Acquired kinking of the hair: A methodologic approach

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Volume 15 Number 5, Part 2 November, 1986 Walking dandruff and Cheyletiella dermatitis REFERENCES 1. Powell RF, Palmer SM, Palmer CH, et al: Cheyletiella dermatitis. Int J Dermatol 16:679-682, 1977. 2. Fox JG, Reed C: CheyletielIa infestation of cats and their owners. Arch Dermatol 114:1233-1234, 1978. 3. Cohen SR: Cheyletiella dermatitis. Arch Dermatol 116: 435-437, 1980. 4. Lee BW: Cheyletiella dermatitis. Arch Dermatol 117: 677-678, ~98l. 5. Shelley ED, Shelley WB, Pula JF, et al: The diagnostic challenge of non-burrowing mite bites Cheyletiella yas- gull. JAMA 251:2690-2691, 1984. 6. Ayalew L, Vaillancourt M: Observations on an outbreak of infestation of dogs with Cheyletiella yasguri and its public health implications. Can Vet J 17:184-191, 1976. 7. Thomsett LR: Mite infestations of man contracted from dogs and cats. Br Med J 3:93-95, 1968. 8. Hewitt M, Walton GS, Waterhouse M: Pet animal infes- tations and human skin lesions. Br J Dermatol 85:215- 225, 1971. 9. Bjarke T, Hellgren L, Orstadius K: Cheyletiella para- sitovoreLr dermatitis in man. Aeta Derm Venereol (Stockh) 53:2t7-224, 1973. 10. van Bronswijk JEMH, de Kreek EJ: Cheyletiella (Acari: Cheyletiellidae) of dog, cat and domesticated rabbit: A review. J Med Entomol 13:315-327, 1976. 11. Hewitt M, Turk SM: Cheyletiella sp. in the personal environment. Br J Dem~atol 90:679-683, 1974. 12. Twiston Davies JH: Another acarine disease. Br J Der- mate/50:243-244, 1938. 13. van Bronswijk 3EMH, Jansen LH, Ophof AJ: Invasion of a house by the dog parasite Cheyletiella yasguri (Smi- ley 1965): A mite causing prurigo in man. Dermatologica 145:338-343, 1972. 14. Kunkle GA, Miller WH: Cheyletiella infestation in hu- mans. Arch Dermatol 116:1345, 1980. Acquired kinking of the hair: A methodologic approach RaOl E. Balsa, M.D.,* Stella Maris Ingratta, M.D.,** and Alberto G. Alvarez, Ph.D.*** La Plata, Argentina A case of acquired, nonprogressive kinking of the hair, a rare abnormality, is studied by means of optical microscopy and scanning electron microscopy. In addition, a standard of measure is proposed for estimation of the number of cuticular ceils per diameter. The criteria for confirmation of the diagnosis of this case are also described. (J AM ACAD DERMA'rOL 15: [[33-ll36, 1986.) In 1932, Wise and Sulzberger r identified kink- ing of the hair for the first time, and in 1955 Knieret a described it as allotrichia circumscripta symmetrica. It was in 1969 that Coupe and Johnston 3 reported the structural changes of this disease. From the Department of Medicine* and the Dermatology Service,** San Roque Hospital, and the Principal Research Group, Cindeca, CONICET,*** National University of La Plata. Reprint requests to: Dr. Rat~l E. Balsa, Department of Dermatology, San Roque Hospital, Casilla de Correos 350, C. P. 1900La Plata, Argentina. Taking into account these previous findings, we propose the following method for the diagnosis of this condition: 1. Clinical examination a. Hair dull and woolly in appearance, in a cir- cumbseribed area only (acquired) b. Frontal, temporal, and parietal topography (at least at the outset) c. Absence of previous trauma and of detersive action by shampoos 2. Observation by the naked eye a. Hairs twisted in a tortuous and irregular way b. First twist appearing at 3 and 4 cm from the emergence of ttle hair 1133

Transcript of Acquired kinking of the hair: A methodologic approach

Volume 15 Number 5, Part 2 November, 1986

Walking dandruff and Cheyle t i e l l a dermatitis

REFERENCES

1. Powell RF, Palmer SM, Palmer CH, et al: Cheyletiella dermatitis. Int J Dermatol 16:679-682, 1977.

2. Fox JG, Reed C: CheyletielIa infestation of cats and their owners. Arch Dermatol 114:1233-1234, 1978.

3. Cohen SR: Cheyletiella dermatitis. Arch Dermatol 116: 435-437, 1980.

4. Lee BW: Cheyletiella dermatitis. Arch Dermatol 117: 677-678, ~98l.

5. Shelley ED, Shelley WB, Pula JF, et al: The diagnostic challenge of non-burrowing mite bites Cheyletiella yas- gull. JAMA 251:2690-2691, 1984.

