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Endocrinology
Immunology
Internal Medicine

Autoimmune Polyglandular Syndrome (APS)

High EvidenceUpdated: 2025-12-24

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Red Flags

  • Addisonian crisis (hypotension, hyponatraemia, hyperkalaemia)
  • Hypoglycaemia in T1DM with undiagnosed adrenal insufficiency
  • Severe hypocalcaemia with tetany or seizures
  • Myxoedema coma (severe hypothyroidism)
  • Thyroid storm in undiagnosed Graves' disease
  • Mucocutaneous candidiasis refractory to treatment
Overview

Autoimmune Polyglandular Syndrome (APS)

1. Clinical Overview

Summary

Autoimmune Polyglandular Syndromes (APS) are a group of rare disorders characterised by the coexistence of multiple autoimmune endocrine gland failures, often accompanied by non-endocrine autoimmune manifestations. The syndromes result from immune-mediated destruction of endocrine organs, leading to hormone deficiencies requiring lifelong replacement therapy. APS is categorised into distinct types based on clinical presentation, genetic background, and age of onset. Early recognition and systematic screening are essential to prevent life-threatening complications such as Addisonian crisis, severe hypoglycaemia, or hypocalcaemic seizures.

Key Facts

  • Prevalence: APS-1: 1:100,000; APS-2: 1:20,000 (more common)
  • Genetics: APS-1: Autosomal recessive (AIRE gene); APS-2: Polygenic (HLA-DR3/DR4)
  • Peak onset: APS-1: Childhood (before age 10); APS-2: Adulthood (30-40 years)
  • Sex ratio: APS-1: Equal; APS-2: Female predominance (3:1)
  • Core components: Addison's disease is central to APS-2
  • Screening principle: If one autoimmune endocrinopathy exists, screen for others annually
  • Life-threatening risk: Undiagnosed adrenal insufficiency in patients with T1DM
  • Classic triad (APS-1): Chronic mucocutaneous candidiasis, hypoparathyroidism, Addison's disease
  • Classic triad (APS-2): Addison's disease, autoimmune thyroid disease, Type 1 diabetes
  • Treatment: Hormone replacement for each deficiency

Clinical Pearls

"The Rule of Three": APS-1 presents with the triad of Candidiasis, Hypoparathyroidism, and Addison's — usually appearing in that order during childhood. If you see one, look for the others.

"Schmidt's Syndrome Warning": APS-2 (Schmidt syndrome) classically presents as Addison's + T1DM + Thyroid disease. A patient with T1DM and vitiligo should be screened for adrenal insufficiency and thyroid dysfunction.

"The Dangerous Duo": Undiagnosed adrenal insufficiency in a patient with T1DM can be life-threatening. Cortisol deficiency impairs gluconeogenesis, causing refractory hypoglycaemia, while reducing catecholamine response.

"Annual Screen": Any patient with one autoimmune endocrine condition should have annual TFTs, glucose, and clinical assessment for adrenal insufficiency.

"AIRE Gene = APS-1": Mutations in the AIRE (Autoimmune Regulator) gene cause failure of central tolerance in the thymus, leading to multi-organ autoimmunity in childhood.

Why This Matters Clinically

Autoimmune polyglandular syndromes represent a significant diagnostic challenge due to their variable presentation and potential for life-threatening complications. Missing the diagnosis of adrenal insufficiency in a patient presenting with another endocrinopathy can be fatal. Systematic screening and a high index of suspicion in patients with any autoimmune endocrine disorder can prevent morbidity and mortality.[1,2]


2. Epidemiology

Incidence & Prevalence

SyndromePrevalenceIncidence
APS-1 (APECED)1:100,000 (general population)Higher in Finland (1:25,000), Iran, Sardinia
APS-2 (Schmidt)1-2:100,0001.4-2.0 per 100,000/year
APS-3More common than APS-2Variable, linked to thyroid autoimmunity

