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In a case of a 26-year-old African Ainhum-Syndrome is reported and compared with a Pseudo-Ainhum-Syndrome by a 44-year-old German woman with chronical Polyneuropathy. Short description of both syndromes.
Dactylolysis spontanea is an idiopathic condition affecting the fifth toe, and sometimes other toes, that is frequently bilateral, with lesions in different stages. Between 1977 and 1999, a total 6000 radiographic studies of the feet were reviewed in a mainly African American population in Chicago, Illinois. After an initial screening based on the Cole criteria, 581 patients were selected and re-examined, and amplification techniques were performed. After reviewing the complementary exams, 102 patients were diagnosed with dactylolysis spontanea or ainhum. Soft-tissue constriction was the most frequently presented radiological sign on the initial screening. Kurtosis at the digit plantar fold and marked rotation of the fifth toe were normal findings in asymptomatic patients. Demographics, comorbidities, and radiological findings were analyzed in the selected population. Associated diseases occurring in these patients appeared to have no specific etiologic correlation with ainhum. African Americans and the dark-skinned population are affected exclusively by this condition, presumably due to the fibrogenic tendency of these individuals. Early diagnosis and accurate staging of ainhum are facilitated by radiological examination of the feet. The findings suggest that this condition is underdiagnosed and overlooked because its low prevalence and variable clinical presentations that might mimic more common etiologies, including localized trauma.
Ainhum is the spontaneous autoamputation of toes as a result of the formation of a constricting band. It usually affects the fifth toe bilaterally and predominantly affects blacks in tropical regions, but has been uncommonly reported in temperate regions as well, including the United States. Of the 29 cases identified in the American literature since 1960, only 6 were published in the internal medicine literature. Because of the rarity of ainhum and consequent lack of attention in the clinical literature, this condition may be relatively unknown and therefore unrecognized by practicing internists. A patient with ainhum is described and the clinical features of this rare disease are emphasized to facilitate recognition and appropriate management.
Ainhum (dactylolysis spontanea) is a poorly understood disease characterized by spontaneous autoamputation of the fifth toe. Its incidence is highest among blacks and in the tropical and subtropical climates. The present study is the first to present certain gross and semimicroscopic findings. Moreover, scanning electron microscopic and histochemical data are presented and these reportings are believed to be unique. Hyperkeratosis and parakeratosis, together with elongation of the epidermal rete pegs and acanthosis, are observed and these findings are associated with the presence of numerous fibroblasts and wound repair phenomena. The leukocytic infiltrate is identified as being principally of T lymphocyte type, suggesting the existence of an unknown immunologic response. Relevant intraoperative data, radiographs, and light microscopy, as well as transmission electron microscopy, have been reported in this study. The spontaneous amputation of the fifth toe, designated as dactylolysis spontanea, has no known etiology, and no treatment short of amputation. The worldwide incidence of this condition is rare. Two cases have been observed at the Baltimore Veterans Administration Medical Center in the last 5 years. One patient is in an early stage of the disease. The present study involves the single instance in which amputation was elected as the treatment of choice. Because the occurrence of ainhum (dactylolysis spontanea) is rare, discussion of the disease is lacking in medical literature. It is the intent of the present study to be the definitive analysis of clinical, surgical, and surgical-pathologic data of this rare disease. Unique applications of semimicroscopic, histochemical, and light microscopic findings, as well as transmission and electron microscopy, will be presented. Speculation as to the implications of immunologic and/or wound repair responses as implicated in ainhum needs to be explored.
Ainhum or dactylolysis spontanea is characterized by the occurrence of a circular constriction located at the root of the 5th toe, rarely of a finger. It deepens, the toe becomes disabled and finally amputation occurs, spontaneously or by surgery. It is a tropical disease of the adult, the origin of which remains obscure. It seems frequent in Niger. 7 cases have been histologically studied. Classical features have been found, including hyperkeratosis, chronic dermatitis, osteoporosis with cortical bone resorption. This study has also revealed in all cases complete ligamental destruction. Moreover, vegetal foreign body granulomas have been found in 4 cases, lying deep in the dermis and in one case in a synovial membrane. Ainhum is compared with pseudo-ainhum seen in genodermatosis, in yaws and in congenital strictures. Among the usually considered etiopathogenetic hypotheses, our findings are in favor of a mechanical and inflammatory origin.
