- Email: [email protected]
Neonatal Hyponatremia in the Setting of Untreated Progressive Hydrocephalus
Case Report
Neonatal Hyponatremia in the Setting of Untreated Progressive Hydrocephalus Megan Obi1 and Kaine Onwuzulike2
Key words Hydrocephalus - Hyponatremia - Neonatal -
Abbreviations and Acronyms CSF: Cerebrospinal fluid EVD: External ventricular drain HC: Head circumference IVH: Intraventricular hemorrhage Na: Sodium NPH: Normal pressure hydrocephalus SIADH: Syndrome of inappropriate secretion of antidiuretic hormone VP: Ventriculoperitoneal From the 1Case Western Reserve School of Medicine, Cleveland, Ohio; and 2Department of Pediatric Neurosurgery, Cleveland Clinic Foundation, Cleveland, Ohio, USA To whom correspondence should be addressed: Kaine Onwuzulike, M.D., Ph.D. [E-mail: [email protected]] Citation: World Neurosurg. (2019) 125:19-22. https://doi.org/10.1016/j.wneu.2019.01.124 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
INTRODUCTION
- BACKGROUND:
Hyponatremia in the neonatal population is an uncommon occurrence and can be associated with significant increase in morbidity and mortality. In the neonatal population, it is typically associated with an excess of antidiuretic hormone and rarely has been found to be associated with hydrocephalus, short of being caused by the subsequent treatment of hydrocephalus.
- CASE
DESCRIPTION: We present a case report of a patient with neonatal hydrocephalus, secondary to intraventricular hemorrhage in the setting of prematurity, treated at our institution, in whom sodium levels reached a nadir as head circumference peaked and subsequent treatment of hydrocephalus resolved the associated hyponatremia.
- CONCLUSIONS:
Hydrocephalus secondary to intraventricular hemorrhage is a notable complication in the premature neonatal population. Physicians should be aware of its potential association with hyponatremia and consider early neurosurgical intervention when other etiologies of the electrolyte disturbance cannot be identified.
subsequently normalized on treatment of the underlying hydrocephalus. CASE DESCRIPTION The female patient is born via spontaneous vaginal delivery at 34 1/7 weeks to a 32 year old gravida 9 para 9 mother with minimal prenatal care. Postnatal course was complicated by respiratory distress and bilateral
Hyponatremia in the neonatal population, especially in low birthweight infants, is associated with significant long-term complications such as longer hospital length of stay, poor growth, and developmental delays. Subsequent delays in diagnosis and treatment can result in increased mortality.1-7 Neurologic symptoms associated with persistent hyponatremia include vomiting, weakness, apathy, and seizures.8 Neonatal hyponatremia is uncommon, but is typically associated with an excess of antidiuretic hormone for which etiologies for this phenomenon are still not well described in literature.9 The association with hydrocephalus is even more uncommon, and has rarely been reported. Here, we describe the case of a neonatal patient with untreated neonatal posthemorrhagic hydrocephalus and an associated worsening hyponatremia that
WORLD NEUROSURGERY 125: 19-22, MAY 2019
pleural effusions requiring intubation, mechanical ventilation, and chest tube placement. Nutrition was maintained on parenteral nutrition with Enfaport (Mead Johnson & Company, LLC, Chicago, Illinois, USA) and supplementation with injectable lipid emulsion and dextrose. Head circumference (HC) and birthweight was 32 cm (79.56 percentile) and 2.39 kg (70.68 percentile), respectively. Neurology, and
Figure 1. Second day of life initial head ultrasound of the coronal plane showing the lateral ventricle with significant intraventricular hemorrhage.
www.journals.elsevier.com/world-neurosurgery
19
CASE REPORT MEGAN OBI AND KAINE ONWUZULIKE
NEONATAL HYPONATREMIA DUE TO HYDROCEPHALUS
Figure 2. Day 20 of life head ultrasound of the coronal plane showing the lateral and third ventricle with significant ventriculomegaly and resolving hemorrhage with peak head circumference.
