HaematologyMyeloid DisordersMyeloid Proliferative Neoplasms (MPN)

Primary Myelofibrosis

Primary myelofibrosis is a chronic myeloproliferative neoplasm characterized by clonal hematopoietic stem cell proliferation with progressive bone marrow fibrosis, extramedullary hematopoiesis, and peripheral blood cytopenias with leukoerythroblastic changes.

Primary Myelofibrosis (PMF)

Anatomy & Function: The Bone Marrow and Spleen

Understanding PMF requires understanding where blood is made and why it moves.

Etiology

A. Primary Myelofibrosis — Driver Mutations

The pathogenesis of PMF centres on acquired somatic mutations in haematopoietic stem cells that constitutively activate the JAK-STAT signalling pathway.

Pathophysiology

This is the core of understanding PMF. Let's build it step by step.

Classification

C. Risk Stratification (IPSS / DIPSS / DIPSS-Plus)

Risk stratification systems for primary myelofibrosis: IPSS (at diagnosis), DIPSS/DIPSS-Plus (at follow-up) [2].

Clinical Features

PMF has a clinical course divided into two phases: prefibrotic (cellular) phase and fibrotic (overt) phase [5]. The clinical features differ by phase but overlap considerably.

Differential Diagnosis of Primary Myelofibrosis

The differential diagnosis of PMF is really about two clinical presentations that bring a patient to attention:

  1. The patient with massive splenomegaly — what else causes this?
  2. The patient with pancytopenia and a leukoerythroblastic blood film — what else causes this?
  3. The patient with bone marrow fibrosis on biopsy — what else causes fibrosis?

We need to systematically work through each of these because PMF sits at the intersection of all three.


References

[1] Lecture slides: Block A - Splenomegaly: common causes of splenomegaly; myeloproliferative diseases.pdf (pp. 22, 27, 29–31) [2] Senior notes: Maksim Medicine Notes.pdf (pp. 170–171) [3] Senior notes: Block A - Splenomegaly: common causes of splenomegaly; myeloproliferative diseases.pdf (pp. 29–31) [4] Senior notes: MBBS Final MB (Medicine) (Felix PY Lai).pdf (pp. 1450–1451) [5] Senior notes: Ryan Ho Haemtology.pdf (pp. 75, 77–78) [7] Senior notes: Ryan Ho Haemtology.pdf (pp. 83) [8] Lecture slides: GC 086. Splenomegaly.pdf (p. 38) [9] Senior notes: Block A - Splenomegaly: common causes of splenomegaly; myeloproliferative diseases.pdf (p. 27) [10] Senior notes: Block A - High white cell count: acute and chronic leukaemia; bone marrow transplantation; immunogenetics.pdf (p. 22) [11] Senior notes: Block A - An old man with bone pain and anaemia: multiple myeloma; monoclonal gammopathy.pdf (p. 12) [12] Senior notes: MBBS Final MB (Surgery) (Felix PY Lai).pdf (p. 130) [13] Senior notes: MBBS Final MB (Pediatrics) (Felix PY Lai).pdf (p. 772) [14] Senior notes: Adrian Lui Pediatrics Notes.pdf (p. 369) [15] Senior notes: Block A - Family history of anaemia: inherited causes of anaemia; haemolytic anaemia; aplastic anaemia.pdf (pp. 7–8) [16] Senior notes: Ryan Ho Fundamentals.pdf (pp. 390, 398)

WHO 2016 (Revised 4th Edition) Diagnostic Criteria

The WHO criteria separate PMF into two entities based on the degree of marrow fibrosis [4][5]:

Investigation Modalities with Key Findings and Interpretations

3. Bone Marrow Examination

Bone marrow examination is necessary to demonstrate marrow fibrosis [5] and is mandatory for the diagnosis of PMF. It consists of two components:

8. Prognostic Risk Stratification Tools

These are not diagnostic tests but are integral to the workup because they guide management (especially the decision for allogeneic HSCT).

Risk stratification systems for primary myelofibrosis: IPSS (at diagnosis), DIPSS/DIPSS-Plus (at follow-up) [2].

Treatment Modalities in Detail

1. Allogeneic Haematopoietic Stem Cell Transplantation (HSCT) — The Only Cure

Allogeneic stem cell transplantation is the only potentially curative procedure for PMF [3].

2. JAK Inhibitors — Disease-Modifying Symptom Control

Targeted JAK2 inhibitors decrease spleen size and constitutional symptoms [3].

Treatment of primary cause, e.g., JAK2 inhibitor in primary myelofibrosis [16].

The name tells you the mechanism: JAK = Janus Activated Kinase inhibitor — blocks the constitutively active JAK-STAT signalling pathway that drives the disease.

Important Concept

JAK inhibitors are NOT curative. They do not significantly reduce the mutant allele burden or reverse marrow fibrosis. They work primarily by suppressing the cytokine storm (reducing spleen size, improving constitutional symptoms, and improving quality of life). They are given regardless of JAK2 mutation status because the JAK-STAT pathway is activated in all PMF subtypes (JAK2, CALR, and MPL mutations all converge on this pathway) [2].

