CLINICAL RECORD
The Journal of Laryngology & Otology (2013), 127, 1148–1151. © JLO (1984) Limited, 2013 doi:10.1017/S0022215113002181
Posterior orbital cellulitis: case report and literature review K ALLAN, H ATKINSON, F AGADA ENT, Head and Neck Surgery Department, York Teaching Hospitals NHS Trust, Scarborough Hospital, UK
Abstract Introduction: It is rare for isolated sphenoid sinusitis to cause orbital cellulitis. We present a rare case of posterior orbital cellulitis, so caused, together with a review of the relevant literature. Case report: A 29-year-old woman presented with a 6-week history of progressive, unilateral, retro-orbital and periorbital right eye pain. On examination, the only finding was reduced visual acuity in the right eye. A computed tomography scan demonstrated right frontal and sphenoid sinus opacification. Sphenoidotomy and frontal sinus trephination were subsequently performed, following failure to respond to intravenous antibiotics. After surgery, the patient’s vision returned to normal. Conclusion: Isolated sphenoid sinusitis is rare but can cause significant visual disturbance and permanent loss of vision. Vague symptoms unsupported by clinical signs at presentation are a feature of posterior orbital cellulitis. The presented case highlights the problem, and the need for a high index of clinical suspicion even in the absence of firm clinical signs, in order to prevent permanent visual loss. Key words: Sphenoid Sinus; Sinusitis; Orbital Cellulitis; Diagnosis
Introduction Sinusitis in adults is common, but orbital complications are usually associated with paediatric rather than adult patients. There is a debate as to the difference between orbital apex syndrome and posterior orbital cellulitis. In the latter condition, a segment of the optic nerve in the superior orbital fissure is affected. This paper reports a rare occurrence of isolated sphenoid sinusitis with associated presentation of posterior orbital cellulitis. The subtlety of eye signs seen in the condition necessitates a high index of clinical suspicion in order to avoid missing the diagnosis.
Case report A 29-year-old woman presented as an acute medical admission with a 6-week history of progressive, severe, retroorbital and peri-orbital right eye pain. The pain was throbbing in nature, increasing in severity, and associated with headaches, nausea and vomiting. The patient’s vision was assessed by the ophthalmologists, revealing blurring, reduced acuity and associated photophobia. There were no other signs of meningism. Initial examination demonstrated right-sided ptosis and tenderness on palpation of the orbit. The patient had demonstrably reduced acuity but maintained normal colour vision. Inflammatory markers were within normal limits. An urgent magnetic resonance imaging brain scan was performed. This showed right sphenoid sinusitis, with sphenoidal and frontal sinus mucosal thickening (Figure 1). Accepted for publication 8 April 2013
FIG. 1 Axial magnetic resonance image showing right sphenoid sinusitis, with opacification and right sphenoid and frontal sinus mucosal thickening.
First published online 29 October 2013
CLINICAL RECORD
Computed tomography (CT) scanning of the sinuses was requested to further define the mucosal disease. This demonstrated right frontal and sphenoid sinus opacification only, with no evidence of maxillary, ethmoidal or contralateral disease (Figure 2). Following the microbiologist’s advice, the patient was started on intravenous Timentin (GSK, Uxbridge, UK). After 48 hours of IV antibiotics, her vision continued to deteriorate, and her ophthalmalgia and photophobia were unchanged. Ophthalmological reassessment revealed reduced acuity: 6/36 for the right eye, compared with 6/6 for the left. Important negative findings included no visible proptosis or chemosis, no demonstrable relative afferent pupillary defect, no reduction in external ocular movements, and no
1149 diplopia. The patient’s optic discs were healthy, with no swelling or venous engorgement. The patient began to develop red desaturation. Two diagnoses were considered by ophthalmology at this point: orbital soft tissue inflammation secondary to sinus infection, and idiopathic orbital inflammatory disease (a diagnosis of exclusion). Right-sided sphenoidotomy and frontal sinus trephination were performed urgently. Pus under pressure drained rapidly from the right sphenoid sinus and was sent for culture and sensitivity tests. The frontal sinus was full of polypoidal mucosa. Microbial culture grew Gram-positive bacilli only; culture of the frontal sinus sample grew no organisms after 48 hours.
