Following a Mycoplasmal Infection

BritishJournalofOphthalmology 1993; 77:673-676 673

CASE REPORTS Br J Ophthalmol: first published as 10.1136/bjo.77.10.673 on 1 October 1993. Downloaded from

Bilateral optic neuropathy and white dot syndrome following a mycoplasmal infection

M Cordonnier, L E Caspers-Velu, C Jacquemin, C Van Nechel, M Tombroff

Little has been written about Mycoplasma virus, influenza A and B, para-influenza, pneumoniae and ophthalmology. It is mentioned syncytial respiratory virus, enterovirus and as an agent that may cause conjunctivitis.'2 adenovirus, HIV, HBS, syphilis, psittacosis, Salzmann et al3 recently stressed the fact that ornithosis, Legionella, Chlamydia, Yersinia, and Mycoplasma pneumoniae should be considered in Borrelia. However, on 2 days of December - that the differential diagnosis of any febrile illness is, 2 weeks after the flue-like illness, the anti- that is accompanied by uveitis or optic disc Mycoplasma antibody titre was 1/320, and the oedema. The current report describes a case of cold haemagglutinin titre was 1/128. One bilateral optic disc swelling and choroidal lesions month later, the anti-Mycoplasma antibody titre occurring after a mycoplasmal infection. was higher than 1/1280. This level stayed unchanged in March and June 1992. The cold haemagglutinin titre dropped to 1/8 at the Case report beginning of January 1992 and to ½/2 in March A 52-year-old woman, without previous medical 1992. history, was admitted on 1 December 1991 for Methanol was absent in blood. A mito- sudden blindness in the left eye being preceded chondrial DNA study did not show any mutation 24 hours before by the same event in the right responsible for Leber optic neuropathy. eye. This blindness was accompanied by photopsias. The patient also complained of headache, numbness, and paraesthesia ofall four RADIOLOGICAL FINDINGS limbs. She did not drink, or smoke, and took no The chest x ray was normal. Cerebral magnetic medication on a regular basis. She had just resonance imaging performed the day after

recovered from a flu-like illness that lasted 8 days admission showed multiple hyperintense foci http://bjo.bmj.com/ during which she took minicycline, amoxy- bilaterally and symmetrically in the supra- cilin, and aspirin. Three days before the loss of tentorial white matter, away from the ventricles. vision, she had returned to work, although she All of them were enhanced by contrast. They still had a slightly productive cough. were observed every month until May 1992. At Physical examination was unremarkable, that time, cerebral magnetic resonance imaging included pulmonary and ear, nose, and throat had returned to normal.

examination. Neurological examination showed on September 28, 2021 by guest. Protected copyright. very brisk myotatic reflexes of the four limbs. There were no meningeal signs. Besides bilateral OTHER INVESTIGATIONS blindness and pupillary areflexia, other cranial Lower limb somaesthesic evoked potentials nerves were unaffected. The fundus showed showed bilateral slowing of conduction velocity bilateral hyperaemic and blurred papillae, within December 1991. Acoustic evoked potentials Neuro-Ophthalmology out any haemorrhage or exudate. were normal at that time. Six months later, the Unit, Hopital Erasme, somaesthesic evoked potentials had returned to Cliniques Universitaires normal. de Bruxelles, Brussels, Belgium SUCCESSIVE BLOOD AND CEREBROSPINAL FLUID N Cordonnier FINDINGS C Van Nechel Two lumbar punctures revealed an increased OPHTHALMIC FINDINGS Hopital E Cavell, level of protein (71 mg/100 ml on 2 days of Throughout 10 months of follow up, visual Brussels, Belgium December and 79 mg/100 ml 1 month later) with acuity was reduced to no light perception in the Ophthalmology no cells. Protein electrophoresis (agar and eye. In the left visual Department right eye, acuity developed L E Casper-Velu electrofocusing) was normal as was glucose very slowly from no light perception to slight concentration. Blood analysis showed no inflam- perception ofhand movements at 50 cm. Radiology Department matory component. There were no LE cells or Bilateral papilloedema was initially described, C Jacquemin antinuclear antibodies. Immunoglobulins were without haemorrhage or exudate. Biomicro- Internal Medicine normal. Protein electrophoresis and immuno- scopic examination 1 month after admission Department electrophoresis were normal. Circulating found the initial optic atrophy but a normal M Tombroff immune complexes were found (fivefold the retinal and vitreous aspect. Between the third Correspondence to: Dr M Cordonnier. normal maximal level). Early and late viral and and the fourth month after admission, biomicro- tests were Accepted for publication bacteriological negative for infectious scopic examination revealed bilateral optic 28 April 1993 mononucleosis, herpes simplex, cytomegalo- atrophy and disclosed retinal thickening, involv- 674. Cordonnier, Caspers-Velu,Jfacquemin, Van Nechel, Tombroff

