HISTORY. Surgical Neurology International 2015, 6:174.

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1 Surgicl Neurology Interntionl OPEN ACCESS For entire Editoril Bord visit : Editor: Jmes I. Ausmn, MD, PhD University of Cliforni, Los Angeles, CA, USA Cse Report Intrcrnil lstomycotic scess mimicking mlignnt rin neoplsm: Successful tretment with voriconzole nd surgery Knik Aror, Ross L. Dwkins 1, Dvid F. Buer 2, Cheryl A. Plmer 3, Jmes R. Hckney 4, Jmes M. Mrkert 1 Deprtments of Neurology, 1 Neurosurgery nd 4 Pthology, University of Alm t Birminghm, Birminghm, Alm, 2 Drtmouth Hitchcock Medicl Center, Lenon, New Hmpshire, 3 Deprtment of Pthology, University of Uth, Slt Lke City, Uth, USA E mil: Knik Aror kror@umc.edu; *Ross L. Dwkins ross.dwkins.md@gmil.com; Dvid F. Buer dvid.f.uer@hitchcock.org; Cheryl A. Plmer cheryl.plmer@pth.uth.edu; Jmes R. Hckney jrhckney@umc.edu; Jmes M. Mrkert jmrkert@umc.edu *Corresponding uthor Received: 01 July 15 Accepted: 09 Septemer 15 Pulished: 20 Novemer 15 Astrct Bckground: Cererl lstomycosis is rrely reported disese, nd in the sence of ssocited, underlying systemic infection, poses gret dignostic difficulty. Mgnetic resonnce imging cn sometimes provide equivocl informtion when trying to pinpoint dignosis. Clssiclly, cererl lstomycosis hs een treted with mphotericin B. Voriconzole is newer trizole ntifungl with potentil s follow up tretment of lstomycosis of the centrl nervous system fter initil therpy with mphotericin B. Cse Description: We descrie one such cse of cererl lstomycotic scess, presenting in the sence of ny systemic disese, which ws initilly thought to e neoplsm. It ws successfully treted y surgicl resection followed y sequentil mphotericin B nd voriconzole. The ptient did well with voriconzole therpy nd ws followed for voriconzole tolernce with liver function tests, which continued to e stle t 8 months pst the initition of therpy. At 12 months postopertively, the ptient ws doing well nd showed grdul improvement in visul field cut, with no sign of recurrent infection. Conclusions: Isolted cererl lstomycosis cn present dignostic chllenge. In the sence of systemic infection, surgicl resection followed y ntifungl therpy is logicl tretment pln. Access this rticle online Wesite: DOI: / Quick Response Code: Key Words: Blstomycotic scess, cererl lstomycosis, voriconzole INTRODUCTION Blstomycosis is n uncommon ut often serious mycosis, endemic in the Southestern nd the Centrl United Sttes. Approximtely, 800 hospitliztions occur nnully in the United Sttes for lstomycosis, of which 6% result in deth. [7] It is primrily disese of the lung nd is chrcterized y suppurtive nd grnulomtous lesions in the lungs, skin, ones, nd genito urinry trct. [9,12,15,16] Centrl nervous system (CNS) involvement is rre, reported in out 4 10% of cses. [9] When present, it is lmost invrily ssocited with the involvement of other orgns, nd it is often the result of hemtogenous dissemintion from pulmonry source. [15,17] Cses presenting with solitry or multiple intrcrnil mss This is n open ccess rticle distriuted under the terms of the Cretive Commons Attriution-NonCommercil-ShreAlike 3.0 License, which llows others to remix, twek, nd uild upon the work non-commercilly, s long s the uthor is credited nd the new cretions re licensed under the identicl terms. For reprints contct: reprints@medknow.com How to cite this rticle: Aror K, Dwkins RL, Buer DF, Plmer CA, Hckney JR, Mrkert JM. Intrcrnil lstomycotic scess mimicking mlignnt rin neoplsm: Successful tretment with voriconzole nd surgery. Surg Neurol Int 2015;6: Surgicl Neurology Interntionl Pulished y Wolters Kluwer - Medknow

