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Parathyroid adenoma, parathyroid crisis, and acute pancreatitis in an adolescent
Volume 83 Number 2
12. Monnens, L., and Schretlen, E.: Haemolyticuremic syndrome, Lancet 2: 735, 1968. I3. Gilchrist, G. S., and Liberman, E.: Haemolytic-uremic syndrome and hepariu therapy, Lancet 2: 1069, 1969. 14. Kaplan, B. S., Katz, J., Krawitz, S., and Lurie, A . : An analysis of the results of therapy in 67 cases of the hemolytic-uremic syndrome, J. PEDIATR.78: 420, 1971.
Parathyroid adenoma, parathyroid criJis, and acute pancreatitis in an adolescent Fredrie Daum, M.D., John F. Rosen, M.D.,* and Scott J. Boley, M.D., Bronx, N. Y.
T ~ E V I R S T case of f a t a l p r i m a r y hyperparathyroidism was reported by Dawson and Struthers 1 in 1923. Subsequently, such cases have been referred to as acute parathyroid intoxication , parathyroid crisis, acute hyperparathyroidism, parathormone intoxication, parathyrotoxicosis, calcium intoxication, parathyroi d poisoning, and hyperparathyroid crisis. Payne and Fitchett, 2 in a review of hyperparathyroid crisis, accepted only those cases in which there was an increase in serum calcium greater than 15 mg. per cent, progression of gastrointestinal, cardiovascular, a n d / o r central nervous system disturbances, and a rising blood urea nitrogen concentration or other evidence of renal failure. Twenty of their 70 patients survived. Of 94 cases of parathyroid crisis reviewed in the From the Division of Adolescent Medicine, Department o[ Pediatrics, and Pediatric Surgical Service, Department of Surgery, Montefiore Hospital and Medical Center, and the Albert Einstein College of Medicine. Reprint address: Scoll I. Boley, M.D., Montefiore Hospital and Medical Center, Department o[ Surgery, 111 E. 210th St., Bronx, N. Y. 10467. "aRedpient ol a grant #ore the John A. Hart[oral Foundation, Inc.
Brie[ clinical and laboratory observations
275
15. Katz, J., Lurie, A., and Kaplan, B.: Haemolytic-uremic syndrome and heparin therapy, Lancet 2: 700, 1969. 16. Brain, M. C.: The hemolytlc-uremic syndrome, Sem. Hematol. 6: 162, 1969. 17. Gianantonio, C. A., Vitacco, M., Mendilaharzu, F., and Gallo, G.: The hemolyticuremic syndrome, J. PEnlATR. 72: 757, 1968.
literature, only four are in children. Three of these four children had parathyroid crises resulting from a parathyroid adenoma3-5; all survived. T h e fourth patient inadvertently received high doses of parenteral parathyroid h o r m o n e for six days. 6 W h e n the error was recognized and therapy with parathyroid h o r m o n e was discontinued, there was spontaneous recovery. Although hypercalcemia and parathyroid crisis have been associated with acute pancreatitis in adults with parathyroid adenomas, this relationship has not been appreciated previously in children. O u r case represents only the fourth child under 15 years of age to be reported with parathyroid crisis and the first such child with documented clinical and chemical evidence of acute pancreatitis.
