- Email: [email protected]
Sleep disorders in Marfan's syndrome
1359
the onchocercal blind are in communities with a CMFL of 10 mf/s prevalence of 55% and over, among 15% of the
meningococcaemia we decided to infuse tissue plasminogen activator (t-PA) in low dose (0-02-0-04 mg/kg/h for 22 h)2 in an
population living in a savanna onchocerciasis-endemic area at high
attempt to preserve function of the dominant hand. The colour and perfusion of the digits of the hand improved with erythematous margins developing and less discoloration, and the right hand was clearly better perfused than the left hand. There was no change in coagulation profile during the infusion. 24 h later, with right hand clearly warmer and less discoloured than the left hand, a left radial arterial cannula was inserted and t-PA was infused at 0-02 mg/kg/h for 11hours. The only abnormality at the time of infusion was a fall in fibrinogen from 1-7to 1-04 g/1. The child recovered, the skin loss and blistering of digits responding to conservative treatment. She now has full function of both hands. Purpura fulminans secondary to sepsis has been described in meningococcaemia, pneumococcal infection, and many other infections. It is associated with DIC, which develops subsequent to endotoxin-mediated endothelial cell injury, elaboration of thromboplastins, and fibrin deposition.4 The skin lesions in meningococcaemia are consistent with those seen in other forms of purpura fulminans.s Local fibrinolysis depends primarily on endothelial cell production oft-PA.6 Septic shock associated DIC leads to characteristic disturbances of fibrinolytic regulation with an early t-PA-induced fibrinolytic response to widespread fibrin deposition that is poorly sustained and followed by increasing inhibition of fibrinolysis and progressive fibrin deposition? Local or systemic infusions of t-PA may address the imbalance between fibrinolysis and its inhibition in this setting. Certainly preservation of function of extremities must be the main aim of therapy once survival in a case of fulminant meningococcaemia is likely. Early use of local infusions of t-PA may help to lessen the morbidity of this devastating condition.
or more or a
Screening communities with a CMFL of 4-10 mf/s or a prevalence of 40-55% increases the pick-up rate to 97% but doubles the population covered to 30% (figure). Onchocercal blindness does not occur in communities with a CMFL below 05 mf/s or a prevalence of 20% or less. These findings underline the importance of epidemiological mapping in onchocerciasis endemic areas,8 before starting any ivermectin mass treatment to control onchocerciasis as a public health problem. Such maps identify the areas and communities that need to have a sustained annual treatment, and also identifies communities that willingly accept treatment. Although ivermectin is supplied free of charge, the cost of its delivery cannot be overlooked even with simplified logistics. An efficient delivery risk of onchocerciasis.
strategy is therefore essential to keep costs down to enhance sustainability. These analyses indicate that before ivermectin mass treatment to control onchocerciasis as a public health problem, it should be clear that the activity will have to be long term. Targeting of the treatment is essential to achieve the most cost-effective and sustainable strategy.
