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Epidemiology of Hexachlorobenzene-Induced Porphyria in Turkey: Clinical and Laboratory Follow-up After 25 Years

Epidemiology of Hexachlorobenzene-Induced Porphyria in Turkey: Clinical and Laboratory Follow-up... Abstract • Cutanea tarda and mixed porphyric symptoms resulted from accidental ingestion of hexachlorobenzene (HCB)-treated seed grain in Turkey 25 years ago and involved about 4,000 persons with a 10% mortality. Twenty-five years later, 161 patients, 63 women and 98 men, with histories of HCB exposure as children or young adults were studied. Many patients had persistent abnormal porphyrin metabolism and prominent neurological, dermatologic, and orthopedic symptoms and signs. Thyromegaly was seen in 60% of the women and 27% of the men. The children born to porphyric mothers 25 years ago all died of pembe yara secondary to maternal milk and transplacental transfer of HCB. Current lactation specimens of porphyric patients show high HCB levels, up to 3.12 ppm, but the infant offspring appear normal. References 1. Cam S: Une nouvelle dermatose epidemique des enfants . Ann Derm Syph 1960;87:393-397. 2. Cam S: Cases of skin porphyria related to hexachlorobenzene intoxication . Saglik Dergisi 1958;32:215-216. 3. Cam S: Cutaneous porphyria related to intoxication . Dirim 1959;34:11-15. 4. Dogramaci I, Wray JE, Ergene T, et al: Porphyria turcica: A survey of 592 cases of cutaneous porphyria seen in southeastern Turkey . Turk J Pediatr 1962;4:138. 5. Cetingil AI, Ozen MA: Toxic porphyria . Blood 1960;16:1002-1011. 6. Schmid R: Cutaneous porphyria in Turkey . N Engl J Med 1960;263:397-398.Crossref 7. Cam S, Nigogosyan G: Acquired toxic porphyria cutanea tarda due to hexachlorobenzene: Report of 348 cases caused by this fungicide . JAMA 1963;183:88-91.Crossref 8. Kantemir I, Giner S, Kayaalp O: Investigations with hexachlorobenzene and organic mercury compounds . Turk Hig Tecr Biyol Derg 1960;20:19. 9. Ockner RK, Schmid R: Acquired porphyria in man and rat due to hexachlorobenzene intoxication . Nature 1961;189:499.Crossref 10. Wray JD, Muftu Y, Dogramaci I: Hexachlorobenzene as a cause of porphyria turcica . Turk J Pediatr 1962;4:132. 11. Peters HA, Johnson SAM, Cam S, et al: Hexachlorobenzene-induced porphyria: Effect of chelation on the disease, porphyrin and metal metabolism . Am J Med Sci 1966;251:314-322.Crossref 12. Bleiberg J, Wallen M, Brodkin R, et al: Industrially acquired porphyria . Arch Dermatol 1964;89:793-797.Crossref 13. Rimington C: Quantitative determinations of porphobilinogen and porphyrins in urine and porphyrins in faeces and erythrocytes. Assoc Clin Path Broadsheet No. 70 (revision of No. 36), August 1971. 14. Cripps DJ, Peters HA: Fluorescing erythrocytes and porphyrin screening test on urine, stool, and blood . Arch Dermatol 1967;96:712-720.Crossref 15. Mauzerall D, Granick S: The occurrence and determination of delta aminolevulinic acid and porphobilinogen in the urine . J Biol Chem 1956;219:435. 16. Piepkorn MW, Hamernyik P, Labbe RF: Modified erythrocyte uroporphyrinogen I synthase assay, and its clinical interpretation . Clin Chem 1978;24:1751. 17. Thomas JF (ed): Manual of Analytical Methods for the Analysis of Pesticide Residues in Human Environmental Samples , section 12-D (2), Dec 2, 1974. Research Triangle Park, NC, US Environmental Protection Agency Health Effects Research Laboratory, 1974. 18. Donald GF, Hunter GA, Roman W, et al: Cutaneous porphyria, favourable results in 12 cases treated by chelation . Aust J Dermatol 1965;8:97-115.Crossref 19. MacGregor AG, Nicholas REH, Rimington C: Porphyria cutanea tarda: Investigation of case, including isolation of some hitherto undescribed porphyrins . Arch Intern Med 1952;90:483-504.Crossref 20. Schrumpf A: Porphyria improved after treatment with BAL . Acta Med Scand 1953;145:338-340.Crossref 21. Woods SM, Peters HA, Johnson SAM: Cutaneous porphyria with porphobilinogenuria: A review and report of a case treated by chelation . Arch Dermatol 1958;77:559-567.Crossref 