CASE REPORT A 12-year-old girl was admitted January 24, 1972, with a 5 day history of intractable vomiting, nonradiating, crampy, periumbilical pain, a weight loss of 20 pounds, low-grade fever and chills, and decreased frequency of urination and bowel movements. Significant past history revealed several episodes of vomiting and abdominal pain dating back to 1968. There was no history of abdominal trauma, jaundice, drug abuse, parasitic infestation, or significant infectious disease. Physical examination revealed a thin female with a blood pressure of 110/70 ram. Hg, pulse rate 120 per minute, respiratory rate 12 per minute, rectal temperature 99.8 ~ F., weight 84 pounds, and height 62 inches. Skin turgor was poor and mucous membranes were dry. No neck masses were palpated. The abdomen was scaphoid with slight muscle guarding in the right upper quadrant, tenderness in the left upper quadrant, and generalized rebound tenderness. Laboratory data revealed a hematocrit of 48
276
Brie[ clinical and laboratory observations
per cent and a white blood cell count of 18,600 with a shift to the left. Blood urea nitrogen was 75 mg. per cent. Serum electrolyte concentrations were normal, but the serum amylase value was 1,800 Somogyi units. Therapy consisted of continuous nasogastric suction and intravenous hydration. The serum calcium concentration at the time of admission was greater than 15 rag. per cent. A repeat determination was greater than 21 mg. per cent with an ionized calcium value of 11 rag. per cent.* An electrocardiogram was normal. The serum amylase value was now 516 Somogyi units. The hematocrit had fallen to 42 per cent. Vital signs were unchanged. Forty-eight hours after admission, the patient's abdominal pain began to radiate to the back. At the same time, serum amylase was 178 Somogyi units, blood urea nitrogen 27 rag. per cent, total serum calcium 18 mg. per cent, ionized calcium 9.5 rag. per cent, phosphorus 2.2 rag. per cent, magnesium 1.4 mEq. per liter, and alkaline phosphatase, 3.8 Bessey-Lowry units. ~ Radiographs revealed subperiosteal resorption of bone in the hands and along the medial proximal left humerus. Primary hyperparathyroidism with associated acute pancreatitis was diagnosed, and hypertonic saline was infused over an 8 hour period without decrease in the serum calcium concentration. Calcitonin was unavailable and because of potential complications isotonic sodium sulfate, ethylenediaminetetraacetate, and furosemide were contraindicated. A nodule was palpated on the right side of the neck, and 72 hours after admission a surgical exploration of the neck was performed. Frozen sections of a 1.5 x 2.5 cm. mass excised from the inferior posterior border of the right thyroid gland revealed a parathyroid adenoma. The right and left superior parathyroid glands were grossly normal. Biopsy of the left inferior parathyroid gland revealed normal parathyroid tissue. The mediastinum was not explored. Twenty-four hours after the operation, the abdominal findings had disappeared. Forty-eight hours after the operation, concentrations of serum calcium and phosphorus had returned to normal. The blood urea nitrogen decreased to 10 mg. per cent. Urinary concentration of amylase remained normal, and she was discharged without complications.
*Serum ionized calcium (Ca++) was measured by means of a flow-through calcium selective electrode (Model 99-20, Orion Research Corporation, Cambridge, Mass.).
The Journal o[ Pediatrics August 1973
DISCUSSION
Verger and associates 7 found parathyroid adenoma to be the most c o m m o n etiology of primary hyperparathyroidism in children. In 1969, Canlorbe and associates s compiled 46 cases of parathyroid adenoma in children 15 years of age or less reported since 1926. No mention was made of patients evaluated by J u d d and associates ~ in 1966 or Verger in 1968. Subsequently, only three other cases have been described. TM 11 Thus, to date, there are only 52 reports of parathyroid adenoma in children, including the present case. Of the 51 previously reported cases, three of the children had symptoms and signs of parathyroid crisis as previously defined. Each of them had surgical excision of a solitary parathyroid adenoma. O f note is that, although recurrent episodes of abdominal pain and vomiting were always present, apparently panereatitis was not suspected. Although M c C a n n had alluded to this relationship as early as 1934, and James and Richards, 1~ in 1956, referred to "abdominal pain being an almost constant accompanying symptom," it was not until 1957 that Cope and associates 1~ emphasized the relationship between primary hyperparathyroidism and pancreatitis. Although it is now estimated that 10 to 20 per cent of all adults with primary hyperparathyroidism have associated pancreatitis, until this report there had been no mention of the simultaneous occurrence of these two entities in children. Several postulates concerning the mechanism of this interrelationship have been previously entertained. 1'~-1~ O u r patient's history and roentgenologic survey indicate recurrent episodes of pancreatitis and longstanding primary hyperparathyroidism. O u r experience suggests that symptoms and signs previously attributed to parathyroid crisis may be, in part, the result of associated pancreatitis. The hypovolemia and corresponding elevation of blood urea nitrogen commonly seen with parathyroid crisis can more clearly be explained by the marked loss in plasma volume associated with acute pancreatitis. T h e abdominal pain and pernicious vomiting similarly are characteristic of panereatitis. It is our impression that
Volume 83 Number 2
p a n c r e a t i c i n f l a m m a t i o n is far more c o m m o n in c h i l d r e n with p r i m a r y h y p e r p a r a t h y r o i d ism than was previously believed. A conflict remains as to the most a p p r o p r i ate t h e r a p y for p a r a t h y r o i d crisis resulting from p a r a t h y r o i d adenoma. W h i l e there are advocates of p r o t r a c t e d efforts to lower the serum c a l c i u m prior to the operation, it remains the impression of most t h a t there should be little delay in emergency p a r a thyroid g l a n d e x p l o r a t i o n and excision of the adenoma. 2 T o date, there have been no reports of c a r d i a c a r r h y t h m i a s or operative complications in children u n d e r g o i n g surgery with h y p e r c a l c e m i a . T h e benign i n t r a - a n d postoperative course of our own p a t i e n t supports the safety and desirability of p r o m p t surgical intervention.