Onchocerciasis Control Programme in West Africa, BP549,
Ouagadougou,
Burkina Faso
K. Y. DADZIE J. REMME B. BOATIN G. DE SOLE E. S. ALLEY O. BA E. M. SAMBA
MA, Diallo S, Diop IM, Larivière M, Porta M. Efficacy of and tolerance of ivermectin in human onchocerciasis. Lancet 1982; ii: 171-73. 2. De Sole G, Remme J, Awadzi K, et al. Adverse reactions after large scale treatment of onchocerciasis with ivermectin: combined results from eight community trials. Bull WHO 1989; 67: 707-19. 3. Dadzie KY, Remme J, Alley ES, De Sole G. Changes in ocular onchocerciasis four and twelve months after community-based treatment with ivermectin in a holoendemic onchocerciasis focus. Trans R Soc Trop Med Hyg 1990; 84: 103-08. 4. Remme J, Baker RHA, De Sole G, et al. A community trial of ivermectin in the onchocerciasis focus of Asubende, Ghana I: effect on the microfilarial reservoir and the transmission of Onchocerca volvulus. Trop Med Parasitol 1989; 40: 367-74. 5. Remme J, De Sole G, Dadzie KY, et al. Large scale use of ivermectin and its epidemiological consequences. Acta Leidensia 1990; 59: 177-92. 6. Remme J, Dadzie KY, Rolland A, Thylefors B. Ocular onchocerciasis and intensity of infection in the community I: West African savanna. Trop Med Parasitol 1989; 40: 340-47. 7. Dadzie KY, Remme J, Rolland A, Thylefors B. Ocular onchocerciasis and intensity of infection in the community II: West African rainforest foci of the vector Simulium yahense. Trop Med Parasitol 1989; 40: 348-54. 8. De Sole G, Giese J, Keita FM, Remme J. Detailed epidemiological mapping of three onchocerciasis foci in West Africa. Acta Trop 1990; 48: 203-13. 1. Aziz
Tissue plasminogen activator for gangrene in fulminant meningococcaemia SIR,-A 13-year-old right-handed girl presented with a 24-hour history of fever, myalgia, headache, and abdominal pain. When seen in our emergency room she was shocked with confusion, agitation, pallor, and peripheral acrocyanosis. Her pulse was weak and
Intensive Care Unit, Adelaide Children’s Hospital, North Adelaide, South Australia 5506, Australia
S. R. KEELEY N. T. MATTHEWS M. BUIST
MN, Langer B, Hoshino S, et al. Pathogenesis of cutaneous lesions in acute meningococcaemia in humans: light, immunoflourescent and electron-microscopic studies in skin biopsy specimens. J Infect Dis 1976; 133: 506. 2. Anderson BJ, Keeley SR, Johnson ND. Prothrombinex induced thrombosis and its management with regional plasminogen activator in hepatic failure. Med J Aust 1990; 153: 352-56. 3. Chu DZJ, Blaisdell FW. Purpura fulminans. Am J Surg 1982; 143: 356-62. 4. Malik AB. Pulmonary micro-embolism. Physiol Rev 1983; 63: 1140-1207 5. Adcock, Hicks. Dermatopathology of skin necrosis. Semin Thromb Hemost 1990; 16: 1. Sotto
284-92. 6. Collen D. On the regulation and control of fibrinolysis. Thromb Hemost 1980; 43: 77-89. 7. Francis RB, Seyfert U. Tissue plasminogen activator antigen and activity in DIC clinico-pathologic correlations. J Lab Clin Med 1987; 110: 541-547.
Sleep disorders in Marfan’s syndrome
the peripheral blood smear, and further tests indicated disseminated intravascular coagulation (DIC) with macroscopic fibrinolysis and a
SIR,-Marfan’s syndrome is inherited as an autosomal dominant trait, and is defined on the basis of characteristic changes in several connective tissue systems: musculoskeletal, ocular, cardiovascular, skin, central nervous, and respiratory.1 The syndrome of obstructive sleep apnoea is a common disorder, especially in middle-aged overweight males.2 Patients experience a sleepinduced occlusion of the pharyngeal airway, which results in multiple apnoeic episodes. Sleep apnoea occurs in association with systemic disorders such as hypothyroidism3 and acromegaly,4 as well as diseases that cause localised narrowing of the upper airway.5 Patients with sleep apnoea are often obese, short-necked, and male.