ing both posterior poles as well as white spots in the middle periphery at the level of the choroid (Fig 1). Fluorescein angiography revealed multiple punctate early hyperfluorescence spots Br J Ophthalmol: first published as 10.1136/bjo.77.10.673 on 1 October 1993. Downloaded from (Fig 2), without cystoid pattern. Seven months after admission, these yellow-white spots were still more numerous in both mid peripheries (Fig 3). Very tiny white spots were also disseminated on both posterior poles (Fig 4). The retinal thickening was not apparent. Fluoroangio- graphy revealed early mottled hyperfluorescence of the whole posterior pole and mid periphery (Figs 5A, SB) accentuating later (Figs SC, SD). The visual evoked potentials were flat. Figure I Red-free picture Electroretinography performed 5 months after ofthe leftfundus (4 months admission was normal. Electro-oculography was after the acute illness): white spots are visible in mid- impossible owing to absence of adequate periphery. fixation.

TREATMENT During the acute phase, intravenous prednisolone was given (100 mg/day) for 10 days and then gradually tapered off until the end of March. Erythromycin was added during the second week of treatment (1-5 g/day) for 11 days. Because no visual improvement occurred, a third treatment was tried at the beginning of January: 400 mg/kg/day intravenous immunoglobin (Sandoglobin) for 1 week. This was done twice at 3 week interval, and led to a slight improvement in the visual acuity of the left eye (the patient started to see shadows instead of no light Figure 2 perception). Fluoroangiographic picture ofthe left posterior pole: multiple hyperfluorescent spots. Comment Neurological complications of mycoplasmal infection are rare. Ponka4 found 4 8% of central nervous system manifestation in hospitalised patients with serologically verified Mycoplasma http://bjo.bmj.com/ pneumoniae infection. Clyde'6 suggested several criteria that should be met in proving the relationship between neurological syndromes and mycoplasmal infections: epidemic period of mycoplasmal infection, appropriate age ofthe patient (greatest Figure 3 Leftfundus (7 incidence between 5-40 years, while any age on September 28, 2021 by guest. Protected copyright. months after the acute illness):yellow-white group may be involved), appropriate season choroidal spots in mid- (more common in autumn and early winter), periphery. compatible clinical illness (febrile tracheo- bronchitis, influenza-like syndrome, or atypical pneumonia), and serological findings compatible with mycoplasmal infection (fourfold or greater rise in specific antibody titre between properly timed acute and convalescent phase sera; fourfold or greater rise or fall in cold haemagglutinins titre). This case fulfils the criteria for linking the neurological disease to Mycoplasma pneumoniae. - There is presently in Europe a Mycoplasma epidemic that started in 1991.7 - This patient had, during the autumn, an influenza-like syndrome that is compatible with a mycoplasmal infection. - Neurological symptoms appeared within 14 days of the acute illness. Figure 4 Red-free - There was a more than fourfold rise in anti- photograph ofthe right Mycoplasma antibody titre in paired serums fundus: tiny white spots are disseminated on the posterior made at 1 month intervals, the first serum being pole. taken 2 weeks after the influenza-like syndrome. Bilateral optic neuropathy and white dot syndromefollowing a mycoplasmal infection 675 Br J Ophthalmol: first published as 10.1136/bjo.77.10.673 on 1 October 1993. Downloaded from

Fig SA Fig SB

Fig SC Fig5D

Figure S Early and latefluoroangiographic picture ofthe rightposteriorpole (A-C) andperipapillary area (B-D). http://bjo.bmj.com/

