Saturday, March 23, 2013

Adrenocortical Insufficiency


                     

Adrenocortical Insufficiency

Deficient production of cortisol or aldosterone result from
lesions of the hypothalamus,
pituitary gland,
adrenal cortex
symptoms may be
severe
mild,
appear
abruptly
insidiously,
Onset -
infancy
later
May be permanent or temporary.

Corticotropin Deficiency. (ACTH)

hypoplasia or aplasia of the pituitary
These congenital defects are associated with abnormalities of the skull and brain
anencephaly and holoprosencephaly.
adrenals are small with normal structure,
autosomal recessive inheritance
craniopharyngioma, is the most common causes of ACTH deficiency

Adrenal Hypoplasia Congenita.

onset of hypo-adrenalism usually begins
in the neonatal period
may be delayed until 10 yr of age
presenting manifestations
Increasing pigmentation
salt-losing symptoms
low levels of all adrenal steroids

Affects - boys
caused by a mutation of the DAX-1 gene, - located on Xp21.
boys with adrenal hypoplasia congenita (AHC) do not undergo puberty owing to hypogonadotropic hypogonadism (HHG),
both AHC and HHG are caused by the same mutated DAX-1 gene. Cryptorchidism, is seen in these boys, is – a manifestation of HHG.

AHC also occurs in
Duchenne muscular dystrophy
glycerol kinase deficiency
mental retardation
Prenatal diagnosis of AHC is possible.


Inborn Defects of Steroidogenesis.

The most common causes of adrenocortical insufficiency in infancy are the salt-losing forms of congenital adrenal hyperplasia 
21-hydroxylase defect
lipoid adrenal hyperplasia
deficiency of 3b-hydroxysteroid dehydrogenase manifest salt-losing symptoms in the newborn period.
Here there is a deficiency in the synthesis of both cortisol and aldosterone
There are elevated levels of steroids that are formed prior to the enzymatic defect.


Hemorrhage into Adrenal Glands.

1.         Occur in the neonatal period because of difficult labor or asphyxia.
Scrotal hematoma may be the presenting sign.
Identified by calcification of the adrenal gland

2.         Waterhouse-Frederickson syndrome - shock resulting from meningococcemia

3.         Adrenal hemorrhage may also occur because of child abuse.

Abrupt Cessation of Administration of Corticotropin or a Corticosteroid.

Likely to occur if steroid is been given in large doses for a long time to patients( asthma, Nephrotic syndrome) who are subjected to stressful situations such as severe infections or surgical procedures.

CLINICAL MANIFESTATIONS.

At birth -
Symptoms characteristic of salt loss-
-         Failure to thrive
-         Vomiting
-         Lethargy
-         anorexia
-         dehydration
-         Circulatory collapse may be fatal.

In older children with Addison disease,
onset is more gradual
-         muscular weakness
-         lassitude
-         anorexia
-         loss of weight
-         general wasting
-         low blood pressure
-         Abdominal pain may simulate an acute surgical condition,
-         an intense craving for salt.
-         adrenal crisis – may be fatal - suddenly becomes cyanotic, the skin is cold, and the pulse is weak and. blood pressure falls, and respirations rapid and labored.
-         crises is precipitated by infection, trauma, excessive fatigue, or drugs such as barbiturates, laxatives, thyroid hormone, or insulin.

Increased pigmentation of the skin should make you think of possibility of adrenocortical insufficiency.
This manifestation occurs in due to excessive secretion of ACTH
-         primary adrenal hypoplasia
-         adrenoleukodystrophy
-         Addison disease.

Pigmentation first on the face and hands
Most intense around the genitals, umbilicus, axillae, nipples, and joints
Scars may be especially pigmented
Areas of depigmentation (vitiligo) may be interspersed with dark areas.
The exposed areas of the skin are the most intensely affected
failure of a suntan to disappear may be the first clue to the condition.
buccal mucosa -pigmentation is bluish brown.
Other features-
hypoglycemia, in the neonate with congenital adrenal hypoplasia.

Pigmentation does not occur in patients with a deficiency of ACTH.
Hypoglycemia is the usual presenting manifestation.
Hyperkalemia does not occur because of preserved aldosterone secretion, but hyponatremia may be present.

LABORATORY FINDINGS.

  1. serum sodium and chloride are low
  2. levels of potassium elevated
  3. increased plasma Renin activity
  4. Urinary excretion of sodium and chloride is increased
  5. urinary potassium is decreased
  6. pH & ABG - acidosis.
  7. Plasma Urea is elevated if dehydration is present.
  8. Hypoglycemia
  9. Blood eosinophils - increased in number.
  10. X ray of the abdomen - calcifications in the area of the adrenals – in  hemorrhage, adrenal cysts, or tuberculosis
  11. Ultrasonography of the abdomen                                   ]
  12. computed tomography of the abdomen              ] --       to show adrenals
  13. magnetic resonance imaging of the abdomen                  ]
  14. X ray Chest – Microcardia - A small and narrow shadow of the heart reflects hypovolemia.
  15. Electrocardiography- changes reflect potassium levels
  16. measurement of the plasma or serum levels of cortisol before and after administration of ACTH-            resting levels are low
No increase occurs after administration of ACTH.
17. Measurement of plasma or serum levels of cortisol precursors - in infants in whom congenital adrenal hyperplasia is suspected.

