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Apr 19, 2011 - Choroid plexus papilloma (CPP), a neoplasm of choroid plexus in the cerebral ventricles, is usually accompanied by hydrocephalus.
Childs Nerv Syst (2011) 27:1153–1156 DOI 10.1007/s00381-011-1459-0

CASE REPORT

Catastrophic electrolyte imbalance caused by excessive production and overdrainage of cerebrospinal fluid in an infant with choroid plexus papilloma Ji Hoon Phi & Choong Ho Shin & Kyu-Chang Wang & Sung-Hye Park & Seung-Ki Kim

Received: 11 March 2011 / Accepted: 7 April 2011 / Published online: 19 April 2011 # Springer-Verlag 2011

Introduction

Case report

Choroid plexus papilloma (CPP), a neoplasm of choroid plexus in the cerebral ventricles, is usually accompanied by hydrocephalus. Obstruction of cerebrospinal fluid (CSF) pathways by the neoplasm may contribute to the development of hydrocephalus, but overproduction of CSF from the tumor is also a major factor in its development [1, 2]. In fact, overproduction of CSF is a unique phenomenon of tumors and of choroid plexus hyperplasia [3]. The chemical and osmotic composition of CSF is different from that of plasma. The sodium content of CSF is nearly identical to that of plasma, but the level of potassium in CSF is less than the level in plasma [4]. Infusions of hypotonic saline with physiologic potassium supplements are commonly used to maintain volume and electrolyte homeostasis in infants and children. If overlooked, this common practice can lead to a severe electrolyte imbalance in patients with CPP when excessive CSF is drained for a short period.

A 7-month-old male baby was brought to the emergency room because of vomiting and lethargy that had persisted for a week. The baby's medical history was otherwise unremarkable. On physical examination, the baby was lethargic but responsive to verbal and visual stimuli. The baby weighed 9.9 kg and had a head circumference of 50 cm. Cranial sutures were separated, and the anterior fontanel was wide and bulging. Engorgement of scalp veins was observed. Limb muscle tone was reduced, but no definite weakness was found. Serum sodium and potassium levels were 133 and 5.0 mmol/L, respectively. A computed tomography scan showed a huge mass in the third ventricle and severe hydrocephalus. Magnetic resonance imaging also showed a well-defined, enhancing mass with a cauliflower shape and internal papillary structures (Fig. 1). The child was diagnosed with choroid plexus tumor and accompanying hydrocephalus. Emergent ventriculostomy was performed to reduce intracranial pressure (ICP). The initial ICP was measured as 24 mmHg during the procedure. After ventriculostomy, the patient was hemodynamically stable. He was alert without focal neurological deficits. The amount of CSF drainage was large considering the age and body weight of the patient. During the 24 h following ventriculostomy, a total of 1,176 mL of CSF (average 49 mL/h) was drained. The patient was fed on breast milk and was administered supplementary fluid via an intravenous line at a rate of 30 mL/h. The supplementary fluid was hypotonic saline containing 5% dextrose, Na+ 34.2 mmol/L (0.2% saline), K+ 20.1 mmol/L, and Cl− 54.3 mmol/L. The patient's input and output were unbalanced to volume loss, but he could feed on breast milk every 2~3 h. Twenty-four hours after the ventriculostomy, the patient's level of consciousness deteriorated rapidly, and the patient

J. H. Phi : K.-C. Wang : S.-K. Kim (*) Division of Pediatric Neurosurgery, Seoul National University Children’s Hospital, 101 Daehangno, Jongno-gu, 110-744, Seoul, Republic of Korea e-mail: [email protected] C. H. Shin Department of Pediatrics, Seoul National University Children’s Hospital, Seoul, Republic of Korea S.-H. Park Department of Pathology, Seoul National University Children’s Hospital, Seoul, Republic of Korea

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Fig. 2 a An intraoperative photograph shows the gross papillary contour of the tumor, mimicking a normal choroid plexus. b A microscopic photograph of a pathological specimen also shows tumor cells arranged in papillary structures

Fig. 1 Axial (a) and sagittal (b) magnetic resonance images of the tumor. A well-defined, enhancing mass is located in the third ventricle. The mass has a typical cauliflower shape and internal papillary structures

developed ventricular ectopic beats that progressed into ventricular tachycardia and hemodynamic collapse. A brief seizure was observed. Serum laboratory exam at the time of hemodynamic collapse revealed severe hyponatremia and hyperkalemia (Na+ 116 mmol/L, K+ 7.6 mmol/L, and Cl− 93 mmol/L). The electrolyte profile of CSF at that time was as follows: Na+ 128 mmol/L, K+ 2.7 mmol/L, and Cl− 98 mmol/L (Table 1). Emergent intubation and cardioversion Table 1 Comparison of the serum, urine, and CSF electrolyte profiles of the patient at the time of catastrophe

