THE EDITORphocytosis in her blood. Antinuclear antibodies andimmunecomplexeswerestill present....
2
Journal of Neurology, Neurosurgery, and Psychiatry 1990;53:925-928 LETTERS TO THE EDITOR Unilateral proptosis secondary to par- tially thrombosed giant carotid ophthalmic artery aneurysm A wide variety of conditions have been des- cribed as a cause of unilateral proptosis. The case we describe had unilateral proptosis due to partially thrombosed giant carotid ophthalmic artery aneurysm, the diagnosis of which was confirmed with angiography and magnetic resonance imaging (MRI). So far as we are aware, unilateral proptosis secondary to giant partially thrombosed aneurysm of the carotid ophthalmic artery demonstrated on MR has not been previously reported. A 70 year old woman was admitted com- plaining of a gradual bulging of the right eye and loss of vision over a period of six months. The bulging of the right eye was painless and nonpulsatile. Except for cholelithiasis, she had no other past significant illness. On examination, the right eye showed nonpul- satile proptosis with complete loss of percep- tion to light. Left eye had 6/18 vision with glasses. Fundus examination of the right eye showed primary optic atrophy. No other neurological deficit was found. Plain radiograph of the skull and orbits did not reveal any abnormality. Contrast enhanced computerised tomogra- phy (CT) of the head and orbit showed a huge retrobulbar mass of the right side with mixed density with the area of enhancement associated with proptosis of the right eye. Based on the CT findings, an aneurysm with partial thrombosis was suggested and angiogram was advised. A digital substrac- tion right carotid angiogram revealed an aneurysm at the origin of the ophthalmic artery from the internal carotid artery above the cavernous sinus with non filling of the ophthalmic artery (fig 1). MR was performed with a 1 5-T superconducting system (Mag- netom, Siemens) using spin echo (SE) tech- nique. It showed a flow void phenomenon on all SE sequences in the right carotid artery and patent portion of the lumen of the aneurysm. It was associated with laminated thrombus of mixed stages in the clotted Figure 1 Right common carotid angiogram, lateral projection show aneurysm at the origin of the ophthalmic artery (arrow). X ~ ~~~~~~~~ ~~~~~~~~~ !: .f _ Figure 2 First-echo image of axial MR through the orbits (SE 2800/28) shows patent portion of the aneurysm arising from right internal carotid artery. Note the laminated thrombus of mixed stages (arrows) around the patent aneurysm causing proptosis. portion of the lumen. It measured 3-8 x 4 x 3 cm and filled the whole right retrobulbar space with proptosis (fig 2). Based on MRI and angiography, the diagnosis of right par- tially thrombosed giant carotid ophthalmic artery aneurysm was established. Carotid ophthalmic artery aneurysms arise in the first 2 mm of the internal carotid artery above the cavernous sinus and below the origin of the posterior communicating artery. The aneurysm frequently originates beneath the optic nerve and may extend into and dilate the optic canal.' Due to their dual component nature, partially thrombosed giant aneurysms have a characteristic appearance on spin echo MRI and identification of the signal intensity pattern is specific for this lesion.2 Our case showed all the characteristic features of a partially thrombosed giant aneurysm. A more complete dileneatide of the giant aneurysm components and associated extra aneurysmal parenchymal abnormalities is possible with MRI compared with either CT or angiogra- phy. RK GUPTA Institute of Nuclear Medicine and Allied Sciences, Lucknow Road, India VS MEHTA* NK MISRAt Department of Neurosurgery* and Neuroradiology,t All India Institute of Medical Sciences, New Delhi, India 1 Peris JB, Ross Russell RW. Giant aneurysms of the carotid system presenting as visual field defect. J Neurol Neurosurg Psychiatry 1980;43:1053-64. 2 Atlas SW, Grossman RI, Goldberg HI, et al. Partially thrombosed giant intracranial aneurysms: correlation of MR and pathologic findings. Radiology 1987;162:111-4. Pituitary abscess with recurrent aseptic meningitis Since the first description of pituitary abscess by Simmonds in 1914,' less than 40 cases have been adequately reported; The diagnosis has rarely been made before operation, the radiological findings being those of a pituitary tumour. We report a patient in whom the diagnosis of pituitary abscess was made pre- operatively, to draw attention to the associa- tion with aseptic lymphoplasmocytic menin- 925 gitis, which provided a clue to the diagnosis. This association has been observed only twice before.24 In 1983 a 67 year old woman