Locked-in Syndrome Due to Metastatic Pontomedullary Tumor Case Report
A 21-year-old man presented with an extremely rare case of locked-in syndrome caused by a metastatic brainstem tumor manifesting as quadriplegia, lower cranial nerve pareses, and irregular respiration. Cranial magnetic resonance imaging revealed a large pontomedullary tumor. An emergency operation w...
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| Published in | Neurologia medico-chirurgica Vol. 43; no. 10; pp. 497 - 500 |
|---|---|
| Main Authors | , |
| Format | Journal Article |
| Language | English |
| Published |
Japan
The Japan Neurosurgical Society
01.01.2003
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0470-8105 1349-8029 1349-8029 |
| DOI | 10.2176/nmc.43.497 |
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| Abstract | A 21-year-old man presented with an extremely rare case of locked-in syndrome caused by a metastatic brainstem tumor manifesting as quadriplegia, lower cranial nerve pareses, and irregular respiration. Cranial magnetic resonance imaging revealed a large pontomedullary tumor. An emergency operation was performed via a posterior fossa approach and the tumor was grossly totally removed. The histological diagnosis was malignant melanoma. The clinical status of the patient remained unchanged and he died on postoperative day 34 of diffuse bronchopneumonia. Locked-in syndrome is characterized by quadriplegia, lower cranial nerve paralysis, and mutism but with maintenance of consciousness, as well as vertical eye movements and eyelid blinking. This case suggests that locked-in syndrome should be considered in any patient seemingly comatose or stuporous, this syndrome may be due to a pontomedullary tumor, and malignant melanoma metastasis should be considered in the differential diagnosis of patients who present with brainstem tumor. |
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| AbstractList | A 21-year-old man presented with an extremely rare case of locked-in syndrome caused by a metastatic brainstem tumor manifesting as quadriplegia, lower cranial nerve pareses, and irregular respiration. Cranial magnetic resonance imaging revealed a large pontomedullary tumor. An emergency operation was performed via a posterior fossa approach and the tumor was grossly totally removed. The histological diagnosis was malignant melanoma. The clinical status of the patient remained unchanged and he died on postoperative day 34 of diffuse bronchopneumonia. Locked-in syndrome is characterized by quadriplegia, lower cranial nerve paralysis, and mutism but with maintenance of consciousness, as well as vertical eye movements and eyelid blinking. This case suggests that locked-in syndrome should be considered in any patient seemingly comatose or stuporous, this syndrome may be due to a pontomedullary tumor, and malignant melanoma metastasis should be considered in the differential diagnosis of patients who present with brainstem tumor. A 21-year-old man presented with an extremely rare case of locked-in syndrome caused by a metastatic brainstem tumor manifesting as quadriplegia, lower cranial nerve pareses, and irregular respiration. Cranial magnetic resonance imaging revealed a large pontomedullary tumor. An emergency operation was performed via a posterior fossa approach and the tumor was grossly totally removed. The histological diagnosis was malignant melanoma. The clinical status