Hematology · PANCE / PANRE

Paroxysmal Nocturnal Hemoglobinuria (PNH)

Acquired clonal stem cell disorder with complement-mediated intravascular hemolysis, thrombosis, and cytopenias.

Also known as: PNH, paroxysmal nocturnal hemoglobinuria, Marchiafava-Micheli

Overview

Acquired clonal hematopoietic stem cell disorder caused by somatic mutation in the PIGA gene, resulting in deficiency of glycosylphosphatidylinositol (GPI)-anchored proteins (CD55, CD59) on blood cells. Loss of complement regulators triggers intravascular hemolysis, hypercoagulability, and bone marrow dysfunction.

Epidemiology

Rare — incidence ~1-2 per million per year. Median age at diagnosis 30s-40s. No sex predominance. Strongly associated with aplastic anemia and other bone marrow failure syndromes.

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Risk factors

  • Pre-existing aplastic anemia or MDS
  • No known inherited predisposition (mutation is acquired/somatic)

Pathophysiology

Somatic mutation in PIGA (X-linked) within a hematopoietic stem cell impairs synthesis of the GPI anchor. Progeny cells lack GPI-anchored complement regulators CD55 (decay-accelerating factor) and CD59 (membrane inhibitor of reactive lysis). Unregulated activation of the alternative complement pathway lyses red cells (intravascular hemolysis). Free hemoglobin scavenges nitric oxide → smooth muscle dystonia (esophageal spasm, abdominal pain, erectile dysfunction) and platelet activation contributing to thrombosis.

Clinical presentation

Symptoms

  • Fatigue and dyspnea from chronic anemia
  • Episodic dark/cola-colored urine — classically first morning void (overnight acidosis enhances complement activity)
  • Abdominal, back, or chest pain from smooth muscle dystonia
  • Dysphagia, erectile dysfunction
  • Unexplained thrombosis — particularly hepatic veins (Budd-Chiari), portal/mesenteric, cerebral venous sinuses, dermal venules

Signs / physical exam

  • Pallor, jaundice
  • Hepatomegaly with ascites (Budd-Chiari)
  • Cytopenias (anemia ± leukopenia, thrombocytopenia)

Classic findings

Coombs-negative intravascular hemolytic anemia with hemoglobinuria, iron deficiency from urinary loss, and atypical-site venous thrombosis.

Differential diagnosis

  • Autoimmune hemolytic anemia — Positive direct Coombs; PNH is Coombs-negative
  • Aplastic anemia — Pancytopenia with hypocellular marrow; PNH and AA frequently coexist — screen all AA patients
  • Myelodysplastic syndrome — Dysplastic morphology, cytogenetic abnormalities; flow cytometry can distinguish
  • Hereditary or acquired TTP/HUS — Schistocytes, thrombocytopenia, renal failure; ADAMTS13 activity
  • March hemoglobinuria / mechanical hemolysis — Hemoglobinuria after vigorous exercise or mechanical valve; no clonal flow defect

Diagnostic workup

Diagnostic criteria

Flow cytometric demonstration of a GPI-deficient clone (≥1% of granulocytes by FLAER) in a patient with hemolysis, cytopenias, or thrombosis. Clinical category assigned: classic PNH (florid hemolysis, large clone), PNH in setting of marrow failure, or subclinical PNH (clone <1% without symptoms).

Labs

  • CBC — anemia ± pancytopenia, often with elevated reticulocytes
  • Markers of intravascular hemolysis: LDH markedly elevated, haptoglobin undetectable, indirect bilirubin elevated, free plasma hemoglobin
  • Urinalysis — hemoglobinuria and hemosiderinuria (urinary iron loss → iron deficiency)
  • Direct antiglobulin test — NEGATIVE
  • Flow cytometry on peripheral blood — DIAGNOSTIC: deficiency of CD55 and CD59 on RBCs; FLAER (fluorescein-labeled aerolysin) binding test on granulocytes/monocytes is most sensitive
  • Bone marrow biopsy if cytopenias or suspected aplastic anemia

