International Journal of Pediatric Otorhinolaryngology Extra
Volume 1, Issue 4 , Pages 310-314, December 2006

Diplopia in childhood secondary to sphenoidal sinusitis

Inonu University, Pediatric Neurology, Malatya, Turkey

Received 13 August 2006; received in revised form 23 September 2006; accepted 24 September 2006.

Article Outline

Summary 

Ocular palsies are seldom seen in pediatric neurology practice whereas more common in adults. A variety of neurological and muscular disorders play role in etiology. Sphenoid sinusitis, which is very rare in childhood period, can cause serious neurological complications because of its anatomically close location to some vital organs. Sphenoid sinusitis should be ruled out in differential diagnosis of diplopia in children.

Keywords: Sphenoidal sinusitis, Diplopia, Childhood

 

Back to Article Outline

1. Introduction 

Diplopia is a common subjective complaint, or it may be elicited during the course of the examination. It is usually the first symptom of many systemic disorders, or may occur in conjunction with some muscular and neurological disorders. Ocular myasthenia, thyroid ophthalmopathy, diabetic ophthalmoplegia, mitochondrial diseases (i.e., cytochrome c oxidase deficiency) and oculopharyngeal muscular dystrophy are important muscular pathologies. In addition, encephalopathy with multiple infarcts, post-traumatic diplopia, carotid cavernous sinus fistula, frontal sinus mucocele, midbrain hemorrhage, basal meningitis and cerebral tumor are common intracranial pathologies [1].

Sinusitis is the inflammation of the mucosa lining paranasal sinuses. It is more commonly diagnosed in toddlers and school-aged children. Facial pain, headache, decrease in sense of smell and nasal congestion are usual symptoms. Infectious complications like brain abscess, meningitis and subdural empyema have been occasionally reported. In addition, neurological complications like cavernous sinus thrombophlebitis, paralysis of third, fourth and sixth cranial nerves, optic neuritis, vision loss and diplopia are rarely reported in pediatric population [2], [3], [4], [5], [6].

We, herewith, report a pediatric case who presented with diplopia which is a very rare complication of sphenoidal sinusitis.

Back to Article Outline

2. Case report 

A 10-year-old male subject was admitted to pediatric outpatient clinic with the complaint of double vision for the last 2 months. Detailed history revealed irregular headache periods for the last year that gradually increased within last 2 months. There was no remarkable detail in own and familial history. Physical examination showed no pathology other than mild restriction in outer gaze of left eye. In assessing binocular look, he complained double vision both on left outer gaze and after 1.5m in primary position. Complete blood count, C-reactive protein, erythrocyte sedimentation rate, routine serum biochemistry tests and thyroid hormones were all within normal limits. In order to exclude idiopathic intracranial hypertension, we performed lumbar puncture and perimetric visual field examination which both showed no pathology. Two weeks of oral pyridostigmine therapy for the presumptive diagnosis of ocular myasthenia showed no clinical beneficial effect.

Cranial magnetic resonance imaging was within normal limits except unilateral sphenoidal pathology suggestive of sinusitis (Fig. 1). Bearing in mind that sphenoidal sinusitis may occasionally spread neighbouring sixth cranial nerve, and thereby cause inflammation and diplopia, we prescribed parenteral amoxicilline-clavulanate suspension for 3 weeks. He reported gradual recovery in diplopia complaint, and complete resolution of symptoms after 4 weeks. Repeated neurological imaging in his follow-up revealed almost completely resolved sphenoidal sinus infection (Fig. 2).

  • View full-size image.
  • Fig. 2. 

    After treatment, sphenoidal sinus has normal structure and consistency in axial CT images (bone window). In addition, bony septa lying in sphenoidal sinus attracts attention.

Back to Article Outline

3. Discussion 

Ocular palsies, more frequent complaint in adults than children, are not uncommon in daily neurology practice. A variety of neurological and muscular disorders have been reported in etiology [1]. The most common presenting symptom is cranial nerve palsies that are attributable to frontal [1] and sphenoidal [7] sinusitis, particularly in adults. The etiological evaluation of the abducens nerve palsy in our patient revealed isolated sphenoidal sinusitis (ISS) which is a rare phenomenon in childhood period.

Sphenoidal sinuses, consist a couple of mucus cells, are covered with ciliated pseudo-stratified epithelium. Therefore, there is a lower drainage rate when compared to remaining paranasal sinuses. The sphenoidal sinuses are located at the apex of the nasal cavity, and this location results in a paucity of specific symptoms and signs [2]. Although underdiagnosed clinically, the incidence of acute ISS is around 3% of all sinusitis cases [8], [9]. Delay in the diagnosis of the condition, which frequently occurs, further facilitates intracranial complications [3]. As the development of sphenoidal sinuses are usually after 6 years of age, the complications are almost always observed in school-aged children as in our case [4], [5].

Steroid therapy, cocaine abuse, immunosuppressive therapy, infected water entry during swimming, craniofacial radiotherapy, obstruction of the sinus ostium and maxillofacial trauma are all predisposing factors for ISS [6]. However, we did not document any risk factor in our patient history and physical examination.

Headache on initial evaluation, which is typically non-localizing and refractory to medical therapy, is almost always the presenting symptom in ISS [10]. The ocular symptoms like visual loss or ocular nerve palsies follow headache [3]. This subject also complained of headache and horizontal diplopia due to involvement of abducens nerve. Similarly, Ada et al. [6] recently reported an adolescent girl with unilateral sixth nerve palsy secondary to ISS.

