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| ARTICLES |
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| Year : 1978 | Volume
: 26
| Issue : 4 | Page : 43-47 |
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A recurrent lacrimal gland tumour-clinically malignant (A case report)
RK Tiwari, H Ram
Deptt. of Ophthalmology, P.M.C. & Hospital, Patna (Bihar), India
Correspondence Address:
R K Tiwari
Department of Ophthalmology, Patna Medical College & Hospital, Patna (Bihar)
India
 Source of Support: None, Conflict of Interest: None  | Check |
PMID: 437865 
How to cite this article:
Tiwari R K, Ram H. A recurrent lacrimal gland tumour-clinically malignant (A case report). Indian J Ophthalmol 1978;26:43-7 |
A recurrent lacrimal gland tumour of mixed pleomorphic type on the right side with clinically malignant mainfestations in the form of indefinite orbital ache and vague headache in lower right frontal region, marked anaemia and rapid progress after an insidious start in a female of about twenty nine years of age is being reported with its management with a view to enrich the literature of this rare disease in the third decade.
The histopathological classification of such rare lacrimal gland tumours has been very contentious on account of diverse hypotheses of their origin[4],[13],[14] and incidence[9],[10],[12] varied clinical course[3],[6] and features, mysterious and unpredicted transformation of benign into malignant[2],[4],[11] confusing relation of recurrences[2] with malignant transformations and different methods of managements.
| Case report | |  |
C. D. a female aged 29 years of Bholana Chak, Islampur, Patna was admitted in the Department of ophthalmology on 26.11.77 with complaints of marked protrusion of right eye ball with congestion, swelling of conjunctiva and mucoid discharge since about two years with gross diminution of vision and pain in the upper part of orbit and lower frontal region, gradually increasing in severity, since about four months.
She was operated for a swelling in this region of right eye, which was gradually increasing in size after first detection at the age of 23 years, about five years back in the department of ophthalmology at Patna Medical College Hospital but no relevant record was available.
On examination-General health average, with marked anaemia; Other systems-Nothing abnormal was detected. Left eye was normal. Right eye [Figure - 1] showed rubbery hard oedematous swelling of upper lid and subcutaneous tissues in the lateral 2/3rd part of eye brow region which seemed appreciably displaced upwards as compared to the normal left side. Lower lid was almost completely hidden by the proptosed conjunctiva and globe. The proptosed tissues encroached upon the outer surface of lateral wall of nose from root to about 1 cm. above the alae nasi.
Proptosis was down and in with complete immobility in the upper and outer direction and almost complete in the upper medial, medial and lower directions. Posterior extent of tumour could not be assertained though its location between orbital wall and eye ball was appreciated. The tenderness of mild degree was demonstrable. Medial and lateral canthi were full. Upper fornix was full and shelving especially in the lateral 1/3rd. Conjunctiva markedly chemosed with dessication and nercrosis and appeared associated with mucopurulent discharge. Limbus was not visible. Cornea was hazy and lustreless all round with dessiated and eroded epithelium in the medial half that corresponded with pus in the anterior chamber. Iris was muddy with exudation at places with hazy pupillary margin and absent light reflex. Media was markedly hazy. Visionperception of hand movement at x meter from lateral side was present which was lost after 3 days of admission. Tension-on digital palpation, was found to be markedly raised and the proptosis was irreducible. On measurement, the proptosis was about 39mm. from the lateral orbital margin.
Blood examinations were normal except haemeglobulin which Has 50% of normal.
Plane X-ray showed enlargement of the orbital out line with raising of the level of the superior orbital rim as compared to that of the left eye and increase of soft tissue shadow in right orbit [Figure - 2].
Management-Patient was given penicillin and streptomycin by I.M. injection, multivitamins, liver extract and Vit B 12 Injection, Blood (750 C.C. packed Cells) in transfusion with 150 C.C. packed Cells intcrmittantly. Chemosed conjunctiva punctured with multiple stab incisions in the earlier stage to relieve chemosis but in about 2-3 days, the chemosis again came to the same original level.
Preoperative radiation of 4500r was given which after administration of about 2000r, caused a regression of the proptosis-Proptosis further regressed after two weeks of the radiation treatment with its measurement of about 30 mm. only remaining.
