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Oedipism: bilateral self-enucleation
Case Reports
127
[9] Schnell RG. Drug-induced dyskinesia treated with intravenous diazepam. J Fla Med Assoc 1972;59:22 - 3. [10] Guala A, Mittino D, Fabbrocini P, Ghini T. Familial metoclopramideinduced dystonic reactions. Mov Disord 1992;7:385 - 6.
Oedipism: bilateral self-enucleation A 35-year-old man was previously diagnosed with a manic depressive disorder leading to a suicide attempt 15 years earlier. He also had a history of substance abuse (marijuana usage and moderate consumption of alcohol) and was treated with diazepam (4 mg/d). Six months before his current presentation, he felt that he was born again as a Christian, and this was associated with increasing religious preoccupations using the bible. On the night before his admission, he had a special religious experience of being able to look into the next life, and there he saw himself as a child molester. To avoid these visions, he decided to remove his eyes using his hands. On arrival at hospital, there was significant bilateral periorbital bruising and swelling but no active bleeding. Both enucleated eyes were attached to a long stalk of the optic nerve, and part of the optic chiasm could clearly be identified (Fig. 1). The patient was sedated with intravenous haloperidol and diazepam, and an urgent computed tomographic scan was performed (Fig. 2), which showed no intracranial hemorrhages. The patient was then taken to the operating room for orbital exploration. Bilateral cerebrospinal fluid (CSF) leaks were identified, and so, the annulus of Zinn was oversown. An orbital implant was placed in both orbits and attached to the recti muscles. The postoperative recovery was uneventful. On a psychiatric evaluation, the patient was diagnosed as suffering a manic phase of atypical manic depressive psychosis with schizophreniform features and was hospitalized in the psychiatric department for several months. Oedipism is the medical term used to describe the rare act of self-enucleation. It is based on the story of Oedipus, who removed both his eyes after the death of his mother, as an expression of guilt for committing incest.
Fig. 2 Axial computed tomographic scan of the brain and orbits at the level of the optic canals. The lateral and medial recti muscles are present (white arrows), but no eye globes are seen. There is no obvious intracranial pathology.
As in our case, Oedipism generally occurs in patients with psychotic diseases such as schizophrenia and drug-induced psychosis [1- 4]. The immediate patient management requires orbital exploration, control of possible CSF leak, and socket reconstruction. In addition, neurologic monitoring and imaging is required to identify and treat any life-threatening intracranial complications, such as intracranial or subarachnoid hemorrhage, CSF leakage, and bacterial meningitis [5]. Long-term psychiatric therapy is mandatory to prevent further self-mutilation and suicide attempts [2]. Igal Leibovitch MD George Pietris MBBS, FRANZCO Robert Casson MBBS, FRANZCO Department of Opthalmology and Visual Sciences Royal Adelaide Hospital University of Adelaide Adelaide 5000, South Australia E-mail address: [email protected] Dinesh Selva MBBS, FRANZCO Department of Opthalmology and Visual Sciences Royal Adelaide Hospital University of Adelaide Adelaide 5000, South Australia Departments of Surgery and Medicine University of Adelaide Adelaide 5000, Australia doi:10.1016/j.ajem.2005.08.019
References Fig. 1 The self-enucleated globes with a long stalk of the optic nerve extending to the optic chiasm (arrows).
[1] Wilson WA. Oedipismus. Am J Ophthalmol 1955;40:563 - 7. [2] Krauss HR, Yee RD, Foos RY. Autoenucleation. Surv Ophthalmol 1984; 29:179 - 87.
128
Case Reports
[3] Jones NP. Self-enucleation and psychosis. Br J Ophthalmol 1990; 74:571 - 3. [4] Aung T, Yap EY, Fam HB, Law NM. Oedipism. Aust N Z J Ophthalmol 1996;24:153 - 7. [5] Khan JA, Buescher L, Ide CH, Pettigrove B. Medical management of self-enucleation. Arch Ophthalmol 1985;103:386 - 9.