FIG. 2 (a) Coronal computed tomography (CT) scan showing right sphenoiditis, with clear ethmoidal sinuses. (b) Axial CT showing right frontal sinus opacification. (c) Axial CT showing sphenoid sinusitis, with clear ethmoid sinuses. (d) Axial CT showing clear maxillary sinuses.
1150 Post-operatively, the patient’s visual acuity improved slowly. Three months after surgery, her vision had returned to normal.
Discussion The sphenoid sinus is lined with pseudostratified epithelium, with fewer mucus-secreting cells compared with other paranasal sinuses.1 Predisposing factors for sphenoid sinusitis include anatomical variation, radiotherapy, immunosuppression and malignancy.2–4 In the presented case, however, no predisposing factor was found. Headache is usually the most common initial symptom of acute sphenoiditis.1 In our patient, there was significant right orbital pain that was predominately retro-bulbar. Sphenoiditis is most often non-specific and poorly localised, due to its inaccessible position. Symptoms are often non-specific.2 Rhinorrhoea and post-nasal drip are common symptoms, with purulent exudates frequently seen below the middle and superior turbinates.3 Visual loss as the presenting symptom is extremely rare, with few reports in the literature. The current report highlights an unusual presentation of a rare complication of sphenoiditis. In our patient, ophthalmalgia and visual change, including blurring and loss of vision, were the most prominent features. Interestingly, this did not appear to be due to direct involvement of the optic nerve. Two forms of visual loss are seen as a complication of sinusitis: (1) visual loss as a complication of orbital cellulitis, with orbital soft tissue involvement characterised by proptosis, oedema, chemosis and ophthalmoplegia;5 and (2) visual loss as a component of ‘orbital apex syndrome’, characterised by involvement of vessels and nerves passing through the superior orbital fissure.2,6 In the fully developed orbital apex syndrome, visual loss is accompanied by complete ophthalmoplegia (with paresis of the IIIrd, IVth and VIth cranial nerves). However, there can be visual loss alone, with no, or only minimal, orbital inflammatory signs. This is thought to be due to a segment of the optic nerve being affected, and is considered as ‘partial’ or ‘posterior’ orbital cellulitis. Reports of sinusitis causing orbital apex syndrome are relatively rare. The term orbital apex syndrome is applied when all structures of the optic canal and superior orbital fissure are affected. When there are minimal signs of orbital inflammation, the term ‘posterior orbital cellulitis’ has been applied. In this context, Slavin and Glaser described three cases of spheno-ethmoiditis causing irreversible visual loss with minimal inflammation. They defined this condition as a clinical syndrome in which early, severe vision loss overshadows accompanying orbital inflammatory signs.7 However, there are no reports describing fronto-sphenoiditis presenting in this way. A study in China assessed patients presenting with orbital apex syndrome as a complication of sphenoid disease. All the patients required sphenoid surgery, and all had visual symptoms at presentation but were misdiagnosed until imaging results became available.8 As in the present case, the authors of the Chinese study emphasised that the diagnosis of orbital apex syndrome can be easily missed, due to its relatively low incidence, non-specific orbital signs and lack of supporting clinical evidence. The absence of nasal symptoms does not preclude sphenoiditis. Theoretically, any of the structures related to the
K ALLAN, H ATKINSON, F AGADA
