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Neurology India, Vol. 54, No. 4, October-December, 2006, pp. 421-424 Case Report Multiple simultaneous intracerebral hemorrhages following accidental massive lumbar cerebrospinal fluid drainage: Case report and literature review Ruiz-Sandoval José L, Campos Ariel, Romero-Vargas Samuel, Jiménez-Rodríguez María I, Chiquete Erwin Department of Neurology and Neurosurgery, Hospital Civil de Guadalajara Fray Antonio Alcalde and the Department of Neurosciences; Centro Universitario de Ciencias de la Salud. Guadalajara, Jalisco Code Number: ni06144 Abstract Multiple simultaneous intracerebral hemorrhages (ICH) are uncommon. We report the case of an 80-year-old woman with previous diagnosis of normal pressure hydrocephalus and who was brought to our hospital with altered mental status and urinary incontinence. Medical history of hypertension, hematological disorders or severe head trauma was absent. Platelet count and coagulation profile were unremarkable. An initial head computed tomography (CT) showed sulcal enlargement and ventricular dilatation, but no evidence of ICH. A tap test indicated as a guide to case selection for shunt surgery accidentally resulted in cerebrospinal fluid (CSF) overdrainage. The patient presented sudden neurological deterioration, with sluggishly responsive pupils and generalized tonic-clonic seizures. A new head CT demonstrated multiple supra and infratentorial ICH. The patient became comatose and had a fatal course. Hence, CSF overdrainage may either cause or precipitate multiple simultaneous ICHs, affecting both the infratentorial and supratentorial regions.Keywords: Cerebrospinal fluid, intracranial hemorrhage, intracranial hypotension, lumbar drainage, neurological examination Introduction Lumbar cerebrospinal fluid (CSF) drainage has several diagnostic and therapeutic indications, with well documented hazardous consequences including overdrainage, acute pneumocephalus, brain collapse and neurological deterioration.[1],[2],[3] Intracerebral hemorrhage (ICH) has been reported after lumbar puncture and lumboperitoneal shunts, sometimes related to other conditions.[4],[5],[6] We report the case of a woman in whom CSF overdrainage resulted in multifocal ICH. To the best of our knowledge, this is the first report on massive CSF drainage as a cause of multiple simultaneous ICHs.Case Report An 80-year-old woman was brought to our hospital with altered mental status and urinary incontinence, as her main complaints. The history revealed that in the previous two months she suffered from cognitive impairment and gait disturbance, which motivated her caregivers to seek medical attention in another hospital. After clinical evaluation and a head computed tomography (CT) scan, she was given a diagnosis of normal pressure hydrocephalus. She was not on anticoagulation or antiplatelet therapy. Medical history of hypertension, hematological disorders or severe head trauma was absent. The neurological examination at presentation to our hospital revealed a conscious woman with spatial disorientation and bilateral hyperreflexia. Focal neurological signs were absent. Laboratory findings were normal, including platelet count (152 x 10 9/liter) and coagulation profile (PT: 90% of control, APTT: 27 seconds, fibrinogen: 225 mg/dl). Blood pressure was below 130/90 mmHg during her hospital stay. A head CT scan performed in our center showed ventriculomegaly, sulcal enlargement and diffuse white matter disease, with chronic bilateral subcortical infarctions [Figure - 1]. No evidence of ICH was found; nevertheless, a laminar collection of blood in the posterior interhemispheric fissure was observed, suggestive of being secondary to previous head trauma for which we had no knowledge on history-taking. In spite of this finding, a tap test was indicated as a guide to case selection for shunt surgery, since no mass effect was observed. The procedure was performed by a physician in training without supervision. Cerebrospinal fluid was clear, with opening pressure of 150 mmH 2 O. The catheter was not withdrawn on time and CSF continued to flow for almost 30 min, until the fluid initiated to drain bloody, with a final CSF collection of 250 mL, as measured in a graduated flask. After the procedure the patient presented sudden neurological deterioration, with pupils sluggishly reacting to light and generalized tonic-clonic seizures. A new head CT was practiced 18h after CSF overdrainage, showing multiple infra and supratentorial ICHs with irruption into the ventricular system [Figure - 2]. The patient became comatose, requiring ventilatory assistance. Replacement of CSF volume could not be practiced. Two days later the patient developed pneumonia, which resulted in sepsis and death in one week more.Discussion Multiple simultaneous ICHs is defined as the presence of two or more intracerebral hemorrhages affecting different arterial territories, without continuity between them and with identical CT density profiles.