Objective: We report an adult case of nasopharyngeal carcinoma treated with radical chemo-radiotherapy, with subsequent development of a histologically proved temporal cavernous haemangioma within the radiation field. Method: Case report and review of the current literature concerning radiation-induced, secondary, space-occupying lesions. Conclusion: The increasing role of radiotherapy in nasopharyngeal carcinoma treatment, together with improved patient survival, is likely to lead to radiation-induced, secondary, space-occupying lesions being encountered more frequently. We emphasise the need to be vigilant for this important but relatively rare complication, which has significant associated morbidity

Ellenbogen, JR

Joshi, SM

Kitchen, N

UCL Discovery

The Journal of Laryngology & Otologyhttp://journals.cambridge.org/JLOAdditional services for The Journal of Laryngology & Otology:Email alerts: Click hereSubscriptions: Click hereCommercial reprints: Click hereTerms of use : Click hereDevelopment of cavernous haemangioma following radical chemo­radiotherapy for nasopharyngeal carcinomaJ R Ellenbogen, S M Joshi and N KitchenThe Journal of Laryngology & Otology / Volume 123 / Issue 08 / August 2009, pp 925 ­ 927DOI: 10.1017/S0022215108003605, Published online: 17 September 2008Link to this article: http://journals.cambridge.org/abstract_S0022215108003605How to cite this article:J R Ellenbogen, S M Joshi and N Kitchen (2009). Development of cavernous haemangioma following radical chemo­radiotherapy for nasopharyngeal carcinoma. The Journal of Laryngology & Otology, 123, pp 925­927 doi:10.1017/S0022215108003605Request Permissions : Click hereDownloaded from http://journals.cambridge.org/JLO, IP address: 144.82.107.164 on 10 Oct 2012Clinical RecordDevelopment of cavernous haemangioma following radicalchemo-radiotherapy for nasopharyngeal carcinomaJ R ELLENBOGEN, S M JOSHI, N KITCHENAbstractObjective: We report an adult case of nasopharyngeal carcinoma treated with radical chemo-radiotherapy, withsubsequent development of a histologically proved temporal cavernous haemangioma within the radiation field.Method: Case report and review of the current literature concerning radiation-induced, secondary,space-occupying lesions.Conclusion: The increasing role of radiotherapy in nasopharyngeal carcinoma treatment, together withimproved patient survival, is likely to lead to radiation-induced, secondary, space-occupying lesions beingencountered more frequently. We emphasise the need to be vigilant for this important but relatively rarecomplication, which has significant associated morbidity.Key words: Cavernous Hemangioma; Radiotherapy; Nasopharyngeal CarcinomaIntroductionAlthough nasopharyngeal carcinoma (NPC) is a rare tumour,it accounts formore than 95percent of nasopharyngealmalig-nancies in adults and 20 to 35 per cent of those in children.1 Itis a highly morbid disease with a poor survival outcome. Theuse of radiotherapy to treat head and neck tumours is increas-ing, either as primary or adjunctive therapy. The risk ofradiation-induced, secondary, space-occupying lesions is agrowing topic of investigation and concern.We report an adult case of NPC treated with radicalchemo-radiotherapy, with subsequent development of ahistologically proved cavernous haemangioma within theradiation field. Such a case has not previously beenreported in the literature. The increasing role of radiother-apy in NPC treatment, and improved patient survival rates,are likely to lead to these secondary tumours been encoun-tered more frequently. We also review the current litera-ture on this condition.Case reportA 57-year-old woman presented in April 2000 with painand deafness in the left ear and left facial numbness.Examination under anaesthesia and imaging at that stageshowed an extensive nasopharyngeal carcinoma (NPC)eroding through the pterygoid plate and invading the sphe-noid and the posterior ethmoid sinus, with a lymph nodepresent in the retropharynx. Biopsies confirmed an undiffer-entiated carcinoma of nasopharyngeal type, and furthermagnetic resonance imaging (MRI) identified extensivedisease throughout the sphenoid, extending to the pituitaryand middle cranial fossa. The disease did however appearto be extradural. Unusually for such an extensive primarytumour, the patient had no cervical lymphadenopathy.Thepatientwent on to receive radical chemo-radiotherapy.Following her treatment, further biopsies taken in June 2000confirmed that she had residual disease within thenasopharynx.The patient underwent further full adjuvant chemother-apy treatment with cisplatin and 5-fluorouracil. At thispoint, residual tumour was resected and gold grains wereimplanted at the primary site.Following this treatment, the patient remained disease-free. However, she suffered sequelae as a consequence ofher radical treatment, namely, pain in the trigeminalnerve distribution on the left side, deafness in the left earand loss of vision in the left eye.In 2001, computed