sphenoid can be involved. Proetz listed 13 structures adjacent to the sphenoid sinus which may be affected: cranial nerves II, III, IV, V and VI; dura mater; pituitary gland; cavernous sinus; internal carotid artery; the sphenopalatine ganglion and artery; and the pterygoid canal and nerve.9 In the presented case, the presence of visual loss in the absence of inflammatory signs would suggest a diagnosis of posterior orbital cellulitis, with involvement of a segment of the optic nerve only. • Posterior orbital cellulitis presenting with visual loss and peri-orbital pain is reported • Imaging showed right frontal and sphenoid sinus opacification • Urgent right sphenoidotomy and frontal sinus trephination were performed, following poor antibiotics response and worsening symptoms • A high index of clinical suspicion is necessary for diagnosis • Early, aggressive treatment is imperative to avert irreversible blindness Various dilemmas may attend the diagnosis of acute sphenoiditis. Flexible nasendoscopy may reveal purulent drainage from the sphenoid ostium of the nasopharynx, but this was not apparent in the present case. The diagnostic study of choice is high resolution CT. This investigation may reveal the presence of fluid or opacification. Staphylococcus aureus is the most common cause of acute sphenoiditis, followed by streptococcal species. Gram-negative bacteria and anaerobes are occasionally cultured.10 Fungi, especially aspergillus, must be considered in all patients, particularly the immunocompromised. In the presented case, microbial cultures of samples obtained intraoperatively were relatively unremarkable. Uncomplicated cases of acute sphenoiditis may resolve with antibiotic therapy, if diagnosed and treated early. If symptoms continue or progress, immediate surgical drainage of the sinus is indicated, as seen in the present case.
Conclusion Acute fronto-sphenoiditis is uncommon, especially with no ethmoidal or maxillary sinus involvement, as in the present case. A high index of clinical suspicion is necessary, as it is well documented that sphenoiditis may present with subtle symptoms and elusive physical findings. Early, aggressive treatment is imperative in order to avert irreversible blindness. Surgery is the key to preventing significant morbidity when antibiotics fail to abate visual loss. References 1 Van Alyea OE. Sphenoid sinus: anatomic study, with consideration of the clinical significance of the structural characteristics of the sphenoid sinus. Arch Otolaryngol 1941;34: 225–53 2 Holt GR, Standefer JA, Brown WE Jr, Gates GA. Infectious diseases of the sphenoid sinus. Laryngoscope 1984;94:330–5 3 Lew D, Southwick FS, Montgomery WW, Weber AL, Baker AS. Sphenoid sinusitis. A review of 30 cases. N Engl J Med 1983;309:1149–54 4 Pearlman SJ, Lawson W, Biller HF, Friedman WH, Potter GD. Isolated sphenoid sinus disease. Laryngoscope 1989;99:716–20
1151
CLINICAL RECORD
5 Chandler JR, Langenbrunner DJ, Stevens ER. The pathogenesis of orbital complications in acute sinusitis. Laryngoscope 1970; 80:1414–28 6 Kjoer I. A case of orbital apex syndrome in collateral pansinusitis. Acta Ophthalmologica 1945;23:357–66 7 Slavin ML, Glaser JS. Acute severe irreversible visual loss with sphenoethmoiditis – ‘posterior orbital cellulitis’. Arch Ophthalmol 1987;105:345–8 8 Guo YF, Yuan YM, Liu ZP, Xu BC, Guan J. Clinical analysis of sphenoid disease manifesting as orbital apex syndrome [in Chinese]. Zhonghua Er Bi Yan Hou Tou Jing Wai Ke Za Zhi 2005;40:688–90 9 Proetz AW. The sphenoid sinus. Br Med J 1948;2:243–5 10 Brook I. Bacteriology of acute and chronic sphenoid sinusitis. Ann Otol Rhinol Laryngol 2002;111:1002–4
Address for correspondence: Dr K Allan, ENT, Head and Neck Surgery Department, York Teaching Hospitals NHS Trust, Scarborough Hospital, Woodlands Drive, Scarborough YO12 6QL, UK E-mail: [email protected]
Dr K Allan takes responsibility for the integrity of the content of the paper Competing interests: None declared
The Journal of Laryngology & Otology (2013), 127, 1148–1151. © JLO (1984) Limited, 2013 doi:10.1017/S0022215113002181
Posterior orbital cellulitis: case report and literature review K ALLAN, H ATKINSON, F AGADA ENT, Head and Neck Surgery Department, York Teaching Hospitals NHS Trust, Scarborough Hospital, UK
