- Email: [email protected]
Bilateral orbital hemorrhage in a newborn
than in dizygotic twins. They also identified that the concordance in location of hemangioma in monozygotic twins was no different than that in dizygotic twins, which suggests that cutaneous hemangioma in twins and in general is not regulated by genetic factors. They did not find a relationship of prematurity to the development of hemangioma. Combining the information on the twins study8 with the information on the familial form of hemangioma,5 it appears that heritable infantile hemangioma is exceedingly rare. However, data may be insufficient to make this conclusion, because most parents are unaware of their medical history at their own birth, and their parents are usually not present to provide information. Additionally, most physicians presume that cutaneous hemangioma is sporadic, so a family history of the disease is not questioned. In summary, multiple factors may be related to the development of cutaneous hemangioma. We found this tumor in premature triplets whose parents had a history of a similar tumor. This suggests that hereditary and environmental factors (prematurity) may have played a role. We recommend that a family history be obtained in all cases of cutaneous capillary hemangioma of infancy. REFERENCES
1. Drolet BA, Esterly NB, Frieden IJ. Hemangiomas in children. N Engl J Med 1999;341:173–181. 2. Shields JA, Shields CL. Atlas of eyelid and conjunctival tumors. Philadelphia: Lippincott Williams & Wilkins, 1999: 106 –111. 3. Burton BK, Schulz CJ, Angle B, Burd LI. An increased incidence of haemangiomas in infants born following chorionic villus sampling (CVS). Prenatol Diagn 1995;15:209 –215. 4. Amir J, Metzker A, Krikler R, Reisner SH. Strawberry hemangioma in preterm infants. Pediatr Dermatol 1986;3:331–332. 5. Blei F, Walter J, Orlow SJ, Marchuk DA. Familial segregation of hemangiomas and vascular malformations as an autosomal dominant trait. Arch Dermatol 1998;134:718 –722. 6. Physicians Desk Reference. Montvale, New Jersey: Medical Economics Company, 1998:1214. 7. Walter JW, Blei F, Anderson JL, Orlow SJ, Speer MC, Marchuk DA. Genetic mapping of a novel familial form of infantile hemangioma. Am J Med Genet 1999;82:77– 83. 8. Cheung DS, Warman ML, Mulliken JB. Hemangioma in twins. Ann Plast Surg 1997;38:269 –274.
Bilateral Orbital Hemorrhage in a Newborn Essam M. Ezzadin, MD, Don Liu, MD, Waleed Al-Rashed, MD, and Claude Jacquemin, MD PURPOSE: To report a case of severe bilateral proptosis resulting from orbital hemorrhage in a newborn and to discuss the differential diagnoses and management.
VOL. 129, NO. 4
FIGURE 1. (Top) A 13-day-old male infant with bilateral proptosis, which is more prominent in the left eye. (Bottom) Marked improvement 14 days later.
Case report of a 13-day-old male infant with bilateral proptosis since birth. The proptosis was monitored with clinical examinations and computed tomography as well as magnetic resonance imaging (MRI) scans, and it was managed with antibiotic ointment and patching. The MRI scans demonstrated bilateral subperiosteal orbital hemorrhage. RESULTS: Proptosis decreased, and there was successful, complete recovery without untoward sequelae in 14 days; follow-up indicated no late complications at age 1 year. CONCLUSION: Spontaneous orbital hemorrhage, unilateral or bilateral, is uncommon in an otherwise healthy newborn without apparent history of birth trauma. Magnetic resonance imaging scans are helpful in making the diagnosis of subperiosteal hemorrhage, and conservative management is advised. (Am J Ophthalmol 2000;129: 531–533. © 2000 by Elsevier Science Inc. All rights reserved.) METHOD:
Accepted for publication Nov 18, 1999. From the Oculoplastic and Orbit Division (E.M.E., D.L., W.A-R.) and the Diagnostic Imaging Department (C.J.), King Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia. Inquiries to Essam M. Ezzadin, MD, King Khaled Eye Specialist Hospital, PO Box 7191, Riyadh, 11462 Saudi Arabia; fax: 966-1-4829311; e-mail: [email protected]
BRIEF REPORTS
531
FIGURE 2. Magnetic resonance imaging scans. (Left) T1-weighted spin echo sequence shows a bilateral orbital roof hypersignal lesion in subperiosteal location (top arrow). The posterior pole of the eyeball is evident (bottom arrow). Note the hyposignal appearance of the posterior vitreous gel, which contrasts with the bright signal of the orbital fat, a characteristic signal on T1-weighted sequences. (Right) A T2-weighted sequence shows the subperiosteal lesion. The hypersignal appearance of the lesion (on both T1 and T2) is characteristic of hemorrhages in the late subacute phase, where methemoglobin is in extracellular location.