tomography scanning identified anarea in the left temporal lobe suggestive of necrosis second-ary to radiotherapy, and MRI scans taken between May2006 and early 2007 showed that this area had developedinto a large, cystic mass (Figure 1). In June 2006, a positronemission tomography scan, performed to exclude recur-rence, showed no uptake within this region, despitenormal uptake elsewhere in the brain.In April 2007, the patient was admitted to The NationalHospital for Neurology andNeurosurgery for a left temporalcraniotomy for resection of the cystic tumour. At that time,she complained of her continued left facial pain, unsteadygait and decreased memory. Intra-operatively, a large,cystic lesion in the left temporal lobe with what appearedto be a cavernous haemangioma at the base was excised.Histological analysis confirmed a cavernous haemangiomaand gliotic brain tissue.After surgery, the patient’s condition improved bothphysically and emotionally, and her pain was less intrusive.DiscussionThe typical radiation field for nasopharyngeal carcinoma(NPC) treatment encompasses the adjacent skull base,sphenoid sinus, middle cranial fossa and the soft tissueFrom the Department of Neurosurgery, The National Hospital for Neurology and Neurosurgery, London, UK.Accepted for publication: 2 July 2008. First published online 17 September 2008.The Journal of Laryngology & Otology (2009), 123, 925–927.# 2008 JLO (1984) Limiteddoi:10.1017/S0022215108003605925juxtaposed to the nasopharynx.1 Patients undergoing a fullcourse of radiotherapy for NPC will receive a dose of 66–70 Gy to this large area. This is above the 55 Gy at whichthe dose–response relationship between radiation doseand secondary induced tumour risk has been demonstratedto increase.2The common complications of radiotherapy to the headand neck have been well documented. Some rarer compli-cations which have been reported after NPC radiotherapy,with potentially serious consequences, include temporalbone necrosis and abscess formation,3 temporal bonetumours,4 sarcomas,2 and, in the current case, cavernoushaemangioma. The five-year incidence of secondaryprimary tumours following radiotherapy for NPC hasbeen shown to be 5.8 per cent.5Cavernous haemangiomas are vascular malformationscomposed of dilated, thin-walled capillaries consisting ofa single layer of endothelium and differing quantities ofsubendothelial fibrous stroma, with absence of smoothmuscle and elastic fibres. These sinusoidal-type vesselsare immediately in apposition to each other, without anyrecognisable intervening neural parenchyma. They canvary in size from punctate to several centimetres across,and can occur anywhere in the intracranial parenchyma,as well as in the spinal cord and on cranial and spinalnerve roots.Although a benign vascular developmental malfor-mation, cavernous haemangiomas have been identified tooccur as a result of radiation therapy. Radiation-induced,de novo formation of cavernous haemangiomas is rarelyreported, but recently has been observed with increasingfrequency. The majority of such lesions have been observedin the male paediatric population.6 One study demon-strated a sixfold prevalence rate of cavernous haemangio-mas in children aged between two to 11 years at the timeof radiotherapy, compared with that cited in the literaturefor this population. The latency interval between radiother-apy and cavernous haemangioma development has beenfound to be three to 102 months (median 37 months).7Cavernous haemangiomas have also been reported tooccur in the radiation field of children, seven to 19 yearsafter radiation treatment for paediatric brain tumours(medulloblastomas, ependymoma and midbrain astrocy-toma).8 A review of de novo cavernous malformations ofthe central nervous system arising after radiotherapyidentified a mean latency period until lesion detection of8.9 years after a mean radiation dose of 60.45 Gy, acrossall primary tumours in adults and children.6 Radiation-induced vascular malformations have been found to occurin a noticeably shorter time interval, compared with otherbenign, radiation-induced tumours.9Although rare, these secondary tumours are associatedwith significant morbidity. Patients with cavernous hae-mangiomas may remain asymptomatic and unaware oftheir vascular lesion; however, they may alternativelypresent with symptoms of intracranial haemorrhage, sei-zures, progressive or transient neurological deficits, andheadaches. The risk of haemorrhage has been suggestedto be between 0.25 and 0.7 per cent per lesion per year.10,11The pathophysiologicalmechanisms of radiation-inducedcavernous haemangioma formation are not clearly under-stood. Various authors have suggested mechanisms predis-posing to cavernous malformation secondary to radiation,including: genetic mutation; growth factor up-regulation;radiation-induced vessel wall necrosis; and vessel wallchanges including cell swelling, hyalinisation, fibrosis andmineralisation.6,12 However, such theories are yet to