Abstract Introduction: It is rare for isolated sphenoid sinusitis to cause orbital cellulitis. We present a rare case of posterior orbital cellulitis, so caused, together with a review of the relevant literature. Case report: A 29-year-old woman presented with a 6-week history of progressive, unilateral, retro-orbital and periorbital right eye pain. On examination, the only finding was reduced visual acuity in the right eye. A computed tomography scan demonstrated right frontal and sphenoid sinus opacification. Sphenoidotomy and frontal sinus trephination were subsequently performed, following failure to respond to intravenous antibiotics. After surgery, the patient’s vision returned to normal. Conclusion: Isolated sphenoid sinusitis is rare but can cause significant visual disturbance and permanent loss of vision. Vague symptoms unsupported by clinical signs at presentation are a feature of posterior orbital cellulitis. The presented case highlights the problem, and the need for a high index of clinical suspicion even in the absence of firm clinical signs, in order to prevent permanent visual loss. Key words: Sphenoid Sinus; Sinusitis; Orbital Cellulitis; Diagnosis
Introduction Sinusitis in adults is common, but orbital complications are usually associated with paediatric rather than adult patients. There is a debate as to the difference between orbital apex syndrome and posterior orbital cellulitis. In the latter condition, a segment of the optic nerve in the superior orbital fissure is affected. This paper reports a rare occurrence of isolated sphenoid sinusitis with associated presentation of posterior orbital cellulitis. The subtlety of eye signs seen in the condition necessitates a high index of clinical suspicion in order to avoid missing the diagnosis.
Case report A 29-year-old woman presented as an acute medical admission with a 6-week history of progressive, severe, retroorbital and peri-orbital right eye pain. The pain was throbbing in nature, increasing in severity, and associated with headaches, nausea and vomiting. The patient’s vision was assessed by the ophthalmologists, revealing blurring, reduced acuity and associated photophobia. There were no other signs of meningism. Initial examination demonstrated right-sided ptosis and tenderness on palpation of the orbit. The patient had demonstrably reduced acuity but maintained normal colour vision. Inflammatory markers were within normal limits. An urgent magnetic resonance imaging brain scan was performed. This showed right sphenoid sinusitis, with sphenoidal and frontal sinus mucosal thickening (Figure 1). Accepted for publication 8 April 2013
FIG. 1 Axial magnetic resonance image showing right sphenoid sinusitis, with opacification and right sphenoid and frontal sinus mucosal thickening.
First published online 29 October 2013
CLINICAL RECORD
Computed tomography (CT) scanning of the sinuses was requested to further define the mucosal disease. This demonstrated right frontal and sphenoid sinus opacification only, with no evidence of maxillary, ethmoidal or contralateral disease (Figure 2). Following the microbiologist’s advice, the patient was started on intravenous Timentin (GSK, Uxbridge, UK). After 48 hours of IV antibiotics, her vision continued to deteriorate, and her ophthalmalgia and photophobia were unchanged. Ophthalmological reassessment revealed reduced acuity: 6/36 for the right eye, compared with 6/6 for the left. Important negative findings included no visible proptosis or chemosis, no demonstrable relative afferent pupillary defect, no reduction in external ocular movements, and no
1149 diplopia. The patient’s optic discs were healthy, with no swelling or venous engorgement. The patient began to develop red desaturation. Two diagnoses were considered by ophthalmology at this point: orbital soft tissue inflammation secondary to sinus infection, and idiopathic orbital inflammatory disease (a diagnosis of exclusion). Right-sided sphenoidotomy and frontal sinus trephination were performed urgently. Pus under pressure drained rapidly from the right sphenoid sinus and was sent for culture and sensitivity tests. The frontal sinus was full of polypoidal mucosa. Microbial culture grew Gram-positive bacilli only; culture of the frontal sinus sample grew no organisms after 48 hours.