6. Ayalew L, Vaillancourt M: Observations on an outbreak of infestation of dogs with Cheyletiella yasguri and its public health implications. Can Vet J 17:184-191, 1976.

7. Thomsett LR: Mite infestations of man contracted from dogs and cats. Br Med J 3:93-95, 1968.

8. Hewitt M, Walton GS, Waterhouse M: Pet animal infes-

tations and human skin lesions. Br J Dermatol 85:215- 225, 1971.

9. Bjarke T, Hellgren L, Orstadius K: Cheyletiella para- sitovoreLr dermatitis in man. Aeta Derm Venereol (Stockh) 53:2t7-224, 1973.

10. van Bronswijk JEMH, de Kreek EJ: Cheyletiella (Acari: Cheyletiellidae) of dog, cat and domesticated rabbit: A review. J Med Entomol 13:315-327, 1976.

11. Hewitt M, Turk SM: Cheyletiella sp. in the personal environment. Br J Dem~atol 90:679-683, 1974.

12. Twiston Davies JH: Another acarine disease. Br J Der- mate/50:243-244, 1938.

13. van Bronswijk 3EMH, Jansen LH, Ophof AJ: Invasion of a house by the dog parasite Cheyletiella yasguri (Smi- ley 1965): A mite causing prurigo in man. Dermatologica 145:338-343, 1972.

14. Kunkle GA, Miller WH: Cheyletiella infestation in hu- mans. Arch Dermatol 116:1345, 1980.

Acquired kinking of the hair: A methodologic approach RaOl E. Balsa, M . D . , * Stella Maris Ingratta, M . D . , * * and Alber to G. Alvarez , Ph .D.*** La Plata, Argentina

A case of acquired, nonprogressive kinking of the hair, a rare abnormality, is studied by means of optical microscopy and scanning electron microscopy. In addition, a standard of measure is proposed for estimation of the number of cuticular ceils per diameter. The criteria for confirmation of the diagnosis o f this case are also described. (J AM ACAD DERMA'rOL 15: [ [33 - l l36 , 1986.)

In 1932, Wise and Sulzberger r identified kink- ing o f the hair for the first t ime, and in 1955

Knieret a descr ibed it as allotrichia circumscripta symmet r ica . It was in 1969 that Coupe and

Johns ton 3 reported the structural changes o f this

disease.

From the Department of Medicine* and the Dermatology Service,** San Roque Hospital, and the Principal Research Group, Cindeca, CONICET,*** National University of La Plata.

Reprint requests to: Dr. Rat~l E. Balsa, Department of Dermatology, San Roque Hospital, Casilla de Correos 350, C. P. 1900 La Plata, Argentina.

Taking into account these previous f indings, we propose the fo l lowing m e t h o d for the d iagnos is o f this condit ion:

1. Clinical examination a. Hair dull and woolly in appearance, in a cir-

cumbseribed area only (acquired) b. Frontal, temporal, and parietal topography (at

least at the outset) c. Absence of previous trauma and of detersive

action by shampoos 2. Observation by the naked eye

a. Hairs twisted in a tortuous and irregular way b. First twist appearing at 3 and 4 cm from the

emergence of ttle hair

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Journal of the American Academy of

Dermatology

Fig. 1. Tuft of hair with loss of normal appearance and gloss.

.

4.

