Eur Radiol (2009) 19: 1560–1563DOI 10.1007/s00330-008-1130-7 INTERPRETATION CORNER

Alberto MunozJesus Ruiz-ContrerasAna JimenezIrene MatéMarta CalvoMiguel VillafruelaGloria del-Pozo

Received: 23 February 2008Revised: 23 April 2008Accepted: 27 May 2008# European Society of Radiology 2008

Bilateral tuberculous otomastoiditis

in an immmunocompetent 5-year-old child: CT

and MRI findings (2009: 3b)

Abstract Bilateral tuberculous mas-toiditis (TOM) in an immunocompe-tent child is a very uncommon form oftuberculous infection presentation.This report shows the CT and MR

imaging of bilateral tuberculous oto-mastoiditis consisting of aggressivesigns of middle ear and mastoidinvolvement with bony destructionand periauricular collections with nosigns of brain involvement. Differen-tial diagnosis at pediatric age ofdestructive lesions such as mainlyaggressive forms of histiocytosis isunderscored. This form of bilateralTOM at this early age has not beendescribed from a radiologicalperspecitve.

Keywords Bilateral tuberculousotomastoiditis . Chronic supurativeotitis . MR imaging .Immunocompromised patients

A. Munoz . J. Ruiz-Contreras .A. Jimenez . I. Maté . M. Calvo .M. Villafruela . G. del-PozoHospital Universitario “12 Doce deOctubre”, Universidad Complutense,Madrid, Spain

J. Ruiz-Contreras . A. JimenezImmunodeficency Unit,Madrid, Spain

A. Jimenez . I. MatéPediatrics,Madrid, Spain

Introduction

Bilateral TOM is a rare cause of chronic supurative otitismedia and is more commonly seen in non-immunocom-petent adults. In immunocompetent individuals, there is aconsiderable delay before the diagnosis because of its lowprevalence and insidious clinical signs, which can lead toirreversible complications. Although the diagnosis relieson the microbiological identification of Mycobacteriumspecies, radiological studies may suggest specific signs ofaggressiveness that can suggest uncommon causes ofmiddle ear and mastoid cavity involvement early on.Furthermore, MR imaging can provide new additionalinformation regarding tissue relaxation times of chronicinflammatory soft tissue deposition.

Case report

A 3-year-old boy with a history of bilateral retroauriculartumefaction for the past 3 months came to his primary carepediatrician and was diagnosed with bacterial cervical lym-phadenitis; he then was treated with several courses of anti-inflammatory agents and antibiotics with no clear resolutionand persistence of intermittent fever and tumefaction.

Two weeks later he was brought to the emergency wardof our hospital because of a new episode of 2-day durationconsisting on a new left retroauricular tumefaction with earpain and fever. Otoscopy showed tympanic perforation andthick purulent discharge. Seen by the ENT service, thechild was clinically diagnosed to have acute mastoiditis,and surgical drainage was performed. Samples for bacterial

M. Calvo . M. VillafruelaOtorhinolaryngology,Madrid, Spain

G. del-PozoPediatric Radiology,Madrid, Spain

A. Munoz (*)Hospital Universitario – Radiology,Glorieta de Malag s/n,Madrid, 28041, Spaine-mail: almugo@teleline.esTel.: +34-91-3908650

cultures and pathology were taken. Thereafter, a 7-daycourse of IV antibiotics was given with clear clinicalimprovement. The patient was discharged on an extra weekof oral antibiotics.

However, the patient continued to have fever, and revisioncarried out some days later revealed not only worsening ofthe left ear, but also a new retroauricular tumefaction on theright side and fever. With the diagnosis of bilateralotomastoiditis, the service of Pediatric Hematology andOncology was consulted to rule out histiocytosis. The childwas then admitted to the Immunodeficiency Unit for furtherdiagnostic studies, and treatment with intravenous anti-biotics was continued.