FIG. 2 (a) Coronal computed tomography (CT) scan showing right sphenoiditis, with clear ethmoidal sinuses. (b) Axial CT showing right frontal sinus opacification. (c) Axial CT showing sphenoid sinusitis, with clear ethmoid sinuses. (d) Axial CT showing clear maxillary sinuses.
1150 Post-operatively, the patient’s visual acuity improved slowly. Three months after surgery, her vision had returned to normal.
Discussion The sphenoid sinus is lined with pseudostratified epithelium, with fewer mucus-secreting cells compared with other paranasal sinuses.1 Predisposing factors for sphenoid sinusitis include anatomical variation, radiotherapy, immunosuppression and malignancy.2–4 In the presented case, however, no predisposing factor was found. Headache is usually the most common initial symptom of acute sphenoiditis.1 In our patient, there was significant right orbital pain that was predominately retro-bulbar. Sphenoiditis is most often non-specific and poorly localised, due to its inaccessible position. Symptoms are often non-specific.2 Rhinorrhoea and post-nasal drip are common symptoms, with purulent exudates frequently seen below the middle and superior turbinates.3 Visual loss as the presenting symptom is extremely rare, with few reports in the literature. The current report highlights an unusual presentation of a rare complication of sphenoiditis. In our patient, ophthalmalgia and visual change, including blurring and loss of vision, were the most prominent features. Interestingly, this did not appear to be due to direct involvement of the optic nerve. Two forms of visual loss are seen as a complication of sinusitis: (1) visual loss as a complication of orbital cellulitis, with orbital soft tissue involvement characterised by proptosis, oedema, chemosis and ophthalmoplegia;5 and (2) visual loss as a component of ‘orbital apex syndrome’, characterised by involvement of vessels and nerves passing through the superior orbital fissure.2,6 In the fully developed orbital apex syndrome, visual loss is accompanied by complete ophthalmoplegia (with paresis of the IIIrd, IVth and VIth cranial nerves). However, there can be visual loss alone, with no, or only minimal, orbital inflammatory signs. This is thought to be due to a segment of the optic nerve being affected, and is considered as ‘partial’ or ‘posterior’ orbital cellulitis. Reports of sinusitis causing orbital apex syndrome are relatively rare. The term orbital apex syndrome is applied when all structures of the optic canal and superior orbital fissure are affected. When there are minimal signs of orbital inflammation, the term ‘posterior orbital cellulitis’ has been applied. In this context, Slavin and Glaser described three cases of spheno-ethmoiditis causing irreversible visual loss with minimal inflammation. They defined this condition as a clinical syndrome in which early, severe vision loss overshadows accompanying orbital inflammatory signs.7 However, there are no reports describing fronto-sphenoiditis presenting in this way. A study in China assessed patients presenting with orbital apex syndrome as a complication of sphenoid disease. All the patients required sphenoid surgery, and all had visual symptoms at presentation but were misdiagnosed until imaging results became available.8 As in the present case, the authors of the Chinese study emphasised that the diagnosis of orbital apex syndrome can be easily missed, due to its relatively low incidence, non-specific orbital signs and lack of supporting clinical evidence. The absence of nasal symptoms does not preclude sphenoiditis. Theoretically, any of the structures related to the
K ALLAN, H ATKINSON, F AGADA
