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Orbital Socket Pain After Injury
ORBITAL SOCKET PAIN AFTER INJURY FRANCIS C. SUTULA, M. D., AND JOHN J. WElTER, Boston, Massachusetts
Socket secretions, superior sulcus deformities, enophthalmos, unsatisfactory motility, lower eyelid laxity, and migration of the implant are the most common sequelae of enucleation.! Severe orbital socket pain unrelated to the prosthesis is unusual. 1,2 We studied a case of persistent superior orbital socket pain after enucleation. Histological examination demonstrated a neuroma with cartilaginous metaplasia. CASE REPORT A 55-year-old man had excruciating pain in his left orbit which had been enucleated four years previously. During the Korean War, the patient had suffered a shrapnel injury which caused corneal perforation and traumatic cataract. The left globe developed a total retinal detachment and neovascular glaucoma which could not be controlled medically. Twentyfour years after the original injury, the patient had been given a retrobulbar injection of 100% alcohol for intolerable, poorly localized orbital pain. After the injection, the patient noted that the quality and location of the pain had been modified. The vague, aching pain had become a sharp, stabbing sensation confined to the distributions of the supraorbital and supratrochlear nerves. Because of this continued discomfort, the left globe was enucleated and a spherical implant inserted. The pain above the patient's left eyebrow persisted. On four separate occasions over a four-year period, the ocular prostheses had been refitted in an attempt to make him more comfortable. Trial periods without any prosthesis in his socket had also failed to relieve his discomfort. When we examined him four years after the enucleation, we found a well-fitting prosthesis with good superior and inferior fornices and no migration of the implant. Palpation of the superior orbital rim
From the Oculoplastic Service, Department of Ophthalmology, Tufts University-New England Medical Center Hospital, Boston, Massachusetts, and the Boston Veterans Administration Hospital Medical Center, Jamaica Plain, Massachusetts. Reprint requests to F. Sutula, M.D., Department of Ophthalmology, Tufts University-New England Medical Center Hospital, Box 450, 171 Harrison Ave., Boston, MA 02111. 692
M. D.
medially, in the region of the supraorbital notch, produced intense pain which radiated superiorly above his eyebrow. Radiographs of the orbits, including tomography and axial and coronal computed tomography, disclosed no abnormalities. A- and Bscan ultrasonography also showed no abnormalities. The patient chose to undergo exploratory surgery in an attempt to define and alleviate his pain. We performed an anterosuperior orbitotomy. We incised the frontal periosteum both medially and laterally to the supratrochlear notch. The supratrochlear and supraorbital nerves were isolated and retracted with silk sutures. The angular, dorsal nasal, and supraorbital veins were also isolated and retracted. We then lifted the periosteum both laterally and medially to the trochlear notch, keeping the anatomical relationships intact. We freed the arcus marginalis from the superior orbital rim, permitting a smooth subperiorbital dissection. Palpation disclosed a firm nodular lesion involving the supratrochlear nerve 1 em posterior to the orbital rim and 1 cm lateral to the trochlea (Fig. 1). This was excised and kept for examination. We then performed a layered closure. The patient's pain ceased immediately after the operation. Hypesthesia continued in the distribution of the supratrochlear nerve for two months postoperatively. This gradually improved, and the patient has continued to do well and has resumed wearing his ocular prosthesis. Examination of the pathological specimen showed a traumatic neuroma (Fig. 2) with cartilaginous metaplasia (Figs. 3 and 4). DISCUSSION
Severe, persistent socket pain unrelated to the ocular prosthesis is rare after enucleation of the globe 1,2 and should alert the clinician to a possible progressive process. In our case, the prosthesis could not have caused the pain because its removal produced no significant reduction in the pain's intensity. An unusual feature of our patient's pain was that it was best defined after a retrobulbar injection of 100% alcohol. This injection apparently blocked the component of the pain caused by the primary glaucoma, and unmasked the pain secondary to the neuroma. The original shrapnel injury, the retrobulbar injection of alcohol, or the enucleation may have caused the
AMERICAN JOURNAL OF OPHTHALMOLOGY 90:692-696, 1980
VOL. 90, NO. 5
ORBITAL SOCKET PAIN
693
,
neuroma of supratrochlear frontal n.
formation of the neuroma and its associated cartilaginous metaplasia. Although the exact cause of the neuroma remains unclear, it seems to have been present before the retrobulbar injection of alcohol. Amputation neuroma is actually an abortive regeneration of a severed nerve rather than a true neoplasm. The connective tissue components-the endoneurium, perineurium, and Schwarm cellsproliferate. Axon cylinders elongate, branch, and ramify to follow these pathfinders from the proximal end towards the distal portion of the nerve.3-5 If enough soft tissue intervenes, however, the normal guidelines are obliterated and the axons become entangled in a densely fibrous matrix. In the present case, the cartilaginous metaplasia may have blocked the regenerating neurons. The proliferative response in the proximal end may persist for years, producing this clubbed overgrowth. The interval from severance of the nerve to the de-
n.
