Benign focal epilepsy of childhood and gastroesophageal...

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BRIEF COMMUNICATION Benign focal epilepsy of childhood and gastroesophageal reflux ANNE G SHEEHAN MB MRCPI, SHERRY PELENSKY RN, COLIN VAN ORMAN MD FRCPC, STEVEN R MARTIN MD FRCPC AG SHEEHAN, S PELENSKY, C V<A,N ORMAN, SR MARTIN. Benign focal epilepsy of childhood and ~ux. Can J Gastroenterol 1994; 8( l ):45-48. Gastrnesophageal te6m( has~ iwociated with, and implicated in, a number of cond itions, inc~teSl)~ disease (recurrent pneumonia, chronic cough, asthma), sudden infiot dead>. syndrome, dysphagia and centra l nervous disorders. An eight-year-old girl p:.ented with an acute history that suggested gas troesophageal reflux. AD~~l motility study was abnormal and 24 h pH st udy de monstrated~ reflux. Before the manometric study, a seiz ure was ob~erved and ~l\eU(Ological evaluation confirmed the diagnosis of benign focal epilepsy of dilldhood, which was treated with carbamazepine. The symptoms resolved after eight weeks and the repeat reflux investigations were essentially normaL Ompbuyngeal symptoms are common in benign focal ep ilepsy of childhood, a condition which is very responsive to therapy. Sy mptoms suggestive of this dlagnostS - acute onset, with unusual oropha1yngeal sensations, or seizures-occUl;ring mainly at night may initially be confused with gastroesophageal reflux. Benign focal epilepsy of c hildhood should he considered in reflux presenting outside infancy. Key Words: Children, Epilepsy, GastroesophageQlreflux, Manomecry Epilepsie partielle benigne infantile et reflux gastro,oesophagien RESUME : Le reflux gastro-oesophagien a &l associe a divers etats morbides, n otamment a ccrtaines affections respiratoires (pneumonie rec idivante, toux chron iquc, asthme), syndrome de mort subite du nourrisson, dysphagie et affec- tions tlu systeme nerveux central. Une fillette de huit ans a presente un tab leau aigu indicatcur de reflux gastro-oesophagien. l'epreuve de motilite oeso- phagienne s'est reve lec anormale et la mesure du pH sur 24 heures a confirme le reflux gascro-oesop hagien. Avant !es epreuves de manometrie, une convulsion a etc observee et l'evaluat inn neurologique subseque nte a confirme un diagnostic d'epilcpsie particlle benigne infantile qui a ete traitee avec de la carbamazepine. Les symptomes sont rcmres clans l'ordre apres huit semaines et a la rep rise, les cpreuvcs sur le reflux sc sonc revelee~ essentiellement nonnalcs. Les symptomes Department of Pediatrics, Alberta Children's Hospital; and University of Calgary, Calgary, Alberta Corres/JOndence: Dr SR Martin, Gastroenterowgie/Nutrwon, I lopital Ste-Justine, 3175 Core Ste Catherine, Montreal, Quebec l-l3T 1C5. Telephone (514) 345-4626, Fax (514) 345-4999 Received for publication October 15, 1992. Accepted]uly 15, 1993 CAN J GASTROENTEROL VOL 8 No 1 JANUARY/FEBRUARY 1994 B ENIGN FOCAL EPILEPSY OF CHILD- hood (BFEC) is defined by five main characteristics. It is age-related, beginning between the ages of two and 13 years, with a spontaneous remission during adolescence ( 1). It occurs in otherwise normal children without neurological or intellectual deficit; in the majority of cases the seizures are partia l, with motor signs frequently as- sociated with somatosensory symp- toms, and are sleep-related in 75% of patients. The incerictal electroencepha- logram displays a spike focus locate<l in the centrotemporal (rolandic) area with normal background act ivity. A pati ent is presented with BFEC, whose clinical history and marked oro- pharyngeal symptoms initially sug- gested gastroesophageal reflux which was confirmed in a 24 h pH study. Clinical symptoms and documented re- flux resolved with seizure therapy. We suggest that gastroesophageal reflux may occur in BFEC and may account for the marked oropha ryngeal symptoms described in this condition. CASE PRESENTATION An eight-year-old Caucasian girl presented to the gastrocnterology clinic of Alberta Children's Hospital with a seven-week history of 'retching episodes', abdominal pain and choking spells. The patient described episodes beginning with an unusual sensation arising in her stomach, which would 45

Transcript of Benign focal epilepsy of childhood and gastroesophageal...

