690
Letters to the Editor
The Journal of Pediatrics April 1976
REFERENCES
1.
2. 3.
4. 5.
Perry TL, Hansen S, Tischler B, and Bunting R: Determination o f heterozygosity for phenylketonuria on the a m i n o acid analyzer, Clin C h e m Acta 18:51, 1967. Dubowitz V: Intellectual impairment in muscular dystrophy, Arch Dis Child 40:296, 1965. Prosser EJ, M u r p h y EG, and T h o m p s o n M W : Intelligence and the gene for D u c h e n n e muscular dystrophy, Arch Dis Child 44:221, 1969. Rosman NP and K a k u l a r BA: Mental deficiency associated with muscular dystrophy, Brain 89:769, 1966. Rosman NP: The cerebral defect a n d m y o p a t h y in Duchenne muscular dystrophy, Neurology 20:329, 1970.
Self-induced subcutaneous emphysema in an adolescent To the Editor: Subcutaneous e m p h y s e m a of the extremities is a serious problem and is usually related to obvious trauma a n d / o r the presence of anerobic organisms in the soft tissues. A n u n u s u a l case of self-induced subcutaneous e m p h y s e m a o f the extremities is reported in an emotionally disturbed adolescent. CASE REPORT
Patient T. R., a 15-year-old black female, was first admitted to the Children's Hospital Medical Center in October, 1974, with a two-week history of weakness in her legs and episodes o f falling. Three weeks before admission she had moved from New Orleans to live with her father w h o m she had not seen in nine years. The patient appeared tired, weak, and in no acute distress. There were n u m e r o u s small scars on the face and trunk. There was a Grade 2/6 systolic m u r m u r heard best along the left lower sternal border. The neurologic examination revealed occasional but not always consistent falling to the left on t a n d e m walking. She was oriented, right-handed, had normal cranial nerve function, bilaterally absent or hypoactive reflexes in all extremities, and inconsistent responses to vibration and position sense in the legs. Muscle strength was normal. Pertinent laboratory data included normal CBC, urinalysis, erythrocyte sedimentation rate, serum concentrations of electrolytes and serum muscle enzyme values. Examination of cerebrospinal fluid on admission was normal as were serum screen testing for drugs, ECG, chest roentgenogram, and electromyogram. The cardiac m u r m u r was thought to be functional. Additional social history revealed that she had first lived with several relatives in Cincinnati and the rural south and then with the mother who allegedly beat her. She had recently expressed a strong desire to live with her father who had a history of paralytic poliomyelitis which resulted in atrophy of the left arm and leg. The patient's falling and weakness slowly improved during a two-week hospital stay. Physical medicine and psychiatric consultations each suggested a hysterical conversion reaction. She was readmitted one month later with a six-day history of swelling and pain in the left arm. On examination there was crepitus in the left hand, forearm, and arm, almost to the shoulder. There was a small, healing puncture wound on the dorsum o f the left hand. Otherwise the physical a n d laboratory examinations were unre-
Fig. 1. Soft tissue air in the left arm and elbow. markable. Roentgenograms of the left a r m revealed no evidence of fracture or radiopaque foreign body. There was significant air within the soft tissues of the arm in the area of the elbow medially and anteriorly (Fig. 1). The right arm was normal radiologically; a chest roentgenogram now showed s u b c u t a n e o u s air along the left axilla and lateral chest wall. The patient vehemently denied needle use. She acknowledged considerable difficulty with her family a n d peers at school and said several boys had been sticking pins in her. The soft tissue air subsided in four days and she was again discharged. Twice in January, 1975, she was seen in the emergency room with