Brief clinical and laboratory observations
6. 7.
8. 9. 10. 11. 12. 13.
REFERENCES 1. Dawson, J. W., and Struthers, J. W.: Generalized psteitis fibrosa with parathyroid tumor and metastatic calcification, Edinburgh Med. J. 30: 421, 1923. 2. Payne, R. L., Jr., and Fitchett, C. W.: Hyperparathyroid crisis: Survey of the literature and a report of two additional cases, Ann. Surg. 161: 737, t965. 3. Fentz, V.: Hypertensive encephalopathy in a child, Acta Neurol. Scan& 38: 307, 1962. 4. Harmon, M.: Parathyroid adenoma in a child, Am. J. Dis. Child. 91: 313, 1956. 5. Reinfrank, R. F., and Edwards, T. L.: Hyper-
Dienceploa l ic autonomic epilepsy caused by a neoplasm
14.
15.
16.
277
calcemic crisis in a child, J. A. M. A. 468: 178, 1961. Lowenberg, H., and Ginsburg, T. M.: Acute hypercaleemia: Report of a case, J. A. M. A. 99: 1166, 1932. Verger, P., Couraud, L., Guillard, J. M., Sandier, B., Azanza, X., and Gambier, P.: Hyperparathyroidie majeure par adenome parathyroidien chez l'enfant, Ann. Pediatr. 17: 17, 1971). Canlorbe, P., Lagrue, G., and Bader, J. C.: Adenomes parathyroidiens de l'enfant operes, Ann. Pediatr. 16: 573, 1969. Judd, D. R., Iteimburger, I., and Johnston, C., Jr.: Parathyroid adenoma, Ann. Surg. 164: 1077, 1966. Bjernulf, A., Hall, K., Sjogren, I., and Werner, I.: Primary hyperparathyroidism in children, Acta Paediatr. Scand. 59: 249, 1970. Ghita, N., Bercovici, M., and Dimitriu, V.: Consideratii asupra hiperparatlroidismului primar la eopil, Pediatria 18: 451, 1969. James, P. R., and Richards, P. G.: Parathyroid crisis, Arch. Surg. 72: 553, 1956. Cope, O., Culver, P. J., Mixter, C. G., and Nardi, G. L.: Pancreatitis, a diagnostic clue to hyperparathyroidism, Ann. Surg. 145: 857, 1957. Haverbaek, B. J , Dyce, B., Hallie, B., and Edmondson, H. A.: Trypsin, trypsinogen, and trypsin inhibitor in human pancreatic juice, Am. J. Med. 29: 424, 1960. Patoyan, E., Paloyan, D., and Harper, A. V.: The role of glucagon hypersecretion in the relationship of pancreatitis and hyperparathyroidism, Surgery 62: 167, 1967. Hennkson, C.: Acute hyperparatbyroidism and acute panereatitis, Acta Pathol. Microbiol. Scand. 50: 42, 1960.
nausea, a n d vomiting. L e n n o x 1 stated that such " a u t o n o m i c seizures seem relatively benign with a strong tendency to fade with the years." Recently, a 20-month-old child presented with a u t o n o m i c epilepsy a n d was found to h a v e an astroblastoma of the diencephalon.'
Gail E. Solomon, M.D., N e w York, N. Y. CASE REPORT seizures are characterized by attacks of flushing, irregular respiration, altered h e a r t rate, labile blood pressure and body t e m p e r a t u r e , diaphoresis, dilated pupils,
A U T O N O M I C
From the Departments o[ Neurology and Pediatrics, Cornell University MgdicaI College. Reprint address: Department o[ Neurology, Cornell University Medical College, 1300 York Ave., New York, N. Y. 10021.
A 20-month-old white girl was evaluated at The New York Hospital with a history of facial flushing, diaphoresis, gagging, and salivation of 3 months' duration, followed by the appearance of circumoral pallor, staring, and then lying down without actual loss of consciousness. These "spells" lasted from one to two minutes and occurred as often as five to six times per day. On one occasion the patient slept for ten minutes after the end of the "spell."
