Acute sphenoid sinusitis presenting as aseptic meningitis: a pachymeningitis syndrome

Internutionul Journttl of Pediatric Otorhinolu~ngologv, Elsevier Biomedical Press

4

( 1982)77-X 1

77

Acute sphenoid sinusitis presenting as aseptic meningitis: a pachymeningitis syndrome Itzhak Brook *, Gary D. Overturf, Evan A. Steinberg Donald B. Hawkins Depurtments

of Pediutrics und the Conmutwuhle Angeles Couniy-USC

Disuse

and

Serorce, lJnioer.si!,~ of Southern Culiforniu. Los

Medicul Center, Los Angeles. CA 90023 {U.S.A.) (Received June 1st. 1981) (Accepted July 27th. 1981)

Introduction

A bacterial infection or abscess adjacent to the meninges can produce symptoms and cerebrospinal fluid (CSF) pleocytosis similar to those of meningitis. Effective treatment, however, requires the administration of different antibiotics than those used routinely for bacterial meningitis. In addition, surgery may be indicated. This report describes a case of sphenoid sinusitis mimicking acute aseptic (viral) meningitis.

Case Report

A 14.5-year-old Caucasian female, presented on 8-21-75 to Los Angeles CountyUniversity of Southern California Medical Center, with a 3-day history of gradual onset of sharp constant frontal headache, vomiting, photophobia and slight blurring of vision. She denied fever, chills, history of previous sinus disease, ear infections, head injuries or exposure to known infections. Upon admission to the hospital physical exam revealed a small thin Caucasian female with temperature 99,4”F, pulse 1lO/min, blood pressure 105/75 mm Hg. Tympanic membranes were normal. Pupils were 2 mm each, round, regular and reactive to light, although somewhat sluggishly; accommodation was normal; extraocular movements were normal; fundi were unremarkable. Her neck was slightly stiff, with positive Brudzinski sign. Neurological examination was normal. Initial laboratory studies (Table I) included CBC with 11,000 WBC/mm3, 95% segs, 2% lymphs, 3% monos, Hgb 12.7 Gm/dl and urinalaysis with specific gravity *

Corresponding author and address for reprints: Itzhak Brook. M.S., M.D.. Naval Institute, National Naval Medical Center, Bethesda, MD 20814 (U.S.A.).

Medical

Research

I

ADM

982 93% 86 36 neg. normal vomiting

Cells/mm3 % PMNS Glucose (mg/dl) Protein (mg/dl) Culture Neurological

AND NEUROLOGICAL

CSF

CSF CHANGES

TABLE DURING

300 79% 89 32 neg. normal vomiting

12 h

FINDINGS

42 69% 74 31 neg. retro-orbital pain tender RT. Sinuses

day

SINUSITIS

4th-6th

OF SPHENOID

162 26% RI 33 neg. normal vomiting

36 h

COURSE

anisocoria decreased visual acuity, O.D. temporal hemianopsia, bruit, O.D.

N.D.

7th- 10th day

of 1.020, trace albumin, small acetone, no sugar, 0.2 WBC/hpf and O-l RBC/hpf. Cerebrospinal fluid (CSF) had 982 WBC/mm3, 93% PMNS, sugar 86 mg/dl, protein 36 mg/dl; gram stain was negative for bacteria. Cultures of blood were obtained.