Page 1: Benign focal epilepsy of childhood and gastroesophageal refluxdownloads.hindawi.com/journals/cjgh/1994/725067.pdf · BRIEF COMMUNICATION Benign focal epilepsy of childhood and gastroesophageal

BRIEF COMMUNICATION

Benign focal epilepsy of childhood and

gastroesophageal reflux

ANNE G SHEEHAN MB MRCPI, SHERRY PELENSKY RN, COLIN VAN ORMAN MD FRCPC, STEVEN R MARTIN MD FRCPC

AG SHEEHAN, S PELENSKY, C V<A,N ORMAN, SR MARTIN. Benign focal epilepsy of childhood and ~ux. Can J Gastroenterol 1994; 8( l ):45-48. Gastrnesophageal te6m( has~ iwociated with, and implicated in, a number of conditions, inc~teSl)~ disease (recurrent pneumonia, chronic cough, asthma), sudden infiot dead>. syndrome, dysphagia and central nervous disorders. An eight-year-old girl p:.ented with an acute history that suggested gastroesophageal reflux. AD~~l motility study was abnormal and 24 h pH study demonstrated~ reflux. Before the manometric study, a seizure was ob~erved and ~l\eU(Ological evaluation confirmed the diagnosis of benign focal epilepsy of dilldhood, which was treated with carbamazepine. The symptoms resolved after eight weeks and the repeat reflux investigations were essentially normaL Ompbuyngeal symptoms are common in benign focal epilepsy of childhood, a condition which is very responsive to therapy. Symptoms suggestive of this dlagnostS - acute onset, with unusual oropha1y ngeal sensations, or seizures-occUl;ring mainly at night may initially be confused with gastroesophageal reflux. Benign focal epilepsy of childhood should he considered in reflux presenting outside infancy.

Key Words: Children, Epilepsy, GastroesophageQlreflux, Manomecry

Epilepsie partielle benigne infantile et reflux gastro,oesophagien

RESUME : Le reflux gastro-oesophagien a &l associe a divers etats morbides, notamment a ccrtaines affections respiratoires (pneumonie recidivante, toux chroniquc, asthme), syndrome de mort subite du nourrisson, dysphagie e t affec­tions tlu systeme nerveux central. Une fillette de huit ans a presente un tableau a igu indicatcur de reflux gastro-oesophagien. l'epreuve de motilite oeso­phagienne s'est revelec anormale et la mesure du pH sur 24 heures a confirme le reflux gascro-oesophagien. Avant !es epreuves de manometrie, une convulsion a etc observee et l'evaluatinn neurologique subsequente a confirme un diagnostic d'epilcpsie partic lle benigne infantile qui a ete traitee avec de la carbamazepine. Les symptomes sont rcmres clans l'ordre apres huit semaines et a la reprise, les cpreuvcs sur le reflux sc sonc revelee~ essentiellement nonnalcs. Les symptomes

Department of Pediatrics, Alberta Children's Hospital; and University of Calgary, Calgary, Alberta

Corres/JOndence: Dr SR Martin, Gastroenterowgie/Nutrwon, I lopital Ste-Justine, 3175 Core Ste Catherine, Montreal, Quebec l-l3T 1C5. Telephone (514) 345-4626, Fax (514) 345-4999

Received for publication October 15, 1992. Accepted]uly 15, 1993

CAN J GASTROENTEROL VOL 8 No 1 JANUARY/FEBRUARY 1994

BENIGN FOCAL EPILEPSY OF CHILD­

hood (BFEC) is defined by five main characteristics. It is age-related, beginning between the ages of two and 13 years, with a spontaneous remission during adolescence ( 1). It occurs in otherwise normal children without neurological or intellectual deficit; in the majority of cases the seizures are partial, with motor signs frequently as­sociated with somatosensory symp­toms, and are sleep-related in 75% of patients. The incerictal electroencepha­logram displays a spike focus locate<l in the centrotemporal (rolandic) area with normal background activity.

A patient is presented with BFEC, whose clinical history and marked oro­pharyngeal symptoms initially sug­gested gastroesophageal reflux which was confirmed in a 24 h pH study. C linical symptoms and documented re­flux resolved with seizure therapy. We suggest that gastroesophageal reflux may occur in BFEC and may account for the marked oropharyngeal symptoms described in this condition.

