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Lingual cavernous hemangioma in a Nepalese boy—‘A Difficult Associate!!!’

Lingual cavernous hemangioma in a Nepalese boy—‘A Difficult Associate!!!’ Hemangiomas are benign tumors comprising of ectatic blood vessels. Although common in the head and neck region, those occurring within the oral cavity and tongue are exceedingly rare. We report a 5-year-old boy with a swelling over the anter- ior third of tongue post failed conservative therapy eventually treated with surgical excision and confirmed histologically as a cavernous hemangioma probably first of its kind reported from Nepal. lowing, speech, recurrent blistering with pain and bleeding INTRODUCTION with minor trauma. He was unable to contain it within his Based on Mulliken and Glovacki Classification (1982), vascular mouth necessitating to sleep open-mouthed. He had also lesions are categorized broadly into two types: hemangiomas developed multiple dental caries with it. and vascular malformations, former being the commonest He had been managed conservatively with beta-blockers lesions of infancy and childhood. Hemangiomas are classified and steroids elsewhere for years with no appreciable benefit. based upon histology into capillary and cavernous types and General examination was normal. Oral examination revealed a upon vessel involvement into high flow (arterial and arterio- well demarcated swelling involving the anterior third of tongue venous) and low flow types (capillary or venous) [1]. extending dorso-ventrally (occupying mostly the ventral sur- Most hemangiomas of head and neck appear few weeks face) and measuring 6×5×3cm , red in color with a purplish post natally, attain rapid growth, enter a phase of involution hue. It had a smooth surface, with fine granularity. It was par- and eventual spontaneous resolution by 5–8 years [2]. However, tially compressible and soft in consistency as shown in Fig. 1. in some they may persist and pose treatment challenges. We Also noted were multiple dental caries. Examination of the hereby present a boy who had a persistent and expanding lin- neck and rest of the systems was unremarkable. gual hemangioma presenting with macroglossia despite A working clinical diagnosis of macroglossia secondary to attempted conservative measures. lingual hemangioma was made and partial glossectomy using an inverted V incision was performed as shown in Fig. 2. CASE REPORT The limbs of V were approximated to reconstruct the A 5-year-old boy presented with an unsightly and protuberant residual tongue as shown in Fig. 3 swelling over the anterior third of tongue that had started as a At 10 day outpatient follow up, improved swallowing, small nodule soon after birth and gradually progressed in size speech clarity and good healing was observed as shown in after 9 months of age. It was associated with difficulty in swal- Fig. 4. Received: August 8, 2018. Accepted: October 19, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted re-use, distribution, and reproduction in any medium, provided the original work is properly cited. 