developed severe frontal headaches progressively wor- sening over several days. Her temperature was 38-5°C and she showed signs of menin- gism. The cerebrospinal fluid (CSF) was xanthochromic with a lymphoplasmocytic pleiocytosis and increased protein content, but bacteriological and virological investiga- tions were negative, including tests for tuber- culosis. Computed tomography (CT) showed moderate enlargement of the subarachnoid spaces. Viral meningitis was presumed and symptoms disappeared gradually with symp- tomatic treatment. In a second episode a month later, she lapsed into a coma for two days. The laboratory findings were similar, treatment was not modified and she recovered after several weeks. One year later, headaches, slowed thinking and meningism recurred. Her sedimentation rate was raised. She had anaemia, polymor- phonuclear leucopaenia and a relative lym- phocytosis. She had circulating immune complexes and antinuclear antibodies, but tests for anti DNA antibodies were negative. Tests of thyroid function showed that TSH was low, with low free T3 but normal free T4. Her CSF had an increased protein content (12 mg/ml) and lymphoplasmocytosis (60 lymphocytes and 15 plasmocytes/ml), but glucose and chloride concentrations were normal. The CT was unchanged. She again recovered without specific treatment. Six months later, she still had a lym- phocytosis in her blood. Antinuclear antibodies and immune complexes were still present. Her free T3 had fallen and she was treated with 0-1 mg L-thyroxine daily. Dur- ing the following months, she experienced several episodes of mental slowing, vertigo, orthostatic hypotension with falls and head- aches but without hyperthermia or signs of meningism. She was referred to the Depart- ment of Neurology, University of Liege, in February 1987. She was moderately obese and hypertensive with brittle hair and thin eyebrows. There was no pubic hair and her pretibial skin was infiltrated. She had a subjective bilateral global diminution of vision, with no apparent hemianopia, but the rest of the neurological examination was normal. Routine blood and urine tests were normal. Antinuclear antibodies were still positive (1/20), but anti DNA antibodies and circu- lating immune complexes were negative. Endocrine tests showed normal basal thyroid function (with 0-1 mg L-thyroxine daily) with undetectable TSH and evidence of hypopituitarism. Injection test of TRH did not increase TSH values. There was only a slight increase in prolactin values (from 110 to 190 clIU/ml). Follicle-stimulating hor- mone (2 9 mIU/ml) and luteinising hormone (< 2-5 mIU/ml) were subnormal for a post menopausal woman and Gn RH did not increase FSH and LH values. Cortisol and ACTH were below the minimal detectable values throughout the circadian cycle. Free urinary cortisol was low at 3 pg/day. CRF test was followed by no increase of ACTH or cortisol. Urinary gravimetry was normal. Ophthalmological examination showed senile bilateral cataract with severe reduction of visual acuity, but no defect in her visual fields. Plain radiographs of the skull showed an enlarged sella with thinning of the right part of the floor; abnormal calcification was on April 4, 2021 by guest. Protected by copyright. http://jnnp.bmj.com/ J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.53.10.925-a on 1 October 1990. Downloaded from
Transcript of THE EDITORphocytosis in her blood. Antinuclear antibodies andimmunecomplexeswerestill present....
-
Journal of Neurology, Neurosurgery, and Psychiatry 1990;53:925-928
LETTERS TOTHE EDITOR
Unilateral proptosis secondary to par-tially thrombosed giant carotidophthalmic artery aneurysm
A wide variety of conditions have been des-cribed as a cause of unilateral proptosis. Thecase we describe had unilateral proptosis dueto partially thrombosed giant carotidophthalmic artery aneurysm, the diagnosis ofwhich was confirmed with angiography andmagnetic resonance imaging (MRI). So far aswe are aware, unilateral proptosis secondaryto giant partially thrombosed aneurysm ofthecarotid ophthalmic artery demonstrated onMR has not been previously reported.A 70 year old woman was admitted com-
plaining of a gradual bulging of the right eyeand loss of vision over a period of six months.The bulging of the right eye was painless andnonpulsatile. Except for cholelithiasis, shehad no other past significant illness. Onexamination, the right eye showed nonpul-satile proptosis with complete loss of percep-tion to light. Left eye had 6/18 vision withglasses. Fundus examination of the right eyeshowed primary optic atrophy. No otherneurological deficit was found. Plainradiograph of the skull and orbits did notreveal any abnormality.
