[Skip to Content]
[Skip to Content Landing]

Acute Myelopathy in a Man With Cutaneous Papules

Educational Objective
Based on this clinical scenario and the accompanying image, understand how to arrive at a correct diagnosis.
1 Credit CME

A 46-year-old man presented with acute-onset weakness of his bilateral lower limbs for 3 days, accompanied by ascending numbness to just below his nipples. Three weeks prior, he had developed acute urinary retention. Neurological examination revealed weakness in bilateral lower limbs (Medical Research Council grade 4 on the right and grade 3 on the left), extensor plantar responses, and a sensory level at T6. Magnetic resonance imaging of his thoracic spine was performed 10 days after the onset of weakness and did not show any signal abnormalities or restricted diffusion. Cerebrospinal fluid analysis findings were remarkable for elevated protein (0.14 g/dL [to convert to grams per liter, multiply by 10]) but white cell count (5 cells/mm3) and glucose levels (54.05 mg/dL [to convert to millimoles per liter, multiply by 0.0555]) were normal. Cytology and flow cytometry were negative for malignancy. Antibodies to aquaporin 4 and myelin oligodendrocyte glycoprotein were not detected. The patient developed a sudden worsening of weakness in his legs 2 weeks after initial presentation. Repeated thoracic spine imaging showed an area of T2 hyperintensity in the left posterolateral cord at T7, which did not enhance with contrast or show restricted diffusion (Figure 1A). Notably, he had a 4-year history of an asymptomatic papular skin eruption with more lesions developing in the last 4 months. These were distributed on the neck, trunk, and limbs, sparing his face, scalp, and genitalia (Figure 1B). Findings of extensive workup for malignancy, including full-body computed tomography scan, paraneoplastic antibody panel, and systemic autoimmune diseases (antinuclear antibody, antidouble stranded DNA, and myositis antibody panel), were negative. The skin papules were biopsied.

Please finish quiz first before checking answer.

You answered correctly!

Read the answer below and download your certificate.

You answered incorrectly.

Read the discussion below and retake the quiz.

A. Malignant atrophic papulosis

The differential diagnoses to consider in a patient who has cutaneous papules and myelopathy can include infectious (eg, secondary syphilis, HIV), ischemic (eg, malignant atrophic papulosis), autoimmune (eg, systemic lupus erythematosus, Behçet disease), and paraneoplastic (eg, scleromyxedema with paraproteinemia) causes.

The diagnosis of malignant atrophic papulosis (MAP) or Köhlmeier-Degos disease was supported by the characteristic cutaneous eruption and histological findings of wedge-shaped dermal collagen degeneration, interstitial mucin deposition, small–vessel angiopathy with luminal thrombi, perivascular and perineural lymphocytic infiltration, and interface vacuolar alteration of the epidermis (Figure 2). These findings, indicative of microvascular injury, can also be seen in the setting of Degos-like diseases, namely dermatomyositis, lupus erythematosus, and scleroderma. Negative autoimmune markers and the absence of characteristic clinical features excluded these differential diagnoses. Scleromyxedema is an unusual condition of dermal mucin deposition and fibroblast proliferation and is typically associated with a monoclonal gammopathy and other systemic manifestations. Histopathological findings of microvascular injury in the patient are not consistent with this diagnosis. Lues maligna is a rare but severe form of secondary syphilis that has been described in patients who have HIV co-infection—cutaneous lesions start out as papules that ulcerate to become well-demarcated necrotic plaques. Syphilis was ruled out with negative findings on serum, cerebrospinal fluid treponemal, and nontreponemal antibody tests, and the absence of spirochetes on skin biopsy. The acute and stepwise deterioration of this patient’s thoracic myelopathy is consistent with a vascular event.

Survey Complete!

Sign in to take quiz and track your certificates

Buy This Activity
Our websites may be periodically unavailable between 12:00am CT March 25, 2023 and 4:00pm CT March 26, 2023 for regularly scheduled maintenance.

JN Learning™ is the home for CME and MOC from the JAMA Network. Search by specialty or US state and earn AMA PRA Category 1 Credit(s)™ from articles, audio, Clinical Challenges and more. Learn more about CME/MOC

CME Disclosure Statement: Unless noted, all individuals in control of content reported no relevant financial relationships. If applicable, all relevant financial relationships have been mitigated.

Article Information

Corresponding Author: Shermyn Neo, MBBS, Department of Neurology, National Neuroscience Institute, 11 Jalan Tan Tock Seng, Singapore 308433 (shermyn.neo.x.m@singhealth.com.sg).

Published Online: November 15, 2021. doi:10.1001/jamaneurol.2021.4194

Correction: This article was corrected on January 31, 2022, to fix an arrowhead that was incorrectly located.

Conflict of Interest Disclosures: None reported.