The most common complication of sphenoidal sinusitis is meningitis [11]. Any surrounding tissue adjacent to sphenoid sinus may be infected. As a result of close anatomical relationship with sphenoid sinuses, cranial nerves II–IV, dura meter, pituitary gland, cavernous sinus, internal carotid artery, sphenopalatine artery, pterigopalatine nerve have been reported to be infected by dissemination [7]. Complications like orbital cellulitis, orbital abscess, orbital apex syndrome, blindness, meningitis, epidural and subdural abscesses, cerebral infarcts, pituitary abscess, cavernous sinus thrombosis and internal carotid artery thrombosis have been published in literature [12]. Our patient only suffered diplopia because of the isolated involvement of sixth cranial nerve.

Clinical suspicion is very important to reach the diagnosis because the symptoms, history and physical examination are not specific for sphenoidal sinusitis. High-resolution axial and coronal computed tomography (CT) is recommended for the diagnosis of sphenoidal sinusitis and the probable intracranial complications [13]. However, cranial magnetic resonance imaging is superior to CT in terms of detecting the involvement of cranial nerves, cavernous sinus, surrounding neurovascular tissue and the presence of tumor [9].

The most common pathogens in the etiology of sphenoidal sinusitis are Staphylococcus aureus, Streptococcus pneumoniae and some aerobic or anaerobic Streptococcus spp. Fungi, particularly Aspergillus spp., should be kept in mind in immunosuppressed patients [8], [14]. Uren and Berkowitz [15] reported eight children with ISS that five of them had been treated successfully with medical therapy. Remaining three subjects, either unresponsive to medical therapy or complicated cases, had undergone endoscopic sphenoidotomy. Parenteral antibiotic therapy should be administered in the beginning since this infection may cause serious even fatal complications. The antibiotic therapy should be completed to a total of 3–4 weeks. The topical decongestants and irrigation with saline solution are recommended as adjunctive therapy [3]. As this subject did not suffer any immunosuppressive condition, we commenced parenteral amoxicilline-clavulanate therapy for the probable pathogens, Staphylococcus spp. and Streptococcus spp. After 3 weeks of therapy, we observed complete clinical and radiological recovery.

Sphenoidal sinusitis is rarely experienced in childhood period but might result in serious neurological complications. We should rule out paranasal sinusitis, particularly ISS, in differential diagnosis of diplopia and focal neurological deficits.

Back to Article Outline

References 

  1. Batocchi AP, Evoli A, Majolini L, Lo Monaco M, Padua L, Ricci E, et al. Ocular palsies in the absence of other neurological or ocular symptoms: analysis of 105 cases. J. Neurol. 1997;244:639–645
  2. Steadman CD, Salmon AH, Tomson CR. Isolated sphenoid sinusitis complicated by meningitis and multiple cerebral infarctions in a renal transplant recipient. Nephrol. Dial. Transplant. 2004;19:242–244
  3. Tan HK, Ong YK. Acute isolated sphenoid sinusitis. Ann. Acad. Med. Singapore. 2004;33:656–659
  4. Saitoh A, Beall B, Nizet V. Fulminant bacterial meningitis complicating sphenoid sinusitis. Pediatr. Emerg. Care. 2003;19:415–417
  5. Kadioglu HH, Sengul G, Malcok UA. Sphenoid sinusitis disguised by precocious puberty. Int. J. Pediatr. Otorhinolaryngol. 2005;69:275–278
  6. Ada M, Kaytaz A, Tuskan K, Guvenc MG, Selçuk H. Isolated sphenoid sinusitis presenting with unilateral VIth nerve palsy. Int. J. Pediatr. Otorhinolaryngol. 2004;68:507–510
  7. Proetz AW. The sphenoid sinus. Br. Med. J. 1948;2:243–245
  8. Lew D, Southwick FS, Montgomery WW, Weber AL, Baker AS. Sphenoid sinusitis. A review of 30 cases. N. Engl. J. Med. 1983;309:1149–1154
  9. Jacquier A, Facon F, Vidal V, Pascal T, Chapon F, Dessi P, et al. Sphenoid sinusitis. J. Neuroradiol. 2003;30:211–218
  10. Lawson W, Reino AJ. Isolated sphenoid sinus disease: an analysis of 132 cases. Laryngoscope. 1997;107:1590–1595
  11. Younis RT, Anand VK, Childress C. Sinusitis complicated by meningitis: current management. Laryngoscope. 2001;111:1338–1342
  12. Urquhart AC, Fung G, McIntosh WA. Isolated Sphenoiditis: a diagnostic problem. J. Laryngol. Otol. 1989;103:526–527
  13. Xenos C, Rosenfeld JV, Kleid SM. Intracranial extension of sphenoid sinusitis. Head Neck. 1995;17:346–350
  14. Wyllie JW, Kern EB, Djalilian M. Isolated sphenoid sinus lesions. Laryngoscope. 1973;83:1252–1265
  15. Uren BA, Berkowitz RG. Isolated inflammatory sphenoid sinus disease in children. Ann. Otol. Rhinol. Laryngol. 2003;112:370–372

PII: S1871-4048(06)00090-6

doi:10.1016/j.pedex.2006.09.005

International Journal of Pediatric Otorhinolaryngology Extra
Volume 1, Issue 4 , Pages 310-314, December 2006

Article Tools

* Image Usage & Resolution