Demographics

FactorAPS-1APS-2
Age of onsetChildhood (median 5-7 years)Adulthood (20-60 years, peak 30-40)
Sex ratio1:1Female predominance 3:1
Genetic inheritanceAutosomal recessivePolygenic, HLA-associated
Ethnic clustersFinnish, Iranian Jews, SardiniansNo specific ethnic predominance

Risk Factors

FactorRelative RiskNotes
Family history of autoimmune disease5-10xFirst-degree relatives
HLA-DR3 haplotype3-5xAPS-2
HLA-DR4 haplotype2-3xAPS-2, especially with T1DM
Female sex3xFor APS-2
AIRE gene mutations>5%Diagnostic for APS-1
Existing autoimmune endocrinopathyVariableTriggers screening protocol
Other autoimmune diseases2-3xVitiligo, pernicious anaemia, coeliac disease

3. Pathophysiology

Mechanism

Step 1: Genetic Predisposition

  • APS-1: Mutations in AIRE gene (chromosome 21) cause defective negative selection in thymus
  • APS-2: HLA class II associations (DR3-DQ2, DR4-DQ8) predispose to autoimmunity
  • Polymorphisms in CTLA-4, PTPN22, and other immune regulatory genes contribute
  • Environmental triggers (infections, molecular mimicry) may initiate disease

Step 2: Loss of Immune Tolerance

  • AIRE gene normally promotes expression of tissue-specific antigens in thymic medullary epithelial cells
  • Defective AIRE leads to failure of central tolerance — autoreactive T cells escape thymic deletion
  • In APS-2, peripheral tolerance mechanisms fail with HLA-restricted antigen presentation
  • Regulatory T cell dysfunction contributes to loss of self-tolerance

Step 3: Organ-Specific Autoimmunity

  • Autoreactive T lymphocytes infiltrate target endocrine organs
  • B cells produce organ-specific autoantibodies
  • Cytotoxic CD8+ T cells and cytokines mediate tissue destruction
  • Gradual loss of functional endocrine tissue over months to years

Step 4: Endocrine Gland Failure

  • Progressive destruction leads to hormone deficiency
  • Adrenal cortex: Cortisol and aldosterone deficiency (Addison's disease)
  • Thyroid: Hypo- or hyperthyroidism (Hashimoto's or Graves')
  • Pancreatic beta cells: Insulin deficiency (Type 1 diabetes)
  • Parathyroids: PTH deficiency (hypoparathyroidism)
  • Gonads: Premature ovarian failure or testicular failure

Step 5: Multi-System Manifestations

  • Non-endocrine autoimmune features develop (vitiligo, alopecia, pernicious anaemia)
  • APS-1: Ectodermal dystrophy, hepatitis, keratitis
  • Disease expression varies even within families with same mutations
  • Sequential development of conditions over years to decades

Classification

TypeAlternative NamesCore FeaturesGenetics
APS-1APECED (Autoimmune Polyendocrinopathy-Candidiasis-Ectodermal Dystrophy)Chronic mucocutaneous candidiasis + Hypoparathyroidism + Addison's (2 of 3 required)AIRE gene mutation (autosomal recessive)
APS-2Schmidt syndrome (if Addison's + thyroid)Addison's disease + Autoimmune thyroiditis and/or Type 1 diabetesPolygenic (HLA-DR3, DR4)
APS-3—Autoimmune thyroid disease + Other autoimmune disease (excluding Addison's/hypoparathyroidism)Polygenic
APS-4—Combinations not fitting Types 1-3Variable

Autoantibody Targets

Target OrganAutoantibodyClinical Significance
Adrenal cortex21-hydroxylase (CYP21A2) antibodiesMarker for adrenal autoimmunity (>0% of autoimmune Addison's)
ThyroidTPO antibodies, Thyroglobulin antibodies, TSH receptor antibodiesHashimoto's or Graves' disease
Pancreatic isletsGAD65, IA-2, ZnT8, insulin antibodiesType 1 diabetes
ParathyroidNALP5, CaSR antibodiesHypoparathyroidism
Gastric parietal cellsIntrinsic factor antibodiesPernicious anaemia
GonadsSteroidogenic enzyme antibodiesPremature ovarian failure