subsequently neurosurgery, were consulted for expanding germinal matrix hemorrhage with initial head ultrasound on day 2 of life, demonstrating bilateral grade II intraventricular hemorrhage (IVH) (Figure 1) that subsequently progressed to grade III IVH on repeat head ultrasound the following day. Neurologic status remained intact and HC stable while patient’s respiratory problems were actively managed. On day 19 of life, HC was noted to be persistently elevated at 33.5 cm with tense, bulging fontanelles, mild hypertonia, and worsening anasarca with scalp pitting
edema. Creatinine was 0.26 mg/dL, previously ranging from 0.73e0.21 mg/dL since birth. On day 20 of life, HC had increased to 35 cm, brain magnetic resonance imaging demonstrated communicating hydrocephalus with worsening resistive indices, decreased diastolic flow in the anterior carotid arteries, and dilation of the ventricular system (Figure 2). Serum sodium (Na) levels at the time were 121 mmol/L (previously ranging from 136e152 mmol/ L), requiring correction with half intravenous 3% Na. Day 21 of life, HC
Figure 3. Postoperative 33rd day of life head ultrasound of the coronal plane showing the lateral and third ventricles with reduction of ventriculomegaly after external ventricular drain placement.
20
www.SCIENCEDIRECT.com
remained stable at 35 cm, however Na level continued to decline with a nadir of 116 mmol/L. Creatinine levels remained stable at 0.22 mg/dL. The patient continued to demonstrate mild sun-setting and hypotonicity, but remained arousable without any pathologic movements, abnormal pupil dilation, or significant gastrointestinal symptoms. A right external ventricular drain (EVD) was placed with removal of about 10e 15 cc/kg of cerebrospinal fluid (CSF) daily and Na correction attempted with full intravenous 3% Na. Postoperatively, on day 22 of life, the patient demonstrated flat and soft anterior fontanelle, HC was down 2 cm, and she became normonatremic at 140 mmol/L (Figure 3). Electrolytes continued to remain stable, creatinine levels persisted at 0.25 mg/dL, and EVD was replaced with a ventriculoperitoneal (VP) shunt on day 29 of life (Figure 4). To date, the patient has not required any additional neurosurgical procedures, has demonstrated no clinical signs of elevated intracranial pressure, electrolytes have continued to remain stable, and shunt has remained patent (Figure 5). DISCUSSION Germinal matrix IVH is one of the most commonly diagnosed brain disorders in preterm infants, with an incidence of about 15%-20% in very-low-birthweight infants, describing an inverse relationship between IVH incidence and gestational age.10,11 The Papile grading system is a widely used scale to describe IVH severity, with a grading scale of I-IV.12 IVH subsequently serves as the most common cause of acquired hydrocephalus in infants, and is thought to be due to subsequent fibrosis, arachnoid granulations, and deposition of proteins in perivascular spaces to inhibit CSF resorption.11,13 Clinical presentation of posthemorrhagic hydrocephalus in infants can include increasing orbitofrontal HC, fontanelle fullness, and splaying of sutures as well as signs of increased ICP (i.e., apnea, lethargy, decreased activity, and bradycardia).11 To date, multiple studies have demonstrated an association between hyponatremia and hydrocephalus in patients. However, to the best of our knowledge, those studies have predominantly focused on adult populations, as well as normal pressure hydrocephalus (NPH) or
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.01.124
CASE REPORT MEGAN OBI AND KAINE ONWUZULIKE
NEONATAL HYPONATREMIA DUE TO HYDROCEPHALUS
mmol/L
160 Bolus of Hypertonic Saline 140
120
117 mmol/L
External Ventricular Drain Placed
100
91.22%
Na (mmol/L)
80
PercenƟle
Head Circumference (%) * 60
40
20
0 0
5
10
15
20
25
30
35
40
Day of Life
Figure 4. Graphic representation of the progression of sodium and the corresponding head circumference over the course of the patient’s initial days of life. Depicted is the point at which head circumference percentile peaked and sodium concentration was at its nadir. *Percentiles based off the Fenton growth chart for premature girls. Na, sodium.