1 Downloaded from https://academic.oup.com/jscr/article-abstract/2018/10/rjy283/5142659 by Ed 'DeepDyve' Gillespie user on 16 October 2019 2 A.L. Shrestha and S.B. Paudel Figure 5: Microscopic appearance of the lingual cavernous hemangioma stained with eosin and hematoxylin at four times magnification. Figure 1: Appearance of the lingual cavernous hemangioma on clinical examination. Figure 6: Microscopic appearance of the lingual cavernous hemangioma stained with eosin and hematoxylin at 10 times magnification. The histopathology report showed mucosa lined by strati- Figure 2: The dorsal and ventral appearance of the lingual hemangioma imme- fied squamous epithelium, with underlying stroma showing diately after excision. dilated vascular channels lined by endothelial cells and con- taining red blood cells in their lumen confirming the diagnosis of lingual cavernous hemangioma as shown in Figs 5 and 6. DISCUSSION Of the majority of hemangiomas that tend to occur in the region of head and neck, the ones that occur in the tongue deserve special consideration not merely due to the rarity of occurrence but also due to associated issues with swallowing, breathing, susceptibility to trivial trauma and cosmesis [3]. Like Figure 3: On table appearance of the specimen nearing excision and after reconstruction of the residual tongue. other hemangiomas most of these glossal lesions appear 2–4 weeks after birth, growing rapidly till 6–8 months age and then following a usual course of involution in 70% of patients to a state of near disappearance by 5–8 years [3]. Less commonly, these lesions may be associated with Sturge–Weber syndrome, Osler–Weber–Rendu syndrome and Blue rubber bleb nevus syn- drome [3]. The characteristic history and local examination findings aided by histopathological studies confirms the diag- nosis in most cases. The treatment modalities include watchful observation, intralesional and systemic steroids, beta blockers, embolization, sclerotherapy and surgical excision [3]. The choice of therapy depends upon factors like age, size, location, lesion hemodynamics and response to the modality used [2, 3]. Having treated earlier with systemic steroids and beta blockers with unsuccessful outcome, we opted to proceed with surgical Figure 4: Dorsal and ventral appearance of the reconstructed residual tongue at excision for our patient with a successful and satisfactory outpatient follow up after 10 days. outcome. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/10/rjy283/5142659 by Ed 'DeepDyve' Gillespie user on 16 October 2019 Lingual cavernous hemangioma in a Nepalese boy—‘A Difficult Associate!!!’ 3 ACKNOWLEDGEMENTS REFERENCES The authors would like to thank the staff of the Operation Room, the 1. Kripal K, Rajan S, Ropak B, Jayanti I. Cavernous hemangioma General wardand thePediatric IntensiveCareUnitofthe hospital of the tongue. Case Rep Dent 2013;2013:898692. for providing support and helping in management of the patient. 2. Pranitha V, Puppala N, Deshmukh SN, Jagadesh B, Anuradha S. Cavernous hemangioma of tongue: management of two cases. J Clin Diagn Res;2014 8(10):ZD15–17. CONFLICT OF INTEREST STATEMENT 3. Khanduri S, Agrawal D, Varshney G, Singh N. Haemangioma None declared. of tongue: a rare case report. JOOMR 2015;3:25–7. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Lingual cavernous hemangioma in a Nepalese boy—‘A Difficult Associate!!!’