of the patient remained unchanged and he died on postoperative day 34 of diffuse bronchopneumonia. Locked-in syndrome is characterized by quadriplegia, lower cranial nerve paralysis, and mutism but with maintenance of consciousness, as well as vertical eye movements and eyelid blinking. This case suggests that locked-in syndrome should be considered in any patient seemingly comatose or stuporous, this syndrome may be due to a pontomedullary tumor, and malignant melanoma metastasis should be considered in the differential diagnosis of patients who present with brainstem tumor.A 21-year-old man presented with an extremely rare case of locked-in syndrome caused by a metastatic brainstem tumor manifesting as quadriplegia, lower cranial nerve pareses, and irregular respiration. Cranial magnetic resonance imaging revealed a large pontomedullary tumor. An emergency operation was performed via a posterior fossa approach and the tumor was grossly totally removed. The histological diagnosis was malignant melanoma. The clinical status of the patient remained unchanged and he died on postoperative day 34 of diffuse bronchopneumonia. Locked-in syndrome is characterized by quadriplegia, lower cranial nerve paralysis, and mutism but with maintenance of consciousness, as well as vertical eye movements and eyelid blinking. This case suggests that locked-in syndrome should be considered in any patient seemingly comatose or stuporous, this syndrome may be due to a pontomedullary tumor, and malignant melanoma metastasis should be considered in the differential diagnosis of patients who present with brainstem tumor. |
| Author | INCI, Servet ÖZGEN, Tuncalp |
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| BackLink | https://www.ncbi.nlm.nih.gov/pubmed/14620202$$D View this record in MEDLINE/PubMed |
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| Cites_doi | 10.1016/S0360-3016(00)01355-9 10.1016/S0003-9993(21)01692-0 10.3171/jns.1998.88.1.0011 10.1016/0022-510X(83)90127-2 10.1111/j.1600-0404.1986.tb03303.x 10.1212/WNL.36.1.80 10.1002/ana.410120413 10.1093/brain/64.4.273 10.1007/BF00306266 10.1212/WNL.21.11.1140 10.1001/archneur.1982.00510150015004 10.1136/jnnp.42.11.1062 10.1212/WNL.26.2.180 10.1007/BF00314463 10.1212/WNL.26.12.1185 10.1002/(SICI)1097-0142(19961015)78:8<1781::AID-CNCR19>3.3.CO;2-Z 10.1016/0303-8467(84)90289-0 10.1002/ana.410010614 10.1007/BF00313183 10.1001/archneur.1982.00510190053017 10.1212/WNL.29.2.147 10.1161/01.STR.17.4.758 |
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| References | 21) Reznik M: Neuropathology in seven cases of locked-in syndrome. J Neurol Sci 60: 67-78, 1983 12) McCusker EA, Rudick RA, Honch GW: Recovery from the locked-in syndrome. Arch Neurol 39: 145-147, 1982 22) Sampson JH, Carter JH Jr, Friedman AH, Seigler HF: Demographics, prognosis, and therapy in 702 patients with brain metastases from malignant melanoma. J Neurosurg 88: 11-20, 1998 23) Tijssen CC, Ter Bruggen JP: Locked-in syndrome associated with ocular bobbing. Acta Neurol Scand 73: 444-446, 1986 18) Patterson JR, Grabois M: Locked-in syndrome: a review of 139 cases. Stroke 17: 758-764, 1986 20) Pogacar S, Finelli PF, Lee HY: Locked-in syndrome caused by a metastasis. R I Med J 66: 147-150, 1983 17) Nussbaum ES, Djalilian HR, Cho KH, Hall WA: Brain metastases. Histology, multiplicity, surgery and survival. Cancer 78: 1781-1788, 1996 7) Ellerhorst J, Strom E, Nordone E, McCutcheon