obstructive etiologies of hydrocephalus.2,14-16 Additionally, hyponatremia in previous studies has been found to be a result of treatment of hydrocephalus rather than as a consequence of worsening hydrocephalus.17-21 Chou et al.2 established an understanding of the prevalence of this association and determined that hyponatremia in NPH patients was clinically more common than had previously been reported, and that those at risk included hypertensives and those with nasogastric tubes during hospitalization. In addition, previous reports have found a
correlation between NPH and syndrome of inappropriate secretion of antidiuretic hormone (SIADH) as well as polydipsia in patients.22 Studies that have involved the pediatric population have demonstrated a correlation between hyponatremia as a subsequent complication of frequent drainage of CSF, but not as a direct consequence of hydrocephalus.17,19 Tenbrock et al.,20 for example, discovered an association between use of a ventricular access device (specifically the Rickham reservoir) for serial liquor punctures in preterm infants with IVH, and
Figure 5. T2-weighted magnetic resonance imaging scans of the axial plane showing the lateral (A) and third ventricles (B) at 1-year follow-up demonstrating a reduction in ventricular size.
WORLD NEUROSURGERY 125: 19-22, MAY 2019
the subsequent development of hyponatremia with improvement after adequate fluid resuscitation. Our patient thus serves as a unique case of hydrocephalus associated hyponatremia in that her serum Na levels seemed to reach a nadir as her hydrocephalus worsened, with near immediate postsurgical resolution of hyponatremia. Although it may be difficult to distinguish whether the improvement in Na was entirely due to surgical correction with the EVD or hypertonic saline administration, it should be noted that the Na continued to decline despite the initial 3% bolus and only began to correct after EVD placement. In addition, although previous cases have suggested a potential renal etiology of SIADH contributing to hyponatremia, our patient’s creatinine levels remained low-normal and were unlikely related to the hyponatremia. Moreover, the patient was adequately receiving appropriate weight-based maintenance fluids, had no gastrointestinal problems, and was on no medications with a known side effect of hyponatremia, ruling out other potential hypovolemic and euvolemic causes of hyponatremia. Only 2 other studies known to us demonstrate improvement of hyponatremia with treatment of hydrocephalus with VP shunt, but those cases described elderly individuals with NPH.14,16 Although this case establishes precedence of the phenomenon, our patient describes a new population as well as etiology. No definitive mechanism has been determined for the association between hydrocephalus and hyponatremia. It is our hypothesis that elevated third ventricle pressure induces mechanical compression on the hypothalamus resulting in disturbances in endocrinology, one being hyponatremia. Wise et al.23 found that blockage of VP shunts resulted in hyponatremia, and subsequently resolved when the blockage was removed and the third ventricle size reduced, and Yoshino et al.16 used magnetic resonance imaging to detect third ventricle enlargement in patients with hyponatremia, both serving as additional evidence to the proposed theory. Additionally, studies observing SIADH in patients with Shy-Dragger and myotonic dystrophy found that patients additionally had enlarged third ventricles as well as atrophied hypothalamuses.24,25 Our patient did not have a significant number of additional studies performed to
www.journals.elsevier.com/world-neurosurgery
21
CASE REPORT MEGAN OBI AND KAINE ONWUZULIKE
NEONATAL HYPONATREMIA DUE TO HYDROCEPHALUS
pinpoint the pathophysiologic mechanism for her symptoms, but given our intervention, a reasonable correlation can be drawn between her worsening hydrocephalus and hyponatremia. However, to the best of our knowledge, there are currently no additional neonatal reports to note third ventricular enlargement as a cause of hyponatremia in the setting of no other internal or external causes. Thus, further research still needs to be performed to better understand this phenomenon and potentially confirm our proposed hypothesis.
5. Marcialis MA, Dessi A, Pintus MC, Irmesi R, Fanos V. Neonatal hyponatremia: differential diagnosis and treatment. J Matern Fetal Neonatal Med. 2011;24(suppl 1):75-79.