Mycotic aneurysm of superior mesenteric artery branch presenting as pulsatile abdominal mass Superior mesenteric artery aneurysms (SMAAs) are an uncommon but lethal entity. A 29-year-old woman presented with 3 days of fever and severe periumbilical pain, intensified with meals. She denied any history of trauma, operation, alcohol consumption, intravenous drug abuse, or cardiovascular disease. She was thinly-built, normotensive, and febrile (39.48C). Physical examination showed a systolic heart murmur and a mobile, tender, pulsatile, bruits-producing 3 4 cm mass in the umbilical area. Laboratory results were unremarkable except leukocyte count 18.50 103/lL and serum amylase 381 U/L. Ultrasound identified a normal-caliber aorta and an aneurysmal anechoic mass beneath the umbilicus. Abdominal computed tomography exhibited a saccular aneurysm, 3 4 cm in diameter, but could not show the communication with SMA clearly (Fig. 1). Echocardiography demonstrated bicuspid aortic valves with moderate regurgitation, but found no vegetation. In view of her previous contrast medium allergy history during an intravenous pyelogram for renal stones, we deferred the angiography. At emergent laparotomy, an aneurysm encountered at the ileal branch of SMA (Fig. 2) was treated successfully by simple ligation and excision. Microbiologic studies of aneurysm tissue and blood cultures both grew Streptococcus viridans. After a 2-week course of intravenous antibiotics, she was discharged and remained well after 10 years. Superior mesenteric artery aneurysms, the third most common (5.5%) visceral artery aneurysms, remain a challenge to recognize and treat [1]. Mycotic aneurysms
Fig. 1 Computed tomography of the abdomen showing a saccular SMA aneurysm (arrows), 3 4 cm in diameter.
Fig. 2 Intraoperative photograph showing a 3 4 cm aneurysm (arrows) localized at the ileal branch of the SMA.
account for 50% to 60% of SMAAs. Other identified etiologies include atherosclerosis, arterial dysplasia, collagen vascular disorders, arteritis, and trauma [1-3]. Most nonmycotic SMAAs affect patients older than 60 years [1]. Contrarily, mycotic aneurysms often occur in patients younger than 50 years as a consequence of bacterial endocarditis, and nonhemolytic Streptococcus is the most common pathogen. The possible pathogenesis involves bacterial seeding via blood, lymphatics, or direct invasion, followed by vessel wall destruction and aneurysm formation [4]. In our patient, the valvular abnormality may contribute toward the bacteremia and aneurysm formation, despite absence of endocardial vegetation. Visceral artery aneurysms are often asymptomatic, but most SMAAs present with significant abdominal pain [1,5]. Although the nonspecific manifestations may be mistaken for pancreatitis, perforated viscus, ruptured ovarian cysts, or appendicitis, a triad of fever, abdominal pain, and a pulsatile abdominal mass should indicate mycotic aneurysms [6,7]. Physical examination is rarely diagnostic because most lesions are small, but imaging studies are straightforward, including ultrasonography, computed tomography, or angiography [8,9]. Conventional angiography is still the criterion standard because of its diagnostic and therapeutic role. Because SMAAs have a definite rupture risk (38%-50%) [2,10] and high resultant mortality rate (40%-60%) [11], elective surgery remains the mainstay of therapy and carries a mortality rate of less than 15% [1]. Therapeutic options include arterial ligation, aneurysmectomy, vascular reconstruction, and transcatheter embolization, often with attempts to preserve end-organ perfusion [3,10]. After surgery, a 6-week course of organism-specific antibiotic therapy is needed [4]. Despite their rarity, mycotic SMAAs should be considered in differential diagnosis in patients with fever, abdominal pain, and a pulsatile abdominal mass. Timely diagnosis depends on a high suspicion and advanced imaging modalities. Aggressive medical and surgical intervention should not be delayed because of its definite risk of rupture and death.
