International Journal of Pediatric Otorhinolaryngology, @ ElsevierlNorth-Holland Biomedical Press
A RARE COMPLICATION
D. PETROVIC,
FOLLOWING
B. PETROVIC-MINIC
Railroad Hospital; ENT Department, Physiology, Belgrade (Yugoslavia)
1 (1979)
117-123
FRONTOETHMOID
117
INJURY
and V. BOROVAC Medical Faculty
and Institute
for Pathological
(Originally presented at the First World Congress of Pediatric Otorhinolaryngology, Sirmione, Italy on April 26-30th, 1977)
in
The oldest records on craniocerebral trauma come from Egypt, dating back to 3000 B.C. Part of a hand-written copy, probably written about 1700 B.C., of a hieroglyphic treatise from that period has been preserved as the Edwin Smith papyrus [ 71. Although head injuries are so common and probably represent the only type of brain damage recognized for centuries, our understanding of their pathology is still very incomplete. Injuries where the frontal bone and the base of the skull are involved are those head injuries where special concern is needed. These injuries are considered as a task for the otorhinolaryngologist to fulfill [ 51. When the head is struck, the brain with its appendages in its semirigid, uneven and partially divided housing is subject to varying degrees of acceleration, deceleration, rotation, compression, expansion and swirling motions about its attachments. When the dura is penetrated the brain structure may be ruptured by bone fragments driven in by the agent of injury. Following the impact, secondary physical and biochemical changes, local or general, can extend the brain damage [l]. Known complications following injuries of the frontobasal region have been the subject of numerous papers in the literature and have their place in the standard textbooks of otolaryngology; therefore we omit these as being well known matter. Presenting a case of a 16-year-old boy who sustained severe head injury followed by the appearance of transitory secondary hyperaldosteronism more than 10 years ago, we attempt to add one, though rare, complication to the list of already published ones, that follow injury to the head. The syndrome of primary hyperaldosteronism described by Conn in 1955 [2] consists of arterial hypertension and polyuria resistant to antidiuretic hormone, as well as muscular weakness followed by a low level of blood potassium and alkalosis. This syndrome is usually caused by an increased level of aldosterone, most frequently as a consequence of a benign tumor named aldosteronoma or aldosterone’s tumor. Increased, or to some extent mismatched,
118
production of aldosterone has been recorded in some forms of general edema in periods of salt-restricted diet or hypovolemic shock. Secondary hyperaldosteronism has the same clinical characteristics, as well as a disturbance in the electrolyte balance being a basic cause of the disorder. There is a lack of evidence of secondary hyperaldosteronism following head injuries in the literature. The nervous system and the endocrine system form a complementary “wire and wireless” communications network regulating a broad array of metabolic processes. Normally, signals from higher centers impinge on the hypothalamus, thereby modulating the synthesis and secretion of neurohumoral substances which either regulate the release of anterior pituitary hormones or pass directly to the posterior pituitary. Once these hormones are released into the circulation they in turn stimulate the secretion of hormones by specific target glands or act directly on other tissues of the body [4]. It has been fairly well established that the diencephalon controls many functions of the pituitary gland. Concerning the hypothalamo-pituitaryadrenocortical axis it appears that this action is mediated through a hypothalamic neurohumor which represents the final direct stimulus for the release of ACTH from the adenohypophysis. This action is also controlled by the level of the target hormone. The most sensitive area for a corticoid blocking action appears to be the anterior part of the median eminence and the postoptic area, but the region between the median eminence and the supraoptic and paraventricular nuclei is involved as well. It is conceivable that corticosteroids suppress the elaboration or release of such a factor. It is true that there is still an open question whether this is a hi&chemical interference or an inhibition of nervous impulse transmission of afferents of the above-mentioned cells or a blockade of the impulses providing the signal for the release of the factor from the store in the infundibular process. Although a relationship with the neurosecretory material is strongly suggested by the spatial arrangement of the effective implants, it is quite possible that corticoids act in another way [ 61. This is a well established and sufficiently proven pattern of action which might be employed in explanation of the pathological physiology of the transitory registered secondary hyperaldosteronism we were to deal with. The main problem was to explain regulation of adrenal cortex production, because there was an evident lack of influence of pituitary hormones in the chain of action. Recently published studies on the regulation of cellular and extracellular thirst seem to provide a possible key for understanding this complex mechanism. The neural system subserving cellular and extracellular thirst is preserved within the brain in distinct pathways through the lateral hypothalamus. One pathway projects caudally from the preoptic region through the mid-lateral hypothalamus, whereas water intake to cellular thirst stimuli is mediated through pathways passing through the far-lateral hypothalamus.