12. Monnens, L., and Schretlen, E.: Haemolyticuremic syndrome, Lancet 2: 735, 1968. I3. Gilchrist, G. S., and Liberman, E.: Haemolytic-uremic syndrome and hepariu therapy, Lancet 2: 1069, 1969. 14. Kaplan, B. S., Katz, J., Krawitz, S., and Lurie, A . : An analysis of the results of therapy in 67 cases of the hemolytic-uremic syndrome, J. PEDIATR.78: 420, 1971.
Parathyroid adenoma, parathyroid criJis, and acute pancreatitis in an adolescent Fredrie Daum, M.D., John F. Rosen, M.D.,* and Scott J. Boley, M.D., Bronx, N. Y.
T ~ E V I R S T case of f a t a l p r i m a r y hyperparathyroidism was reported by Dawson and Struthers 1 in 1923. Subsequently, such cases have been referred to as acute parathyroid intoxication , parathyroid crisis, acute hyperparathyroidism, parathormone intoxication, parathyrotoxicosis, calcium intoxication, parathyroi d poisoning, and hyperparathyroid crisis. Payne and Fitchett, 2 in a review of hyperparathyroid crisis, accepted only those cases in which there was an increase in serum calcium greater than 15 mg. per cent, progression of gastrointestinal, cardiovascular, a n d / o r central nervous system disturbances, and a rising blood urea nitrogen concentration or other evidence of renal failure. Twenty of their 70 patients survived. Of 94 cases of parathyroid crisis reviewed in the From the Division of Adolescent Medicine, Department o[ Pediatrics, and Pediatric Surgical Service, Department of Surgery, Montefiore Hospital and Medical Center, and the Albert Einstein College of Medicine. Reprint address: Scoll I. Boley, M.D., Montefiore Hospital and Medical Center, Department o[ Surgery, 111 E. 210th St., Bronx, N. Y. 10467. "aRedpient ol a grant #ore the John A. Hart[oral Foundation, Inc.
Brie[ clinical and laboratory observations
275
15. Katz, J., Lurie, A., and Kaplan, B.: Haemolytic-uremic syndrome and heparin therapy, Lancet 2: 700, 1969. 16. Brain, M. C.: The hemolytlc-uremic syndrome, Sem. Hematol. 6: 162, 1969. 17. Gianantonio, C. A., Vitacco, M., Mendilaharzu, F., and Gallo, G.: The hemolyticuremic syndrome, J. PEnlATR. 72: 757, 1968.
literature, only four are in children. Three of these four children had parathyroid crises resulting from a parathyroid adenoma3-5; all survived. T h e fourth patient inadvertently received high doses of parenteral parathyroid h o r m o n e for six days. 6 W h e n the error was recognized and therapy with parathyroid h o r m o n e was discontinued, there was spontaneous recovery. Although hypercalcemia and parathyroid crisis have been associated with acute pancreatitis in adults with parathyroid adenomas, this relationship has not been appreciated previously in children. O u r case represents only the fourth child under 15 years of age to be reported with parathyroid crisis and the first such child with documented clinical and chemical evidence of acute pancreatitis.
