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Year : 2019  |  Volume : 14  |  Issue : 2  |  Page : 110-112

Resolution of idiopathic intracranial hypertension symptoms by surgery for obstructive sleep apnea in a pediatric patient

1 Department of Neurology, Neurology Clinic, Yozgat City Hospital, Yozgat, Turkey
2 Department of Ophthalmology, Ophthalmology Clinic, Yozgat City Hospital, Yozgat, Turkey

Date of Web Publication20-Aug-2019

Correspondence Address:
Dr. Halil Onder
Neurology Clinic, Yozgat City Hospital, Yozgat.
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jpn.JPN_30_19

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How to cite this article:
Onder H, Aksoy M. Resolution of idiopathic intracranial hypertension symptoms by surgery for obstructive sleep apnea in a pediatric patient. J Pediatr Neurosci 2019;14:110-2

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Onder H, Aksoy M. Resolution of idiopathic intracranial hypertension symptoms by surgery for obstructive sleep apnea in a pediatric patient. J Pediatr Neurosci [serial online] 2019 [cited 2023 Dec 1];14:110-2. Available from: https://www.pediatricneurosciences.com/text.asp?2019/14/2/110/264745

Dear Editor,

A 10-year-old boy presented to our ophthalmology polyclinic with complaints of blurry vision and transient visual obscurations. On ophthalmological assessment, Frisen stage 1 papilledema was determined [Figure 1]. Other examinations including visual acuity, color vision, and visual field were in normal ranges. Automated Humphrey visual field test also showed a normal visual field. Hence, the patient was referred to the neurology department for further examinations. He was 135cm in height and 35kg in weight with a body mass index of 20kg/m2. He had no complaints of headache, tinnitus, or double vision. Other neurological examinations were within normal limits. Cranial magnetic resonance imaging showed normal findings. Therefore, with a provisional diagnosis of idiopathic intracranial hypertension (IIH), lumbar puncture (LP) was performed. LP opening pressure was high (280mm H2O) and biochemical investigations of the cerebrospinal fluid were in normal ranges. With the final diagnosis of IIH, acetazolamide 2 × 250mg was initiated. Following medication, no significant improvement in patient’s visual symptoms or fundus examination results occurred. Besides, an apparent metabolic acidosis occurred because of acetazolamide usage in the 2nd month of the treatment, which enabled to continue acetazolamide. On the contrary, during reevaluations on the 2nd month of follow-up, a significant hypernasal speech was recognized, and it was learned that the patient had sleep apnea episodes for a long time (which was obvious over the last 3 years as it could be learned from his parents). Hence, the patient was referred to the otorhinolaryngology department. Otorhinolaryngology examination revealed significant septal deviation and hypertrophy of the concha. The Epworth Sleepiness Scale (ESS) score was calculated as 12. Polysomnography was suggested, however, could not be performed due to socioeconomic limitations. Clinically, obstructive sleep apnea syndrome (OSAS) was considered. However, as a result of interdepartmental consultations and recent literature data pointing to an association between OSAS and IIH in terms of a causal relationship, surgery was predicted to improve papilledema also. Hence, septoplasty surgery in conjunction with conchoplasty was planned. Following the surgery, apnea episodes dramatically resolved and repeated ESS score was 0; a rapid and progressive improvement in the patient’s visual symptoms was also achieved. Post-op optic coherence tomography (OCT) showed a significant resolution in the average retinal nerve fiber measurements [Figure 2]. Furthermore, LP was repeated, which showed the resolution of LP opening pressure to 150mm H2O.
Figure 1: Fundus photography showing Frisen stage 1 papilledema at admission. (A) Right eye. (B) Left eye

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Figure 2: Figure showing recovery in the average retinal nerve fiber measurements. (A) At admission. (B) At 10th month of follow-up (4 months after surgery). RNFL = average retinal nerve fiber measurements

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   Discussion Top

The association of OSAS and cerebral vasodilatation–linked intracranial pressure increments is a recognized entity among specialists.[1] However, there is no consensus about the issues of the association between IIH and OSAS, the exact responsible mechanisms, or the causal relationship between these entities.[2] Nevertheless, an increasing evidence (at the level of case reports) supporting the role of OSAS as an etiological agent in the pathophysiology of IIH is available.[3],[4],[5] Herein, we present a remarkable patient with IIH, who recovered significantly via the surgery of OSAS. The improvement was shown by the resolution of the OCT measurements as well as LP opening pressure measurements. However, a major limitation may be that an objective evaluation in the interval period before surgery and after medical treatment may complicate the mechanisms of recovery of IIH. Also, the medical treatment duration was too short and insufficient (due to side effects) to obtain an optimal treatment response. Besides, the patient and his parents stated a clear resolution of the visual symptoms after the surgery, which may also support the role of OSAS surgery in the recovery of IIH. In our previous report, we had shown the recovery of a malignant coursed IIH after surgery for OSAS in detail via serial OCT measurements and fundus examination results.[3] Through the presentation of this report and related literature, we reemphasize the importance of interrogation of OSAS in patients with IIH (also in the pediatric population as in adult patients), as treatment of OSAS may potentially relieve the primary etiology leading to their total cure.

Another important point may be that our patient did not have headache in contrast to the classical presentations of IIH. IIH without headache has been reported rarely in literature.[6] These patients were particularly reported to be more likely to present with severe visual loss.[6] Hence, the authors remarked that the management of IIH is difficult in the absence of headache as none of the major clinical signs for the monitorization of the patients is present.[6] In this context, we would also like to emphasize that detailed ophthalmological evaluation is the critical stage for early diagnosis as well as better visual outputs in these specific patients. Future illustrations of these rare patients might contribute to our understanding of the pathophysiology of IIH as well as OSAS. The results of these studies may also provide crucial remarks to be kept in mind in clinical practice.

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   References Top

Sanner BM, Konermann M, Tepel M, Groetz J, Mummenhoff C, Zidek W Platelet function in patients with obstructive sleep apnoea syndrome. Eur Respir J 2000;16:648-52.  Back to cited text no. 1
Wardly D, Wolford LM, Veerappan V Idiopathic intracranial hypertension eliminated by counterclockwise maxillomandibular advancement: a case report. Cranio 2017;35:259-67.  Back to cited text no. 2
Onder H, Ergun O, Kaygisiz M, Daltaban IS Total improvement after surgery for obstructive sleep apnea syndrome in a patient with concurrent malignant idiopathic intracranial hypertension. J Neurosurg 2018:1-5.  Back to cited text no. 3
Javaheri S, Qureshi Z, Golnik K Resolution of papilledema associated with OSA treatment. J Clin Sleep Med 2011; 7:399-400.  Back to cited text no. 4
Kalyoussef E, Brooks NO, Quraishi H, Turbin R, Frohman L Idiopathic intracranial hypertension in a child with obstructive sleep apnea cured by tonsillectomy/adenoidectomy. J Neuroophthalmol 2013;33:413-4.  Back to cited text no. 5
Lim M, Kurian M, Penn A, Calver D, Lin JP Visual failure without headache in idiopathic intracranial hypertension. Arch Dis Child 2005;90:206-10.  Back to cited text no. 6


  [Figure 1], [Figure 2]

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