CASE REPORT

Hiccups: An Unusual Manifestation of an Abdominal Aortic Aneurysm /

Robert J. Stine, MD* Sally J. Trued, MDt Baltimore, Maryland

A patient with hiccups was found to have an abdominal aortic aneurysm that subsequently ruptured. We believe that a leaking abdominal aortic aneurysm led to an ileus-induced distention of the splenic flexure of the colon with consequent diaphragmatic irritation and phrenic nerve stimulation. This led to persistent hiccups as a result of repetitive stimulation of the reflex arc mediating hiccups. Persistent hiccups require investigation for an underlying organic etiology, and a leaking abdominal aortic aneursym should be included in the differential diagnosis. Stine RJ, Trued SJ: Hiccups: an unusual manifestation of an abdominal aortic aneurysm. JACEP 8:368-370, September 1979. hiccups; aortic aneurysm, abdomina/

INTRODUCTION

Hiccups are repetitive involuntary spasmodic contractions of the inspiratory muscles, particularly the diaphragm, followed by abrupt closure of the glottis, which terminates the inflow of air and produces the characteristic sound. 1-3 Unlike vomiting, hiccups apparently serve no useful function. 1 Transient hiccups are usually benign. Persistent hiccups, however, may be a sign of serious underlying organic disease. 3 We describe what is, to our knowledge, the first reported case of hiccups as a presenting manifestation of an abdominal aortic aneurysm. CASE REPORT

A 70-year-old man with a history of h y p e r t e n s i o n presented to the emergency department at Baltimore City Hospital with unrelenting hiccups of three days duration and left shoulder pain. He had postprandial emesis during this time but no chest pain, abdominal pain, pulmonary symptoms, or bowel abnormalities. There was no history of alcohol abuse. One year ago the patient was hospitalized elsewhere with abdominal pain, emesis, and hiccups of less severity. An upper gastrointestinal series at that time revealed a markedly deformed duodenal bulb. He was treated with antacids and his symptoms resolved. On current physical examination, the patient had hiccups but was in no acute distress. Blood pressure was 170/98 mm Hg; pulse rate, 108 beats/min; respiratory rate, 18/min; and temperature, 38.2 C (100.7 F). Abnormal physical From the D'ivision of Emergency Medicine, the Baltimore City Hospital,* and the Department of Emergency Medicine, the Johns Hopkins Hospital;t and the Division of Emergency Medicine, The Johns Hopkins Uni,Jersity School of Medicine; Baltimore, Maryland.*t Address for reprints: RobertJ. Stine, MD, Division of Emergency Medicine, Baltimore City Hospital, 4940 Eastern Avenue, Baltimore, Maryland 21224. 8:9 (September) 1979

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findings were l i m i t e d to the abdomen a n d c o n s i s t e d of h y p o a c t i v e bowel sounds a n d a t e n d e r a o r t a palpable to th~ l e f t of t h e m i d l i n e , w i t h a w i d t h ' o f a p p r o x i m a t e l y 7 cm. Results of the rectal examination were w i t h i n n o r m a l l i m i t s and the stool guaia~ negative. An a b d o m i n a l aortic a n e u r y s m was suspected and confirmed by an emergency sonogram. Additional labor~tory d a t a included a hematocrit r e a d i n g of 44 ml/dl; WBC count of 26,800/cu m m with a shift to the left 0;n t h e differential count; serum electrolytes within normal limits; blood u r e a nitrogen, 23 mg/dl; blood glucQse, 151 mg/dl; s e r u m amylase, 58 Sbmogyi units/dl; and serum bilirubin, 1.0 mg/dl. A n electrocardiog r a m showed a sinus t a c h y c a r d i a , left a n t e ~ o r hemoblock, a n d nonspecific ST-T wave changes. C h e s t and abd o m i n a l x-ray films showed conside r a b l e gas in the splenic flexure of the ~olon and a n e l e v a t e d left hemid i a p h r a g m (Figure). The p a t i e n t was a d m i t t e d to the hospital. Six hours a f t e r admission he developed ieft lower q u a d r a n t abdominal pain with an associated palpable tense mass and became hypotensive with a blood pressure of 68/50 m m Hg. His h e m a t o c r i t reading was 36%. The p a t i e n t was t a k e n i m m e d i a t e l y to the o p e r a t i n g room, a n d an e x p l o r a t o r y l a p a r o t o m y rev e a l e d a r u p t u r e d aortic a n e u r y s m w i t h a 7 cm i n f r a r e n a l t e a r of the left lateral portion of the aorta. The peritoneal cavity was filled with blood, a n d t h e r e was a n e x t e n s i v e r e t r o p e r i t o n e a l h e m a t o m a . A n ane u r y s m e c t o m y was p e r f o r m e d , a n d a d a c r o n g r a f t w a s i n s e r t e d . Posto p e r a t i v e l y , t h e p a t i e n t w a s rec u p e r a t i n g s a t i s f a c t o r i l y . On t h e 13th hospital day, however, he suffered a c a r d i o p u l m o n a r y a r r e s t and could not be resuscitated. A n autopsy was not obtained. DISCUSSION Hiccups are reflex in origin. The a f f e r e n t l i m b of t h e r e f l e x a r c is t h o u g h t to include the vagus nerve, t h e p h r e n i c n e r v e , ~nd t h e 6 t h t h r o u g h 12th t h o r a c i c s e g m e n t s of the s y m p a t h e t i c chain. 1-3 The center of the reflex arc is located in the cervical cord a t t h e level of the t h i r d t h r o u g h f i f t h c e r v i c a l s.egments. 2 Connections to h i g h e r centers likely exist. 1 Efferent i m p u l s e s are transm i t t e d p r i n c i p a l l y by t h e p h r e n i c nerve. 1-3 Other efferent p a t h w a y s to t h e r e s p i r a t o r y i n t e r c o s t a l muscles and the glottis l i k e l y are involved. 2,3 48/369

