Trichinellosis is a worldwide zoonotic disease caused by a nematode Trichinella spiralis.
We studied a case of Trichinella spiralis infection with severe eye involvement, febrile
condition, generalised malaise and muscular weakness in a young female patient.
Comprehensive ophthalmologic, infectologic, neurological and immunologic examinations
including electro diagnostic tests and CT scan of the head were performed, but the
diagnosis was confirmed only by histological examination of biopsy specimens of skeletal
muscle. The patient did not respond to standard corticosteroid therapy and improved
only after pulse doses of 1000 mg methylprednisolon. Although most authors recommend
moderately high doses of corticosteroids in the treatment of Trichinellosis, in
severe cases extremely high doses might be necessary

B. Vojniković

G. Zamolo

N. Brnčić

R. Budiselić

S. Njirić

S. Novak

English

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Coll. Antropol. 25 Suppl. (2001) 131–135UDC 616.995.132-085Original scientific paperSevere Trichinellosis Cured withPulse Doses of GlucocorticoidsB. Vojnikovi}1, N. Brn~i}2, G. Zamolo3, R. Budiseli}4, S. Njiri}1and S. Novak41 Department of Ophthalmology, Clinical Hospital Center, Rijeka, Croatia2 Department of Infectious diseases, Clinical Hospital Center Rijeka, Croatia3 Institute of Pathology, Medical faculty Rijeka, Croatia4 Department of Internal Medicine, Clinical Hospital Center, Rijeka, CroatiaA B S T R A C TTrichinellosis is a worldwide zoonotic disease caused by a nematode Trichinella spi-ralis. We studied a case of Trichinella spiralis infection with severe eye involvement, fe-brile condition, generalised malaise and muscular weakness in a young female patient.Comprehensive ophthalmologic, infectologic, neurological and immunologic examina-tions including electro diagnostic tests and CT scan of the head were performed, but thediagnosis was confirmed only by histological examination of biopsy specimens of skele-tal muscle. The patient did not respond to standard corticosteroid therapy and impro-ved only after pulse doses of 1000 mg methylprednisolon. Although most authors recom-mend moderately high doses of corticosteroids in the treatment of Trichinellosis, insevere cases extremely high doses might be necessary.IntroductionTrichinellosis is a worldwide zoonoticdisease closely related to cultural and di-etary habits caused by a nematode Tri-chinella spp. Human infection is acquiredthrough ingestion of undercooked meatcontaining infective encysted larvae1. Tra-umatic and immunological alterations areresponsible for the main clinical features,including diarrhea, febrile syndrome, myal-gias, oculopalpebral signs and eosinophi-lia.2 There has been an increase in thenumber oh human Trichinellosis cases inCroatia associated with war in the formerYugoslavia.Case ReportA 25-year old female was admitted toOphthalmologic facility after a 2-day pe-riod of eyelid oedema with spiking feverand suspicion on bilateral orbital cellu-litis. Physical examination was normalexcept her febrile condition and eye dis-131Received for publication June 4, 2001.turbances. The lids, conjunctiva and ex-traocular muscles were inflamed with se-vere conjuctival vascular congestion andbilateral protrusion (Figure 1). Fundusexamination showed bilateral vasculitiswith retinal haemorrhages (Figure 2).Orbital computerised tomography (CT)revealed inflamed and extraordinary thickextraocular muscles (Figure 3). Labora-tory parameters on admission showed:Erythrocyte sedimentation rate (ESR) 27,hemoglobin 13,.3 g/dL, white blood cells(WBCs) 7.5  109/L with 76% neutrophils,13% lymphocytes, 5% eosinophils, 4% mo-nocytes, 2% basophils, alanine transami-nase (ALT) 41.2 IU/L (normal < 31), as-partat transaminase (AST) 30.7 IU/L (nor-mal < 31.0) lactic dehydrogenase (LDH)230.5 IU/L (normal < 225), creatine kina-se (CK) 212.3 IU/L (normal < 145.0). Al-kaline phosphatase, amylase, bilirubin,urea, creatinine, prothrombine time, elec-trolytes, blood glucose, urin analysisshowed normal values.Following admission, antibiotic ther-apy along with oral 64 mg methyl-pred-nisolon and