CASE REPORT A 12-year-old girl was admitted January 24, 1972, with a 5 day history of intractable vomiting, nonradiating, crampy, periumbilical pain, a weight loss of 20 pounds, low-grade fever and chills, and decreased frequency of urination and bowel movements. Significant past history revealed several episodes of vomiting and abdominal pain dating back to 1968. There was no history of abdominal trauma, jaundice, drug abuse, parasitic infestation, or significant infectious disease. Physical examination revealed a thin female with a blood pressure of 110/70 ram. Hg, pulse rate 120 per minute, respiratory rate 12 per minute, rectal temperature 99.8 ~ F., weight 84 pounds, and height 62 inches. Skin turgor was poor and mucous membranes were dry. No neck masses were palpated. The abdomen was scaphoid with slight muscle guarding in the right upper quadrant, tenderness in the left upper quadrant, and generalized rebound tenderness. Laboratory data revealed a hematocrit of 48
276
Brie[ clinical and laboratory observations
per cent and a white blood cell count of 18,600 with a shift to the left. Blood urea nitrogen was 75 mg. per cent. Serum electrolyte concentrations were normal, but the serum amylase value was 1,800 Somogyi units. Therapy consisted of continuous nasogastric suction and intravenous hydration. The serum calcium concentration at the time of admission was greater than 15 rag. per cent. A repeat determination was greater than 21 mg. per cent with an ionized calcium value of 11 rag. per cent.* An electrocardiogram was normal. The serum amylase value was now 516 Somogyi units. The hematocrit had fallen to 42 per cent. Vital signs were unchanged. Forty-eight hours after admission, the patient's abdominal pain began to radiate to the back. At the same time, serum amylase was 178 Somogyi units, blood urea nitrogen 27 rag. per cent, total serum calcium 18 mg. per cent, ionized calcium 9.5 rag. per cent, phosphorus 2.2 rag. per cent, magnesium 1.4 mEq. per liter, and alkaline phosphatase, 3.8 Bessey-Lowry units. ~ Radiographs revealed subperiosteal resorption of bone in the hands and along the medial proximal left humerus. Primary hyperparathyroidism with associated acute pancreatitis was diagnosed, and hypertonic saline was infused over an 8 hour period without decrease in the serum calcium concentration. Calcitonin was unavailable and because of potential complications isotonic sodium sulfate, ethylenediaminetetraacetate, and furosemide were contraindicated. A nodule was palpated on the right side of the neck, and 72 hours after admission a surgical exploration of the neck was performed. Frozen sections of a 1.5 x 2.5 cm. mass excised from the inferior posterior border of the right thyroid gland revealed a parathyroid adenoma. The right and left superior parathyroid glands were grossly normal. Biopsy of the left inferior parathyroid gland revealed normal parathyroid tissue. The mediastinum was not explored. Twenty-four hours after the operation, the abdominal findings had disappeared. Forty-eight hours after the operation, concentrations of serum calcium and phosphorus had returned to normal. The blood urea nitrogen decreased to 10 mg. per cent. Urinary concentration of amylase remained normal, and she was discharged without complications.
*Serum ionized calcium (Ca++) was measured by means of a flow-through calcium selective electrode (Model 99-20, Orion Research Corporation, Cambridge, Mass.).
The Journal o[ Pediatrics August 1973
DISCUSSION
Verger and associates 7 found parathyroid adenoma to be the most c o m m o n etiology of primary hyperparathyroidism in children. In 1969, Canlorbe and associates s compiled 46 cases of parathyroid adenoma in children 15 years of age or less reported since 1926. No mention was made of patients evaluated by J u d d and associates ~ in 1966 or Verger in 1968. Subsequently, only three other cases have been described. TM 11 Thus, to date, there are only 52 reports of parathyroid adenoma in children, including the present case. Of the 51 previously reported cases, three of the children had symptoms and signs of parathyroid crisis as previously defined. Each of them had surgical excision of a solitary parathyroid adenoma. O f note is that, although recurrent episodes of abdominal pain and vomiting were always present, apparently panereatitis was not suspected. Although M c C a n n had alluded to this relationship as early as 1934, and James and Richards, 1~ in 1956, referred to "abdominal pain being an almost constant accompanying symptom," it was not until 1957 that Cope and associates 1~ emphasized the relationship between primary hyperparathyroidism and pancreatitis. Although it is now estimated that 10 to 20 per cent of all adults with primary hyperparathyroidism have associated pancreatitis, until this report there had been no mention of the simultaneous occurrence of these two entities in children. Several postulates concerning the mechanism of this interrelationship have been previously entertained. 1'~-1~ O u r patient's history and roentgenologic survey indicate recurrent episodes of pancreatitis and longstanding primary hyperparathyroidism. O u r experience suggests that symptoms and signs previously attributed to parathyroid crisis may be, in part, the result of associated pancreatitis. The hypovolemia and corresponding elevation of blood urea nitrogen commonly seen with parathyroid crisis can more clearly be explained by the marked loss in plasma volume associated with acute pancreatitis. T h e abdominal pain and pernicious vomiting similarly are characteristic of panereatitis. It is our impression that
Volume 83 Number 2
p a n c r e a t i c i n f l a m m a t i o n is far more c o m m o n in c h i l d r e n with p r i m a r y h y p e r p a r a t h y r o i d ism than was previously believed. A conflict remains as to the most a p p r o p r i ate t h e r a p y for p a r a t h y r o i d crisis resulting from p a r a t h y r o i d adenoma. W h i l e there are advocates of p r o t r a c t e d efforts to lower the serum c a l c i u m prior to the operation, it remains the impression of most t h a t there should be little delay in emergency p a r a thyroid g l a n d e x p l o r a t i o n and excision of the adenoma. 2 T o date, there have been no reports of c a r d i a c a r r h y t h m i a s or operative complications in children u n d e r g o i n g surgery with h y p e r c a l c e m i a . T h e benign i n t r a - a n d postoperative course of our own p a t i e n t supports the safety and desirability of p r o m p t surgical intervention.