F i g . Chest x-ray film showing collection of gas in the splenic flexure of the colon and elevation of the left hernidiaphragm. Hiccups r e s u l t from s t i m u l a t i o n of the reflex arc a n d can be classified as c e n t r a l or p e r i p h e r a l in origin depending upon the site 0fstimulation. 2 C e n t r a l causes include diseases of the c e n t r a l n e r v o u s s y s t e m (eg, encephalitis, meningitis, neurosypl/ilis, cerebrovascular insufficiency or accident, tumor, injury, deg e n e r a t i v e disease); 2-4 metabolic and electrolyte d i s t u r b a n c e s (eg, uremia, alcohol i n t o x i c a t i o n , h y p o k a l e m i a , hypocalcemia);2,3, 5 psychogenic factors; 2-4 a n d c e r t a i n d r u g s (eg, sulfonamides, short-acting barbiturates).2, 6 Peripheral etiologies include those processes t h a t s t i m u l a t e the afferent fibers of the vagus nerve, phrenic nerve, and sympathetic chain from T6 t h r o u g h T12. They include lesions in the h e a d and neck (eg, e n l a r g e d g l a n d , t u m o r , c y s t , aneurysm, scalenus anticus syn-

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drome, h a i r in t h e e x t e r n a l a u d i t o r y canal);3,4, 7 lesions in t h e thorax (eg, a d e n o p a t h y , t u m o r , a n e u r y s m , cardiomegaly, myocardial infarction, pericarditis, pneumonia, pleurisy, esophagitis, diaphragmatic hern i a ) ; 2-4 i n t r a - a b d o m i n a l d i s o r d e r s (eg, peritonitis, abscess, cho]ecystitis, p a n c r e a t i t i s , g a s t r i c i r r i t a t i o n or dilatation, gastritis, peptic ulcer, t u m o r , i n t e s t i n a l d i s t e n t i o n o r obstruction, u r i n a r y t r a c t dis0rder); 2-4 and surgical and postoperative states. 2-4 Abdominal aortic aneurysms m a y be a s y m p t o m a t i c and discovered o n l y on r o u t i n e ph:~sical e x a m i n a tion. The m a j o r i t y of n o n l e a k i n g abd o m i n a l a n e u r y s m s , h o w e v e r , pres e n t w i t h one o r m o r e of s e v e r a l common symptoms: g a s t r o i n t e s t i n a l d i s t u r b a n c e s (eg, a b d o m i n a l p a i n or discomfort, n a u s e a and/or vomiting, constipation or diarrhea, anorexia '