2 Surgicl Neurology Interntionl 2015, 6:174 lesions in the sence of systemic infection re extremely rre, though they hve een documented nd hve often een mistken for rin neoplsms. [2,3,15,19] Once dignosed, intrcrnil lstomycosis hs clssiclly een treted with mphotericin B deoxycholte or lipid formultions of mphotericin B. [18] Voriconzole, n zole ntifungl, hs shown promise s tretment option for CNS lstomycosis. Clinicl experience with voriconzole s prt of the tretment regimen for CNS lstomycosis is limited. [1,2,4,8,14] This report discusses cse of 37 yer old, otherwise helthy, nondietic, nd immunocompetent, white mn presenting with n isolted left temporo occipitl mss lesion, identified s Blstomyces dermtitidis, nd successfully treted with surgicl resection, followed y the sequentil use of mphotericin B nd voriconzole. HISTORY A 37 yer old, midextrous txidermist presented to ophthlmology clinic t the University of Alm t Birminghm Hospitl with severl yer history of visul floters of the right eye nd intermittent decresed hering on the left. He complined of worsening symptoms over the previous 8 months with intermittent senstions of pressure ehind the right eye, difficulty with lnce nd multion, difficulty reding, nd mild hedches. He lso descried poorly defined prolems with cognition. He did not report the impirment of speech, lnguge, or comprehension. His pst medicl nd surgicl histories were unremrkle. He denied the use of tocco or intrvenous drugs nd indicted infrequent lcohol use. Exmintion On physicl exmintion, significnt findings included smll right inferior qudrntnopi to confronttionl exmintion. Automted visul fields documented the presence of congruous inferior qudrntnopi. His neurologic exmintion ws otherwise norml. No norml skin findings were present. Work up An mgnetic resonnce imging (MRI) of the rin ws otined nd reveled two closely ssocited lrge, heterogeneous, left temporo occipitl lesions with mss effect. The lesions were heterogeneously enhncing on T1 weighted imges, nd T2 weighted imges showed chnges consistent with edem extending through the temporl nd occipitl loes. Diffusion weighted imging demonstrted equivocl incresed signl intensity within the lesions. A low pprent diffusion coefficient (ADC) ws not seen on the ADC mp, s is typicl with cererl scesses. While closely ssocited with the convexity dur nd the tentorium, the msses ppered to e intr xil, nd were interpreted s most consistent with mlignnt gliom or metsttic lesion [Figure 1]. Of note, his lood profile indicted mild leukocytosis ( /mm 3 ) nd predominntly neutrophilic (74%). Lumr puncture ws deferred ecuse of intrcrnil mss effect. Body imging did not revel extrcrnil lesions nd urine cultures were negtive. Opertion nd initil tretment For dignosis nd therpy, the ptient ws tken to the operting room for neuronvigtion guided resection of the mss vi temporo occipitl crniotomy with ultrsound ssistnce in delineting the orders of the lesion. The intr opertive specimen ppered to e fungl scess on frozen section evlution. The scess ws resected [Figure 2] nd the ptient ws strted on intrvenous lipid complex mphotericin B nd flucytosine, which he received for pproximtely 3 weeks. The microiology cultures demonstrted B. dermtitidis. The mphotericin B nd flucytosine were stopped nd the ptient ws electively prescried orl voriconzole 200 mg twice dy. Neuropthology Histopthology reveled grnulomtous process with encpsulted yest forms. Numerous multinucleted gint cells were present, some of which contined fungl orgnisms, which were smll, refrctile, nd vrily sized. A CD163 stin demonstrted undnt mcrophges, nd lso highlighted the lrger, multinucleted histiocytic gint cells [Figure 3]. The fungl orgnisms were positive for oth periodic cid Schiff nd Gomori s methenmine silver [Figure 4] stins. There ws n undnce of T cells, with slight predominnce of the CD4 helper/inducer suset s compred to the CD8 cytotoxic/suppressor suset [Figure 5]. Nturl killer c Figure 1: Preopertive xil imges. T1-weighted imge fter gdolinium dministrtion (), demonstrting multiloculted ringenhncement. Diffusion- weighted imge demonstrtes very smll mount of incresed signl (), ut decresed pprent diffusion coefficient vlue is not ovious (c). Findings re consistent with mlignnt neoplsm, either primry or metsttic, ut lso with scess