a swollen left arm. In early February she appeared at the Cincinnati General Hospital. Again she had pain, swelling, and crepitus of the left arm and now in the left leg. Several probable needle puncture marks were seen on the d o r s u m of the left h a n d and in the left medial ankle area. She was admitted to the surgical service where serious consideration was given to the possibility of an anaerobic infection. White blood count was normal. Repeated blood cultures for aerobic and anaerobic organisms were negative. Roentgenograms again showed air in the soft tissues o f the forearm to the elbow and in the leg from ankle to mid-thigh. The patient again insisted she had not inflicted needle marks on herself nor injected air. An open surgical biopsy of the soft tissue of the left mid-thigh was performed which was normal and from which no organisms grew. The attending physicians as well as the family were now convinced that the problem was self-induced. A 12 ml syringe and needle, identical to those used at Children's Hospital Medical Center, was found in her room at home. Psychologic testing revealed a full scale IQ of 61. The psychologist felt she was very depressed. She was a person of at least borderline intelligence whose functioning was impaired by emotional problems. The patient was hospitalized for six weeks a n d a behavior
Volume 88 Number 4, part 1
modification program begun. Residential placement in an adolescent group home was r e c o m m e n d e d but the parents objected. After discharge she was returned by the father to the mother in New Orleans. Our only follow-up is that she was admitted to Charity Hospital in May, 1975, for subcutaneous e m p h y s e m a of the left arm. DISCUSSION An extensive review of the literature revealed three similar cases. One was a 28-year-old American housewife who had three hospital admissions before she admitted that the soft tissue air was self-induced? A 36-year-old Czech male used a cutting tool to instill air into the subcutaneous tissue of the scrotum and subsequently sutured the wound with a sewing needle and thread. -~A 16-year-old Greek adolescent girl was reported who had unexplained air in the right thigh; self-induction was considered? N o n e of these patients as well as the present one were drug abusers. Subcutaneous e m p h y s e m a of the extremities is u n c o m m o n ; the major cause is gas gangrene associated with severe trauma. In rare cases subcutaneous emphysema m a y develop after tears of the gastrointestinal, genitourinary, and respiratory systems and the escape of gas-tbrming organisms into subcutaneous tissues. If its etiology is not readily apparent, self-induction should be seriously considered. Joseph L. Rauh, M.D. Division of Adolescent Medicine Children's Hosp#al Medical Center Cincinnati, Ohio 45229 REFERENCES 1.
Gershwin ME, Gude JK, and Petrally J: Factitious subcutaneous emphysema, A n n Intern Med 75:585, 1971. Srch M, and Beran J: Sevstbeschadigung durch Verletzung des Hodensackes and subcutanes e m p h y s e m , Zrechtmed 72:73, 1973. Balas P, Oeconomidis M, Tzamouranis D, and Tripolitis A; Spontaneous subcutaneous e m p h y s e m a , A m J Surg 127:755, 1974.
"'Hypothermia'" in a cool mist tent To the Editor: A recent admission to the Ventura County Hospital called our attention to an unexpected and potentially lethal complication o f the "cool mist tent." CASE REPORT A one-month-old female infant presented in the emergency room with slow, shallow respirations and very cold skin. The child had been seen by a physician one week prior to admission and an upper respiratory infection had been diagnosed. Treatment included instructions for the parents to construct a "cool mist tent" at home. The parents placed a hollow, wooden door on top of the crib, a heavy plastic shower curtain over the sides, and aimed a standard cool mist vaporizer filled with tap water through a small opening. After five days of this treatment the