CONCLUSIONS
9. Zieg J. Evaluation and management of hyponatraemia in children. Acta Paediatr. 2014;103: 1027-1034.
Our aim in presenting this case was to describe a rare and surgically reversible cause of hyponatremia. For patients, especially preterm infants, with IVH and other concurrent medical complications in whom hyponatremia may have various causes, it is pertinent to consider hydrocephalus, notably associated with third ventricular enlargement, as a formidable contribution to hyponatremia, for which ventricular decompression may offer resolution. REFERENCES 1. Brunsvig PF, Os I, Frederichsen P. [Hyponatremia. A retrospective study of occurrence, etiology and mortality]. Tidsskr Nor Laegeforen. 1990;110: 2367-2369.
6. Rafael Valdivia L, Ferrandiz Quiroz J. [Hyponatremia as a possible mortality factor in cirrhotic patients hospitalised in the Guillermo Almenara Irigoyen State Hospital, 2003-2005]. Rev Gastroenterol Peru. 2007;27:37-46. 7. Reyes Dominguez A, Briones Perez de la Blanca E. [Intrahospital mortality and hyponatremia]. An Med Interna. 1996;13:616-617. 8. Greenberg JH, Tufro A, Marsenic O. Approach to the treatment of the infant with hyponatremia. Am J Kidney Dis. 2015;65:513-517.
10. Ahn SY, Shim SY, Sung IK. Intraventricular hemorrhage and post hemorrhagic hydrocephalus among very-low-birth-weight infants in Korea. J Korean Med Sci. 2015;30(suppl 1):S52-58. 11. Robinson S. Neonatal posthemorrhagic hydrocephalus from prematurity: pathophysiology and current treatment concepts. J Neurosurg Pediatr. 2012;9:242-258. 12. Papile LA, Burstein J, Burstein R, Koffler H. Incidence and evolution of subependymal and intraventricular hemorrhage: a study of infants with birth weights less than 1,500 gm. J Pediatr. 1978;92:529-534. 13. Tully HM, Dobyns WB. Infantile hydrocephalus: a review of epidemiology, classification and causes. Eur J Med Genet. 2014;57:359-368.
2. Chou CY, Liu JH, Wang SM, et al. Hyponatraemia in patients with normal pressure hydrocephalus. Int J Clin Pract. 2009;63:457-461.
14. Jung KH, Chu K, Jeong SW, Hong YH, Park KI, Roh JK. Idiopathic normal pressure hydrocephalus predominantly with prolonged fever and hyponatremia. Neurology. 2003;61:554-556.
3. Garcia Segura A, Gadea Ruiz C, Oliva Fanlo B, et al. [Hyponatremia upon admission in patients over 65 years of age. Relation with medium length of stay and hospital mortality]. An Med Interna. 1994;11:487-489.
15. Kim JH, Kang JK, Lee SA. Hydrocephalus and hyponatremia as the presenting manifestations of primary CNS lymphoma. Eur Neurol. 2006;55:39-41.
4. Goldberg A, Hammerman H, Petcherski S, et al. Hyponatremia and long-term mortality in survivors of acute ST-elevation myocardial infarction. Arch Intern Med. 2006;166:781-786.
22
www.SCIENCEDIRECT.com
16. Yoshino M, Yoshimi Y, Taniguchi M, Nakamura S, Ikeda T. Syndrome of inappropriate secretion of antidiuretic hormone associated with idiopathic normal pressure hydrocephalus. Intern Med. 1999; 38:290-292.
17. Caglar MK, Deniz FE, Ozer I, Sezer T, Senayli A. An unusual cause of hyponatremia: ventricular drainage. Neurol India. 2006;54:109. 18. Lang SS, Bauman JA, Aversano MW, et al. Hyponatremia following endoscopic third ventriculostomy: a report of 5 cases and analysis of risk factors. J Neurosurg Pediatr. 2012;10:39-43. 19. MacMahon P, Cooke RW. Hyponatraemia caused by repeated cerebrospinal fluid drainage in post haemorrhagic hydrocephalus. Arch Dis Child. 1983; 58:385-386. 20. Tenbrock K, Kribs A, Roth B, Speder B. Hyponatraemia as a consequence of serial liquor punctures in preterm infants with a ventricular access device after posthaemorrhagic hydrocephalus. Arch Dis Child Fetal Neonatal Ed. 2003;88:F351. 21. Vaicys C, Fried A. Transient hyponatriemia complicated by seizures after endoscopic third ventriculostomy. Minim Invasive Neurosurg. 2000;43: 190-191. 22. Peterson DT, Marshall WH. Letter: Polydipsia and inappropriate secretion of antidiuretic hormone associated with hydrocephalus. Ann Intern Med. 1975;83:675-676. 23. Wise BL. Inappropriate secretion of ADH caused by obstruction of ventriculoatrial shunts. J Neurosurg. 1968;28:429-433. 24. Nagumo Furumoto drome of hormone. 177-181.