The boy was born in Venezuela and had been living inSpain for the past 8 months. He had no remarkable pastmedical history. Vaccinations were up to date, includingBCG administered at birth. He had no known contact withtuberculosis. No contact with pets or with farm or wildanimals in Venezuela or Spain was reported. The parentsdenied ingestion of non-controlled food or non-pasteurizeddairy products. A purified protein derivative (PPD), done inVenezuela 1 month prior to the child’s coming to Spain, wasnegative. On physical examination, the child had a goodgeneral appearance, with no signs of respiratory distress orwasting. Heart and lung auscultations were normal. Abdom-inal examwas also normal, with no tenderness or masses; theliver and spleen were not felt. Bilateral submandibularlymphadenopathy was present along with bilateral retro-auricular tumefaction, with displacement of both pinna. Thearea was tender, hot, and red. Otoscopy was then normal. Noother abnormalities were found.

Laboratory tests such as CBC showed 9,700 whitecells with a differential count of 46% neutrophils, 40%lymphocytes, 7.3% monocites, and 5.8% eosinophils.Other tests showed: hemoglobin 10.3 g/dl; MCV 67.3 fl;RDW 16.7%; C reactive protein (CRP): 6.8 mg/dl and ESR60 mm/h. Renal and liver function tests were normal. Nobacteria were isolated in cultures from otic aspirate. Oticaspiration for bacterial culture, blood cultures, serologiesfor HIV, syphilis, CMV, toxoplasma, and three stoolsamples for parasites and ova were taken, and all werenegative. Immunologic studies were normal. A Mantouxtest showed a 20-mm induration after 48 h. Cranial CT(Fig. 1) and MRI (Fig. 2) with specific studies of mastoidand tympanic cavities were performed showing filling ofboth cavities, with signs of osteitis, periauricular collec-tions, and neck adenoapthies. No abnormalities of thebrain, including the pituitary gland or hypothalamus, werefound. The pathology results of the sample taken in thesurgical draining of the ear were then available anddemonstrated non-necrotizing granuloma. With theseresults, a diagnosis of TOM was considered as the mostlikely diagnostic option, so gastric aspirates on 3consecutive days were collected, and a plain chest X-rayand body CT scan were done to rule out more extensivetuberculous involvement. All imaging studies were nega-

tive. Mycobacterium tuberculosis grew in gastric aspiratesseveral weeks later. Antituberculous treatment with fourdrugs (isoniazide, rifampicin, etambuthol, and pirazina-mide) was then started with progressive clinical improve-ment in later and final clinical resolution of the symptoms.

Discussion

Radiologic reports of TOM are scant and mainly based onCT findings. Rho et al. [1] have recently reported the CTfindings of TOM in adults focusing on its differentialdiagnosis with cholesteatomatous otitis. CT findings ofTOM in this study were soft tissue in the entire middle earcavity, preservation of mastoid air cells without scleroticchange, and soft tissue extension or mucosal thickening ofthe external auditory canal (EAC) with intact scutum.These CT findings were consistent or not consistent withother reports [2, 3].

On radiological grounds, in adults and in the early stagesof the disease, soft tissue is noted in the tympanic cavity,and erosion of the ossicles and of the walls of the tympaniccavity takes place. Destruction of the inner ear structures,retroauricular abscesses, and mastoid fistula can be seen inthe advanced stages [4]. CT is the first imaging modalityfor the evaluation of middle ear diseases. However, inpatients with neurologic complications, such as facial nerveand labyrinth involvement, only contrast-enhanced MRIcan demonstrate the affected areas [5]. MR has theadvantage of superior soft tissue discrimination and isparticularly useful for assessing soft tissue planes aroundthe skull base and abnormalities of the medullary cavity ofbone. Highly sensitive but nonspecific MR findings ofosteomyelitis include marrow T1 hypointensity and T2hyperintensity [6]. MR imaging can also demonstrate far

Fig. 1 Axial contrast-enhanced CT of the temporal bones (bone-tissue algorithm) shows filling of both tympanic and mastoidcavities by soft tissue material, erosions of both mastoid septa, largeright osseous defect (arrows), and bilateral periauricular soft tissueswelling

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better than CT concomitant intracranial tuberculous dis-ease, such tuberculomas, hydrocephalus, and base of theskull involvement.