Moreover, there was an elevated titre of cold They include multiple evanescent white dot haemagglutinins 2 weeks after the influenza-like syndrome (MEWDS), multifocal choroiditis and syndrome and a more than fourfold fail 4 weeks panuveitis, acute retinal pigment epithelitis,

later. acute posterior multifocal placoid pigment on September 28, 2021 by guest. Protected copyright. The description of an optic neuropathy after epitheliopathy (APMPPE), and subretinal mycoplasmal infection is not new and we have fibrosis and uveitis syndrome. found several such cases in the published reports None of these entities seems to correspond to (eight to 19), most of them having optic disc our case: unlike in MEWDS, the white dots of swelling. According to the literature, the con- our case are choroidal rather than deep retinal sequences may vary from irreversible blindness and are not evanescent. The other entities have to complete recovery. As in our case, the optic obviously different ophthalmoscopic and fluoro- neuropathy usually accompanies other neuro- angiographic features and we will not consider logical findings (meningoencephalitis, peripheral them in detail. The delayed appearance (3 and/or cranial nerve neuropathy, cerebellar months after admission) of these white choroidal ataxia, transverse myelitis). The pathogenesis of dots and retinal thickening is surprising. The central nervous system involvement is still corticosteroid treatment and the immunological unknown but is presently regarded as an disorder caused by Mycoplasma may have immunological response to extraneural infection: favoured another infection that was clinically Mycoplasma infection may induce the develop- silent but induced a white dot syndrome. On the ment of antibodies for normal tissues and other hand, both choroidal dots and retinal Mycoplasma itselfinfluences the activity ofT and thickening appeared when steroid dosage was B lymphocytes.20 very low (10 mg prednisolone every other day) There are several entities that have been and 1 month after intravenous immunoglobulin characterised as essentially presenting at some treatment was over. If these fundus anomalies time with multiple white dots in the fundus, are related to some immunological disorder usually in the deeper layers of the posterior induced by Mycoplasma itself, steroids may have segment. Nussenblatt2" gathers these entities protected the eye from the early development of together under the name of'white dot syndrome'. this white dot syndrome. Intravenous immuno- 676 Cordonnier, Caspers-Velu,Jfacquemin, VanNechel, Tombroff

9 Novelli VM, MarshallWC. Opticdisc swelling andMycoplasma globulin therapy may also have played a protec- pneumoniae. PediatrInfectDis 1984; 3: 597. tive role through its immunoregulatory effect.22 10 Michel D, Laurent B, Granouillet R, Gaudin-Terrasse OD.