Fig. 1 (SutuIa and Weiter). Schematic view of left orbit shows the location of the neuroma.
velopment of the neuroma can range from 16 months to 50 years." Although amputation, or traumatic, neuroma is common in many parts of the body, reported cases in the orbit are rare. Henderson' did not come across such a tumor in his orbital survey. Duke-Elder and Macfault suggested that it rarely occurs in the orbit because the orbital nerves are small and there are usually no irritating factors. Blodi 6 reported an amputation neuroma in the orbit after enucleation for retinoblastoma in 1949. The neuroma was found in a specimen exenterated after recurrence of the retinoblastoma. The present case is unusual in that the neuroma formed in a nerve not known to have suffered trauma or amputation. Orbital cartilage is normally found at the trochlea and occasionally at the caruncle. 7,8 Orbital chondromas are extremely rare. Dahlin," in a review of 3,987 osseous and cartilaginous tumors, did not find any orbital chondromas. Henderson'?
694
AMERICAN JOURNAL OF OPHTHALMOLOGY
NOVEMBER, 1980
Fig. 2 (Sutula and Welter). Histologic section of neuroma. Top, Low-power view shows axon cylinders encased in connective tissue (hematoxylin and eosin, X 25). Bottom, Higher-power view of axon cylinders (hematoxylin and eosin, x 2(0).
Fig. 3 (Sutula and Weiter). Histologic section shows neuroma in approximation to cartilaginous metaplasia (hematoxylin and eosin, x 25).
Fig. 4 (Sutula and Weiter). Histologic section of cartilaginous metaplasia showing disorderly cellular grouping (hematoxylin and eosin, x 2(0).
695
696
AMERICAN JOURNAL OF OPHTHALMOLOGY
noted that these tumors are extremely rare in the orbit and that most cartilaginous growth occurring around the rim of the orbit is likely to be cartilaginous metaplasia incorporated into the fibromas. A neuroma contains fibrous connective tissue in addition to Schwann cells, perineurium, and endoneurium. We know that this tissue is capable of differentiating into many mesenchymal elements, including cartilage, under irritating influences. This case is unique for several reasons. Both neuroma and cartilaginous metaplasia are rare in the orbit; an orbital manifestation with both tissue types has not been reported to our knowledge. Additionally, the neuroma arose from a nerve that was not traumatized by enucleation. The supratrochlear nerve, not the nasociliary nerve, was involved. SUMMARY
Total retinal detachment and neovascular glaucoma occurred after a penetrating injury to the left globe that left the eye blind and painful. A retrobulbar injection of alcohol and enucleation modified but did not relieve the orbital discomfort. At surgery, we discovered a neuroma with cartilaginous metaplasia involving the
NOVEMBER, 1980
supratrochlear nerve. Excision of this mass produced relief of symptoms. This is, to our knowledge, the first reported case of a neuroma with cartilaginous metaplasia involving the supratrochlear nerve. REFERENCES 1. Soli, D. B.: The anophthalmic socket. In Soli, D. B. (ed.): Management of Complications in Ophthalmic Plastic Surgery. Birmingham, Aesculapius Publishing ce., 1976, pp. 315-343. 2. Callahan, A.: Reconstructive Surgery of the Eyelids and Ocular Adnexa. Birmingham, Aesculapius Publishing ce., 1966, pp. 305-325. 3. Hogan, M. J., and Zimmerman, L. E.: Ophthalmic Pathology. Philadelphia, W. B. Saunders Co., 1962, p. 753. 4. Duke-Elder, S., and MacFaul, P. A.: The Ocular Adnexa. Lacrimal, Orbital and Para-Orbital Diseases. In Duke-Elder, S. (ed.): System of Ophthalmology, vol. 13, pt. 2. St. Louis, C. V. Mosby, 1974, p. 1123. 5. Henderson, J. W.: Orbital Tumors. Philadelphia, W. B. Saunders Co., 1973, p. 316. 6. Blodi, F. C.: Amputation neuroma in the orbit. Am. J. Ophthalmol. 32:929, 1949. 7. Whitnall, S. E.: Anatomy of the Human Orbit and Accessory Organs of Vision. London, Oxford University Press, 1932, p. 277. 8. Duke-Elder, S.: Normal and Abnormal Development. Congenital Deformities. In System of Ophthalmology, vol. 3, pt. 2. St. Louis, C. V. Mosby, 1964, p. 862. 9. Dahlin, D. C.: Bone Tumors. Springfield, Charles C Thomas, 1967, p. 28. 10. Henderson, J. W.: Orbital Tumors, Philadelphia, W. B. Saunders Co., 1973, p. 240.