J: Whole brain irradiation for patients with metastatic melanoma: a review of 87 cases. Int J Radiat Oncol Biol Phys 49: 93-97, 2001 4) Chase TN, Moretti IL, Prensky AL: Clinical and electroencephalographic manifestations of vascular lesions of the pons. Neurology 18: 358-368, 1968 25) Yang CCS, Lieberman JS, Hong CZ: Early smooth horizontal eye movement: a favorable prognostic sign in patients with locked-in syndrome. Arch Phys Med Rehabil 70: 230-232, 1989 13) Merlo A, Weder B, Ketz E, Matter L: Locked-in state in Borrelia burgdorferi meningitis. J Neurol 236: 305-306, 1989 15) Murphy MJ, Brenton DW, Aschenbrener CA, Van Gilder JC: Locked-in syndrome caused by a solitary pontine abscess. J Neurol Neurosurg Psychiatry 42: 1062-1065, 1979 24) Virgile RS: Locked-in syndrome. Clin Neurol Neurosurg 86: 275-279, 1984 1) Bauer G, Gerstenbrand F, Hengl R: Involuntary motor phenomena in the locked-in syndrome. J Neurol 223: 191-198, 1980 2) Bauer G, Prugger M, Rumpl E: Stimulus evoked oral automatism in the locked-in syndrome. Arch Neurol 39: 435-436, 1982 19) Plum F, Posner JB: The Diagnosis of Stupor and Coma. Philadelphia, FA Davis, 1966, pp 92-93 5) Cherington M, Stears J, Hodges J: Locked-in syndrome caused by a tumour. Neurology 26: 180-182, 1976 8) Forti A, Ambrosetto G, Amore M: Locked-in syndrome in multiple sclerosis with sparing of the ventral portion of the pons. Ann Neurol 12: 393-394, 1982 11) Masuzawa H, Sato J, Kamitani H, Kamikura T, Aoki N: Pontine gliomas causing locked-in syndrome. Childs Nerv Syst 39: 256-259, 1993 10) Keane JR: Locked-in syndrome after head and neck trauma. Neurology 36: 80-82, 1986 16) Nordgren RE, Markesbery WR, Fukuda K, Reeves AG: Seven cases of cerebromedullospinal disconnection: The locked-in syndrome. Neurology 21: 1140-1148, 1971 3) Britt RH, Herrick MK, Hamilton RD: Traumatic locked-in syndrome. Ann Neurol 1: 590-592, 1977 9) Hawkes CH, Bryan-Smith L: Locked-in syndrome caused by a tumour. Neurology 26: 1185, 1976 6) Crains H, Oldfield RC, Pennybacker JB: Akinetic mutism with an epidermoid cyst of third ventricle. Brain 64: 273-290, 1941 14) Messert B, Orrison WW, Hawkins MJ: Central pontine myelinolysis: considerations on etiology, diagnosis and treatment. Neurology 29: 147-160, 1979 McCusker EA, Rudick RA, Honch GW (12) 1982; 39 Pogacar S, Finelli PF, Lee HY (20) 1983; 66 Merlo A, Weder B, Ketz E, Matter L (13) 1989; 236 Cherington M, Stears J, Hodges J (5) 1976; 26 Britt RH, Herrick MK, Hamilton RD (3) 1977; 1 Forti A, Ambrosetto G, Amore M (8) 1982; 12 Ellerhorst J, Strom E, Nordone E, M (7) 2001; 49 Bauer G, Gerstenbrand F, Hengl R (1) 1980; 223 Chase TN, Moretti IL, Prensky AL (4) 1968; 18 Messert B, Orrison WW, Hawkins MJ (14) 1979; 29 Yang CCS, Lieberman JS, Hong CZ (25) 1989; 70 Crains H, Oldfield RC, Pennybacker (6) 1941; 64 Sampson JH, Carter JH Jr, Friedman (22) 1998; 88 Virgile RS (24) 1984; 86 Hawkes CH, Bryan-Smith L (9) 1976; 26 Masuzawa H, Sato J, Kamitani H, Kam (11) 1993; 39 Keane JR (10) 1986; 36 17 Patterson JR, Grabois M (18) 1986; 17 Plum F, Posner JB (19) 1966 Murphy MJ, Brenton DW, Aschenbrener (15) 1979; 42 Tijssen CC, Ter Bruggen JP (23) 1986; 73 Reznik M (21) 1983; 60 Bauer G, Prugger M, Rumpl E (2) 1982; 39 Nordgren RE, Markesbery WR, Fukuda (16) 1971; 21 |