Brief clinical and laboratory observations
6. 7.
8. 9. 10. 11. 12. 13.
REFERENCES 1. Dawson, J. W., and Struthers, J. W.: Generalized psteitis fibrosa with parathyroid tumor and metastatic calcification, Edinburgh Med. J. 30: 421, 1923. 2. Payne, R. L., Jr., and Fitchett, C. W.: Hyperparathyroid crisis: Survey of the literature and a report of two additional cases, Ann. Surg. 161: 737, t965. 3. Fentz, V.: Hypertensive encephalopathy in a child, Acta Neurol. Scan& 38: 307, 1962. 4. Harmon, M.: Parathyroid adenoma in a child, Am. J. Dis. Child. 91: 313, 1956. 5. Reinfrank, R. F., and Edwards, T. L.: Hyper-
Dienceploa l ic autonomic epilepsy caused by a neoplasm
14.
15.
16.
277
calcemic crisis in a child, J. A. M. A. 468: 178, 1961. Lowenberg, H., and Ginsburg, T. M.: Acute hypercaleemia: Report of a case, J. A. M. A. 99: 1166, 1932. Verger, P., Couraud, L., Guillard, J. M., Sandier, B., Azanza, X., and Gambier, P.: Hyperparathyroidie majeure par adenome parathyroidien chez l'enfant, Ann. Pediatr. 17: 17, 1971). Canlorbe, P., Lagrue, G., and Bader, J. C.: Adenomes parathyroidiens de l'enfant operes, Ann. Pediatr. 16: 573, 1969. Judd, D. R., Iteimburger, I., and Johnston, C., Jr.: Parathyroid adenoma, Ann. Surg. 164: 1077, 1966. Bjernulf, A., Hall, K., Sjogren, I., and Werner, I.: Primary hyperparathyroidism in children, Acta Paediatr. Scand. 59: 249, 1970. Ghita, N., Bercovici, M., and Dimitriu, V.: Consideratii asupra hiperparatlroidismului primar la eopil, Pediatria 18: 451, 1969. James, P. R., and Richards, P. G.: Parathyroid crisis, Arch. Surg. 72: 553, 1956. Cope, O., Culver, P. J., Mixter, C. G., and Nardi, G. L.: Pancreatitis, a diagnostic clue to hyperparathyroidism, Ann. Surg. 145: 857, 1957. Haverbaek, B. J , Dyce, B., Hallie, B., and Edmondson, H. A.: Trypsin, trypsinogen, and trypsin inhibitor in human pancreatic juice, Am. J. Med. 29: 424, 1960. Patoyan, E., Paloyan, D., and Harper, A. V.: The role of glucagon hypersecretion in the relationship of pancreatitis and hyperparathyroidism, Surgery 62: 167, 1967. Hennkson, C.: Acute hyperparatbyroidism and acute panereatitis, Acta Pathol. Microbiol. Scand. 50: 42, 1960.
nausea, a n d vomiting. L e n n o x 1 stated that such " a u t o n o m i c seizures seem relatively benign with a strong tendency to fade with the years." Recently, a 20-month-old child presented with a u t o n o m i c epilepsy a n d was found to h a v e an astroblastoma of the diencephalon.'
Gail E. Solomon, M.D., N e w York, N. Y. CASE REPORT seizures are characterized by attacks of flushing, irregular respiration, altered h e a r t rate, labile blood pressure and body t e m p e r a t u r e , diaphoresis, dilated pupils,
A U T O N O M I C
From the Departments o[ Neurology and Pediatrics, Cornell University MgdicaI College. Reprint address: Department o[ Neurology, Cornell University Medical College, 1300 York Ave., New York, N. Y. 10021.
A 20-month-old white girl was evaluated at The New York Hospital with a history of facial flushing, diaphoresis, gagging, and salivation of 3 months' duration, followed by the appearance of circumoral pallor, staring, and then lying down without actual loss of consciousness. These "spells" lasted from one to two minutes and occurred as often as five to six times per day. On one occasion the patient slept for ten minutes after the end of the "spell."