Letters to the Editor
69 1
infant had cool hands and feet and was feeding poorly. On the day of admission she had slow respirations and felt cold to the touch. Physical examination revealed a temperature o f 22.8 ~ C measured with a rectal probe inserted 6 cm, pulse 70, respirations 15, and a blood pressure of 62 systolic. The skin was cold, mottled, and cyanotic; the baby responded sluggishly to stimulation and had a weak cry. The heart sounds were distant and she had rhinorrhea. The remainder of the examination was normal. Laboratory evaluation included normal chest roentgenogram, complete blood count, urinalysis, electrolytes, and glucose. Arterial blood gases on room air revealed P% 64, Pco~ 53, and p H 7.28. Electrocardiogram demonstrated sinus bradycardia. The infant was placed in an isolette and warmed to 37 ~ C over four hours. Both respiratory and pulse rate returned to normal as temperature increased, and no untoward effects were noted. DISCUSSION Several physiologic changes occur with hypothermia that could explain this apparently well-tolerated low temperature. Metabolically, all biochemical reactions are slowed with hypothermia according to the van't Hoff-Arrhenius law which states that for each 10 degree centigrade decrease in body temperature the rate o f biochemical reactions slows two- to three-fold? This decreased metabolic rate explains at least in part how the infant could tolerate the bradycardia and hypopnea present on admission. Other effects of hypothermia include': (1) Cardiac arrhythmias--most commonly bradycardia and atrial fibrillation, (2) progressive central nervous system depression leading to loss of consciousness and abolition of reflexes, (3) hypoxemia and respiratory acidosis, owing to the decreased respiratory rate and t h e increased solubility of CO2, (4) depression of glucose metabolism which can lead to hyperglycemia, but which m a y be offset by, and (5) decreased re-absorption of glucose and other substances by the kidney, owing to a direct effect on the enzymatic activity in the distal tubules. A recent article by Tafari and Gentz ~ discusses whether a hypothermic infant should be rapidly or slowly rewarmed. The authors studied 30 infants with severe hypothermia and concluded there was no increased risk with rapid rewarming. Our case concurs with their experience. Perhaps the most important point to be m a d e from this case report, though, is to remind us as physicians to be more aware of the potential complications o f our treatment. Even such a standard treatment as the cool mist tent, which is so c o m m o n l y used in hospitalized patients, in this case resulted in near tragedy. The actual temperature in this home-constructed tent with a commercial cool mist vaporizer was 14 ~ C! James ,1. Grady, M.D. Jacquelyn Bamman, M.D. General Hospital 3291 Loma Vista Rd. Ventura, Calif. 93003 REFERENCES 1. Meriwther WD, and G o o d m a n RM: Severe accidental hypothermia with survival after rapid re-warming, A m J Med 53:505, 1972. Tafari N, and Gentz J: Aspects on re-warming newborn infants with severe accidental hypothermia, Acta Pediatr Scand 63:595, 1974.
Letters to the Editor
The Journal of Pediatrics April 1976
REFERENCES
1.
2. 3.
4. 5.
Perry TL, Hansen S, Tischler B, and Bunting R: Determination o f heterozygosity for phenylketonuria on the a m i n o acid analyzer, Clin C h e m Acta 18:51, 1967. Dubowitz V: Intellectual impairment in muscular dystrophy, Arch Dis Child 40:296, 1965. Prosser EJ, M u r p h y EG, and T h o m p s o n M W : Intelligence and the gene for D u c h e n n e muscular dystrophy, Arch Dis Child 44:221, 1969. Rosman NP and K a k u l a r BA: Mental deficiency associated with muscular dystrophy, Brain 89:769, 1966. Rosman NP: The cerebral defect a n d m y o p a t h y in Duchenne muscular dystrophy, Neurology 20:329, 1970.