A
(Figure 1, bottom). The last follow-up examination, at 1 year of age, demonstrated no recurrence or complications. Unilateral and bilateral proptosis has been reported in newborns.1– 4 In one reported case, needle biopsy of the orbit and orbital exenteration for possible tumor were considered; the patient had deep x-ray treatment; fortunately, spontaneous improvement occurred, and 20 days later, the author reported that the eye appeared normal.1 A second infant was managed as an outpatient with resolution of bilateral proptosis after 32 days.2 Another infant, managed by Kundert, had bilateral proptosis that resolved by 30 days.3 Kundert stated that the absolute diagnosis of that case was difficult without exploration or histopathological studies.3 Recently, Munoz and Weatherhead4 reported a 4-day-old neonate with right proptosis and unilateral orbital hemorrhage, confirmed by surgical exploration. Munoz and Weatherhead tried to exclude the possibility of a neoplasm by exploring the orbit and found an old blood clot; no details are given regarding the timing of this intervention and other clinical and laboratory evidence that necessitated such a decision. An orbital hematoma should be suspected in a newborn with proptosis, despite the absence of a clotting or vascular disorder and despite a history of uncomplicated delivery. Spontaneous orbital hemorrhage is uncommon in an otherwise healthy newborn without apparent history of birth trauma. Diagnosis is greatly helped by MRI scans, which can characterize all stages of blood degradation, thus avoiding unnecessary surgical exploration.5 Oxidative denaturation of oxyhemoglobin gives rise to degradation products, mainly methemoglobin and hemosiderin, and both display specific MRI behavior. The high signal on
13-DAY-OLD MALE INFANT HAD BILATERAL PROPTOSIS
since birth, more severe on the left side, which resulted in a corneal epithelial defect (Figure 1, top). He was the product of normal pregnancy and delivery, with no history of trauma to the head. There was no family history of systemic disease. Pupils were reactive without relative afferent defect. Tonometry readings were RE: 15 mm Hg and LE: 30 mm Hg. No hyperthermia, tenderness, or specific lesion was detected on orbital palpation, but resistance to retropulsion was appreciated. Results of complete blood count with differentials, sickling test, glucose-6-phosphate dehydrogenase test, prothrombin time, activated partial thromboplastin time, and hemoglobin electrophoresis were normal. Initially, orbital computed tomography (CT) scans were performed, which showed bilateral high-density heterogenic extraconal masses suggestive of hemorrhage but did not rule out other causes. Differential diagnoses included teratoma, craniosynostosis, neuroblastoma, leukemic infiltrate, pseudotumor, metastatic tumors, Wilms tumor, and spontaneous orbital hemorrhage. Also, infant abuse should be kept in mind as one of the differentials. Antibiotic ointment and patching were applied to the left eye. Intraocular pressure was monitored, and the pupils and proptosis were observed carefully. Orbital exploration and needle biospy was contemplated, but only if proptosis increased or afferent pupillary defect occurred. The patient was admitted to the hospital, and improvement was evident by the fourth postadmission day. Magnetic resonance imaging (MRI) scans confirmed bilateral subperiosteal hemorrhage (Figure 2). The patient was discharged on the 14th day with normal eye findings 532
AMERICAN JOURNAL
OF
OPHTHALMOLOGY
APRIL 2000
both T1 and T2 spin echo sequences is characteristic (as in our patient) of methemoglobin in extracellular location. The heme iron is in the ferric state (Fe⫹⫹⫹), causing shortening of T1, and surrounded by water molecules, causing elongation of T2. This MRI behavior of hematomas can be present from a few days until several weeks after the sudden bleeding phenomenon, a condition often encountered in clinical practice. This type of signal is pathognomonic of organized hematomas in the late subacute phase. We managed bilateral proptosis with subperiosteal orbital hemorrhage conservatively, based on a lack of eyelid ecchymosis, palpable lesion, and laboratory findings. The corneal exposure was treated while anticipating other options. One can argue that blood aspiration might accelerate resolution; however, with an uncertain diagnosis, it is considered unwise to insert a needle in an orbit. Magnetic resonance imaging scans are helpful in making the diagnosis of subperiosteal hemorrhage, and conservative management is advised.