time of 182min. Widespread organ dysfunction ensued with oliguric renal failure, persistent acidosis, and respiratory failure necessitating intubation and ventilation. She became stable after haemofiltration and an adrenaline infusion. Neisseria rnmingitidis was cultured from blood. After 48 h it became clear that the child was likely to survive. At that time she had a confluent purpuric discoloration of all digits of the hands from the metacarpophalangeal joints. This was associated with absent capillary filling and coldness of the overlying skin. Her right hand was much worse than her left, presumably due to the Presence of a right radial artery catheter. Because microvessel thrombosis is a prominent feature in the skin lesions of fulminant
An association with Marfan’s syndrome has never been described before, and sleep apnoea in the tall, thin, marfanoid person would seem unusual. We report 6 patients with Marfan’s syndrome, all of whom were found to have obstructive sleep apnoea. The first patient was referred to our sleep disorders service because of excessive daytime sleepiness. When he was found to have sleep apnoea, 4 patients were randomly recruited from the Marfan clinic at our institution to investigate the possibility of an association between the two conditions. The sixth patient was referred to our sleep disorders service with snoring, and he was also found to have Marfan’s syndrome. All 6 patients fulfilled diagnostic criteria for this condition.’ They all underwent standard polysomnography.6
thready at 120/min and her blood pressure was 80/50 mm Hg. She had an evolving petechial/purpuric rash over her flanks and forehead. She
was
given
intravenous colloid solution and transferred
to
intensive care. Investigations indicated a poor prognosis (Hb 129
g/1, white cells 1900/11, platelets 8000/ul); diplococci were seen on clotting
1360
DATA FROM SLEEP STUDIES
biopsy although their histological findings (no abnormality in 3, koilocytosis in 1, CIN grade I in 10) would not usually be regarded as sufficient grounds for such a procedure. Our findings suggest that in our study also some "unnecessary" cone biopsies were done. Although loop diathermy excision seems a much better technique than laser ablation, the possibility of over-treatment cone
should be borne in mind. Department of Obstetrics & Gynaecology, George Eliot Hospital,
Calculated respiratory variables were apnoea plus hypopnoea index (AHI; the number of apnoeas and hypopnoeas per hour of sleep), apnoea duration, and minimum arterial O2 saturation during each apnoeic episode. Standard definitions of apnoea and hypopnoea were applied, and sleep recordings were scored in 60 s epochs and staged according to previously established criteria.6 No patient received either sedatives or alcohol before the sleep study. All patients were snorers. Of the 4 who were randomly recruited, 3 had definite symptoms of sleep apnoea. 2 patients were premenopausal females, and 4 were males. The mean age was 33 years (range 15-43). The average body mass index (BMI) was 21-2 kg/m2 (range 18-0-25-9). Only 1 patient had abnormal spirometry. All 6 had obstructive sleep apnoea (AHI > 5). The average AHI was 23 (range [SEM] 9-48 [14.72]), and the average length of apnoeas for the group was 23 s (range 10-43 [9.3]) (table). Our preliminary study suggests that there may be a high frequency of sleep apnoea in patients with Marfan’s syndrome. We suggest that this association may be relevant to the poor outlook of patients with this condition, because of apnoea-induced haemodynamic changes that may stress the weakened aortic root. Additionally, we suggest that a floppy, easily collapsible pharynx is responsible for the observed apnoeic episodes. We believe that patients with Marfan’s syndrome should be evaluated for coexistent sleep apnoea. We thank Dr J. G. Richards and Dr J. Hwa of the Marfan clinic, and acknowledge the technical help of the nursing staff of the sleep disorders unit.
Department of Respiratory Medicine, Sleep Disorders Unit, Royal Prince Alfred Hospital, and Department of Medicine, University of Sydney, Sydney 2006, Australia
PETER A. CISTULLI COLIN E. SULLIVAN
1. Pyeritz RE. The Marfan syndrome Am Fam Physician 1986; 34: 83-94. 2. Guilleminault C, Dement WC. 235 cases of excessive daytime sleepiness. J Neurol Sci 1977; 31: 13-27. 3. Grunstein RR, Sullivan CE. Sleep apnea and hypothyroidism: mechanisms and management. Am J Med 1988; 85: 775-79. 4. Mezon BJ, West P, MacLean JP, et al. Sleep apnea in acromegaly. Am J Med 1980; 69: 615-18. 5. Mangat D, Orr WC, Smith RO. Sleep apnea, hypersomnolence, and upper airway obstruction secondary to adenotonsillar enlargement. Arch Otolaryngol 1977; 103: 383-86. 6. Guilleminault C, van den Hoed J, Miller M. Clinical overview of the sleep apnea syndromes. In: Guilleminault C, Dement W, eds. Sleep apnea syndromes. New York: Alan R. Liss, 1978: 1-12.