TREATMENT.

Treatment - for adrenal crisis must be immediate and vigorous.
  1. Blood sample should be obtained before therapy for determination of levels of   ACTH,
                cortisol
                aldosterone
                PRA(plasma renin activity)
                17a-hydroxyprogesterone
                adrenal androgens.
  2. Intravenous administration of 5% glucose in 0.9% saline solution - to correct the hypoglycemia and the sodium loss.
  3. water-soluble form of hydrocortisone- should be given intravenously. - large doses can be given safely. - 25 mg for infants and 75 mg for older children -given intravenously at 6-hr intervals for the first 24 hr.  - dose may be reduced during the next 24 hr
  4. After the first 48 hr, if oral intake is satisfactory, intravenous fluids may be discontinued
  5. corticosteroid given orally as cortisol in doses of 5–20 mg at 8-hr intervals.
  6. Florinef (9a-fluorohydrocortisone), a mineralocorticoid, added orally at 0.05–0.3 mg daily.

chronic replacement therapy for aldosterone and cortisol deficiencies.
cortisol given orally in daily doses of 5–10 mg/24 hr in two or three divided doses
Fluorhydrocortisone is continued orally in doses of 0.05–0.3 mg daily.
During situations of stress, such as infection or operative procedures, the dose of hydrocortisone should be increased.

Overdosage with fluorhydrocortisone results in
hypertension
cardiac enlargement
edema
because of excessive retention of sodium chloride and water
Excessive loss of potassium may produce weakness or paralysis

elevated levels of very long chain fatty acids are diagnostic of adrenoleukodystrophy. antiadrenal antibodies suggests an autoimmune pathogenesis;
Infants with congenital adrenal hypoplasia should undergo chromosomal analysis to search for a mutation or deletion of the Xp21 region
elevated levels of creatine phosphokinase indicate an association with Duchenne muscular dystrophy
elevated levels of triglycerides suggest glycerol kinase deficiency.

                       

                       

Deficiency of 21-hydroxylase
accounts for 90% of affected patients.

This P450 enzyme (P450c21) hydroxylates progesterone and 17-hydroxyprogesterone (17-OHP)        to yield
                        11-deoxycorticosterone (DOC) and 11-deoxycortisol

There steroid 21-hydroxylase genes are on the short arm of chromosome 6

classic disorder occurs in salt-wasting and simple virilizing forms.

Newborn screening programs
            Uses -capillary heel blood on filter paper disks
75% of affected infants have the salt-losing, virilizing form
25% have the simple virilizing form

CLINICAL MANIFESTATIONS.

The clinical manifestations in CAH depend on
            which hormones are deficient
            and which are overproduced
Most patients with CAH have the defect in 21-hydroxylation
            75% of infants are salt losers,

Non-Salt–Losing Congenital Adrenal Hyperplasia.

In the male with 21-hydroxylase deficiency,
            clinical manifestations are of premature isosexual development.
The infant isnormal at birth,
-         signs of sexual and somatic precocity may appear within the first 6 mo of life
-         evident at 4–5 yr of age or later.
-         Enlargement of the penis, scrotum, and prostate
-         appearance of pubic hair
-         development of acne and a deep voice
-         Muscles are well developed
-         bone age is advanced for chronological age.
-         premature closure of the epiphyses causes growth to stop early
-         testes appear small in contrast to the enlarged penis
-         Mental development is normal
-         abnormal physical development may result in behavioral problems.

In the female, CAH due to 21-hydroxylase deficiency results in
-         female pseudohermaphroditism
-         there is almost always evidence of masculinization at birth.
-         enlargement of the clitoris and varying degrees of labial fusion.
-         vagina usually has a common opening with the urethra (urogenital sinus).
-         The clitoris may be so enlarged that it resembles a penis,
-         because the urethra opens below this organ, a mistaken diagnosis of hypospadias and cryptorchidism is sometimes made.
-         complete labial fusion, a phallic urethra, and an external meatus at the tip of the penis.
-         internal genital organs are of a normal female
-         Salt losing may or may not be seen

After birth
-         Pubic and axillary hair develop prematurely
-         acne appears
-         voice assumes a masculine quality.
-         tall for their age
-         ossification is advanced;
-          body build of a boy
-         the internal genitals are female
-         breast development and menstruation do not occur
-         erroneously reared as males
LABORATORY FINDINGS

salt-losing disease
-         low serum concentrations of sodium and chloride
-         elevated levels of potassium and blood urea nitrogen.
-         Plasma levels of renin are elevated
-         serum aldosterone is inappropriately low for the renin level.
-         In classic 21-hydroxylase deficiency, serum levels of 17-OHP are elevated
-         Blood levels of cortisol are usually low in patients with the salt-losing type of disease.
-         Levels of urinary 17-ketosteroids and pregnanetriol are elevated
DIAGNOSIS.