K, Kita K, Kitano K, Simoe Y, H. Shy-Drager syndrome and the syninappropriate secretion of antidiuretic Rinsho Shinkeigaku (Clin Neurol). 1992;32:
25. Tamaoka A, Nishimura Y. A case of myotonic dystrophy associated with SIADH. Rinsho Shinkeigaku (Clin Neurol). 1986;26:1146-1151.
Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 12 December 2018; accepted 14 January 2019 Citation: World Neurosurg. (2019) 125:19-22. https://doi.org/10.1016/j.wneu.2019.01.124 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.01.124
Neonatal Hyponatremia in the Setting of Untreated Progressive Hydrocephalus Megan Obi1 and Kaine Onwuzulike2
Key words Hydrocephalus - Hyponatremia - Neonatal -
Abbreviations and Acronyms CSF: Cerebrospinal fluid EVD: External ventricular drain HC: Head circumference IVH: Intraventricular hemorrhage Na: Sodium NPH: Normal pressure hydrocephalus SIADH: Syndrome of inappropriate secretion of antidiuretic hormone VP: Ventriculoperitoneal From the 1Case Western Reserve School of Medicine, Cleveland, Ohio; and 2Department of Pediatric Neurosurgery, Cleveland Clinic Foundation, Cleveland, Ohio, USA To whom correspondence should be addressed: Kaine Onwuzulike, M.D., Ph.D. [E-mail: [email protected]] Citation: World Neurosurg. (2019) 125:19-22. https://doi.org/10.1016/j.wneu.2019.01.124 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
INTRODUCTION
- BACKGROUND:
Hyponatremia in the neonatal population is an uncommon occurrence and can be associated with significant increase in morbidity and mortality. In the neonatal population, it is typically associated with an excess of antidiuretic hormone and rarely has been found to be associated with hydrocephalus, short of being caused by the subsequent treatment of hydrocephalus.
- CASE
DESCRIPTION: We present a case report of a patient with neonatal hydrocephalus, secondary to intraventricular hemorrhage in the setting of prematurity, treated at our institution, in whom sodium levels reached a nadir as head circumference peaked and subsequent treatment of hydrocephalus resolved the associated hyponatremia.
- CONCLUSIONS:
Hydrocephalus secondary to intraventricular hemorrhage is a notable complication in the premature neonatal population. Physicians should be aware of its potential association with hyponatremia and consider early neurosurgical intervention when other etiologies of the electrolyte disturbance cannot be identified.
subsequently normalized on treatment of the underlying hydrocephalus. CASE DESCRIPTION The female patient is born via spontaneous vaginal delivery at 34 1/7 weeks to a 32 year old gravida 9 para 9 mother with minimal prenatal care. Postnatal course was complicated by respiratory distress and bilateral
Hyponatremia in the neonatal population, especially in low birthweight infants, is associated with significant long-term complications such as longer hospital length of stay, poor growth, and developmental delays. Subsequent delays in diagnosis and treatment can result in increased mortality.1-7 Neurologic symptoms associated with persistent hyponatremia include vomiting, weakness, apathy, and seizures.8 Neonatal hyponatremia is uncommon, but is typically associated with an excess of antidiuretic hormone for which etiologies for this phenomenon are still not well described in literature.9 The association with hydrocephalus is even more uncommon, and has rarely been reported. Here, we describe the case of a neonatal patient with untreated neonatal posthemorrhagic hydrocephalus and an associated worsening hyponatremia that
WORLD NEUROSURGERY 125: 19-22, MAY 2019
pleural effusions requiring intubation, mechanical ventilation, and chest tube placement. Nutrition was maintained on parenteral nutrition with Enfaport (Mead Johnson & Company, LLC, Chicago, Illinois, USA) and supplementation with injectable lipid emulsion and dextrose. Head circumference (HC) and birthweight was 32 cm (79.56 percentile) and 2.39 kg (70.68 percentile), respectively. Neurology, and
Figure 1. Second day of life initial head ultrasound of the coronal plane showing the lateral ventricle with significant intraventricular hemorrhage.