parabulbar injections of me-thyprednisolon acetate (Depo-Medrol) at40 mg was initiated. In spite of adminis-trated therapy, the high fever persistedfor another days, her condition was wors-ening with the appearance of headache,generalized malaise, dysphagia, weak-ness of masseters and neck muscles. Onfifth hospital day, neurological, infecto-logical and immunological examinationwere performed.Anamnestic epidemiological data di-rected to Trichinellosis and some similarcases in neighboring was categoric nega-tive. In spite of normal eosinophils, nega-tive epidemiological history, and non-re-spond to corticosteroid therapy, serologicexamination for Trichinellosis was indi-cated. In meantime, high fever persisted,weakness of muscles was progressing tolumbal region and patient was unable towalk. Electromyoneurographic (EMNG)132B. Vojnikovi} et al.: Pulse Doses of Corticoids, Coll. Antropol. 25 Suppl. (2001) 131–135Fig. 1. Conjunctival vascular congestion over athickened medial rectus muscleFig. 3. Orbital axial CT scan prior to therapyshowing bilateral proptosis and enormousextraocular muscles enlargementFig. 2. Fundus photography showing vasculitisand retinal haemorrhagesanalysis revealed a primary myogenicpattern. Electrocardiogram (ECG) exami-nation was normal. On 13. hospital dayshe was transferred to Department forimmunologic disorders with the diagnosisof possible acute polymyositis syndrome.Pulse dose of 1000 mg methylprednisolonwas started and fever critically fell down.Enzyme-linked immunosorbent assay(ELISA) IgM Trichinella test was positiveand IgH highly suspicious.Elevated serum creatine kinase andlactic dehydrogenase levels indicated con-siderable muscle involvement.3Pathohistological skin (Figure 4) andstriated muscle (Figure 5) biopsy findingwas as follows:»Histologically skin is built of normalepidermis with slightly hyaline thickenedbasal membrane. Around teleangiectaticcapillaries in papillary dermis fairly ex-tensive mononuclear infiltration can benoticed.Muscle tissue is built of variably widemuscle fibres among which cysts with en-capsulated parasites are found. Amongmuscle fibres in perimisium inflamed in-filtrates and eosinophil granulocytes arefound. Histological finding indicates Tri-chinellosis.«In that way definitive diagnosis wasestablished.On further inquiry, it was revealedthat the patient had eaten smoked porkmeat in Dalmatia two weeks prior to theonset of symptoms.Following initiating pulse dose of cor-ticosteroids, gradual but constant impro-vement of clinical and laboratory findingswas observed. Two weeks later orbital CTscan showed completely normalized find-ings of extraocular muscles (Figure 6). Af-ter 28 in-hospital days the patient wasdischarged and steroids were taperedover the next two months.133B. Vojnikovi} et al.: Pulse Doses of Corticoids, Coll. Antropol. 25 Suppl. (2001) 131–135Fig. 4. Striated muscle (HE) showing encapsu-lated larvae of Trichinella spiralis ( 400)Fig. 5. Skin (HE) – perivascular mononuclearinfiltrate in dermis ( 400).Fig. 6. Orbital axial CT scan after corticoste-roid pulse therapy – normalization of findingsDiscussionThe patient here described presentedwith severe immunologic reaction to Tri-chinella spiralis infestation.Although ocular involvement is com-mon in Trichinellosis, in this case ocularclinical features were particularly strik-ing, especially in the initial phase of thedisease. Therefore the patient was emer-gently admitted to Eye clinic with suspi-cion on bilateral orbital cellulitis.Eyelid oedema, conjunctival congesti-on and subconjuctival haemorrhages andbilateral protrusion of the eye balls due toenormous extraocular muscles enlarge-ment have been present. Consequentlyall the ductions and the versions havebeen reduced causing diplopia due to re-strictive myopathy.On ophthalmoscopic examination thetypical traits of ocular pathology includ-ing toxic vasculitis and retinal haemor-rhages mostly in the macular region havebeen found.In spite of started corticosteroid ther-apy (64 kg methyl prednisolon orally)proptosis persisted and there was stillpain on attempted moving the globe. Theclinical status