Our patient presents with a rather striking form ofbilateral middle ear and mastoid bone involvement. On CT,signs of complicated otomastoiditis (CO) consisting offilling of the middle ear and mastoid bone air cells withextensive bony erosion and intratympanic and periauricularcollections were seen. On MR, the tympanic cavity and themastoid bone were filled with low T1 and T2 signalmaterial that diffusely enhanced on postcontrast T1-weighted sequences. Additional extensive periauricular,intratympanic, and subtemporal collections were observed.Most of these findings could be seen on other morecommon infectious causes of CO. However, low T2 signalwith cerebroid-like appearance and extensive coalescentmaterial that enhances on post-contrast studies is nottypically seen in common inflammatory diseases of themiddle ear and petrous bone, but rather hyperintense T2collections. Such findings may help in the diagnosis ofother less frequent diseases, such as tuberculous involve-ment, and may reflect the histologic composistion ofcaseation necrosis. Finally, on radiological grounds,despite CT exquisitely demonstrating the destructivechanges of the bony mastoid septa and tympanic cavityas well as the main bulky soft tissue collections, MRdepicted additional extensive soft tissue involvement andsubtemporal enhancement. Furthermore, it excluded mem-branous labyrinthine involvement or other brain andmeningeal complications.

Differential diagnosis in cases of children with a bilateraldestructive otomastoid process is limited and includesother infections, particularly in HIV-positive and othernon-immunocompetent patients, such as histiocytosis and

malignancies, particularly leukemia (bilateral granulocyticsarcoma).

In our case, the age of the patient, the lack of any historyof neoplasia, and the paucity of infection’s signs, such aselevated ESR, led us to considerer a chronic skull infection.However, the bilateral otomastoid destructive changes andthe rarity of bilateral TOM in children led us to also includehistiocytosis involvement. Few reports on this subject havebeen made on MR imaging when histiocytosis involves thepetrous bone. Extensive, sometimes bilateral, mastoiddestruction with enhancing soft tissue and high T2hyperintense lesions within the tissue are the mainradiological signs [7]. These findings were not seen inour patient, but rather iso-hypointense signal on the mainMR sequences, which further diffusely enhances on post-contrast T1-weighted studies.

TOM is considered a prototype disease for most of thetrue granulomas that can occur in the temporal bone.Histologic studies of tuberculous lesions generally revealcaseous foci that are surrounded by epithelioid or histio-cytic cells, multinucleated inflammatory cells, and mixedacute and chronic inflammatory cells, as well as fibrousgranulation tissue. Definitive diagnosis, however, requiresidentification of the acid-fast bacilli by either culture orappropriate tissue staining. In our particular case, thesamples taken from the surgical draining demonstratednon-necrotizing granulomas, but were negative for theidentification of the bacilli, and Mycobacterium tubercu-losis only grew from the gastric aspirates. The rate ofpositive microbiological diagnosis from otic samples islow. Recently, Nishiike et al. [2] described that smears ofpatient’s otic secretion for acid- and alcohol-fast bacilli werepositive in only 3 of 12 patients. Six microscopic examina-tions showed that acid-fast bacilli should be present in only