Outpatient loop diathermy conisation SIR,—We agree with Mr Byme and colleagues’ (April 13, p 917) outpatient loop diathermy excision. They point out that one major advantage of the technique is the availability of comments on
histological material for a confirmatory diagnosis, which is not so with simple ablative procedures. We have also commented thus.1 Byme et al say that loop diathermy is "likely soon to replace laser vaporisation". Again, we agree. In a prospective randomised trial of almost 200 patients loop diathermy proved better than laser vaporisation in respect of haemorrhage rate, patient comfort, and operative speed, although recurrence of disease rates were much the same.2 The technique Byme et al describe is similar to that reported earlier, and we concur that this is a safe and effective technique. A word of caution seems appropriate, however. From Byme and colleagues’ report, it seems that 14/50 (28%) patients underwent
Nuneaton, CV10 1 DJ, UK
JEFFREY H. PHIPPS
Department of Obstetrics & Gynaecology, Watford General Hospital, Herts
P. C. GUNASEKERA
Phipps JH, Gunasekera PC, Lewis BV. Large loop diathermy excision of the transformation zone in cervical dysplasia. J Obstet Gynaecol 1990; 10: 542-44. 2. Gunasekera PC, Phipps JH, Lewis BV. Large loop excision of the transformation zone compared to carbon dioxide laser in cervical dysplasia: a superior mode of treatment. Br J Obstet Gynaecol 1991; 97: 995-98. 3. Mor-Yosef S, Lopez A, Pearson S, Monaghan J. Loop diathermy cone biopsy. Obstet Gynecol 1990; 75: 884-86. 1.
Benign aseptic (Mollaret’s) meningitis after genital herpes SIR,-Mollaret’s meningitis is a benign aseptic meningitis characterised by headache, meningism, malaise, and fever, which recurs at intervals ranging from weeks to months. The cause is unknown. Herpes simplex virus 1 (HSV 1) was isolated from the cerebrospinal fluid (CSF) of 1 patient with Mollaret’s meningitis,! and intrathecal synthesis of antibody to an HSV-2-specific protein has been shown in othersInow report 3 patients with recurrent episodes of aseptic meningitis that developed shortly after the appearance of symptoms of recrudescent genital herpes. Case 1-A 43-year-old woman first presented with aseptic meningitis in 1974, and had yearly recurrences requiring hospital admission up to 1979. She experienced a further attack in March, 1990. 1-2 days before these episodes, she felt a burning sensation on the sole of her left foot, herpetic vesicles erupted on her buttocks and thigh. Attacks of meningitis were not associated with visible skin eruptions. CSF examination has shown only a mixed cell pleocytosis during each symptomatic period. She has had no further recurrence of meningitis since she was given acyclovir 200 mg five times daily to be taken from the onset of prodromal symptoms. Case 2-A 34-year-old woman had three episodes of aseptic meningitis over a six month period up to August, 1990. She complained of severe headaches, meningismus, malaise, fever, myalgias, and arthralgias on each occasion. CSF examination during the initial attack revealed 235 white cells/mm3 (97% mononuclear cells), 89 g/1 protein, and glucose 2-8 mmol/1. Each episode was preceded by a severe herpetic eruption affecting her left buttock, hip, and leg. These outbreaks date back to 1983 and have increased in frequency from two per year to over ten per year. High-dose acyclovir improved her symptoms and she has not had any further attacks of meningitis since starting acyclovir 200 mg three times daily. Case 3-A 47-year-old woman noted severe headache, hallucinations, impaired memory, low-grade fever, myalgias, and arthralgias for three weeks in December, 1985. In August, 1986, and July, 1988, she had recurrences of these symptoms without hallucinations. CSF examination in July, 1988, revealed a normal opening pressure, 352 white blood cells/mm3 (91% polymorphonuclear cells), 19-9 