www.journals.elsevier.com/world-neurosurgery
19
CASE REPORT MEGAN OBI AND KAINE ONWUZULIKE
NEONATAL HYPONATREMIA DUE TO HYDROCEPHALUS
Figure 2. Day 20 of life head ultrasound of the coronal plane showing the lateral and third ventricle with significant ventriculomegaly and resolving hemorrhage with peak head circumference.
subsequently neurosurgery, were consulted for expanding germinal matrix hemorrhage with initial head ultrasound on day 2 of life, demonstrating bilateral grade II intraventricular hemorrhage (IVH) (Figure 1) that subsequently progressed to grade III IVH on repeat head ultrasound the following day. Neurologic status remained intact and HC stable while patient’s respiratory problems were actively managed. On day 19 of life, HC was noted to be persistently elevated at 33.5 cm with tense, bulging fontanelles, mild hypertonia, and worsening anasarca with scalp pitting
edema. Creatinine was 0.26 mg/dL, previously ranging from 0.73e0.21 mg/dL since birth. On day 20 of life, HC had increased to 35 cm, brain magnetic resonance imaging demonstrated communicating hydrocephalus with worsening resistive indices, decreased diastolic flow in the anterior carotid arteries, and dilation of the ventricular system (Figure 2). Serum sodium (Na) levels at the time were 121 mmol/L (previously ranging from 136e152 mmol/ L), requiring correction with half intravenous 3% Na. Day 21 of life, HC
Figure 3. Postoperative 33rd day of life head ultrasound of the coronal plane showing the lateral and third ventricles with reduction of ventriculomegaly after external ventricular drain placement.
20
www.SCIENCEDIRECT.com
remained stable at 35 cm, however Na level continued to decline with a nadir of 116 mmol/L. Creatinine levels remained stable at 0.22 mg/dL. The patient continued to demonstrate mild sun-setting and hypotonicity, but remained arousable without any pathologic movements, abnormal pupil dilation, or significant gastrointestinal symptoms. A right external ventricular drain (EVD) was placed with removal of about 10e 15 cc/kg of cerebrospinal fluid (CSF) daily and Na correction attempted with full intravenous 3% Na. Postoperatively, on day 22 of life, the patient demonstrated flat and soft anterior fontanelle, HC was down 2 cm, and she became normonatremic at 140 mmol/L (Figure 3). Electrolytes continued to remain stable, creatinine levels persisted at 0.25 mg/dL, and EVD was replaced with a ventriculoperitoneal (VP) shunt on day 29 of life (Figure 4). To date, the patient has not required any additional neurosurgical procedures, has demonstrated no clinical signs of elevated intracranial pressure, electrolytes have continued to remain stable, and shunt has remained patent (Figure 5). DISCUSSION Germinal matrix IVH is one of the most commonly diagnosed brain disorders in preterm infants, with an incidence of about 15%-20% in very-low-birthweight infants, describing an inverse relationship between IVH incidence and gestational age.10,11 The Papile grading system is a widely used scale to describe IVH severity, with a grading scale of I-IV.12 IVH subsequently serves as the most common cause of acquired hydrocephalus in infants, and is thought to be due to subsequent fibrosis, arachnoid granulations, and deposition of proteins in perivascular spaces to inhibit CSF resorption.11,13 Clinical presentation of posthemorrhagic hydrocephalus in infants can include increasing orbitofrontal HC, fontanelle fullness, and splaying of sutures as well as signs of increased ICP (i.e., apnea, lethargy, decreased activity, and bradycardia).11 To date, multiple studies have demonstrated an association between hyponatremia and hydrocephalus in patients. However, to the best of our knowledge, those studies have predominantly focused on adult populations, as well as normal pressure hydrocephalus (NPH) or
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.01.124
CASE REPORT MEGAN OBI AND KAINE ONWUZULIKE
NEONATAL HYPONATREMIA DUE TO HYDROCEPHALUS
mmol/L
160 Bolus of Hypertonic Saline 140
120
117 mmol/L
External Ventricular Drain Placed
100
91.22%
Na (mmol/L)
80
PercenƟle
Head Circumference (%) * 60
40
20
0 0
5
10
15
20
25
30
35
40
Day of Life
Figure 4. Graphic representation of the progression of sodium and the corresponding head circumference over the course of the patient’s initial days of life. Depicted is the point at which head circumference percentile peaked and sodium concentration was at its nadir. *Percentiles based off the Fenton growth chart for premature girls. Na, sodium.