22. Woods SM, Peters HA, Johnson SAM: Chelation therapy in cutaneous porphyria: A review and report of a five-year recovery . Arch Dermatol 1961;84:920-927.Crossref 23. Epstein JH, Epstein NN, Greenlee M: Epidermolysis bullosa acquisita (tardive) and porphyria cutanea tarda . Arch Dermatol 1959;80:713-724.Crossref 24. Galambos JT, Peacock LB: The use of chelating agents in the treatment of acute porphyria . Ann Intern Med 1959;50:1056-1061.Crossref 25. Gulbrandsen R, Wigmostad KR: Porphyria acuta, 2 tilfellemed fremtredende neuropsykiatriske sumptomer, behandlet med BAL . Tidsskr Nor Laegeforen 1958;9:420. 26. Luby ED, Ware JG, Senf R, et al: Stress and the precipitation of acute intermittent porphyria . Psychosom Med 1959;21:34-39.Crossref 27. McCabe ES: Case report: Recovery in a case of ascending landry paralysis due to porphyria . Am Practit 1955;6:878-880. 28. Painter JT, Morrow EJ: Porphyria: Its manifestations and treatment with chelating agents . Tex J Med 1959;55:811-818. 29. Paul KG, Thyresson N: The effect of BAL on a case of 'cutaneous porphyria.' Acta Derm 1954;34:403-408. 30. Redeker AG, Sterling RE, Archer B: Porphyria cutanea tarda: Report of a case effectively treated with dimercaprol (BAL) . Arch Intern Med 1959;104:779-782.Crossref 31. Schirger A, Martin WJ, Goldstein NP, et al: Orthostatic hypotension in association with acute exacerbations of porphyria . Mayo Clin Proc 1962;37:7-11. 32. Treanor WJ, Rupe CE: Porphyria, 'an enzyme lesion': Review of basic and clinical aspects . Henry Ford Hosp Bull 1957;3:162-170. 33. Harmel-Tourneur L, Dropsy G: Porphyrie cutanee tardive traitee par le BAL . Bull Soc Fr Dermatol Syphiligr 1959;66:543-546. 34. Roman W: Some observations on the treatment of porphyrias with caEDTA . Enzymologia 1967;32:37-82. 35. Dogramaci I, Duzgunes O, Ergene T, et al: A possible genetic factor in the etiology of porphyria turcica . Turk J Pediatr 1962;4:193-200. 36. Cripps DJ, Gocmen A, Peters HA, et al: Experimental pembe yara: Hexachlorobenzene intoxication in neonates. Read before the Society for Investigative Dermatology, San Francisco, April 1981. 37. Kroger M: Insecticide residues in human milk . J Pediatr 1972;80:401-405.Crossref 38. Courtney KD: Hexachlorobenzene (HCB): A review . Environ Res 1979;20:225-266.Crossref 39. Cabral JR, Shubik P, Mollner T, et al: Carcinogenic activity of hexachlorobenzene in hamsters . Nature 1977;269:510-511.Crossref 40. Knauf V, Hobson W: Hexachlorobenzene ingestion by female rhesus monkeys: Tissue distribution and clinical symptomatology . Bull Environ Contam Toxicol 1979;21:243-248.Crossref 41. Berwick P: 2,4,-Dichlorophenoxyacetic acid poisoning in man: Some interesting clinical and laboratory findings . JAMA 1970;214:1114-1117.Crossref 42. Eberstein A, Goodgold J: Experimental myotonia induced in denervated muscles by 2,4-D . Muscle Nerve 1979;2:364-368.Crossref 43. Albers JW, Robertson WC, Daube JR: Electrodiagnostic findings in acute porphyric neuropathy . Muscle Nerve 1978;1:292-296.Crossref 44. Bick M: Chlorinated hydrocarbon residues in human body fat . Med J Aust 1967;1:1127-1130. 45. Brady MN, Siyali DS: Hexachlorobenzene in human body fat . Med J Aust 1972;1:158-161. 46. Siyali DS: Hexachlorobenzene and other organochloride pesticides in human blood . Med J Aust 1972;2:1063-1066. 47. Burns JE, Miller FM, Gomes ED, et al: Hexachlorobenzene exposure from contaminated DCPA in vegetable spraymen . Arch Environ Health 1974;29:192-194.Crossref 48. Isensee AR, Holden ER, Woolson EA, et al: Soil persistence and aquatic bioaccumulation potential of hexachlorobenzene (HCB) . J Agric Food Chem 1976;24:1210-1214.Crossref 49. Laseter JS, Bartell CK, Laska AL, et al: An Ecological Study of Hexachlorobenzene (HCB) publication 560/6-76-009. Environmental Protection Agency, Office of Toxic Substances, 1976. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Neurology American Medical Association

Epidemiology of Hexachlorobenzene-Induced Porphyria in Turkey: Clinical and Laboratory Follow-up After 25 Years