of the patient progressi-vely deteriorated.Orbital CT scanning revealed enlargedextraocular muscles but no retrobulbarexpanding process or orbital infiltration.At this point the most likely diagnosessuggested by CT scan seemed orbitalmyositis or endocrine ophthalmopathy.Besides constant fever during the co-urse of the disease, there were also my-algia, dysphagia4, muscular weakness andgeneralised malaise so acute polymyosi-tis syndrome was suspected. Trichinello-sis was also included in the differentialdiagnosis and testing in that directionwas started. Serious diagnostic problemwas the absence of corresponding epide-miological data, at least in the beginningof investigation.5As in all cases when doubt remains,muscle biopsy was indicated and it dem-onstrated the presence of Trichinella spi-ralis encapsulated larvae in striated mus-cle specimen. Serologic tests supportedthe diagnosis, too.In the meantime corticosteroid pulsetherapy was started as well as parabul-bar injections of methyl prednisolon-ace-tate at 40 mg. Only then patient prompt-ly started to improve.ConclusionTreatment of Trichinellosis is defini-tely quite difficult. Since the manifesta-tion of the disease is of variable intensity(depending upon the extent of invasionand the immune response of the host),the treatment is related to severity of thedisease. Anthelmintic therapy is usedonly for an early period, probably in thefirst 3 days following infection.6 If appliedit prevents subsequent muscular inva-sion but has little if any effect on musclelarvae. In the treatment of symptomaticTrichinellosis most authors recommendoral prednisolon at 30 to 60 mg/day for aperiod of 10–14 days.7,8,9 They are used intreatment of acute stage of Trichinellosissince they suppress symptoms and signsof immediate type hypersensitivity andshorten the duration of the disease.But in this case the patient did not re-spond to such therapy and even got worsedeveloping most disturbing clinical sta-tus. Therefore, unconventionally highdosed corticosteroid therapy was startedand only at his point improvement wasobserved.It is concluded that the decision aboutthe treatment should be made on individ-ual basis.134B. Vojnikovi} et al.: Pulse Doses of Corticoids, Coll. Antropol. 25 Suppl. (2001) 131–135R E F E R E N C E S1. TARATUTO, A. L., Brain Pathol., 7 (1997)663.— 2. DWORKIN, M. S., H. R. GAMBLE, D. S. ZAR-LENGA, P.O. TENNICAN, J. Infect. Dis., (1996) 663.— 3. CHURCHILL, D. R., C. MORRIS, A. FAKOYA,J. Infect, 33 (1996) 103. — 4. WANG, Z. Q., J. CUI, F.WU, F. R. MAO, X. X. JIN, Acta Trop., 71 (1998) 255.— 5. NIKOLIC, S., M. VUJOSEVIC, M. SASIC, J.POLUGA, S. MISIC, Lj. NAJDANOVIC, Srp., Arh.,126 (1998) 209. — 6. DENNETT, X., S. J. SIEJKA, J.R. ANDREWS, I. BEVERIDGE, D. M. SPRATT, Med.J. Aust., 168 (1998) 226. — 7. KOCIECKA, W., Vet.Parasit., 93 (2000) 365. — 8. PRIN, L., S. DUBU-CQUOI, Med. Trop., 58 (1998) 433. — 9. JASMER, D.P., J.Cell Biol., 21 (1993) 785.B. Vojnikovi}Clinical Hospital Center Rijeka, Department of Ophthalmology, Kre{imirova 42,51000 Rijeka, CroatiaTE@AK OBLIK TRIHINELOZE IZLIJE^EN VISOKIM DOZAMAGLUKOKORTIKOIDAS A @ E T A KTrihineloza je zoonoza ra{irena u cijelom svijetu uzrokovana nematodom Trichinelaspiralis. Prikazan je slu~aj mlade djevojke zara`ene Trihinellom spiralis s te{kom sli-kom o~ne patologije, febrilitetom i progresivnom slabo{}u skeletne muskulature. Izvr-{eni su temeljiti oftalmolo{ki, infektolo{ki, neurolo{ki i imunolo{ki pregledi uklju~uju}ielektrodijagnosti~ne pretrage kao i CT glave, ali je kona~na dijagnoza potvr|ena tekna temelju patohistolo{kog nalaza biopsije uzorka skeletnog mi{i}a. Pacijentica nijereagirala na uobi~ajene doze kortikosteroida, a do pobolj{anja je do{lo tek nakon pri-mjene pulsnih doza od 1000 mg metilprednizolona. Iako ve}ina autora preporu~ujeumjereno visoke doze kortikosteroida u lije~enju trihineloze, te{ki slu~ajevi mogu po-nekad zahtijevati ekstremno visoke doze.135B. Vojnikovi} et al.: Pulse Doses of Corticoids, Coll. Antropol. 25 Suppl. (2001) 131–135

Severe Trichinellosis Cured with Pulse Doses of Glucocorticoids

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