besubstantiated.. The increasing role of radiotherapy innasopharyngeal carcinoma (NPC) treatment,together with improved patient survival, is likely tolead to radiation-induced, secondary,space-occupying lesions being encountered morefrequently. This report describes an adult case of NPC treatedwith radical chemo-radiotherapy, with subsequentdevelopment of a cavernous haemangioma withinthe radiation field. The authors emphasise the need to be vigilant forthis important but relatively rare complication,which has significant associated morbidityIt is debatable whether radiation-induced cavernous hae-mangiomas develop from previously overlooked precursorsor are, in fact, de novo malformations. The fact that theyoccur within the radiation field, as in our case, supportsthe theory that they arise de novo.2 Other supporting evi-dence lies in the fact that, in children less than 10 years ofage, the length of the latency period between radiationand cavernous haemangioma formation has been shown tobe dependent on the total radiation dose.13 In children,the higher the radiation dose, over 30 Gy, the shorter thelatency period; in adults, cavernous haemangiomas havebeen shown to develop only above a 30 Gy threshold.13 Itis unclear why cavernous malformations occur more fre-quently in children than adults after radiotherapy. Theimmature brain may be more sensitive to radiotherapy-induced changes than the mature brain.14ConclusionWe report an adult case of nasopharyngeal carcinomatreated with radical chemo-radiotherapy, with subsequentdevelopment of a histologically proved temporal cavernoushaemangioma within the radiation field. It is likely that,FIG. 1Axial, T2-weighted magnetic resonance imaging brain scan,demonstrating the lesion.J R ELLENBOGEN, S M JOSHI, N KITCHEN926with the increasing use of radiotherapy and the associatedincrease in life expectancy, radiation-induced secondarytumours will become more commonplace. It is vital thatclinicians remain vigilant for this important but relativelyrare complication, as early detection can enable timelyintervention and a reduction in potentially significantmorbidity.References1 Jeyakumar A, Brickman TM, Jeyakumar A, Doerr T.Review of nasopharyngeal carcinoma. Ear Nose ThroatJ 2006;85:168–742 King AD, Ahuja AT, Teo P, Tse GMK, Kew J. Radiationinduced sarcomas of the head and neck following radio-therapy for nasopharyngeal carcinoma. Clin Radiol 2000;55:684–93 Cheng KM, Chan CM, Fu YT, Ho YW, Tsang YW, LeeMK et al. Brain abscess formation in radiation necrosis ofthe temporal lobe following radiation therapy for naso-pharyngeal carcinoma. Acta Neurochir (Wien) 2000;142:435–414 Goh YH, Chong VF, Low WK. Temporal bone tumours inpatients irradiated for nasopharyngeal neoplasms. J Laryn-gol Otol 1999;113:222–85 Kong L, Lu JJ, Hu C, Guo X, Wu Y, Zhang Y. The risk ofsecondary primary tumors in patients with nasopharyngealcarcinoma after definitive radiotherapy. Cancer 2006;107:1287–936 Nimjee SM, Power CJ, Bulsara KR. Review of the litera-ture on de novo formation of cavernous malformations ofthe central nervous system after radiation therapy. Neuro-surg Focus 2006;21:1–67 Burn S, Gunny R, Phipps K, Gaze M, Haywood R. Inci-dence of cavernoma development in children after radio-therapy for brain tumors. J Neurosurg 2007;106:379–838 Baumgartner JE, Ater JL, Ha CS, Kuttesch JF, Leeds NE,Fuller GN et al. Pathologically proven cavernous angiomasof the brain following radiation therapy for pediatric braintumours. Pediatr Neurosurg 2003;39:201–79 Amirjamshidi A, Abbassioun K. Radiation-inducedtumors of the central nervous system occurring in child-hood and adolescence. Childs Nerv Syst 2000;16:390–710 Robinson JR, Awad IA, Little JR. Natural history of thecavernous angioma. J Neurosurg 1991;75:709–1411 Curling OD, Kelly DL, Elster AD, Craven TE. An analysisof the natural history of cavernous angiomas. J Neurosurg1991;75:702–812 Furuse M, Miyatake SL, Kuroiwa T. Cavernous malfor-mation after radiation therapy for astrocytoma in adultpatients: report of 2 cases. Acta Neurochir (Wien) 2005;147:1097–10113 Heckl S, Achoff A, Kunze S. Radiation-induced cavernoushemangiomas of the brain. Cancer 2002;94:3285–9114 Gaensler EH, Dillon WP, Edwards MS, Larson DA,Rosenau W, Wilson CB. Radiation-induced telangiectasiain the brain simulates cryptic vascular malformations atMR imaging. Radiology 1994;193:629–36Address for correspondence:Mr N Kitchen,Consultant Neurosurgeon,The National Hospital for Neurology & Neurosurgery,London, WC1N 3BG, UK.E-mail: neil.kitchen@uclh.nhs.ukMr N Kitchen takes responsibility for the integrity ofthe content of the paper.Competing interests: None declaredCLINICAL RECORD 927

Development of cavernous haemangioma following radical chemo-radiotherapy for nasopharyngeal carcinoma

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Development of cavernous haemangioma following radical chemo-radiotherapy for nasopharyngeal carcinoma

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