4. Clinical Presentation

Symptoms by System

SystemSymptoms
Adrenal (Addison's)Fatigue (100%), weakness, weight loss, salt craving, postural dizziness, nausea, abdominal pain
Thyroid (Hypothyroidism)Fatigue, cold intolerance, weight gain, constipation, dry skin, depression
Thyroid (Hyperthyroidism)Weight loss, heat intolerance, tremor, palpitations, anxiety, diarrhoea
Pancreas (T1DM)Polyuria, polydipsia, weight loss, fatigue
ParathyroidMuscle cramps, paraesthesias, tetany, seizures
Mucocutaneous (APS-1)Oral thrush, nail dystrophy, skin candidiasis

Signs

SystemPhysical Signs
Adrenal insufficiencyHyperpigmentation (buccal mucosa, palmar creases, scars), hypotension, cachexia
HypothyroidismBradycardia, dry skin, delayed relaxation of reflexes, periorbital oedema
HyperthyroidismTachycardia, tremor, lid lag, warm moist skin, goitre, exophthalmos (Graves')
HypoparathyroidismChvostek's sign, Trousseau's sign, tetany
T1DMWeight loss, may have lipodystrophy at injection sites
VitiligoDepigmented patches
AlopeciaPatchy or total hair loss

APS-1 Specific Features

FeatureFrequencyNotes
Chronic mucocutaneous candidiasis75-100%Usually first manifestation (often before age 5)
Hypoparathyroidism80-85%Second component to appear
Addison's disease70-75%Usually develops by age 15
Alopecia30-40%May be areata or totalis
Primary gonadal failure45-60% (females)Premature ovarian insufficiency
Vitiligo10-25%Patchy depigmentation
Autoimmune hepatitis10-15%Can be severe
Enamel hypoplasia70%Ectodermal dystrophy feature
Nail dystrophy50%Ectodermal dystrophy
Keratitis15-25%Can cause visual impairment

APS-2 Specific Features

FeatureFrequencyNotes
Addison's disease100%Defining feature
Autoimmune thyroid disease70%Hashimoto's or Graves'
Type 1 diabetes50%May precede or follow Addison's
Pernicious anaemia5-10%B12 deficiency
Vitiligo5-10%Depigmentation
Primary gonadal failure5-10%Less common than APS-1
Coeliac disease2-5%Associated autoimmunity

Red Flags

[!CAUTION] Red Flags — Seek Urgent Assessment:

  • Hypotension, hyponatraemia, hyperkalaemia — suspect Addisonian crisis
  • Recurrent unexplained hypoglycaemia in T1DM — screen for adrenal insufficiency
  • Seizures or tetany with hypocalcaemia — urgent IV calcium needed
  • Altered consciousness with hypothermia — myxoedema coma
  • High fever, tachycardia, agitation in known thyroid disease — thyroid storm
  • Treatment-refractory oral or systemic candidiasis in a child — consider APS-1

5. Clinical Examination

Structured Approach

General Inspection:

  • Skin pigmentation (hyperpigmentation of Addison's vs vitiligo patches)
  • Body habitus (wasting suggests adrenal or metabolic disease)
  • Alopecia (areata or totalis)
  • Oral examination (candidiasis, hyperpigmented buccal mucosa)

Cardiovascular:

  • Blood pressure (postural hypotension in Addison's)
  • Heart rate (bradycardia in hypothyroidism, tachycardia in hyperthyroidism)
  • Signs of heart failure

Neck:

  • Thyroid examination (goitre, nodularity)
  • Thyroidectomy scar
  • Lymphadenopathy

Hands:

  • Tremor (hyperthyroidism)
  • Palmar crease pigmentation (Addison's)
  • Nail dystrophy (APS-1)
  • Vitiligo

Neurological:

  • Chvostek's sign (tap facial nerve → facial twitch = hypocalcaemia)
  • Trousseau's sign (blood pressure cuff → carpal spasm = hypocalcaemia)
  • Peripheral neuropathy (B12 deficiency, diabetes)
  • Deep tendon reflexes (delayed in hypothyroidism)