Additional Contributions: We thank the patient for granting permission to publish this information. We thank Lee Shapiro, MD (Albany Medical College, Albany, New York), Cynthia Magro, MD (Weill Cornell Medicine, New York, New York), Peter A. Merkel, MD (University of Pennsylvania, Philadelphia), and Robert G. Micheletti, MD (University of Pennsylvania), for providing expert advice on the investigation and management of Degos disease. They did not receive any forms of compensation for their advice.

References
1.
Magro  CM , Poe  JC , Kim  C ,  et al.  Degos disease: a C5b-9/interferon-α-mediated endotheliopathy syndrome.   Am J Clin Pathol. 2011;135(4):599-610. doi:10.1309/AJCP66QIMFARLZKIPubMedGoogle ScholarCrossref
2.
Subbiah  P , Wijdicks  E , Muenter  M , Carter  J , Connolly  S .  Skin lesion with a fatal neurologic outcome (Degos’ disease).   Neurology. 1996;46(3):636-640. doi:10.1212/WNL.46.3.636PubMedGoogle ScholarCrossref
3.
Matsuura  F , Makino  K , Fukushima  T ,  et al.  Optic nerve and spinal cord manifestations of malignant atrophic papulosis (Degos disease).   J Neurol Neurosurg Psychiatry. 2006;77(2):260-262. doi:10.1136/jnnp.2005.066134PubMedGoogle ScholarCrossref
4.
Label  LS , Tandan  R .  Myelomalacia and hypoglycorrhachia in malignant atrophic papulosis.   Neurology. 1983;33(7):936-939. doi:10.1212/wnl.33.7.936Google ScholarCrossref
5.
Theodoridis  A , Konstantinidou  A , Makrantonaki  E , Zouboulis  CC . Malignant and benign forms of atrophic papulosis (Köhlmeier-Degos disease).  Br J of Dermatol. 2013;170(1):110-115. doi:10.1111/bjd.12642
6.
Theodoridis  A , Makrantonaki  E , Zouboulis  CC . Malignant atrophic papulosis (Köhlmeier-Degos disease): a review.  Orphanet J Rare Dis. 2013;8:10. doi:10.1186/1750-1172-8-10
7.
Guo  Y-F , Pan  W-H , Cheng  R-H , Yu  H , Liao  W-Q , Yao  Z-R .  Successful treatment of neurological malignant atrophic papulosis in child by corticosteroid combined with intravenous immunoglobulin.   CNS Neurosci Ther. 2014;20(1):88-91. doi:10.1111/cns.12196PubMedGoogle ScholarCrossref
8.
Magro  CM , Wang  X , Garrett-Bakelman  F , Laurence  J , Shapiro  LS , DeSancho  MT . The effects of eculizumab on the pathology of malignant atrophic papulosis.  Orphanet J Rare Dis. 26;8:185. doi:10.1186/1750-1172-8-185
9.
Richardson  PJ , Ottaviani  S , Prelle  A , Stebbing  J , Casalini  G , Corbellino  M .  CNS penetration of potential anti-COVID-19 drugs.   J Neurol. 2020;267(7):1880-1882. doi:10.1007/s00415-020-09866-5PubMedGoogle ScholarCrossref
AMA CME Accreditation Information

Credit Designation Statement: The American Medical Association designates this Journal-based CME activity activity for a maximum of 1.00  AMA PRA Category 1 Credit(s)™. Physicians should claim only the credit commensurate with the extent of their participation in the activity.

Successful completion of this CME activity, which includes participation in the evaluation component, enables the participant to earn up to:

  • 1.00 Medical Knowledge MOC points in the American Board of Internal Medicine's (ABIM) Maintenance of Certification (MOC) program;;
  • 1.00 Self-Assessment points in the American Board of Otolaryngology – Head and Neck Surgery’s (ABOHNS) Continuing Certification program;
  • 1.00 MOC points in the American Board of Pediatrics’ (ABP) Maintenance of Certification (MOC) program;
  • 1.00 Lifelong Learning points in the American Board of Pathology’s (ABPath) Continuing Certification program; and
  • 1.00 CME points in the American Board of Surgery’s (ABS) Continuing Certification program

It is the CME activity provider's responsibility to submit participant completion information to ACCME for the purpose of granting MOC credit.

Close
Want full access to the AMA Ed Hub?
After you sign up for AMA Membership, make sure you sign in or create a Physician account with the AMA in order to access all learning activities on the AMA Ed Hub
Buy this activity
Close
Want full access to the AMA Ed Hub?
After you sign up for AMA Membership, make sure you sign in or create a Physician account with the AMA in order to access all learning activities on the AMA Ed Hub
Buy this activity
Close
With a personal account, you can:
  • Access free activities and track your credits
  • Personalize content alerts
  • Customize your interests
  • Fully personalize your learning experience
Education Center Collection Sign In Modal Right
Close

Name Your Search

Save Search
With a personal account, you can:
  • Access free activities and track your credits
  • Personalize content alerts
  • Customize your interests
  • Fully personalize your learning experience
Close
Close

Lookup An Activity

or

My Saved Searches

You currently have no searches saved.

Close

My Saved Courses

You currently have no courses saved.

Close