119
Fig. 1.
Fig. 2
120
These findings suggest that the deficit in drinking to both types of homeostatic thirst signals observed after large bilateral lesions of the lateral hypothalamus may be due to destruction of both the far- and the mid-lateral pathways [ 31. We feel that the above-mentioned mechanism of regulation and pattern of action could be employed in explanation of such a rare complication following frontoethmoid injury. A 16-year-old country boy D.M. (4447/921) was admitted as an emergency case on 14th July, 1966 after being struck by hind hooves. First aid was given at the site of injury and, as an emergency, he was admitted only a few hours after the injury. On admittance a painful bruise on the forehead region was recorded with no evidence of injury on other parts of the head. The upper right eyelid was swollen, closing the eye. In the medial two-thirds of the right eyebrow there was a lacerated wound going across the nasal root. Exploration of the wound gave evidence of an open anterior ethmoidal labyrinth. Beyond the head of the left eyebrow there was a lacerated wound exposing the anterior frontal sinus wall at its bottom. This is a more or less classical description of soft tissue changes with obligatory hematoma underneath showing the site of impact as well as the dimensions and shape of the blow (Figs. 1 and 2). Operative findings were severe: numerous fractures of bony structures were found. The anterior walls of both frontal sinuses were multiply fractured and impressed as well as frontal parts of both upper jaws. There was evidence of a multiple fracture line in the anterior ethmoid labyrinth as well as fracture lines on the posterior walls of the frontal sinuses with impressions of bony fragments toward the anterior fossa. Massive hematomas covering the anterior parts of the dura were noted. The surgeon performed standard procedures and a few days later the operative procedure was repeated, with neurosurgical assistance providing therapeutic measures concerning the dura (sawing) and identifying changes of malatia on anterior poles. After surgery there was an uneventful course of recovery lasting 11 days. Then we noticed some disturbances in thirst regulation. The boy was drinking an enormous quantity of water (up to 4 liters). He was gaining weight and generalized edema, most prominent in the penoscrotal region, was noticed. A consultant thought of secondary diabetes insipidus (published as a complication of the craniocerebral trauma, although rare). Blood potassium level at that time was within normal limits, as was pH. Since there was no response to energetic therapy, the endocrinologists suggested transfer to the Endocrinological Clinic, where he was admitted on the 10th of August. On admittance generalized edema, particularly developed at the lower parts and penoscrotal region, was present. Blood pressure was recorded as 120/75 mm Hg compared with the mean value of 80/50 mm Hg registered previously. The endocrinologist suspected the possibility that a case of posttraumatic secondary hyperaldosteronism was in question, and directed further investigations into this direction. Blood potassium level showed interesting fluctuations, diminishing every day to its lowest level at 2.49 mEq (11th
121 Blood
mEq
level
of pOtaSSlum
Post traumatic
days
Fig. 3.
M Sodium x- - - +K Chloride
mEq 150 140 130 I*
’
=
-
=
=
=
=
l
110 J-__-x
100
90
:
-x’--
x--_~----x--__r_--__-~---
.’
2’
70 80 1
10 t
17
19
21
23
25 Post
27
29
traumatic
31
33
35
days
Fig. 4.
ml/24 4000
-
3000
-
2000
-
h
Urine
1
,6
Fig. 5.