The right column is the normal ranges for serum electrolytes of our institution

Na+ K+ Cl− Total CO2

were performed, and the patient regained a sinus rhythm and normal hemodynamic profiles. Sodium bicarbonate and calcium were administered to ameliorate hyperkalemia and its adverse effects. The main fluid was changed to normal saline (Na+ 154 mmol/L, 0.9% saline) without a potassium supplement. Hypertonic saline (Na+ 500 mmol/L, 3% saline) was added to the fluid supply to counteract hyponatremia. With gradual normalization of hyponatremia, hyperkalemia, and volume loss, the patient fully regained consciousness. No further seizure or focal neurological deficits were found. The tumor removal was delayed for 3 days. The tumor was approached via an interhemispheric transcallosal transforaminal route. During the operation, a reddish hypervascular tumor with a gross papillary structure was observed (Fig. 2). The tumor was completely removed, and pathological examination revealed it to be a CPP. Two weeks after the operation, the patient developed symptomatic subdural

Serum (mmol/L)

Urine (mmol/L)

CSF (mmol/L)

116 7.6 93 3

19 103.5 70 –

129 2.7 98 24

Normal ranges for serum (mmol/L) 135–145 3.5–5.5 98–110 24–31

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hygroma that required a subduroperitoneal shunt. The patient was discharged without focal neurological deficits.

of the tumor itself also helps prevent electrolyte imbalance due to CSF overdrainage.

Discussion

Conclusion

Overproduction of CSF causing hydrocephalus is a wellknown phenomenon of CPP, although obstruction of CSF pathways may also cause hydrocephalus in some patients [5–7]. In 1974, Eisenberg et al. [2] calculated the rate of CSF formation to be 1.4 mL/min, or more than 2 L/day, in a 5-month-old baby with CPP and hydrocephalus using a ventricular perfusion technique. Considering the role of normal choroid plexus in CSF production and the large size of CPP, CSF production rates many times greater than normal are not uncommon with CPP [8]. For instance, CSF production of 5 L/day has been reported for a patient with bilateral CPPs [3]. Infants are vulnerable to water and electrolyte imbalances. Even a day of starvation or repeated vomiting can cause severe water and electrolyte imbalance in this population. Relative to their body weight, infants have a larger body surface area than adults and require a greater volume of maintenance fluid for their body weight. To date, hypotonic saline (25~75 mmol/L of sodium and 5% dextrose) with a physiologic potassium supplement (20 mmol/L) has been widely used as a maintenance fluid in pediatrics [9–12]. However, many recent studies criticized this tradition of prescribing hypotonic saline for sick children because of the risk of developing iatrogenic hyponatremia which is often fatal [13–15]. Instead, the use of isotonic saline as a maintenance fluid has been advocated [12, 16]. Hypotonic saline with a potassium supplement may be especially inappropriate in situations in which the patient loses fluid with a different ionic composition to that of extracelluar fluid (ECF). CPP is most commonly found in infants and young children [17]. Preoperative drainage of excessive CSF is frequently required to control intracranial pressure in patients with CPP [18]. CSF is an isotonic fluid with an approximate sodium concentration of 138 mmol/L, but the potassium content of CSF (2.8 mmol/L) is less than that of ECF (4.5 mmol/L) [4]. This was also true for the patient discussed in this report (see Table 1). The total body water and plasma volumes, respectively, are about 70% and 6% of body weight for infants, which correspond to about 7 and 0.6 L for our patient [19]. Therefore, loss of a large amount of CSF (e.g., more than 1 L/day) is a serious condition and may elicit a catastrophic electrolyte imbalance if combined with infusion of hypotonic saline with a physiologic potassium supplement. To prevent this catastrophe, it is imperative to monitor serum electrolyte levels closely during drainage of a large amount of CSF, and it is recommended to use isotonic saline with minimal potassium supplement. Early surgical removal

Excessive drainage of CSF combined with infusion of hypotonic saline with a physiologic potassium supplement can cause a catastrophic electrolyte imbalance in small infants with choroid plexus papilloma. Acknowledgment This study was supported by a grant from the National R&D Program for Cancer Control, Ministry of Health and Welfare, Republic of Korea (Grant no. 0520300; to Seung-Ki Kim). Financial disclosure The authors report no financial disclosure.

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