127
[9] Schnell RG. Drug-induced dyskinesia treated with intravenous diazepam. J Fla Med Assoc 1972;59:22 - 3. [10] Guala A, Mittino D, Fabbrocini P, Ghini T. Familial metoclopramideinduced dystonic reactions. Mov Disord 1992;7:385 - 6.
Oedipism: bilateral self-enucleation A 35-year-old man was previously diagnosed with a manic depressive disorder leading to a suicide attempt 15 years earlier. He also had a history of substance abuse (marijuana usage and moderate consumption of alcohol) and was treated with diazepam (4 mg/d). Six months before his current presentation, he felt that he was born again as a Christian, and this was associated with increasing religious preoccupations using the bible. On the night before his admission, he had a special religious experience of being able to look into the next life, and there he saw himself as a child molester. To avoid these visions, he decided to remove his eyes using his hands. On arrival at hospital, there was significant bilateral periorbital bruising and swelling but no active bleeding. Both enucleated eyes were attached to a long stalk of the optic nerve, and part of the optic chiasm could clearly be identified (Fig. 1). The patient was sedated with intravenous haloperidol and diazepam, and an urgent computed tomographic scan was performed (Fig. 2), which showed no intracranial hemorrhages. The patient was then taken to the operating room for orbital exploration. Bilateral cerebrospinal fluid (CSF) leaks were identified, and so, the annulus of Zinn was oversown. An orbital implant was placed in both orbits and attached to the recti muscles. The postoperative recovery was uneventful. On a psychiatric evaluation, the patient was diagnosed as suffering a manic phase of atypical manic depressive psychosis with schizophreniform features and was hospitalized in the psychiatric department for several months. Oedipism is the medical term used to describe the rare act of self-enucleation. It is based on the story of Oedipus, who removed both his eyes after the death of his mother, as an expression of guilt for committing incest.
Fig. 2 Axial computed tomographic scan of the brain and orbits at the level of the optic canals. The lateral and medial recti muscles are present (white arrows), but no eye globes are seen. There is no obvious intracranial pathology.
As in our case, Oedipism generally occurs in patients with psychotic diseases such as schizophrenia and drug-induced psychosis [1- 4]. The immediate patient management requires orbital exploration, control of possible CSF leak, and socket reconstruction. In addition, neurologic monitoring and imaging is required to identify and treat any life-threatening intracranial complications, such as intracranial or subarachnoid hemorrhage, CSF leakage, and bacterial meningitis [5]. Long-term psychiatric therapy is mandatory to prevent further self-mutilation and suicide attempts [2]. Igal Leibovitch MD George Pietris MBBS, FRANZCO Robert Casson MBBS, FRANZCO Department of Opthalmology and Visual Sciences Royal Adelaide Hospital University of Adelaide Adelaide 5000, South Australia E-mail address: [email protected] Dinesh Selva MBBS, FRANZCO Department of Opthalmology and Visual Sciences Royal Adelaide Hospital University of Adelaide Adelaide 5000, South Australia Departments of Surgery and Medicine University of Adelaide Adelaide 5000, Australia doi:10.1016/j.ajem.2005.08.019
References Fig. 1 The self-enucleated globes with a long stalk of the optic nerve extending to the optic chiasm (arrows).
[1] Wilson WA. Oedipismus. Am J Ophthalmol 1955;40:563 - 7. [2] Krauss HR, Yee RD, Foos RY. Autoenucleation. Surv Ophthalmol 1984; 29:179 - 87.
128
Case Reports
[3] Jones NP. Self-enucleation and psychosis. Br J Ophthalmol 1990; 74:571 - 3. [4] Aung T, Yap EY, Fam HB, Law NM. Oedipism. Aust N Z J Ophthalmol 1996;24:153 - 7. [5] Khan JA, Buescher L, Ide CH, Pettigrove B. Medical management of self-enucleation. Arch Ophthalmol 1985;103:386 - 9.