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References (53)

Publisher
American Medical Association
Copyright
Copyright © 1982 American Medical Association. All Rights Reserved.
ISSN
0003-9942
eISSN
1538-3687
DOI
10.1001/archneur.1982.00510240006002
Publisher site
See Article on Publisher Site

Abstract

Abstract • Cutanea tarda and mixed porphyric symptoms resulted from accidental ingestion of hexachlorobenzene (HCB)-treated seed grain in Turkey 25 years ago and involved about 4,000 persons with a 10% mortality. Twenty-five years later, 161 patients, 63 women and 98 men, with histories of HCB exposure as children or young adults were studied. Many patients had persistent abnormal porphyrin metabolism and prominent neurological, dermatologic, and orthopedic symptoms and signs. Thyromegaly was seen in 60% of the women and 27% of the men. The children born to porphyric mothers 25 years ago all died of pembe yara secondary to maternal milk and transplacental transfer of HCB. Current lactation specimens of porphyric patients show high HCB levels, up to 3.12 ppm, but the infant offspring appear normal. References 1. Cam S: Une nouvelle dermatose epidemique des enfants . Ann Derm Syph 1960;87:393-397. 2. Cam S: Cases of skin porphyria related to hexachlorobenzene intoxication . Saglik Dergisi 1958;32:215-216. 3. Cam S: Cutaneous porphyria related to intoxication . Dirim 1959;34:11-15. 4. Dogramaci I, Wray JE, Ergene T, et al: Porphyria turcica: A survey of 592 cases of cutaneous porphyria seen in southeastern Turkey . Turk J Pediatr 1962;4:138. 5. Cetingil AI, Ozen MA: Toxic porphyria . Blood 1960;16:1002-1011. 6. Schmid R: Cutaneous porphyria in Turkey . N Engl J Med 1960;263:397-398.Crossref 7. Cam S, Nigogosyan G: Acquired toxic porphyria cutanea tarda due to hexachlorobenzene: Report of 348 cases caused by this fungicide . JAMA 1963;183:88-91.Crossref 8. Kantemir I, Giner S, Kayaalp O: Investigations with hexachlorobenzene and organic mercury compounds . Turk Hig Tecr Biyol Derg 1960;20:19. 9. Ockner RK, Schmid R: Acquired porphyria in man and rat due to hexachlorobenzene intoxication . Nature 1961;189:499.Crossref 10. Wray JD, Muftu Y, Dogramaci I: Hexachlorobenzene as a cause of porphyria turcica . Turk J Pediatr 1962;4:132. 11. Peters HA, Johnson SAM, Cam S, et al: Hexachlorobenzene-induced porphyria: Effect of chelation on the disease, porphyrin and metal metabolism . Am J Med Sci 1966;251:314-322.Crossref 12. Bleiberg J, Wallen M, Brodkin R, et al: Industrially acquired porphyria . Arch Dermatol 1964;89:793-797.Crossref 13. Rimington C: Quantitative determinations of porphobilinogen and porphyrins in urine and porphyrins in faeces and erythrocytes. Assoc Clin Path Broadsheet No. 70 (revision of No. 36), August 1971. 14. Cripps DJ, Peters HA: Fluorescing erythrocytes and porphyrin screening test on urine, stool, and blood . Arch Dermatol 1967;96:712-720.Crossref 15. Mauzerall D, Granick S: The occurrence and determination of delta aminolevulinic acid and porphobilinogen in the urine . J Biol Chem 1956;219:435. 16. Piepkorn MW, Hamernyik P, Labbe RF: Modified erythrocyte uroporphyrinogen I synthase assay, and its clinical interpretation . Clin Chem 1978;24:1751. 17. Thomas JF (ed): Manual of Analytical Methods for the Analysis of Pesticide Residues in Human Environmental Samples , section 12-D (2), Dec 2, 1974. Research Triangle Park, NC, US Environmental Protection Agency Health Effects Research Laboratory, 1974. 