20
I
22
I
I
i
I
,
I
1
24
26
26
30
32
34
36
post traumatic
days
122
August, 1966) with a subsequent raising of level to 4.56 mEq (16th August, 1976) (Fig. 3). Such a potassium balance had, as its natural consequence, the following: (1) on the ECG taken on 13th August, 1966 the cardiologist found micro R wave in Dl-D3 as well as in V4-V6, and slightly negative in Vl-V2, thus leading to the conclusion of a myocardial lesion; (2) registered blood pH taken during this period was 7.50, suggesting metabolic alkalosis (Figs. 4 and 5). During endocrinological treatment the patient lost weight, edema disappeared and after correction of the metabolic disorder there was evidence of stormy neurological disturbances occurring three weeks after the injury, with striking signs pointing to basal arachnoiditis, known as a frequent complication after craniocerebral injury. Upon being cured the patient left hospital and we arranged long-term follow-up. The recovery period was rather fast, with no disturbances, thus allowing him a normal way of living. We saw him some ten and half years after being injured and he was free of any disorders, with all laboratory and clinical findings being within normal limits. When analyzing the chain of causes and disturbances we found that there was only one possibility, since there were no other etiological factors recorded previously, at the moment of injury or afterwards. All the parameters taken pointed to the occurrence of transitory secondary hyperaldosteronism following severe impact of the forehead. The pattern of action as well as specified spatial determination and reaction of the far- and mid-lateral hypothalamic structures which were doubtless involved when the head was struck led, through a rather complicated mechanism of action, to an extremely rare complication following craniocerebral trauma. We feel that the order of action was: disturbance of thirst signal production which might have led to oligemia, generalized edema and, finally, to transitory secondary hyperaldosteronism all having as a principal cause sustained craniocerebral trauma. The clinical course also showed metabolic disturbances, and long-term follow-up allowed the conclusion of such an etiology which confirms by itself the opinion that one of the important insights gained in the neurobiological sciences during the past decade is recognition of the high degree of functional interdependence between the nervous end endocrine centers. REFERENCES Caveness, F.W., Posttraumatic sequelae. In P. Vinken and G.W. Bruyn (Eds.), Handbook of Clinical Neurology, Vol. 23, North-Holland, Amsterdam, 1975, pp. 209-219. Cecil, L. and Loeb, F.R., A Textbook of Medicine, 1959,1960,758 pp. Kucharczyk, J. and Mogensen, G., Exp. Neurol., 53 (1976) 3’71-376. The Neurosciences, Second Study Program, Rockefeller University Press, New York, 1970, pp. 519-529.
123 5 Scott-Brown, Disease of the Ear, Nose and Throat, Vol. 1, Butterworths, London, 1965, pp. 104-121. 6 Smelik, P.G. and Sawyer, C.H., Hypothalamic structures involved in the adrenocortical feedback action on pituitary corticotrophin. In W. Bargmann and J.P. Schade (Eds.), Lectures on the Diencephalon, Progress in Brain Res., Vol. 5, Elsevier, Amsterdam, 1964, pp. 132-135. 7 Verjaal, A., and van? Hooft, F., Commotio and Contusio Cerebri. In P. Vinken and G.W. Bruyn (Eds.), Handbook of Clinical Neurology, Vol. 23, North-Holland, Amsterdam, 1975, pp. 417-444.
A RARE COMPLICATION
D. PETROVIC,
FOLLOWING
B. PETROVIC-MINIC
Railroad Hospital; ENT Department, Physiology, Belgrade (Yugoslavia)
1 (1979)
117-123
FRONTOETHMOID
117
INJURY
and V. BOROVAC Medical Faculty
and Institute
for Pathological
(Originally presented at the First World Congress of Pediatric Otorhinolaryngology, Sirmione, Italy on April 26-30th, 1977)
in
The oldest records on craniocerebral trauma come from Egypt, dating back to 3000 B.C. Part of a hand-written copy, probably written about 1700 B.C., of a hieroglyphic treatise from that period has been preserved as the Edwin Smith papyrus [ 71. Although head injuries are so common and probably represent the only type of brain damage recognized for centuries, our understanding of their pathology is still very incomplete. Injuries where the frontal bone and the base of the skull are involved are those head injuries where special concern is needed. These injuries are considered as a task for the otorhinolaryngologist to fulfill [ 51. When the head is struck, the brain with its appendages in its semirigid, uneven and partially divided housing is subject to varying degrees of acceleration, deceleration, rotation, compression, expansion and swirling motions about its attachments. When the dura is penetrated the brain structure may be ruptured by bone fragments driven in by the agent of injury. Following the impact, secondary physical and biochemical changes, local or general, can extend the brain damage [l]. Known complications following injuries of the frontobasal region have been the subject of numerous papers in the literature and have their place in the standard textbooks of otolaryngology; therefore we omit these as being well known matter. Presenting a case of a 16-year-old boy who sustained severe head injury followed by the appearance of transitory secondary hyperaldosteronism more than 10 years ago, we attempt to add one, though rare, complication to the list of already published ones, that follow injury to the head. The syndrome of primary hyperaldosteronism described by Conn in 1955 [2] consists of arterial hypertension and polyuria resistant to antidiuretic hormone, as well as muscular weakness followed by a low level of blood potassium and alkalosis. This syndrome is usually caused by an increased level of aldosterone, most frequently as a consequence of a benign tumor named aldosteronoma or aldosterone’s tumor. Increased, or to some extent mismatched,
118