3. Supportive Care for Cytopenias

Supportive for cytopenia [3]:

5. Splenectomy — Surgical Intervention

Splenectomy → sometimes performed to relieve pressure symptoms and decrease transfusion requirements [3].

Surgical treatment if splenomegaly causes problems, e.g., ↑ transfusion requirement [16].

Complications of Primary Myelofibrosis

PMF is a progressive disease whose complications arise from three fundamental processes: (1) progressive marrow failure from fibrosis, (2) extramedullary haematopoiesis and its consequences, and (3) clonal evolution of the malignant stem cell. Additionally, complications can arise from the treatments themselves. Let's work through each systematically.


1. Leukaemic Transformation (Blast Phase / AML)

This is the most feared complication.

PMF has the highest rate of leukaemic transformation among the Ph-negative MPNs — 15% [3]. Other sources report 6–18% [5] or 8% per year [4].

For comparison across MPNs [3][5][8]:

  • PMF: 6–18% (highest)
  • PV: 10% at 10 years, 25% at 25 years (2nd highest) [5]; transformation to acute leukaemia 5% [8]
  • ET: < 5% (15-year cumulative risk) (lowest) [19]

All forms of MPN share the potential to progress to myelofibrosis and blastic transformation [8].

2. Thrombotic Complications

MPN increases risk of both arterial and venous thrombosis because blood is more viscous due to the high cell counts [6].

The JAK2 mutation-positive subtype is especially prone to increasing thrombosis risk [6].

7. Complications of Treatment

References

[2] Senior notes: Maksim Medicine Notes.pdf (pp. 170–171) [3] Senior notes: Block A - Splenomegaly: common causes of splenomegaly; myeloproliferative diseases.pdf (pp. 28–32) [4] Senior notes: MBBS Final MB (Medicine) (Felix PY Lai).pdf (p. 1457) [5] Senior notes: Ryan Ho Haemtology.pdf (pp. 75, 77–78) [6] Senior notes: Block A - Leg swelling and chest pain: deep vein thrombosis; pulmonary embolism; Thrombophilia.pdf (p. 18) [8] Lecture slides: GC 086. Splenomegaly.pdf (pp. 32, 38) [16] Senior notes: Ryan Ho Fundamentals.pdf (pp. 397–398) [18] Senior notes: Block A - High white cell count: acute and chronic leukaemia; bone marrow transplantation; immunogenetics.pdf (p. 34) [19] Senior notes: Block A - Splenomegaly: common causes of splenomegaly; myeloproliferative diseases.pdf (p. 29); MBBS Final MB (Medicine) (Felix PY Lai).pdf (p. 1449) [20] Senior notes: Block A - Fever after a blood transfusion: transfusion and related problems.pdf (p. 27) [21] Senior notes: Maksim Surgery Notes.pdf (p. 153) [22] Senior notes: Ryan Ho Haemtology.pdf (p. 156)

High Yield Summary

  1. PMF is a chronic Ph-negative MPN characterised by bone marrow fibrosis (from reactive fibroblasts, NOT clonal fibroblasts), extramedullary haematopoiesis, and massive splenomegaly.

  2. The megakaryocytes are the culprit — they release PDGF and TGF-β → stimulate fibroblast proliferation → reticulin/collagen deposition → marrow fibrosis.

  3. Driver mutations: JAK2 V617F (60–65%), CALR (20–25%), MPL (7%). All converge on constitutive JAK-STAT activation. Triple-negative = worst prognosis.

  4. Two phases: Pre-fibrotic (↑ WBC and Plt, minimal fibrosis) → Overt fibrotic (pancytopenia + massive splenomegaly).

  5. Classic blood film: Leukoerythroblastic picture + tear-drop RBCs. BM aspirate = dry tap. Trephine biopsy is essential.

  6. Most common symptom: Severe fatigue. Most apparent sign: Massive splenomegaly.

  7. Least common MPN, worst prognosis (median survival 3–5 years). Highest rate of AML transformation (6–18%).

  8. Risk stratification: IPSS (at diagnosis), DIPSS/DIPSS-Plus (dynamic). Parameters: age > 65, constitutional symptoms, Hb < 10, WBC > 25, blasts ≥ 1%.

  9. Only curative treatment: Allogeneic HSCT (limited by age and comorbidities).

  10. MPN thrombosis: JAK2+ MPNs prone to unusual-site thrombosis (e.g., mesenteric vein). Screen for JAK2 in unusual-site clots.

High Yield Summary — DDx of PMF

  1. Before diagnosing PMF, you MUST exclude: CML (BCR-ABL1), PV (elevated RBC mass), ET (megakaryocyte-predominant without fibrosis/atypia), and MDS (dysplastic features).

  2. Massive splenomegaly DDx (exam favourite): CML, PMF, Gaucher disease, Kala-azar, Malaria, Thalassaemia major.

  3. Pancytopenia DDx: Aplastic anaemia (hypocellular marrow, NO splenomegaly) vs PMF (fibrotic marrow, MASSIVE splenomegaly) — feel for the spleen!