Infections aiguees, reentes, et parfois persistantes a Br J Ophthalmol: first published as 10.1136/bjo.77.10.673 on 1 October 1993. Downloaded from Mycoplasma pneumoniae, associees a des manifestations neurologiques. Rev Neurol (Paris) 1981; 137: 6-7. Conclusion 11 BehanPO, FeldmanRG, SegerraJM, DraperIT. Neurological aspects of mycoplasmal infection. Acta Neurol Scand 1986; This case reaffirms that Mycoplasma pneumoniae 74: 314-22. should be considered in the differential diagnosis 12 Yesnick L. Central nervous system complications of primary atypical pneumonia. Arch Intern Med 1956; 97: 93-8. of an optic neuropathy preceded by a febrile 13 Verin Ph, Vildy A, Cales R, Bapt J-B. Une nouvelle etiologie illness. Moreover, it suggests that Mycoplasma de l'oedeme des voies optiquespregeniculees: le Mycoplasma pneumoniae. BullSoc OphtalmolFr 1980; 11: 1009-12. may play a role in the induction of a white dot 14 Lerer RJ, Kalavsky SM. Central nervous system disease syndrome. associated with Mycoplasma pneumoniae infection. Report of five cases and review of the literature. Pediatrics 1973; 52: 658-68. 15 Yamamoto T, Inoue N, Yamashita Y, Shiraishi S, Murai Y. A 1 Murray HW, Masur H, Senterfit LB, Roberts RB. The case of bilateral optic neuritis and polyneuritis associated protean manifestations ofMycoplasma pneumoniae infection with Mycoplasma pneumoniae infection. Rinsho-Shinkeigaku in adults. AmJ Med 1975; 58: 229-42. 1983; 23: 404-9. 2 Tuazon CU, Murray HW. Atypical pneumonias. In: 16 Baker AB, Noran HH. Changes in the central nervous system Pennington JE, ed. Respiratory infections: diagnosis and associated with encephalitis complicating pneumonia. Arch management. 2nd ed. New York: Raven Press, 1988: 346. Intern Med 1945; 76: 146-53. 3 Salzman MB, Sood SK, Slavin ML, Rubin LG. Ocular 17 Robert H, Fischer C, Cloppet H, Trillet M, Schott B. Un cas manifestations of Mycoplasma pneumoniae infection. Clin d'atteinte neurologique (encephalite et nevrite optique) au InfectDis 1992; 14: 1137-9. cours di'une infection a Mycoplasma pneumoniae. Lyon Med 4 Ponka A. Central nervous system manifestations associated 1981; 2:67-70. with serologically verified Mycoplasmapneunoniae infection. 18 Rothstein EL, Kenny GE. Cranial neuropathy, myelo- Scand Infect Dis 1980; 12: 175-84. radiculopathy, and myositis complications of Mycoplasma 5 Clyde WA, Jr. Neurological syndromes and mycoplasmal pneumoiae infection. Arch Neurol 1979; 36: 476-7. infections. Arch Neurol 1980; 37: 65-6. 19 Fisher RS, Clark AW, Wolinksy JS, Parhad IM, Moses H, 6 Clyde WA, Jr. Mycoplasmal diseases of the central nervous Mardiney MR. Postinfectious leukoencephalitis compli- system. In: Scheld WM, Whitley RJ, Durack DT, eds. cating Mycoplasma pneumoniae infection. Arch Neurol 1983; Infections of the central nervous system. New York: Raven 40: 109-13. Press, 1991: 283-93. 20 Mandell GL, Douglas GR, Bennett JE. Principles and practice 7 Anonymous. Mycoplasma pneumoniae. Lancet 1991; 337: of infectious diseases. 3rd ed. Edinburgh: Churchill 651-2. Livingstone, 1990: 1450. 8 Garcia CP, Petri EM, Iturralde RG, Bragado FG, Urech ET, 21 Nussenblatt RB, Palestine AG. Uveitis. Fundamental and Rivero Puente MI MA. Sindrome de Devic complicando una clinicalpractice. Chicago: Year Book 1989: 291-308. infecci6n por Mycoplasma pneumoniae. Rev Clfn Esp 1987; 22 Dwyer JM. Manipulating the immune system with immune 181:29-31. globulin. N EnglJf Med 1992; 326: 107-16. http://bjo.bmj.com/

BritishJournal ofOphthalmology 1993; 77: 676-677

Orbital masquerade: hyperthyroidism and on September 28, 2021 by guest. Protected copyright. cavernous haemangioma of the orbit

Martin L Leib

Orbit and Plastics Service, the Edward S Unilateral exophthalmos is a challenging clinical Harkness Eye Institute, 1982 with a several week history of intermittent Columbia-Presbyterian problem, and hyperthyroidism, statistically, is blurred vision in her right eye. Medical Center and the one of the most common causes in the adult Her ocular history included variable exoph- College of Physicians and population. Surgeons, Columbia thalmos in the right eye that was associated University, New York, The possibility of other orbital inflammations previously with documented hyperthyroidism of USA or neoplasms coexisting with hyperthyroidism 3 years' duration. She had received a short course M L Leib requires emphasis. In this report a case is ofpropylthiouracil and propranolol, and is pres- Correspondence to: presented where the patient had a history Martin L Leib, MD, of ently euthyroid. Director, Orbit and Plastics chemical hyperthyroidism and cavernous On ophthalmic examination, her best cor- Service, The Edward S haemangioma of the orbit previously unrecog- Harkness Eye Institute, rected visual acuity was 20/20+2 right eye and Columbia-Presbyterian nised. 20/15-1 left eye. The pupils were normal and the Medical Center, 635 West 165th Street, New York, New extraocular movements were full. She was ortho- York 10032, USA. phoric at distance and had 6 prism dioptres of Accepted for publication Case report exophoria at near. No diplopia could be elicited. 18 May 1993 A 32-year-old woman presented on 15 September Orbital examination revealed mild resistance