g/l protein, and glucose 22 mmol/1. There was no evidence of sarcoid or connective tissue disease. Extensive microbiological studies were negative, although high serum titres of antibody to HSV 2 were found. CSF examination during a recurrence in February, 1990, showed 30 white blood cells/mm3 (100% lymphocytes), and 20-8 g/dl protein. Computed tomography and magnetic resonance imaging of the skull were normal. Viral isolation studies indicate that HSV 1 and HSV 2 are responsible for approximately 0-5—3% of cases of aseptic meningitis.3 Aseptic meningitis is a well-recognised complication of primary genital herpes.4 In one large study, stiff neck, headache,
the onchocercal blind are in communities with a CMFL of 10 mf/s prevalence of 55% and over, among 15% of the
meningococcaemia we decided to infuse tissue plasminogen activator (t-PA) in low dose (0-02-0-04 mg/kg/h for 22 h)2 in an
population living in a savanna onchocerciasis-endemic area at high
attempt to preserve function of the dominant hand. The colour and perfusion of the digits of the hand improved with erythematous margins developing and less discoloration, and the right hand was clearly better perfused than the left hand. There was no change in coagulation profile during the infusion. 24 h later, with right hand clearly warmer and less discoloured than the left hand, a left radial arterial cannula was inserted and t-PA was infused at 0-02 mg/kg/h for 11hours. The only abnormality at the time of infusion was a fall in fibrinogen from 1-7to 1-04 g/1. The child recovered, the skin loss and blistering of digits responding to conservative treatment. She now has full function of both hands. Purpura fulminans secondary to sepsis has been described in meningococcaemia, pneumococcal infection, and many other infections. It is associated with DIC, which develops subsequent to endotoxin-mediated endothelial cell injury, elaboration of thromboplastins, and fibrin deposition.4 The skin lesions in meningococcaemia are consistent with those seen in other forms of purpura fulminans.s Local fibrinolysis depends primarily on endothelial cell production oft-PA.6 Septic shock associated DIC leads to characteristic disturbances of fibrinolytic regulation with an early t-PA-induced fibrinolytic response to widespread fibrin deposition that is poorly sustained and followed by increasing inhibition of fibrinolysis and progressive fibrin deposition? Local or systemic infusions of t-PA may address the imbalance between fibrinolysis and its inhibition in this setting. Certainly preservation of function of extremities must be the main aim of therapy once survival in a case of fulminant meningococcaemia is likely. Early use of local infusions of t-PA may help to lessen the morbidity of this devastating condition.
or more or a
Screening communities with a CMFL of 4-10 mf/s or a prevalence of 40-55% increases the pick-up rate to 97% but doubles the population covered to 30% (figure). Onchocercal blindness does not occur in communities with a CMFL below 05 mf/s or a prevalence of 20% or less. These findings underline the importance of epidemiological mapping in onchocerciasis endemic areas,8 before starting any ivermectin mass treatment to control onchocerciasis as a public health problem. Such maps identify the areas and communities that need to have a sustained annual treatment, and also identifies communities that willingly accept treatment. Although ivermectin is supplied free of charge, the cost of its delivery cannot be overlooked even with simplified logistics. An efficient delivery risk of onchocerciasis.
strategy is therefore essential to keep costs down to enhance sustainability. These analyses indicate that before ivermectin mass treatment to control onchocerciasis as a public health problem, it should be clear that the activity will have to be long term. Targeting of the treatment is essential to achieve the most cost-effective and sustainable strategy.