Skin:

  • Vitiligo patches
  • Hyperpigmentation pattern
  • Fungal infections (candidiasis)
  • Injection sites (lipodystrophy in T1DM)

Special Tests

TestTechniqueSignificance
Chvostek's signTap facial nerve below zygomaPositive = hypocalcaemia
Trousseau's signInflate BP cuff above systolic for 3 minCarpopedal spasm = hypocalcaemia
Postural BPLying to standing BPDrop >0 mmHg systolic = autonomic dysfunction/adrenal insufficiency
Thyroid palpationAssess size, nodules, tendernessGoitre, nodule detection
Visual acuitySnellen chartKeratitis screening in APS-1

6. Investigations

First-Line Investigations

InvestigationRationaleExpected Findings
9 AM serum cortisolAdrenal function screeningLow (<00 nmol/L) suggests insufficiency; high-normal (>00 nmol/L) excludes
TFTs (TSH, free T4)Thyroid functionHigh TSH + low T4 = hypothyroidism; Low TSH + high T4 = hyperthyroidism
Fasting glucose / HbA1cDiabetes screeningElevated in T1DM
Calcium (corrected)Parathyroid functionLow in hypoparathyroidism
PTHParathyroid functionLow/inappropriately normal with low calcium = hypoparathyroidism
FBCAnaemia screeningMacrocytic anaemia (B12 deficiency), normocytic anaemia (chronic disease)
U&EElectrolyte disturbanceHyponatraemia, hyperkalaemia in Addison's
Vitamin B12, folatePernicious anaemiaLow B12 with macrocytic anaemia

Confirmatory Investigations

InvestigationIndicationInterpretation
Short Synacthen TestSuspected adrenal insufficiencyCortisol fails to rise to >50 nmol/L at 30 min = Addison's
Adrenal autoantibodies (21-hydroxylase)Confirm autoimmune aetiologyPositive in >0% autoimmune Addison's
TPO antibodiesAutoimmune thyroiditisPositive in Hashimoto's
GAD65, IA-2 antibodiesT1DM autoimmune markersPositive supports autoimmune T1DM
Intrinsic factor antibodiesPernicious anaemiaPositive = autoimmune gastritis
AIRE gene testingSuspected APS-1Confirms diagnosis if mutations found

Imaging

ModalityIndicationFindings
Adrenal CTDistinguish autoimmune from other causesSmall, atrophic adrenals (autoimmune); enlarged (infiltrative/haemorrhage/TB)
Thyroid ultrasoundGoitre, nodule evaluationHeterogeneous, hypoechoic in Hashimoto's
DEXA scanOsteoporosis screeningReduced BMD (corticosteroid excess, hypogonadism)

Genetic Testing

TestIndication
AIRE gene sequencingSuspected APS-1, childhood onset, mucocutaneous candidiasis
HLA typingResearch/risk stratification for APS-2

