A case of idiopathic methemoglobinemia treated by ascorbic acid and methylene blue

King, E. J.; White, John; Gilchrist, Margaret

doi:10.1002/path.1700590119

Cited by 16 publications

(4 citation statements)

References 6 publications

(2 reference statements)

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“…That the deficiency can be corrected by methylene blue has been reported by a number of investigators (32,45,47) and is further supported by the data in Table IV, Part B. The concentration of methemoglobin in the erythrocytes from the patient with congenital methemoglobinemia was increased by incubating the cells with sodium nitrite.…”

Section: Methodssupporting

confidence: 69%

The Reduction of Methemoglobin in Human Erythrocytes Incubated With Purine Nucleosides*†

Jaffé¹

1959

J. Clin. Invest.

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Section: Methodssupporting

confidence: 69%

The Reduction of Methemoglobin in Human Erythrocytes Incubated With Purine Nucleosides*†

Jaffé¹

1959

J. Clin. Invest.

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“…G. Ball. reversion was inhibited by M/300 iodoacetate. The beneficial therapeutic effect of methylene blue has been observed clinically for 15 years in secondary types of methemoglobinemia (32)(33)(34) and has been recently reported in congenital methemoglobinemia (8,14). The marked therapeutic effect of methylene blue in our patient suggests that it replaces some constituent of normal cells that is necessary for the enzymatic reduction of methemoglobin and that this is lacking in the cells of our patient.…”

mentioning

confidence: 54%

“…The congenital type of methemoglobinemia is present from birth and is characterized by the constancy of the level of methemoglobin, by the relatively mild associated symptoms, and by polycythemia with no evidence of excessive blood destruction. There are 15 verified cases of congenital methemoglobinemia in the literature (1)(2)(3)(4)(5)(6)(7)(8)(9)(10) and 10 additional cases of idiopathic methemoglobinemia (11)(12)(13)(14)(15) in which definite information as to the congenital aspect was lacking. In all of these cases the abnormalpigmentwas identified spectroscopically.…”

mentioning

confidence: 99%

Congenital Methemoglobinemia. A Clinical and Biochemical Study of a Case 1

Eder¹,

Finch²,

McKee³

1949

J. Clin. Invest.

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Congenital methemoglobinemia is an unusual condition in which a large amount of intracellular hemoglobin exists as methemoglobin-pigment in which the hemoglobin iron is in the ferric state and incapable of carrying oxygen. The congenital type of methemoglobinemia is present from birth and is characterized by the constancy of the level of methemoglobin, by the relatively mild associated symptoms, and by polycythemia with no evidence of excessive blood destruction. There are 15 verified cases of congenital methemoglobinemia in the literature (1-10) and 10 additional cases of idiopathic methemoglobinemia (11)(12)(13)(14)(15) in which definite information as to the congenital aspect was lacking. In all of these cases the abnormalpigmentwas identified spectroscopically. Laboratory data: Hinton test for syphilis was negative; RBC was 6.5 million per mm.3; hematocrit was 58 per cent; Hb was 19 gms. per 100 cc.; WBC was 7,850 per mm.' with a normal differential count; reticulocytes were 2.2 per cent. Hypotonic saline fragility was normal. Bone marrow puncture showed only a normoblastic hyperplasia, consistent with his polycythemia. Urine and stool examinations were negative. Plasma proteins were 7.8 gms. per 100 cc. with 4.4 gms. of albumin; serum bilirubin was 0.5 mgm. per 100 cc. Venous pressure was 100 cm. of water; circulation time was 14 sec. (decholin) ; vital capacity was 4 liters. Bromsulfalein excretion test showed a retention of 5 per cent of the dye in the serum in 30 min. EKG was normal; BMR was -15 and -12 per cent. X-ray of the chest showed normal lung fields with a heart of normal size and shape. X-ray of the large bowel showed no abnormalities. Spectroscopic examination of the blood showed the absorption bands characteristic of methemoglobin. By difference between total pigment and oxygen capacity it was estimated that 45 per cent of the total blood pigment was in the form of methemoglobin. Subsequent determinations of methemoglobin were done by the colorimetric method of Evelyn and Malloy (16). This method was checked by the carbon monoxide capacity method of Van Slyke and Hiller (17) and found to be accurate to + 0.2 gm. in our hands.After a control period of one week in which the level of methemoglobin remained constant he was given 90 mgm. of methylene blue intravenously. In 10 minutes half of the methemoglobin disappeared and by 30 minutes over 95 per cent had disappeared. He was then able to exercise vigorously without headaches and breathlessness which had previously so limited his activity. During the course of the following five years he was treated with ascorbic acid and with methylene blue by mouth. It was found that 240 mgm. of methylene blue in enteric coated tablets per day were well tolerated and were sufficient to keep him free of cyanosis. Coincident with this treatment polycythemia and reticulocytosis subsided. No toxic effects from this long-sustained treatment have been observed.

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“…10 Thirdly, there was no his¬ tory of exposure to substances that can cause methemoglobinemia, such as con¬ taminated well water,11 aniline dye in diapers,|| phenytoin,14 or coal tar. 10 Thirdly, there was no his¬ tory of exposure to substances that can cause methemoglobinemia, such as con¬ taminated well water,11 aniline dye in diapers,|| phenytoin,14 or coal tar.…”

Section: Commentmentioning

confidence: 99%

Congenital Methemoglobinemia in the Newborn Period

Dine¹

1956

Arch Pediatr Adolesc Med

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A case of idiopathic methemoglobinemia treated by ascorbic acid and methylene blue

Cited by 16 publications

References 6 publications

The Reduction of Methemoglobin in Human Erythrocytes Incubated With Purine Nucleosides*†

The Reduction of Methemoglobin in Human Erythrocytes Incubated With Purine Nucleosides*†

Congenital Methemoglobinemia. A Clinical and Biochemical Study of a Case 1

Congenital Methemoglobinemia in the Newborn Period

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