After 4 weeks from the last day of radiation when Hb% was found to be 70%, leteral orbitotomy operation was performed under general anaesthesia with incision of skin starting from a point just lateral to pulley of superior obique along the eye brow line going along the lateral orbital wall to the level of outer canthus and turning backwards horizontally with-a gently curve at the angle, The bony margin of upper and outer angle and lateral 1/3rd of the upper border was found very weak and eroded which was nibbled away and removed. Periosteum was incised from lateral wall, lower frontal region and in the zygomatic bone in the usual way after first cleaning the temporal fossa only retracting the temporal muscle and fascia.
A rectangular-piece of bone of the lateral orbital wall and zygomatic bone was removed after making bone cuts with a dental drill in the upper and lower part and joining the two along the line starting from the drill opening in the upper frontal part to a pointjust anterior to the inferior orbital fissure and emerging through the lower drill cut. Prior to the removal of this piece, drill holes were made-two on the zygomatic bone and two correspondingly opposite on the lateral orbital wall and one on the upper part of lateral orbital border and one on the correspondingly opposite point on the bone above. On the separation of the orbital periosteum from lateral wall, much of it in the lacrimal gland region was found adherent to the tumour mass. Adherent periosteum was not separated from the mass but the tumour mass-closely attached from the lacrimal gland region to the orbital apex along with its full medial extent was excised in one piece and kept in the Ringer's solution. Bony walls were normal. There was much less of bleeding. Remaining periosteum was sutured with 5/0 chromic catgut. Bone replaced and stitched with 4/0 chromic catgut through the drill holes Temporal muscle, fascia and peritosteum and subcutaneous tissues stitched with 6/0 chromic catgut in separate convenient layers with drain in the lower and outer part. Pressure bandage applied after cleaning and dressing of wound with antibiotic powder.
The lacrimal gland tumour kept in Ringer's solution was about 38 mm, x 30 mm, in anteroposterior and horizontal direction respectively and about 15-18 mm. in thickness in its largest zone in the anterior part. It was dirty grey [Figure - 3]. The tumour was firm to feel with surrounding periosteum adherent. Its extension in the posterior portion near orbital apex was narrow in all dimensions.
On section, the cut surface appeared white and partly honeycombed. Microscopic examination: Many sections showed 9/10th of the tissue to be necrotic and did not take stain which might be because of radiation effect. In one section, masses of epithelial cells lying in a mucinous matrix were found which did not exhibit malignant changes (slide photograph no. 4- x 300).
Histological diagnosis-Pleomorphic adenoma showing benign features and radiation burn of the lacrimal gland.
Patient was kept on higher antibiotics, Vit. C and B complex. There was fever of 100°C on the 1st post operative day. Drain was removed after 36 hours. Alternate skin stitches were removed on 5th day while outer stitches were removed on 7th day. Evisceration of the ocular contents was done on 7th day with frill excision of sclera. Conjunctival stitch after evisceration was removed on 5th day and conjunctiva healed fully by 7th day. A little collection of fluid was found below the skin incision just posterior to the lower part of lateral orbital wall outside the outer canthal plane it was drained from the posterior end of the healed skin incision and wound healed with few dressings. In the third week of operation patient was given a temporary artificial prosthesis and watched. In about 15 days time, the swelling of lid subsided to a great extent and went on subsiding till 7 weeks whereafter only lax tissues of upper lid remained slighly swollen and hanging down which were repaired.
| Discussion | | |
The present case with most of the clinical features of mixed lacrimal gland tumours of benign type eg. marked proptosis down in, restricted mobility up and out, transcient diplopia, fullness and evertion of upper formix by rubbery hard nodular mass extending up to about 6 mm. from limbus, swelling of lids and raising of eye brow upward in lateral part, history of recurrence with insidious[3] progress since more than 2 years, has been, because of its uncommon association with indefinite orbital ache and vague headache in lower and outer frontal region and developing anaemia in the patientominous signs potentially suggestive of malignancy[1],[3],[5] and because of the fact that malignancy usually supervenes in recurrences which are reported even after complete excision showing a potentially malignant implicationfrom the initial stage of management, planned to be managed with this very back ground and to avoid operative dissemination and excessive haemorrhage, preoperative radiation by Silva by two fields size 6 x 6 cm. right lateral orbital and of the total 4500 r in 4 weeks time was administered which was, as expected, followed by a regression of proptosis by about 8-9 mm. The presumed malignant character of the tumour and hazy media with more than half of anterior chamber filled with pus with dessicated and ulcerated cornea were the main reasons why the lateral orbitotomy for removal of the tuncour was not done at the first stage.
