Vogt-Koyanagi-Harada disease during pregnancy

rence.4 5 Because the results reported byseveral authors cannot be equally comparedbecause of differences in their detailed recordsof courses of diseases, it is hard to select aunique factor affecting the ophthalmic findingsin these patients.

Recently, massive corticosteroid treatmentof VKH was clinically reinvestigated.6Yamamoto et al6 found a similarity in theprognosis of patients treated with non-steroidal anti-inflammatory agents and thosetreated with massive doses of systemiccorticosteroids. Therefore, it was difficult todetermine ifthe remission ofthe disease duringpregnancy m our patient was spontaneous ordue to the pregnancy.

Although a good clinical result has been

obtained without the systemic administrationof high doses of corticosteroids or topicalcorticosteroids on the basis of careful examina-tions of a patient with VKH in early pregnancy,further discussions will be required about itstreatment during pregnancy.

1 Sugiura S. Vogt-Koyanagi-Harada disease. Jpn J Ophthalmol1978; 22: 9-35.

2 Steahly LP. Vogt-Koyanagi-Harada syndrome and preg-nancy. Ann Ophthalmol 1990; 22: 59-62.

3 Snyder DA, Tessler HH. Vogt-Koyanagi-Harada syndrome.Am J Ophthalmol 1980; 90: 69-75.

4 Friedman Z, Granat M, Neumann E. The syndrome ofVogt-Koyanagi-Harada and pregnancy. Metab PediatrOphthalmol 1980; 4: 147-9.

5 Satoh S, Koh M, Tamura H. Harada's disease in pregnancy,a case report. Folia OphthalmolJpn 1986; 37: 46-50.

6 Yamamoto T, Hayakawa K, Hatano H, Kamata K, Sasaki T.Clinical course of Harada's disease, a critical evaluation ofmassive corticosteroid therapy. Jpn J Clin Ophthalmol1983; 37: 137-42.

British Journal of Ophthalmology 1995; 79: 95-96

Fatal bilateral necrotising fasciitis of the eyelids

Department ofOphthalmology, King'sCollege Hospital,Denmark Hill, LondonSE5 9RSD KentP L AtkinsonEW G Davies

Department ofMedical Microbiology,King's College SchoolofMedicine andDentistry, BessemerRoad, London SE5 9PJB Patel

Correspondence to:Dr D Kent, Department ofOphthalmology, Link 8Z,Royal Liverpool UniversityHospital, Prescot Street,Liverpool L7 8XP.Accepted for publication5 September 1994

D Kent, P L Atkinson, B Patel, EW G Davies

Necrotising fasciitis is an uncommon softtissue infection usually affecting the trunk,perineum, or legs after surgery or trauma.Infection spreads rapidly along subcutaneousfascial planes and produces overlying skinnecrosis. Head and neck involvement is rare,with scalp and neck fasciitis almost always aftertrauma or dental sepsis. Midfacial and peri-orbital infection is notable in that a history oftrauma may be absent or such trauma may beminor. We report a fatal case ofbilateral necro-tising fasciitis of the eyelids with no history ofpreceding trauma in which Streptococcuspyogenes (j3 haemolytic streptococcus

Figure 1 Severe bilateral gangrene of the periorbital skin and subcutaneous tissue.

Lancefield group A) waseyelids, blood, and throat.

cultured from the

Case reportA 40-year-old man with a history of alcoholicliver disease presented with a 24 hour historyof increasing bilateral painful periocular andfacial swelling. On examination he wasafebrile, mildly jaundiced with ascites andhepatomegaly. The lid skin was markedlyswollen with a small patch of dusky skin on theright lower lid. Intravenous benzylpenicillinand flucloxacillin were commenced and thefacial swelling began to subside over the next24 hours so the eyes could be opened revealingnormal globes. However, within 48 hours thelid skin became dusky, and progressed to franklid gangrene (Fig 1). Investigations revealedanaemia (haemoglobin 9-7 g/dl), a white bloodcount of 8*2X 109/1, thrombocytopenia(82X109/l) and an abnormal clotting screen(INR 2.4). Streptococcus pyogenes, sensitive topenicillin and erythromycin, was isolated fromthe cultures of the eyelids, blood, and throat. Adiagnosis of necrotising fasciitis was made.Clinically the patient began to deteriorate andsurgical debridement was delayed until 7 daysafter presentation. Under local anaesthesia allnecrotic periorbital skin and subcutaneoustissue were debrided revealing healthy orbicu-laris muscle beneath (Fig 2). However, renaland circulatory failure developed and, despiteintensive supportive measures, he died 15 daysafter his admission.

