LETTERTOTHEEDITOR

Chronic inflammatory demyelinating polyneuropathy afterautologous peripheral blood stem cell transplantation

Dear Editor,

Autologous peripheral blood stem cell transplanta-

tion (APBSCT) has found a place in the treatment of a

variety of haematological disorders, including lympho-

mas, leukaemias and multiple myeloma. Neuromuscular

complications have rarely been reported after APBSCT,

including axonal neuropathy (Boiron et al., 1994), brachial

plexopathy, and polyradiculopathy (Rabinstein et al.,

2003). We report a patient with chronic inflammatory

demyelinating polyneuropathy (CIDP) after autologous

stem cell transplantation for multiple myeloma.

A 62-year-old man presented with a 6-week history

of low-back pain without neurological signs. Blood

tests revealed anaemia (Hb 7.4 g/dL), raised erythro-

cyte sedimentation rate (ESR) (143 mm/h), an immuno-

globulin-A k paraprotein (36.1 g/L), a skeletal survey

showed mottled lucencies throughout the skull vault,

and a bone marrow examination indicated 51% plasma

cells with reduced marrow reserve. These findings

were diagnostic of multiple myeloma.

He was treated with the C-Dex-Thal regime (cyclo-

phosphamide 500 mg/week, dexamethasone 40 mg/

day for 4 days and thalidomide 100 mg/day).

Appropriate counselling including a written description

of the adverse effects of thalidomide and signed con-

sent were undertaken before commencing thalidomide.

Two months into treatment he complained of tingling

numbness in hands and feet, which was ascribed to

thalidomide neuropathy, and the dosage was reduced

to 50 mg/day with symptom resolution. After six cycles

of chemotherapy, he was judged to have made a good

response (paraprotein level almost undetectable), and

he underwent APBSCT; thalidomide was stopped

prior to transplantation. Peripheral blood stem cells

were mobilized with ESHAP [etoposide, cisplatin, cytar-

abine, and methylprednisolone] with CD34 yield of

5.64–5.94 � 106/kg. Following melphalan 200 mg/m2,

half the stem cells were infused.

Approximately 3 weeks later, he developed pins and

needles in his feet and then hands accompanied by

progressive weakness causing difficulty with fine finger

manipulative tasks including holding even light objects.

These symptoms were ascribed to ‘severe thalidomide

neuropathy,’ and a routine neurological appointment

was requested. When seen 2 months later, his symp-

toms had steadily improved spontaneously. Aside from

some distal small muscle wasting and absence of distal

reflexes, neurological examination was normal.

Nerve conduction studies (NCS) showed a severe

demyelinating sensorimotor neuropathy in all limbs.

Lower limb sensory responses were unrecordable;

upper limb sensory amplitudes were reduced with pro-

longed sensory latencies. There was marked motor

conduction slowing with temporal dispersion (median

nerve 26 m/s, normal range (NR) > 49 m/s and tibial

nerve 30 m/s, NR > 41 m/s) but no conduction block.

F-wave responses were markedly prolonged in all limbs.

Electromyography of tibialis anterior was normal. These

findings were consistent with a diagnosis of CIDP

(Report from an Ad Hoc Subcommittee of the

American Academy of Neurology AIDS Task Force,

1991). In view of his continuing clinical improvement,

lumbar puncture for cerebrospinal fluid protein concen-

tration was not thought justified, and he declined intra-

venous immunoglobulin treatment.

This patient developed CIDP 3–4 weeks after

APBSCT for multiple myeloma. Although this might

represent a chance concurrence, we think a causal

relationship more likely. CIDP has been reported in a

child 4 years after allogeneic bone marrow transplanta-

tion (Adams et al., 1995), but we have not identified any

prior adult case of CIDP following APBSCT. Temporary

imbalances in the mechanisms of immune regulation

known to occur after immune reconstitution have

been suspected in the pathogenesis of post-transplan-

tation neuropathies (Rabinstein et al., 2003). Both cell-

mediated and antibody-mediated immune responses to

glycolipid or myelin protein antigens have been impli-

cated in the pathogenesis of CIDP (Said, 2002).

Address correspondence to: Dr. Gary Peters, Walton Center forNeurology and Neurosurgery, Lower Lane, Liverpool, L9 7LJ, UK.Tel: þ44-0151-525-3611; Fax: þ44-0151-529-5513; E-mail:gary.peters@thewaltoncentre.nhs.uk

Journal of the Peripheral Nervous System 10:384–385 (2005)

� 2005 Peripheral Nerve Society 384 Blackwell Publishing

Although initially ascribed to thalidomide neuropathy,

our patient’s illness differed both clinically and neuro-

physiologically from this condition, which is usually a distal

sensory axonal neuropathy, but which in severe cases

can cause irreversible weakness (Cavaletti et al., 2004).

Thalidomide treatment guidelines have been published

(Powell and Gardner-Medwin, 1994; Zeldis et al., 1999),

some of which recommend baseline and serial NCS in all

patients. However, in practice, not all patients receiving

thalidomide have baseline NCS (36% in one survey;

Chave et al., 2001). The absence of baseline and serial

NCS in our patient makes it impossible to say whether

his pre-transplant paraesthesia may have been due to

thalidomide, and hence, incidental to the post-transplant

changes or the initial symptoms of CIDP.

Hence, this case broadens the reported neuro-

pathic complications observed following APBSCT. It

also shows the importance of adhering to guidelines

recommending baseline and serial NCS in patients

receiving thalidomide not only for diagnostic purposes

but also for differential diagnostic purposes.

Sincerely,

Gary Peters, Andrew J. Larner,

Walton Center for Neurology and Neurosurgery, UK

AcknowledgementWe thank Dr. Brian Tedman for neurophysiological

studies.

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Letter to the Editor Journal of the Peripheral Nervous System 10:384–385 (2005)

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