A
63-YEAR-OLD MAN WHO UNDERWENT A HEART TRANS-
BRIEF REPORTS
533
REFERENCES
1. Windham RE. A case of exophthalmos in a newborn infant. Am J Ophthalmol 1942;25:1236. 2. Harley RD. Exophthalmos in the newborn. Am J Ophthalmol 1943;26:1314 –1315. 3. Kundert KF. Bilateral exophthalmos in the newborn. Am J Ophthalmol 1951;34:1173. 4. Munoz M, Weatherhead R. Orbital hemorrhage in a newborn. Am J Ophthalmol 1989;108:206 –208. 5. Polito E, Leccisotti A. Diagnosis and treatment of orbital hemorrhagic lesions. Ann Ophthalmol 1994;26:85–93.
Hyphema Caused by a Metallic Intraocular Foreign Body During Magnetic Resonance Imaging Christopher N. Ta, MD, and R. Wayne Bowman, MD PURPOSE: To report a 63-year-old man with a retained intraocular foreign body who developed a hyphema during magnetic resonance imaging (MRI) of the brain. METHODS: Case report and review of the current literature on ocular injury caused by intraocular foreign bodies when subjected to an electromagnetic field. RESULTS: Our patient underwent a brain MRI, and the intraocular foreign body caused a hyphema and increased Accepted for publication Nov 23, 1999. From the Department of Ophthalmology, Stanford University Medical Center, Stanford, California (C.N.T.), and the Department of Ophthalmology,, Southwestern Medical School, University of Texas, Dallas, Texas (R.W.B.). Inquiries to Christopher N. Ta, MD, Department of Ophthalmology, Stanford University Medical Center, Boswell Bldg, Rm A-157, 300 Pasteur Dr, Stanford, CA 94305-5308; fax: (650) 723-7918.
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intraocular pressure. The presence and location of the intraocular foreign body were determined by computed tomography (CT). CONCLUSION: Magnetic resonance imaging can cause serious ocular injury in patients with ferromagnetic intraocular foreign bodies. This case demonstrates the importance of obtaining an occupational history, and, when indicated, a skull x-ray or CT to rule out intraocular foreign body before an MRI study. (Am J Ophthalmol 2000;129:533–534. © 2000 by Elsevier Science Inc. All rights reserved.)
plant 11 months prior to admission presented with a 5-day history of fever, headaches, and change in mental status. A brain magnetic resonance imaging (MRI) study was obtained. Immediately after the MRI, the patient complained of sudden pain and loss of vision in the left eye that occurred while he was in the MRI scanner. The patient was a metal worker but denied any intraocular injury. On examination at the bedside after the MRI, his visual acuity with a near card (with correction) was RE: 20/30 and LE: counting fingers at 3 inches. The intraocular pressure was RE: 12 mm Hg and LE: 30 mm Hg. His right pupil was reactive. His left pupil was minimally reactive at 6 mm. No afferent pupillary defect was detected. Examination of the right eye was normal except for a moderate cataract. The left eye had a 2-mm paracentral corneal scar and a 50% hyphema. The lens had moderate nuclear sclerosis. Fundus examination revealed an attached retina with no vitreous hemorrhage. A metallic intraocular foreign body was suspected. An orbital computed tomography (CT) scan revealed a radiodense intraocular foreign body in the inferior anterior chamber in the left eye (Figure 1). The right orbit had a foreign body just outside the globe in the inferior temporal quadrant (Figure 2). Treatment included bedrest, elevation of the head of bed 30 degrees, scopolamine, prednisolone acetate, timolol, and brimonidine. By hospital day 5, the left eye had an intraocular pressure of 14 with a 1-mm blood clot and retinal flame hemorrhages. There were also scattered subconjunctival hemorrhages in both eyes. Unfortunately, our patient developed multiorgan failure and died. The cause of death is unknown, but sepsis or thrombocytopenic thrombotic purpura was suspected. Several studies show that ferromagnetic bodies have significant movements when subjected to MRI, with the potential to cause intraocular injury. Lagouros and associates showed that all ferromagnetic materials (such as a BB pellet, a staple, or a screw) move during an MRI study in vitro when suspended in gelatin.1 Three of four ferromagnetic bodies implanted in the vitreous of rabbits showed movement that caused retinal holes, detachment, and