Optical microscopy a. Sharp reduction in the diameter of the shaft,

conceivably because of the short-segment twists of flattened hair

b. Broadened sections alternating with flattened ones

Morphologic changes observed by scanning electron microscopy a. Partial twisting of the hair on its longitudinal

axis b. Spindle-shaped broadening with eventual fis-

sures c. Sporadic sectors of pili canaliculati d. Increase in the number of cuticular cells per

diameter in the twisted sections

CASE REPORT

A white 25-year-old man had an alteration of the normal characteristics of his hair in a tuft situated in the left frontotemporoparietal region, with loss of nor- mal orientation (Fig. 1). The rest of the hair presented no malformation. This anomaly, detected about 2 years previously, did not show progression.

Family history was investigated but was noncontrib- utory. The hair was tortuous, the twisting being more evident in the distal section. The patient reported that he had had his hair cut at 3 or 4 cm from its emergence so that the alterations would not be noticed. A tendency

Fig. 2. Evident reduction in shaft diameter.

toward breakage was not detected; the growth rate of the hair in that area was similar to that of the normal region.

On naked-eye examination, hairs presented an irreg- ular, twisted, and tortuous appearance.

On optical microscopy examination, periodic reduc- tions in the diameter of the shaft were detectable (Fig. 2), together with some broadened and other flattened sections. At the same time, twists of hair appeared every 3 mm.

When examined by scanning electron microscopy approximately every 3 ram, hairs showed twists about 1 mm long, the number of these twists varying ac- cording to the length of the hairs. In each curl the shaft described a rotation of 180 degrees on its longitudinal axis (Fig. 3). Sporadic spindle-shaped, broadened sec- tors of the shaft were observed (Fig. 4), with occasional fractures (Fig. 5). These flattened sections were 0.05 rnm long, and in them the cylindric shape of the shaft became flat without rotation (genuine flatness). Some sections--not all--revealed hairs with characteristics of pili canaliculati.

Blood test results were normal, including determi- nation of the copper level. A biopsy of the scalp at the level of the affected hairs revealed that the epidermis, dermis, and sebaceous glands presented no morphologic alterations, nor did the shafts until they emerged from the scalp.

DISCUSSION

We have already described acquired kinky hair. What is noteworthy about our case is that it was not progressive during a period of 2 years, in- cluding a whole year of our fol low-up during this time. It presented no alterations in c o l o r - - o n l y in texture.

The differential diagnosis obliges us to exclude

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Kinking hair 1135

Fig. 3. Twisting of hair on its longitudinal axis with a 180-degree rotation (scanning electron microscopy).

~? ~ ii ;i;i!ii i! i iiiii!i!i ¸!!~

~ ?~ i!!iiii~!ii!ii~ !!ii!;;ili!iiiii!?!ii;iiiiii!i:~i

i i iii !!! i !~iiiil ii ; ~ iii i ilil !ili ~i! ~iii! ~il ~il !~ii!iill ~I!IIIIII i ~I~I~; ii

iii i !!iiill ...... ii!ii!i

i I ...................... /i Fig. 5. Spontaneous fracture at the level of broadened section (scanning electron microscopy).

Fig. 4. Broadening of hair shaft.

this anomaly from others that compose the pili torti syndromes from birth, such as the syndromes of Menkes, 4 of Bj6rnstad, 5 and of Crandall 6 and the classic pili torti without the development of syn- dromic vincula. 7-9 In addition, we should exclude the so-called woolly hair abnormalities, such as

hereditary or familial woolly hair nevus, straight hair nevus, whisker hair syndrome, *'m'~I tricho- dento-osseous syndrome, 12 and pseudomonile- thrix. J3,~4

~Ferrando J, Gratacos MR, Fontamau R: Wooly hair: Estudio his- tol6gico y ultraestructural ea euatro casos. Aeta Derm Sifilogr (Madrid) 1979;70:203-13.

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This anomaly should also be differentiated f rom others in which the altered shaft presents dissimilar character is t ics , such as bayonet -shaped hair, *,Ls in which the alteration is spindle shaped and solely distal, and spunglass hair. ~6 The latter hair disorder is different f r om kinky hair because spun-glass hair is a mal format ion of the shaft f r om its bulb and is not iceable in histologic samples with erasure o f cuticular cells , is which does not occur in kinky hair.