Hospital course For 3 days she had intermittent fever to 103°F and complained of severe, non-localized headaches. Ophthalmic exam remained normal. CSF after admission revealed a shift to a predominantly lymphocytic pleocytosis (Table I). On the fourth hospital day her headache decreased and vomiting resolved but she complained of pain behind and around the left eye. Physical exam revealed tenderness of the left periorbital area without signs of inflammation. On day 5, the temperature had declined to lOl”F, but the left frontal sinus was tender to palpation. Radiographs of the paranasal sinuses were interpreted as normal. On day 6 she complained of right eye pain. Blood cultures from the night of admission and from the fifth hospital day had yielded Staphylococcus aureus. WBC at this time was 7,800/mm3 with 64% PMNs and 2% bands; ESR was 53 mm/h. Treatment with oxacillin was initiated, 1 g i.v. every 4 h. On the seventh day her right pupil was noted to be dilated and poorly reactive to light. She had diminished visual acuity in the right eye, with a right temporal hemianopsia, right exotropia, and a blurred right optic nerve disc margin. The following day, although still dilated, the right pupil was slightly more reactive to light and the right exotropia was no longer noted; however poor visual acuity persisted in this eye. On the ninth day her visual status was unchanged; the right fundus showed a hyperemic disc with elevation of the margins; the left fundus was normal. A brain scan demonstrated an area of increased uptake in the right anterior subcalvarial area. On the tenth day she became afebrile. However, bruits were noted in the left temporal area and over the right globe. She described tenderness of the right eye on movement. At the end of two weeks of hospitalization, a bilateral carotid angiogram revealed distal spasm of both internal carotid arteries in the cavernous portion. with lateral displacement of the right internal carotid. Sphenoid sinus clouding was also noted. Tomograms were obtained; both sphenoid sinuses and posterior ethmoid sinuses were noted to be clouded. Upon review the original sinus radiographs were felt to be normal, despite the abnormalities noted on the tomograms. Phenylephrine, 0.25% and pseudoephedrine 30 mg p.o. every 8 h were added to treatment over the next 12 days, her vision progressively improved; on the 29th day her visual acuity was 20/25 on the right and 20/15 on the left; color vision was intact; visual field testing demonstrated a right inferonasal cut. Oxacillin was discontinued after 24 days because of neutropenia and eosinophilia. Institution of vancomycin and subsequently clindamycin, were associated with generalized urticaria, and all antibiotic therapy was discontinued. On the 40th hospital day a bilateral intranasal sphenoidotomy and ethomidotomy were per-

formed. Purulent material was drained from both sphenoid sinuses but neither ethmoid sinus yielded purulent material. Culture of surgical specimens grew Streptococcus viridans, gamma Streptococci, Staphylococcus albus, and Neisseria sp. Biopsy of the sphenoid sinus revealed only chronic inflammation and granulation tissue. Postoperatively cephalothin 1.20 g, i.v. every 4 h was administered for 5 days followed by cephalexin 250 mg p.o. every 6 h. She was discharged on the seventh postoperative day. Her visual acuity was 20/20 in each eye, although her right pupil remained sluggishly reactive to light; color perception was intact; extraocular movements were intact.

Discussion This patient was initially thought to have meningitis. An early clinical course characterized by resolution of headache, a falling CSF white cell count, and increasing percentage of CSF mononuclear cells, negative CSF culture and gram stains, all appeared compatible with the expected progression of aseptic meningitis [4]. However, the CSF in sphenoid sinus infection may be abnormal [ 1,6], mimicking the findings in aseptic menigitis. The white cell count may be highly variable, from 10 to 1700, but is usually below 500; the differential ranges from 5% to 99% polymorphonuclear cells. Although CSF protein may be mildly elevated, sugar is normal and gram stain and bacterial culture are negative. A localized suppurative process, adjacent to the dura mater, with CSF pleocytosis, has been previously referred to as pachymeningitis indicating dural inflammation [lo]. This syndrome has been described with infection of frontal, ethmoidal or sphenoidal sinuses, mastoiditis, osteomyelitis or cranial or vertebral bones, and epidural abscess. Spread of the infection, when unhindered by effective antimicrobial therapy, follows well-defined anatomic pathways and leads to a variety of complications of which meningitis has been the most common [7]. Meningeal infection may be the result of septicemia or venous septic thrombosis. Direct extension of infection through bone and dura mater may occur or infection may be spread by regional pathways such as the pia arachnoid sheath of the optic nerve or perineural sheath of the olfactory nerves [8]. The symptoms of pachymeningitis are generally those of local compression, and it should be considered in the differential diagnosis of aseptic meningitis, particularly when there is a history of sinus disease, cranial trauma, chronic ear infections, or when there are signs of focal neurologic deficit, purulent nasal or otic discharge, middle ear disease, or failure to improve as expected. The sphenoid sinus central location in the skull places it adjacent to dura mater, optic chiasm, pituitary gland, opthalmic veins, internal carotid artery, cavernous sinus, cranial nerves II, III, IV, VI and the ophthalmic and maxillary divisions of V. Ophthalmologic problems similar to the patient described have been among the most commonly reported complications of sphenoid sinus disease [1,2]. The orbital apex syndrome is one such constellation and includes external and internal ophthalmoplegia, optic neuritis, and ophthalmic nerve sensory complaints ranging from