Contrast enhanced computerised tomogra-phy (CT) of the head and orbit showed a hugeretrobulbar mass of the right side with mixeddensity with the area of enhancementassociated with proptosis of the right eye.Based on the CT findings, an aneurysm
with partial thrombosis was suggested andangiogram was advised. A digital substrac-tion right carotid angiogram revealed ananeurysm at the origin of the ophthalmicartery from the internal carotid artery abovethe cavernous sinus with non filling of theophthalmic artery (fig 1). MR was performedwith a 1 5-T superconducting system (Mag-netom, Siemens) using spin echo (SE) tech-nique. It showed a flow void phenomenon onall SE sequences in the right carotid arteryand patent portion of the lumen of theaneurysm. It was associated with laminatedthrombus of mixed stages in the clotted
Figure 1 Right common carotid angiogram,lateral projection show aneurysm at the originof the ophthalmic artery (arrow).
X ~ ~~~~~~~~~~~~~~~~~!: .f _
Figure 2 First-echo image of axial MRthrough the orbits (SE 2800/28) shows patentportion of the aneurysm arisingfrom rightinternal carotid artery. Note the laminatedthrombus ofmixed stages (arrows) around thepatent aneurysm causing proptosis.
portion of the lumen. It measured 3-8 x 4 x3 cm and filled the whole right retrobulbarspace with proptosis (fig 2). Based on MRIand angiography, the diagnosis of right par-tially thrombosed giant carotid ophthalmicartery aneurysm was established.
Carotid ophthalmic artery aneurysms arisein the first 2 mm of the internal carotid arteryabove the cavernous sinus and below theorigin of the posterior communicating artery.The aneurysm frequently originates beneaththe optic nerve and may extend into and dilatethe optic canal.' Due to their dual componentnature, partially thrombosed giant aneurysmshave a characteristic appearance on spin echoMRI and identification of the signal intensitypattern is specific for this lesion.2 Our caseshowed all the characteristic features of apartially thrombosed giant aneurysm. A morecomplete dileneatide of the giant aneurysmcomponents and associated extra aneurysmalparenchymal abnormalities is possible withMRI compared with either CT or angiogra-phy.
RK GUPTAInstitute of Nuclear Medicine and
Allied Sciences, Lucknow Road, IndiaVS MEHTA*NK MISRAt
Department of Neurosurgery* andNeuroradiology,t All India Institute of Medical
Sciences,New Delhi, India
1 Peris JB, Ross Russell RW. Giant aneurysms ofthe carotid system presenting as visual fielddefect. J Neurol Neurosurg Psychiatry1980;43:1053-64.
2 Atlas SW, Grossman RI, Goldberg HI, et al.Partially thrombosed giant intracranialaneurysms: correlation ofMR and pathologicfindings. Radiology 1987;162:111-4.
Pituitary abscess with recurrent asepticmeningitis
Since the first description of pituitary abscessby Simmonds in 1914,' less than 40 cases havebeen adequately reported; The diagnosis hasrarely been made before operation, theradiological findings being those ofa pituitarytumour. We report a patient in whom thediagnosis of pituitary abscess was made pre-operatively, to draw attention to the associa-tion with aseptic lymphoplasmocytic menin-
925
gitis, which provided a clue to the diagnosis.This association has been observed only twicebefore.24
In 1983 a 67 year old woman developedsevere frontal headaches progressively wor-sening over several days. Her temperaturewas 38-5°C and she showed signs of menin-gism. The cerebrospinal fluid (CSF) wasxanthochromic with a lymphoplasmocyticpleiocytosis and increased protein content,but bacteriological and virological investiga-tions were negative, including tests for tuber-culosis. Computed tomography (CT) showedmoderate enlargement of the subarachnoidspaces. Viral meningitis was presumed andsymptoms disappeared gradually with symp-tomatic treatment. In a second episode amonth later, she lapsed into a coma for twodays. The laboratory findings were similar,treatment was not modified and she recoveredafter several weeks.One year later, headaches, slowed thinking
and meningism recurred. Her sedimentationrate was raised. She had anaemia, polymor-phonuclear leucopaenia and a relative lym-phocytosis. She had circulating immunecomplexes and antinuclear antibodies, buttests for anti DNA antibodies were negative.Tests of thyroid function showed that TSHwas low, with low free T3 but normal free T4.Her CSF had an increased protein content(12 mg/ml) and lymphoplasmocytosis (60lymphocytes and 15 plasmocytes/ml), butglucose and chloride concentrations werenormal. The CT was unchanged. She againrecovered without specific treatment.
