Download TYPE OF ARTICLE : Case report

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TITLE FILE :
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TYPE OF ARTICLE : Case report
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TITLE OF THE ARTICLE: Intramuscular cavernous hemangioma of medial rectus
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muscle in paediatric age group
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AUTHORS:
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1. Dr. Anuj Mehta : Professor.
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2. Dr. Sangeeta Abrol : Professor
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3. Dr. Shalini Butola : Senior Resident
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4. Dr. Mayuresh Naik : Senior resident
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5. Dr. Anju Kumari : Senior Resident
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DEPARTMENT AND INSTITUTION : Department of Ophthalmology, V.M.M.C &
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Safdarjung Hospital, Ring Road, Ansari Nagar, NewDelhi- 110029
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CORRESPONDING AUTHOR:
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Name : Dr. Naik Mayuresh P.
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Address: Room No.430 of Eye OPD, 4th floor of OPD building, V.M.M.C &
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Safdarjung Hospital, Ansari Nagar, Ring road Newdelhi-110029.
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Phone numbers: +91-8287344576
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E-mail address: [email protected]
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KEY WORDS :
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Cavernous hemangioma ; Paediatric hemangioma ; Medial rectus ; Intramuscular
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hemangioma
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TOTAL NUMBER OF PAGES :
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WORD COUNTS :
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FOR ABSTRACT : 155
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FOR ARTICLE : 780
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SOURCES OF SUPPORT : NONE
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FUNDING : NONE
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PRESENTATION AT A MEETING : NONE
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CONFLICTING INTEREST : NONE
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ACKNOWLEDGEMENT : NONE
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CONTRIBUTORSHIP : All the uthors were involved in the concept and design of
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the study, data acquisition, data analysis and interpretation, draftinf manuscript,
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technical support and final review of the manuscript.
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ABSTRACT :
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An 11 year old male child presented with a mass on nasal aspect of right eye for last 2 years.
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Extraocular movements were decreased in right eye on levoversion , levoelevation and
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levodepression .
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Local examination revealed a bluish mass with irregular surface and ill-defined
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marginslocated in medial rectus muscle. The mass was 10 x 20mm in size , firm , nodular ,
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non-tender , non-pulsatile , non-compressible and non-reducible.
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MRI of the orbit revealed a well-defined mass of approximately 23 x 13 mm along the medial
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rectus(MR) muscle . It was hyperintense on T2W images with very minimal contrast
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enhancement.A provisional diagnosis of hemangioma or lymphangioma with intralesional
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haemorrhage was made.
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During surgical excision, the mass was found to be encapsulated by MR fibres. The MR
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fibres were separated , the mass measuring 20 x 8 x 6.5mm was removed and sent for
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histopathology. The histopathological examination revealed an intramuscular cavernous
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hemangioma.
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MANUSCRIPT:
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CASE REPORT :
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An 11 year old male child presented to our hospital with a mass on nasal aspect of
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right eye for last 2 years. It was associated with redness over the lesion which was
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relieved on topical medications. There was no associated pain , lacrimation and
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photophobia . It was associated with binocular diplopia on levoversion for last 4
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months . There was no history of sudden increase in size of mass , pain , forward
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protrusion of eyeball or diminution of vision .
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On systemic examination , no abnormality was detected . On ocular examination , the
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best corrected visual acuity was 6/6 in both eyes . His anterior segment and fundus
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examination was normal. Extraocular movements were decreased in right eye on
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levoversion
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base 100mm was 15mm in both eyes.
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Local examination revealed a bluish mass with irregular surface and ill-defined
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margins located in medial rectus muscle. The mass became more prominent on
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dextroversion. The mass was 10 x 20mm in size , firm
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non-pulsatile , non-compressible and non-reducible.(Fig.1)
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USG B-Scan revealed a heterogenous mass approximately 23 x 13mm in extraconal
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compartment close to insertion of medial rectus(MR) muscle with few hyperechoic
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flecks. (Fig. 2) MRI of the orbit revealed a well-defined mass of approximately 23 x
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13 mm along the medial rectus(MR) muscle . It was isointense on T1W and
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hyperintense on T2W images with very minimal contrast enhancement (Fig. 3). Patient
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initially consulted a local ophthalmic surgeon elsewhere, where it was initially
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diagnosed as cysticercosis and treated with albendazole and oral steroids.
, levoelevation and levodepression . Exophthalmometry with Hertel’s for
, nodular , non-tender ,
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A
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haemorrhage was made. As patient had already received a course of oral steroids
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without any response and there was no evidence of active flow in the lesion, a
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decision to do excision biopsy was taken.
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During surgical excision, the mass was found to be encapsulated by MR fibres. The
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MR fibres were separated and the mass was removed completely. The MR fibres
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were sutured together with 6-0 vicryl suture. the mass measuring 20 x 8 x 6.5mm
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was removed and sent for histopathology. The histopathological examination revealed
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an intramuscular cavernous hemangioma.(Fig. 4.) The patient was started on oral
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provisional
diagnosis
of
hemangioma
or
lymphangioma
with
intralesional
steroids for two weeks to ameliorate the post-operative edema and fibrosis (Fig. 5).