Onchocerciasis Control Programme in West Africa, BP549,
Ouagadougou,
Burkina Faso
K. Y. DADZIE J. REMME B. BOATIN G. DE SOLE E. S. ALLEY O. BA E. M. SAMBA
MA, Diallo S, Diop IM, Larivière M, Porta M. Efficacy of and tolerance of ivermectin in human onchocerciasis. Lancet 1982; ii: 171-73. 2. De Sole G, Remme J, Awadzi K, et al. Adverse reactions after large scale treatment of onchocerciasis with ivermectin: combined results from eight community trials. Bull WHO 1989; 67: 707-19. 3. Dadzie KY, Remme J, Alley ES, De Sole G. Changes in ocular onchocerciasis four and twelve months after community-based treatment with ivermectin in a holoendemic onchocerciasis focus. Trans R Soc Trop Med Hyg 1990; 84: 103-08. 4. Remme J, Baker RHA, De Sole G, et al. A community trial of ivermectin in the onchocerciasis focus of Asubende, Ghana I: effect on the microfilarial reservoir and the transmission of Onchocerca volvulus. Trop Med Parasitol 1989; 40: 367-74. 5. Remme J, De Sole G, Dadzie KY, et al. Large scale use of ivermectin and its epidemiological consequences. Acta Leidensia 1990; 59: 177-92. 6. Remme J, Dadzie KY, Rolland A, Thylefors B. Ocular onchocerciasis and intensity of infection in the community I: West African savanna. Trop Med Parasitol 1989; 40: 340-47. 7. Dadzie KY, Remme J, Rolland A, Thylefors B. Ocular onchocerciasis and intensity of infection in the community II: West African rainforest foci of the vector Simulium yahense. Trop Med Parasitol 1989; 40: 348-54. 8. De Sole G, Giese J, Keita FM, Remme J. Detailed epidemiological mapping of three onchocerciasis foci in West Africa. Acta Trop 1990; 48: 203-13. 1. Aziz
Tissue plasminogen activator for gangrene in fulminant meningococcaemia SIR,-A 13-year-old right-handed girl presented with a 24-hour history of fever, myalgia, headache, and abdominal pain. When seen in our emergency room she was shocked with confusion, agitation, pallor, and peripheral acrocyanosis. Her pulse was weak and
Intensive Care Unit, Adelaide Children’s Hospital, North Adelaide, South Australia 5506, Australia
S. R. KEELEY N. T. MATTHEWS M. BUIST
MN, Langer B, Hoshino S, et al. Pathogenesis of cutaneous lesions in acute meningococcaemia in humans: light, immunoflourescent and electron-microscopic studies in skin biopsy specimens. J Infect Dis 1976; 133: 506. 2. Anderson BJ, Keeley SR, Johnson ND. Prothrombinex induced thrombosis and its management with regional plasminogen activator in hepatic failure. Med J Aust 1990; 153: 352-56. 3. Chu DZJ, Blaisdell FW. Purpura fulminans. Am J Surg 1982; 143: 356-62. 4. Malik AB. Pulmonary micro-embolism. Physiol Rev 1983; 63: 1140-1207 5. Adcock, Hicks. Dermatopathology of skin necrosis. Semin Thromb Hemost 1990; 16: 1. Sotto
284-92. 6. Collen D. On the regulation and control of fibrinolysis. Thromb Hemost 1980; 43: 77-89. 7. Francis RB, Seyfert U. Tissue plasminogen activator antigen and activity in DIC clinico-pathologic correlations. J Lab Clin Med 1987; 110: 541-547.
Sleep disorders in Marfan’s syndrome
the peripheral blood smear, and further tests indicated disseminated intravascular coagulation (DIC) with macroscopic fibrinolysis and a
SIR,-Marfan’s syndrome is inherited as an autosomal dominant trait, and is defined on the basis of characteristic changes in several connective tissue systems: musculoskeletal, ocular, cardiovascular, skin, central nervous, and respiratory.1 The syndrome of obstructive sleep apnoea is a common disorder, especially in middle-aged overweight males.2 Patients experience a sleepinduced occlusion of the pharyngeal airway, which results in multiple apnoeic episodes. Sleep apnoea occurs in association with systemic disorders such as hypothyroidism3 and acromegaly,4 as well as diseases that cause localised narrowing of the upper airway.5 Patients with sleep apnoea are often obese, short-necked, and male.