Self-induced subcutaneous emphysema in an adolescent To the Editor: Subcutaneous e m p h y s e m a of the extremities is a serious problem and is usually related to obvious trauma a n d / o r the presence of anerobic organisms in the soft tissues. A n u n u s u a l case of self-induced subcutaneous e m p h y s e m a o f the extremities is reported in an emotionally disturbed adolescent. CASE REPORT
Patient T. R., a 15-year-old black female, was first admitted to the Children's Hospital Medical Center in October, 1974, with a two-week history of weakness in her legs and episodes o f falling. Three weeks before admission she had moved from New Orleans to live with her father w h o m she had not seen in nine years. The patient appeared tired, weak, and in no acute distress. There were n u m e r o u s small scars on the face and trunk. There was a Grade 2/6 systolic m u r m u r heard best along the left lower sternal border. The neurologic examination revealed occasional but not always consistent falling to the left on t a n d e m walking. She was oriented, right-handed, had normal cranial nerve function, bilaterally absent or hypoactive reflexes in all extremities, and inconsistent responses to vibration and position sense in the legs. Muscle strength was normal. Pertinent laboratory data included normal CBC, urinalysis, erythrocyte sedimentation rate, serum concentrations of electrolytes and serum muscle enzyme values. Examination of cerebrospinal fluid on admission was normal as were serum screen testing for drugs, ECG, chest roentgenogram, and electromyogram. The cardiac m u r m u r was thought to be functional. Additional social history revealed that she had first lived with several relatives in Cincinnati and the rural south and then with the mother who allegedly beat her. She had recently expressed a strong desire to live with her father who had a history of paralytic poliomyelitis which resulted in atrophy of the left arm and leg. The patient's falling and weakness slowly improved during a two-week hospital stay. Physical medicine and psychiatric consultations each suggested a hysterical conversion reaction. She was readmitted one month later with a six-day history of swelling and pain in the left arm. On examination there was crepitus in the left hand, forearm, and arm, almost to the shoulder. There was a small, healing puncture wound on the dorsum o f the left hand. Otherwise the physical a n d laboratory examinations were unre-
Fig. 1. Soft tissue air in the left arm and elbow. markable. Roentgenograms of the left a r m revealed no evidence of fracture or radiopaque foreign body. There was significant air within the soft tissues of the arm in the area of the elbow medially and anteriorly (Fig. 1). The right arm was normal radiologically; a chest roentgenogram now showed s u b c u t a n e o u s air along the left axilla and lateral chest wall. The patient vehemently denied needle use. She acknowledged considerable difficulty with her family a n d peers at school and said several boys had been sticking pins in her. The soft tissue air subsided in four days and she was again discharged. Twice in January, 1975, she was seen in the emergency room with a swollen left arm. In early February she appeared at the Cincinnati General Hospital. Again she had pain, swelling, and crepitus of the left arm and now in the left leg. Several probable needle puncture marks were seen on the d o r s u m of the left h a n d and in the left medial ankle area. She was admitted to the surgical service where serious consideration was given to the possibility of an anaerobic infection. White blood count was normal. Repeated blood cultures for aerobic and anaerobic organisms were negative. Roentgenograms again showed air in the soft tissues o f the forearm to the elbow and in the leg from ankle to mid-thigh. The patient again insisted she had not inflicted needle marks on herself nor injected air. An open surgical biopsy of the soft tissue of the left mid-thigh was performed which was normal and from which no organisms grew. The attending physicians as well as the family were now convinced that the problem was self-induced. A 12 ml syringe and needle, identical to those used at Children's Hospital Medical Center, was found in her room at home. Psychologic testing revealed a full scale IQ of 61. The psychologist felt she was very depressed. She was a person of at least borderline intelligence whose functioning was impaired by emotional problems. The patient was hospitalized for six weeks a n d a behavior
Volume 88 Number 4, part 1
modification program begun. Residential placement in an adolescent group home was r e c o m m e n d e d but the parents objected. After discharge she was returned by the father to the mother in New Orleans. Our only follow-up is that she was admitted to Charity Hospital in May, 1975, for subcutaneous e m p h y s e m a of the left arm. DISCUSSION An extensive review of the literature revealed three similar cases. One was a 28-year-old American housewife who had three hospital admissions before she admitted that the soft tissue air was self-induced? A 36-year-old Czech male used a cutting tool to instill air into the subcutaneous tissue of the scrotum and subsequently sutured the wound with a sewing needle and thread. -~A 16-year-old Greek adolescent girl was reported who had unexplained air in the right thigh; self-induction was considered? N o n e of these patients as well as the present one were drug abusers. Subcutaneous e m p h y s e m a of the extremities is u n c o m m o n ; the major cause is gas gangrene associated with severe trauma. In rare cases subcutaneous emphysema m a y develop after tears of the gastrointestinal, genitourinary, and respiratory systems and the escape of gas-tbrming organisms into subcutaneous tissues. If its etiology is not readily apparent, self-induction should be seriously considered. Joseph L. Rauh, M.D. Division of Adolescent Medicine Children's Hosp#al Medical Center Cincinnati, Ohio 45229 REFERENCES 1.