With all those conditions that are due to hered- i tary or familial trends having been discarded, our pat ient should be described as a true example o f acquired k inky hair if we consider the similarities be tween this case and the findings of Rebora and Ouarrera. ~v

It is only by means o f e lec t ron scanning mi- c roscopy that we can now define anomalies that were impossible to categorize in the past. ~s,tg

Measurement of an anomalous hair was carried out and compared with that o f a normal hair in order to establish, by scanning microscopy, a unit o f measure o f the cuticular cells that might help in the study o f the various hair anomalies. We suggest the use of this unit, which we have des- ignated as cuticular cells per diameter (cpd). For this purpose, the count should start f rom the first appearance o f a whole cut icular cell, including the visible section of the last one , whether the cell is complete or not.

This s tandard always requires the transfer, at the same site, o f the hair d iameter along the length o f the hair and the count of the cuticular ceils in that section, fo l lowing a line. In our count o f normal hairs the rat io was 8 to 10 cpd. In regard to ac- quired k inky hair, the var ia t ion depended on the section under study. The sect ion without twists coincided with the normal number o f cuticular cells per diameter, whereas in the areas displaying twisting of hair, the count rose to be tween 14 and 16 cpd.

*Pierard G: Structure du poll en baionnette. Arch Beiges Dermatol 1972;28:350-61.

REFERENCES

1. Wise F, Sulzberger MD. Acquired progressive kinking of the scalp hair accompanied by changes in its pigmen- tation: correlation of an unidentified group of cases pre- senting circumscribed areas of kinky hair. Arch Dermatol Syph 1932;25:99-110.

2. Knierer W. Allotrichia circumscripta symmetrica cap- illitii (Pubeshaarnaevus). Dermatol Wschrift 1955; 132:794.

3. Coupe RL, Johnston MM. Acquired progressive kinking of the hair. Arch Dermatol 1969;100:191-5.

4. Dank DM, Campbell PE, Stevens BJ, et al. Menkes' kinky hair syndrome: inherited defect in copper ab- sorption with widespread effects. Pediatrics 1972;50: 188-200.

5. Ebling FJ, Rook A. Hair. In: Rook A, Wilkinson DS, Ebling FJG, eds. Textbook of dermatology. 3d ed. Ox- ford: Blackwell Scientific Publications, Ltd, 1979:1801.

6. Crandall BF. A familial syndrome of deafness, alopecia, and hypogonadism. J Pediatr 1973;82:461-5.

7. Munro DD, Darley C. Hair. In: Fitzpatrick RB, Eisen AZ, Wolff K, et al, eds. Dermatology in general medicine. 2d ed. New York: McGraw-Hill Book Co, 1979:467.

8. Dawber R, Comaish S. Scanning electron microscopy of normal and abnormal hair shafts. Arch Dermatol 1970; 101:316-22.

9, Zaun H. Pathologie der Haare. In Schnyder UW, editor. Histopathologie tier Haut. 2d ed. Berlin: Springer-Verlag, 1978:501-3.

10. Norwood OT. Whisker hair. Arch Dermatol 1979; 115:930-1.

11. Norwood OT. Whisker hair: an update. Cutis 1981; 27:651-2.

12. Lichtenstein J, Watson R, Jorgenson R, et al. The tricho- dento-osseous (TDO) syndrome. Am J Hum Genet 1972;24:569-81.

13. Bentley PB, Bayles MAH. A previously undescribed he- reditary hair anomaly (pseudomonilethrix). Br J Der- matol 1973;89:159-67.

14. Bentley PB, Bayles MAH. Pseudomonilethrix [Letter to Editor] Br J Dermatol 1975;92:113-5.

15. Pierard GE. Structure et interpretation pathogSnique: dys- trophies acquises du cheveu. Ann Dermatol Syph (Paris) 1975;102:137-43.

16. Ferrando J, Gratacos MR, Fontamau R. Sindrome de los eabellos impeinables. Med Cutan Ibero Lat Am 1977; 5:39-46.

17. Rebora A, Guarrera M. Acquired progressive kinking of the hair. J AM ACAD DERMATOL 1985;12:933-6.

18. Caputo R, Ceccarelli B. Study of normal hair and of some malformations with a scanning electron micro- scope. Arch Klin Exp Dermatol 1969;234:242-9.

19. Bottons E, Wyatt E, Comaish S. Progressive changes in cuticular pattern along the shafts of human hair as seen by scanning electron microscopy. Br J Dermatol 1972; 86:379-84.