7. Management

Management Algorithm

              AUTOIMMUNE POLYGLANDULAR SYNDROME
                           ↓
┌─────────────────────────────────────────────────────────────┐
│              INITIAL ASSESSMENT                             │
│  - Full endocrine panel (cortisol, TFTs, glucose, Ca, PTH) │
│  - Autoantibody screen (21-OH, TPO, GAD, IF)               │
│  - AIRE gene testing if childhood onset/APS-1 suspected    │
└─────────────────────────────────────────────────────────────┘
                           ↓
┌─────────────────────────────────────────────────────────────┐
│          TREAT EACH COMPONENT                               │
├─────────────────────────────────────────────────────────────┤
│  ADDISON'S DISEASE:                                         │
│  ➤ Hydrocortisone 15-25 mg/day (divided doses)             │
│  ➤ Fludrocortisone 50-200 mcg/day                          │
│  ➤ SICK DAY RULES: Double/triple dose when unwell          │
│  ➤ Emergency injection kit + MedicAlert bracelet           │
├─────────────────────────────────────────────────────────────┤
│  HYPOTHYROIDISM:                                            │
│  ➤ Levothyroxine (start low 25-50 mcg if adrenal insuffi-  │
│    ciency; ALWAYS replace steroids FIRST)                   │
├─────────────────────────────────────────────────────────────┤
│  TYPE 1 DIABETES:                                           │
│  ➤ Insulin therapy (basal-bolus or pump)                   │
│  ➤ Beware: Adrenal insufficiency → recurrent hypoglycaemia │
├─────────────────────────────────────────────────────────────┤
│  HYPOPARATHYROIDISM:                                        │
│  ➤ Calcium supplements (1-3 g/day in divided doses)        │
│  ➤ Active vitamin D (alfacalcidol/calcitriol)              │
│  ➤ Target: Low-normal calcium to avoid hypercalciuria      │
├─────────────────────────────────────────────────────────────┤
│  MUCOCUTANEOUS CANDIDIASIS (APS-1):                         │
│  ➤ Topical/oral antifungals (fluconazole, nystatin)        │
│  ➤ Prophylactic if recurrent                               │
└─────────────────────────────────────────────────────────────┘
                           ↓
┌─────────────────────────────────────────────────────────────┐
│          ANNUAL SCREENING PROTOCOL                          │
│  - TFTs (TSH, fT4)                                         │
│  - Fasting glucose / HbA1c                                 │
│  - 9 AM cortisol (if symptoms or new fatigue)              │
│  - Calcium, PTH                                            │
│  - FBC, B12 (pernicious anaemia)                           │
│  - Coeliac serology if GI symptoms                         │
│  - Gonadal function assessment if fertility concerns       │
└─────────────────────────────────────────────────────────────┘

Hormone Replacement Protocols

ConditionFirst-Line TreatmentMonitoring
Addison's diseaseHydrocortisone 15-25 mg/day (divided: 10 mg AM, 5 mg lunch, 5 mg PM) + Fludrocortisone 50-200 mcg dailyClinical (BP, weight, pigmentation), electrolytes, renin
HypothyroidismLevothyroxine 1.6 mcg/kg/day (start low if cardio risk)TSH every 6-8 weeks until stable, then annually
Type 1 diabetesInsulin (basal-bolus MDI or pump)HbA1c every 3-6 months, CGM encouraged
HypoparathyroidismCalcium 1-3 g/day + Calcitriol 0.25-1 mcg/daySerum calcium, phosphate, urinary calcium, renal function
Premature ovarian failureHRT (oestrogen + progesterone if uterus intact)Bone health, cardiovascular risk, fertility counselling

Critical Treatment Considerations

ScenarioActionRationale
Starting levothyroxine in adrenal insufficiencyReplace steroids FIRST, then levothyroxineThyroxine increases cortisol metabolism → precipitates Addisonian crisis
Sick day rules (Addison's)Double hydrocortisone if febrile/unwell; triple if vomitingPhysiological stress requires increased cortisol
Surgery preparationIV hydrocortisone 100 mg preop + 50-100 mg q8hPrevent perioperative adrenal crisis
Hypoglycaemia in T1DM + Addison'sScreen for adrenal insufficiency; reduce insulinCortisol needed for gluconeogenesis

Patient Education

  • MedicAlert bracelet: Essential for all patients with adrenal insufficiency
  • Emergency injection kit: IM hydrocortisone for emergencies when oral intake impossible
  • Sick day card: Instructions for dose adjustment during illness
  • Annual screening adherence: Even if well, new autoimmune conditions can develop

8. Complications

Immediate (Hours)

ComplicationIncidenceClinical FeaturesManagement
Addisonian crisisVariable in undiagnosedHypotension, shock, hyponatraemia, hyperkalaemia, hypoglycaemiaIV hydrocortisone 100 mg bolus then 50-100 mg q6h, IV fluids
Hypoglycaemic comaT1DM patientsConfusion, seizures, unconsciousnessIV glucose, glucagon, assess for adrenal insufficiency
Hypocalcaemic tetany/seizuresHypoparathyroidismCarpopedal spasm, laryngospasm, generalised seizuresIV calcium gluconate 10%, monitor ECG
Thyroid stormUndiagnosed Graves'High fever, tachycardia, deliriumPTU, beta-blockers, steroids, supportive care