We describe the case of an Italian Caucasian man with ainhum involving both big toes. Ainhum or dactylolysis spontanea is characterized by the development of a constricting band around a toe, which progresses to spontaneous autoamputation. It usually affects the fifth toe bilaterally, but in rare cases other toes may be involved. The disease occurs in Black people living in tropical regions but occasionally has been reported in persons having fair skin.
A 39 year old woman is reported who has congenital constrictions on several fingers and toes as well as on both lower legs. Ainhum and Ainhum-like syndromes are briefly discussed.
A 23-year-old young woman presented with recurrent episodes of painful bruising along with linear erosions on the accessible areas of the body of nine years duration with a pseudo-ainhum of her left nipple for the past three months. Her case history included repeated visits to various physicians at different centers and an extensive investigative profile. A diagnosis of autoerythrocyte sensitization was made on the basis of the clinical history, dermatological examination complemented by a positive autoerythrocyte sensitization test, psychiatric evaluation and absence of any organic cause for her ailment. She was placed on psychiatric management and has remained symptom-free after six months follow-up. The case is reported for its rarity, as well as for the association of autoerythrocyte sensitization syndrome with frank dermatitis artefacta and pseudo-ainhum, which to the best of our knowledge has not yet been reported in the literature.
Ainhum (dactolysis spontanea) is most frequently seen involving the fifth toes of black men. An atypical case involving the second toes bilaterally in a white male is presented. The differential diagnosis, radiological findings and pathological appearance of this rare entity are discussed.
Ainhum is a rare constricting band disease, usually affecting digits and resulting in spontaneous amputation. Prompt symptomatic relief and latter partial resolution of the fibrotic band followed intralesional steroid injection in the case presented.
Pseudoainhum is a rare acquired or congenital disorder characterized by progressive development of a fibrotic band on a finger or toe until spontaneous autoamputation occurs. Ainhum is an idiopathic disease involving the fifth toes of black people mainly in tropical zones. Based on experience in five cases and a review of the literature, the authors describe clinical features of these diseases and analyze various etiological hypotheses including infectious (mycosis, mycobacteria), traumatic (plants), vascular (andysplasia), neurological (polyneuritis), and genetic (keratodermia) mechanisms.
AINHUM which has been described as one of the conditions to be differentiated from leprosy was met in the field in a village of Andhra Pradesh. The condition was found in a young adult male involving both the little toes. It was found to be slow progressive associated with pain.
Ainhum is an ancient disease described in 1821 by Messum and its origin is still unknown. It is a tropical or semitropical disease seen in Blacks, Whites and Indians which affects the 5th and sometimes the 4th toe and causes spontaneous amputation. Females are more often affected than males, and there is a tendency to geographical clustering of cases and familial occurrence.
An 11-year-old Turkish boy who has suffered from palmoplantar keratosis since his first year of life is presented. He is the only one of a large family to be affected. The diffuse keratosis extends to the back of the hands and feet and still has a progressive course. At the age of 6 he developed a symmetric high-tone acoustic impairment and at 10, an ainhum-like constricting band around the fifth digit of the left hand. This constellation of symptoms is highly characteristic for mutilating keratoma (Vohwinkel's disease), which is a rare disorder of keratinization. The majority of cases in the literature have had an autosomal dominant pattern of inheritance, although sporadic cases like this have also been reported as well. If constricting band proceeds to the point where spontaneous amputation seems imminent, a therapy with orally administered retinoids should be considered.
Epidermal nevus syndrome is an unusual occurrence of epidermal nevi with central nervous system (CNS), eye, and skeletal developmental abnormalities. We report an as yet undocumented association of generalized epidermal nevus with striate keratoderma, ainhum-like digital constriction, and autoamputation of the right fifth toe in a 10-year-old girl. The excellent response to oral acitretin with softening of the constrictions is also highlighted.
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