3 Surgicl Neurology Interntionl 2015, 6:174 Figure 2: Postopertive xil T1-weighted imge fter gdolinium dministrtion, demonstrting resection of the lesions with postopertive chnges nd no evidence of residul enhncement c Figure 3: Numerous gint cells re present in the grnulomtous inflmmtion (), mny of which contin intrcytoplsmic fungl orgnisms (). A CD163 stin shows undnt mcrophges (c) Figure 4: The fungl orgnisms re positive with oth periodic cid- Schiff () nd Gomori s methenmine silver stins () cells were rrely present [Figure 6]. The scess displyed shrp order with the underlying gliotic rin tissue. Follow up The ptient did well with voriconzole therpy nd ws followed up for possile voriconzole toxicity with liver function tests, which continued to e stle t 8 months pst the initition of therpy. Due to the development of photosensitivity nd some res of cutneous erythem, voriconzole ws stopped fter 7 months of therpy nd switched to orl fluconzole 800 mg dily. This ws continued for 3 months. At 12 months postopertively, the ptient ws doing well nd showed grdul improvement in his visul field deficit. His MRI t this time showed smll loulted res of enhncement tht were interpreted s postsurgicl chnge nd not recurrence of the scess [Figure 7]. All ntifungl therpy ws stopped. The ptient hs continued to do well t 2½ yers postopertively with stle right inferior qudrntnopi on ophthlmologic exmintion. DISCUSSION Blstomycosis is systemic fungl infection cused y dimorphic fungus B. dermtitidis. A disese of worldwide Figure 5: There is n undnce of oth CD4 helper T cells () nd CD8 cytotoxic T cells () distriution, it is found to e endemic in the Ohio nd Mississippi river vlleys of the United Sttes, s well s Centrl Cnd. It is primrily disese of the lung, though secondry dissemintion to skin, ones genito urinry trct, nd rrely CNS hs een documented. [6,13,15] CNS involvement is most frequently secondry to systemic lstomycosis nd my mnifest s cute or chronic meningitis, spinl msses, nd intrcrnil mss lesions, which re usully multiple. [2,9,12,15,16] Solitry lesions without systemic disese s descried in this cse report hve een rrely descried in the literture. [2,3,19] Solitry, or in this cse, multiple enhncing rin lesions re difficult or impossile to dignose without direct exmintion of the tissue nd wrrnt surgicl excision since skin tests re unrelile nd ttempts to culture the orgnism from spinl fluid re futile. [10,12] Dignosis of the fungl species y culture my tke 3 4 weeks. The guidelines pulished y the Mycosis Study Group under the uspices of the Infectious Disese Society of Americ in 2008 recommend the intrvenous mphotericin B lipid formultions s first line tretment for CNS lstomycosis. [5] The use of mphotericin

4 Surgicl Neurology Interntionl 2015, 6:174 Figure 6: The CD56 () nd CD57 () stins show tht rre nturl killer cells re present B is often limited y nephrotoxity with prolonged use; therefore follow up with nother clss of ntifungls the zoles (fluconzole, itrconzole, nd voriconzole) fter initil therpy with mphotericin B is lso recommended. Voriconzole is rod spectrum trizole ntifungl, which hs excellent lood rin penetrtion independent of meningel inflmmtion. [11] It hs emerged s potentil first line tretment option for CNS lstomycosis. The clinicl experience with