Mycotic aneurysm of superior mesenteric artery branch presenting as pulsatile abdominal mass Superior mesenteric artery aneurysms (SMAAs) are an uncommon but lethal entity. A 29-year-old woman presented with 3 days of fever and severe periumbilical pain, intensified with meals. She denied any history of trauma, operation, alcohol consumption, intravenous drug abuse, or cardiovascular disease. She was thinly-built, normotensive, and febrile (39.48C). Physical examination showed a systolic heart murmur and a mobile, tender, pulsatile, bruits-producing 3 4 cm mass in the umbilical area. Laboratory results were unremarkable except leukocyte count 18.50 103/lL and serum amylase 381 U/L. Ultrasound identified a normal-caliber aorta and an aneurysmal anechoic mass beneath the umbilicus. Abdominal computed tomography exhibited a saccular aneurysm, 3 4 cm in diameter, but could not show the communication with SMA clearly (Fig. 1). Echocardiography demonstrated bicuspid aortic valves with moderate regurgitation, but found no vegetation. In view of her previous contrast medium allergy history during an intravenous pyelogram for renal stones, we deferred the angiography. At emergent laparotomy, an aneurysm encountered at the ileal branch of SMA (Fig. 2) was treated successfully by simple ligation and excision. Microbiologic studies of aneurysm tissue and blood cultures both grew Streptococcus viridans. After a 2-week course of intravenous antibiotics, she was discharged and remained well after 10 years. Superior mesenteric artery aneurysms, the third most common (5.5%) visceral artery aneurysms, remain a challenge to recognize and treat [1]. Mycotic aneurysms
Fig. 1 Computed tomography of the abdomen showing a saccular SMA aneurysm (arrows), 3 4 cm in diameter.
Fig. 2 Intraoperative photograph showing a 3 4 cm aneurysm (arrows) localized at the ileal branch of the SMA.
account for 50% to 60% of SMAAs. Other identified etiologies include atherosclerosis, arterial dysplasia, collagen vascular disorders, arteritis, and trauma [1-3]. Most nonmycotic SMAAs affect patients older than 60 years [1]. Contrarily, mycotic aneurysms often occur in patients younger than 50 years as a consequence of bacterial endocarditis, and nonhemolytic Streptococcus is the most common pathogen. The possible pathogenesis involves bacterial seeding via blood, lymphatics, or direct invasion, followed by vessel wall destruction and aneurysm formation [4]. In our patient, the valvular abnormality may contribute toward the bacteremia and aneurysm formation, despite absence of endocardial vegetation. Visceral artery aneurysms are often asymptomatic, but most SMAAs present with significant abdominal pain [1,5]. Although the nonspecific manifestations may be mistaken for pancreatitis, perforated viscus, ruptured ovarian cysts, or appendicitis, a triad of fever, abdominal pain, and a pulsatile abdominal mass should indicate mycotic aneurysms [6,7]. Physical examination is rarely diagnostic because most lesions are small, but imaging studies are straightforward, including ultrasonography, computed tomography, or angiography [8,9]. Conventional angiography is still the criterion standard because of its diagnostic and therapeutic role. Because SMAAs have a definite rupture risk (38%-50%) [2,10] and high resultant mortality rate (40%-60%) [11], elective surgery remains the mainstay of therapy and carries a mortality rate of less than 15% [1]. Therapeutic options include arterial ligation, aneurysmectomy, vascular reconstruction, and transcatheter embolization, often with attempts to preserve end-organ perfusion [3,10]. After surgery, a 6-week course of organism-specific antibiotic therapy is needed [4]. Despite their rarity, mycotic SMAAs should be considered in differential diagnosis in patients with fever, abdominal pain, and a pulsatile abdominal mass. Timely diagnosis depends on a high suspicion and advanced imaging modalities. Aggressive medical and surgical intervention should not be delayed because of its definite risk of rupture and death.