Socket secretions, superior sulcus deformities, enophthalmos, unsatisfactory motility, lower eyelid laxity, and migration of the implant are the most common sequelae of enucleation.! Severe orbital socket pain unrelated to the prosthesis is unusual. 1,2 We studied a case of persistent superior orbital socket pain after enucleation. Histological examination demonstrated a neuroma with cartilaginous metaplasia. CASE REPORT A 55-year-old man had excruciating pain in his left orbit which had been enucleated four years previously. During the Korean War, the patient had suffered a shrapnel injury which caused corneal perforation and traumatic cataract. The left globe developed a total retinal detachment and neovascular glaucoma which could not be controlled medically. Twentyfour years after the original injury, the patient had been given a retrobulbar injection of 100% alcohol for intolerable, poorly localized orbital pain. After the injection, the patient noted that the quality and location of the pain had been modified. The vague, aching pain had become a sharp, stabbing sensation confined to the distributions of the supraorbital and supratrochlear nerves. Because of this continued discomfort, the left globe was enucleated and a spherical implant inserted. The pain above the patient's left eyebrow persisted. On four separate occasions over a four-year period, the ocular prostheses had been refitted in an attempt to make him more comfortable. Trial periods without any prosthesis in his socket had also failed to relieve his discomfort. When we examined him four years after the enucleation, we found a well-fitting prosthesis with good superior and inferior fornices and no migration of the implant. Palpation of the superior orbital rim
From the Oculoplastic Service, Department of Ophthalmology, Tufts University-New England Medical Center Hospital, Boston, Massachusetts, and the Boston Veterans Administration Hospital Medical Center, Jamaica Plain, Massachusetts. Reprint requests to F. Sutula, M.D., Department of Ophthalmology, Tufts University-New England Medical Center Hospital, Box 450, 171 Harrison Ave., Boston, MA 02111. 692
M. D.
medially, in the region of the supraorbital notch, produced intense pain which radiated superiorly above his eyebrow. Radiographs of the orbits, including tomography and axial and coronal computed tomography, disclosed no abnormalities. A- and Bscan ultrasonography also showed no abnormalities. The patient chose to undergo exploratory surgery in an attempt to define and alleviate his pain. We performed an anterosuperior orbitotomy. We incised the frontal periosteum both medially and laterally to the supratrochlear notch. The supratrochlear and supraorbital nerves were isolated and retracted with silk sutures. The angular, dorsal nasal, and supraorbital veins were also isolated and retracted. We then lifted the periosteum both laterally and medially to the trochlear notch, keeping the anatomical relationships intact. We freed the arcus marginalis from the superior orbital rim, permitting a smooth subperiorbital dissection. Palpation disclosed a firm nodular lesion involving the supratrochlear nerve 1 em posterior to the orbital rim and 1 cm lateral to the trochlea (Fig. 1). This was excised and kept for examination. We then performed a layered closure. The patient's pain ceased immediately after the operation. Hypesthesia continued in the distribution of the supratrochlear nerve for two months postoperatively. This gradually improved, and the patient has continued to do well and has resumed wearing his ocular prosthesis. Examination of the pathological specimen showed a traumatic neuroma (Fig. 2) with cartilaginous metaplasia (Figs. 3 and 4). DISCUSSION
Severe, persistent socket pain unrelated to the ocular prosthesis is rare after enucleation of the globe 1,2 and should alert the clinician to a possible progressive process. In our case, the prosthesis could not have caused the pain because its removal produced no significant reduction in the pain's intensity. An unusual feature of our patient's pain was that it was best defined after a retrobulbar injection of 100% alcohol. This injection apparently blocked the component of the pain caused by the primary glaucoma, and unmasked the pain secondary to the neuroma. The original shrapnel injury, the retrobulbar injection of alcohol, or the enucleation may have caused the
AMERICAN JOURNAL OF OPHTHALMOLOGY 90:692-696, 1980
VOL. 90, NO. 5
ORBITAL SOCKET PAIN
693
,
neuroma of supratrochlear frontal n.
formation of the neuroma and its associated cartilaginous metaplasia. Although the exact cause of the neuroma remains unclear, it seems to have been present before the retrobulbar injection of alcohol. Amputation neuroma is actually an abortive regeneration of a severed nerve rather than a true neoplasm. The connective tissue components-the endoneurium, perineurium, and Schwarm cellsproliferate. Axon cylinders elongate, branch, and ramify to follow these pathfinders from the proximal end towards the distal portion of the nerve.3-5 If enough soft tissue intervenes, however, the normal guidelines are obliterated and the axons become entangled in a densely fibrous matrix. In the present case, the cartilaginous metaplasia may have blocked the regenerating neurons. The proliferative response in the proximal end may persist for years, producing this clubbed overgrowth. The interval from severance of the nerve to the de-
n.