-         adrenal tumor may be palpable
-         may be seen on IVP by displacement of the adjacent kidney.
-         Ultrasonography, computed tomography (CT), or magentic resonance imaging (MRI) may be necessary if hormonal studies have ruled out CAH.
-         Urinary 17-ketosteroid excretion and plasma levels of dehydroepiandrosterone sulfate (DHEAS) are elevated with CAH and cortical tumors,
-         very high values favor the diagnosis of neoplasm.
Administration of hydrocortisone quickly reduces elevated steroid levels to normal in patients with CAH but does not do so in those with a virilizing tumor.
By inhibiting secretion of corticotropin, corticosteroids reduce the stimulation of the adrenals in patients with CAH, whereas adrenocortical tumors are not subject to pituitary regulation.

In males with CAH,
-         testes are small for the degree of virilization,
-         in true precocious puberty or with Leydig cell tumors, the testes are enlarged for age.

Females with CAH -adrenal cortical steroid levels elevated.

Molecular techniques can now be used for genetic counseling for all forms of CAH.

Prenatal Diagnosis and Treatment.

Prenatal diagnosis of 21-hydroxylase

in 1st trimester
            DNA analysis and HLA genotyping of chorionic villus cells.

In 2nd trimester
            1.         measuring 17-OHP and androstenedione in amniotic fluid
            2.         HLA typing and DNA analysis of amniotic fluid cells.

Prenatal treatment
-         maternal dexamethasone administration
-         dexamethasone, readily crosses the placenta,
-         First-trimester chorionic villus biopsy is performed to determine the sex and genotype of the fetus
-         therapy is continued only if the fetus is a female.
-         Maternal side effects - edema, excessive weight gain, hypertension, glucose intolerance, cushingoid facial features, and severe striae with permanent scarring.
DNA analysis of chorionic villus cells can be used for the prenatal diagnosis of all forms of CAH.

TREATMENT.

glucocorticoids inhibits excessive production of androgens
prevents progressive virilization.
-         hydrocortisone (10–20 mg/m2 /24 hr) orally in two or three divided doses
-         Infants require 2.5–5 mg two to three times daily
-         children 5–10 mg two to three times daily.
-         Doses must be individualized by monitoring growth and hormonal levels.
-         salt-losing disease- require a mineralocorticoid and sodium supplementation in addition to the glucocorticoid. - therapy with 9a-fluorohydrocortisone (0.05–0.3 mg daily) and sodium chloride, 1–3 g
-         non–salt-losing disease may also manifest elevated plasma renin activity and require a mineralocorticoid.

Serum levels of 17-OHP, androstenedione, testosterone, and renin,
            measured at 8–9 A.M
            prior to taking the morning medication
            monitor for signs of
                        cortisol or androgen excess
                        growth and weight gain
                        pubertal development
                        osseous maturation
administration of hydrocortisone must be continued indefinitely
Increased doses are indicated during periods of stress such as infection or surgery because they have defective adrenal reserve.

The enlarged clitoris of female infants - requires surgical correction
Timing is 6–12 mo.
Vaginoplasty and correction of the urogenital sinus also done at the time of clitoral surgery
complete sexual gratification, including orgasm, can be achieved.
menarche occurs at the appropriate age
delay is related to suboptimal dose.

sex assignment of infants with intersex - -easy to make a female.
                       

Addison Disease.

Destruction of the adrenal cortex during childhood
            Tuberculosis
            Autoimmune destruction
Usually the medulla is not destroyed
All adrenal cortical function is lost
Antiadrenal antibodies seen in plasma
Addison disease as a component of two syndromes,
            inherited as an autosomal recessive disorder,
            gene  in chromosome 21q22.3
Type I autoimmune polyendocrinopathy - autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy.
            Chronic mucocutaneous candidiasis is the first manifestation,
            Hypoparathyroidism
            Addison disease
Other features -
            gonadal failure
            alopecia
            vitiligo
            keratopathy
            enamel hypoplasia
            nail dystrophy
            intestinal malabsorption
            chronic active hepatitis
            Hypothyroidism
            type I diabetes mellitus
                       

Type II autoimmune polyendocrinopathy
            Addison disease
            autoimmune thyroid disease or insulin-dependent diabetes.
            Gonadal failure
            Vitiligo
            alopecia
            chronic atrophic gastritis
            pernicious anemia
            HLA-D3 and HLA-D4 predominate in these patients
            common in females
            Antiadrenal antibodies, steroid cell antibodies, and antibodies to 21-OH, 17a-OH, and P450 scc enzymes are also found in these patients.
                       


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