voriconzole in treting CNS lstomycosis in humns is limited. [1,2,4,8,14] Review of the literture reveled tht mny were cses of systemic lstomycosis with secondry CNS involvement, which hd een treted with mphotericin B nd other zoles on multiple occsions efore inititing tretment with voriconzole. Furthermore, most involved only medicl mngement nd surgicl excision ws not the prt of the tretment pln. Our ptient, electively strted on voriconzole fter surgicl excision nd course of mphotericin B, showed fvorle response nd continued to improve 12 months postopertively. While n isolted visul field deficit cn e difficult to detect if it is sutle, this cse report emphsizes the importnce of full visul field exmintion s prt of neurologicl exmintion. While we do not elieve tht the scess ws relted to some of the ptient s initil complints, such s the visul floters nd decresed hering on the left, we re certin tht the lesion does explin the visul field deficits found on exmintion. A congruous nd right sided qudrntnopi fits with lesion in the left occipitl loe, which ws the loction of our ptient s scess. Furthermore, our ptient hd improvement in visul fields fter the resection of the scess. MRI evlution of lstomycosis cn e difficult, nd these lesions cn pper consistent with intrcrnil mlignncies or other infectious conditions. In ddition, it is rre, ut possile, to hve n intrcrnil fungl scess without systemic disese. Our ptient hd no risk fctors for infection nor ny evidence of ny systemic mlignncy nd thus surgicl tretment ws the logicl pproch to oth dignosis nd tretment. Figure 7: Axil T1-weighted imge fter gdolinium dministrtion showing complete resolution of lesions with chronic encephlomlci t 1 yer postsurgery nd fter completing ntifungl tretment Finncil support nd sponsorship Nil. Conflicts of interest There re no conflicts of interest. REFERENCES 1. Bkleh M, Aksmit AJ, Tleyjeh IM, Mrshll WF. Successful tretment of cererl lstomycosis with voriconzole. Clin Infect Dis 2005;40:e Briol JR, Perry P, Ppps PG, Proi L, Sheley W, Wright PW, et l. Blstomycosis of the centrl nervous system: A multicenter review of dignosis nd tretment in the modern er. Clin Infect Dis 2010;50: Bell RM, Strshk RJ, Sty JR, Hr JM. Solitry intrcrnil lstomycotic scess. Wis Med J 1983;82: Borgi SM, Fuller JD, Sri A, El Helou P. Cererl lstomycosis: A cse series incorporting voriconzole in the tretment regimen. Med Mycol 2006;44: Chpmn SW, Dismukes WE, Proi LA, Brdsher RW, Ppps PG, Threlkeld MG, et l. Clinicl prctice guidelines for the mngement of lstomycosis: 2008 updte y the Infectious Diseses Society of Americ. Clin Infect Dis 2008;46: Chpmn SW. Blstomyces dermtitidis. In: Mndell GL, Dougls GR, Bennett JE, editors. Infectious Diseses nd Their Etiologic Agents. Principles nd Prctice of Infectious Disese. 3 rd ed. New York: Churchill Livingstone; p Chu JH, Feudtner C, Heydon K, Wlsh TJ, Zoutis TE. Hospitliztions for endemic mycoses: A popultion sed ntionl study. Clin Infect Dis 2006;42: Freifeld A, Proi L, Andes D, Bddour LM, Blir J, Spellerg B, et l. Voriconzole use for endemic fungl infections. Antimicro Agents Chemother 2009;53: Gonye EF. The spectrum of primry lstomycotic meningitis: A review of centrl nervous system lstomycosis. Ann Neurol 1978;3: Krvitz GR, Dvies SF, Eckmn MR, Srosi GA. Chronic lstomycotic meningitis. Am J Med 1981;71: Lutsr I, Roffey S, Troke P. Voriconzole concentrtions in the cererospinl fluid nd rin tissue of guine pigs nd immunocompromised ptients. Clin Infect Dis 2003;37: Morgn D, Young RF, Chow AW, Mehringer CM, Itshi H. Recurrent intrcererl lstomycotic grnulom: Dignosis nd tretment. Neurosurgery 1979;4: Morse HG, Nichol WP, Cook DM, Blnk NK, Wrd TT. Centrl nervous

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