production of aldosterone has been recorded in some forms of general edema in periods of salt-restricted diet or hypovolemic shock. Secondary hyperaldosteronism has the same clinical characteristics, as well as a disturbance in the electrolyte balance being a basic cause of the disorder. There is a lack of evidence of secondary hyperaldosteronism following head injuries in the literature. The nervous system and the endocrine system form a complementary “wire and wireless” communications network regulating a broad array of metabolic processes. Normally, signals from higher centers impinge on the hypothalamus, thereby modulating the synthesis and secretion of neurohumoral substances which either regulate the release of anterior pituitary hormones or pass directly to the posterior pituitary. Once these hormones are released into the circulation they in turn stimulate the secretion of hormones by specific target glands or act directly on other tissues of the body [4]. It has been fairly well established that the diencephalon controls many functions of the pituitary gland. Concerning the hypothalamo-pituitaryadrenocortical axis it appears that this action is mediated through a hypothalamic neurohumor which represents the final direct stimulus for the release of ACTH from the adenohypophysis. This action is also controlled by the level of the target hormone. The most sensitive area for a corticoid blocking action appears to be the anterior part of the median eminence and the postoptic area, but the region between the median eminence and the supraoptic and paraventricular nuclei is involved as well. It is conceivable that corticosteroids suppress the elaboration or release of such a factor. It is true that there is still an open question whether this is a hi&chemical interference or an inhibition of nervous impulse transmission of afferents of the above-mentioned cells or a blockade of the impulses providing the signal for the release of the factor from the store in the infundibular process. Although a relationship with the neurosecretory material is strongly suggested by the spatial arrangement of the effective implants, it is quite possible that corticoids act in another way [ 61. This is a well established and sufficiently proven pattern of action which might be employed in explanation of the pathological physiology of the transitory registered secondary hyperaldosteronism we were to deal with. The main problem was to explain regulation of adrenal cortex production, because there was an evident lack of influence of pituitary hormones in the chain of action. Recently published studies on the regulation of cellular and extracellular thirst seem to provide a possible key for understanding this complex mechanism. The neural system subserving cellular and extracellular thirst is preserved within the brain in distinct pathways through the lateral hypothalamus. One pathway projects caudally from the preoptic region through the mid-lateral hypothalamus, whereas water intake to cellular thirst stimuli is mediated through pathways passing through the far-lateral hypothalamus.
119
Fig. 1.
Fig. 2
120
These findings suggest that the deficit in drinking to both types of homeostatic thirst signals observed after large bilateral lesions of the lateral hypothalamus may be due to destruction of both the far- and the mid-lateral pathways [ 31. We feel that the above-mentioned mechanism of regulation and pattern of action could be employed in explanation of such a rare complication following frontoethmoid injury. A 16-year-old country boy D.M. (4447/921) was admitted as an emergency case on 14th July, 1966 after being struck by hind hooves. First aid was given at the site of injury and, as an emergency, he was admitted only a few hours after the injury. On admittance a painful bruise on the forehead region was recorded with no evidence of injury on other parts of the head. The upper right eyelid was swollen, closing the eye. In the medial two-thirds of the right eyebrow there was a lacerated wound going across the nasal root. Exploration of the wound gave evidence of an open anterior ethmoidal labyrinth. Beyond the head of the left eyebrow there was a lacerated wound exposing the anterior frontal sinus wall at its bottom. This is a more or less classical description of soft tissue changes with obligatory hematoma underneath showing the site of impact as well as the dimensions and shape of the blow (Figs. 1 and 2). Operative findings were severe: numerous fractures of bony structures were found. The anterior walls of both frontal sinuses were multiply fractured and impressed as well as frontal parts of both upper jaws. There was evidence of a multiple fracture line in the anterior ethmoid labyrinth as well as fracture lines on the posterior walls of the frontal sinuses with impressions of bony fragments toward the anterior fossa. Massive hematomas covering the anterior parts of the dura were noted. The surgeon performed standard procedures and a few days later the operative procedure was repeated, with neurosurgical assistance providing therapeutic measures concerning the dura (sawing) and identifying changes of malatia on anterior poles. After surgery there was an uneventful course of recovery lasting 11 days. Then we noticed some disturbances in thirst regulation. The boy was drinking an enormous quantity of water (up to 4 liters). He was gaining weight and generalized edema, most prominent in the penoscrotal region, was noticed. A consultant thought of secondary diabetes insipidus (published as a complication of the craniocerebral trauma, although rare). Blood potassium level at that time was within normal limits, as was pH. Since there was no response to energetic therapy, the endocrinologists suggested transfer to the Endocrinological Clinic, where he was admitted on the 10th of August. On admittance generalized edema, particularly developed at the lower parts and penoscrotal region, was present. Blood pressure was recorded as 120/75 mm Hg compared with the mean value of 80/50 mm Hg registered previously. The endocrinologist suspected the possibility that a case of posttraumatic secondary hyperaldosteronism was in question, and directed further investigations into this direction. Blood potassium level showed interesting fluctuations, diminishing every day to its lowest level at 2.49 mEq (11th
121 Blood
mEq
level
of pOtaSSlum
Post traumatic
days
Fig. 3.