neuralgia to anesthesia [5]. This syndrome arises from compression of the nerves and vessels which course through the superior sphenoid fissure. The patient in this case fulfilled all criteria, but lacked external ophthalmoplegia. Other features of sphenoid sinus infection include reto-oribtal headache [ 1,2] and fluctuating neurological signs and symptoms [6]. Perhaps the most common feature of sphenoid sinus infection is the difficulty in diagnosis. Yune and co-workers state the small size of the sphenoid sinus limits the usefulness of routine radiographic procedures [ 111. They recommended tomography if a lesion of the sphenoid is suspected. Others have described the angiographic signs of sphenoid sinus infection as segmented narrowing and lateral deviation of the intracavemous internal carotid artery [6]. The lack of value of routine sinus radiography in sphenoid sinus disease, as well as the usefulness of carotid angiography and tomography, are well demonstrated by this case report. A variety of organisms have been implicated in sphenoid sinus infecton including such unusual bacteria as Proteus mirabilis and enterococci, as well as pneumococci, streptococci, hemophilus and staphylococcal organisms [ 1,6,9]. Importantly, the etiologic agent may be resistant to the antibiotics used routinely for bacterial meningitis as was the S. aureus isolated from the blood of our patient. Lastly, surgical drainage of the sphenoid sinus is usually necessary; surgical techniques have been reviewed elsewhere 131.

References I Alper, M.G., Mucoceles of the sphenoid sinus: neuroophthalmologic manifestations, Trans. Amer. ophthal. Sot., 74 (1976) 53. 2 Bregeat, P., Ophthalmic manifestations of sphenoidal mucoceles, Ann. Ophthal.. 9 (1977) 259. 3 Calcaterra. T.C. and Rand, R.W., Current adjuncts for surgery of the sphenoid sinus and pituitary gland, Laryngoscope, 86 (1976) 1692. 4 Feigin, R.D. and Shackelford, P.G., Value of repeat lumbar puncture in the differential diagnosis of meningitis, N. Engl. J. Med., 289 (1973) 571. 5 Kronschnabel, E.F., Orbital apex syndrome due to sinus infection, Laryngoscope, 84 (1974) 353. 6 Nelson, D.A., Holloway, W.J., Kara-Eneff. S.C. and Goldenberg. HI.. Neurological syndromes produced by sphenoid sinus abscess, Neurology, I7 ( 1967) 98 I. 7 Teed, R.W.. Meningitis from the sphenoid sinus. Arch. Otolaryng., 28 (1938) 589. 8 Turner, A.L. and Reynolds, F.E., Intracranial Pyogenic Diseases, Oliver and Boyd. London, 1931. p. II. 9 Van Cauwenberge, P., Bacteriological findings in sinusitis (1963-1975) Stand. J. infect. Dis., Suppl.. 9 (I 976) 72. IO Wechsler, I.S., A Textbook of Clinical Neurology, Saunders, Philadelphia, 1932, 2nd edn., p. 169. 1I Yune. H.Y.. Holden, R.W. and Smith, J.A.. Normal variations and lesions of the sphenoid sinus. Amer. J. Roentgenol., 124 (1975) 129.