Early (Days-Weeks)

ComplicationClinical FeaturesManagement
Electrolyte disturbanceHyponatraemia, hyperkalaemiaSteroid replacement, IV fluids
Metabolic derangementKetoacidosis (T1DM)Insulin, fluids, potassium
Infection (candidiasis)Oral/oesophageal thrushAntifungals

Late (Months-Years)

ComplicationIncidenceNotes
Osteoporosis20-30%Due to glucocorticoid replacement, hypogonadism
Cardiovascular diseaseIncreasedAssociated with thyroid disease, diabetes
New autoimmune conditionsOngoing riskRequires annual screening
InfertilityVariablePremature ovarian failure, testicular failure
Adrenal crisis during intercurrent illnessPreventablePatient education on sick day rules
Visual impairment (APS-1)15-25%Keratitis, cataracts
Hepatitis (APS-1)10-15%Monitor LFTs

9. Prognosis & Outcomes

Natural History

SyndromePrognosisLife Expectancy
APS-1Chronic, lifelong; accumulation of components over timeReduced if complications (hepatitis, adrenal crisis, malignancy)
APS-2Variable; depends on adequacy of hormone replacementNear-normal with good management

Prognostic Factors

Good PrognosisPoor Prognosis
Early diagnosis and treatmentDelayed diagnosis with crisis presentation
Good treatment adherencePoor compliance with replacement therapy
Regular screening and follow-upMissed appointments / lost to follow-up
Access to specialist careLimited healthcare access
Education on sick day rulesLack of patient education

Treatment Outcomes

ConditionOutcome with Treatment
Addison's diseaseExcellent QoL with adequate replacement; persistent fatigue in some
HypothyroidismNormal life with levothyroxine
T1DMGood glycaemic control reduces microvascular complications
HypoparathyroidismSymptomatic improvement; monitoring for renal calcification needed

10. Evidence & Guidelines

Key Guidelines

GuidelineOrganisationYearKey Points
Primary Adrenal Insufficiency GuidelinesEndocrine Society2016Hydrocortisone preferred; stress dosing protocols; fludrocortisone for all PAI
Hypothyroidism ManagementATA/AACE2012Levothyroxine monotherapy; TSH targets
Type 1 Diabetes StandardsADA2024Individualised HbA1c targets; CGM recommended
Hypoparathyroidism GuidelinesEndocrine Society2022Calcium + active vitamin D; avoid hypercalciuria

Landmark Trials and Evidence

AIRE Gene Discovery (1997)

  • Finnish-German Consortium identified AIRE mutations in APS-1
  • Established genetic basis for disease
  • Enabled diagnostic testing
  • PMID: 9425231

Betterle et al. Natural History Study (2002)

  • n=195 patients with autoimmune Addison's
  • 50% developed additional autoimmune diseases over follow-up
  • Established need for systematic screening
  • PMID: 12456856

Husebye et al. Clinical Registry (2009)

  • Norwegian registry of APS patients
  • Documented spectrum of complications
  • Informed screening protocols
  • PMID: 19246518

Bornstein et al. (2016) — Endocrine Society PAI Guidelines

  • Comprehensive management recommendations
  • Stress dosing protocols
  • Quality of life considerations
  • PMID: 26760044

Evidence Strength

InterventionLevelEvidence Source
Hydrocortisone replacement1aMultiple RCTs, meta-analyses
Fludrocortisone for mineralocorticoid replacement1bRCTs
Annual screening for new components2aCohort studies, expert consensus
Sick day rules education2bObservational studies, case series
AIRE gene testing for APS-11aDiagnostic accuracy studies

11. Patient/Layperson Explanation

What is Autoimmune Polyglandular Syndrome?

Autoimmune Polyglandular Syndrome (APS) is a condition where your immune system, which normally protects you from infections, mistakenly attacks the glands that make important hormones. This leads to several hormone-producing glands not working properly at the same time.

Why does it happen?