sphenoid can be involved. Proetz listed 13 structures adjacent to the sphenoid sinus which may be affected: cranial nerves II, III, IV, V and VI; dura mater; pituitary gland; cavernous sinus; internal carotid artery; the sphenopalatine ganglion and artery; and the pterygoid canal and nerve.9 In the presented case, the presence of visual loss in the absence of inflammatory signs would suggest a diagnosis of posterior orbital cellulitis, with involvement of a segment of the optic nerve only. • Posterior orbital cellulitis presenting with visual loss and peri-orbital pain is reported • Imaging showed right frontal and sphenoid sinus opacification • Urgent right sphenoidotomy and frontal sinus trephination were performed, following poor antibiotics response and worsening symptoms • A high index of clinical suspicion is necessary for diagnosis • Early, aggressive treatment is imperative to avert irreversible blindness Various dilemmas may attend the diagnosis of acute sphenoiditis. Flexible nasendoscopy may reveal purulent drainage from the sphenoid ostium of the nasopharynx, but this was not apparent in the present case. The diagnostic study of choice is high resolution CT. This investigation may reveal the presence of fluid or opacification. Staphylococcus aureus is the most common cause of acute sphenoiditis, followed by streptococcal species. Gram-negative bacteria and anaerobes are occasionally cultured.10 Fungi, especially aspergillus, must be considered in all patients, particularly the immunocompromised. In the presented case, microbial cultures of samples obtained intraoperatively were relatively unremarkable. Uncomplicated cases of acute sphenoiditis may resolve with antibiotic therapy, if diagnosed and treated early. If symptoms continue or progress, immediate surgical drainage of the sinus is indicated, as seen in the present case.
Conclusion Acute fronto-sphenoiditis is uncommon, especially with no ethmoidal or maxillary sinus involvement, as in the present case. A high index of clinical suspicion is necessary, as it is well documented that sphenoiditis may present with subtle symptoms and elusive physical findings. Early, aggressive treatment is imperative in order to avert irreversible blindness. Surgery is the key to preventing significant morbidity when antibiotics fail to abate visual loss. References 1 Van Alyea OE. Sphenoid sinus: anatomic study, with consideration of the clinical significance of the structural characteristics of the sphenoid sinus. Arch Otolaryngol 1941;34: 225–53 2 Holt GR, Standefer JA, Brown WE Jr, Gates GA. Infectious diseases of the sphenoid sinus. Laryngoscope 1984;94:330–5 3 Lew D, Southwick FS, Montgomery WW, Weber AL, Baker AS. Sphenoid sinusitis. A review of 30 cases. N Engl J Med 1983;309:1149–54 4 Pearlman SJ, Lawson W, Biller HF, Friedman WH, Potter GD. Isolated sphenoid sinus disease. Laryngoscope 1989;99:716–20
1151
CLINICAL RECORD
5 Chandler JR, Langenbrunner DJ, Stevens ER. The pathogenesis of orbital complications in acute sinusitis. Laryngoscope 1970; 80:1414–28 6 Kjoer I. A case of orbital apex syndrome in collateral pansinusitis. Acta Ophthalmologica 1945;23:357–66 7 Slavin ML, Glaser JS. Acute severe irreversible visual loss with sphenoethmoiditis – ‘posterior orbital cellulitis’. Arch Ophthalmol 1987;105:345–8 8 Guo YF, Yuan YM, Liu ZP, Xu BC, Guan J. Clinical analysis of sphenoid disease manifesting as orbital apex syndrome [in Chinese]. Zhonghua Er Bi Yan Hou Tou Jing Wai Ke Za Zhi 2005;40:688–90 9 Proetz AW. The sphenoid sinus. Br Med J 1948;2:243–5 10 Brook I. Bacteriology of acute and chronic sphenoid sinusitis. Ann Otol Rhinol Laryngol 2002;111:1002–4
Address for correspondence: Dr K Allan, ENT, Head and Neck Surgery Department, York Teaching Hospitals NHS Trust, Scarborough Hospital, Woodlands Drive, Scarborough YO12 6QL, UK E-mail: [email protected]
Dr K Allan takes responsibility for the integrity of the content of the paper Competing interests: None declared