Fig. 2 (a-c) MR imaging at a similar level. a and b: Axial SE T1-weighted (500/12/2; TR/TE/ex) (a) and FSE T2-weighted(6,000/100/2; TR/TE/ex) MRs (b) show respectively extensiveiso- and low intensity soft tissue material filling both temporal andmastoid bone sparing the inner ears and right middle ear andperiauricular T2-hyperintense collections (asterisks). c: Axialenhanced T1-weighted spin echo sequences (500/12/2; TR/TE/ex)

with fat saturation technique show bilateral soft tissue enhancementthat fills external and middle ear cavities, mastoid cells, andperiauricular soft tissues. Notice right middle ear and periauriculartissue collections (arrows). Potential intratemporal facial nerveenhancement cannot be differentiated from background soft tissueenhancement. Notice the lack of enhancement of labyrinthinestructures

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one granuloma. Also Rho et al. report [1] only 5 of our 19patients had positive results for acid-fast bacilli staining onhistologic examination. However, most patients showedtypical caseation necrosis and Langerhans giant cells.

The pathogenesis of TOM is postulated to involve threemechanisms: (1) aspiration of mucus through the eusta-chian tube, (2) hematogenous dissemination from othertuberculosis foci, or (3) direct implantation through theEAC and a tympanic membrane perforation [8, 9]. The factthat our patient had bilateral otomastoid involvement andthe positive culture taken from the gastric aspirates leads usto suspect that in this particular case the first mechanism isresponsible for the infection, although we cannot rule outcompletely the cervical lymphadenopathy present in ourpatient as the primary infection source. Furthermore,

tuberculous lymphadenopathy is the most commonmanifestation of this disease in the head and neck, affecting5%–10% of all patients with tuberculosis [10]. In Rho etal.’s 10-year experience [1], they obtained hystopathologicdiagnosis of cervical tuberculous lymphadenitis in morethan 100 patients by sonographically guided biopsy. In oneof their cases, a 5-year-old male patient complained of leftotorrhea and ipsilateral cervical lymphadenopathy, and thediagnosis was made by tissue biopsy of the cervicallymphanedopathy.

In uncomplicated cases TOM is usually treated withantituberculous drugs. Surgery is indicated for complicatedcases, such as those involving subperiosteal abscess ofthe mastoid, facial palsy, postauricular fistulas, or CNSextension of infection.

References

1. Rho MH, Kim DW, Kim SS et al(2007) Tuberculous otomastoiditis onhigh-resolution temporal bone CT:comparison with nontuberculous oto-mastoiditis with and without choleste-atoma. Am J Neurora 28:493–496

2. Nishiike S, Irifune M, Doi K et al(2003) Tuberculous otitis media: clini-cal aspect of 12 cases. Ann Otol RhinolLaryngol 12:935–938

3. Vaamonde P, Castro C, Garcia-Soto Net al (2004) Tuberculous otitis media: asignificant diagnostic challenge. Oto-laryngol Head Neck Surg 130:759–766

4. de Castro CC, de Barros NG, de SousaCampos ZM, Cerri CC (1995) CT scansof cranial tuberculousis. Radiol ClinNorth Am 33:753–769

5. Kinoshita T, Ishii K, Okitsu T et al(2001) Facial nerve palsy: evaluationby contrast-enhanced MR imaging.Clin Radiol 56:926–932

6. Chang PC, Fischbein NJ, Holliday RA(2003) Central skull base osteomyelitisin patients without otitis externa: im-aging findings. Am J Neuroradiol24:1310–1316

7. Bonafeé A, Joomye H, Jaeger P et al(1994) Hitiocytosis X of the petrousbone in the adult: MRI. Neuroradiology36:330–333

8. Greenfield BJ, Selesnick SH, Fisher Let al (1995) Aural tuberculosis. Am JOtol 16:175–182

9. Hyams VJ (1982) Granulomatous dis-orders of the temporal bone. Otolaryn-gol Clin North Am 15:515–518

10. Cleary KR, Batsakis JG (1995) Myco-bacterial disease of the head and neck:current perspective. Ann Otol RhinolLaryngol 104:830–833

Precisely correct answer was received by closing date from:

Luiz Coelho, Curitiba, BrazilMarc De Baets, Lugano, Switzerland

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