Acute Crit Care.  2024 Nov;39(4):647-651. 10.4266/acc.2021.01081.

Hydranencephaly in a newborn due to occupational toluene exposure during pregnancy: a case report

• Aydın B
• Botan E
• Gülensoy B
• Akyol S

• KMID: 2563555
• DOI: http://doi.org/10.4266/acc.2021.01081

Abstract

The etiopathogenesis of hydranencephaly remains unclear; however, exposure to toxic substances during pregnancy likely increases hydranencephaly risk. Head computed tomography (CT) was performed in a neonate 9 hours post-delivery because the anterior fontanelle was large and there were clinical signs of encephalopathy. Head CT revealed a lack of both cerebral hemispheres and significant cystic enlargement, while the cerebellar hemispheres and pons were found to have developed normally. History-taking revealed that the mother worked in the automotive industry, specifically in the car paint cleaning business and was exposed to toluene during the pregnancy. The patient was diagnosed with hydranencephaly, central diabetes insipidus and central hypothyroidism. Due to the increased head circumference and tense anterior fontanelle, a ventriculoperitoneal shunt was placed. Toluene exposure during pregnancy should be considered among the causes of hydranencephaly. Furthermore, central diabetes insipidus and central hypothyroidism may develop in such cases.

Keyword

hydranencephaly, hypothyroidism; newborn, toluene

Figure

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  Figure 1. Cranial computed tomography (A) and cranial magnetic resonance imaging (B) taken due to head circumference on the 1 postnatal day.

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  Figure 2. Head circumference of the patient by week. 8 Week: first ventriculoperitoneal shunt insertion; 22 week: ventriculoperitoneal shunt revision.

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  Figure 3. Cranial computed tomography (A) and clinical (B) image of the patient after ventriculoperitoneal shunt insertion.

Reference

1. Khalid M, Khalid S, Zaheer S, Redhu N. Hydranencephaly: a rare cause of an enlarging head size in an infant. N Am J Med Sci. 2012; 4:520–2.

2. Quek YW, Su PH, Tsao TF, Chen JY, Ng YY, Hu JM, et al. Hydranencephaly associated with interruption of bilateral internal carotid arteries. Pediatr Neonatol. 2008; 49:43–7.

3. Sandoval JI, De Jesus O. Hydranencephaly. In: StatPearls [Internet]. StatPearls Publishing;2022.

4. Pavone P, Praticò AD, Vitaliti G, Ruggieri M, Rizzo R, Parano E, et al. Hydranencephaly: cerebral spinal fluid instead of cerebral mantles. Ital J Pediatr. 2014; 40:79.

5. Plantaz D, Joannard A, Pasquier B, Bost M, Beaudoing A. Hydranencephaly and congenital toxoplasmosis: apropos of 4 cases. Pediatrie. 1987; 42:161–5.

6. Hannigan JH, Bowen SE. Reproductive toxicology and teratology of abused toluene. Syst Biol Reprod Med. 2010; 56:184–200.

7. Laslo-Baker D, Barrera M, Knittel-Keren D, Kozer E, Wolpin J, Khattak S, et al. Child neurodevelopmental outcome and maternal occupational exposure to solvents. Arch Pediatr Adolesc Med. 2004; 158:956–61.

8. Demır M, Cicek M, Eser N, Yoldaş A, Sısman T. Effects of acute toluene toxicity on different regions of rabbit brain. Anal Cell Pathol (Amst). 2017; 2017:2805370.

9. Filley CM, Halliday W, Kleinschmidt-DeMasters BK. The effects of toluene on the central nervous system. J Neuropathol Exp Neurol. 2004; 63:1–12.

10. Omar AT 2nd, Khu KJO. Hydranencephaly complicated by central diabetes insipidus: report of two cases and systematic review of literature. Childs Nerv Syst. 2019; 35:1165–71.