Six months later, she still had a lym-phocytosis in her blood. Antinuclearantibodies and immune complexes were stillpresent. Her free T3 had fallen and she wastreated with 0-1 mg L-thyroxine daily. Dur-ing the following months, she experiencedseveral episodes of mental slowing, vertigo,orthostatic hypotension with falls and head-aches but without hyperthermia or signs ofmeningism. She was referred to the Depart-ment of Neurology, University of Liege, inFebruary 1987. She was moderately obeseand hypertensive with brittle hair and thineyebrows. There was no pubic hair and herpretibial skin was infiltrated. She had asubjective bilateral global diminution ofvision, with no apparent hemianopia, but therest of the neurological examination wasnormal.
Routine blood and urine tests were normal.Antinuclear antibodies were still positive(1/20), but anti DNA antibodies and circu-lating immune complexes were negative.Endocrine tests showed normal basal thyroidfunction (with 0-1 mg L-thyroxine daily)with undetectable TSH and evidence ofhypopituitarism. Injection test of TRH didnot increase TSH values. There was only aslight increase in prolactin values (from 110to 190 clIU/ml). Follicle-stimulating hor-mone (2 9 mIU/ml) and luteinising hormone(< 2-5 mIU/ml) were subnormal for a postmenopausal woman and Gn RH did notincrease FSH and LH values. Cortisol andACTH were below the minimal detectablevalues throughout the circadian cycle. Freeurinary cortisol was low at 3 pg/day. CRF testwas followed by no increase of ACTH orcortisol. Urinary gravimetry was normal.
Ophthalmological examination showedsenile bilateral cataract with severe reductionof visual acuity, but no defect in her visualfields. Plain radiographs of the skull showedan enlarged sella with thinning of the rightpart of the floor; abnormal calcification was
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Letters to the editor
...~~~~~~~..,., :,,
Figure 1 Sphenoidal mucosa exhibiting anextensive inflammatory infiltrate composedmainly by lympho-monocytic cells (HE, x100).
not observed. CT showed an enhancing 7-8mm mass in the right posterior part of thesella turcica. A pituitary abscess was suspec-ted and operation was performed.Transphenoidal exploration showed that
the mucous membrane of the sphenoid sinuswas thickened with necrotic zones in its rightposterior portion. The sellar floor was verythin, the capsule of the mass in the sella wasincised and yellow purulent material exuded.Cultures ofthe purulent material were sterile,but Staphylococcus epidermidis was found inthe mucosa of the sphenoid. Histologicalexamination of the tissue removed failed toshow evidence of an adenoma or othertumour tissue. The sphenoid mucous mem-brane was covered by a ciliated columnarepithelium with calciform cells. The underly-ing chorion was very oedematous, and inten-sely infiltrated by lympho-monocytic cells(fig 1). A fragment of the sella mass showedan organised conjunctivo-vascular tissuesurrounding a central cavity (fig 2), infiltratedby lympho-monocytic cells and polynuclears.Another fragment showed necrotic cells withpolynuclear cells, red cells and fibrin deposi-tion. The histopathological diagnosis wascompatible with a non-specific chronicsphenoidal sinusitis and an organisedpituitary abscess.
Antibiotics and corticosteroid replacementtherapy was started, she recovered rapidly;her headaches disappeared gradually and twoyears after surgery, there has been norecurrence of her symptoms.Domingue and Wilson reported seven
cases of pituitary abscess and reviewed the 50previously described cases,' since the firstdescription by Simmonds.' Only 29 of thesecases had adequate clinical details. Thirteenmore cases have been reported by severalinvestigators. Six of those 42 patients hadrecurrent episodes of purulent or asepticmeningitis."' Zorub et al drew attention tothe clinical clues that should lead to suspicionof the diagnosis before operation: meningealsigns, fever, blood and CSF leukocytosis.6Suspicion of the diagnosis before operationshould lead to appropriate antibiotic therapy
t. H S;.#'i35
Figure 2 Intrasellar fragment composed by acollagenous tissue more cellular in the outermembrane (upper part), large fibroblasts andoedematous spaces infiltrated by lympho-monocytic cells and polynuclears (inner core ofthe lesion) (HE, x 100).
