Chan, DTM

Choy, CBY

Fang, XHB

Mok, MY

Journal of the Formosan Medical Association

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Title Invasive fungal rhinosinusitis presenting as Bell's palsy in akidney and liver transplant recipientAuthor(s) Mok, MY; Fang, XHB; Choy, CBY; Chan, DTMCitation Journal of the Formosan Medical Association, 2017, v. 116, p.910-911Issued Date 2017URL http://hdl.handle.net/10722/247339Rights This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.Journal of the Formosan Medical Association (2017) 116, 910e911Available online at www.sciencedirect.comScienceDirectjournal homepage: www.jfma-onl ine.comCORRESPONDENCEInvasive fungal rhinosinusitis presenting asBell’s palsy in a kidney and liver transplantrecipientMaggie Ming Yee Mok a,*, Benjamin Xin Hao Fang b,Bo Ying Choy a, Tak Mao Chan aa Division of Nephrology, Department of Medicine, Queen Mary Hospital, University of Hong Kong,Hong Kongb Department of Radiology, Queen Mary Hospital, University of Hong Kong, Hong KongReceived 11 October 2016; accepted 17 April 2017A 73 year-old man with type II diabetes mellitus, hepatitis Crelated liver cirrhosis received a live-donor liver transplantin 2004. A year later, he was diagnosed to have mesangio-proliferative glomerulonephritis with deteriorating renalfunction unresponsive to a course of corticosteroid. Herequired regular hemodialysis in 2006. He received acadaveric renal transplant in 2008. The post-transplantcourse was uneventful. His maintenance immunosuppres-sion included prednisolone 5 mg daily, cyclosporin A (troughlevel around 70 mg/L) and mycophenolate sodium 180 mgtwice daily. Serum creatinine level had been stable at100e110 mmol/L. He presented with symptoms of sinusitisin 2014, and was treated with amoxicillin/clavulanate375 mg thrice daily for a week, intranasal corticosteroids,and nasal irrigation. Intermittent use of intranasal corti-costeroids was continued for recurrent symptoms. Sixmonths later, he presented with right-sided facial palsy ofsudden onset and right ear pain. Physical examinationshowed lower motor neuron type of right cranial nerve (CN)VII palsy without additional neurological abnormality. Hewas afebrile with normal white cell count. There were no* Corresponding author. Professorial Block 405, Queen Mary Hos-pital, University of Hong Kong, 102 Pokfulam Road, Hong Kong. Fax:þ852 2855 1143.E-mail address: maggiemymok@gmail.com (M.M.Y. Mok).http://dx.doi.org/10.1016/j.jfma.2017.04.0120929-6646/Copyright ª 2017, Formosan Medical Association. PublishedCC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/vesicles around the right auricle. Bell’s palsy (House-Brackmann Grade V) was diagnosed and he was treatedwith prednisolone 60 mg daily for one week. MagneticResonance Imaging of the brain (Fig. 1) performed a weeklater revealed right mastoid effusion, otitis media andmaxillary sinusitis, and also superior sagittal sinus throm-bosis. Right myringotomy and maxillary sinus antrostomywere performed immediately, which showed a fungal balllesion. Histological examination revealed fungal hyphaewith leucocyte infiltrates, but fungal culture was negative.By internal transcribed spacer sequencing, the fungus wasidentified as Aspergillus species. Endoscopic sinusdebridement was performed twice. He was treated withintravenous anidulafungin for 4 months and oral vor-iconazole was extended for 15 months. He is continued onoral voriconazole as interval imaging still demonstratesinusitis. His CN VII palsy had complete recovery.This patient illustrates an unusual presentation offungal rhinosinusitis masquerading as Bell’s palsy. Invasivefungal infection is relatively uncommon in kidney trans-plant recipients. An incidence rate of 1.3% at 1-year hasbeen reported, compared with 4.7% in liver transplantrecipients.1 Common risk factors include diabetes melli-tus, high-dose corticosteroid therapy, recent or prolongedantibiotic use, long-term hospitalization, neutropenia, andgraft failure.2 Diabetes mellitus is also associated with aby Elsevier Taiwan LLC. This is an open access article under the4.0/).Figure 1 Post-gadolinium T1-weighted fat saturated mag-netic resonance image showing retention and mucosalenhancement and thickening over right maxillary sinus inkeeping with sinusitis. Abnormal enhancement was also notedover right middle ear and petrous part of right temporal bonein keeping with otitis media and inflammatory changesinvolving the temporal bone.Fungal rhinosinusitis Bell’s palsy transplant recipient 911higher risk of Bell’s palsy, especially in those greater than40 years-old.3 This patient was exposed to a short course ofhigh-dose systemic corticosteroid prior to the diagnosis.Whether the prior course of antibiotics and prolongeduse of intranasal corticosteroid inhalation might haveprecipitated the fungal infection, or the latter had beenpresent from the start when the patient presented withsinusitis remained speculative.4 Aspergillus and Mucoralesare the two micro-organisms most commonly associatedwith invasive fungal rhinosinusitis. Both show a tendencyfor vascular invasion, giving rise to tissue infarction andthrombosis, as demonstrated in our patient. Other poten-tial consequences include bony erosion, complicationsaffecting the eyeball or surrounding cranial nerves, andmeningoencephalitis. Fungal rhinosinusitis is thus aserious complication which could result in mortality.Treatment should be rapid and aggressive, involving bothsurgical debridement and prolonged systemic anti-fungaltreatment.5Conflicts of interestThe authors have no conflicts of interest relevant to thisarticle.References1. Pappas PG, Alexander BD, Andes DR, Hadley S, Kauffman CA,Freifeld A, et al. Invasive fungal infections among organ trans-plant recipients: results of the Transplant-Associated InfectionSurveillance Network (TRANSNET). Clin Infect Dis 2010;50(8):1101e11.2. Trabelsi H, Neji S, Sellami H, Yaich S, Cheikhrouhou F,Guidara R, et al. Invasive fungal infections in renal transplantrecipients: about 11 cases. J Mycol Med 2013;23(4):255e60.3. Yanagihara N, Hyodo M. Association of diabetes mellitus andhypertension with Bell’s palsy and Ramsay Hunt syndrome. AnnOtol Rhinol Laryngol Suppl 1988;137:5e7.4. Chang GH, Wang WH. Intranasal fungal (Alternaria) infectionrelated to nasal steroid spray. Am J Otolaryngol 2013;34(6):743e5.5. Epstein VA, Kern RC. Invasive fungal sinusitis and complicationsof rhinosinusitis. Otolaryngol Clin North Am 2008;41(3):497e524. viii.

Invasive fungal rhinosinusitis presenting as Bell's palsy in a kidney and liver transplant recipient

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Invasive fungal rhinosinusitis presenting as Bell's palsy in a kidney and liver transplant recipient

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