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Kent, Atkinson, Patel, Davies

*S*t@+^r'to80,et,i'4ts o s; X ~ I - *, . 'I ., .................

Figure 2 Appearance following debnidement of all necrotic tissue.

.~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~------;----iM.

Figure 3 Intense acute inflammatory response with piolymorphonuclear leucocytesinfiltrating between striated muscle fibres (haematoxylin and eosin, x 83).

Histology of the debrided tissue showed fullthickness necrosis of the skin and underlyingfat with a diffuse heavy infiltrate of neutrophilswith sparse Gram positive cocci (Fig 3).Subsequent typing of the organism showed itto be a Streptococcus pyogenes type Ml, T1.

CommentNecrotising fasciitis is a soft tissue infectioncaused by group A ,B haemolytic Streptococcuseither alone or in combination with other organ-isms, most commonly with Staphylococcusaureus.1 Progression is rapid, with initialerythema and tenderness becoming frankgangrene in as little as 24 hours. Spread along

subcutaneous planes results in necrosis offascia, fat, and overlying skin but usually withpreservation of underlying muscle. Systemicupset is characteristically severe with fever,neutropenia, and septicaemia which mayprogress to multiple organ failure and death.

Necrotising fasciitis without precedingtrauma or surgery is rare, but has beenreported in three cases of periorbital infec-tion.2 3 In this case infection could haveoccurred via unnoticed skin trauma or sepsis,or possibly haematogenous spread from thepharynx.

Invasive disease due to group A Streptococcushas been reported more frequently since thelate 1980s and this may be due to a change inserotype distribution, with the more invasivetypes Ml, 3, and 18 being more prevalent (Mprotein is an organism cell wall constituent4).The rapid progression and spread seen innecrotising fasciitis may well be related to theexotoxins produced by the Streptococcus suchas pyrogenic exotoxin, streptokinase, andhyaluronidase.

Successful treatment depends on earlyrecognition of the condition, early surgicaldebridement, and the currently recommendedantimicrobial treatment of high parenteraldoses of benzylpenicillin with the addition ofclindamycin in severe cases.5 Furthermore, itmust be remembered that the subcutaneousspread is often more extensive than theinvolvement of the overlying skin while thepreservation of the orbicularis and the eyelidmargins will simplify reconstructive surgery.

Although rare, particularly without a historyof trauma, prompt recognition of this poten-tially fatal infection is necessary for successfultreatment. The apparent change in thespectrum of the disease caused by Streptococcuspyogenes may be related to the prevalent Mserotypes and the ability of the organisms toproduce pyrogenic exotoxins. If theseserotypes become more prominent theninvasive Streptococcus pyogenes may becomemore common.The authors thank the Streptococcal Reference Laboratory, 61Colindale Avenue, London NW9 5HT for the typing of theorganism and Dr P J O'Donnell, consultant pathologist,Department of Histopathology, King's College Hospital for hisassistance in the preparation of the pathology illustration.

1 Stevens DL. Invasive group A streptococcal infections.Clin Infect Dis 1992; 14: 2-13.

2 Kronish JW, McLeish WM. Eyelid necrosis and periorbitalnecrotising fasciitis. Report of a case and review of theliterature. Ophthalmology 1991; 98: 92-8.

3 Rose GE, Howard DJ, Watts MR. Periorbital necrotisingfasciitis. Eye 1991; 5: 736-40.

4 Schwartz B, Facklam RR, Breiman RF. Changing epidemi-ology of group A streptococcal infection in the USA. Lancet1990; 336: 1167-71.

5 Invasive group A streptococcal infections in Gloucestershire.CDR Weekly 1994; 4: 97.

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