CASE PRESENTATION An eight-year-old Caucasian girl

presented to the gastrocnterology clinic of Alberta Children's Hospital with a seven-week history of 'retching episodes', abdominal pain and choking spells. The patient described episodes beginning with an unusual sensation arising in her stomach, which would

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SHEEI JAN er al

oropharynges sont frequents clans l'cpilcpsie partielle bcnigne infantile, une maladie qui repond tres bien au traitement. Les symptomes qui concordenr avec ce diagnostic sont le declenchement rapide, les sensations oropharyngees inhabi­tuelles ou !es convulsions survemmt surtout la nuit et la possibilite d'une confu­sion initiate accompagnant le reflux gastro-oesophagien. L'epilepsie partielle benigne infantile doit etre envisagce lorsqu'il y a reflux, si l'enfant n'est plus un nourrisson.

TABLE 1 Esophageal manometry and 24 h pH study results before and following eight weeks of therapy with carbamazepine

At presentation After therapy Normal values

Esophageal manometry LESP (mmHg) 5 4 30.6±9.2 (reference 2)

UESP (mmHg) 120 30 21.3±7 .4 (reference 3)

24 h pH study Mean ± so• Mean ± 2S0

%time pH <4 31 3 2.3±1.9 6.1

Number of refluxes 164 50 11.1±6.1 23.3

Longest reflux 124 4 12.6±9.4 31.4 (mins)

Refluxes >5 mins 13 0 2. 1±1.8 5.7

LESP Lower esophageal sphincter pressure; UESP Upper esophageal sphincter pressure. 'Reference 17

migrate to the back of her throat. She would often notice sour tasting fluid in her mouth and begin to retch and cough. These episodes lasted 10 to 15 mins and occurred at any hour, but mostly at night, often waking her from sleep. They gradually increased in fre­quency to JO to 20 times daily.

The patient's physical examination was essentially unremarkable. Her weight was 30 kg (above the 50th per­centile), her height was 134.5 cm (on the 90th percentile) and she had nor­mal vital signs.

Investigations revealed a normal complete blood count, electrolytes, glucose, blood urea nitrogen, creat­inine, total protein, albumin, liver function tests, amylase and lipase. Ur­inalysis was unremarkable. A barium swallow demonstrated no evidence of anatomical obstruction, showing only mild gastroesophageal reflux.

The patient's initial symptoms, which preceded the retching and coughing, suggested that gastroeso­phageal reflux was precipitating these episodes, so an esophageal motility study, 24 h pH study (Table 1) and esophageal biopsy were carried out without sedation. On insertion of the motility catheter, however, the patient

developed central cyanosis with twitching movements of her eyelids lasting 2 mins, during which she was unresponsive to voice. The catheter was withdrawn and after she had recov­ered from the episode the motility study was carried out without problem. T he lower esophageal sphincter pressure was extremely low. Normal peristaltic swallow waves were demonstrated in the esophageal body but the upper esophageal sphincter showed ex­tremely high pressures (Table 1). The 24-hour pH study proved co be grossly abnormal (Table 1). Esophageal bi­opsy, from 3 cm above the lower esophageal sphincter, was normal.

In view of the seizure activity that had occurred during the study, a neuro­logical opinion was sough t, and an elec­troencephalogram and computerized tomographic (CT) scan of the brain were performed. The electroencepha­logram was abnormal with a sharp spike focus in the right central region, the field of distribution extending into the right midtemporal and midfrontal ar­eas; CT scan was normal. The findings were considered to be diagnostic of BFEC and the patient was started on carbamazepine. Because of the abnor­mal pH study and the concern that

acidification of the esophagus might be stimulating seizures, she was also started on ranitidine. In the following weeks, the patient's seizure episodes gradually reduced in frequency and se­verity, and eight weeks later had stopped completely.

Two weeks after disconttnuing rarn ­tidine, the motility study and 24 h pH study were repeated. The lower esopha­geal sphincter pressure was again low but the upper esophageal sphincter pressure was normal. Repeat 24 h pH study on this occasion was entirely dif­ferent from the previous one, the only abnormality being nn increase in mnn­ber of reflux episodes. An esophageal biopsy was not repeated because it was previously normal. The patient remains asymptomatic on carbamazepine.

DISCUSSION Primary gastrocsophageal reflux is

commonly seen in infancy with an esti­mated incidence of l :500 ( 4 ). The ma­jority of these patients have functional reflux which may be perceived as a de­velopmental d isorder which resolves in most patients by age four years ( 5). A small percentage of these patients may develop pathological gastroesophageal reflux associated with dysphagia, eso­phagitis, strictures, poor growth, respi­ratory problems or Sandifer's syndrome, and these patients usually require chronic medical therapy or surgical in­tervention (5,6).