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DISCUSSION :
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Intramuscular
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congenital neoplasms which remain unremarkable for long but may start growing in
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second and third decade of life(1,2). Allen and Enzinger classified them into three
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types based on histopathological features into capillary (50%) , cavernous (29%) and
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mixed (21%) . IMHs are rare tumors(3). Less than 1% of hemangiomas occur in
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skeletal muscles and more than 20% occur in head and neck region. The IMHs in
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extraocular muscles are very rare(1,2,3). Only six such cases are recorded in literature.
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(Table 1).
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Christensen et al reported a case of 21 year old female with 10 year history of
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proptosis. She was operated thrice in 9 yrs without reaching a diagnosis. Enucleation
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of the eye ball with excision of affected muscle was done which revealed mixed
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IMH involving four of the six muscles(4).
hemangiomas(IMH)
are
non-metastasizing,
benign,
hamartomatous,
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Kiratli et al reported two cases of isolated IMH of extraocular muscles ; one in a
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3yr old child with involvement of lateral rectus muscle and the other in 40 yr old
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man with involvement of medial rectus muscle. Both of them presented with proptosis
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and lid swelling. On histopathological examination, the child was diagnosed as having
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capillary type IMH and the adult man was diagnosed as having mixed type IMH(5).
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In our case, the medial rectus was involved. The mass could be excised completely
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and on histopatholgical examination, a purely cavernous type of IMH was noted. In
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six cases reported till date, 2 cases had mixed type, 2 had capillary and 2 had
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cavernous type(in adult 63 yr old patient and 31 yr old pregnant female) on
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histopathological examination(4,5,6,7). Our case was unique because it is the first case
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of a purely cavernous hemangioma in a child.
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The armamentarium of management options narrows down to just two ; medically by
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systemic steroids and surgically by complete excision. Systemic steroids can reduce
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the bulk of the tumor but IMHs of extraocular muscles are insensitive to systemic
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corticosteroids because of encapsulation and presence of cavernous elements which are
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resistant to corticosteroids
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surrounding healthy tissue, is the treatment of choice for management of such lesions,
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although recurrences ranging from 9-28% have been described even after wide
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resection of a
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was noticed even after 3 years of regular follow-up.
(3)
. Open surgical excision, including an adequate rim of
cuff of normal muscle around the tumor(9). In our case, no recurrence
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FIGURE LEGEND :
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Fig. 1 : Pre-operative status showing prominence of the hemangioma on dextroversion
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Fig. 2 : USG B-scan of orbit swing hheterogenous mass in extraconal compartment
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close to insertion of medial rectus
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Fig. 3 : MRI scan of orbit MRI of the orbit revealed a well-defined mass of
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approximately 23 x 13 mm along the medial rectus muscle
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Fig. 4 : Histopathological section showing cavernous hemangioma
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Fig. 5 : Post-operative status after excision of hemangioma
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TABLE LEGEND :
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Table 1 : Review of cases of ‘Intramuscular hemangiomas’ in extraocular muscles
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REFERENCES :
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1. Lee H.C., Lee S.J., Kim Y.D. Intramuscular hemangioma of upper lid. J. Korean
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Ophthalmol. Soc. 2003;44:2428-2433.
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2. Rossiter J.L., Hendrix R.A., Tom L.W.C., Potsic W.P.S. Intramuscular hemangioma
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of head and neck. Laryngoscope 1993;108:18-26.
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3. Allen P.W., Enzinger F.M.. Hemangioma of skeletal muscle; an analysis of 89
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cases. Cancer 1972;8-22.
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4. ChristensenS.R., Borgesen S.E., Heegard S., Prause J.U. Orbital intramuscular
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hemangioma. Acta Ophthalmol Scand. 2002;80:336-339.
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5. Kiratli H., Bilgic S., Calgar M., Soylemezoglu. F. Intramuscular hemangioma of
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extraocular muscles. Ophthalmology 2003;110:564-580.
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6. Kim S.H., Hyung S.H., Rho B.K., Lee S.E., BAek S.H. a case of intramuscular
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hemangioma
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Soc;2006:196-198.
presenting
with
large
angle
hypertropia.
J
Korean
Ophthalmol.
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7. Charles N.C., Belliappa S., Patel P. Intramuscular Hemangioma of the Inferior
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ObliqueA Rare Cause of Extraocular Muscle Enlargement. JAMA Ophthalmol.
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2014;132(1):122-124.
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8. Giudice M., Piazza C., Bolzoni A., Peretti G. Head and neck intramuscular
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hemangioma
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Otorhinolaryngol. 2003;260:498-501.
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9. Lee BH et al. Orbital intramuscular hemangioma enlarging during pregnancy.
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Ophthal Plast Reconstr Surg 2009;25:491-3.
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:
report
of
two
cases
with
unusual
localisation.
Eur.
Arch.