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April 1959

Rectal, Ocular, and Submaxillary Pain: A Familial Autonomic Disorder Related to Proctalgia Fugax: Report of a Family

Author Affiliations

San Francisco
Senior Medical Student (Dr. Hayden) and Assistant Professor (Dr. Grossman), Department of Pediatrics, University of California Medical Center.

AMA Am J Dis Child. 1959;97(4):479-482. doi:10.1001/archpedi.1959.02070010481013

Introduction  This paper is concerned with an unusual hereditary idiopathic, presumably autonomic, syndrome characterized by very brief episodes of excruciating rectal pain associated with flushing of the buttocks and legs, ocular pain and flushing of the eyelid and periorbital skin, and submaxillary pain. The rectal pain appears to be similar, though not identical, to that described in the condition known as proctalgia fugax (first described by Thaysen, in 1935).1 To our knowledge, this syndrome has not been previously reported.

Report of Family  Infant.—This 9-month-old white boy was first seen in the Pediatric Clinic at the University of California Medical Center on Dec. 13, 1956, with the chief complaint of episodes of rectal pain and flushing of the lower extremities identical to that which his father and a number of his father's blood relatives had suffered during childhood. His past history revealed that he had been well and healthy