vrile, focl eosinophilic infiltrtion, edem, nd vrily dilted foveole with focl regenertive epithelil typi (Figure 3). There were lso frgments of firinopurulent exudte mixed with rownish, iron negtive pigment (Perl s stin) nd undnt PAS, Grocott s, nd Grm positive rod- Figure 2 Gstroscopy. Deformed stomch, with rottion like ppernce. There re inflmed res with greenish rown mteril over the surfce. like nd filmentous cteri, morphologiclly consistent with Actinomyces (Figure 4). The orgnisms were overlooked in the first iopsy. The second iopsy ws performed ecuse dignosis of mlignncy ws still in suspicion, despite the negtive result of the first iopsy. A revisit to the first iopsy confirmed negtivity for mlignncy ut reveled the presence of orgnisms identicl to those noted in the second iopsy. Culturing of gstric contents following the second gstroscopy, yielded only Streptococcus viridns with no Actinomyces identified. However, neroic culture ws not specificlly ordered y the clinicin. Consequently Actinomyces, known to e strictly neroic, were not detected. Despite the negtive culture, the typicl morphology of the orgnisms in tissue sections confirmed y positive Grocott, PAS nd Grm stining ws considerd sufficient Figure 1 Rdiologic findings.. Plin dominl X-ry showing mrkedly dilted stomch. Contrst CT-scn showing significntly distended stomch with thickened wll nd norml configurtion. Note lso ir-fluid level.

3 Al-Oidy et l. BMC Clinicl Pthology (2015) 15:2 Pge 3 of 5 Figure 3 Gstroscopic iopsies.. Inflmed trophic mucos with dilted foveole nd pigmented firinopurulent inflmmtory exudte. H & E x 100. Perl s stin showing iron negtive rownish pigment. x 400. for dignosis with no necessity for confirmtion y repet culturing under neroic conditions. The ptient ws then mnged conservtively in the hospitl. A third gstroscopic iopsy two weeks lter reveled chronic trophic gstritis with no Actinomyces detected, nd the ptient ppered in good helth sttus. A pln ws set up to strt him on the pproprite ntiiotic therpy for ctinomycosis with follow up gstroscopy fter one month. However, the ptient chose to continue tretment somewhere else. So he ws dischrged on his request nd never showed up gin in our institution. Discussion Actinomycosis in humn is most commonly cused y Actinomyces isrelii [1,3,21-26] which is n endogenous commensl present in the orl nd GI-trct flor [9,10,12,22,27]. Actinomycetes typiclly invde injured mucos with opportunistic infection occuring if there is rek in the mucosl rrier. Fctors tht precipitte intr-dominl ctinomycosis include GI surgery, inflmmtion, nd viscerl perfortion [28,29]. However, in most cses of gstric ctinomycosis, it hs een impossile to trce the mechnism y which Actinomyces hd reched the gstric wll [30]. Our ptient hd no pst c d Figure 4 Filmentous nd rod-like cteri consistent with Actinomyces in gstroscopic iopsies.. H & E x 400. PAS x 1000 c. Grm x 1000 d. Grocott s x 1000.