and the use of a transphenoidal approach, toavoid contamination of the intracranial com-partment. The mortality rate of patients whodevelop meningitis is quite high (450,o).'Our patient had no associated adenoma or
tumour, as reported in several instances (10cases reviewed).6 No evidence for an infil-trative lesion (leukaemia, lymphoma, sar-coidosis, granuloma) was found. Thepurulent material was sterile, that was also thecase in 50"0 of previous reports. However,cultures for anaerobics were not usually per-formed. The pathogenesis of the abscess inour patient remains unclear. Chronic sphen-oidal sinusitis might have been responsible,but it is not easy to decide if the pituitaryabscess was primary or secondary to thepresence of sinusitis. Purulent sphenoidalsinusitis can spread to the sella, sometimeswith osteomyelitis, sometimes with thrombo-phlebitis of the cavernous sinus and menin-gitis. A pituitary abscess may also arise byseptic metastasis from a remote source ofinfection.This case illustrates that the diagnosis of
pituitary abscess should be suspected whenprogressive panhypopituitarism is associatedwith recurrent episodes of meningitis and aneroded or enlarged sella. We suggest thatskull radiographs should always be obtainedwhen meningitis is established, especially inrecurrent episodes.
DANIEL GUILLAUMEACHILLE STEVENAERT*
THIERRY GRISARPIERRE DOYEN
MICHEL REZNIKtDepartment of Neurology,
Department of Neurosurgery* andDepartment of Neuropathology,t
University of Liige, CHU, Liege, Belgium
Correspondence to: Dr Guillaume, Department ofNeurology, University of Liege, CHU B33, 4000LIEGE, Belgium.
1 Domingue JN, Wilson CB. Pituitary abscesses.Report of seven cases and review of theliterature. J Neurosurg 1977;46:601-8.
2 Emile J, Degos C, Szekely AM. Meningite aigueaseptique d liquide clair, avec rechutes,associee a un abces de la loge hypophysaire Apropos d'un cas. Rev Neurol 1969;121:189-95.
3 Lindholm J, Rasmussen P, Korsgaard 0.Intrasellar or pituitary abscess. J Neurosurg1973;38:616-9.
4 Rudwan PA. Pituitary abscess. Neuroradiology1977;12:243-8.
5 Simmonds M. Zur Pathologie der Hypophysis.Verh. Dtsch. Ges Pathol 1914;17:208-12.
6 Zorub DS, Martinez AJ, Nelson PB, Man-TaiL. Invasive pituitary adenoma with abscessformation: case report. Neurosurgery1979;5:718-22.
Transient global amnesia followingingestion of chloroquine
Transient global amnesia (TGA) is a tem-porary disturbance of short term memory ofunknown aetiology. Neither an epileptic' nora vascular, transient ischaemic cause havebeen confirmed. It has rarely been describedafter ingestion of clioquinol derivatives,2 andpossibly triazolam,' after trauma, withtumours of the temporal lobe associated withmigraine.'A 62 year old healthy business man was
planning to leave for Zaire on the day of hisadmission. He had used chloroquine on ajourney to Central America two yearspreviously, apparently without ill effect. Themorning of his departure, he took 300 mg ofchloroquine. Three hours later he felt ill. Hecould remember nothing about his travellingplans. He did not recall the name of thecolleague he was planning to travel with. Heforgot things which had happened onlyminutes before.On admission, he was oriented to time and
place. He could remember his birthday andsome events from the past. However, hisimmediate recall was considerably disturbed.This spontaneously subsided about threehours later. Routine biochemical andhaematological parameters were normal. Acerebral CT scan, cardiovascular examin-ation that included ECG and digital intraven-ous angiography ofthe extracranial portion ofcarotid and vertebral arteries, were normal.Peak values of chloroquine are reached
after three hours. Although TGA has neverbeen associated with this product, itschemical structure is related to clioquinol,which has been. The retinopathy andneuromyopathy of chloroquine are wellknown. Admittedly, these unwanted effectsoccur after ingestion of large doses for exten-ded periods. Although a chance associationcannot be excluded, we suspect that theingestion ofchloroquine in this man caused ortriggered the appearance of TGA.
PATRICK CRASJEAN-JACQUES MARTINLaboratory of Neuropathology,
Born-Bunge Foundation and Department ofNeurology,
University of Antwerp,Universiteitsplein 1, Wilrijk, Belgium
1 Cole AJ, Gloor P, Kaplan R. Transient globalamnesia: the electroencephalogram at onset.Ann Neurol 1987;22:771-2.
2 Mumenthaler M. Transient global amnesia afterclioquinol: five personal observations fromoutside Japan. J Neurol Neurosurg Psychiatry1979;42: 1084-90.
3 Morris HH and Estes ML. Traveler's amnesia.Transient global amnesia secondary totriazolam. JAMA 1987;258:945-6.
4 Miller JW, Petersen RC, Metter EJ, MillikanCH, Yanagihara T. Transient global amnesia:clinical characteristics and prognosis.Neurology (Minneapolis) 1987;37:733-7.
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