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Oxford University Press
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Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018.
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2042-8812
DOI
10.1093/jscr/rjy283
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Abstract

Hemangiomas are benign tumors comprising of ectatic blood vessels. Although common in the head and neck region, those occurring within the oral cavity and tongue are exceedingly rare. We report a 5-year-old boy with a swelling over the anter- ior third of tongue post failed conservative therapy eventually treated with surgical excision and confirmed histologically as a cavernous hemangioma probably first of its kind reported from Nepal. lowing, speech, recurrent blistering with pain and bleeding INTRODUCTION with minor trauma. He was unable to contain it within his Based on Mulliken and Glovacki Classification (1982), vascular mouth necessitating to sleep open-mouthed. He had also lesions are categorized broadly into two types: hemangiomas developed multiple dental caries with it. and vascular malformations, former being the commonest He had been managed conservatively with beta-blockers lesions of infancy and childhood. Hemangiomas are classified and steroids elsewhere for years with no appreciable benefit. based upon histology into capillary and cavernous types and General examination was normal. Oral examination revealed a upon vessel involvement into high flow (arterial and arterio- well demarcated swelling involving the anterior third of tongue venous) and low flow types (capillary or venous) [1]. extending dorso-ventrally (occupying mostly the ventral sur- Most hemangiomas of head and neck appear few weeks face) and measuring 6×5×3cm , red in color with a purplish post natally, attain rapid growth, enter a phase of involution hue. It had a smooth surface, with fine granularity. It was par- and eventual spontaneous resolution by 5–8 years [2]. However, tially compressible and soft in consistency as shown in Fig. 1. in some they may persist and pose treatment challenges. We Also noted were multiple dental caries. Examination of the hereby present a boy who had a persistent and expanding lin- neck and rest of the systems was unremarkable. gual hemangioma presenting with macroglossia despite A working clinical diagnosis of macroglossia secondary to attempted conservative measures. lingual hemangioma was made and partial glossectomy using an inverted V incision was performed as shown in Fig. 2. CASE REPORT The limbs of V were approximated to reconstruct the A 5-year-old boy presented with an unsightly and protuberant residual tongue as shown in Fig. 3 swelling over the anterior third of tongue that had started as a At 10 day outpatient follow up, improved swallowing, small nodule soon after birth and gradually progressed in size speech clarity and good healing was observed as shown in after 9 months of age. It was associated with difficulty in swal- Fig. 4. Received: August 8, 2018. Accepted: October 19, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted re-use, distribution, and reproduction in any medium, provided the original work is properly cited. 1 Downloaded from https://academic.oup.com/jscr/article-abstract/2018/10/rjy283/5142659 by Ed 'DeepDyve' Gillespie user on 16 October 2019 2 A.L. Shrestha and S.B. Paudel Figure 5: Microscopic appearance of the lingual cavernous hemangioma stained with eosin and hematoxylin at four times magnification. Figure 1: Appearance of the lingual cavernous hemangioma on clinical examination. Figure 6: Microscopic appearance of the lingual cavernous hemangioma stained with eosin and hematoxylin at 10 times magnification. The histopathology report showed mucosa lined by strati- Figure 2: The dorsal and ventral appearance of the lingual hemangioma imme- fied squamous epithelium, with underlying stroma showing diately after excision. dilated vascular channels lined by endothelial cells and con- taining red blood cells in their lumen confirming the diagnosis of lingual cavernous hemangioma as shown in Figs 5 and 6. DISCUSSION Of the majority of hemangiomas that tend to occur in the region of head and neck, the ones that occur in the tongue deserve special consideration not merely due to the rarity of occurrence but also due to associated issues with swallowing, breathing, susceptibility to trivial trauma and cosmesis [3]. Like Figure 3: On table appearance of the specimen nearing excision and after reconstruction of the residual tongue. other hemangiomas most of these glossal lesions appear 2–4 weeks after birth, growing rapidly till 6–8 months age and then following a usual course of involution in 70% of patients to a state of near disappearance by 5–8 years [3]. Less commonly, these lesions may be associated with Sturge–Weber syndrome, Osler–Weber–Rendu syndrome and Blue rubber bleb nevus syn- drome [3]. The characteristic history and local examination findings aided by histopathological studies confirms the diag- nosis in most cases. The treatment modalities include watchful observation, intralesional and systemic steroids, beta blockers, embolization, sclerotherapy and surgical excision [3]. The choice of therapy depends upon factors like age, size, location, lesion hemodynamics and response to the modality used [2, 3]. Having treated earlier with systemic steroids and beta blockers with unsuccessful outcome, we opted to proceed with surgical Figure 4: Dorsal and ventral appearance of the reconstructed residual tongue at excision for our patient with a successful and satisfactory outpatient follow up after 10 days. outcome. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/10/rjy283/5142659 by Ed 'DeepDyve' Gillespie user on 16 October 2019 Lingual cavernous hemangioma in a Nepalese boy—‘A Difficult Associate!!!’ 3 ACKNOWLEDGEMENTS REFERENCES The authors would like to thank the staff of the Operation Room, the 1. Kripal K, Rajan S, Ropak B, Jayanti I. Cavernous hemangioma General wardand thePediatric IntensiveCareUnitofthe hospital of the tongue. Case Rep Dent 2013;2013:898692. for providing support and helping in management of the patient. 2. Pranitha V, Puppala N, Deshmukh SN, Jagadesh B, Anuradha S. Cavernous hemangioma of tongue: management of two cases. J Clin Diagn Res;2014 8(10):ZD15–17. CONFLICT OF INTEREST STATEMENT 3. Khanduri S, Agrawal D, Varshney G, Singh N. Haemangioma None declared. of tongue: a rare case report. JOOMR 2015;3:25–7.

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Journal of Surgical Case Reports – Oxford University Press

Published: Oct 1, 2018

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