M Sodium x- - - +K Chloride
mEq 150 140 130 I*
’
=
-
=
=
=
=
l
110 J-__-x
100
90
:
-x’--
x--_~----x--__r_--__-~---
.’
2’
70 80 1
10 t
17
19
21
23
25 Post
27
29
traumatic
31
33
35
days
Fig. 4.
ml/24 4000
-
3000
-
2000
-
h
Urine
1
,6
Fig. 5.
20
I
22
I
I
i
I
,
I
1
24
26
26
30
32
34
36
post traumatic
days
122
August, 1966) with a subsequent raising of level to 4.56 mEq (16th August, 1976) (Fig. 3). Such a potassium balance had, as its natural consequence, the following: (1) on the ECG taken on 13th August, 1966 the cardiologist found micro R wave in Dl-D3 as well as in V4-V6, and slightly negative in Vl-V2, thus leading to the conclusion of a myocardial lesion; (2) registered blood pH taken during this period was 7.50, suggesting metabolic alkalosis (Figs. 4 and 5). During endocrinological treatment the patient lost weight, edema disappeared and after correction of the metabolic disorder there was evidence of stormy neurological disturbances occurring three weeks after the injury, with striking signs pointing to basal arachnoiditis, known as a frequent complication after craniocerebral injury. Upon being cured the patient left hospital and we arranged long-term follow-up. The recovery period was rather fast, with no disturbances, thus allowing him a normal way of living. We saw him some ten and half years after being injured and he was free of any disorders, with all laboratory and clinical findings being within normal limits. When analyzing the chain of causes and disturbances we found that there was only one possibility, since there were no other etiological factors recorded previously, at the moment of injury or afterwards. All the parameters taken pointed to the occurrence of transitory secondary hyperaldosteronism following severe impact of the forehead. The pattern of action as well as specified spatial determination and reaction of the far- and mid-lateral hypothalamic structures which were doubtless involved when the head was struck led, through a rather complicated mechanism of action, to an extremely rare complication following craniocerebral trauma. We feel that the order of action was: disturbance of thirst signal production which might have led to oligemia, generalized edema and, finally, to transitory secondary hyperaldosteronism all having as a principal cause sustained craniocerebral trauma. The clinical course also showed metabolic disturbances, and long-term follow-up allowed the conclusion of such an etiology which confirms by itself the opinion that one of the important insights gained in the neurobiological sciences during the past decade is recognition of the high degree of functional interdependence between the nervous end endocrine centers. REFERENCES Caveness, F.W., Posttraumatic sequelae. In P. Vinken and G.W. Bruyn (Eds.), Handbook of Clinical Neurology, Vol. 23, North-Holland, Amsterdam, 1975, pp. 209-219. Cecil, L. and Loeb, F.R., A Textbook of Medicine, 1959,1960,758 pp. Kucharczyk, J. and Mogensen, G., Exp. Neurol., 53 (1976) 3’71-376. The Neurosciences, Second Study Program, Rockefeller University Press, New York, 1970, pp. 519-529.
123 5 Scott-Brown, Disease of the Ear, Nose and Throat, Vol. 1, Butterworths, London, 1965, pp. 104-121. 6 Smelik, P.G. and Sawyer, C.H., Hypothalamic structures involved in the adrenocortical feedback action on pituitary corticotrophin. In W. Bargmann and J.P. Schade (Eds.), Lectures on the Diencephalon, Progress in Brain Res., Vol. 5, Elsevier, Amsterdam, 1964, pp. 132-135. 7 Verjaal, A., and van? Hooft, F., Commotio and Contusio Cerebri. In P. Vinken and G.W. Bruyn (Eds.), Handbook of Clinical Neurology, Vol. 23, North-Holland, Amsterdam, 1975, pp. 417-444.