[7],[8] This is a rare presentation of the hemorrhagic cerebrovascular disease, accounting for 0.6 to 2.8% of the cases of nontraumatic, nonaneurysmal ICH.[7],[8] The main causative factors are hypertension, cerebral amyloid angiopathy and forms of vasculitis, among other conditions [Table - 1]. There is a strong preponderance for the supratentorial space, especially affecting the basal ganglia (thus denouncing the hypertensive nature seen in most cases).[8] However, most of the knowledge regarding multiple simultaneous ICHs is derived from case reports, which are possibly the type of communications subject to the strongest reporting bias. Therefore, the clinical picture, outcome and even the putative causes may vary more than is reflected in case reports. Since most of the causative factors previously attributed to multiple simultaneous ICHs were excluded in the case presented here and given that neurological deterioration as well as the hemorrhagic findings in the second head CT began immediately after CSF overdrainage, it seems reasonable to think that this procedure was the cause or at least, a precipitating factor of multifocal ICH. To our knowledge, this patient had a cause of multiple simultaneous ICHs not previously reported [Table - 1]. In the present case, the putative pathophysiological mechanism that led to multiple simultaneous ICHs points to a continuous and massive lumbar CSF evacuation resulting in a reduction of CSF volume with the associated lowering in intraspinal and intracranial pressure, which eventually increased the transmural pressure gradient of the vessels, leading to a secondary wall stress rupture.[1] Advanced age and the presence of diffuse white matter disease could be the other important contributing factors.[9] The widespread and prolonged degeneration of the intracerebral arterioles in older people may also predispose to the development of multiple ICHs. Unfortunately, amyloid angiopathy or other age-related cerebrovascular conditions were not completely excluded in our patient because no cerebral biopsy was performed. Moreover, we were not able to obtain a necropsy. Since amyloid angiopathy is very common in older people and is also an important cause of multiple simultaneous ICHs [Table - 1], our patient might have had an underlying susceptibility (e.g., amyloid angiopathy) of presenting ICH, which in turn was precipitated by CSF overdrainage. Nevertheless, the association of CSF overdrainage with ICH in this patient seems clear, either as an independent causative or precipitating factor. Indeed, the laminar collection of blood over the left parietal convexity and the posterior interhemispheric space seen in the head CT performed at presentation to our hospital [Figure - 1] need comments. We were not told about the antecedent of head trauma that might explain this abnormality; however, considering the gait instability that the patient was presenting, falls that might have caused mild head trauma cannot be discarded. Nevertheless, even though delayed traumatic ICH exists,[10],[11] it is mainly associated with severe head trauma and would hardly cause more than two ICHs affecting both the infratentorial and supratentorial regions. When a tap test is indicated, intermittent lumbar or continuous CSF drainage at controlled rate are safe strategies in avoiding overdrainage,[1],[2] especially because the lower threshold of CSF volume compatible with life in humans is rather unknown.[2] In our patient, an advanced age, sulcal enlargement and ventricular dilatation allowing a large CSF volume might have permitted such drainage of the fluid (250 mL in 30 min). Any time lumbar CSF drainage is indicated as diagnostic procedure, it is necessary to be warned about an excessive rate of CSF drainage, which should not exceed 20-25 mL/h.[12] When used as a guide to case selection for a shunting procedure in normal pressure hydrocephalus[2] or as treatment of CSF fistula,[12] lumbar CSF drainage of 40 to 50 mL per session is considered safe and effective.[1],[2] Another concern with respect to the case discussed here is the medical error that led to this catastrophe. This complication has the possibility to be repeated, especially in teaching hospitals in which physicians in training perform without expert supervision. Appropriate measures were taken in our center to avoid another accident like this. Excessive work must not be an exception of a tight supervision to junior doctors. In conclusion, CSF overdrainage can either cause or precipitate multiple simultaneous ICHs, affecting both the infratentorial and supratentorial regions. References
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