obstructive etiologies of hydrocephalus.2,14-16 Additionally, hyponatremia in previous studies has been found to be a result of treatment of hydrocephalus rather than as a consequence of worsening hydrocephalus.17-21 Chou et al.2 established an understanding of the prevalence of this association and determined that hyponatremia in NPH patients was clinically more common than had previously been reported, and that those at risk included hypertensives and those with nasogastric tubes during hospitalization. In addition, previous reports have found a
correlation between NPH and syndrome of inappropriate secretion of antidiuretic hormone (SIADH) as well as polydipsia in patients.22 Studies that have involved the pediatric population have demonstrated a correlation between hyponatremia as a subsequent complication of frequent drainage of CSF, but not as a direct consequence of hydrocephalus.17,19 Tenbrock et al.,20 for example, discovered an association between use of a ventricular access device (specifically the Rickham reservoir) for serial liquor punctures in preterm infants with IVH, and
Figure 5. T2-weighted magnetic resonance imaging scans of the axial plane showing the lateral (A) and third ventricles (B) at 1-year follow-up demonstrating a reduction in ventricular size.
WORLD NEUROSURGERY 125: 19-22, MAY 2019
the subsequent development of hyponatremia with improvement after adequate fluid resuscitation. Our patient thus serves as a unique case of hydrocephalus associated hyponatremia in that her serum Na levels seemed to reach a nadir as her hydrocephalus worsened, with near immediate postsurgical resolution of hyponatremia. Although it may be difficult to distinguish whether the improvement in Na was entirely due to surgical correction with the EVD or hypertonic saline administration, it should be noted that the Na continued to decline despite the initial 3% bolus and only began to correct after EVD placement. In addition, although previous cases have suggested a potential renal etiology of SIADH contributing to hyponatremia, our patient’s creatinine levels remained low-normal and were unlikely related to the hyponatremia. Moreover, the patient was adequately receiving appropriate weight-based maintenance fluids, had no gastrointestinal problems, and was on no medications with a known side effect of hyponatremia, ruling out other potential hypovolemic and euvolemic causes of hyponatremia. Only 2 other studies known to us demonstrate improvement of hyponatremia with treatment of hydrocephalus with VP shunt, but those cases described elderly individuals with NPH.14,16 Although this case establishes precedence of the phenomenon, our patient describes a new population as well as etiology. No definitive mechanism has been determined for the association between hydrocephalus and hyponatremia. It is our hypothesis that elevated third ventricle pressure induces mechanical compression on the hypothalamus resulting in disturbances in endocrinology, one being hyponatremia. Wise et al.23 found that blockage of VP shunts resulted in hyponatremia, and subsequently resolved when the blockage was removed and the third ventricle size reduced, and Yoshino et al.16 used magnetic resonance imaging to detect third ventricle enlargement in patients with hyponatremia, both serving as additional evidence to the proposed theory. Additionally, studies observing SIADH in patients with Shy-Dragger and myotonic dystrophy found that patients additionally had enlarged third ventricles as well as atrophied hypothalamuses.24,25 Our patient did not have a significant number of additional studies performed to
www.journals.elsevier.com/world-neurosurgery
21
CASE REPORT MEGAN OBI AND KAINE ONWUZULIKE
NEONATAL HYPONATREMIA DUE TO HYDROCEPHALUS
pinpoint the pathophysiologic mechanism for her symptoms, but given our intervention, a reasonable correlation can be drawn between her worsening hydrocephalus and hyponatremia. However, to the best of our knowledge, there are currently no additional neonatal reports to note third ventricular enlargement as a cause of hyponatremia in the setting of no other internal or external causes. Thus, further research still needs to be performed to better understand this phenomenon and potentially confirm our proposed hypothesis.