4 Al-Oidy et l. BMC Clinicl Pthology (2015) 15:2 Pge 4 of 5 history of dominl surgery or trum. However, he ws on prolonged mediction for non-ischemic crdiomyopthy, premture ventriculr contrctions, renl impirment, hypertension, nd dyslipidemi. Such extensive mediction my hve cused physicl or functionl gstric mucosl dmge tht fcilitted entry of the orgnisms into the gstric wll. Numerous drugs, cting through vrious mechnisms, hve een ssocited with gstric mucosl dmge [31]. Age relted mucosl trophy my hve lso contriuted to diminished mucosl resistnce. The rrity of gstric involvement y ctinomycosis hs een ttriuted to the high lumenl cidity of the stomch. As result of the low gstric ph, the orgnisms my e killed or growth is inhiited [9]. The usul presenting clinicl mnifesttions of gstric ctinomycosis re low-grde fever, epigstric pin, weight loss, nd upper GI leeding [1,3,10,12,20]. One ptient developed symptoms of gstric outlet ostruction [19]. The durtion of symptoms rnged from two weeks to severl yers [3,8,9,11,19]. Our ptient presented with cute diffuse dominl pin, dominl distension, constiption nd vomiting for two dys durtion, with no history of fever. The clinicl impression ws tht of gstric outlet ostruction. There is no specific rdiologicl or endoscopic ppernce for gstric ctinomycosis. CT findings hve mostly demonstrted n infiltrtive lesion with diffuse gstric wll thickening. The ppernce suggested denocrcinom or lymphom of the stomch [2,20,32]. In our cse, contrst CT-scn showed significntly distended stomch with thickened wll nd norml configurtion. Similr to rdiologic studies, the endoscopic findings of the disese my simulte gstric neoplsm nd include sumucosl tumor-like or infiltrtive lesions nd, occsionlly, mucosl ulcertion [13]. A non-infectious etiology ws initilly suspected in our ptient sed on rdiologic nd endoscopic findings, possily denocrcinom or lymphom, nd the gstric outlet ostruction susequently interpreted s functionl due to sence of peristltic movement consequent to infiltrtion of the gstric wll y ctinomycosis. An ssocited prlytic ileus due to cute pncretitis my e n lterntive explntion for the ostruction s suggested y elevted serum lipse nd mylse levels. Such ostruction my hve lso contriuted to the gstric locliztion of the ctinomycosis, so tht the clinicl mnifesttions my e consequence of cute pncretitis with secondry gstric overinfection y Actinomyces, fcilitted y the mucosl dmge. Becuse of the sumucosl locliztion of the inflmmtory process, gstroscopic iopsy specimens usully revel nonspecific inflmmtory chnges [3,14,18,19]. In most cses, the dignosis ws mde fter surgery nd histopthologicl exmintion of the resected specimen [9,12,19,20,22]. Only two cses hve een reported in which the dignosis of gstric ctinomycosis ws mde on microscopic evlution of gstroscopic iopsy specimen [6,8]. In our cse, the dignosis ws, likewise, estlished through histologic exmintion of gstroscopic iopsies in which undnt PAS, Grocott s, nd Grm positive rod-like nd filmentous cteri, morphologiclly consistent with Actinomyces were identified. The presence of rownish, iron negtive pigment in the firinopurulent inflmmtory exudte (tht ws lso visile endoscopiclly) lerted us to the possiility of ctinomycosis which ws estlished y pproprite specil stining tht reveled the microorgnisms. It is well known tht the min sources of nturl pigments re plnts nd microorgnisms, including Actinomycets [33]. Culturing is negtive in most cses of gstric ctinomycosis (>76%) [19,24,25]. In our cse, culturing yielded only Streptococcus viridns, nother endogenous eroic/neroic fculttive commensl present in the orl nd GI-trct flor [34]. Despite the negtive culture, the typicl morphology of the orgnisms in tissue sections confirmed y positive Grocott, PAS nd Grm stining ws considered sufficient for the dignosis of Actinomyces infection with no necessity for culture confirmtion. Most neroic cteri recovered from clinicl infections re found mixed with other neroic orgnisms [35]. Polymicroil infections re known to e more pthogenic for experimentl nimls thn re those involving single orgnisms [35]. Whether Streptococcus viridns, known to e n orgnism of low virulence, hd contriuted to the gstritis in our cse remins uncler. Primry gstric ctinomycosis is n indolent infection. If the disese is recognized, the prognosis is good ecuse ntiiotic tretment, prticulrly penecillin is very effective [4,19]. Our ptient received 2 doses of IV levofloxcin nd 3 doses of IV metronidzole nd ppered in good helth sttus, two weeks fter dignosis. Conclusions Althuogh extremely rre, primry gstric ctinomycosis should e considered in the differentil dignosis of rdiologic nd gstroscopic diffuse gstric wll thickening nd sumucosl tumor-like or infiltrtive lesions, prticulrly in ptients with history of dominl surgery or trum or those receiving extensive mediction. A high level of suspicion is required y the pthologist to chieve dignosis in gstroscopic iopsies. Sutle chnges such s the presence of pigmented inflmmtory exudte should lert the pthologist to perform pproprite specil stins to revel the custive orgnism. Consent Written informed consent ws otined from the ptient for puliction of this cse report nd ny ccompnying imges. A copy of the written consent is ville for review y the Editor of this journl.

5 Al-Oidy et l. BMC Clinicl Pthology (2015) 15:2 Pge 5 of 5 Competing interests The uthors declre tht they hve no competing interests. Authors contriutions KA-O shred in nlysis of histologic, clinicl nd rdiologic findings nd significntly contriuted to drfting of the rticle. FA shred in nlysis of histologic findings nd contriuted to drfting of the rticle. AAN shred in interprettion of histologic findings nd performed criticl review of the rticle. RA provided nd interpreted clinicl, endoscopic nd rdiologicl findings. ZA shred in nlysis of histologic findings nd performed criticl review of the rticle. MAS performed interprettion nd nlysis of histologic findings, closely supervised progress of the study nd completed drfting nd editing of the rticle. All uthors hve red nd pproved the finl mnuscript. Acknowledgements The uthors cknowledge Dr Amni Al Nemer, Assistnt Professor, Microiology Deprtment, University of Dmmm for reviewing the results of H & E nd specil stining of micro-orgnisms in tissue sections. The uthors lso cknowledge the services of Mr Shkir Ahmed nd Mrs Mri Rosrio Lzro from the histopthology lortory of the University of Dmmm, Sudi Ari for conducting the histology technicl work. Author detils 1 Pthology Deprtment, College of Medicine, University of Dmmm, P.O. Box 1982, Dmmm 31441, Sudi Ari. 2 Deprtment of Internl Medicine, College of Medicine, University of Dmmm, P.O. Box 1982, Dmmm 31441, Sudi Ari. 3 King Fhd Hospitl of the University, University of Dmmm, P.O. Box 2208, Al-Khor 31952, Sudi Ari. Received: 7 April 2014 Accepted: 9 Ferury 2015 References 1. Choi MM, Bek JH, Lee JN, Prk S, Lee WS. Clinicl fetures of dominopelvic ctinomycosis: report of twenty cses nd literture review. Yonsei Med J. 2009;50: Isik B, Aydin E, Sogutlu G, Ar C, Yilmz S, Kirimlioglu V. 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Synergistic effects of neroic ccocci, Bcteroides, Clostridi, Fusocteri, nd neroic cteri on mouse nd induction of sustnces scess. J Infect Dis. 1984;149: Sumit your next mnuscript to BioMed Centrl nd tke full dvntge of: Convenient online sumission Thorough peer review No spce constrints or color figure chrges Immedite puliction on cceptnce Inclusion in PuMed, CAS, Scopus nd Google Scholr Reserch which is freely ville for redistriution Sumit your mnuscript t