Fig. 1 (SutuIa and Weiter). Schematic view of left orbit shows the location of the neuroma.
velopment of the neuroma can range from 16 months to 50 years." Although amputation, or traumatic, neuroma is common in many parts of the body, reported cases in the orbit are rare. Henderson' did not come across such a tumor in his orbital survey. Duke-Elder and Macfault suggested that it rarely occurs in the orbit because the orbital nerves are small and there are usually no irritating factors. Blodi 6 reported an amputation neuroma in the orbit after enucleation for retinoblastoma in 1949. The neuroma was found in a specimen exenterated after recurrence of the retinoblastoma. The present case is unusual in that the neuroma formed in a nerve not known to have suffered trauma or amputation. Orbital cartilage is normally found at the trochlea and occasionally at the caruncle. 7,8 Orbital chondromas are extremely rare. Dahlin," in a review of 3,987 osseous and cartilaginous tumors, did not find any orbital chondromas. Henderson'?
694
AMERICAN JOURNAL OF OPHTHALMOLOGY
NOVEMBER, 1980
Fig. 2 (Sutula and Welter). Histologic section of neuroma. Top, Low-power view shows axon cylinders encased in connective tissue (hematoxylin and eosin, X 25). Bottom, Higher-power view of axon cylinders (hematoxylin and eosin, x 2(0).
Fig. 3 (Sutula and Weiter). Histologic section shows neuroma in approximation to cartilaginous metaplasia (hematoxylin and eosin, x 25).
Fig. 4 (Sutula and Weiter). Histologic section of cartilaginous metaplasia showing disorderly cellular grouping (hematoxylin and eosin, x 2(0).
695
696
AMERICAN JOURNAL OF OPHTHALMOLOGY
noted that these tumors are extremely rare in the orbit and that most cartilaginous growth occurring around the rim of the orbit is likely to be cartilaginous metaplasia incorporated into the fibromas. A neuroma contains fibrous connective tissue in addition to Schwann cells, perineurium, and endoneurium. We know that this tissue is capable of differentiating into many mesenchymal elements, including cartilage, under irritating influences. This case is unique for several reasons. Both neuroma and cartilaginous metaplasia are rare in the orbit; an orbital manifestation with both tissue types has not been reported to our knowledge. Additionally, the neuroma arose from a nerve that was not traumatized by enucleation. The supratrochlear nerve, not the nasociliary nerve, was involved. SUMMARY
Total retinal detachment and neovascular glaucoma occurred after a penetrating injury to the left globe that left the eye blind and painful. A retrobulbar injection of alcohol and enucleation modified but did not relieve the orbital discomfort. At surgery, we discovered a neuroma with cartilaginous metaplasia involving the
NOVEMBER, 1980
supratrochlear nerve. Excision of this mass produced relief of symptoms. This is, to our knowledge, the first reported case of a neuroma with cartilaginous metaplasia involving the supratrochlear nerve. REFERENCES 1. Soli, D. B.: The anophthalmic socket. In Soli, D. B. (ed.): Management of Complications in Ophthalmic Plastic Surgery. Birmingham, Aesculapius Publishing ce., 1976, pp. 315-343. 2. Callahan, A.: Reconstructive Surgery of the Eyelids and Ocular Adnexa. Birmingham, Aesculapius Publishing ce., 1966, pp. 305-325. 3. Hogan, M. J., and Zimmerman, L. E.: Ophthalmic Pathology. Philadelphia, W. B. Saunders Co., 1962, p. 753. 4. Duke-Elder, S., and MacFaul, P. A.: The Ocular Adnexa. Lacrimal, Orbital and Para-Orbital Diseases. In Duke-Elder, S. (ed.): System of Ophthalmology, vol. 13, pt. 2. St. Louis, C. V. Mosby, 1974, p. 1123. 5. Henderson, J. W.: Orbital Tumors. Philadelphia, W. B. Saunders Co., 1973, p. 316. 6. Blodi, F. C.: Amputation neuroma in the orbit. Am. J. Ophthalmol. 32:929, 1949. 7. Whitnall, S. E.: Anatomy of the Human Orbit and Accessory Organs of Vision. London, Oxford University Press, 1932, p. 277. 8. Duke-Elder, S.: Normal and Abnormal Development. Congenital Deformities. In System of Ophthalmology, vol. 3, pt. 2. St. Louis, C. V. Mosby, 1964, p. 862. 9. Dahlin, D. C.: Bone Tumors. Springfield, Charles C Thomas, 1967, p. 28. 10. Henderson, J. W.: Orbital Tumors, Philadelphia, W. B. Saunders Co., 1973, p. 240.