1. Drolet BA, Esterly NB, Frieden IJ. Hemangiomas in children. N Engl J Med 1999;341:173–181. 2. Shields JA, Shields CL. Atlas of eyelid and conjunctival tumors. Philadelphia: Lippincott Williams & Wilkins, 1999: 106 –111. 3. Burton BK, Schulz CJ, Angle B, Burd LI. An increased incidence of haemangiomas in infants born following chorionic villus sampling (CVS). Prenatol Diagn 1995;15:209 –215. 4. Amir J, Metzker A, Krikler R, Reisner SH. Strawberry hemangioma in preterm infants. Pediatr Dermatol 1986;3:331–332. 5. Blei F, Walter J, Orlow SJ, Marchuk DA. Familial segregation of hemangiomas and vascular malformations as an autosomal dominant trait. Arch Dermatol 1998;134:718 –722. 6. Physicians Desk Reference. Montvale, New Jersey: Medical Economics Company, 1998:1214. 7. Walter JW, Blei F, Anderson JL, Orlow SJ, Speer MC, Marchuk DA. Genetic mapping of a novel familial form of infantile hemangioma. Am J Med Genet 1999;82:77– 83. 8. Cheung DS, Warman ML, Mulliken JB. Hemangioma in twins. Ann Plast Surg 1997;38:269 –274.
Bilateral Orbital Hemorrhage in a Newborn Essam M. Ezzadin, MD, Don Liu, MD, Waleed Al-Rashed, MD, and Claude Jacquemin, MD PURPOSE: To report a case of severe bilateral proptosis resulting from orbital hemorrhage in a newborn and to discuss the differential diagnoses and management.
VOL. 129, NO. 4
FIGURE 1. (Top) A 13-day-old male infant with bilateral proptosis, which is more prominent in the left eye. (Bottom) Marked improvement 14 days later.
Case report of a 13-day-old male infant with bilateral proptosis since birth. The proptosis was monitored with clinical examinations and computed tomography as well as magnetic resonance imaging (MRI) scans, and it was managed with antibiotic ointment and patching. The MRI scans demonstrated bilateral subperiosteal orbital hemorrhage. RESULTS: Proptosis decreased, and there was successful, complete recovery without untoward sequelae in 14 days; follow-up indicated no late complications at age 1 year. CONCLUSION: Spontaneous orbital hemorrhage, unilateral or bilateral, is uncommon in an otherwise healthy newborn without apparent history of birth trauma. Magnetic resonance imaging scans are helpful in making the diagnosis of subperiosteal hemorrhage, and conservative management is advised. (Am J Ophthalmol 2000;129: 531–533. © 2000 by Elsevier Science Inc. All rights reserved.) METHOD:
Accepted for publication Nov 18, 1999. From the Oculoplastic and Orbit Division (E.M.E., D.L., W.A-R.) and the Diagnostic Imaging Department (C.J.), King Khaled Eye Specialist Hospital, Riyadh, Saudi Arabia. Inquiries to Essam M. Ezzadin, MD, King Khaled Eye Specialist Hospital, PO Box 7191, Riyadh, 11462 Saudi Arabia; fax: 966-1-4829311; e-mail: [email protected]
BRIEF REPORTS
531
FIGURE 2. Magnetic resonance imaging scans. (Left) T1-weighted spin echo sequence shows a bilateral orbital roof hypersignal lesion in subperiosteal location (top arrow). The posterior pole of the eyeball is evident (bottom arrow). Note the hyposignal appearance of the posterior vitreous gel, which contrasts with the bright signal of the orbital fat, a characteristic signal on T1-weighted sequences. (Right) A T2-weighted sequence shows the subperiosteal lesion. The hypersignal appearance of the lesion (on both T1 and T2) is characteristic of hemorrhages in the late subacute phase, where methemoglobin is in extracellular location.