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and/or weight loss) t h o u g h t to be due to bowel compression or vascular ins u f f i c i e n c y ; s b a c k p a i n from e n croachment of the a n e u r y s m a l mass on the vertebral bodies; 9 and sensation or discovery of a pulsatile mass by the p a t i e n t 2 A r u p t u r e d or sign i f i c a n t l y l e a k i n g "aneurysm commonly presents with the t r i a d of abdominal pain, expanding pulsatile abdominal mass, and hypotension2 There are also a n u m b e r of unusual p r e s e n t a t i o n s for which to consider the diagnosis of abdominal aortic a n e u r y s m . These include symptoms of s m a l l bowel (usually duodenal) compression or obstruction from the e n l a r g i n g mass; s g a s t r o i n t e s t i n a l h e m o r r h a g e from aorto-enteric fistula formation, u s u a l l y occurring at the t h i r d portion of the duodenum;9 jaundice from common duct compression; s s u p e r i o r m e s e n t e r i c a r t e r y thrombosis with a malabsorptionlike syndrome; l° inferior mesenteric a r t e r y t h r o m b o s i s w i t h colon ischemia a n d diarrhea; 1° groin, hip, or scrotal pain; 9 i n t e r m i t t e n t claudication from i n c r e a s i n g t h r o m b o s i s of the a n e u r y s m ; ~° aorto-caval f i s t u l a formation w i t h cardiac failure a n d lower e x t r e m i t y edema; TM peripheral n e r v e s y m p t o m s from a n e u r y s m a l compression of l u m b a r roots or leakage into a tight fascial c o m p a r t m e n t such as the psoas sheath; 9 splenic r u p t u r e from t r a c t i o n on the capsule; ~ a n d s e x u a l d y s f u n c t i o n i n

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males from i n t e r n a l iliac artery occlusion, lo Our p a t i e n t presented with hiccups of three days duration a n d left shoulder pain. He was found to have a n a b d o m i n a l aortic a n e u r y s m t h a t s u b s e q u e n t l y r u p t u r e d . We believe t h a t his hiccups were a m a n i f e s t a tion of a l e a k i n g aortic a n e u r y s m ; his low-grade fever and leukocytosis support this diagnosis. We postulate that the l e a k i n g a n e u r y s m led to a local i l e u s w i t h d i s t e n t i o n of the splenic flexnre of the colon and resultant diaphragmatic irritation, phrenic nerve s t i m u l a t i o n , and initiation of the reflex arc m e d i a t i n g hiccups. Less likely but possible mecha n i s m s c a u s i n g hiccups in this individual include i r r i t a t i o n of the diap h r a g m by blood from the l e a k i n g a n e u r y s m with consequent stimulat i o n of t h e p h r e n i c n e r v e ; d i r e c t s t i m u l a t i o n of the vagus nerve by the ileus-induced dilation of the splenic flexure of the colon; and direct stimulation of the s y m p a t h e t i c c h a i n by t h e a n e u r y s m i t s e l f or the consequent retroperitoneal irritation.

CONCLUSION This case emphasizes t h a t hiccups are not always benign. Persist e n t hiccups d e m a n d a search for underlying organic disease. They may be an u n u s u a l presenting manifestation of a l e a k i n g a b d o m i n a l a o r t i c aneurysm.

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REFERENCES 1. Davis JN: An experimental study of hiccup. Brain 93:851-872, 1970. 2. Salem MR, Baraka A, Rattenborg CC, et al: Treatment of hiccups by pharyngeal stimulation in anesthetized and conscious subjects. J A M A 202:126-130, 1967. 3. Souadjian JV, Cain JC: Intractable hiccup. Etiologic factors in 220 cases. Postgrad Med 43:72-77, 1968. 4. Samuels L: Hiccup. A ten year review of anatomy, etiology, and treatment. Can Med Assoc J 67:315-322, 1952. 5. Thorne MG: Hiccup and heart block. Br Heart J 31:397-399, 1969.

6. Eisenstadt HB: A case of hiccups. J A M A 202:915, 1967.

7. Cardi E: Hiccups associated with hair in the external auditory canal - - successful treatment by manipulation. N Engl J Med 265:286, 1961. 8. Sondheimer FK, Steinberg I: Gastrointestinal manifestations of abdominal aortic aneurysms. A m J Roentgenol 92:1110-1122, 1964. 9. Gore I, Hirst AE: Arteriosclerotic aneurysms of the abdominal aorta: a review. Prog Cardiovasc Dis 16:113-145, 1973. 10. Hardy JD, Timmis HH: Abdominal aortic aneurysms: special problems. A n n Surg 1"/3:945-965, 1971. 11. Askew AR, Wilmshurst CC: Abdominal aortic aneurysm presenting with splenic rupture, and subsequent paraplegia. Vasc Surg 7:253-257, 1973.

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Hiccups: an unusual manifestation of an abdominal aortic aneurysm.

CASE REPORT Hiccups: An Unusual Manifestation of an Abdominal Aortic Aneurysm / Robert J. Stine, MD* Sally J. Trued, MDt Baltimore, Maryland A pati...
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