Imaging

  • Doppler ultrasound or MR venography of abdomen if Budd-Chiari or portal vein thrombosis suspected
  • MR/MRV brain for cerebral venous sinus thrombosis

Diagnostic algorithm

flowchart TD
  A[Coombs-negative<br/>hemolytic anemia<br/>± thrombosis<br/>± cytopenias] --> B[Flow cytometry<br/>peripheral blood]
  B --> C[FLAER on granulocytes<br/>CD55/CD59 on RBCs]
  C -->|GPI-deficient<br/>clone present| D[PNH diagnosis]
  D --> E[Meningococcal vax<br/>+ prophylaxis]
  E --> F[Eculizumab or<br/>ravulizumab]
  F --> G{Breakthrough<br/>hemolysis?}
  G -->|Yes| H[Pegcetacoplan<br/>or iptacopan]
  G -->|No| I[Continue + monitor]
  D --> J{Marrow failure?}
  J -->|Yes| K[Allo-HSCT<br/>only cure]
PNH diagnostic flow and treatment ladder anchored on terminal complement inhibition.

Treatment

First-line

  • Terminal complement inhibition — eculizumab (anti-C5 monoclonal antibody) or ravulizumab (long-acting anti-C5)
  • Meningococcal vaccination (MenACWY + MenB) at least 2 weeks before C5 inhibitor initiation; antibiotic prophylaxis (penicillin or ciprofloxacin) for vaccine lag period
  • Folic acid supplementation; iron repletion as needed
  • Therapeutic anticoagulation for documented thrombosis

Classic hemolytic PNH

  • Eculizumab or ravulizumab
  • Add proximal complement inhibitor (pegcetacoplan — C3 inhibitor) for breakthrough hemolysis on anti-C5 therapy
  • Newer agents: iptacopan (oral factor B inhibitor), danicopan (factor D inhibitor) for residual hemolysis

PNH with bone marrow failure

  • Immunosuppression (ATG + cyclosporine) for associated aplastic anemia
  • Allogeneic hematopoietic stem cell transplant — only curative option

Thrombosis

  • Long-term anticoagulation (warfarin or DOAC — apixaban, rivaroxaban) PLUS complement inhibition
  • Consider catheter-directed thrombolysis or TIPS for acute Budd-Chiari

Second-line / adjunct

  • Allogeneic stem cell transplantation — only curative therapy; reserved for transplant-eligible patients with marrow failure or refractory disease
  • Primary thromboprophylaxis with anticoagulation may be considered for large clones (>50%) without complement inhibition

Complications

  • Thrombosis (leading cause of mortality) — Budd-Chiari, portal, cerebral, mesenteric, dermal
  • Chronic kidney disease from recurrent hemoglobinuria and microvascular thrombosis
  • Pulmonary hypertension from chronic intravascular hemolysis
  • Progression to aplastic anemia, MDS, or AML
  • Meningococcal sepsis (Neisseria meningitidis) in patients on terminal complement inhibitors

PANCE pearls

  • Despite the name, hemolysis is continuous — nocturnal hemoglobinuria reflects concentration of overnight breakdown products.
  • Unexplained thrombosis at unusual sites (especially hepatic veins) in a young adult should prompt PNH screening.
  • Screen all aplastic anemia and unexplained Coombs-negative hemolysis patients with flow cytometry.
  • Anti-C5 therapy controls intravascular hemolysis but extravascular (C3-mediated) hemolysis can persist — pegcetacoplan or oral factor B/D inhibitors address this.
  • Meningococcal vaccination is mandatory before starting eculizumab/ravulizumab; document and provide patient wallet card.

References

  • International PNH Interest Group — Parker C et al. Diagnosis and management of paroxysmal nocturnal hemoglobinuria. Blood 2005; 106:3699-3709 (with subsequent updates).
  • FDA approvals — Eculizumab (Soliris) approved 2007; ravulizumab (Ultomiris) 2018; pegcetacoplan (Empaveli) 2021; iptacopan (Fabhalta) 2023.

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