A
(Figure 1, bottom). The last follow-up examination, at 1 year of age, demonstrated no recurrence or complications. Unilateral and bilateral proptosis has been reported in newborns.1– 4 In one reported case, needle biopsy of the orbit and orbital exenteration for possible tumor were considered; the patient had deep x-ray treatment; fortunately, spontaneous improvement occurred, and 20 days later, the author reported that the eye appeared normal.1 A second infant was managed as an outpatient with resolution of bilateral proptosis after 32 days.2 Another infant, managed by Kundert, had bilateral proptosis that resolved by 30 days.3 Kundert stated that the absolute diagnosis of that case was difficult without exploration or histopathological studies.3 Recently, Munoz and Weatherhead4 reported a 4-day-old neonate with right proptosis and unilateral orbital hemorrhage, confirmed by surgical exploration. Munoz and Weatherhead tried to exclude the possibility of a neoplasm by exploring the orbit and found an old blood clot; no details are given regarding the timing of this intervention and other clinical and laboratory evidence that necessitated such a decision. An orbital hematoma should be suspected in a newborn with proptosis, despite the absence of a clotting or vascular disorder and despite a history of uncomplicated delivery. Spontaneous orbital hemorrhage is uncommon in an otherwise healthy newborn without apparent history of birth trauma. Diagnosis is greatly helped by MRI scans, which can characterize all stages of blood degradation, thus avoiding unnecessary surgical exploration.5 Oxidative denaturation of oxyhemoglobin gives rise to degradation products, mainly methemoglobin and hemosiderin, and both display specific MRI behavior. The high signal on
13-DAY-OLD MALE INFANT HAD BILATERAL PROPTOSIS
since birth, more severe on the left side, which resulted in a corneal epithelial defect (Figure 1, top). He was the product of normal pregnancy and delivery, with no history of trauma to the head. There was no family history of systemic disease. Pupils were reactive without relative afferent defect. Tonometry readings were RE: 15 mm Hg and LE: 30 mm Hg. No hyperthermia, tenderness, or specific lesion was detected on orbital palpation, but resistance to retropulsion was appreciated. Results of complete blood count with differentials, sickling test, glucose-6-phosphate dehydrogenase test, prothrombin time, activated partial thromboplastin time, and hemoglobin electrophoresis were normal. Initially, orbital computed tomography (CT) scans were performed, which showed bilateral high-density heterogenic extraconal masses suggestive of hemorrhage but did not rule out other causes. Differential diagnoses included teratoma, craniosynostosis, neuroblastoma, leukemic infiltrate, pseudotumor, metastatic tumors, Wilms tumor, and spontaneous orbital hemorrhage. Also, infant abuse should be kept in mind as one of the differentials. Antibiotic ointment and patching were applied to the left eye. Intraocular pressure was monitored, and the pupils and proptosis were observed carefully. Orbital exploration and needle biospy was contemplated, but only if proptosis increased or afferent pupillary defect occurred. The patient was admitted to the hospital, and improvement was evident by the fourth postadmission day. Magnetic resonance imaging (MRI) scans confirmed bilateral subperiosteal hemorrhage (Figure 2). The patient was discharged on the 14th day with normal eye findings 532
AMERICAN JOURNAL
OF
OPHTHALMOLOGY
APRIL 2000
both T1 and T2 spin echo sequences is characteristic (as in our patient) of methemoglobin in extracellular location. The heme iron is in the ferric state (Fe⫹⫹⫹), causing shortening of T1, and surrounded by water molecules, causing elongation of T2. This MRI behavior of hematomas can be present from a few days until several weeks after the sudden bleeding phenomenon, a condition often encountered in clinical practice. This type of signal is pathognomonic of organized hematomas in the late subacute phase. We managed bilateral proptosis with subperiosteal orbital hemorrhage conservatively, based on a lack of eyelid ecchymosis, palpable lesion, and laboratory findings. The corneal exposure was treated while anticipating other options. One can argue that blood aspiration might accelerate resolution; however, with an uncertain diagnosis, it is considered unwise to insert a needle in an orbit. Magnetic resonance imaging scans are helpful in making the diagnosis of subperiosteal hemorrhage, and conservative management is advised.