In addition, in infancy- but more so in childhood - gastroesophageal reflux may occur as a secondary phenomenon in other <lisease states, cg, cystic fibrosis (7) and motility disorders of the esophagus (8), but most commonly in association with central nervous system disorders (9). A recent report described the association of gastroesophageal re­flux with brain-stem glioma in three infants, the suggested mechanism being that the tumour infiltration of the brain-stem impaired neurogenical con­tro l of esophageal motility, resulting in gastroesophageal reflux (10). This re­port <lcscri bes gastroesophageal reflux in association with untreated BFEC, fur­ther supporting the central influence on esophageal function.

RFEC' is the most common type of

46 CAN J GASTROENTEROI VOL 8 N O l jANUARY/FEBRl.,ARY 1994

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partial minor epilepsy in childhood, ac­counting for 11 .5 to 25% of the epilep­sies of ~chool-age children (11 ). Oro­pharyngeal symptoms arc reporred by more than half rhe patients and include hypcrsalivation with inability to swal­low, 'sounds from the throat', gurgling noise as if the child were about to

vom1r, moven1l'nts nf the mouth and speech arrest Jue to tonic or clonic phenomena mvolving the mouth (anJ probably the larynx) (1,11 ). In the case descnhed here, we documented severe gastroesophageal reflux over 24 h be­fore commencing therapy for seizures due to Bf-TC. A repeat stuJy after eight weeb of anricon vu lsant therapy dem­onstrated a dramatic reduction in num­ber and duration of reflux episodes (Ta­ble I). Jc was fortuitous that during insertion of the motility catheter, the patient developed an episode of eyelid twi tching and cyanosis which her mother had not previously described (possibly since most episodes occurred in sleep). The very high upper esopha­geal sphincter pressure demonstrated afterwards was no longer evident on repeat study e ight weeks after car­bamazepine therapy. Lability of upper esophageal sphincter pressure has been described previously ( 12- 14 ); reduc­tiorn, in pressure occur wi th sedation and increases occur with esophageal acidification, arousal, emotional st ress and 111creased abdom111al and tho racic. pressure Jue to stra ining. We cannot discount the possible effects of a seizure within the previous 10 or 15 mins, al-

ACKNOWLEDGEMENTS: The authors thank Chem Br(x1ks an<l Bev Harley for their assistance m preparing the manuscript.

REFERENCES l. Lrnseau P, Duche B. BeniJ,?"n <.:h1l<lhnod

epi lepsy with centrotemporal spike~. CleveClm J Med 1989;56:17-21.

2. Moroz SP, Espinoza J, CumminJ,?' WA, Diamant NE Lower esophageal sphincter function in children with an<l wichout ga.,troesophageal reflux. Gastroenterology l 97 6; 71 :236-41.

}. Sondheimer JM. Upper esophageal sphincter an<l phar,ngoesophagcal motor function in infants with an<l without gastrnesophageal reflux. Gastroenterology 1983;85:301-5.

4. Boyle J Gastroesophageal reflux m the

though the patient was not drowsy and was not experiencing any symptoms du ring the study. The measured upper sphincter pressure was well above that previously dcscriheJ in studies of fac­tors increasing upper esophageal sphincter pressure, and the tnplc lu­men, continuously perfused catheter system used here ha::- hcen thought to

underestimate the true sphincter pres­sure compared with sleeve catheters ( l 2-14). Perhaps high upper esopha­geal sphincter pressure explaim the 'lump in rhe throat' feeling and the hypersal1vatilln that is descnbed 111 this form of partial epilepsy. Possibly it is a protective mechamsm to prevent aspi­ration from gastroesophageal reflux during these episodes.

The pathophysiological mecha­nisms of gastroesophageal reflux are multifactorial. Enological mechanisms include a low basal sphincter pressure, inappropriate sphincter rclaxanon which is enher synchronous or asyn­chronous with swallowing, and tran­sient increases in intra-abdominal or gastric pressure alone or in comh111a­tion with the above mechanisms (15,16). Resting tone in the lower esophageal sphinc ter 1s influenced hy neural factors. The typical electroen­cephalogram seen in BFEC is that of centrotemporal spikes on normal ha<.k­ground activ ity. Perhaps discharges from this area md1recrly lead to map­propriatc relaxation of the lower esophageal sphmcter, resulting m re­flux, and an associated e levation in up-

pediatric patient. Gastmenternl Clin Nllrth Am 1989;18:315-37.

