Evidence map›Paper›PMID 28509131›Full record

ArticleCEN case reports2017

Growth hormone therapy for a patient with idiopathic Fanconi syndrome and growth hormone deficiency.

Takayuki Okamoto, Yasuyuki Sato, Takeshi Yamazaki, Asako Hayashi, Toshiyuki Takahashi

Open access · bronzeAbstract read
In one paragraph

Article in CEN case reports, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact, top 99% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed, 1 citations in OpenAlex.

  1. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

5 authors at 2 institutions in 1 country.

Takayuki OkamotoDepartment of Pediatrics, Hokkaido University Hospital, N15, W7, Sapporo, 060-8638, Japan. okamon@med.hokudai.ac.jp.ORCID http://orcid.org/0000-0002-6029-451X
Yasuyuki SatoDepartment of Pediatrics, Hokkaido University Hospital, N15, W7, Sapporo, 060-8638, Japan.
Takeshi YamazakiDepartment of Pediatrics, Hokkaido University Hospital, N15, W7, Sapporo, 060-8638, Japan.
Asako HayashiDepartment of Pediatrics, Hokkaido University Hospital, N15, W7, Sapporo, 060-8638, Japan.
Toshiyuki TakahashiDepartment of Pediatrics, Hokkaido University Hospital, N15, W7, Sapporo, 060-8638, Japan.
Hokkaido University Hospital · JPHokuyukai Neurology Hospital · JP

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Idiopathic Fanconi syndrome (FS) is characterized by a generalized dysfunction of the renal proximal tubules. Patients with FS often exhibit growth retardation due to complex factors, such as hypophosphatemia, metabolic acidosis, disturbed vitamin D metabolism and hypokalemia. To date, one FS patient has been reported to exhibit growth failure due to growth hormone deficiency (GHD), but the long-term clinical course of recombinant human GH (rhGH) therapy has not been reported. At 10 months of age, the patient was admitted to our hospital due to growth failure. Blood and urinary biochemical abnormalities, such as hypophosphatemia, metabolic acidosis, glycosuria and low-molecular-weight proteinuria, indicated a generalized dysfunction of the renal proximal tubules. The presence of cystinosis, collagen diseases, toxic agents and metabolic diseases were excluded. These features are compatible with idiopathic FS. Treatment with high-dose alkali, potassium citrate, phosphate buffer, hydrochlorothiazide and vitamin D supplement was initiated. The biochemical abnormalities achieved nearly normal values, and the patient's height was within -2.5 SD at the age of 2 years. However, his height did not continue to increase at the same rate and gradually declined to -2.9 SD at 4 years of age. GH stimulation test demonstrated GHD. After initiation of rhGH therapy, his height improved to -2.0 SD at the age of 9 years with no adverse effects. In conclusion, we report the case of a patient with FS and GHD who continued rhGH therapy for 5 years. The differential diagnosis of GHD should also be considered for FS patients with short stature.

Indexed as

GHDGrowth hormone deficiencyIdiopathic Fanconi syndromeRecombinant human growth hormone

Identifiers

PMID28509131
PMCPMC5438813
OpenAlexW2586868318

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.