A
63-YEAR-OLD MAN WHO UNDERWENT A HEART TRANS-
BRIEF REPORTS
533
REFERENCES
1. Windham RE. A case of exophthalmos in a newborn infant. Am J Ophthalmol 1942;25:1236. 2. Harley RD. Exophthalmos in the newborn. Am J Ophthalmol 1943;26:1314 –1315. 3. Kundert KF. Bilateral exophthalmos in the newborn. Am J Ophthalmol 1951;34:1173. 4. Munoz M, Weatherhead R. Orbital hemorrhage in a newborn. Am J Ophthalmol 1989;108:206 –208. 5. Polito E, Leccisotti A. Diagnosis and treatment of orbital hemorrhagic lesions. Ann Ophthalmol 1994;26:85–93.
Hyphema Caused by a Metallic Intraocular Foreign Body During Magnetic Resonance Imaging Christopher N. Ta, MD, and R. Wayne Bowman, MD PURPOSE: To report a 63-year-old man with a retained intraocular foreign body who developed a hyphema during magnetic resonance imaging (MRI) of the brain. METHODS: Case report and review of the current literature on ocular injury caused by intraocular foreign bodies when subjected to an electromagnetic field. RESULTS: Our patient underwent a brain MRI, and the intraocular foreign body caused a hyphema and increased Accepted for publication Nov 23, 1999. From the Department of Ophthalmology, Stanford University Medical Center, Stanford, California (C.N.T.), and the Department of Ophthalmology,, Southwestern Medical School, University of Texas, Dallas, Texas (R.W.B.). Inquiries to Christopher N. Ta, MD, Department of Ophthalmology, Stanford University Medical Center, Boswell Bldg, Rm A-157, 300 Pasteur Dr, Stanford, CA 94305-5308; fax: (650) 723-7918.
VOL. 129, NO. 4
intraocular pressure. The presence and location of the intraocular foreign body were determined by computed tomography (CT). CONCLUSION: Magnetic resonance imaging can cause serious ocular injury in patients with ferromagnetic intraocular foreign bodies. This case demonstrates the importance of obtaining an occupational history, and, when indicated, a skull x-ray or CT to rule out intraocular foreign body before an MRI study. (Am J Ophthalmol 2000;129:533–534. © 2000 by Elsevier Science Inc. All rights reserved.)
plant 11 months prior to admission presented with a 5-day history of fever, headaches, and change in mental status. A brain magnetic resonance imaging (MRI) study was obtained. Immediately after the MRI, the patient complained of sudden pain and loss of vision in the left eye that occurred while he was in the MRI scanner. The patient was a metal worker but denied any intraocular injury. On examination at the bedside after the MRI, his visual acuity with a near card (with correction) was RE: 20/30 and LE: counting fingers at 3 inches. The intraocular pressure was RE: 12 mm Hg and LE: 30 mm Hg. His right pupil was reactive. His left pupil was minimally reactive at 6 mm. No afferent pupillary defect was detected. Examination of the right eye was normal except for a moderate cataract. The left eye had a 2-mm paracentral corneal scar and a 50% hyphema. The lens had moderate nuclear sclerosis. Fundus examination revealed an attached retina with no vitreous hemorrhage. A metallic intraocular foreign body was suspected. An orbital computed tomography (CT) scan revealed a radiodense intraocular foreign body in the inferior anterior chamber in the left eye (Figure 1). The right orbit had a foreign body just outside the globe in the inferior temporal quadrant (Figure 2). Treatment included bedrest, elevation of the head of bed 30 degrees, scopolamine, prednisolone acetate, timolol, and brimonidine. By hospital day 5, the left eye had an intraocular pressure of 14 with a 1-mm blood clot and retinal flame hemorrhages. There were also scattered subconjunctival hemorrhages in both eyes. Unfortunately, our patient developed multiorgan failure and died. The cause of death is unknown, but sepsis or thrombocytopenic thrombotic purpura was suspected. Several studies show that ferromagnetic bodies have significant movements when subjected to MRI, with the potential to cause intraocular injury. Lagouros and associates showed that all ferromagnetic materials (such as a BB pellet, a staple, or a screw) move during an MRI study in vitro when suspended in gelatin.1 Three of four ferromagnetic bodies implanted in the vitreous of rabbits showed movement that caused retinal holes, detachment, and