Gershwin ME, Gude JK, and Petrally J: Factitious subcutaneous emphysema, A n n Intern Med 75:585, 1971. Srch M, and Beran J: Sevstbeschadigung durch Verletzung des Hodensackes and subcutanes e m p h y s e m , Zrechtmed 72:73, 1973. Balas P, Oeconomidis M, Tzamouranis D, and Tripolitis A; Spontaneous subcutaneous e m p h y s e m a , A m J Surg 127:755, 1974.
"'Hypothermia'" in a cool mist tent To the Editor: A recent admission to the Ventura County Hospital called our attention to an unexpected and potentially lethal complication o f the "cool mist tent." CASE REPORT A one-month-old female infant presented in the emergency room with slow, shallow respirations and very cold skin. The child had been seen by a physician one week prior to admission and an upper respiratory infection had been diagnosed. Treatment included instructions for the parents to construct a "cool mist tent" at home. The parents placed a hollow, wooden door on top of the crib, a heavy plastic shower curtain over the sides, and aimed a standard cool mist vaporizer filled with tap water through a small opening. After five days of this treatment the
Letters to the Editor
69 1
infant had cool hands and feet and was feeding poorly. On the day of admission she had slow respirations and felt cold to the touch. Physical examination revealed a temperature o f 22.8 ~ C measured with a rectal probe inserted 6 cm, pulse 70, respirations 15, and a blood pressure of 62 systolic. The skin was cold, mottled, and cyanotic; the baby responded sluggishly to stimulation and had a weak cry. The heart sounds were distant and she had rhinorrhea. The remainder of the examination was normal. Laboratory evaluation included normal chest roentgenogram, complete blood count, urinalysis, electrolytes, and glucose. Arterial blood gases on room air revealed P% 64, Pco~ 53, and p H 7.28. Electrocardiogram demonstrated sinus bradycardia. The infant was placed in an isolette and warmed to 37 ~ C over four hours. Both respiratory and pulse rate returned to normal as temperature increased, and no untoward effects were noted. DISCUSSION Several physiologic changes occur with hypothermia that could explain this apparently well-tolerated low temperature. Metabolically, all biochemical reactions are slowed with hypothermia according to the van't Hoff-Arrhenius law which states that for each 10 degree centigrade decrease in body temperature the rate o f biochemical reactions slows two- to three-fold? This decreased metabolic rate explains at least in part how the infant could tolerate the bradycardia and hypopnea present on admission. Other effects of hypothermia include': (1) Cardiac arrhythmias--most commonly bradycardia and atrial fibrillation, (2) progressive central nervous system depression leading to loss of consciousness and abolition of reflexes, (3) hypoxemia and respiratory acidosis, owing to the decreased respiratory rate and t h e increased solubility of CO2, (4) depression of glucose metabolism which can lead to hyperglycemia, but which m a y be offset by, and (5) decreased re-absorption of glucose and other substances by the kidney, owing to a direct effect on the enzymatic activity in the distal tubules. A recent article by Tafari and Gentz ~ discusses whether a hypothermic infant should be rapidly or slowly rewarmed. The authors studied 30 infants with severe hypothermia and concluded there was no increased risk with rapid rewarming. Our case concurs with their experience. Perhaps the most important point to be m a d e from this case report, though, is to remind us as physicians to be more aware of the potential complications o f our treatment. Even such a standard treatment as the cool mist tent, which is so c o m m o n l y used in hospitalized patients, in this case resulted in near tragedy. The actual temperature in this home-constructed tent with a commercial cool mist vaporizer was 14 ~ C! James ,1. Grady, M.D. Jacquelyn Bamman, M.D. General Hospital 3291 Loma Vista Rd. Ventura, Calif. 93003 REFERENCES 1. Meriwther WD, and G o o d m a n RM: Severe accidental hypothermia with survival after rapid re-warming, A m J Med 53:505, 1972. Tafari N, and Gentz J: Aspects on re-warming newborn infants with severe accidental hypothermia, Acta Pediatr Scand 63:595, 1974.