  4. Leukoerythroblastic picture = marrow infiltration until proven otherwise. Causes: PMF, metastatic cancer, leukaemia/lymphoma, severe sepsis/haemolysis.

  5. Pre-PMF vs ET: Both can present with thrombocytosis. Pre-PMF has megakaryocyte atypia, higher LDH, higher risk of progression. BM biopsy is the only way to distinguish.

  6. Dry tap on BM aspirate: Think PMF, hairy cell leukaemia, metastatic cancer. Always proceed to trephine biopsy.

  7. Acute myelofibrosis (rare AML variant): rapid onset, pancytopenia, NO splenomegaly — unlike chronic PMF.

High Yield Summary — Diagnosis of PMF

  1. WHO 2016 criteria: ALL 3 major + ≥ 1 minor for both pre-PMF and overt PMF. The key difference is fibrosis grade (≤ 1 for pre-PMF; ≥ 2 for overt PMF).

  2. Major criteria: (1) Megakaryocyte atypia ± fibrosis, (2) Exclusion of CML/PV/ET/MDS, (3) Clonal marker or no reactive cause.

  3. Minor criteria: Anaemia, WBC ≥ 11, splenomegaly, ↑LDH, ± leukoerythroblastosis (overt PMF only). Must be confirmed on 2 consecutive measurements.

  4. PBS hallmarks: Leukoerythroblastic picture + tear-drop RBCs — indicates marrow infiltration.

  5. BM aspirate = dry tap (classic). Trephine biopsy is mandatory — shows megakaryocyte atypia + fibrosis. Reticulin = silver stain; Collagen = trichrome stain.

  6. Molecular testing: JAK2 V617F (60–65%), CALR (20–25%), MPL (7%). BCR-ABL1 must be negative. Can be done on peripheral blood.

  7. Exclusion of CML is the first molecular step — if BCR-ABL1 positive, it is CML by definition.

  8. Risk stratification: IPSS at diagnosis, DIPSS/DIPSS-Plus dynamically. 5 parameters: age > 65, constitutional symptoms, Hb < 10, WBC > 25, blasts ≥ 1%.

High Yield Summary — Management of PMF

  1. Only curative treatment: Allogeneic HSCT — reserved for Int-2/High risk patients who are fit and have a donor. Feasibility limited by age (median 67) and comorbidities.

  2. Management is predominantly palliative if not eligible for HSCT.

  3. JAK inhibitors (ruxolitinib first-line): reduce spleen size and constitutional symptoms; work regardless of JAK2 mutation status (all driver mutations converge on JAK-STAT). NOT curative. Worsen anaemia and thrombocytopenia (on-target effect).

  4. Pacritinib (selective JAK2 inhibitor, approved 2022): specifically for patients with Plt < 50 where ruxolitinib is too myelosuppressive.

  5. Supportive care: Transfusion ± iron chelation, EPO, folic acid, allopurinol (gout prophylaxis), luspatercept (TGF-β inhibitor for anaemia).

  6. Hydroxyurea: Cytoreductive agent for leukocytosis/thrombocytosis/splenomegaly; S/E include myelosuppression, mucocutaneous ulcers, peripheral neuropathy, teratogenicity.

  7. Splenectomy: For refractory symptomatic splenomegaly; high perioperative mortality (6.3%); requires pre-splenectomy vaccination (Pneumococcus, Meningococcus, Hib). Does NOT impair haematopoiesis because EMH in spleen is ineffective.

  8. Risk stratification guides management: IPSS at diagnosis; DIPSS/DIPSS-Plus dynamically; MIPSS70 for transplant decisions.

High Yield Summary — Complications of PMF

  1. AML transformation: PMF has the highest blast transformation rate (6–18%) among Ph-negative MPNs. Secondary AML from PMF has a dismal prognosis — resistant to standard chemotherapy.

  2. Thrombohaemorrhagic complications: JAK2-positive MPN causes thrombosis in unusual sites (mesenteric, portal, hepatic veins — Budd-Chiari). Paradoxically, extreme thrombocytosis can cause acquired vWD → bleeding.

  3. Portal hypertension: From hepatic EMH infiltration + increased splenic blood flow + portal vein thrombosis. Leads to ascites, variceal bleeding.

  4. Cord compression: EMH can occur at any site, including spine → neurological emergency.

  5. Transfusion haemosiderosis: 200 mg iron per unit of blood, only 1 mg/day excreted → iron overload in liver (fibrosis, HCC), heart (failure), endocrine organs (DM, hypogonadism).

  6. Post-splenectomy OPSI: Encapsulated organisms (Pneumococcus, Meningococcus, Hib). Vaccinate pre-splenectomy + prophylactic penicillin. Mnemonic: "Some Nasty Killers Have Some Capsule Protection."

  7. HSCT complications: GVHD (acute/chronic), VOD, graft rejection, infections, secondary malignancy, endocrine dysfunction, infertility, cataract. HSCT is not always a silver bullet — relapse can be 30–40%.

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