In APS, the immune system becomes confused and produces antibodies that attack your own glands. This can run in families and is related to your genes. In children, a specific gene called AIRE is often involved. In adults, a combination of genes makes some people more likely to develop these problems.

What are the symptoms?

Symptoms depend on which glands are affected:

  • Adrenal glands: Tiredness, weakness, weight loss, low blood pressure, salt cravings, darkening of the skin
  • Thyroid gland: Tiredness, weight changes, feeling too hot or cold
  • Pancreas: Increased thirst and urination, weight loss (diabetes)
  • Parathyroid glands: Muscle cramps, tingling in fingers and toes

How is it treated?

Each hormone deficiency is treated by replacing the missing hormone:

  • Steroid tablets (hydrocortisone) for adrenal problems
  • Thyroid hormone tablets for thyroid problems
  • Insulin injections for diabetes
  • Calcium and vitamin D for parathyroid problems

With proper treatment, most people can live normal, active lives.

What to expect?

This is a lifelong condition, but with regular medication and check-ups, you can manage your symptoms well. You will need to take medications every day and see your doctor regularly for blood tests.

When to seek help urgently

Seek immediate medical attention if you experience:

  • Severe weakness, dizziness, or fainting (especially if unwell)
  • Vomiting and unable to keep medications down
  • Muscle cramps or twitching that doesn't stop
  • Very fast heartbeat with high temperature
  • Confusion or reduced consciousness

Always carry a steroid emergency card and wear a medical alert bracelet.


12. References

Primary Guidelines

  1. Bornstein SR, Allolio B, Arlt W, et al. Diagnosis and Treatment of Primary Adrenal Insufficiency: An Endocrine Society Clinical Practice Guideline. J Clin Endocrinol Metab. 2016;101(2):364-389. PMID: 26760044

  2. Brandi ML, Bilezikian JP, Shoback D, et al. Management of Hypoparathyroidism: Summary Statement and Guidelines. J Clin Endocrinol Metab. 2016;101(6):2273-2283. PMID: 26943718

Key Studies

  1. Finnish-German APECED Consortium. An autoimmune disease, APECED, caused by mutations in a novel gene featuring two PHD-type zinc-finger domains. Nat Genet. 1997;17(4):399-403. PMID: 9425231

  2. Betterle C, Dal Pra C, Mantero F, Zanchetta R. Autoimmune adrenal insufficiency and autoimmune polyendocrine syndromes: autoantibodies, autoantigens, and their applicability in diagnosis and disease prediction. Endocr Rev. 2002;23(3):327-364. PMID: 12050123

  3. Husebye ES, Perheentupa J, Rautemaa R, Kämpe O. Clinical manifestations and management of patients with autoimmune polyendocrine syndrome type I. J Intern Med. 2009;265(5):514-529. PMID: 19382991

  4. Kahaly GJ. Polyglandular autoimmune syndromes. Eur J Endocrinol. 2009;161(1):11-20. PMID: 19411300

  5. Michels AW, Gottlieb PA. Autoimmune polyglandular syndromes. Nat Rev Endocrinol. 2010;6(5):270-277. PMID: 20309000

Reviews & Resources

  1. Husebye ES, Anderson MS, Kämpe O. Autoimmune Polyendocrine Syndromes. N Engl J Med. 2018;378(12):1132-1141. PMID: 29562162

  2. Magitta NF, Bøe Wolff AS, Johansson S, et al. A coding polymorphism in NALP1 confers risk for autoimmune Addison's disease and type 1 diabetes. Genes Immun. 2009;10(2):120-124. PMID: 18946481

  3. Society for Endocrinology. Emergency management of acute adrenal insufficiency (adrenal crisis) in adult patients. 2020. sofe.org/adrenal-crisis