5. Came IJ. A H1s1nncal Review of thl· Clm1cal Cnnscquen<.:l'\ of H1aral I lcrma (p.irt ,al thorauc st111rn1d1) ,md Gasrrnes11phageal Reflux. In, Gellin SS, ed. Gastroesophageal Reflux. Rep,m of the Ros, Confl'rence on Ped1atrn: Research. C11lumhu,. Ross, 1979· 1-l 2.

6. Carrn-Sm1th AC, Mad1id.1 11, Bui:nl'r JD, Gall DG, Swtt RB. The role of gastmc,ophageal reflux m pe<lmtnc <lysphag1a. J Pe<liatr Gastrocnteml Nutr l991;12:l59-65.

7. Scott RB, O'LiuJ.!hlm EV. Gall DG. Oastrocsophagl'al reflux in pauents with cystt( fihrosis. J Ped1.1rr 1985;106:22.3-7.

8. Mahony MJ, Migltavan:a M, '>ptt: L,

CAN J GASTROENTEROI. VOL 8 NO I JANUARY/FFRRUARY 1994

Gastroesophageal reflux and epilepsy

per esophageal sphincter pressure. lt is possible that centrally ml.!diated me(han1sms play a higger role 111 the pachophys1ology of gastrocsophageal reflux rhan is realized; iJcally, m dus case, chi~ might have bl.!en examined wtth simultaneous electrocnci.!phalo­gram and esophageal pl I mon1tonng. Due to the frequency llf sei:urcs, we were unable ro ohtam pt:nmsston to perform these further studies, but they should be considered 1f the opportunity arise:,, 111 future <.<1scs.

CONCLUSIONS C,astroesophageal reflux, whteh 1s

common in infancy, b,s commonly presents in childhood. In the appro­priate settmg, one should consider the possibility llf an associated under lying disease state. HFE< 1s the most common form of p,irtial t:pilepsy in childhood and, 111 the absence of ob­served seizures, the clinical history of ornpharynge,11 symptoms may suggest gastroe,ophagcal reflux. In the pre­sented GN:, reflux was do<.umenced 111 associa tion with RFEC which rcsolve<l with trl'Htmcnt of the epilepsy. G iven the like lihood of central effects on esophageal functmn , further studies arc required of oth l.!r ch ildren with BFEC or cnmplcx partial ~e1zurcs with oropharyngeal phenomena to evalu­ate the prc~cncc of gastroesophageal reflux anJ to ohra111 more precise in­

formation on the effoct of seizure ac­tivity on upper esophagl.'al sph111ctcr pressure.

Milla PJ. M11tor disorder, of the oe,ophagll', m gastr11esophageal reflux. Arch Dis Ch ild 1988;6 3: I H3-8.

9. Jolley SG, I krhst JJ, John,on DG, M;itl,1k ME, Boni LS. Surgery 111

childrL·n with gast rocsophageal reflux an<l re,p1ralnr} symptnm,. J Pc,li.itr 1980;96; 194-8.

10. Mahony MJ, Kennedy JD, Leaf H, Matthew DJ, Milla PJ. Rram stem gl1oma pre,entmg as J.?'ilstroesnphagcal rdlux. Arch Dis Child 1987;62:731-7.

11. A1cardi J. Simple partial seizures. In: Epilepsy m Ch1l<lren {lnternauonal Review of Chil<l NeurokiJ,?'y Scnes). New Y,irk: Raven Press, l986:l l9-128.

12. Davidson O P, Dent J, WillmgJ. M1,111wrmg of upper esophageal ,phmcrer pressure m children. Gut 1991;32:607 11.

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SHEEHAN ec al

13. Cook lJ, Dent J, Shannon S, Collins SM. Measurement of upper esophageal sphincter pressure: Effect of acute emotional stress. Gastroenterology 1987:93:526-32.

14. Kahrilas PJ, Dodds WJ, Dent), Haeberle B, Hogan WJ, Arndorfer RC.

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Effect of sleep, spontaneous gasrroesophageal reflux and a meal on upper esophageal sphincter pressure in normal human volunteers. Gastrocnterology 1987;92:466-71.

15. Milla PJ. Reflux vomiting. Arch Dis Child. 1990;65:996-9.

16. Werlin SL, Dodds WS, Hogan WJ, er al. Mechanics of gastrocsophageal reflux in children. J Pe<liatr I 980;97:244-9.

17. Euler AR, Byrne WJ. Twency-four hour esophageal intraluminal pH probe testing: A comparative analysis. Gastroenterology 1981;80:957-61.

CAN J GASTROENTEROL VOL 8 Nol JANUARY/FEBRUARY 1994

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