18. Donald GF, Hunter GA, Roman W, et al: Cutaneous porphyria, favourable results in 12 cases treated by chelation . Aust J Dermatol 1965;8:97-115.Crossref 19. MacGregor AG, Nicholas REH, Rimington C: Porphyria cutanea tarda: Investigation of case, including isolation of some hitherto undescribed porphyrins . Arch Intern Med 1952;90:483-504.Crossref 20. Schrumpf A: Porphyria improved after treatment with BAL . Acta Med Scand 1953;145:338-340.Crossref 21. Woods SM, Peters HA, Johnson SAM: Cutaneous porphyria with porphobilinogenuria: A review and report of a case treated by chelation . Arch Dermatol 1958;77:559-567.Crossref 22. Woods SM, Peters HA, Johnson SAM: Chelation therapy in cutaneous porphyria: A review and report of a five-year recovery . Arch Dermatol 1961;84:920-927.Crossref 23. Epstein JH, Epstein NN, Greenlee M: Epidermolysis bullosa acquisita (tardive) and porphyria cutanea tarda . Arch Dermatol 1959;80:713-724.Crossref 24. Galambos JT, Peacock LB: The use of chelating agents in the treatment of acute porphyria . Ann Intern Med 1959;50:1056-1061.Crossref 25. Gulbrandsen R, Wigmostad KR: Porphyria acuta, 2 tilfellemed fremtredende neuropsykiatriske sumptomer, behandlet med BAL . Tidsskr Nor Laegeforen 1958;9:420. 26. Luby ED, Ware JG, Senf R, et al: Stress and the precipitation of acute intermittent porphyria . Psychosom Med 1959;21:34-39.Crossref 27. McCabe ES: Case report: Recovery in a case of ascending landry paralysis due to porphyria . Am Practit 1955;6:878-880. 28. Painter JT, Morrow EJ: Porphyria: Its manifestations and treatment with chelating agents . Tex J Med 1959;55:811-818. 29. Paul KG, Thyresson N: The effect of BAL on a case of 'cutaneous porphyria.' Acta Derm 1954;34:403-408. 30. Redeker AG, Sterling RE, Archer B: Porphyria cutanea tarda: Report of a case effectively treated with dimercaprol (BAL) . Arch Intern Med 1959;104:779-782.Crossref 31. Schirger A, Martin WJ, Goldstein NP, et al: Orthostatic hypotension in association with acute exacerbations of porphyria . Mayo Clin Proc 1962;37:7-11. 32. Treanor WJ, Rupe CE: Porphyria, 'an enzyme lesion': Review of basic and clinical aspects . Henry Ford Hosp Bull 1957;3:162-170. 33. Harmel-Tourneur L, Dropsy G: Porphyrie cutanee tardive traitee par le BAL . Bull Soc Fr Dermatol Syphiligr 1959;66:543-546. 34. Roman W: Some observations on the treatment of porphyrias with caEDTA . Enzymologia 1967;32:37-82. 35. Dogramaci I, Duzgunes O, Ergene T, et al: A possible genetic factor in the etiology of porphyria turcica . Turk J Pediatr 1962;4:193-200. 36. Cripps DJ, Gocmen A, Peters HA, et al: Experimental pembe yara: Hexachlorobenzene intoxication in neonates. Read before the Society for Investigative Dermatology, San Francisco, April 1981. 37. Kroger M: Insecticide residues in human milk . J Pediatr 1972;80:401-405.Crossref 38. Courtney KD: Hexachlorobenzene (HCB): A review . Environ Res 1979;20:225-266.Crossref 39. Cabral JR, Shubik P, Mollner T, et al: Carcinogenic activity of hexachlorobenzene in hamsters . Nature 1977;269:510-511.Crossref 40. Knauf V, Hobson W: Hexachlorobenzene ingestion by female rhesus monkeys: Tissue distribution and clinical symptomatology . Bull Environ Contam Toxicol 1979;21:243-248.Crossref 41. Berwick P: 2,4,-Dichlorophenoxyacetic acid poisoning in man: Some interesting clinical and laboratory findings . JAMA 1970;214:1114-1117.Crossref 42. Eberstein A, Goodgold J: Experimental myotonia induced in denervated muscles by 2,4-D . Muscle Nerve 1979;2:364-368.Crossref 43. Albers JW, Robertson WC, Daube JR: Electrodiagnostic findings in acute porphyric neuropathy . Muscle Nerve 1978;1:292-296.Crossref 44. Bick M: Chlorinated hydrocarbon residues in human body fat . Med J Aust 1967;1:1127-1130. 45. Brady MN, Siyali DS: Hexachlorobenzene in human body fat . Med J Aust 1972;1:158-161. 46. Siyali DS: Hexachlorobenzene and other organochloride pesticides in human blood . Med J Aust 1972;2:1063-1066. 47. Burns JE, Miller FM, Gomes ED, et al: Hexachlorobenzene exposure from contaminated DCPA in vegetable spraymen . Arch Environ Health 1974;29:192-194.Crossref 48. Isensee AR, Holden ER, Woolson EA, et al: Soil persistence and aquatic bioaccumulation potential of hexachlorobenzene (HCB) . J Agric Food Chem 1976;24:1210-1214.Crossref 49. Laseter JS, Bartell CK, Laska AL, et al: An Ecological Study of Hexachlorobenzene (HCB) publication 560/6-76-009. Environmental Protection Agency, Office of Toxic Substances, 1976.

Journal

Archives of NeurologyAmerican Medical Association

Published: Dec 1, 1982

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