  4. Addison's Disease Self-Help Group. Patient resources. addisonsdisease.org.uk

  5. British Thyroid Foundation. Patient information. btf-thyroid.org


13. Examination Focus

High-Yield Exam Topics

TopicKey Points
APS classificationType 1 = AIRE gene, childhood, candidiasis/hypopara/Addison's; Type 2 = HLA-associated, adult, Addison's + thyroid + T1DM
AIRE geneAutoimmune Regulator; mutations cause failure of central tolerance in thymus
21-hydroxylase antibodiesMarker for autoimmune adrenal insufficiency (>0% positive)
Treatment prioritiesALWAYS replace steroids before starting levothyroxine
Sick day rulesDouble hydrocortisone if febrile; triple if vomiting; IM injection if unable to take oral
Addisonian crisis managementIV hydrocortisone 100 mg stat, IV 0.9% saline, treat underlying cause

Sample Viva Questions

Q1: A 35-year-old woman with Type 1 diabetes presents with recurrent hypoglycaemia despite reducing insulin doses. What should you consider?

Model Answer: Consider concurrent adrenal insufficiency (APS-2). Cortisol is essential for gluconeogenesis and counter-regulatory response to hypoglycaemia. Check 9 AM cortisol and perform Short Synacthen Test. Screen for 21-hydroxylase antibodies. If confirmed, start hydrocortisone replacement which may allow normalisation of insulin doses.

Q2: Why must steroids be replaced before starting levothyroxine in a patient with both adrenal insufficiency and hypothyroidism?

Model Answer: Levothyroxine increases metabolic rate and cortisol clearance. In a patient with undiagnosed or undertreated adrenal insufficiency, starting levothyroxine precipitates Addisonian crisis by depleting already low cortisol reserves. Always confirm adequate steroid replacement before initiating thyroid hormone therapy.

Q3: A 7-year-old presents with recurrent oral thrush and hypocalcaemia. What is the likely diagnosis and what investigation would you perform?

Model Answer: This presentation is highly suggestive of APS-1 (APECED). The triad includes chronic mucocutaneous candidiasis (usually first), hypoparathyroidism, and Addison's disease. Perform AIRE gene sequencing for confirmation. Screen for adrenal insufficiency with 9 AM cortisol and Short Synacthen Test. Annual screening for new components is essential.

Common Exam Errors

ErrorCorrect Approach
Starting levothyroxine before steroidsAlways replace steroids first
Missing adrenal insufficiency in T1DM with hypoglycaemiaScreen for Addison's if unexplained hypoglycaemia
Forgetting screening in patients with single autoimmune endocrinopathyAnnual TFTs, glucose, symptoms review
Not educating about sick day rulesAll patients with Addison's need written sick day guidance
Misclassifying APS typesAPS-1 = AIRE/childhood/candidiasis; APS-2 = adult/HLA/no candidiasis

Last Reviewed: 2025-12-24 | MedVellum Editorial Team


Medical Disclaimer: MedVellum content is for educational purposes and clinical reference. Clinical decisions should account for individual patient circumstances. Always consult appropriate specialists.

Last updated: 2025-12-24

At a Glance

EvidenceHigh
Last Updated2025-12-24

Red Flags

  • Addisonian crisis (hypotension, hyponatraemia, hyperkalaemia)
  • Hypoglycaemia in T1DM with undiagnosed adrenal insufficiency
  • Severe hypocalcaemia with tetany or seizures
  • Myxoedema coma (severe hypothyroidism)
  • Thyroid storm in undiagnosed Graves' disease
  • Mucocutaneous candidiasis refractory to treatment

Clinical Pearls

  • **"The Rule of Three"**: APS-1 presents with the triad of Candidiasis, Hypoparathyroidism, and Addison's — usually appearing in that order during childhood. If you see one, look for the others.
  • **"Annual Screen"**: Any patient with one autoimmune endocrine condition should have annual TFTs, glucose, and clinical assessment for adrenal insufficiency.
  • **"AIRE Gene = APS-1"**: Mutations in the AIRE (Autoimmune Regulator) gene cause failure of central tolerance in the thymus, leading to multi-organ autoimmunity in childhood.
  • **Red Flags — Seek Urgent Assessment:**
  • - Hypotension, hyponatraemia, hyperkalaemia — suspect Addisonian crisis

Guidelines

  • NICE Guidelines
  • BTS Guidelines
  • RCUK Guidelines