Malignant Atrophic Papulosis 

  • Author: L Campbell Levy, MD; Chief Editor: Julian Katz, MD   more...
 
Updated: Jan 8, 2010
 

Background

Kohlmeier described a case of malignant atrophic papulosis (MAP) as a form of thromboangiitis obliterans in 1941.[1] It was recognized as a distinct clinical entity by Degos in 1942, hence the name.[2] Since that time, 2 distinct clinical patterns have been recognized. A malignant variant affects multiple organ systems and results in death (most commonly from intestinal perforation) within a 2-year period. A benign form that is limited to the skin has a prolonged survival and low morbidity. A rare familial form has also been described, which also has a benign prognosis.

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Pathophysiology

Malignant atrophic papulosis is a multisystem disorder involving small-caliber blood vessels. The disease is characterized by narrowing and occlusion of the lumen by intimal proliferation and thrombosis, which leads to ischemia and infarction of the involved organ systems. Malignant atrophic papulosis is different from other vasculitides in that inflammation is not a prominent component of the disease. Malignant atrophic papulosis may involve the gastrointestinal and genitourinary tracts, central and peripheral nervous systems, skin, heart, lungs, eyes, pancreas, adrenals, and kidneys. The disease involves the skin alone in 37% of cases. The gastrointestinal tract is involved in about 50% of cases, and neurologic involvement occurs in approximately 20% of patients.

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Epidemiology

Frequency

International

Malignant atrophic papulosis is a rare disease, with approximately 200 cases reported to date.

Most of the cases are sporadic, although a benign familial variant has been described. There have been approximately 30 reports involving 10 families. Only 4 of the 30 cases had systemic involvement.

Mortality/Morbidity

The morbidity and mortality of malignant atrophic papulosis depend upon the extent of disease involvement. The benign cutaneous variant occurs in approximately 4-15% of cases. Most patients with the cutaneous-limited variant, who were monitored for over a decade, have not suffered significant morbidity. With systemic disease, the reported mean survival is approximately 2 years, but there is a wide variation, from less than 1 year to more than 12 years. The main causes of morbidity and mortality are bowel infarction, bowel perforation, CNS infarction and hemorrhage, and pleuropericardial disease. The benign and malignant variants are clinically indistinguishable initially but become distinct once systemic complications arise. Lack of systemic involvement at 2 years after diagnosis portends a better prognosis.

Race

Most cases of malignant atrophic papulosis reported from Europe and North America have been in whites. The disease has also been reported from Japan, India, and Africa.

Sex

Malignant atrophic papulosis affects both sexes. A slight male predominance has been reported but has not been substantiated.

Age

The disease predominantly affects young adults, but cases have been described in infants and children. Moss et al reported the case of a 6-month-old infant who was admitted to the emergency department with bilateral subdural fluid collections and skin ulcers that resembled cigarette burns.[3] Due to the infant's presentation, child abuse was suspected; however, during the child's admission, his neurologic condition continued to deteriorate, with progressive cerebral infarctions, and his skin ulcers revealed failure to heal. Histology confirmed the diagnosis of Degos disease. The child received palliative care and died 8 weeks after presentation.[3]

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Contributor Information and Disclosures
Author

L Campbell Levy, MD  Fellow, Section of Gastroenterology and Hepatology, Department of Internal Medicine, Dartmouth Hitchcock Medical Center

L Campbell Levy, MD is a member of the following medical societies: Alpha Omega Alpha

Disclosure: Nothing to disclose.

Coauthor(s)

Robert J MacNeal, MD  Staff Physician, Department of Dermatology, Critical Care Fellowship Reviewer, Dartmouth-Hitchcock Medical Center; Supervising Medical Officer, Veterans Administration Hospital, White River Junction, Vermont

Robert J MacNeal, MD is a member of the following medical societies: Alpha Omega Alpha

Disclosure: Nothing to disclose.

Brian E Lacy, MD, PhD  Associate Professor of Medicine, Dartmouth Medical School; Director of GI Motility Laboratory, Department of Gastroenterology, Dartmouth Hitchcock Medical Center

Brian E Lacy, MD, PhD is a member of the following medical societies: Alpha Omega Alpha, American College of Gastroenterology, American Gastroenterological Association, American Motility Society, and American Society for Gastrointestinal Endoscopy

Disclosure: Nothing to disclose.

Hemant Pande, MD  Consulting Staff, Department of Gastroenterology, Leesville Surgical Clinic and Digestive Disease Center

Hemant Pande, MD is a member of the following medical societies: American College of Gastroenterology, American Gastroenterological Association, and American Society for Gastrointestinal Endoscopy

Disclosure: Nothing to disclose.

Lawrence J Cheskin, MD  Director, Johns Hopkins Weight Management Center; Associate Professor, Health, Behavior & Society, Johns Hopkins Bloomberg School of Public Health; Joint Appointment, Department of Medicine, Division of Gastroenterology, Johns Hopkins University School of Medicine; International Health/Human Nutrition, JH Bloomberg School of Public Health

Lawrence J Cheskin, MD is a member of the following medical societies: American College of Gastroenterology, American College of Physicians, and American Gastroenterological Association

Disclosure: Medifast Salary Employment; Mushroom Council Grant/research funds research grant; Medifast Honoraria Board membership

Specialty Editor Board

David Eric Bernstein, MD  Chief, Section of Hepatology, North Shore University Hospital, Director, Associate Professor, Department of Internal Medicine, Division of Hepatology, New York University School of Medicine

David Eric Bernstein, MD is a member of the following medical societies: American Association for the Study of Liver Diseases, American College of Gastroenterology, American College of Physicians, American Gastroenterological Association, and American Society for Gastrointestinal Endoscopy

Disclosure: Nothing to disclose.

Francisco Talavera, PharmD, PhD  Senior Pharmacy Editor, eMedicine

Disclosure: eMedicine Salary Employment

Douglas M Heuman, MD, FACP, FACG, AGAF  Chief of Hepatology, Hunter Holmes McGuire Department of Veterans Affairs Medical Center; Professor, Department of Internal Medicine, Division of Gastroenterology, Virginia Commonwealth University School of Medicine

Douglas M Heuman, MD, FACP, FACG, AGAF is a member of the following medical societies: American Association for the Study of Liver Diseases, American College of Physicians, and American Gastroenterological Association

Disclosure: Nothing to disclose.

Alex J Mechaber, MD, FACP  Associate Dean for Undergraduate Medical Education, Associate Professor of Medicine, University of Miami Miller School of Medicine

Alex J Mechaber, MD, FACP is a member of the following medical societies: Alpha Omega Alpha, American College of Physicians-American Society of Internal Medicine, and Society of General Internal Medicine

Disclosure: Nothing to disclose.

Chief Editor

Julian Katz, MD  Clinical Professor of Medicine, Drexel University College of Medicine; Consulting Staff, Department of Medicine, Section of Gastroenterology and Hepatology, Hospital of the Medical College of Pennsylvania

Julian Katz, MD is a member of the following medical societies: American College of Gastroenterology, American College of Physicians, American Gastroenterological Association, American Geriatrics Society, American Medical Association, American Society for Gastrointestinal Endoscopy, American Society of Law, Medicine & Ethics, American Trauma Society, Association of American Medical Colleges, and Physicians for Social Responsibility

Disclosure: Nothing to disclose.

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