| References_xml | – reference: 24) Virgile RS: Locked-in syndrome. Clin Neurol Neurosurg 86: 275-279, 1984 – reference: 25) Yang CCS, Lieberman JS, Hong CZ: Early smooth horizontal eye movement: a favorable prognostic sign in patients with locked-in syndrome. Arch Phys Med Rehabil 70: 230-232, 1989 – reference: 16) Nordgren RE, Markesbery WR, Fukuda K, Reeves AG: Seven cases of cerebromedullospinal disconnection: The locked-in syndrome. Neurology 21: 1140-1148, 1971 – reference: 18) Patterson JR, Grabois M: Locked-in syndrome: a review of 139 cases. Stroke 17: 758-764, 1986 – reference: 21) Reznik M: Neuropathology in seven cases of locked-in syndrome. J Neurol Sci 60: 67-78, 1983 – reference: 8) Forti A, Ambrosetto G, Amore M: Locked-in syndrome in multiple sclerosis with sparing of the ventral portion of the pons. Ann Neurol 12: 393-394, 1982 – reference: 17) Nussbaum ES, Djalilian HR, Cho KH, Hall WA: Brain metastases. Histology, multiplicity, surgery and survival. Cancer 78: 1781-1788, 1996 – reference: 13) Merlo A, Weder B, Ketz E, Matter L: Locked-in state in Borrelia burgdorferi meningitis. J Neurol 236: 305-306, 1989 – reference: 19) Plum F, Posner JB: The Diagnosis of Stupor and Coma. Philadelphia, FA Davis, 1966, pp 92-93 – reference: 6) Crains H, Oldfield RC, Pennybacker JB: Akinetic mutism with an epidermoid cyst of third ventricle. Brain 64: 273-290, 1941 – reference: 14) Messert B, Orrison WW, Hawkins MJ: Central pontine myelinolysis: considerations on etiology, diagnosis and treatment. Neurology 29: 147-160, 1979 – reference: 22) Sampson JH, Carter JH Jr, Friedman AH, Seigler HF: Demographics, prognosis, and therapy in 702 patients with brain metastases from malignant melanoma. J Neurosurg 88: 11-20, 1998 – reference: 4) Chase TN, Moretti IL, Prensky AL: Clinical and electroencephalographic manifestations of vascular lesions of the pons. Neurology 18: 358-368, 1968 – reference: 1) Bauer G, Gerstenbrand F, Hengl R: Involuntary motor phenomena in the locked-in syndrome. J Neurol 223: 191-198, 1980 – reference: 12) McCusker EA, Rudick RA, Honch GW: Recovery from the locked-in syndrome. Arch Neurol 39: 145-147, 1982 – reference: 5) Cherington M, Stears J, Hodges J: Locked-in syndrome caused by a tumour. Neurology 26: 180-182, 1976 – reference: 15) Murphy MJ, Brenton DW, Aschenbrener CA, Van Gilder JC: Locked-in syndrome caused by a solitary pontine abscess. J Neurol Neurosurg Psychiatry 42: 1062-1065, 1979 – reference: 20) Pogacar S, Finelli PF, Lee HY: Locked-in syndrome caused by a metastasis. R I Med J 66: 147-150, 1983 – reference: 23) Tijssen CC, Ter Bruggen JP: Locked-in syndrome associated with ocular bobbing. Acta Neurol Scand 73: 444-446, 1986 – reference: 7) Ellerhorst J, Strom E, Nordone E, McCutcheon J: Whole brain irradiation for patients with metastatic melanoma: a review of 87 cases. Int J Radiat Oncol Biol Phys 49: 93-97, 2001 – reference: 3) Britt RH, Herrick MK, Hamilton RD: Traumatic locked-in syndrome. Ann Neurol 1: 590-592, 1977 – reference: 10) Keane JR: Locked-in syndrome after head and neck trauma. Neurology 36: 80-82, 1986 – reference: 11) Masuzawa H, Sato J, Kamitani H, Kamikura T, Aoki N: Pontine gliomas causing locked-in syndrome. Childs Nerv Syst 39: 256-259, 1993 – reference: 9) Hawkes CH, Bryan-Smith L: Locked-in syndrome caused by a tumour. Neurology 26: 1185, 1976 – reference: 2) Bauer G, Prugger M, Rumpl E: Stimulus evoked oral automatism in the locked-in syndrome. Arch Neurol 39: 435-436, 1982 – volume: 49 start-page: 93 issn: 0360-3016 year: 2001 ident: 7 publication-title: Int J