time of 182min. Widespread organ dysfunction ensued with oliguric renal failure, persistent acidosis, and respiratory failure necessitating intubation and ventilation. She became stable after haemofiltration and an adrenaline infusion. Neisseria rnmingitidis was cultured from blood. After 48 h it became clear that the child was likely to survive. At that time she had a confluent purpuric discoloration of all digits of the hands from the metacarpophalangeal joints. This was associated with absent capillary filling and coldness of the overlying skin. Her right hand was much worse than her left, presumably due to the Presence of a right radial artery catheter. Because microvessel thrombosis is a prominent feature in the skin lesions of fulminant
An association with Marfan’s syndrome has never been described before, and sleep apnoea in the tall, thin, marfanoid person would seem unusual. We report 6 patients with Marfan’s syndrome, all of whom were found to have obstructive sleep apnoea. The first patient was referred to our sleep disorders service because of excessive daytime sleepiness. When he was found to have sleep apnoea, 4 patients were randomly recruited from the Marfan clinic at our institution to investigate the possibility of an association between the two conditions. The sixth patient was referred to our sleep disorders service with snoring, and he was also found to have Marfan’s syndrome. All 6 patients fulfilled diagnostic criteria for this condition.’ They all underwent standard polysomnography.6
thready at 120/min and her blood pressure was 80/50 mm Hg. She had an evolving petechial/purpuric rash over her flanks and forehead. She
was
given
intravenous colloid solution and transferred
to
intensive care. Investigations indicated a poor prognosis (Hb 129
g/1, white cells 1900/11, platelets 8000/ul); diplococci were seen on clotting
1360
DATA FROM SLEEP STUDIES
biopsy although their histological findings (no abnormality in 3, koilocytosis in 1, CIN grade I in 10) would not usually be regarded as sufficient grounds for such a procedure. Our findings suggest that in our study also some "unnecessary" cone biopsies were done. Although loop diathermy excision seems a much better technique than laser ablation, the possibility of over-treatment cone
should be borne in mind. Department of Obstetrics & Gynaecology, George Eliot Hospital,
Calculated respiratory variables were apnoea plus hypopnoea index (AHI; the number of apnoeas and hypopnoeas per hour of sleep), apnoea duration, and minimum arterial O2 saturation during each apnoeic episode. Standard definitions of apnoea and hypopnoea were applied, and sleep recordings were scored in 60 s epochs and staged according to previously established criteria.6 No patient received either sedatives or alcohol before the sleep study. All patients were snorers. Of the 4 who were randomly recruited, 3 had definite symptoms of sleep apnoea. 2 patients were premenopausal females, and 4 were males. The mean age was 33 years (range 15-43). The average body mass index (BMI) was 21-2 kg/m2 (range 18-0-25-9). Only 1 patient had abnormal spirometry. All 6 had obstructive sleep apnoea (AHI > 5). The average AHI was 23 (range [SEM] 9-48 [14.72]), and the average length of apnoeas for the group was 23 s (range 10-43 [9.3]) (table). Our preliminary study suggests that there may be a high frequency of sleep apnoea in patients with Marfan’s syndrome. We suggest that this association may be relevant to the poor outlook of patients with this condition, because of apnoea-induced haemodynamic changes that may stress the weakened aortic root. Additionally, we suggest that a floppy, easily collapsible pharynx is responsible for the observed apnoeic episodes. We believe that patients with Marfan’s syndrome should be evaluated for coexistent sleep apnoea. We thank Dr J. G. Richards and Dr J. Hwa of the Marfan clinic, and acknowledge the technical help of the nursing staff of the sleep disorders unit.
Department of Respiratory Medicine, Sleep Disorders Unit, Royal Prince Alfred Hospital, and Department of Medicine, University of Sydney, Sydney 2006, Australia
PETER A. CISTULLI COLIN E. SULLIVAN
1. Pyeritz RE. The Marfan syndrome Am Fam Physician 1986; 34: 83-94. 2. Guilleminault C, Dement WC. 235 cases of excessive daytime sleepiness. J Neurol Sci 1977; 31: 13-27. 3. Grunstein RR, Sullivan CE. Sleep apnea and hypothyroidism: mechanisms and management. Am J Med 1988; 85: 775-79. 4. Mezon BJ, West P, MacLean JP, et al. Sleep apnea in acromegaly. Am J Med 1980; 69: 615-18. 5. Mangat D, Orr WC, Smith RO. Sleep apnea, hypersomnolence, and upper airway obstruction secondary to adenotonsillar enlargement. Arch Otolaryngol 1977; 103: 383-86. 6. Guilleminault C, van den Hoed J, Miller M. Clinical overview of the sleep apnea syndromes. In: Guilleminault C, Dement W, eds. Sleep apnea syndromes. New York: Alan R. Liss, 1978: 1-12.