5. Marcialis MA, Dessi A, Pintus MC, Irmesi R, Fanos V. Neonatal hyponatremia: differential diagnosis and treatment. J Matern Fetal Neonatal Med. 2011;24(suppl 1):75-79.
CONCLUSIONS
9. Zieg J. Evaluation and management of hyponatraemia in children. Acta Paediatr. 2014;103: 1027-1034.
Our aim in presenting this case was to describe a rare and surgically reversible cause of hyponatremia. For patients, especially preterm infants, with IVH and other concurrent medical complications in whom hyponatremia may have various causes, it is pertinent to consider hydrocephalus, notably associated with third ventricular enlargement, as a formidable contribution to hyponatremia, for which ventricular decompression may offer resolution. REFERENCES 1. Brunsvig PF, Os I, Frederichsen P. [Hyponatremia. A retrospective study of occurrence, etiology and mortality]. Tidsskr Nor Laegeforen. 1990;110: 2367-2369.
6. Rafael Valdivia L, Ferrandiz Quiroz J. [Hyponatremia as a possible mortality factor in cirrhotic patients hospitalised in the Guillermo Almenara Irigoyen State Hospital, 2003-2005]. Rev Gastroenterol Peru. 2007;27:37-46. 7. Reyes Dominguez A, Briones Perez de la Blanca E. [Intrahospital mortality and hyponatremia]. An Med Interna. 1996;13:616-617. 8. Greenberg JH, Tufro A, Marsenic O. Approach to the treatment of the infant with hyponatremia. Am J Kidney Dis. 2015;65:513-517.
10. Ahn SY, Shim SY, Sung IK. Intraventricular hemorrhage and post hemorrhagic hydrocephalus among very-low-birth-weight infants in Korea. J Korean Med Sci. 2015;30(suppl 1):S52-58. 11. Robinson S. Neonatal posthemorrhagic hydrocephalus from prematurity: pathophysiology and current treatment concepts. J Neurosurg Pediatr. 2012;9:242-258. 12. Papile LA, Burstein J, Burstein R, Koffler H. Incidence and evolution of subependymal and intraventricular hemorrhage: a study of infants with birth weights less than 1,500 gm. J Pediatr. 1978;92:529-534. 13. Tully HM, Dobyns WB. Infantile hydrocephalus: a review of epidemiology, classification and causes. Eur J Med Genet. 2014;57:359-368.
2. Chou CY, Liu JH, Wang SM, et al. Hyponatraemia in patients with normal pressure hydrocephalus. Int J Clin Pract. 2009;63:457-461.
14. Jung KH, Chu K, Jeong SW, Hong YH, Park KI, Roh JK. Idiopathic normal pressure hydrocephalus predominantly with prolonged fever and hyponatremia. Neurology. 2003;61:554-556.
3. Garcia Segura A, Gadea Ruiz C, Oliva Fanlo B, et al. [Hyponatremia upon admission in patients over 65 years of age. Relation with medium length of stay and hospital mortality]. An Med Interna. 1994;11:487-489.
15. Kim JH, Kang JK, Lee SA. Hydrocephalus and hyponatremia as the presenting manifestations of primary CNS lymphoma. Eur Neurol. 2006;55:39-41.
4. Goldberg A, Hammerman H, Petcherski S, et al. Hyponatremia and long-term mortality in survivors of acute ST-elevation myocardial infarction. Arch Intern Med. 2006;166:781-786.
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Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 12 December 2018; accepted 14 January 2019 Citation: World Neurosurg. (2019) 125:19-22. https://doi.org/10.1016/j.wneu.2019.01.124 Journal homepage: www.journals.elsevier.com/worldneurosurgery Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2019 Elsevier Inc. All rights reserved.
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.01.124