The successful and complete operative removal of tumour with relatively greater ease without much bleeding has been possible mostly due to the preoperative radiation. The presence of necrotic pieces of bone on the orbital margin in upper and outer angle without any evidence of any bony erosion elsewhere in the fossa for lacrimal gland weighs more in favour of its causation due to irradiation than due to the tumour mass itself.
As against the much advocated surgical method of removal of a large piece of bone including the lateral wall, lateral border, Zygoma and part of the upper orbital margin[8] the removed bone piece including only a part of the lateral wall including its lateral border with a piece of zygoma just adjacent to lowet part of lateral border by simple drill cuts (not by Gigli's Saw or Stilli's Saw) with replacement of this piece in original place after anchorage by 4/0 chromic catgut sutures passed through drill holes.
Evisceration of the globe which was done on the 7th post operative day, was avoided at the time of lateral orbitomy with a view to allow a blind eye to remain for cosmetic proposes.
The histological examination showed the tumour as one of mixed tumour of benign pleomorphic adenoma type with radiation necrosis.
As per relevant literature, though the pseudo-tumours[5] of lacrimal gland are relatively common the real tumours are relatively rare[4]. Out of the real tumours, the epithelial tumours are the most common types of neoplasms of the gland. The incidence of the tumours has been found to be from 20% & 25% to 5% and 7% of all space occupying lesions of orbits[1],[2],[4],[15]
| Summary and conclusion | | |
A case of recurrent pleomorphic adenoma of mixed benign type at the end of 3rd decade with clinically malignant characteristics has been reported with. its management by resuscitative therapy, preoperative radiation, complete surgical removal through lateral orbitotomy after cutting the bone of lateral orbital wall by dental drill artificial prosthesis and correction of post operative ptosis.
On a comprehensive analysis of the whole case, it is concluded that preoperative irradiation in such cases deserves consideration as sound and reasonable step for decreasing the size of tumour mass, lessening vascularity, obviating surgical dissemination and possibly impeding expected future recurrences.
| References | | |
| 1. | Arnold Sorsby 1964, Modern Ophthal., 4,794, 825, Butterworths, London.  |
| 2. | B.N. Consul, O.P. Kulshrestha & Subhash Gupta 1961, Jour. of All India Ophth. Soc., 12, 87. |
| 3. | Benedict & Border's 1930, Amer. J. Ophth., 13, 585. |
| 4. | Das, S.P., 1961, Jour. of All India Ophth. Soc., 30. |
| 5. | Duke-Elder S., 1974, System of Ophthalmology, 13, 643, Henry Kimpton, London. |
| 6. | Fleming, 1949, Brit. J. Ophth., 33, 763. |
| 7. | Godtfredsen, 1948, Brit. Journ. Ophth., 32, 171.
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| 8. | H.B. Stallard, 1973, Eye Surgery, 5, 862, K.M. Varghese Company, Bombay, India. |
| 9. | Hugh Mac Millan Moss, 1962, Amer. J. Ophth., 54,761. |
| 10. | Erving, A.R., Roberts, W.L. and Suda Koff, P.S., 1951, Amer. J. Ophth., 34, 1511. |
| 11. | Lane, 1922, Amer. J. Ophth., 5, 425. |
| 12. | Mckinnery, W.W. and Butz, W.C., 1951, Amer. J. Ophth., 34, 1519. |
| 13. | M.L. Agarwal & R.P. Dhanda. 1961, Jour, of All India Ophth. Soc. 12, 97. |
| 14. | R.G. Kulkarni, D.L., Maria & A.W. Bhawathakur, 1957 Ind. Jour. Ophth., 25, III, 42. |
| 15. | T.B. Johnston, D.V. Davies and F. Davies 1958, 32, Gray's Anatomy. 264. Longmans, Green And Co. London, New Yark. Toronto. |
[Figure - 1], [Figure - 2], [Figure - 3], [Figure - 4]
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