Outpatient loop diathermy conisation SIR,—We agree with Mr Byme and colleagues’ (April 13, p 917) outpatient loop diathermy excision. They point out that one major advantage of the technique is the availability of comments on
histological material for a confirmatory diagnosis, which is not so with simple ablative procedures. We have also commented thus.1 Byme et al say that loop diathermy is "likely soon to replace laser vaporisation". Again, we agree. In a prospective randomised trial of almost 200 patients loop diathermy proved better than laser vaporisation in respect of haemorrhage rate, patient comfort, and operative speed, although recurrence of disease rates were much the same.2 The technique Byme et al describe is similar to that reported earlier, and we concur that this is a safe and effective technique. A word of caution seems appropriate, however. From Byme and colleagues’ report, it seems that 14/50 (28%) patients underwent
Nuneaton, CV10 1 DJ, UK
JEFFREY H. PHIPPS
Department of Obstetrics & Gynaecology, Watford General Hospital, Herts
P. C. GUNASEKERA
Phipps JH, Gunasekera PC, Lewis BV. Large loop diathermy excision of the transformation zone in cervical dysplasia. J Obstet Gynaecol 1990; 10: 542-44. 2. Gunasekera PC, Phipps JH, Lewis BV. Large loop excision of the transformation zone compared to carbon dioxide laser in cervical dysplasia: a superior mode of treatment. Br J Obstet Gynaecol 1991; 97: 995-98. 3. Mor-Yosef S, Lopez A, Pearson S, Monaghan J. Loop diathermy cone biopsy. Obstet Gynecol 1990; 75: 884-86. 1.
Benign aseptic (Mollaret’s) meningitis after genital herpes SIR,-Mollaret’s meningitis is a benign aseptic meningitis characterised by headache, meningism, malaise, and fever, which recurs at intervals ranging from weeks to months. The cause is unknown. Herpes simplex virus 1 (HSV 1) was isolated from the cerebrospinal fluid (CSF) of 1 patient with Mollaret’s meningitis,! and intrathecal synthesis of antibody to an HSV-2-specific protein has been shown in othersInow report 3 patients with recurrent episodes of aseptic meningitis that developed shortly after the appearance of symptoms of recrudescent genital herpes. Case 1-A 43-year-old woman first presented with aseptic meningitis in 1974, and had yearly recurrences requiring hospital admission up to 1979. She experienced a further attack in March, 1990. 1-2 days before these episodes, she felt a burning sensation on the sole of her left foot, herpetic vesicles erupted on her buttocks and thigh. Attacks of meningitis were not associated with visible skin eruptions. CSF examination has shown only a mixed cell pleocytosis during each symptomatic period. She has had no further recurrence of meningitis since she was given acyclovir 200 mg five times daily to be taken from the onset of prodromal symptoms. Case 2-A 34-year-old woman had three episodes of aseptic meningitis over a six month period up to August, 1990. She complained of severe headaches, meningismus, malaise, fever, myalgias, and arthralgias on each occasion. CSF examination during the initial attack revealed 235 white cells/mm3 (97% mononuclear cells), 89 g/1 protein, and glucose 2-8 mmol/1. Each episode was preceded by a severe herpetic eruption affecting her left buttock, hip, and leg. These outbreaks date back to 1983 and have increased in frequency from two per year to over ten per year. High-dose acyclovir improved her symptoms and she has not had any further attacks of meningitis since starting acyclovir 200 mg three times daily. Case 3-A 47-year-old woman noted severe headache, hallucinations, impaired memory, low-grade fever, myalgias, and arthralgias for three weeks in December, 1985. In August, 1986, and July, 1988, she had recurrences of these symptoms without hallucinations. CSF examination in July, 1988, revealed a normal opening pressure, 352 white blood cells/mm3 (91% polymorphonuclear cells), 19-9 g/l protein, and glucose 22 mmol/1. There was no evidence of sarcoid or connective tissue disease. Extensive microbiological studies were negative, although high serum titres of antibody to HSV 2 were found. CSF examination during a recurrence in February, 1990, showed 30 white blood cells/mm3 (100% lymphocytes), and 20-8 g/dl protein. Computed tomography and magnetic resonance imaging of the skull were normal. Viral isolation studies indicate that HSV 1 and HSV 2 are responsible for approximately 0-5—3% of cases of aseptic meningitis.3 Aseptic meningitis is a well-recognised complication of primary genital herpes.4 In one large study, stiff neck, headache,