Radiat Oncol Biol Phys doi: 10.1016/S0360-3016(00)01355-9 – volume: 70 start-page: 230 issn: 0003-9993 year: 1989 ident: 25 publication-title: Arch Phys Med Rehabil doi: 10.1016/S0003-9993(21)01692-0 – volume: 88 start-page: 11 issn: 0022-3085 year: 1998 ident: 22 publication-title: J Neurosurg doi: 10.3171/jns.1998.88.1.0011 – volume: 60 start-page: 67 issn: 0022-510X year: 1983 ident: 21 publication-title: J Neurol Sci doi: 10.1016/0022-510X(83)90127-2 – volume: 73 start-page: 444 issn: 0001-6314 year: 1986 ident: 23 publication-title: Acta Neurol Scand doi: 10.1111/j.1600-0404.1986.tb03303.x – volume: 36 start-page: 80 issn: 0028-3878 year: 1986 ident: 10 publication-title: Neurology doi: 10.1212/WNL.36.1.80 – volume: 12 start-page: 393 issn: 0364-5134 year: 1982 ident: 8 publication-title: Ann Neurol doi: 10.1002/ana.410120413 – volume: 64 start-page: 273 issn: 0006-8950 year: 1941 ident: 6 publication-title: Brain doi: 10.1093/brain/64.4.273 – volume: 39 start-page: 256 issn: 0256-7040 year: 1993 ident: 11 publication-title: Childs Nerv Syst doi: 10.1007/BF00306266 – volume: 21 start-page: 1140 issn: 0028-3878 year: 1971 ident: 16 publication-title: Neurology doi: 10.1212/WNL.21.11.1140 – start-page: 92 year: 1966 ident: 19 publication-title: The Diagnosis of Stupor and Coma – volume: 39 start-page: 145 issn: 0003-9942 year: 1982 ident: 12 publication-title: Arch Neurol doi: 10.1001/archneur.1982.00510150015004 – volume: 42 start-page: 1062 issn: 0022-3050 year: 1979 ident: 15 publication-title: J Neurol Neurosurg Psychiatry doi: 10.1136/jnnp.42.11.1062 – volume: 26 start-page: 180 issn: 0028-3878 year: 1976 ident: 5 publication-title: Neurology doi: 10.1212/WNL.26.2.180 – volume: 236 start-page: 305 issn: 0340-5354 year: 1989 ident: 13 publication-title: J Neurol doi: 10.1007/BF00314463 – volume: 26 start-page: 1185 issn: 0028-3878 year: 1976 ident: 9 publication-title: Neurology doi: 10.1212/WNL.26.12.1185 – volume: 18 start-page: 358 issn: 0028-3878 year: 1968 ident: 4 publication-title: Neurology – volume: 66 start-page: 147 issn: 0360-067X year: 1983 ident: 20 publication-title: R I Med J – ident: 17 doi: 10.1002/(SICI)1097-0142(19961015)78:8<1781::AID-CNCR19>3.3.CO;2-Z – volume: 86 start-page: 275 issn: 0303-8467 year: 1984 ident: 24 publication-title: Clin Neurol Neurosurg doi: 10.1016/0303-8467(84)90289-0 – volume: 1 start-page: 590 issn: 0364-5134 year: 1977 ident: 3 publication-title: Ann Neurol doi: 10.1002/ana.410010614 – volume: 223 start-page: 191 issn: 0340-5354 year: 1980 ident: 1 publication-title: J Neurol doi: 10.1007/BF00313183 – volume: 39 start-page: 435 issn: 0003-9942 year: 1982 ident: 2 publication-title: Arch Neurol doi: 10.1001/archneur.1982.00510190053017 – volume: 29 start-page: 147 issn: 0028-3878 year: 1979 ident: 14 publication-title: Neurology doi: 10.1212/WNL.29.2.147 – volume: 17 start-page: 758 issn: 0039-2499 year: 1986 ident: 18 publication-title: Stroke doi: 10.1161/01.STR.17.4.758 |
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| SubjectTerms | Adult Brain Stem Neoplasms - complications Brain Stem Neoplasms - secondary Humans locked-in syndrome Male malignant melanoma Melanoma - complications Melanoma - secondary pontomedullary tumor Quadriplegia - etiology Skin Neoplasms - pathology |
| Subtitle | Case Report |
| Title | Locked-in Syndrome Due to Metastatic Pontomedullary Tumor |
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